ARTICLE
Auteur(s) : Haruko Uemura, Sinichi
Koba, Masakazu Otu, Noriyuki Misago, Yutaka Narisawa
Division of Dermatology, Department of International
Medicine, Faculty of Medicine, Saga University,
5-1-1 Nabeshima, Saga city, Saga 849-8501, Japan
Leiomyoma is a relatively well-known, smooth muscle-derived
tumor, accounting for approximately 4% of benign soft-tissue tumors
[1]. Cutaneous soft-tissue leiomyomas are classified according to
their localization into superficial leiomyoma, which arises from
the skin or superficial subcutaneous layer and is commonly
encountered in dermatological practice, and deep soft-tissue
leiomyoma (DSTL) which originates from the deep subcutaneous layer.
Superficial leiomyomas include pilar leiomyoma arising from the
erector pili muscle, angioleiomyoma from vascular smooth muscle,
and genital leiomyoma from the smooth muscle of the scrotum, vulva,
or nipple. DSTL arising in subcutaneous soft tissue are very rare.
We report the case of a DSTL that arose in the thigh.
A 52-year-old woman first noticed a subcutaneous mass in the
left thigh about one month prior to the first examination. In the
left femoral region, there was an elastic-soft, subcutaneous mass,
4 cm in diameter, without skin changes. There was no
spontaneous pain, tenderness, or radiating pain, and the mass was
mobile. She did not have other muscular tumors or family history.
MRI revealed a 38×25×19-mm well-defined, subcutaneous mass
partially in contact with the fascia in the deep layer of
subcutaneous adipose tissue in the left anterior thigh. At surgery,
a well-defined, smooth-surfaced mass was observed in the deep layer
of adipose tissue (figure 1A). The mass was
not adherent to the surrounding tissue. There was no relapse.
Histopathologically, the tumor was well-demarcated from the
surrounding tissue, and composed of irregularly intertwining
bundles of spindle-shaped cells with eosinophilic cytoplasm and
fusiform nuclei (figure
1B). A “halo”, characteristic of smooth muscle cells,
was also observed (figure 1C). No features of
malignancy were noted. Immunohistochemistry showed that the tumor
cells were positive for α-SMA, desmin, and vimentin, and negative
for S-100 protein (figures 1D-F). The
histopathological and immunohistochemical findings led to a
diagnosis of deep soft-tissue leiomyoma.
Leiomyoma is a benign smooth muscle tumor with a predilection
for the uterus and skin, and which very rarely arises in deep soft
tissue. Clinically, superficial leiomyomas, which are primarily
encountered in dermatological practice, arise in the dermis and
upper subcutaneous adipose tissue, averaging less than 2 cm in
diameter [2]. On the other hand, DSTL present as a well-defined,
elastic-firm mass, affect persons with a mean age of 37 years, with
a male-to-female ratio of about 1:1, have a predilection for the
lower extremities, and, compared with superficial leiomyomas,
present as a larger mass with a mean diameter of 6.5 cm [3,
4]. They are characteristically painful, and must be differentiated
from other painful tumors such as glomus tumors, angiolipomas,
spiradenomas and schwannomas. In this patient, the subcutaneous
mass was well-defined and was located in the deep subcutaneous
adipose tissue and it was a smaller-than-average size for a
leiomyoma; therefore, the preoperative diagnosis of leiomyoma was
difficult.
Histopathologically, DSTL is well-demarcated and composed of
irregularly intertwining bundles of spindle-shaped cells with
eosinophilic cytoplasm and blunt-ended, fusiform nuclei. Hyaline
material is present in the stroma in many patients [5], and
calcification is observed in more than 50% of patients [6].
Immunohistochemically, these leiomyomas are positive for vimentin,
desmin, and α-SMA, as observed in this patient.
In contrast to superficial leiomyoma, the pathogenesis of DSTL
has not been previously discussed and remains unclear. DSTL may
originate from several large vessels that consist of smooth muscles
in their walls. But in this patient, there were no characteristic
features of frequently encountered angioleiomyoma, such as the
angiocentric proliferation of smooth muscle cells, and the simple
proliferation of smooth muscle cells was observed. These features
were relatively similar to those of leiomyomas in other organs,
such as uterine myoma, and we speculate that the origin of DSTL may
be the ectopic smooth muscles.
Acknowledgements
Conflict of interest: none. Financial support: none.
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