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Journal of Dermatology & Dermatologic Surgery 20 (2016) 7881

Case report

Diuse intramuscular lipoma responsible for monomelic hypertrophy

Krich Sanaa a,, Meziane Mariame a, Amraoui Nissrine a, Abid Hatim b, Mrini Abdelmajid b,
Tizniti Siham c, Mernissi Fatima Zahra a
a
Dermatological Department, Hassan II University Hospital, Fes, Morocco
b
Department of Orthopaedic and Trauma Surgery, Hassan II University Hospital, Fes, Morocco
c
Radiological Department, Hassan II University Hospital, Fes, Morocco

Received 21 March 2015; accepted 28 July 2015

Available online 29 August 2015

Abstract

Background: Lipomas are common benign soft tissue, however intramuscular lipoma, is an often misdiagnosed clinical entity. The
aim of this case is to present an atypical inltrating lipoma and discuss its diagnostic and therapeutic features.
Clinical features: A 24 year-old patient presented to our consultation with a swelling of the right lower limb from childhood and grad-
ually increasing in size. Clinical examination revealed a rm and extended swelling along the leg, thigh and foot giving an aspect of
monomelic hypertrophy. There were no signs of vascular or nerve compression.
Intervention and outcome: Pathological study showed a lipoma. Magnetic resonance imaging (MRI) of the right lower limb objectied
a large inltrating lipoma aecting muscles of the posterior compartment of the thigh and the right leg with no signs of malignancy. The
diagnosis of intramuscular lipoma was done. The surgery was proposed but the patient refused.
Conclusion: This case report described an adult female patient with an unusually intramuscular lipoma where magnetic resonance
imaging was playing an important role in the diagnostic. Indeed the intramuscular is a benign fatty tumor with characteristic appearance
on MRI. Its management should be considered in multidisciplinary collaboration.
2015 The Authors. Production and hosting by Elsevier B.V. on behalf of King Saud University. This is an open access article under the CC BY-
NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

Keywords: Inltrating lipoma; Intramuscular; Monomelic hypertrophy; Magnetic resonance imaging

1. Introduction Intramuscular lipoma is a deep lipoma; its relatively

Lipoma is the most common form of soft-tissue tumor
(Kransdorf, 1995). It is composed of mature adipocytes
(Terzioglu et al., 2004) and is usually localized in the
subcutaneous tissue, but can also occur in deeper locations
(Munk et al., 1997).
Corresponding author. Tel.: +212 615458263.
E-mail address: krichsanaa@hotmail.fr (K. Sanaa).
Peer review under responsibility of King Saud University.
Production and hosting by Elsevier
rare and has been described in many locations, but has
been exceptionally reported as monomelic (Fedell et al.,
1978).
The etiology of this lipoma is unknown. Copcu argued
that in rare cases they may arise following trauma
(Copcu, 2003). This hypothesis was explained by the fact
that blunt trauma is always a cause of inammation in soft
tissues. Mediators secondary to inammation could play a
role in the dierentiation of precursors of mature adipo-
cytes (Mori et al., 2004) and can probably explain focal
lipoma. Another hypothesis was proposed by Mori et al.;
they suggested the association of neurogenic or myogenic
disorder in the lesions and that fat cell overgrowth into
areas of muscle involution could modulate the inltrative

http://dx.doi.org/10.1016/j.jdds.2015.07.005
2352-2410/ 2015 The Authors. Production and hosting by Elsevier B.V. on behalf of King Saud University.
This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
 K. Sanaa et al. / Journal of Dermatology & Dermatologic Surgery 20 (2016) 7881
Figure 1. A rm swelling extended to the posterior leg and thigh with
gluteal fold deviation.
growth of these tumors into the surrounding muscle (Mori
et al., 2004). In addition, sex hormones may inuence the
proliferation and dierentiation of adipocytes and thus
the formation of adipose tissue; the local estradiol concen-
tration in adipose tissue may be a stimulus to the prolifer-
ation of preadipocytes (Gregoire, 2001).
The aim of this article is to describe an exceptional case
of intramuscular lipoma responsible for monomelic hyper-
trophy, and describe its clinical features, dierential diag-
nosis, imaging characteristics and treatment options.
2. Case report
A 24-year-old woman was admitted in dermatological
department for a 14 year history of swelling of the right
lower limb. The patient reported that the lesion has begun
in the right foot at the age of 10 years, was removed by sur-
gery but had recurred 1 year later. The swelling had
increased gradually in size to involve the entire lower limb
(LL). She denies a history of trauma or cancer.
Physical examination noticed a rm and painless swel-
ling of the sole of the right foot predominantly on the prox-
imal part which extended to the posterior leg and thigh
with gluteal fold deviation giving an aspect of monomilic
hypertrophy (Fig 1). We also found two cafe au lait spots
on the trunk. There were no signs of vascular or nerve com-
pression. Laboratory investigation revealed a normal level
of plasma lipid.
79
Pathological examination of the foot objectived typical
and matures adipocytes interspersed with skeletal muscle
bundles (Fig. 2). Magnetic resonance imaging (MRI) of
the right LL showed two large intramuscular lipomas,
involving the muscles of the posterior compartment of
the thigh and the right leg with no signs of malignancy
(Fig. 3a and b). The diagnosis of intramuscular lipoma
was made. A surgical therapy was proposed, but the
patient refused.
3. Discussion
Our case described an unusual case of intramuscular
lipoma from childhood with monomelic hypertrophy.
Intramuscular lipoma was rst described by Paget in the
trapezius muscle in 1856 (Kindblom et al., 1974). This sub-
type of lipoma is rare (1%) and is characterized by inltra-
tion of the muscle tissues or growth between the muscle
bers (Rosai, 1996; Batsakis, 1979). They occur without
gender predilection, generally during middle or late
adulthood (majority between 40 to 60 years) and began
rarely in childhood as in our case (Bjerregaard et al.,
1989; Dionne and Seemayer, 1974).
Intramuscular lipoma is often presented without clinical
symptoms and therefore grows to a large size before detec-
tion unless they compress neurovascular structures
(Terzioglu et al., 2004; Nigri et al., 2008). The remarkable
characteristics of this subtype of lipoma are large dimen-
sions at diagnosis (over than 5 cm), a deep subfascial loca-
tion and a predilection for the large muscle groups of the
shoulder, upper arm, hip and thigh (Kindblom et al.,
1974). They are not always as well circumscribed as the
supercial type (Fletcher and Martin-Bates, 1988).
Monomelic distribution is exceptional, to our knowl-
edge just one case was described in English literature; it
was a report of a girl of 12 years but hypertrophy of her
lower right limb was less developed as in our case (Fedell
et al., 1978).
Several dierential diagnoses must be excluded (Ohguri
et al., 2003). In our case, because of monomelic hypertro-
Figure 2. Mature adipocytes interspersed with skeletal muscle bundles.
80 K. Sanaa et al. / Journal of Dermatology & Dermatologic Surgery 20 (2016) 7881
Figure 3. Coronal T1-weighted (a) and axial T2-Weighted (b) MR imaging showing voluminous lipoma inltrating the posterior compartment of the leg
containing streaky structures corresponding to muscular bers of gastrocnemius muscle.
phy of the LL and the aspect of the foot, we rst eliminated
Proteus syndrome and neurobromatosis by the absence of
other diagnostic criteria.
Radiologically, standard radiography reveal osseous
and cartilaginous changes within the masses, but MRI is
the gold standard for assessing soft tissue masses and elim-
inates liposarcoma (Matsumoto et al., 1999). On MRI
intramuscular lipoma may appear homogeneous or inho-
mogeneous depending upon the amount of intermingled
muscle bers within the fatty mass. In most cases the mar-
gin of the lesion is inltrative but 60% of cases may still
appear to be rather well dened (Matsumoto et al.,
1999). As in our case, on T1 and T2 -weighted images
the lesion demonstrates high signal intensity, similar to
normal fat. Fat-suppressed sequences demonstrate signal
suppression, again similar to normal fat. Linear structures
with low signal intensity on both the T1 and T2- weighted
sequences are seen in most lesions, and represent muscle
bers within the tumor.
Detailed histological examination is essential to charac-
terize intramuscular lipomas and to eliminate liposarcoma
(Matsumoto et al., 2000). It shows mature uni-vacuolated
adipocytes of uniform size interspersed with skeletal muscle
bundles and varying amounts of brous stroma which
forms incomplete capsules in some cases (Kindblom
et al., 1974). In places the fatty tumor completely replaces
the muscle bundles. The fatty cells grow into the endomy-
sial space, and remaining muscle bers show extensive
damage without atypical nucleus, mucous degeneration,
polymorphism, and mitosis (Fletcher and Martin-Bates,
1988).
Surgical excision is the best way to treat intramuscular
lipoma. However, the recurrence rate for inltrating lipo-
mas has been reported to range between 3% and 62.5%
(Su et al., 2011). This dierence may be explained by inad-
equate resection of the tumor or misdiagnosing it as an
intramuscular lipoma or by the fact that this type of lipoma
is not always well circumscribed.
Indications for excision include a mass greater than 5 cm
in size, a subfascial location, a tumor that is growing, clin-
ical features such as pain, rmness, or irregularity, or a
questionable preoperative needle biopsy result. Another
reason for excision includes a patients cosmetic concern
(Serpell and Chen, 2007). In our case, despite the large size
of the tumors and its appearance the patient refused sur-
gery because of risk of recurrence.
We think that lipoma in our patient can be explained by
the hormonal theory because of the occurrence of lesions
during the prepubertal period; but the presence of unilat-
eral involvement could be explained by the presence a local
factor may be exacerbated by surgery on the right foot dur-
ing childhood.
4. Conclusion
Our case described unusual unilateral and large intra-
muscular lipoma located on the muscles of the posterior
compartment of the thigh and the right leg where imaging
was playing an important role in the diagnosis.
Conict of interest
None declared.
Financial support
None.
References
Batsakis, J.G., 1979. Tumors of the Head and Neck: Clinical and
Pathological Considerations, second ed. Williams and Wilkins, Balti-
more, pp. 360364.
Bjerregaard, P., Hagen, K., Daugaard, S., Kofoed, H., 1989. Intramus-
cular lipoma of the lower limb. J. Bone Joint Surg. Br. 71, 812815.
Copcu, E., 2003. Can intramuscular lipoma have a post-traumatic origin?.
Br. J. Dermatol. 149 10771094.
 K. Sanaa et al. / Journal of Dermatology & Dermatologic Surgery 20 (2016) 7881
Dionne, G.P., Seemayer, T.A., 1974. Inltrating lipomas and angiolipo-
mas revisited. Cancer 33, 732738.
Fedell, M.R., Duy, P.E., Rowland, L.P., 1978. Inltrating lipoma: a
cause of monomelic hypertrophy. Muscle Nerve 1 (1), 7580.
Fletcher, C.D., Martin-Bates, E., 1988. Intramuscular and intermuscular
lipoma: neglected diagnoses. Histopathology 12, 275287.
Gregoire, F.M., 2001. Adipocyte dierentiation: from broblast to
endocrine cell. Exp. Biol. Med. 226, 9971002.
Kindblom, L.G., Angervall, L., Stener, B., Wickbom, I., 1974. Intermus-
cular and intramuscular lipomas and hibernomas. A clinical,
roentgenologic, histologic, and prognostic study of 46 cases. Cancer
33, 754762.
Kransdorf, M.J., 1995. Benign soft-tissue tumors in a large referral
population: distribution of specic diagnosis by age, sex, and location.
Am. J. Roentgenol. 164, 395402.
Matsumoto, K., Hukuda, S., Ishizawa, M., et al., 1999. MRI ndings in
intramuscular lipomas. Skeletal Radiol. 28, 145152.
Matsumoto, K., Hukuda, S., Ishizawa, M., Egawa, M., Okabe, H., 2000.
Liposarcoma associated with multiple intramuscular lipomas. Clin.
Orthop. Relat. Res. 373, 202207.
Mori, K., Chano, T., Matsumoto, K., Ishizawa, M., Matsusue, Y., Okabe,
H., 2004. Type-selective muscular degeneration promotes inltrative
growth if intramuscular lipoma. BMC Musculoskelet. Dis. 5, 20.
81
Munk, P.L., Lee, M.J., Janzen, D.L., Connell, D.G., Logan, P.M., Poon,
P.Y., Bainbridge, T.C., 1997. Lipoma and liposarcoma: evaluation
using CT and MR imaging. AJR Am. J. Roentgenol. 169 (2), 589594.
Nigri, G., Dente, M., Valabrega, S., Beccaria, G., Aurello, P., DAngelo,
F., Di Marzo, F., Ramacciato, G., 2008. Giant inframuscular lipoma
disclosed 14 years after a blunt trauma: a case report. J. Med. Case
Rep. 2, 318.
Ohguri, T., Aoki, T., Hisaoka, M., et al., 2003. Dierential diagnosis of
benign peripheral lipoma from well-dierentiated liposarcoma on MR
imaging: is comparison of margins and internal characteristics useful?
AJR Am. J. Roentgenol. 180 (6), 16891694.
Rosai, J., 1996. Akermans surgical pathology, eighth ed. Mosby, St.
Louis, pp. 20532055.
Serpell, J.W., Chen, R.Y., 2007. Review of large deep lipomatous
tumours. ANZ J. Surg. 77 (7), 524529.
Su, Ch., Hung, J.K., Chang, I.L., 2011. Surgical treatment of intramus-
cular, inltrating lipoma. Int. Surg. 96, 5659.
Terzioglu, A., Tuncali, D., Yuksel, A., Bingul, F., Aslan, G., 2004. Giant
lipomas: a series of 12 consecutive cases and a giant liposarcoma of the
thigh. Dermatol. Surg. 30 (3), 463467.