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678 Collagenous gastritis: Gastroesophageal reflux
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Yousef Usta, Denise Millstine, Fredrick Kenny
682 Prostatic ductal adenocarcinoma presenting
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Tafadzwa P Makarawo, Jeffrey L OConnor
686 Chest X-Ray appearance of total opacification
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Victor Kong, Leah Naidoo, Damon Jeetoo,
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745 Hemophilic Arthropathy
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Hamidpour et al.
review article
666
OPEN ACCESS
Abstract
The purpose of this article is to use literature
search engines such as PubMed and SciFinder
to locate scholarly journal articles pertaining
saffron, its preparation, analysis, and its use
in cancer treatments. Saffron comes from the
dried red stigmas of the Crocus sativus L. flower.
Along with its use in cooking and in traditional
medicine, it has numerous applications as an
antitoxic, anti-oxidant, and anti-cancer agent,
due to its secondary metabolites and their
derivatives are safranal, crocins, crocetin and
dymethyl-crocetin. This study demonstrate
that Crocus sativus extract (CSE) and its major
constituents, crocin, and crocetin, significantly
inhibited the growth of certain cancer cells while
not effecting normal cells. Crocus sativus L.
extract should be investigated further as a viable
agent in the treatment of prostate, pancreatic or
other types of cancer.
Keywords: Crocus sativus L., Tumor inhibitor,
Saffron components, Nutritional supplements
*********
Rafie Hamidpour1, Soheila Hamidpour2, Mohsen Hamidpour3,
Mina Shahlari4
Affiliations: 1PhD, President, Pars Bioscience, Leawood,
Kansas, United States; 2MD, Pars Bioscience, Leawood,
Kansas, United States; 3PhD, Pars Bioscience, Leawood,
Kansas, United States; 4BA, Pars Bioscience, Leawood,
Kansas, United States.
Corresponding Author: Rafie Hamidpour, Pars Bioscience
LLC, 14109 Cambridge Lane, Leawood, Kansas, United
States 66224; Ph: (913) 432-0107; Fax: (913) 432-5708;
Email: rafi@parsbioscience.com
Received: 16 November 2012
Accepted: 12 January 2013
Published: 01 December 2013
*********
doi:10.5348/ijcri-2013-12-407-RA-1
Introduction
Saffron is one of the most expensive spices in the
world, derived from the dry stigmata of Crocus sativus L.
a member of the Iridaceae (Iris) family. Saffron is handharvested during the flowering season. This process is
very time consuming which involves picking the stigmata
by hand and then carefully drying the stigmata to produce
a quality product. One stigma of saffron weighs about
2 mg and each flower has three stigmata. In order to
obtain 1 kg of spice 150,000 flowers must be picked [1].
Saffron (Crocus sativus L.) is mostly cultivated
in Spain, Iran, India, Greece, China and some other
European and Asian countries. The quality and chemical
composition of saffron are affected by the region in which
saffron is grown, the drying process, the conditions of
packaging, storage of saffron and the analytical extraction
methods which have been used [1, 2].
The nutritional supplement value of saffron (Crocus
sativus L.) which was provided by pars bioscience LLC
in powder form, to Covance, Madison. WI laboratory
was analyzed and shown to include the following
contents: NL-Proximate (moisture, ash, protein, fat, total
carbohydrates, calories, and calories from fat), results of
these analyzes are given in Table 1. Vitamins (vitamin A,
vitamin C, and folic acid), and minerals (calcium, copper,
iron, magnesium, manganese, phosphorus, potassium,
sodium, and zinc) are detailed in Table 2. The results of
analysis of saffron fatty acid profile are given in Table 3.
The analysis of the saffron was conducted by grinding
and extracting saffron, and analyzing the extract using
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):666670.
www.ijcasereportsandimages.com
Results
(per 100 g serving size)
Moisture
7.7 g
Ash
4.6 g
Protein
15.6 g
Fat
5.5 g
Total Carbohydrates
69.6 g
Calories
363 cal
22.1 cal
Hamidpour et al.
667
Vitamins
Vitamin A
< 100 IU
Vitamin C
< 1.0 mg
Folic Acid
800 mg
Minerals
Calcium
124 mg
Copper
0.908 mg
Iron
23.7 mg
Magnesium
154 mg
Manganese
2.44 mg
Phosphorus
404 mg
Potassium
1750 mg
Sodium
39.0 mg
Zinc
4.15 mg
Analysis
8:0 Caprylic
10:0 Capric
12:0 Lauric
14:0 Myristic
14:1 Myristoleic
15:0 Pentadecanoic
15:1 Pentadecenoic
16:0 Palmitic
16:1 Palmitoleic
17:0 Heptadecanoic
17:1 Heptadecenoic
18:0 Stearic
18:1 Oleic
18:2 Linoleic
18:3 Gamma Linolenic
18:3 Linolenic
20:0 Arachidic
20:1 Eicosenoic
20:2 Eicosadienoic
20:3 Eicosatrienoic
20:4 Arachidonic
22:0 Behenic
Saturated Fat
Monounsaturated Fat
Polyunsaturated Fat
Sum of Fatty Acids
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):666670.
www.ijcasereportsandimages.com
DISCUSSION
Saffron is a very valuable spice with many traditional
medicinal usages. The high amount of carotenoids in
saffron including crocin, crocetin and dimethyl-crocetin
are responsible for some biological functions of saffron.
Most of the studies on the effect of saffron, indicates
the significant inhibitory effects of the Crocus sativus
components on the synthesis of nucleic acids in different
human cancer cell lines [24].
As the studies have shown, diets rich in antioxidants
will lower the risk of several chronic diseases and protect
the body against the development and growth of tumor
cells. Therefore, saffron and its constituents with their
antioxidant properties can act as a protecting agent for
the prevention of some serious diseases like cancer [25].
Crocus sativus L. extract used in several studies were
prepared from stigmas of Crocus sativus. It contains
several pharmacologically active constituents. Saffron
has antioxidant properties; these have been showed in
humans, where saffron (50 mg, twice a day) decreases
the lipoprotein oxidation susceptibility [20]. Also crude
methanol extract of saffron and its compound crocin
have been exhibited high antioxidant and scavenging
properties [21].
The oral administration of the saffron ethanolic
extract (200 mg/kg body wt) increased the life span of
Swiss albino mice intraperitoneally transplanted with
sarcoma-180 (S-180) cells, Ehrlich ascites carcinoma
(EAC) or Daltons lymphoma ascites (DLA) tumors [22],
and it has an inhibitory effect on chemical carcinogenesis
in mice using two stage assay system [16].
Crocetin protects body against free radicals and the
studies have shown its role as an antitumor agent [15]
The effect of crocetin on two different types of animal
tumors, skin papillomas and Rous sarcoma have been
studied and shown that crocetin decreased the number
of tumor cells and delayed the onset of the tumors as
well [23]. A recent study showed that crocetin (20
mg/ kg) reverted the level of lipid peroxidation induced
by Benzo (a) pyrene(B(a)P), also increased the activities
Hamidpour et al.
668
CONCLUSION
In summary, our study suggests that crocin and
crocetin found in Crocus sativus L. may be efficacious
in treating several or certain type of cancer. Considering
the popularity of locally used chemical drugs in cancer
patients, Crocus sativus L. should be investigated further
as a natural viable option in the treatment of prostate,
pancreatic, or other types of cancer.
*********
Author Contributions
Rafie Hamidpour Conception and design, Analysis
and interpretation of data, Critical revision of the article,
Final approval of the version to be published
Soheila Hamidpour Analysis and interpretation of
data, Critical revision of the article, Final approval of the
version to be published
Mohsen Hamidpour Analysis and interpretation of
data, Critical revision of the article, Final approval of the
version to be published
Mina Shahlari Conception and design, Critical revision
of the article, Final approval of the version to be published
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Rafie Hamidpour et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):666670.
www.ijcasereportsandimages.com
REFERENCES
1.
Hamidpour et al.
669
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):666670.
www.ijcasereportsandimages.com
Hamidpour et al.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
670
IJCRI 2013;4(12):671677.
www.ijcasereportsandimages.com
Hamidpour et al.
review article
671
OPEN ACCESS
Abstract
Sage (Salvia) species have been used for centuries,
worldwide as culinary herbs as well as traditional
remedies for the treatment of many common
health complications and their symptoms.
In recent decades, more studies have been
performed on the effect of sage in preventing,
controlling, or curing various diseases naturally
and more safely. A number of sage family members
especially Salvia officinalis (common sage) and
their components have shown to be beneficial in
protecting the body against oxidative stress, free
radical damages, angiogenesis, inflammation,
bacterial and virus infection, etc. Several studies
suggest that sage might potentially provide novel
natural treatments for the relief or cure of many
serious and life-threatening diseases in addition
to treating minor common illnesses such as
depression, dementia, obesity, diabetes, lupus,
autism, heart disease and cancer.
Keywords: Sage (Salvia species),
officinalis, Traditional remedies
Salvia
*********
Rafie Hamidpour1, Soheila Hamidpour2, Mohsen Hamidpour3,
Mina Shahlari4
Affiliations: 1PhD, President, Pars Bioscience, Leawood,
Kansas, United States; 2MD, Pars Bioscience, Leawood,
Kansas, United States; 3PhD, Pars Bioscience, Leawood,
Kansas, United States; 4BA in Biology, Pars Bioscience,
Leawood, Kansas, United States.
Corresponding Author: Rafie Hamidpour, Pars Bioscience
LLC, 14109 Cambridge Lane, Leawood, Kansas, UNITED
STATES. 66224; Ph: (913) 432-0107; Email: rafi@
parsbioscience.com; Fax: (913) 432-5708
Received: 16 November 2013
Accepted: 11 January 2013
Published: 01 December 2013
*********
doi:10.5348/ijcri-2013-12-408-RA-2
Introduction
The genus Salvia, commonly called sage, is the largest
member of Lamiaceae or mint family containing over 900
species throughout the world [1, 2]. The plants are mostly
aromatic, perennial, with flowers in different colors
[3]. Many species of Salvia, including Salvia officinalis
(common sage) are native to Mediterranean region and
some of the Salvia species have been used worldwide as
flavoring spices as well as traditional herbal medicine [3, 4].
Sage tea has been traditionally used for the treatment
of digestive and circulation disturbances, bronchitis,
cough, asthma, angina, mouth and throat inflammations,
depression, excessive sweating, skin, and many other
diseases [57]. Essential oils of Salvia have been used
in the treatment of large range of diseases such as,
nervous system, heart and blood circulation, respiratory,
digestive, metabolic, and endocrine diseases. In addition,
sage essential oil has shown to have carminative,
antispasmodic, antiseptic and astringent properties [8, 9].
The essential oil of Salvia species can have various
compositions depending on the genetic, climates, season,
and environmental factors [10] (Table 1). There are some
chemical compounds such as flavonoids, terpenoids and
essential oils present in different species of Salvia [3].
Essential oils are very important sources for the screening
of anticancer, antimicrobial, antioxidant, and free radical
scavenging agents [11]. Salvia officinalis is considered to
have the highest amount of essential oil compared to the
other species of Salvia [5, 12].
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S. Officinalis2
S. Officinalis3
(1R)- (+)-a-Pinene
3.70
4.50
(-)- Camphene
2.60
5.00
B- Pinene
6.00
5.20
Sabinene
0.30
B- Myrcene
3.00
3.50
a-Terpinene
0.40
(R)-(+) Limonene
1, 8 Cineole
62.0
55.0
Y- Terpinene
0.30
0.50
P-Cymene
0.60
0.60
Terpinolene
0.20
(-)-a-Thujone
1.38
1.80
B-Thujone
0.72
1.50
Camphor
8.0
10.0
(-)-Linalool
0.80
0.80
Linalyl acetate
0.60
0.30
(-)-TransCaryophyllene
2.00
1.00
Monoterpene
1.26
1.10
(+)-Menthol
Borneol
5.00
4.50
a- Terpineol
0.20
Geranyl acetate
0.30
Geraniol
0.10
0.25
Phytol
0.18
Thymol
0.80
0.70
Carvacrol
0.20
0.40
Farnesol
0.20
T r a n s - t r a n s - 0.06
Farnesol
0.15
Total Components
30
45
672
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CONCLUSION
Salvia species may represent natural, safe, and
effective treatments for many diseases and their
symptoms. In recent decades, with the increase of
pharmacological knowledge about the beneficial effects of
sage especially Salvia officinalis, these herbal medicines
with antibacterial, antioxidant, anti-inflammatory, free
radical scavenging and antitumor activities, have found
to be very effective in the development of novel natural
drugs to prevent, control and treat many minor health
Hamidpour et al.
675
*********
Author Contributions
Rafie Hamidpour Substantial contributions to
conception and design, Analysis and interpretation of
data, Revising it critically for important intellectual
content, Final approval of the version to be published
Soheila Hamidpour Analysis and interpretation of data,
Revising it critically for important intellectual content,
Final approval of the version to be published
Mohsen Hamidpour Analysis and interpretation of
data, Revising it critically for important intellectual
content, Final approval of the version to be published
Mina Shahlari Acquisition of data, Drafting the article,
Final approval of the version to be published
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Rafie Hamidpour et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
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www.ijcasereportsandimages.com
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26. Stanojevic D, Comic L, Stefanovic O, Solujic-Sukdolak
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27. Perry NS, Bollen C, Perry EK, Ballard C. Salvia
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30. Akhondzadeh S, Noroozian M, Mohammadi M,
Ohadinia S, Jamshidi AH, Khani M. Salvia officinalis
extract in the treatment of patients with mild to
moderate Alzheimers disease: A double blind,
randomized and placebo-controlled Trial. J Clin
Pharm Ther 2003 Feb;28(1):539.
31. Iuvone T, De Filipis D, Esposito G, DAmico A,
Izzo A. The spice sage and its active ingredient
rosmarinic acid protect PC12 cells from amyloidbeta Peptide-induced neurotoxicity. The Journal of
Pharmacology and Experimental Therapeutics 2006
Jun;317(3):11439.
32. Moss L, Rouse M, Wesnes KA, Moss M. Differential
effects of the aromas of Salvia species on memory and
mood. Hum Psychopharmacol 2010 Jul;25(5):388
96.
33. Christensen KB, Jrgensen M, Kotowska D, Petersen
RK, Kristiansen K, Christensen LP. Activation
of the nuclear receptor PPAR? by metabolites
isolated from sage (Salvia officinalis L.). Journal of
Ethnopharmacology 2010 Oct 28;132(1):12733.
34. Keshavarz M, Bidmeshkipour A, Mostafavi A,
Mansouri K, Mohamadi-Motlagh H. Anti tumor
activity of Salvia officinalis is due to its antiangiogenic, anti-migratory and anti-proliferative
effects. Cell Journal 2011;12(4):47782.
35. Jedinak A, Muckova M, Kostalova D, Maliar T,
Masterova I. Antiprotease and antimetastatic activity
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36. Pedro D, Ramos A, Lima C, Baltazar F, PereiraWilson C. Modulation of DNA damage prevention
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prevention. BMC Proceedings 2010;4:53.
37. Tildesley NT, Kennedy DO, Perry EK, et al. Salvia
lavandulaefolia (Spanish sage) enhances memory in
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healthy young volunteers. Pharmacol Biochem Behav
2003 Jun;75(3):66974.
38. Ninomiya K, Matsuda H, Shimoda H, et al. Carnosic
acid, a new class of lipid absorption inhibitor from
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Usta et al.
CASE REPORT
678
OPEN ACCESS
Abstract
Introduction:
Collagenous
gastritis,
first
described in 1989, is a very rare disease with only
about 50 cases ever reported to date. This is a
histologic diagnosis characterized by a thickened
subepithelial collagen bands in the gastric
mucosa. It is associated with an inflammatory
infiltrate with mixed chronic inflammatory
infiltrate of the lamina propria and surface
epithelial damage. We present an adult case
with symptoms of reflux that does not fit the
recent literature that has combined all adult
collagenous gastritis cases with collagenous
colitis and chronic watery diarrhea. Case Report:
A 55-year-old female presented to us with
chronic heart burn. She was clinically diagnosed
with gastroesophageal reflux disease. Upper
endoscopy was done and she was found to have
gastric and gastroesophageal junction collagen
deposits on biopsy. She did not have collagenous
colitis on colonoscopy biopsies or any other
systemic disease and she denied any lower
gastrointestinal symptoms including diarrhea.
All the blood tests performed were normal.
Conclusion: To the best of our knowledge, this
is the first case of collagenous involvement of
the esophageal gastric junction in addition to
Yousef Usta , Denise Millstine , Fredrick Kenny
Affiliations: 1MD, Internal Medicine, Mayo Clinic Scottsdale,
13400 E. Shea Blvd. Scottsdale, AZ 85259; 2DO, Internal
Medicine, St Joseph Hospital and Medical Center, 350 West
Thomas Rd Phoenix, Arizona, 85013; 3MD, Gastroenterology,
St Joseph Hospital and Medical Center, 350 West Thomas
Rd Phoenix, Arizona, 85013.
Corresponding Author: Yousef Usta, 3302 N 7th St Apt 248.
Phoenix Arizona, 85014 USA; Ph: 6023089575; Email:
yosusta@hotmail.com
1
*********
Usta Y, Millstine D, Kenny F. Collagenous gastritis:
Gastroesophageal reflux disease in an adult patient
without collagenous colitis. International Journal of Case
Reports and Images 2013;4(12):678681.
*********
doi:10.5348/ijcri-2013-12-409-CR-3
Introduction
Collagenous gastritis, first described in 1989 [1], is a
very rare disease with only about 50 cases reported to
date [2]. This is a histologic diagnosis characterized by
thickened subepithelial collagen bands in the gastric
mucosa associated with an inflammatory infiltrates [3].
Recent case reports have started grouping cases into two
major groups with distinct clinical presentations seen
in children and adults, and adult type disease [2, 46].
The two groups are described as children or young adults
presenting with isolated collagenous gastric disease with
anemia and gastric nodularity on endoscopy and the other
distinct group includes older adults with both collagenous
gastritis (CG) and collagenous colitis (CC) associated
with chronic diarrhea and erythema on colonoscopy [6].
The incidence of CC has increased significantly over time
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Usta et al.
679
CASE REPORT
Herein, we present a 55-year-old female with a 10year history of reflux and heartburn. Her past medical
history includes iron deficiency anemia that responded
to iron supplements, osteoporosis, and a resected
acoustic neuroma 20 years before. Family history
was negative for gastrointestinal diseases. She denied
odynophagia, nausea, vomiting, or anorexia.
She
denied fever, chills, diarrhea, weight loss, hematochezia,
or melena. She denied aspirin, nonsteroidal antiinflammatory medications, alcohol abuse, or any toxic
chemical ingestion. All laboratory examinations were
normal including antithyroid antibody, antiparietal
cell antibodies, anti tissue transglutaminase, thyroid
stimulating hormone, erythrocyte sedimentation rate,
complete metabolic panel, and complete blood count.
An esophagoduodenoscopy (EGD) was performed
which showed a grossly normal esophagus and duodenum.
Grossly, the stomach had abnormal vascularity and
atrophy of the mucosa with no bleeding. Biopsies
performed on the duodenum showed no evidence of celiac
disease. Histology of the antrum of the stomach showed
evidence of chemical gastropathy. There was villiform
transformation and muscular stranding along mucosal
congestion with no acute or chronic inflammation.
Biopsy results were negative for Helicobacter Pylori, and
there was no evidence of intestinal metaplasia, abnormal
infiltrate, or neoplasia. Biopsies of the body and fundus
of the stomach showed evidence of chronic atrophic
gastritis, patchy collagenous deposits, focal lymphocytic
gastritis, severe mucosal atrophy and fibrosis, and focal
intestinal metaplasia. Some of the tissue showed an
intense lymphoplasmacytic infiltrate in the lamina propria
which in some places completely replaces the glandular
mucosa. There were a very large number of plasma cells
and beta cells seen but the majority of the cells were T
cells. There was marked deposition of collagen beneath
the surface in many of the areas particularly where there
was significant epithelial destruction seen with trichrome
DISCUSSION
Our case does not fit the recent literature that
has categorized all adult CG cases with CC including
symptoms of chronic watery diarrhea, and other systemic
diseases such as lymphocytic gastritis, celiac disease,
lymphocytic colitis, and autoimmune disorders [5]. To
date, there is no evidence of an association of CG with
a pan systemic collagenous disease. When comparing
to other described biopsy results of CG, our pathology
was very similar. As seen in our case, the collagenous
deposits showed surface epithelial injury and entrapped
inflammatory cells, and the distribution in the antrum
and body was variably patchy and diffuse [5]. She was
started on omeprazole 40 mg by mouth every twelve
hours for treatment of her GERD symptoms. Although
there seems to be no known or proven therapy for CG,
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
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CONCLUSION
To the best of our knowledge, this was the first case
of collagenous involvement of the esophageal gastric
junction in collagenous gastritis. She is the first adult
patient who presented with collagenous gastritis with
upper gastrointestinal symptoms with no manifestations
of collagenous colitis, and no lower gastrointestinal
symptoms including diarrhea which is consistent with
pediatric type categorization of the disease. Considering
the rarity of the disease, we feel that is still too premature
to categorize collagenous gastritis into two distinct adult
type and pediatric type disease. Our patients symptoms
resolved with a proton pump inhibitor, but histologically,
our patient showed no improvement on one year
esophagoduodenoscopy biopsy surveillance.
*********
Usta et al.
680
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Yousef Usta et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
REFERENCES
1. Colletti RB, Trainer TD. Collagenous gastritis.
Gastroenterology 1989 Dec;97(6):15525.
2. Brain O, Rajaguru C, Warren B, Booth J, Travis S.
Collagenous gastritis: Reports and systematic review.
Eur J Gastroenterol Hepatol 2009 Dec;21(12):1419
24.
3. Ravikumara M, Ramani P, Spray CH. Collagenous
gastritis: A case report and review. Eur J Pediatr 2007
Aug;166(8):7693.
4. Suskind D, Wahbeh G, Murray K, Christie D, Kapur
RP. Collagenous gastritis, a new spectrum of disease in
pediatric patients: Two case reports. Cases J 2009 Jun
10;2:7511.
5. Leung ST, Chandan VS, Murray JA, Wu TT. Collagenous
gastritis: Histopathologic features and association with
other gastrointestinal diseases. Am J Surg Pathol 2009
May;33(5):78898.
6. Lagorce-Pages C, Fabiani B, Bouvier R, Scoazec JY,
Durand L, Flejou JF. Collagenous gastritis: A report of
six cases. Am J Surg Pathol 2001 Sep;25(9):11749.
7. Bonderup OK, Hansen JB, Teglbjaerg PS, Christensen
LA, Fallingborg JF. Long-term budesonide treatment
of collagenous colitis: A randomised, double-blind,
placebo-controlled trial. Gut 2009 Jan;58(1):6872.
8. Freeman HJ. Complications of collagenous colitis.
World J Gastroenterol 2008 Mar 21;14(11):16435.
Author Contributions
Yousef Usta Substantial contributions to conception
and design, Acquisition of data, Analysis and
interpretation of data, Drafting the article, Revising
it critically for important intellectual content, Final
approval of the version to be published
Denise Millstine Substantial contributions to
conception and design, Acquisition of data, Analysis
and interpretation of data, Drafting the article, Revising
it critically for important intellectual content, Final
approval of the version to be published
Fredrick Kenny Substantial contributions to
conception and design, Acquisition of data, Analysis
and interpretation of data, Drafting the article, Revising
it critically for important intellectual content, Final
approval of the version to be published
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):678681.
www.ijcasereportsandimages.com
Usta et al.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
681
IJCRI 2013;4(12):682685.
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Makarawo et al.
CASE REPORT
682
OPEN ACCESS
Abstract
Introduction:
Ductal
(endometrioid)
adenocarcinoma is a rare form of prostate
cancer that most often presents with local
obstructive or irritative symptoms such as acute
urinary obstruction and hematuria, often in the
presence of a normal prostate specific antigen
(PSA) value. Hemospermia is a symptom rarely
associated with prostate malignancy. Therefore,
this was not investigated extensively in the
past, requiring only patient reassurance and
empiric antibiotic treatment. Case Report: A
56-year-old male presented with complaints of
hemospermia. His physical examination was
unremarkable and serum PSA was within normal
limits. He was, therefore, reassured and given a
course of antibiotics. However, due to persistent
hemospermia symptoms, he underwent a
flexible cystoscopy which identified a polypoid
lesion within the verumontanum of the prostatic
urethra, which upon resection was found to be
prostatic ductal adenocarcinoma. Following
an initial period of surveillance, the patient
underwent a radical prostatectomy from which
he made an uneventful recovery and remained
disease-free up to the last follow-up six months
before. Conclusion: Hemospermia is a symptom
largely considered to be benign but in some
Tafadzwa P Makarawo1, Jeffrey L OConnor1
Affiliations: 1Providence Hospital and Medical Centers,
16001 West Nine Mile, Rd, Southfield, MI.
Corresponding Author: Dr. Tafadzwa P. Makarawo, MD,
Department of Surgery, Providence Hospital and Medical
Centres, 16001 W Nine Mile Road, Southfield, Michigan,
United States, Tel: +1-248-849-3000, Fax: +1-248-8495324; Email: taffyowm@yahoo.com
Received: 05 June 2012
Accepted: 20 August 2012
Published: 01 December 2013
*********
Makarawo TP, OConnor JL. Prostatic ductal
adenocarcinoma presenting with hemospermia.
International Journal of Case Reports and Images
2013;4(11):682685.
*********
doi:10.5348/ijcri-2013-12-410-CR-4
Introduction
Prostatic ductal (endometrioid) adenocarcinoma is
a rare variant of prostate adenocarcinoma presenting
in 0.136% of all prostate cancers [1]. As with acinar
prostate adenocarcinoma, ductal adenocarcinoma
affects elderly men over the age of sixty-five. However,
in ductal adenocarcinoma, there exist some important
differences in its presentation. Whereas acinar prostate
adenocarcinoma cases are most often diagnosed through
screening by asymptomatic increases in prostate specific
antigen (PSA), ductal adenocarcinoma is known to
present with local obstructive or irritative symptoms such
as acute urinary obstruction and hematuria, often in the
presence of a normal PSA value [1].
Hemospermia is an uncommonly reported symptom
that historically did not warrant further investigation
at presentation [2]. In recent years, there has been a
growing body of evidence in support of its association
with urological malignancy [3]. We add to this body of
evidence a case of prostatic ductal adenocarcinoma
presenting with hemospermia, with only one other case
describing this association in literature [4].
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IJCRI 2013;4(12):682685.
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Makarawo et al.
683
CASE REPORT
A 56-year-old male was presented with complaints
of hemospermia. His PSA was below 0.1, urinalysis was
negative, and digital rectal examination was normal. He
was, therefore, reassured and given a course of antibiotics.
One month later, his symptoms had not resolved
despite a course of antibiotics. Flexible cystoscopy was,
therefore, performed and revealed no evidence of bladder
malignancy but noted an inflammatory-looking polyp
extending off the right portion of the verumontanum.
A transurethral resection of the abnormality
revealed histological features consistent with ductal
adenocarcinoma (Figures 1 and 2). Specifically, tall,
pseudostratified, columnar cells with abundant cytoplasm
arranged in a predominantly papillary pattern were seen
on H&E stain (Figure 1), with PSA immunohistochemistry
staining verifying the prostatic origin of the malignancy
(Figure 2) and distinguishing it from colorectal cancer
invading the prostate.
Following an initial period of active surveillance
elected by the patient, he eventually underwent a radical
prostatectomy. Final pathological evaluation of the
prostate specimen indicated a focus of Gleason score 7
(4+3) ductal adenocarcinoma with two foci of acinar
adenocarcinoma of Gleason score 8 (4+4). Surgical
margins were negative.
The patient had an uneventful postoperative recovery
and remained disease-free on follow-up visits.
DISCUSSION
Ductal adenocarcinoma has unique morphological
and clinical features. Histologically, it is characterized
by the presence of tall, pseudostratified, columnar cells
with abundant cytoplasm arranged in a papillary pattern
[1]. Typically, these lesions grow as exophytic lesions into
the urethra, most commonly around the verumontanum
[1]. The exophytic growth into the urethra accounts for
the clinical presentation of obstruction and hematuria.
Mimickers of ductal adenocarcinoma include prostatic
urethral polyps, hyperplastic benign prostate glands,
High-grade prostatic intraepithelial neoplasia (PIN),
colorectal adenocarcinoma, and papillary urothelial
carcinoma. The diagnosis of ductal adenocarcinomas
is made more challenging by the fact that they may not
always present with an elevated PSA and transrectal
ultrasound biopsy can miss these largely central tumors,
diagnosis often being made incidentally following radical
prostatectomy [1, 5].
Hemospermia is defined as the presence of fresh or
altered blood in the ejaculate [2]. The exact incidence of
hemospermia remains unknown as most ejaculates go
unnoticed during intercourse. The etiology of hemospermia
has been described as largely idiopathic, reported in as
many as 70% of all presenting cases [2], with a variety of
other less common causes described previously (Table 1).
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IJCRI 2013;4(12):682685.
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CONCLUSION
Makarawo et al.
684
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Tafadzwa Makarawo et al. 2013; This article is
distributed under the terms of Creative Commons
attribution 3.0 License which permits unrestricted use,
distribution and reproduction in any means provided
the original authors and original publisher are properly
credited. (Please see www.ijcasereportsandimages.com/
copyright-policy.php for more information.)
REFERENCES
1.
*********
Author Contributions
Tafadzwa Makarawo Substantial contributions to
conception and design, Acquisition of data Drafting the
article, Revising it critically for important intellectual
content, Final approval of the version to be published
Jeffrey OConnor Substantial contributions to
conception and design, Drafting the article, Final approval
of the version to be published
Guarantor
The corresponding author is the guarantor of submission.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):682685.
www.ijcasereportsandimages.com
Makarawo et al.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
685
IJCRI 2013;4(12):686690.
www.ijcasereportsandimages.com
Kong et al.
CASE REPORT
686
OPEN ACCESS
*********
Kong V, Naidoo L, Jeetoo D, Oosthuizen G, Laing G,
Clarke D. Chest X-Ray appearance of total opacification
of the hemithorax following central venous line insertion:
A cautionary tale. International Journal of Case Reports
and Images 2013;4(12):686690.
*********
doi:10.5348/ijcri-2013-12-411-CR-5
Introduction
Central venous line insertion is associated
with significant complications which are often
underappreciated [1]. Mechanical complications,
especially pneumothorax, are common, and injuries
to major structures (including the major vessels)
are reported to be as high as 20% [2]. A chest X-ray
post-insertion of a central venous line is commonly
performed [3]. Technical complications need to be
addressed in a timely fashion, but requires correct
interpretation of radiological findings. We report a
case of a 25-year-old male who had misinterpreted post
insertion chest radiography and a tube thoracostomy
was subsequently planned inappropriately by the
junior surgical resident.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):686690.
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Kong et al.
687
CASE REPORT
A 25-year-old male was presented who had a
laparotomy for a gastric perforation following a stab
injury. Due to poor peripheral venous access, a right
subclavian central venous line was inserted by the
anaesthetic resident intra-operatively. However, several
attempts were undertaken due to difficult positioning of
the patient. Ultrasound guidance was not utilised because
the machine was not available due to maintenance. The
central line remained patent and functional, and the
patient remained well throughout the operation. The
surgery was uneventful and he was discharged to the
general ward.
A routine post procedural chest X-ray (Figure 1)
was done immediately, but this was not reviewed by
the surgical resident until 12 hours later. The chest
X-ray appeared to show that the central venous line
entered the right subclavian vein, and then traversed
the brachiocephalic vein before re-entering the left
subclavian vein. There was no pneumothorax or
hemothorax noted. The patient had no complains and
was not in distress. Therefore, it was decided that the
central line should be removed as soon as the patient
could tolerate orally, likely to be the following day.
However, he began complaining of abdominal pain
and increasing dull pain of the right chest throughout
the night. He had poor respiratory effort, refused chest
physiotherapy and was unable to mobilize out of the bed
due to ongoing pain.
On the following morning, he was noted to be in mild
respiratory distress, with a respiratory rate of 24/min,
shallow pattern, SpO2: 90% (FiO2: 100% via a Hudson
mask with reservoir bag) with marked dullness and
reduce air entry on the right chest. His heart rate was
90/min and blood pressure 105/70 mmHg. Laboratory
results were: hemoglobin 11.5 g/dL, white blood cell
15x103/L. A repeat chest X-ray (Figure 2) showed a
complete opacification of the right hemithorax.
The junior resident who reviewed the chest X-ray
felt it was due to a delayed presentation of iatrogenic
hemothorax related to the insertion of the central
line, and a tube thoracostomy was planned. However,
prior to tube thoracostomy, the patient was eventually
reviewed by the duty attending trauma surgeon who
advised on an emergency computed tomography (CT)
scan of the thorax prior to proceeding. The CT scan
(Figure 3) revealed a complete collapse of the right
lung, with a large mucus plug obstructing the right main
bronchus. An emergency awake flexible bronchoscopy
was performed (under conscious sedation), and a large
amount of mucus was removed using suction. Clinical
condition of the patinent improved immediately. A
subsequent chest X-ray, six hours after the procedure,
showed a complete expansion of the right lung. Following
this, his analgesia regime was further escalated, and he
was treated with aggressive chest physiotherapy, which
was well tolerated. He had an uneventful recovery,
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IJCRI 2013;4(12):686690.
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DISCUSSION
Central venous line insertion is a common procedure
which is frequently performed by hospital doctors, but
is associated with significant complications. Overall
complication rate varies, but it has been reported to be
as high as 20% [1]. Complications are generally divided
into mechanical, thromboembolic and infectious. Of the
mechanical complications, vascular and lung injuries
(hemothorax, pneumothorax) are the most common [4].
The choice of insertion site, however, is often dictated
by the operator, usually dependent on the individual level
of skills and experience [4]. Malpositioning has been
reported in up to 15%, even when inserted by experienced
clinicians [3]. Current evidence appears to suggest that
the subclavian route is more likely to be associated with
malposition in comparison with the internal jugular route
[1]. However, there is no evidence to suggest any difference
in the incidence of hemothorax, pneumothorax and vessel
occlusion between the two different routes [1]. It is also
important to stress that although free venous back flow
must be present after insertion, it does not absolutely
exclude malpositioning or even subsequent migration
[5]. Whilst insertion under ultrasound guidance has
become increasingly common, the traditional anatomical
landmark technique continues to be used frequently [6],
especially in developing countries where the resources
and facilities may be limited.
Death following significant subclavian artery injury
has been reported [7]. The artery is particularly vulnerable
due to its close proximity to the subclavian vein.
Furthermore, this complication appears to be directly
proportional to the number of attempts undertaken. More
than three attempts are associated with a complication
rate of approximately 50% [8].
Several important issues are highlighted in this unusual
case. Firstly, the initial placement position was clearly
incorrect, but this was only recognised 12 hours after
the insertion. Also, because the line appeared functional,
this initially gave a false sense of security. Although some
literatures suggest that chest radiography may not be
absolutely necessary in all cases, especially when inserted
by experienced operators, most clinicians continue to do
so, especially when difficulties are encountered or when
multiple attempts have been made [3]. In the context of
central line insertion, the appearance of pneumothorax
or hemothorax on the chest X-ray is usually obvious,
but erroneous interpretation (as in this case) almost
leads to unnecessary further procedures (such as tube
thoracostomy) being carried out. Whilst the appearance
of total opacification of the hemithorax could be caused
by hemothorax (secondary to vascular injury), another
important diagnosis to consider is complete pulmonary
Kong et al.
688
CONCLUSION
Central venous line insertion is associated with
significant morbidity, and is often under-appreciated.
Although vascular and lung injuries are relatively
common, chest radiographic findings may be easily
misinterpreted. Whilst clinicians must always remain
vigilant of potential complications, it is equally
important to remain open to alternative diagnoses,
especially if the overall clinical picture does not seem
to fit together.
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*********
Author Contributions
Victor Kong Substantial contributions to conception and
design, Acquisition of data, Analysis and interpretation
of data, Drafting the article, Revising it critically for
important intellectual content, Final approval of the
version to be published
Leah Naidoo Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
Damon Jeetoo Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
George Oosthuizen Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
Grant Laing Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
Damian Clarke Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
Kong et al.
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Victor Kong et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
REFERENCES
1.
2.
3.
689
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Kong et al.
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690
IJCRI 2013;4(12):691694.
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Sahoo et al.
CASE REPORT
691
OPEN ACCESS
Abstract
Introduction: Intussusception is uncommon in
adults compared with the pediatric population.
Unlike its pediatric counterpart, intussusception
in adults is associated with obvious pathology.
Small bowel intussusceptions are much less
common than the ileocolic type, with jejunoileal
intussusceptions being amongst the rarest.
There are no reports of double intussusception
in literature. We report a first case of double
telescoping intussusception in an adult where the
proximal ileum (intussusceptum) is telescoping
into distal ileum (intussuscipiens) and this as a
whole is again telescoping into terminal ileum.
Case Report: A 46-year-old woman came to the
emergency ward with a three-day history of pain
abdomen, distension and multiple episodes of
bilious vomiting. The abdomen was distended
and there was rebound tenderness on palpation.
There was a palpable mass just below the
umbilicus. Plain X-ray abdomen showed multiple
air fluid levels. Ultrasonography of abdomen
showed dilated bowel loops, with target sign
giving an impression of ileoileal intussusception.
Intraoperatively double telescoping ileoileal
intussusception was present one foot proximal to
Sahoo Manash Ranjan1, Gowda Manoj Srinivas2, Kaladagi
Raghavendra Mohan2
Affiliations: 1MBBS, MS (General Surgery), Associate
Professor, Department of General Surgery, SCB Medical
college and Hospital, Cuttack, Odisha, India; 2MBBS, MS
(General Surgery), Post Graduate, Department of General
Surgery, SCB Medical college and Hospital, Cuttack,
Odisha, India.
Corresponding Author: Manash Ranjan Sahoo, Associate
Professor Surgery, SCB Medical College and Hospital,
Cuttack, Odisha, India - 753007; Ph: +919937025779;
Email: manash67@gmail.com
Received: 20 February 2013
Accepted: 25 April 2013
Published: 01 December 2013
Laparotomy,
*********
Sahoo MR, Gowda MS, Kaladagi RM. Idiopathic double
telescoping intussusception in an adult. International
Journal of Case Reports and Images 2013;4(12):691694.
*********
doi:10.5348/ijcri-2013-12-412-CR-6
Introduction
Intussusception was first reported in 1674 by Barbette
from Amsterdam and further presented in a detailed
report in 1789 by John Hunter as introssusception.
Intussusception is defined as the telescoping of a
segment of the gastrointestinal tract (intussusceptum)
into an adjacent one (intussuscipiens). This may lead
to intestinal obstruction and loss of blood supply to
the area. More rarely, the intussusceptum may become
strangulated, necrotic, and gangrenous and lead to
sepsis or death. Intussusception is uncommon in adults
compared with the pediatric population. It is estimated
that only 5% of all intussusceptions occur in adults and
approximately 5% of bowel obstructions in adults are the
result of intussusception [1]. Clinically, intussusception
in adults often presents with nonspecific symptoms such
as abdominal pain, nausea, diarrhea, and rectal bleeding.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
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Sahoo et al.
692
CASE REPORT
A 46-year-old woman came to the emergency ward
with a three-day history of abdomen pain, distension
and multiple episodes of bilious vomiting. She also
had complaint of constipation for last two days. There
was no significant past medical history. On physical
examination, she had tachycardia and hypotension;
temperature was normal. The abdomen was distended
and there was rebound tenderness on palpation. There
was a palpable mass just below the umbilicus. Laboratory
investigations showed leukocytosis. Liver function tests
were normal. Plain X-ray abdomen showed multiple air
fluid levels. Ultrasonography of abdomen showed dilated
bowel loops, with target sign giving an impression of ileoileal intussusception. With all these findings suggestive
of acute intestinal obstruction, patient was planned for
urgent exploratory laparotomy. Intraoperatively double
telescoping ileoileal intussusception was present one
foot proximal to ileocecal junction (Figure 1). The bowel
proximal to this area was dilated and the intussusceptum
was gangrenous. There was no intramural mass or any
other type of lead point of intussusceptions (Figure 2).
There was no free fluid and inter bowel adhesions. The
resection of the ileal segment bearing the intussusception
with end ileostomy was done. Even though primary
anastomosis is the standard after resection in this case,
end ileostomy was done due to poor general condition
of the patient and gangrenous bowel segment. The
histopathological study of the specimen did not reveal
any pathology. Patient recovered well in postoperative
period and was discharged on 10th postoperative day.
Patient was doing well during follow-up, and after three
months ileo-ascending anastomosis was done.
DISCUSSION
Adult intussusception is an uncommon clinical
entity encountered by surgeons. Common sites of
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CONCLUSION
Intussusception in adults is very rare and has an
identifiable cause in 80% of cases. We report an unusual
cause of small bowel obstruction in an adult patient,
secondary to double telescoping intussusception with
no lead point. Double telescoping intussusception is
the condition where there will be a proximal segment
intussuscepting into the distal segment and this whole
thing will again intussuscept into the still farther segment
of intestine. Since most cases are secondary to lead point
surgery remains the mainstay of management.
*********
Author Contributions
Sahoo et al.
693
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Manash Ranjan Sahoo et al. 2013; This article is
distributed under the terms of Creative Commons
attribution 3.0 License which permits unrestricted use,
distribution and reproduction in any means provided
the original authors and original publisher are properly
credited. (Please see www.ijcasereportsandimages.com/
copyright-policy.php for more information.)
REFERENCES
1. Zubaidi A, Al-Saif F, Silverman R. Adult
intussusception: A retrospective review. Dis Colon
Rectum 2006 Oct;49(10):15461.
2. Azar T, Berger DL. Adult intussusception. Ann Surg
1997 Aug;226(2):1348.
3. Eisen LK, Cunningham JD, Aufses AH Jr.
Intussusception in adults: Institutional review. J Am
Coll Surg 1999 Apr;188(4):3905.
4. Begos DG, Sandor A, Modlin IM. The diagnosis and
management of adult intussusception. Am J Surg
1997 Feb;173(2):8894.
5. Weilbaecher D, Bolin JA, Hearn D, Ogden W 2nd.
Intussusception in adults. Review of 160 cases. Am J
Surg 1971 May;121(5):5315.
6. Sachs M, Encke A. Entero-enteral invagination of the
small intestine in adults. A rare cause of uncertain
abdomen. Langenbecks Arch Chir 1993;378(5):288
91.
7. Schiud F, van Gansbeke D, Ansey J. Intussusception
in adults--report of 3 cases. Acta Chir Belg 1985 JanFeb;85(1):5560.
8. Nagorney DM, Sarr MG, McIlrath DC. Surgical
management of intussusception in the adult. Ann
Surg 1981 Feb;193(2):2306.
9. Ishii M, Teramoto S, Yakabe M, et al. Small intestinal
intussusceptions caused by percutaneous endoscopic
jejunostomy tube placement. J Am Geriatr Soc 2007
Dec;55(12):20934.
10. Takeuchi K, Tsuzuki Y, Ando T, et al. The diagnosis
and treatment of adult intussusception. J Clin
Gastroenterol 2003 Jan;36(1):1821.
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IJCRI 2013;4(12):691694.
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Sahoo et al.
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694
IJCRI 2013;4(12):695697.
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Dutta et al.
695
Abstract
Introduction: Elephantiasic pretibial myxedema
is a rare manifestation of autoimmune
thyroid disease. Herein, we report a case of
elephantiasic pretibial myxedema. Case Report:
A 45-year-old male presented with bilateral firm
confluent pretibial polypoid hyperpigmented
nodular and plaque lesions. He had a history of
thyroidectomy for toxic goitre in the past and
was not on any thyroxine replacement therapy.
On examination, he had dry skin, myxedematous
appearance and grade 3 clubbing of fingers.
Investigations
revealed
elevated
thyroid
stimulating hormone and thyroid stimulating
hormone receptor stimulating antibody levels.
Histopathology of the skin lesion was suggestive
of pretibial myxedema.
The patient was
started with thyroxine replacement and steroid
occlusive dressing over lesions. His condition
was improved remarkably with treatment.
Conclusion: Elephantiasic pretibial myxedema
is rare. This condition should be recognized
promptly and should be treated with steroid,
Pinaki Dutta1, Viral N Shah2
Affiliations: 1DM, Assistant Professor, Department of
Endocrinology, 4th Floor, F Block, Nehru Hospital, Post
Graduate Institute of Medical Education and Research,
Sector 12, Chandigarh, India; 2DM, Department of
Endocrinology, Post Graduate Institute of Medical Education
and Research, Sector-12, Chandigarh, India.
Corresponding Author: Dr. Pinaki Dutta, DM, Assistant
Professor, Department of Endocrinology, 4th Floor, F
Block, Nehru Hospital, Post Graduate Institute of Medical
Education and Research, Sector 12, Chandigarh, India 160012; Ph: +91-172-2756584; +91-935711477; Fax: +910172-2747944; Email: pinaki_dutta@hotmail.com
Received: 08 February 2013
Accepted: 27 April 2013
Published: 01 December 2013
*********
Dutta P, Shah VN. Pretibial myxedema mimicking
elephantiasis. International Journal of Case Reports
and Images 2013;4(12):695697.
*********
doi:10.5348/ijcri-2013-12-413-CR-7
Introduction
Pretibial myxedema (PTM) is an immune mediated
localized form of thyroid associated dermopathy often
found in patients with autoimmune thyroid disease,
particularly Graves diseases [1]. Rarely, it is also
seen in patients with hypothyroidism [2]. The skin
lesions are diffuse, non-pitting in majority. However,
plaques and nodular forms have also been describe in
literature. Elephantiasis form of (PTM) which is known
as elephantiasis nostra verrucosa (ENV) is extreme rare
form which is found late in the course of Graves disease
[3]. We describe a case who remained hypothyroid for a
long period of time, eventually developed elephantiasic
PTM.
CASE REPORT
A 45-year-oldmale presented with large irregular
nodular swelling over anterior aspects of tibia bilaterally.
These swellings were gradually increasing in size for past
two years. He had a history of thyroidectomy for large
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IJCRI 2013;4(12):695697.
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Dutta et al.
696
DISCUSSION
Pretibial myxedema is an infrequent manifestation
of autoimmune thyroid disease as a result of immune
mediated phenomenon [1]. About 0.54.3% of patients
with a history of thyrotoxicosis have thyroid dermopathy,
and 15% of patients with severe Graves ophthalmopathy
have this cutaneous manifestation [4]. According to
one study, PTM was also found with frequency of 17.1%
in Hashimotos thyroiditis, and 6.5% in idiopathic
hypothyroidism [5]. The elephantiasic pretibial
myxedema constitutes less than 1% of PTM. Our patient
had toxic nodular goitre and underwent thyroidectomy,
subsequent hypothyroidism and eventually developed
PTM.
Classically, histopathology of these lesions shows
increase in levels of glycosaminoglycan (GAG) in the
reticular dermis with excess increase in hyaluronic acid
concentration [6]. The proposed mechanism for this is
thought to be due to fibroblast stimulation by thyroidstimulating hormone receptor (TSH-R) antibodies [6,
7]. Histopathological findings in our case is same as
describe in literature and the level of TSH-R stimulating
antibodies was also elevated suggesting the role of TSH-R
stimulating antibodies in fibroblast proliferation and
synthesis of GAG.
The most commonly describe therapy for PTM is
tight occlusive steroid dressing [6] which has also been
given in our case. The response rate varies with topical
steroid occlusive dressing is 26-50%. Our case responded
dramatically with local steroid as well as thyroxine
replacement.
CONCLUSION
Elephantiasic pretibial myxedema is a rare disease.
This condition should be recognized promptly and should
be treated with steroid occlusive dressing and treatment
of underlying thyroid disease.
ACKNOWLEDGEMENTS
We are grateful to Department of Photography
(Mr. Brij Lal and Mr. Abijeet) for the taking patient
photographs. We also thank Mr. Prakamya Gupta for
manuscript editing.
Figure 2: Biopsy of one of the nodular skin lesions showing
separation of collagen fibers, extensive deposition and stellate
fibroblasts (H&E stain, x400).
*********
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):695697.
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Author Contributions
Pinaki Dutta Substantial contributions to conception
and design, Acquisition of data, Analysis and
interpretation of data, Drafting the article, Revising
it critically for important intellectual content, Final
approval of the version to be published
Viral N Shah Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Pinaki Dutta et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
Dutta et al.
697
REFERENCES
1. Beierwaltes WH. Clinical correlation of pretibial
myxedema with malignant exophthalmos. Ann Intern
Med 1954 May;40(5):96884.
2. Reed Larsen, P, Davies TF, Hay IA. The thyroid gland
in Williams Textbook of Endocrinology 9th Edition
WB Saunders 1998:390498.
3. Humbert P, Dupond JL, Carbillet JP. Pretibial
myxedema: An overlapping clinical manifestation of
autoimmune thyroid disease. The American journal
of medicine 1987 Dec;83(6):11701.
4. Kriss JP. Pathogenesis and treatment of pretibial
myxedema. Endocrinol Metab Clin North Am 1987
Jun;16(2):40915.
5. Salvi M, De Chiara F, Gardini E, et al.
Echocardiographic diagnosis of pretibial myxedema
in patients with autoimmune thyroid disease. Eur J
Endocrinol 1994 Aug;131(2):1139.
6. Schwartz KM, Fatourechi V, Ahmed DD, Pond GR.
Dermopathy of Graves disease (pretibial myxedema):
Long-term outcome. J Clin Endocrinol Metab 2002
Feb;87(2):4386.
7. Fatourechi V, Ahmed DD, Schwartz KM. Thyroid
acropachy: Report of 40 patients treated at a single
institution in a 26-year period. J Clin Endocrinol
Metab 2002 Dec;87(12):543541.
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Khattab et al.
CASE REPORT
698
OPEN ACCESS
Abstract
Introduction:
Currently,
amphetamine
abuse is a widespread problem. While the
use of recreational drugs is more common in
young populations, drug abuse within elderly
populations is increasing, though largely
unrecognized. The exact nature and dose of
recreational drugs are difficult to identify when
a patient presents requiring urgent care, notably
amphetamine-induced acute coronary syndrome
(ACS). Case Report: A 54-year-old male smoker
presented with classical anginal chest pain after
using amphetamines. Electrocardiography (ECG)
showed T-wave inversion in leads I, aVL, and
V4-V6. Laboratory data revealed total creatine
kinase (CK) level 1759 IU/L, CK-MB 87 IU/L,
and a troponin-I of 13.38 ng/mL. A diagnosis
of non-ST elevation myocardial infarction
(NSTEMI) was made. The emergency room
physician commenced the NSTEMI protocol,
but the patient experienced worsening chest
pain unresponsive to nitrates or morphine, with
no change in the ECG pattern. The cardiologist
decided to proceed with percutaneous coronary
intervention. Coronary angiography revealed
100% stenosis in the proximal portion of the left
circumflex artery, with a large clot burden. The
artery was stented with excellent results. Later
testing showed a positive urine toxicology screen
Eyad Khattab , Asaad Shujaa
Affiliations: 1MD, MPH, Demonstrator, Department of
Emergency Medicine, King Khalid University Hospital, King
Saud University, Riyadh, Saudi Arabia. Resident, Department
of Internal Medicine, University of Toledo Medical Center,
Toledo, Ohio, United States of America.
Corresponding Author: Eyad Khattab, P.O. Box 7805,
Riyadh, SAUDI ARABIA 11472; Ph:+966-503327350; Fax:
+966-14672439; Email: eyad.khattab@yahoo.com
1
*********
Khattab E, Shujaa A. Amphetamine abuse and
acute thrombosis of left circumflex coronary artery.
International Journal of Case Reports and Images
2013;4(12):698701.
*********
doi:10.5348/ijcri-2013-12-414-CR-8
Introduction
Currently, amphetamine abuse is a universal problem.
Drug abuse is more common in young population.
However, the use of recreational drugs by elderly
populations is largely unrecognized and ignored, but
increasing [1]. The prevalence of illicit drug use in general
populations is difficult to identify due to the covert nature
of the activity, especially in one of the most conservative
societies, Saudi Arabia, which is tightly surrounded by
tradition and religious conservatism. Furthermore, the
exact nature and dose of recreational drugs are difficult to
establish when a patient presents need urgent care.
As the name suggests, sympathomimetic drugs such
as amphetamine work by activating the sympathetic
nervous system. As a result, heart rate increases and the
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
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Khattab et al.
699
CASE REPORT
A 54-year-old male, known to have bronchial asthma
(mild) well controlled with albuterol inhaler, was brought
by his family to our emergency medicine department three
hours after the onset of pressure like chest pain, which was
central and radiated to the left arm. The patient reported
taking an oral dose of amphetamine about seven hours
before the presentation. The patient told that the chest
pain was neither decreased nor increased by anything.
The pain was associated with shortness of breath, nausea
and vomiting. The patient had no history of palpitation,
cough, wheezing, abdominal pain or back pain. He had no
history of similar symptoms before, such as ischemic heart
disease, diabetes mellitus, hypertension, hyperlipidemia,
or atrial fibrillation. He had no family history of similar
symptoms or coronary artery disease. He was not on
any medication. He was not allergic to anything. He
smoked one pack of cigarettes per day over the last 10
years. He used amphetamine tablets occasionally and
had no history of drinking alcohol. At the time of arrival
to the hospital, his blood pressure was 137/94 mmHg,
heart rate was 77 beat per minute, respiratory rate was
17 breaths per minute, oral temperature was 37.2C and
oxygen saturation was 99% on room air. Generally, he
appeared in severe respiratory distress, but alert and
oriented. Cardiovascular examination revealed first and
second heart sounds were within normal limits, point
of maximum impulse was not displaced, and no rub,
murmur or gallop sounds were exhibited. His lungs were
clear to auscultation bilaterally.
A 12-lead electrocardiogram showed a regular sinus
rhythm (79/min) and T-wave inverted in lead I, aVL, (V4V6). The patient had no previous ECG, echo, stress, or
lipid profile reports for comparison Because of his lack
of prior related symptoms. Laboratory data showed total
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DISCUSSION
Cardiovascular problems related to amphetamine use
are common. However, most of the cases and population
studies in literature concerning drug-induced acute
myocardial infarction (AMI) have identified young
male patients presenting with elevated ST levels and
vasospasms in the left anterior descending artery and/or
right coronary artery. What is unique in the present case
is the age of the patient, the apparent contributing factors
of the AMI, and the location. To our knowledge, there are
no publications of amphetamine-induced NSTEMI with
selective thrombosis of the LCX and very little focus on
oral amphetamine abuse in older adults. According to
a population-based observational study, amphetamine
abuse contributes to AMI among persons aged 18-44
years, but does not address the incidence among those
older than 44 years [5]. Another study of the frequency
of acute coronary symptoms due to amphetamine use
among patients aged between 32-48 years [6]. Thus, the
age of our 54-year-old patient is unique among literature
regarding coronary symptoms due to amphetamine use.
Moreover, the lack of vasospasms and the presence
of thrombosis in our case are unusual in literature. The
mechanism of drug-induced MI is usually associated with
coronary spasms [3]. One exception in literature is the
case of a young male patient who presented with acute
MI and tested positive for amphetamine. An arteriogram
showed acute thrombosis, but early PCI did not help and
the patient died later [3]. Our patient was fortunate to
have survived. Additionally, many cases of drug-induced
AMI show ST-segment elevation, for instance a young
male who presented to the emergency room with chest
pain after taking Captagon and showed ST elevation due
to a narrowing in the proximal portion of the left anterior
descending (LAD) artery [7]. However, LCX occlusions, as
in our case, do not usually result in elevated ST segments
[8].
While LCX occlusions are usually NSTEMI, it is rare
for the cardiovascular problems among amphetamine
users to be located in the LCX artery in the first place.
In contrast, cases of narrowing in the LAD artery [7] and
the right coronary artery due to amphetamine use are not
unusual [9]. In a rare case, screening of the LCX artery
showed thrombosis, but it was secondary to thrombosis
of the larger LAD artery [10]. The underlying reason for
the rarity of amphetamine-induced LCX thrombosis is
speculative, but perhaps it is related to age and pre-existing
cardiovascular issues. Exposure to amphetamine may
trigger AMI thrombotic occlusion at the site of multiple
Khattab et al.
700
CONCLUSION
This case demonstrates why asking questions about
stimulant drugs use is crucial with anginal chest pain
presentation, even for an older patient. Drug abuse
is not only for young adults and men but middle aged,
older persons and women are also susceptible, and can
present unique amphetamine-induced acute coronary
syndrome etiology. In some cases, as in this present case,
sympathomimetic drugs may cause myocardial ischemia
due to blood clot formation.
*********
Acknowledgements
Acknowledgment is owed to Dr. Sabry Elmalah for his
help diagnosing and treating the patient and to Clayton
Chiarelott for proofreading and editing the article.
Author Contributions
Eyad Khattab Conception and design, Acquisition of
data, Analysis and interpretation of data, Drafting the
article, Critical revision of the article, Final approval of
the version to be published
Asaad Shujaa Conception and design, Acquisition of
data, Drafting the article, Final approval of the version to
be published
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Eyad Khattab et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
REFERENCES
1.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):698701.
www.ijcasereportsandimages.com
drug users in northwest England. Ageing Society
2007;27:799810.
2. From AM, Best PJ, Lennon RJ, Rihal CS, Prasad A.
Acute myocardial infarction due to left circumflex
artery occlusion and significance of ST-segment
elevation. Am J Cardiol 2010 Oct 15;106(8):1081-5.
3. El Menyar AA. Drug-induced myocardial infarction
secondary to coronary artery spasm in teenagers and
young adults. Journal of Postgraduate Medicine 2006
Jan-Mar;52(1):516.
4. Heusch G. Heart rate in the pathophysiology of
coronary blood flow and myocardial ischaemia:
Benefit from selective bradycardic agents. British
Journal of Pharmacology 2008 Apr;153(8):1589-601.
5. Westover AN, Nakonezny PA, Haley RW. Acute
myocardial infarction in young adults who abuse
amphetamines. Drug Alcohol Depend 2008 Jul
1;96(1-2):4956.
6. Turnipseed SD, Richards JR, Kirk JD, Diercks
DB, Amsterdam EA. Frequency of acute coronary
Khattab et al.
701
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IJCRI 2013;4(12):702704.
www.ijcasereportsandimages.com
Kumar et al.
702
Abstract
doi:10.5348/ijcri-2013-12-415-CR-9
Introduction:
Hyperthyroidism
causing
pseudotumor cerebri is very rare in children. Case
Report: We are reporting a case of a 17-year-old
girl with uncontrolled Graves disease presenting
with pseudotumor cerebri (PTC). Patient initially
did not respond to common management of
PTC with therapeutic lumbar puncture and
acetazolamide, but improved after treating
hyperthyroidism by radioablation therapy.
Conclusion: We emphasize that uncontrolled
hyperthyroidism should be suspected as a
secondary cause of pseudotumor cerebri in
children and treated aggressively to avoid its
dreaded complications.
Keywords: Pseudotumor cerebri (PTC), Benign
intracranial
hypertension
(BIH),
Graves
disease, Papilledema, Radioablation therapy,
Hyperthyroidism
*********
Kumar A, Kilaikode S, Saenger P. Pseudotumor cerebri
caused by uncontrolled Graves disease in an adolescent.
International Journal of Case Reports and Images
2013;4(12):702704.
*********
Ashutosh Kumar , Sasikumar Kilaikode1, Paul Saenger2
Affiliations: 1MD, Resident, Pediatrics, The Brooklyn Hospital
Center, Brooklyn, New York, USA; 2MD, Attending, Pediatric
Endocrinology, The Brooklyn Hospital Center, Brooklyn,
New York, USA.
Corresponding Author: Sasikumar Kilaikode, 539 85th Street
Apt 1C, Brooklyn, New York, USA. 11209; Ph: 6466249458;
Email: sak9072@nyp.org
1
Introduction
Pseudotumor cerebri (PTC), also known as benign
intracranial hypertension (BIH), is a syndrome of
increased intracranial pressure in the absence of
change in ventricular size, with normal cerebrospinal
fluid (CSF) analysis and normal neuroimaging [1].
Presenting symptoms are similar to those associated with
hydrocephalus and typically include headache, vomiting,
and changes in vision. Neurological examination is
typically normal except papilledema which is almost
always noted in adults, but may be inconsistent in
children and adolescents.
Pseudotumor cerebri can be idiopathic or secondary
to numerous underlying causes, including endocrine,
rheumatologic, immunologic conditions and medications
[1]. Both hypothyroidism and hyperthyroidism have been
reported to cause PTC. Hyperthyroidism causing PTC is
rare. There are only a few case reports of PTC caused by
hyperthyroidism in adults and two case reports in the
pediatric population [2, 3].
We are reporting a case of a 17-year-old girl with
uncontrolled Graves disease presenting with PTC.
CASE REPORT
A 17-year-old African-American female with
congenital HIV controlled on anti-retroviral therapy
(Complera, Gilead Sciences, Inc. Foster City, CA, USA),
mild intermittent asthma and Graves disease diagnosed
five years ago, managed by methimazole with poor
compliance, presented with one day history of severe
pounding bi-frontal headache unrelieved by analgesics.
There was no history of vomiting, fever, visual problems,
weakness of extremities, seizures, neck stiffness, or
trauma. On examination, patient had tachycardia and
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
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Kumar et al.
Prior to
Radioablation
therapy
4 weeks
after
therapy
6 weeks
after
therapy
TSH (IU/mL)
<0.1
<0.1
<0.1
Total T4(g/dL)
17.7
12
10.9
Free T4 ( ng/dL)
2.5
1.7
1.4
Total T3 (ng/dL)
310
230
180
703
CONCLUSION
We would like to emphasize that uncontrolled Graves
disease should be considered as a rare secondary cause
of pseudotumor cerebri in children. Hyperthyroidism
should be treated aggressively to reduce the complications
associated with pseudotumor cerebri, most notably
permanent visual impairment.
*********
Author Contributions
Ashutosh Kumar Conception and design, Acquisition
of data, Analysis and interpretation of data, Drafting the
article, Critical revision of the article, Final approval of
the version to be published
Sasikumar Kilaikode Conception and design,
Acquisition of data, Analysis and interpretation of data,
Drafting the article, Critical revision of the article, Final
approval of the version to be published
Paul Saenger Conception and design, Acquisition of
data, Analysis and interpretation of data, Drafting the
article, Critical revision of the article, Final approval of
the version to be published
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
DISCUSSION
Hyperthyroidism causing PTC is rare in children
with only 2 reported cases [2, 3]. In one report,
hyperthyroidism was associated with hypovitaminosis A.
We excluded other causes of raised intracranial
pressure in our patient and confirmed the diagnosis of
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
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REFERENCES
1. Rudolph CD, Rudolph AM, Lister GE, First LR,
Gershon AA. Rudolphs Pediatrics, 22nd Ed. New
York: McGraw-Hill Medical; 2011. P.2176.
2. Merkenschlager A, Ehrt O, Mller-Felber W, Schmidt
H, Bernhard MK. Reversible benign intracranial
hypertension in a child with hyperthyroidism. J
Pediatr Endocrinol Metab 2008 Nov;21(11):10991.
3. Roos RA, Van der Blij JF. Pseudotumor
cerebri associated with hypovitaminosi A and
hyperthyroidism. Dev Med Child Neurol 1985
Apr;27(2):2468.
Kumar et al.
4.
704
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Tevethia et al.
CASE REPORT
705
OPEN ACCESS
*********
Tevethia HV, Baskaran S, Kadavanu TM, Panchbhaya
RM, Dan S, Siva PK. IGastric malignancy presenting as
adrenal insufficiency: A case report. International Journal
of Case Reports and Images 2013;4(12):705708.
*********
doi:10.5348/ijcri-2013-12-416-CR-10
Introduction
Carcinomas in the lung, breast, and lymphomas are
known to metastasize to the adrenal gland [1]. However,
in most cases they do not attain clinical significance
since these are diagnosed at autopsy. We report a case of
adrenal insufficiency due to bilateral adrenal metastases
as the presenting manifestation of gastric carcinoma.
CASE REPORT
A 72-year old male, without any known comorbidities,
was admitted to our hospital due to altered mental
status, weakness, anorexia, hematemesis and urinary
incontinence for three days. He complained of abdominal
distension and dyspepsia for the last two years. There was
a loss of appetite and weight of about 10 kg. At the time
of admission to hospital, physical examination revealed
blood pressure 110/80 mmHg, tachycardia 120 bpm
with no other specific systemic finding. The patient was
severely dehydrated, presented with decreased reflexes
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
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Tevethia et al.
706
DISCUSSION
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):705708.
www.ijcasereportsandimages.com
Tevethia et al.
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Harsh Vardhan Tevethia et al. 2013; This article
is distributed under the terms of Creative Commons
attribution 3.0 License which permits unrestricted use,
distribution and reproduction in any means provided
the original authors and original publisher are properly
credited. (Please see www.ijcasereportsandimages.com/
copyright-policy.php for more information.)
REFERENCES
1.
2.
3.
4.
5.
CONCLUSION
6.
*********
7.
8.
Author Contributions
Harsh Vardhan Tevethia Acquisition of data, Critical
revision of the article, Final approval of the version to be
published
Bhaskaran S Conception and design, Critical revision of
the article, Final approval of the version to be published
Kadavanu Tony Mathew Conception and design,
Acquisition of data, Drafting the article, Critical revision
of the article, Final approval of the version to be published
Panchbhaya Riyaz Analysis and interpretation of data,
Drafting the article, Final approval of the version to be
published
Sunny DAN Conception and design, Drafting the
article, Critical revision of the article, Final approval of
the version to be published
SIva P K Conception and design, Critical revision of the
article, Final approval of the version to be published
707
9.
10.
11.
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Tevethia et al.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
708
IJCRI 2013;4(12):709713.
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Mohan et al.
CASE REPORT
709
OPEN ACCESS
Abstract
Introduction: Talon cusp is a well delineated
accessory cusp projecting from the cingulum
area of an anterior tooth. It is named so due to
its resemblance with eagles talon, which is the
shape of eagles claw when hooked on to its prey.
Various alterations in shape in talons cusp may
be present like it may be either sharp or spiked,
teat like or may have rounded and smooth tips.
Only two cases have been reported in Indian
population till date. Case Report: Hereby, we
report a unique case of talons cusp in a fouryear-old boy. Conclusion: The occurrence of
talons cusp in mandibular primary dentition is
extremely rare.
Keywords: Cingulum, Accessory, Talon cusp
*********
*********
doi:10.5348/ijcri-2013-12-417-CR-11
Introduction
Talon cusp is a rare dental anomaly, which originates
during the morphodifferentiation stage of tooth
development. It is defined as, an additional cusp that
predominantly projects from the lingual surface of
primary or permanent anterior teeth, is morphologically
well delineated and extends at least half the distance from
cementoenamel junction to incisal edge [1]. Although
it affects both the primary and permanent dentition
but permanent dentition is found to be involved thrice
more commonly than the primary dentition. Several
studies have reported a male and female ratio to be 2:1
[2, 3]. Talon cusp is not entirely innocuous and may
pose difficulties like predisposition to caries, occlusal
interferences and unpleasant esthetics [4]. This article
focusses on an extremely rare case presenting with talon
cusp in mandibular primary dentition.
CASE REPORT
A four-year-old boy was reported to the outpatient
department with a chief complaint of decaying of right
lower back tooth region since 12 months. There was no
remarkable medical or dental history. He was apparently
healthy child born to non-consanguineous parents at full
term. Nothing significant findings were elicited in general
physical examination. Intraoral examination revealed
mixed dentition with good oral hygiene. Dental caries was
present in right lower back teeth (in relation to 73, 74).
Apart from this, an extra cusp was seen projecting from
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):709713.
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Mohan et al.
710
DISCUSSION
More than two centuries ago, William Mitchel
described accessory cusp on the lingual surface of
permanent upper central incisor of a woman and described
it as, process of horn like shape curving from the base
downwards to the cutting edge. In 1972, Mellor and Ripa
named it talon cusp owing to its close resemblance with
an eagles talon [3]. Since then , many names have been
used for this dysmorphic disturbance of teeth, namely,
dens evaginatus, supernumerary cusp, horn, hyperplastic
cingulum, evaginated odontome, cusped cingulum,
accessory cusp and supernumerary lingual tubercle [5].
The usage of multiple terms added up to confusion which
was cleared when Mader suggested that the term talon
cusp should be reserved only for those anomalous cusp
that prominently projected from the lingual surface of
a succedaneous tooth, that are morphologically well
delineated and extend at least half the distance from
cementoenamel junction to the incisal edge. Smaller cusp
like projections in the cingulum should be referred to as
enlarged or prominent cingulum [6].
The prevalence of talon cusp in primary dentition is
extremely low as concluded from the review of literature
from 1977 to 2013 (Table 1). Till date 34 cases of talon
cusp in primary dentition have been reported from all
over the world. Out of these 34 cases, only two are in
the mandibular primary dentition and to the best of
our knowledge, ours is the third reported case of this
uncommon anomaly.
The etiology still remains unclear. Various hypotheses
regarding its etiology have been put forward. Some of
these are; it is supposed to be a consequence of outward
folding of inner enamel epithelial cells or may be due to
hyperactivity of dental lamina [2]. Another hypothesis
suggests genetics to be a causative factor of talon cusp based
on its occurrence in a family. Trauma and other localized
forces on tooth germ have also been held responsible
for talon cusp [2, 4, 10]. Lee proposed that hyperactivity
of cells of tooth germ may lead to development of talon
cusp, which is genetically determined but the degree is
influenced by environmental factors [16].
Clinically, it presents as an accessory cusp that
projects from the cingulum area of anterior teeth and
involves more than the half of the lingual or palatal
surface. Hattab et al. classified talons cusp based on the
degree of formation and extension into three categories.
These are [15]:
Type 1 (True talon): A well-delineated additional
cusp that predominantly projects from the
palatal or lingual surface of an anterior teeth and
extends half way from cementoenamel junction
to the incisal edge (as seen in our patient).
Type 2 (Semi talon): An additional cusp of a
millimetre or more but extending less than half
the distance from cementoenamel junction to
incisal edge. It may blend with palatal surface or
strand away from the crown
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IJCRI 2013;4(12):709713.
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Mohan et al.
711
Table 1: The prevalence of talon cusp in primary dentition from 1977 to 2013.Till date 34 cases of talons cusp in primary dentition
have been reported from all over the world. Out of these 34 cases, only two are in the mandibular primary dentition and to the best of
our knowledge, ours is the third reported case of this uncommon anomaly
S. No
Name of Author
Year
Maxillary
Dentition
Mandibualr
Dentition
Age of Patient
2013
51
4 year+ 2 months
Female
Chinese
S.K.Mallineni et al.[7]
2013
51
2 year+9 months
Male
,,
,,
,,
61
2 year +9 months
Male
,,
Prabhu et al.[8]
2012
62
4 years Male
South Indian
82
5 years Male
,,
S.Hedge et al.[10]
72
Not available
,,
Praveen P et al.
2011
51
8 years Male
,,
A.Kapur et al.[12]
2011
51
4 years male
,,
2008
52
Chinese
10
,,
,,
52
,,
11
,,
,,
52
12
51
14 months Male
New York
13
2003
51
13 months Female
Chinese
14
Gungor H et al.
2000
51
14 months Male
,,
15
51
17 months Male
,,
16
Seadano et al.[15]
1989
Mexican
17
Chawla et al.[15]
1983
North Indian
18
Liu JF &Chen JR
1955
51
12 months
Female
Chinese
19
,,
,,
51
,,
Chinese
20
,,
,,
51
3 years+6 months
Female
Chinese
21
,,
,,
51
,,
Chinese
22
Meon R et al.[14]
1990
51
4 years Male
,,
23
Morin CK et al.
1987
51
12 months Male
,,
24
1986
51
10 months Male
,,
25
,,
,,
51
6.5 years
,,
26
,,
,,
51
9 months Male
,,
27
,,
Not
available
51
13 months Male
,,
28
,,
,,
51
4 years Male
,,
29
,,
,,
51
6 years Male
,,
30
Davis PJ
AH[1,14]
51
5 years Male
31
,,
,,
51
5 years Male
32
Natkin E
1983
51
5 years Male
33
Mass et al.
1978
51
1 year Male
34
51
4 years Female
2011
[11]
[14]
[14]
[14]
[14]
[14]
[14]
&Brooke 1986
,,
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IJCRI 2013;4(12):709713.
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CONCLUSION
In this article, we have reviewed literature regarding
talon cusp in primary dentition and have added
significantly to the present knowledge by documenting a
rare case report.
*********
Author Contributions
Ravi Prakash Sasankoti Mohan Substantial
contributions to conception and design, Acquisition of
data, Analysis and interpretation of data, Drafting the
article, Revising it critically for important intellectual
content, Final approval of the version to be published
Sankalp Verma, Udita Singh Substantial contributions
to conception and design, Acquisition of data, Analysis
and interpretation of data, Revising it critically for
important intellectual content, Final approval of the
version to be published
Neha Agarwal Substantial contributions to conception
and design, Acquisition of data, Analysis and
interpretation of data, Revising it critically for important
intellectual content, Final approval of the version to be
published
Soumi Ghanta Substantial contributions to
conception and design, Acquisition of data, Analysis and
interpretation of data, Revising it critically for important
intellectual content, Final approval of the version to be
published
Kuber Tyagi Substantial contributions to conception and
design, Acquisition of data, Analysis and interpretation
Mohan et al.
712
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Ravi Prakash Sasankoti Mohan et al. 2013; This article
is distributed under the terms of Creative Commons
attribution 3.0 License which permits unrestricted use,
distribution and reproduction in any means provided
the original authors and original publisher are properly
credited. (Please see www.ijcasereportsandimages.com/
copyright-policy.php for more information.)
REFERENCES
1. Davis PJ, Brook AH. The presentation of talon
cusp: Diagnosis, clinical features, association and
possible aetiology. British Dental Journal 1986 Feb
8;160(3):848.
2. Shafer WG, Hine MK, LevyBM. A textbook of oral
pathology. 4th ed. Philadelphia: WB Saunders
1983:401.
3. Mellor JK, Ripa LW. Talon cusp: A clinically
significant anomaly. Oral Surg Oral Med Oral Pathol
1970 Feb;29(2):2258.
4. Goaz PW, White SC. Oral Radiology. Missouri: CV.
Mosby Company 1987;2:447.
5. Gardener DG, Girgs SS. Talon cusps: A dental anomaly
in the Rubinstein-Taybi syndrome. Oral Surg Oral
Med Oral Pathol 1979 Jun;47(6):51921.
6. Marder CL. Talon Cusp. J Am Dent Assoc 1981
Aug;103(2):2446.
7. Mallineni SK, Manan NM, Lee CK, King NM. Talon
cusp affecting primary dentition in two siblings: A
case report. Rom J Morphol Embryol 2013;54(1):211
3.
8. Rachana V Prabhu, Prasanna Kumar Rao, Veena
KM, Prathima Shetty, Laxmikanth Chatra, Prashanth Shenai. Prevalence of Talon cusp in Indian
population. J Clin Exp Dent2012;4(1):e237.
9. Swaminathan Kavitha, Haridoss Selvakumar,
Rajendran Barathan. Mandibular talon cusp in
primary lateral incisor: A rare case report. Case Rep
Dent 2012;2012:670745.
10. Hedge S, Ashok Kumar BR. Mandibular talon cusp:
Report of 2 cases. Int Journal of Paediatric dentistry
2004;9(4):303.
11. Praveen P, Anantharaj A, Karthik Venkataraghavan,
Prathibha Rani, Jaya AR. Talon Cusp In A Primary
Tooth. Journal of dental science and research
2011;2(1):3540.
12. Kapur A, Goyal A, Bhatia S. Talon cusp in a primary
incisor: A rare entity. Journal of Indian society of
Pedodontics and Preventive dentistry 2011 JulSep;29(3):24850.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):709713.
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13. Chun-Kei Lee, Nigel M King, Edward CM L, Shiuyin Cho. Talon cusp in primary dentition-literature
review and report of 3 cases. Journal of clinical
pediatric dentistry 2008;30(4):1536.
14. Yoon RK, Chussid S. Dental management of a talon
cusp on a primary incisor. Pediatric dentistry 2007
Jan-Feb;29(1):515.
15. Hattab FN, Yassin OM, Al-Nimri KS. Talon cusp in
permanent dentition associated with other dental
Mohan et al.
713
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IJCRI 2013;4(12):714718.
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Alleyne-Mike et al.
CASE REPORT
714
OPEN ACCESS
*********
Alleyne-Mike KR, Mohamed KEA, van Wijk L, Botha
FCJ. Unique case of dual gynecological malignancies
co-existing with an uncommon thyroid lymphoma.
International Journal of Case Reports and Images
2013;4(12):714718.
*********
doi:10.5348/ijcri-2013-12-418-CR-12
Introduction
Synchronous primaries often pose unique management
challenges. A careful analysis, discussion of patient and
disease-related factors are required to guide appropriate
management. It has been suggested that the upper female
reproductive tract is an extension of the Mullerian system
and thus carcinogenic exposure to this unit has the potential
to produce multicentric lesions [1]. However, the lower
female genital tract does not originate from the Mullerian
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
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CASE REPORT
A 64-year-old female, with a medical history of
hypertension, ischemic heart disease, and previous total
abdominal hysterectomy for a benign indication, presented
to the gynecology department with a six month history of
a pruritic sore to the right labium minorus. Clinically, the
lesion was 3 cm in diameter and extended partially across
the midline with involvement of the clitoris. Two clinically
palpable, 1 cm ipsilateral mobile inguinal lymph nodes
were noted. On further clinical examination, an abdominal
mass was palpated arising from the pelvis and extending
upwards to above the umbilicus. Additionally, she had
experienced progressive dysphagia for the previous three
months. An enlarged, smooth thyroid gland with bilateral
lobe involvement was present.
Investigations showed an elevated CA-125 558
kU/L and TSH 14 mIU/L. Full blood count, lactate
dehydrogenase (LDH), renal and liver function tests
were normal. HIV and syphilis serology were negative.
Computed tomography (CT) scan of neck showed a
massively enlarged thyroid gland with a maximum
diameter of 9.4 cm (right lobe) and 9.1 cm (left lobe)
(Figure 1). There was associated esophageal compression
and the mass displaced the airway anteriorly without
compromise. The CT scan of chest was negative for
malignancy and CT scan of abdomen showed an
enlarged liver and spleen as well as a central pelvic lesion
extending into the abdomen (Figure 2A) causing right
hydronephrosis and hydroureter (Figure 2B).
Biopsy of the vulva lesion was reported as a nonkeratinizing squamous cell carcinoma (Figure 3A) with
associated lichen sclerosus, while that of the thyroid
gland was consistent with a diffuse large B cell lymphoma
(DLBCL) (Figure 3BD). Repeated cytological aspiration
(biopsies) of the inguinal nodes failed to confirm
malignancy. Further staging investigations included a
bone marrow biopsy which was clear.
The patient was discussed in a multi-disciplinary
clinic with input from clinical oncology, gynecology,
hematology, radiology, pathology and anesthesiology.
After extensive discussion, it was agreed to forgo
surgical management of the groin due to concerns over
potential morbidity which would delay commencement of
chemotherapy. The patient underwent laparotomy during
which a cystic left ovarian tumor was found. There was
no ascites, peritoneal or omental nodules seen during the
abdominal exploration. Peritoneal cytology was obtained
in a standard manner, followed by bilateral salpingo-
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DISCUSSION
In gynecologic oncology, the most frequently
documented synchronous lesions are endometrial and
ovarian malignancies [2]. Synchronous vulva and ovarian
primaries are rare. The DLBCL is an aggressive malignancy
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CONCLUSION
The findings outlined in this case are of unknown
significance due to the rarity of the occurrence. This is
the first documented presentation of such a case and thus
may propel interest in further research into the genetics
of multiple synchronous malignancies. The accumulation
of case reports may ultimately be of benefit for the
systematic analysis of multiple neoplasms as the reasons
for their occurrence are likely multi-factorial.
*********
Author Contributions
Kellie Alleyne-Mike Conception and design, Acquisition
of data, Analysis and interpretation of data, Drafting the
article, Critical revision of the article, Final approval of
the version to be published
Khadiga Elfadil Ahmed Mohammed Analysis and
interpretation of data, Critical revision of the article,
Final approval of the version to be published
Adriaan Leon van Wijk Analysis and interpretation of
data, Critical revision of the article, Final approval of the
version to be published
Francois Cornelius Jacobus Botha Acquisition of data,
Critical revision of the article, Final approval of the
version to be published.
Alleyne-Mike et al.
717
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Kellie Alleyne-Mike et al. 2013; This article is
distributed under the terms of Creative Commons
attribution 3.0 License which permits unrestricted use,
distribution and reproduction in any means provided
the original authors and original publisher are properly
credited. (Please see www.ijcasereportsandimages.com/
copyright-policy.php for more information.)
REFERENCES
1. Eisner RF, Nieberg RK, Berek JS. Synchronous
primary neoplasms of the female reproductive tract.
Gynecol Oncol 1989 Jun;33(3):335-9.
2. Zaino R, Whitney C, Brady MF, DeGeest K, Burger RA,
Buller RE. Simultaneously detected endometrial and
ovarian carcinomas--a prospective clinicopathologic
study of 74 cases: a gynecologic oncology group study.
Gynecol Oncol 2001 Nov;83(2):355-62.
3. Gaarenstroom KN, Kenter GG, Trimbos JB, et al.
Postoperative complications after vulvectomy and
inguinofemoral lymphadenectomy using separate
groin incisions. Int J Gynecol Cancer 2003 JulAug;13(4):522-7.
4. van der Velden J, Fons G, Lawrie TA. Primary groin
irradiation versus primary groin surgery for early
vulvar cancer. Cochrane Database Syst Rev 2011 May
11;(5):CD002224.
5. Petereit DG, Mehta MP, Buchler DA, Kinsella TJ.
Inguinofemoral radiation of N0, N1 vulvar cancer may
be equivalent to lymphadenectomy if proper radiation
technique is used. Int J Radiat Oncol Biol Phys 1993
Nov 15;27(4):963-7.
6. Kalidas H. Influence of inguinal node anatomy
on radiation therapy techniques. Med Dosim
1995;20(4):295-300.
7. Hu XR, Hu YX, Fu HR, et al. Diffuse large B-cell
lymphoma with concurrent gastric adenocarcinoma:
case report and literature review. J Int Med Res
2011;39(5):2051-8.
8. Regauer S. Immune dysregulation in lichen sclerosus.
Eur J Cell Biol 2005 Mar;84(2-3):273-7.
9. Gutierrez-Pascual M, Vicente-Martin FJ, LopezEstebaranz JL. Lichen sclerosus and squamous cell
carcinoma. Actas Dermosifiliogr 2012 Jan;103(1):218.
10. Russo A, Cal V, Bruno L, Rizzo S, Bazan V, Di Fede
G. Hereditary ovarian cancer. Crit Rev Oncol Hematol
2009 Jan;69(1):28-44.
Guarantor
The corresponding author is the guarantor of submission.
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718
IJCRI 2013;4(12):719722.
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Mohammed et al.
CASE REPORT
719
OPEN ACCESS
*********
Mohammed KEA, Mike KRA, Parkes J. Unexpected
brain atrophy following administration of intratumoral
interferon alpha-2b for cystic craniopharyngioma: A case
report. International Journal of Case Reports and Images
2013;4(12):719722.
*********
doi:10.5348/ijcri-2013-12-419-CR-13
Introduction
Craniopharyngiomas (CPs) are of pituitary embryonic
origin and are the third most common intracranial
tumor of childhood; most of these tumors are cystic in
nature. They are histologically benign, but can behave
aggressively due to their location. Surgery has been
the mainstay of treatment, but complete resection is
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720
CASE REPORT
An eight-year-old boy was referred by an
ophthalmologist to the Pediatric Oncology Center
(Cape Town) in 2004. He presented with features of
hypopituitarism and bilateral optic atrophy. His imaging
studies showed a ring enhancing lesion with solid and
cystic components arising from the suprasellar area and
compressing the third ventricle (V3) with associated
hydrocephalus.
A subtotal resection and left subdural shunt was
performed at that time, and a histological diagnosis of CP
was made hormonal replacement was initiated.
A year later following his first recurrence the patient
was treated with repeat debulking resection and at this
time an Ommaya reservoir was placed. Further subtotal
resection to the growing lesion, followed by adjuvant
radiotherapy (54 Gy) was done the following year after he
had turned three years old.
At this point, multi-modality treatment allowed for
disease stability for another three years, when evidence
of disease progression was again noted. This prompted
surgical re-intervention and a debulking procedure was
again performed.
In June 2011, at the age of eight, radiological evidence
of tumor recurrence was again noted (Figure 1AB)
and after multi-disciplinary discussion, treatment with
intracystic IFNa-2b was proposed as an alternative method
of gaining local control and thereby controlling disease
progression. A pre-treatment leakage test was done via
the Ommaya reservoir and this showed extensive leakage
around the catheter into the right ventricle (Figure 2).
There was thus concern over the potential for leakage
during IFNa-2b treatment and the clinical implications
of this were researched. The treating multi-disciplinary
team enlisted further advice from international colleagues
regarding the safety of IFNa-2b administration in such
a situation. Following this advice and the information
obtained through documented research, a decision was
made to proceed with the therapy as intra-ventricular
spillage of IFNa was considered to be innocuous.
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DISCUSSION
Craniopharyngioma is considered to be a benign
chronic condition that is subject to increasing symptoms
and deterioration as was observed in the current case
scenario. The intractable tumor growth and treatment
side effects lead to substantial long-term morbidity
and impaired quality of life for these patients. IFNa has
emerged as quite a promising therapy in the long-term
control of the disease. It appears to be far less neurotoxic
than the previously used intratumoral bleomycin, which
has been fatal in some cases, and its administration
requires less technical expertise than is required when
using beta emitting radionuclides such as yttrium [3].
IFNa is the 1st cytokine produced by recombinant
DNA for treatment of infection and malignant diseases.
It has an important role in the induction of apoptosis; cell
regulation and anti-proliferative effects [1, 3].
The first reports in the early 2000 by Jakacki et al.
looked at the safety and efficacy of systemic IFNa in
the treatment of CP. Tumors with a cardinal cystic
component appear to be more likely to respond to IFNa
than solid tumors. However, they reported significant
side effects, which led to the reduction or interruption of
the treatment [1].
Chamberlain et al. used IFNa as an intrathecal drug
for neoplastic meningitis, and they reported both safety
and feasibility of its administration into the circulating
cerebrospinal fluid system [4]. The side effects were
minimal and did not impede its continuation as in the
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CONCLUSION
This is the first documented case of a possible IFNa
induced brain atrophy. Although a causal association
between IFNa administration and neurotoxicity has not
been elucidated, the gravity of the adverse effect warrants
further investigation of the mechanism of occurrence and
extreme vigilance for similar events in the future.
*********
Author Contributions
Khadiga Elfadil Ahmed Mohammed Conception and
design, Acquisition of data, Analysis and interpretation
of data, Drafting the article, Critical revision of the article,
Final approval of the version to be published
Kellie R Alleyne Mike Acquisition of data, Drafting the
article, Critical revision of the article, Final approval of
the version to be published
Jeannette Parkes Analysis and interpretation of data,
Critical revision of the article, Final approval of the
version to be published
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Khadiga Elfadil Ahmed Mohammed et al. 2013; This
article is distributed under the terms of Creative Commons
attribution 3.0 License which permits unrestricted use,
Mohammed et al.
722
REFERENCES
1.
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IJCRI 2013;4(12):723726.
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Fujii et al.
723
Abstract
Introduction: Cholesterol granuloma of the
breast is a rare benign condition which is
difficult to differentiate from breast cancer. Case
Report: We herein report the case of a 56-yearold Japanese asymptomatic female who, on a
computed tomography examination for sigmoid
colon cancer, was found to have a palpable
nodule in the upper external quadrant of the left
breast. Breast carcinoma could not be ruled out
on the basis of clinical and radiological findings,
but excisional biopsy revealed cholesterol
granuloma. A histological examination implied
that this case of cholesterol granuloma might
have been related with mammary duct ectasia.
Conclusion: Although cholesterol granuloma is
a rare disorder in the breast, this case indicated
that a better awareness of this benign breast
disease is important for avoiding misdiagnosis
and unnecessary surgery.
Keywords: Cholesterol
carcinoma, Ductal ectasia
granuloma,
Breast
*********
doi:10.5348/ijcri-2013-12-420-CR-14
Introduction
Cholesterol granuloma consists of fibrous granulation
tissue containing cholesterol crystals with foreign body
giant cells. Cholesterol granuloma occurs commonly in
the mastoid region and in various organs such as the
peritoneum, kidney, parotid gland, thyroglossal duct,
lymph node, liver, and spleen. However, cholesterol
granuloma of the breast is a very rare tumor-like lesion
and has been rarely reported. We herein report a rare
case of cholesterol granuloma of the breast. Cholesterol
granuloma should be considered in the differential
diagnosis of breast lesion suspected to be malignant [16].
*********
Takaaki Fujii1, Reina Yajima1, Hiroki Morita1, Satoru
Yamaguchi1, Soichi Tsutsumi1, Takayuki Asao1, Hiroyuki
Kuwano1
Affiliations: 1Department of General Surgical Science,
Graduate School of Medicine, Gunma University, Gunma,
Japan.
Corresponding Author: Takaaki Fujii, MD, PhD, Department
of General Surgical Science, Graduate School of Medicine,
Gunma University, 3-39-22 Showa-machi, Maebashi,
Gunma 371-8511, JAPAN; Ph: +81-027-220-8224; Fax:
+81-027-220-8230; Email: ftakaaki@med.gunma-u.ac.jp
Received: 20 June 2013
Accepted: 27 July 2013
Published: 01 December 2013
CASE REPORT
A 56-year-old Japanese asymptomatic female was
admitted to our hospital with a diagnosis of sigmoid colon
cancer. Computed tomography (CT) scan of her chest
and abdomen was done as a preoperative examination of
her colon cancer, and showed a nodule in the left breast
(Figure 1). The patient had no family history of breast
cancer, no history of mammary trauma or biopsy and no
history of lipid metabolism abnormality.
On admission, physical examination revealed a
2.0x2.0 cm palpable nodule which was elastically firm
and mobile in the upper external quadrant of the left
breast with no associated skin findings. There was no
axillary or supraclavicular lymphadenopathy, and no
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Fujii et al.
724
DISCUSSION
We herein report a rare case of cholesterol granuloma
of the breast. Cholesterol granuloma of the breast
is composed of fibrous granulation tissue in which
cholesterol crystals accompanied by giant cells are
deposited. The pathogenesis of cholesterol granuloma is
not fully understood. Physical and infectious factors have
been considered and some reports postulate that they are
caused by a reactive response to an unknown stimulus. In
the breast, cholesterol granuloma is thought to represent
a rare manifestation of duct ectasia. Lipid-rich material
is normally found in ectatic ducts. It is postulated that
leakage of cholesterol crystals from the ectatic ducts
causes a foreign body giant cell reaction which results
in granuloma formation. Mammary duct ectasia is a
disorder of middle-aged women. Mammary duct ectasia
is a frequent disorder of the breast that has a wide variety
of clinical manifestations, and this disorder sometimes
involves an extensive disruption of the dilated duct
and is occasionally noticed as a palpable mass. In rare
instances the mass, microscopically, contains cholesterol
crystals. In the pathologic specimen in this case, a
cystic lesion surrounded by fibrous granulation tissue,
chronic inflammation, cholesterol crystals, and scattered
multinucleated giant cells was observed. The lesion was
considered to be cholesterol granuloma associated with
duct ectasia [16].
The main concern with breast cholesterol granuloma is
performing differential diagnosis. Clinically, it resembles
breast cancer. The clinical and radiological features of
cholesterol granuloma mimic those of a carcinoma of
the breast. Thus, it is difficult to distinguish cholesterol
granuloma from carcinoma of the breast based on the
Figure 2: Mammography showing a 2.0 cm in diameter highdensity shadow lesion, which corresponded to the palpable
mass, with partly ill-defined margins and without specula or
microcalcification.
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725
Author Contributions
Guarantor
Figure 4: The histological evaluation revealed a cystic mass
surrounded by fibrous granulation tissue in which (A) needlelike crystals were deposited, (B) accompanied by giant cells
(H&E stain, 200x).
Conflict of Interest
Authors declare no conflict of interest.
Copyright
imaging results. The diagnosis of cholesterol granuloma
is histologic. It is not difficult to distinguish between
cholesterol granuloma and breast cancer histologically,
and core needle biopsy is thought to be useful for
differential diagnosis. However, it should be noted that
breast cholesterol granuloma could be accompanied by
cancer. Thus, cases in which malignancy cannot be ruled
out clinically, a complete removal of the tumor helps in
making a definitive diagnosis [16].
REFERENCES
1.
CONCLUSION
We here reported a rare case of breast cholesterol
granuloma mimicking breast cancer. Reports on breast
granuloma are rare. However, duct ectasia itself,
which is related with cholesterol granuloma, is a very
frequent disorder. To make an accurate diagnosis of this
rare disorder and rule out the possibility of cancer, a
histological examination is required.
*********
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5.
Fujii et al.
6.
726
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Sayed et al.
CASE REPORT
727
OPEN ACCESS
*********
Sayed A. Severely ectatic left circumflex coronary artery
with fistula to coronary sinus presenting with acute
coronary syndrome. International Journal of Case
Reports and Images 2013;4(12):727730.
*********
doi:10.5348/ijcri-2013-12-421-CR-15
Introduction
An aneurysmal left circumflex coronary artery (LCX)
with associated fistulous connection to the coronary
sinus (CS) is an extremely uncommon coronary artery
anomaly. A few patients have been reported in literature.
The majority of these cases were asymptomatic and
presented late in life. Accurate diagnosis of these fistulas
often requires multiple imaging modalities to delineate
the entire pathway of the fistula. Multiple treatment
options are available including surgery, trans catheter
closure, and conservative management.
CASE REPORT
A 56-year-old male, who was on a skiing trip, was
presented with sudden onset of retrosternal chest
pressure with shortness of breath and diaphoresis.
There was no family history of congenital heart disease
or other inherited abnormalities. Physical Examination
revealed normal vital signs and a grade II continuous
murmur loudest at the left sternal border in the fourth
intercostal space. The electrocardiography showed
inferior ST segment depression and his high sensitivity
troponin-T was 417 ng/L. A posteroanterior and
lateral X-ray of chest showed only mild cardiomegaly.
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728
DISCUSSION
Coronary arteriovenous fistulas (CAFs) are considered
a major coronary anomaly [1], with an incidence of 0.1%
to 0.87% [2, 3]. The majority of cases originate from the
right coronary and left anterior descending coronary
arteries. Coronary artery fistulas originating from the
circumflex coronary artery account for only 18% of all
coronary arterial (venous) fistulas [3]. More than 90% of
the fistulas drain into the right side of the heart with the
most common sites of drainage being the right ventricle,
right atrium, and pulmonary artery [4, 5]. Rarely, they
drain into the CS in 7% and the superior vena cava in
1% of cases [4]. A fistula originating from the circumflex
artery that drains into the coronary sinus is rare. In one
literature review a total of 14 cases of LCX to CS fistula
were reported [6]. Most of these cases were found
incidentally in male and most common presentation
was either dysnpnea on exertion or chest pain, rarely
can remain asymptomatic. Two cases involved inferior
and inferolateral myocardial infarction related to the
CAF with patent epicardial coronary arteries [6]. The
dilation of the coronary artery may relate to prolonged
hyperdynamic circulation from the fistula.
Our patient presented in his late fifties. He was
healthy with no limitation in physical activity. His initial
presentation was one of an acute coronary syndrome
which was confirmed by his electrocardiography and
troponin abnormalities. His symptoms were likely
secondary to significant shunt-induced ischemia triggered
by increased physical activity during skiing. The diagnosis
of the fistula in our patient was established by invasive
coronary angiography and CT coronary angiography.
The management of coronary arteriovenous fistulas
remains controversial. There are significant differences
in the clinical presentation, size and anatomical
connection of coronary artery fistulas, which has led to
a lack of consistent guidelines for treatment [7]. In the
most recent American College of Cardiology/American
Heart Association Guidelines for the Management of
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Sayed et al.
CONCLUSION
Coronary artery fistulas are uncommon and can have
different presentations ranging from asymptomatic to
acute coronary syndromes. The management of coronary
artery fistulae depends on the severity and the clinical
presentation of these congenital anomalies.
*********
Author Contributions
Ahmed Sayed Substantial contributions to conception
and design, Acquisition of data, Analysis and
interpretation of data, Drafting the article, Revising
it critically for important intellectual content, Final
approval of the version to be published
Guarantor
The corresponding
submission.
author
is
the
guarantor
of
Conflict of Interest
Authors declare no conflict of interest.
729
REFERENCES
1.
Copyright
Ahmed Sayed 2013; This article is distributed under
the terms of Creative Commons attribution 3.0 License
which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
ACKNOWLEDGEMENTS
I would like to thank Dr. Wardal for the time and
effort that he spent in the critical revision of this article.
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730
IJCRI 2013;4(12):731734.
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Sarkar et al.
CASE REPORT
731
OPEN ACCESS
Abstract
Introduction
Introduction:
Bronchogenic
cyst
is
an
incidentally detected mediastinal mass which is
usually asymptomatic in most of the cases. The
bronchogenic cyst appears to be a well-defined,
nonenhancing cystic mass with peripheral
calcification and presence of milk of calcium in
some cases. It may be said congenital if it detected
in very early age group. Case Report: A case of
bronchogenic cyst was incidentally detected by
computed tomography scan in a 35-year-old
male presenting with the heaviness in the chest.
Conclusion: The case is reported here to give an
emphasis on its formation and mode of treatment
accordingly due to its so hazardous position in
the mediastinum.
*********
Sarkar A, Pandit N. Bronchogenic cyst: A case report.
International Journal of Case Reports and Images
2013;4(12):731734.
*********
doi:10.5348/ijcri-2013-12-422-CR-16
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Sarkar et al.
732
CASE REPORT
A 35-year-old male patient was presented with sense
of heaviness in chest and shortness of breath even at
rest for past two years. He gave history of intermittent
low grade fever, cough, right sided pain chest since his
adolescence and was frequently treated with antibiotics.
Chest X-ray showed a right sided mediastinal mass with
ipsilateral peripheral pleural calcification in the mid/
lower zone. Electrogram and echocardiography were
within normal limits. The CT scan of thorax showed a
right sided posterior mediastinal, unilocular, cystic mass
lesion having diameter of 10.9x8 cm with a homogeneous,
increased fluid attenuation value of +15 HU (Figure 1). On
contrast study the cyst was thin-walled, nonenhancing
except a small part of its right edge suggesting focal
thickening of inflamed mediastinal pleura or compressed
part of lung adjacent to the cyst (Figure 2). A fluid was
found at its dependent part, both in supine and in prone
position due to presence of a small amount of higher
density fluid (Figure 1, Figure 2 and Figure 3). A focus
of calcification was found in a part its periphery (Figure 3).
Absence of air within the cyst proved that the cyst was
noncommunicating. Right intermediate bronchus was
partially compressed and slightly displaced by the mass
(Figure 4). Associated ipsilateral periopheral pleural
calcification was present. The CT guided aspiration
revealed its content to be whitish, opalescent fluid with
high protein, epithelial cells, amorphous calcium. Culture
report was negative. Fibreoptic bronchoscopy revealed
mucosal thickening and mild narrowing of lumen of right
intermediate bronchus due to extrinsic compression.
Bronchoscopic biopsy specimen revealed inflammatory
changes and no malignant cell was detected.
DISCUSSION
Bronchogenic cysts are the cystic malformations which
are having the respiratory epithelial lining. As it is a space
occupying lesion in the mediastinum, so the appearance
of the symptoms of patients having the bronchogenic
cysts depend on the position and most importantly the
size of the tumor. As they are enlarge, they may produce
symptoms by compression of the surrounding structures.
A few of the cysts might become infected and can rupture
into the nearest bronchus causing mucopurulent sputum,
hemoptysis and fever. Bronchogenic cysts are usually
asymptomatic in early lives. Bronchogenic cysts can be
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Sarkar et al.
733
CONCLUSION
The bronchogenic cysts are rare mediastinal mass
which are very much important in treatment point
of view when it is diagnosed. There are a few cases of
bronchogenic cysts have been reported till now. The case
is presented herein with a hope that it will open new way
of better treatment of such developmental mass having a
knot of complications which is tightening with the delay
of its removal.
*********
Author Contributions
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Aniruddha Sarkar et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
REFERENCES
Figure 4: Axial computed tomography scan of thorax at the
level of carina; mediastinal window, prone position.
1.
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734
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Katsuragi et al.
CASE REPORT
735
OPEN ACCESS
*********
Katsuragi S, Osaki M, Suzuki R, Ikeda T, Toyoda K,
Yoshimatsu J. Reversible cerebral vasoconstriction
syndrome in HELLP syndrome. International Journal of
Case Reports and Images 2013;4(12):735738.
*********
doi:10.5348/ijcri-2013-12-423-CR-17
Introduction
Reversible cerebral vasoconstriction syndrome
(RCVS) is characterized by severe headaches, often
thunderclap headaches, with or without focal deficits
and seizures, and a multifocal constriction of cerebral
arteries, which resolve spontaneously within three
months. This syndrome has a female preponderance
and a mean age of onset of approximately 42 years [1,
2]. The pathophysiology of RCVS is not fully understood.
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Katsuragi et al.
736
CASE REPORT
A 33-year-old primipara women was referred to
our hospital because of severe headache on third day
postpartum following vaginal delivery at 37th week.
Her delivery course at a previous hospital was eventful
because she developed an eclamptic attack just before
delivery and was diagnosed with HELLP syndrome based
on findings of hemolysis, elevation of liver enzymes
(AST/ALT: 1455/663 U/L), and a low platelet count
(40103/L). Her blood pressure on tonic-clonic seizure
was 192/108 mmHg. On admission to our hospital,
the patient was alert and conscious, but had severe
headache over the temples with a positive Kernigs sign.
Computed tomography of head showed hemorrhage in
the left caudate nucleus and magnetic resonance imaging
(MRI) of the corpus callosum on both sides showed fluid
attenuated inversion recovery (FLAIR) and diffusionweighted image (DWI) hyperintensity, and apparent
diffusion coefficient (ADC) map hypointensity. The
anterior cerebral arteries (ACA) and middle cerebral
arteries (MCA) were barely detectable by brain MRA
(Figure 1A). Therapy for brain edema and continuous
intravenous infusion of nicardipine were performed as
conservative management.
On hospital day 2, TCCFI indicated MCA peak
systolic velocities (PSV) of 330 (right side) and 266 (left
side) cm/s (Figure 2). On hospital day 3, nicardipine
was discontinued and replaced by oral administration
of a calcium blocking agent, which maintained blood
pressure at approximately 130/80 mmHg. Headache
was also relieved from hospital day 7. TCCFI on hospital
day 13 showed that the PSV of both MCAs had improved
to approximately 150 cm/s. MRA on hospital day 14
and at 43rd day postpartum revealed progressive
improvement of the lesions in the ACAs and MCAs
(Figure 1BC). On hospital day 17, cerebral angiography
showed improvement of segmental changes of the ACA
and a larger diameter of the MCA compared to that in
previous tests (Figure 3). MRI scan of head at 43rd day
postpartum showed decreased caudate hemorrhage and
disappearance of the lesions in the corpus callosum.
DISCUSSION
The pathophysiology of eclampsia is thought to involve
vasoconstriction [3]. The patient in this case developed
juvenile cerebral hemorrhage during puerperium, and
the observation of multiple reversible stenoses and
Figure 2: Middle cerebral artery peak systolic velocities (MCAPSV) measured by transcranial color flow imaging. Both right
and left MCA-PSVs were severely increased on hospital days
2-3, but had gradually decreased to an almost normal range
by hospital day 13. The gray area indicates the normal velocity
range (100-130 cm/s).
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):735738.
www.ijcasereportsandimages.com
Katsuragi et al.
737
CONCLUSION
The peak velocity of middle cerebral artery by
transcranial color flow imaging was the highest in
reversible cerebral vasoconstriction syndrome cases
that have ever reported, and large areas of cytotoxic
edema disappeared in the chronic stage. These facts
and magnetic resonance angiography/angiography
reflects the vasoconstriction was most severe but rapidly
normalized which was clearly shown by transcranial color
flow imaging. These results further support the idea that
transcranial color flow imaging demonstrates the rapid
change of vasoconstriction even in the most acute type of
reversible cerebral vasoconstriction syndrome.
*********
Author Contributions
Shinji Katsuragi Substantial contributions to
conception and design, Acquisition of data, Analysis
and interpretation of data, Drafting the article, Revising
it critically for important intellectual content, Final
approval of the version to be published
Masato Osaki Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
Rieko Suzuki Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
Tomoaki Ikeda Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
Kazunori Toyoda Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
Jun Yoshimatsu Analysis and interpretation of data,
Drafting the article, Revising it critically for important
intellectual content, Final approval of the version to be
published
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):735738.
www.ijcasereportsandimages.com
Guarantor
Katsuragi et al.
4.
Conflict of Interest
5.
Copyright
Shinji Katsuragi et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
REFERENCES
1.
2.
3.
6.
7.
8.
9.
10.
738
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):739741.
www.ijcasereportsandimages.com
Gonzalez et al.
CASE REPORT
739
OPEN ACCESS
Abstract
Introduction:
Venous air embolism is a
predominantly iatrogenic complication that
occurs when atmospheric gas is introduced
into the systemic venous system. The incidence
of venous air embolism associated with the
injection of contrast for computed tomography
scan reportedly is 1123%. Yet, most patients are
asymptomatic, it can be fatal if the amount of
air is enough to cause hemodynamic instability.
Case report: We report a case of a 20-year-old
female patient who presented with air bubbles
in the right ventricle after an IV contrast
computed tomography of the abdomen. Venous
air embolism is a potential complication after an
IV contrast injection. Conclusion: We conclude
that is imperative that clinicians pay special
attention to the prevention, association, and
early recognition of venous air embolism when
performing imagines studies with IV contrast.
Keywords: Venous air embolism, Intravenous
contrast injector
*********
Gonzalez C, Aquino D, Kanna B. Air bubble in the heart:
An unrecognized complication of IV contrast injector.
International Journal of Case Reports and Images
2013;4(12):739741.
*********
doi:10.5348/ijcri-2013-12-424-CR-18
Introduction
Venous air embolism (VAE) is predominantly an
iatrogenic complication that occurs when atmospheric gas
is introduced into the systemic venous system [1]. Venous
air embolism has been described as a complication of
neurosurgical procedures performed in a seated position,
central venous catheterization, penetrating and blunt
chest trauma, high-pressure mechanical ventilation,
thoracentesis, hemodialysis, and several other invasive
procedures [1]. However, since the advent of relatively
newer radiological technologies with power intravenous
(IV) contrast injectors, it has become an important
contributor to the incidence of VAE [13]. The incidence
of venous air embolism associated with the injection of
contrast for computed tomography (CT) scan reportedly
is 1123% [1]. Venous air embolism associated with IV
contrast injection is now widely recognized as a cause
of VAE, yet a few cases have been reported in literature.
Clinicians should be aware of the association between
VAE and radiologic exams with IV contrast. We report a
case of a 20-year-old female patient presented with air
bubbles in the right ventricle after an IV contrast CT scan
of the abdomen.
CASE REPORT
Received: 18 October 2012
Accepted: 17 November 2012
Published: 01 December 2013
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):739741.
www.ijcasereportsandimages.com
Figure 1: (A, B) Right ventricular air embolism in a 20-yearold female patient: Contrast computed tomography scan of the
abdomen, upper sections showing multiple air bubbles in the
right ventricle without obstruction of right ventricle outflow
tract (arrow).
Gonzalez et al.
740
DISCUSSION
Power contrast injectors have the ability to deliver
400 mL of air in 5 seconds if inappropriately timed
[4]. Although the volume of air required to cause death
is believed to be approximately 200 to 300 mL [2,
5, 6], the amount required to induce hemodynamic
instability is unclear. Most cases of VAE associated with
contrast injection are clinical asymptomatic and often
go undiagnosed [7]. Yet, when the amount of air is large
enough to cause symptoms these may include, but are
not limited to: cardiopulmonary and central nervous
symptoms such as cardiac arrhythmias, chest pain,
electrocardiography changes, dyspnea, altered mental
status, focal deficit, coma, and death; if it is not detected
and treated in time [8]. Other factors associated with the
severity of VAE include the rate of infusion, position of
the patient and the patient baseline health [2, 3, 8].
When acute right ventricular failure secondary to
venous air embolism occurs, it is due to an increase in
the pressure of the pulmonary circulation as a result
of mechanical blood flow obstruction by an air bubble.
Once the right ventricle fails, there may be a secondary
decrease of the venous return to the left ventricle, leading
to decreased cardiac output and systemic cardiovascular
collapse [1].
The management of VAE consists of stopping and
preventing any ongoing air entry, hemodynamic support
with fluid, pressors or inotropes if needed, and increasing
the patients oxygenation. To achieve this, the patient
should be started on 100% oxygen to decrease air bubble
size and placed in a Trendelenburg position that displaces
the air bubbles in the right ventricle to a more buoyant
position [1, 3, 4, 8]. Direct right heart air aspiration and
hyperbaric oxygen also has been described with a good
success rate and should be used when available and
indicated [812].
CONCLUSION
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):739741.
www.ijcasereportsandimages.com
*********
Author Contributions
Carlos Gonzalez Substantial contributions to
conception and design, Acquisition of data, Analysis
and interpretation of data, Drafting the article, Revising
it critically for important intellectual content, Final
approval of the version to be published
Dioben Aquino Acquisition of data, Drafting the article,
Revising it critically for important intellectual content,
Final approval of the version to be published
Balavenkatesh Kanna Acquisition of data, Drafting the
article, Revising it critically for important intellectual
content, Final approval of the version to be published
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Carlos Gonzalez et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
REFERENCES
1.
Gonzalez et al.
741
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):742744.
www.ijcasereportsandimages.com
Clinical image
Rodrguez-Gutirrez et al.
742
OPEN ACCESS
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):742744.
www.ijcasereportsandimages.com
Rodrguez-Gutirrez et al.
743
*********
Rodrguez-Gutirrez R, Rodarte-Shade M, GonzlezGonzlez JG. Unexpected Crohns Disease after a Free
Gastrointestinal Perforation. International Journal of
Case Reports and Images 2013;4(12):742744.
*********
doi:10.5348/ijcri-2013-12-425-CL-19
*********
DISCUSSION
Crohns disease is a chronic illness of uncertain
etiology that is characterized by a transmural
inflammation of the entire gastrointestinal tract. Crohns
complications are fistulae, localized peritonitis, abscesses,
bowel obstructions, gastrointestinal perforation, and
hemorrhage. Of them free gastrointestinal perforation,
which is a life-threatening complication, is the most rare
and described only in 12% of these patients. It is more
common as the initial manifestation of the illness, but
has also been described during the course of the disease
[1]. Most of the patients present instead with an intraabdominal abscesses or fistulae. The most common
site of free perforation is the ileum or jejunum and
chronic steroid use has been proposed as risk factor for
perforation. Pneumoperitoneum, as found in this case,
is an uncommon feature of small intestine perforation
[2]. Diagnosis is made by the characteristic clinical
presentation of acute abdominal pain along with acute
systemic inflammatory response syndrome or sepsis and
the characteristic findings on the chest and abdominal
X-rays or computed tomography. Immediate management
to achieve hemodynamic stability with high-volume
intravenous fluids and sometimes catecholamines along
with broad-spectrum antibiotics is essential. Nevertheless,
urgent surgical management with exploratory laparotomy
is the cornerstone of the treatment. Prognosis compared
to ulcerative colitis perforation is formidable with a
mortality rate of less than 5% if managed promptly [3].
In this case, the steroids that could have treated and
masked the illness for a long time could have also been
the triggering cause of the gastrointestinal perforation.
CONCLUSION
Free gastrointestinal perforation in Crohns disease
is an unusual but life-threatening complication that
Author Contributions
Ren Rodriguez-Gutierrez Served as the principal
investigator and made substantial contributions in the
conception, design and acquisition of the data, Drafting
and revising it critically for important intellectual content,
Approved the final version to be published
Mario Rodarte-Shade Made substantial contributions
in the conception, design and acquisition of the data,
Drafting and revising it critically for important intellectual
content, Approved the final version to be published
Jos Gerardo Gonzlez-Gonzlez Made substantial
contributions in the conception, design and acquisition of
the data, Drafting and revising it critically for important
intellectual content, Approved the final version to be
published
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
Ren Rodriguez-Gutierrez et al. 2013; This article
is distributed under the terms of Creative Commons
attribution 3.0 License which permits unrestricted use,
distribution and reproduction in any means provided
the original authors and original publisher are properly
credited. (Please see www.ijcasereportsandimages.com/
copyright-policy.php for more information.)
REFERENCES
1.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):742744.
www.ijcasereportsandimages.com
Rodrguez-Gutirrez et al.
2.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
744
IJCRI 2013;4(12):745747.
www.ijcasereportsandimages.com
Olubaniyi et al.
clinical image
745
OPEN ACCESS
Hemophilic Arthropathy
Babajide O Olubaniyi, Olubukola Ajala, David C Howlett
case report
A 34-year-old male with hemophilia A presented to
the emergency department with pain and swelling in his
left knee following a minor fall. He was known to suffer
from advanced arthropathy and was awaiting a total knee
replacement. On clinical examination, the left knee was
swollen and tender with a reduced range of movement.
Plain radiograph (antero-posterior and lateral views)
performed (Figures 1 and 2) excluded a fracture or
dislocation but demonstrated classic appearances
of hemophilic arthropathy with severe secondary
degenerative changes. He was discharged with adequate
simple analgesia and outpatient follow-up appointments
with his hematologist and physiotherapist.
DISCUSSION
Hemophilia is an inherited, X-linked recessive
disorder caused by deficiency of plasma clotting factors
VIII, IX or XI. It manifests in males while females
are asymptomatic carriers. Hemophilic arthropathy
Babajide O Olubaniyi1, Olubukola Ajala2, David C Howlett3
Affiliations: 1FRCR, Department of Radiology, Eastbourne
District General Hospital, Kings Drive, Eastbourne, East
Sussex, BN21 2UD, United Kingdom; 2MRCP, Department
of Diabetes and Endocrinology, Western Sussex NHS Trust,
Worthing Hospital, Lyndhurst road, BN11 2DH, United
Kingdom, 3FRCR, Department of Radiology, Eastbourne
District General Hospital, Kings Drive, Eastbourne, East
Sussex, BN21 2UD, United Kingdom.
Corresponding Author: Dr. Babajide O Olubaniyi, Department
of Radiology, Eastbourne District General Hospital, Kings
Drive, Eastbourne, East Sussex, BN21 2UD, United
Kingdom; Tel: +441323417400, Extension 4673; Email: jide.
olubaniyi@nhs.net
Received: 03 June 2013
Accepted: 30 July 2013
Published: 01 December 2013
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):745747.
www.ijcasereportsandimages.com
Olubaniyi et al.
746
CONCLUSION
Hemophilia can cause progressive joint destruction
and loss of function. Early prophylactic treatment in
children with severe hemophilia can prevent or delay
joint destruction.
*********
Olubaniyi BO, Ajala O, Howlett DC. Hemophilic
Arthropathy. International Journal of Case Reports and
Images 2013;4(12):745747.
*********
doi:10.5348/ijcri-2013-12-426-CL-20
*********
Author Contributions
Babajide O Olubaniyi Substantial contributions to
conception and design, Acquisition of data, Analysis
and interpretation of data, Drafting the article, Revising
it critically for important intellectual content, Final
approval of the version to be published
Olubukola Ajala Acquisition of data, Drafting the
article, Revising it critically for important intellectual
content, Final approval of the version to be published
David C Howlett - Substantial contributions to conception
and design, Acquisition of data, Revising it critically for
important intellectual content, Final approval of the
version to be published
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
Copyright
BO Olubaniyi et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):745747.
www.ijcasereportsandimages.com
REFERENCES
1.
Olubaniyi et al.
2.
3.
747
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):748749.
www.ijcasereportsandimages.com
Desai et al.
748
To the Editor,
Plasma cell leukemia is very rare comprising 2%
plasma cell dyscrasias [1]. We report a case of a 49-yearold male with complaints of chest pain, breathlessness,
and acute attacks of gastroenteritis. On examination,
there was marked pallor and hepatosplenomegaly.
X-ray and computed tomography (CT) scan revealed
multiple paratracheal lymph node enlargements.
However, there was no diffuse osteopenia or multiple
osteolytic lesions. Laboratory analysis revealed high
serum calcium 11.5 mg/dL, low hemoglobin 5 g/dL,
normal WBC count, thrombocytopenia 22000/mm3
and elevated erythrocyte sedimentation rate (ESR).
Peripheral smear showed many atypical cells (41%) with
blastoid morphology, giving a blood picture of acute
leukemia. However, on closer inspection many of them
appeared plasmacytoid. There was marked rouleaux
formation of red cells in the background. Bone marrow
aspirate and biopsy also revealed near total replacement
by such atypical cells with plasmacytoid morphology.
Serum electrophoresis revealed a prominent M spike
and immunofixation revealed IgG heavy chain and kappa
light chain immunostaining ruling out the possibility
of Waldenstrms macroglobulinemia. Plasma cell
Parth Desai1, Sagar Dholariya2, Sanjeev Gupta3, Tejinder
Singh4
Affiliations: 1MBBS, Resident Department of Pathology,
MAMC, Maulana Azad Medical College, New Delhi; 2MBBS,
Resident Department of Biochemistry, MAMC, Maulana
Azad Medical College, New Delhi; 3DM Hematopathology,
Professor AIIMS, Maulana Azad Medical College, New
Delhi; 4MD Pathology, Director Professor, MAMC, Maulana
Azad Medical College, New Delhi.
Corresponding Author: Parth Anil Desai, MBBS, Resident,
Department of Pathology, MAMC, Maulana Azad Medical
College, New Delhi; Email: parth1410@gmail.com
Received: 07 May 2013
Accepted: 27 June 2013
Published: 01 December 2013
*********
Desai PA, Dholariya S, Gupta S, Singh T. Plasma cell
leukemia of IgG secreting type: A rare case. International
Journal of Case Reports and Images 2013;4(12):748
749.
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]
IJCRI 2013;4(12):748749.
www.ijcasereportsandimages.com
*********
doi:10.5348/ijcri-2013-12-427-LE-21
*********
Author Contributions
Parth Anil Desai Substantial contributions to conception
and design, Acquisition of data, Drafting the article,
Revising it critically for important intellectual content,
Final approval of the version to be published
Sagar Dholariya Substantial contributions to conception
and design, Acquisition of data, Drafting the article,
Revising it critically for important intellectual content,
Final approval of the version to be published
Sanjeev Gupta Substantial contributions to conception
and design, Acquisition of data, Drafting the article,
Revising it critically for important intellectual content,
Final approval of the version to be published
Tejinder Singh Substantial contributions to conception
and design, Acquisition of data, Drafting the article,
Revising it critically for important intellectual content,
Final approval of the version to be published.
Desai et al.
Copyright
Parth Anil Desai et al. 2013; This article is distributed
under the terms of Creative Commons attribution 3.0
License which permits unrestricted use, distribution and
reproduction in any means provided the original authors
and original publisher are properly credited. (Please see
www.ijcasereportsandimages.com/copyright-policy.php
for more information.)
REFERENCES
1.
Guarantor
The corresponding author is the guarantor of submission.
Conflict of Interest
Authors declare no conflict of interest.
749
IJCRI International Journal of Case Reports and Images, Vol. 4 No. 12, December 2013. ISSN [0976-3198]