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CASE REPORT

Kerion of the Vulva Caused by Trichophyton


mentagrophytes
Amina Bougrine, Catherine Villeneuve-Tang, Danielle Bouffard, Danielle Rouleau, and Suzanne Chartier
Background: Deep dermatophytosis of genital skin is a rare clinical manifestation of infection by a common group of pathogens.
Objective: We emphasize the importance of clinical suspicion and the use of accurate diagnostic methods in the evaluation of
deep dermatophytosis.
Methods: We report a single case of tinea pubis, kerion type, caused by Trichophyton mentagrophytes in an immunocompetent
host.
Results: A 54-year-old female presented with a suppurative infection of the vulva and pubis that was unresponsive to empirical
antibiotic therapy. T. mentagrophytes was isolated. Oral itraconazole was initiated on the basis of clinical suspicion and continued
for a total of 6 weeks.
Conclusion: Accurate diagnosis and treatment of deep dermatophytosis of genital skin rests upon proper identification of the
pathogen. Prompt initiation of treatment with an oral antifungal agent, such as itraconazole, should be undertaken in order to avoid
irreversible scarring alopecia.
Contexte: Les dermatophytoses profondes des parties genitales sont une manifestation clinique rare dinfections causees par un
groupe frequent dagents pathoge`nes.
Objectif: Letude visait a` faire ressortir limportance du doute clinique et du recours a` des examens precis de diagnostic dans
levaluation de dermatophytoses profondes.
Methode: Sera expose ici un cas de teigne du pubis, de type kerion, cause par Trichophyton mentagrophytes, chez un hote
immunocompetent.
Resultats: Une femme de 54 ans a consulte pour une infection suppuree de la vulve et du pubis, refractaire a` lantibiotherapie
empirique. Le champignon T. mentagrophytes a ete isole. Un traitement par litraconazole, par voie orale, reposant sur les doutes
cliniques a ete amorce, et il sest poursuivi pendant 6 semaines en tout.
Conclusions: Le diagnostic exact des dermatophytoses profondes des parties genitales et leur traitement reposent sur
lidentification precise de lagent pathoge`ne. Il faudrait entreprendre rapidement le traitement par un antifongique oral, tel que
litraconazole, afin deviter une alopecie cicatricielle irreversible.

54-YEAR-OLD HAITIAN WOMAN presented to the


emergency department of a university hospital with
acute painful lesions of the vulva and inguinal region.
She was brought to the hospital by ambulance for syncope secondary to pain and dehydration. She was given
a presumptive diagnosis of bacterial folliculitis and

From the Departments of Dermatology, Pathology, and Microbiology,


Centre Hospitalier de lUniversite de Montreal, Montreal, QC.
Address reprint requests to: Amina Bougrine, BSc, MDCM, Centre
Hospitalier de lUniversite de Montreal Dermatology, Hopital St-Luc Pavillion Edouard-Asselin (Porte 1305) 264, boulevard Rene Levesque E.,
Montreal, PQ, H2X 1P1; e-mail: amina.bougrine@gmail.com.

DOI 10.2310/7750.2013.13101
# 2014 Canadian Dermatology Association

206

cellulitis of the vulva. A consultation in dermatology


was requested for a diagnostic opinion after she failed to
respond to a 6-day course of broad-spectrum intravenous
antibiotics.
The patient first noticed a solitary lesion on the vulva
2 weeks previously that had expressed a purulent discharge
on manipulation. After shaving the pubic region, she
noticed the appearance of multiple similar lesions. She
complained of vulvar swelling and pain on walking and
urination. The vulvar lesions were itchy, with minimal
relief obtained with an over-the-counter anti-itch cream.
Her past medical history was unremarkable, aside from a
malarial infection 2 years previously. There were no risk
factors for immunosuppression or sexually transmitted
infections.

Canadian Dermatology Association | Journal of Cutaneous Medicine and Surgery, Vol 18, No 3 (May/June), 2014: pp 206209

Kerion of the Vulva Caused by T. mentagrophytes

On examination, there were erythematous, edematous,


confluent plaques with oozing, boggy nodules involving the
mons pubis, labia majora, and inguinal folds (Figure 1).
Subtle, hyperpigmented, scaly patches were present in the
inguinal folds and the suprapubic region. There was no
significant regional lymphadenopathy. Ill-defined, erythematous, scaly patches were noted on the left hand (Figure 2).
The feet showed xerosis without erythema or interdigital
maceration. The highest fever recorded during hospitalization was 38.0uC.
The white cell count was 17.5 3 109/L with 80%
neutrophils. Swab cultures showed nonpathogenic bacterial colonization of the vulva. The patient was started on
empirical treatment with an oral antifungal because
bacterial cultures were negative and no improvement was
observed after 6 days of intravenous antibiotics. Two
biopsies were taken for routine histology and culture.
Histopathology demonstrated an area of inflammation in
continuity with a dilated hair follicle. The inflammatory
infiltrate in the superficial and deep dermis was composed
of neutrophils, lymphocytes, histiocytes, eosinophils, and
multinucleated giant cells of both the Langhans and
foreign body subtypes (Figure 3). The epidermis exhibited
mild acanthosis. The periodic acidSchiff, Grocott, and
Ziehl stains failed to demonstrate the presence of fungal
organisms. Human immunodeficiency virus (HIV) serologies and a tuberculin skin test were negative.
Trichophyton mentagrophytes was isolated in both the
biopsy and the skin scrapings from the left hand, thus
establishing the diagnosis of tinea profunda of the vulva and
tinea manuum, respectively. A culture of skin scrapings
from the feet was negative for dermatophytes. The patient
was treated with itraconazole 200 mg orally twice daily for

6 weeks, which led to complete resolution of her symptoms.


Nevertheless, the patient developed irreversible scarring
alopecia on areas of the mons pubis and vulva.

Figure 1. Erythema and edema of the mons pubis, as well as boggy,


oozing nodules involving the mons pubis, labia majora, and inguinal
folds. Subtle scaly patches were noted on the lower abdomen and in
the inguinal folds.

Figure 3. Dermal polymorphous inflammatory infiltrate composed of


neutrophils, lymphocytes, histiocytes, eosinophils, and multinucleated
giant cells of both the Langhans and foreign body subtypes.
Hematoxylin-phloxine-saffron stain, 340 original magnification.

Figure 2. Ill-defined, erythematous, scaly patches on the palm and


fingers of the patients left hand.

Discussion
Deep dermatophytosis of the vulva is rare and has been
reported with T. mentagrophytes,1 Microsporum canis,2
Trichophyton verrucosum,3 and Trichophyton erinacei.4 We
report the second case of deep dermatophytosis of the
vulva caused by T. mentagrophytes. In the previously
reported case, the patient had a history of long-standing
topical steroid use for a nonspecific dermatitis. Our
patient, however, had no history of topical steroid use.

Canadian Dermatology Association | Journal of Cutaneous Medicine and Surgery, Vol 18, No 3 (May/June), 2014: pp 206209

207

Bougrine et al

Cases of deep dermatophytosis of the vulva have


been described to evolve from the use of combination
antifungal-steroid products2 and topical steroids1 on
existing superficial dermatophytosis or tinea cruris.
These agents may weaken the skin barrier and produce a
local immunosuppressive effect, allowing for deeper tissue
involvement.2 Of interest, our patient denied the use of
any topical treatments. In our case, we hypothesize that
shaving the pubic region facilitated the deep invasion by
the pathogens.
Although typically confined to the epidermis, dermatophytes may gain access to the dermis with mechanical
breakage of the epidermal barrier, such as that which
occurs with shaving.5 The dermis is more alkaline than the
epidermis and is thus not an ideal growth environment.
Disruption of follicles introduces keratinous material into
the dermis, which may act as a substrate on which the
organisms can thrive.6 Cellular destruction and inflammation secondary to dermatophytic infection may increase
the local acidity of the dermis, thereby favoring continued
proliferation.
As previously mentioned, our patient presented
erythematous, scaly patches on the digits of the left hand.
Superficial cultures revealed the presence of T. mentagrophytes, establishing tinea manuum as the likely source of
inoculation in this patient.
Confusing nosology and nomenclature have hampered
the understanding of deep dermatophytosis. Many different terms have been employed to describe these infections.
Majocchi granuloma, or trichophytic granuloma, is
defined as an uncommon intracutaneous or subcutaneous
granulomatous inflammation arising from local invasion
by a dermatophyte through a ruptured follicle in the
dermis.5
Two types of Majocchi granuloma have been described.
The superficial type usually presents as perifollicular
papules on the legs of otherwise healthy individuals,
frequently secondary to shaving.1,5,6 The deep form
consists of deep subcutaneous nodules occurring on the
scalp, face, or forearms of individuals with immunosuppression as a result of medication, alcoholism, cirrhosis,
diabetes mellitus, leukemia, lymphoma, or Cushing
syndrome.1,5,6
Kerion is considered by some to be a form of
trichophytic granuloma,2 the latter being a generic term
for fungal infections causing deep tissue involvement.
Kerion is characterized by the invasion of terminal hair
bearing skin. Some define kerion by its anatomic location,
namely the scalp and less commonly the beard.2,7
Clinically, kerion often presents acutely with boggy,
208

localized swelling. This infection results from an aggressive


inflammatory response to the organism. Likewise, tinea
profunda results from an excessive inflammatory response
to a dermatophyte infection and is thus analogous to
kerion but in anatomic sites devoid of terminal hair. It
may present with a granulomatous or verrucous morphology or may mimic cutaneous tuberculosis or even
squamous cell carcinoma.7 Thus, based on the clinical
and histologic picture, our case is best described as tinea
pubis, kerion type.
Systemic dissemination is rare but has been reported.6
A unique case of tinea profunda cysticum has been
reported with multiple subcutaneous cystic lesions on the
pubic region, thighs, back, and buttocks in a severely
immunosuppressed patient with T-cell deficiency. 8
Dermatophytes can also cause pseudomycetomas, consisting of tumefactions and subcutaneous abscesses without
formation of sinus tracts or granule discharge.9 This entity
may involve the scalp, the nape of the neck, or hairless
areas such as the palms.
Our case demonstrates once again the delay in diagnosis that often occurs with genital deep dermatophytic
infection, a rare infection by a common pathogen.1
Clinicians should remain alert to this entity when
evaluating suppurative infections of the vulva, which
may present as bacterial cellulitis unresponsive to antibiotic therapy, mycobacterial infection, or even neoplasia.
Diagnosis and treatment depend on adequate isolation and identification of the organism. This is best
performed by culture of a biopsy specimen. Epidermal
changes may not appear in this type of presentation, so
KOH examination of skin scrapings may prove falsely
negative. Fungal stains on routine histology may also
prove falsely negative, as in our experience. Finally,
treatment with oral antifungal therapy, such as itraconazole, should be initiated promptly to avoid irreversible
scarring alopecia.

Acknowledgment
Financial disclosure of authors and reviewers: None
reported.

References
1. Chang SE, Lee DK, Choi JH, et al. Majocchis granuloma of the vulva
caused by Trichophyton mentagrophytes. Mycoses 2005;48:38284,
doi:10.1111/j.1439-0507.2005.01147.x.
2. Margolis DJ, Weinberg JM, Tangoren IA, et al. Triphophytic
granuloma of the vulva. Dermatology 1998;197:6970, doi:10.1159/
000017961.

Canadian Dermatology Association | Journal of Cutaneous Medicine and Surgery, Vol 18, No 3 (May/June), 2014: pp 206209

Kerion of the Vulva Caused by T. mentagrophytes

3. Grunewald S, Paasch U, Graser Y, et al. Vernarbende Tinea profunda


des Mons pubis durch Trichophyton verrucosum. Hautarzt 2006;57:
8113, doi:10.1007/s00105-005-1066-7.
4. Feldmann R, Riegler E, Willinger B, et al. [54-year-old-woman with
perigenital follicular pustules]. J Dtsch Dermatol Ges 2011;24951.
5. Gupta S, Kumar B, Radotra BD, Rai R. Majocchis granuloma
trichyticum in an immunocompromised patient. Int J Dermatol
2000;39:14059, doi:10.1046/j.1365-4362.2000.00894.x.
6. Cho HR, Lee MH, Haw CR. Majocchis granuloma of the scrotum.
Mycoses 2007;50:5202, doi:10.1111/j.1439-0507.2007.01404.x.

7. Sobera JO, Elewski BE. Fungal diseases. In: Bolognia JL, Jorizzo JL,
Rapini RP, editors. Bolognia textbook of dermatology. 2nd ed. Spain:
Mosby Elsevier; 2008. p. 1177.
8. Kobayashi M, Ishida E, Yasuda H, et al. Tinea profunda cysticum
caused by Trichophyton rubrum. J Am Acad Dermatol 2006;54(2
Suppl):S113, doi:10.1016/j.jaad.2005.03.058.
9. Erbagci Z. Deep dermatophytoses in association with atopy and
diabetes mellitus: Majocchis granuloma tricophyticum or dermatophytic pseudomycetoma? Mycopathologia 2001;154:1639, doi:10.
1023/A:1016328001146.

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