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A Rare Location and Unusual Development: Antecubital Leiomyoma.

INTRODUCTION

Leiomyomas are tumors derived from smooth muscle. They represent 4.4% of all benign soft-tissue neoplasms. [1] Leiomyomas are rarely seen in the extremities. Leiomyomas are classified into three groups: cutaneous leiomyomas that arise from the erector pili muscle; vascular leiomyomas that arise from smooth muscle of the vein; and leiomyomas of deep soft tissues. [2] We report a case of leiomyoma that developed on antecubital fossa with an unusual onset.

CASE REPORT

A 52.year.old man presented with a palpable nearly 4 cm x 3 cm, firm, mobile mass on the volar surface of his right forearm present that was present for approximately 3 months [Figure 1]. He had no history of any drug use or comorbidity. He did not complain any pain or tenderness of the mass. On physical examination, both motor and sensory evaluation was normal. Routine blood investigations were within normal limits. The patient declared that he had an intravenous (i.v.) catheterization procedure on the same area which the tumor had developed on approximately 6 months ago. The patient was further evaluated magnetic resonance imaging (MRI). On imaging, a well-circumscribed antecubital fossa.located 5 cm x 4 cm x 3 cm mass was determined on his right elbow MRI with heterogeneous appearance. The tumor was well capsulated and did not involve muscles, local vasculature, or nerves. After obtaining informed consent,the patient underwent surgery. It was easily dissected from the surrounding tissue along with its capsule and removed entirely [Figure 2]. The resected tumor was sent to laboratory for histopathological assessment. Initial microscopic examination revealed a well-circumscribed lesion composed of spindle cells arranged in a whorled pattern. The tumor cells contained elongated nuclei with a moderate amount of eosinophilic cytoplasm. There was no evidence of necrosis, mitoses, or nuclear atypia. Further, immunohistochemical (IHC) staining showed strong reactivity for vimentin, smooth muscle actin (SMA), and desmin and a negative reaction against S.100 that favored a diagnosis of leiomyoma. IHC using antibodies directed against SMA and S.100 protein showed diffuse immunoreactivity for SMA and negative for S.100 [Figures 3 and 4]. On 1.year follow-up of the patient, there was no evidence of recurrence.

DISCUSSION

Leiomyomas are benign soft-tissue neoplasms which are originated from smooth muscle cells. Leiomyomas predominantly occur in adolescence and early adulthood ages even though they can emerge at any age. [1] They are classified as cutaneous leiomyomas, vascular leiomyomas, and leiomyomas of soft tissues. Leiomyomas of the limbs are divided into superficial and deep soft-tissue tumors. Superficial tumors include both cutaneous and subcutaneous lesions. Cutaneous and subcutaneous leiomyomas are independent of age and that there is no predisposition by race or sex. Vascular and deep soft-tissue leiomyomas are more common in the second and third decades of life with a female preponderance. [2] Leiomyomas rarely occur in extremities and are more common the lower limb than in the upper extremity. [3] The deep soft-tissue leiomyomas are further classified into vascular (originating from vessel wall smooth muscle) and nonvascular tumors. [4] It can usually be seen anywhere the smooth muscle exists, including generally the uterus, gastrointestinal tract, urinary tract, skin, and mucous membranes. [5] Leiomyomas in the hand are usually not painful, but that lesions in the arm and the forearm commonly are. Pain in these cases may be because of contraction of smooth muscles or due to compression of cutaneous nerves because of tumor. [6] Since the mass probably did not compress any nerve, there was no pain in our patient.

Our findings for this case were unusual that not only leiomyomata are not commonly found in the elbow but also are particularly uncommon as developing after an i.v. catheter procedure. Two theories have been postulated for the pathogenesis of this rare tumor. Goodman and Bryggs have believed that deep leiomyomas arise from undifferentiated mesenchymal cells or smooth muscle rests. Stout has suggested that these tumors may instead arise from the smooth muscle in the walls of blood vessels. [7,8] We suppose that, in our case, i.v. injection might have initiated leiomyoma formation in the blood vessel smooth muscles as Stout mentioned. Before our report, we could not access any reported previous case of leiomyoma on both forearm and which have developed following an i.v. injection. In our case, leiomyoma has formed on antecubital fossa and after an i.v. catheterization procedure.

Operative excision is the treatment of choice in these cases for both pathologic diagnosis and definite treatment. Billings et al. reported neither recurrences nor metastases after excision of somatic soft-tissue leiomyomas during a mean follow-up of 58.7 months. [9] The imaging features of deep soft-tissue leiomyomas are nonspecific and similar to those of many other soft-tissue neoplasms. The radiological differential diagnosis includes lipoma, leiomyosarcoma, schwannoma or neurofibroma, vascular malformation, hemangioma, and soft-tissue giant.cell tumor of the tendon sheath. [10] Leiomyoma is a disease that has a possibility to reoccur in the long term, and for this reason, a follow-up is absolutely mandatory. [11]

CONCLUSION

The case was presented due to its very rare location and extraordinary pathway to originate from. Soft-tissue leiomyoma of the upper extremity is uncommon, and its appearance in the forearm constitutes a rare issue. It should also be considered in the differential diagnosis of any solitary masses of the extremities.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

REFERENCES

[1.] Lopez.Barea F, Rodriguez.Peralto JL, Burgos E, Gonzalez.Lopez J. Calcified leiomyoma of deep soft tissue. Report of a case in childhood. Virchows Arch 1994;425:217-20.

[2.] Bommireddy B, Gurram V. Deep soft tissue leiomyoma of forearm: A case report and review of literature. J Clin Diagn Res 2016;10:RD03-5.

[3.] Arenas AJ, Urbiola E, Pampliega T, Cordoba A. Deep intramuscular leiomyoma of the lower limb. Acta Orthop Belg 1993;59:211-3.

[4.] Ramachandran R, Rangaswami R, Raja DK, Shanmugasundaram G. Deep soft-tissue leiomyoma of the forearm mimicking a primary bone tumor of the ulna. Radiol Case Rep 2014;9:960.

[5.] James WD, Berger TG, Elston DM. Dermal and subcutaneous tumors. In: Andrews' Diseases of the Skin Clinical Dermatology. 10th ed. Philadelphia: Saunder Elsevier; 2006. p. 626-7.

[6.] Uchida M, Kojima T, Hirase Y, Iizuka T. Clinical characteristics of vascular leiomyoma of the upper extremity: Report of 11 cases. Br J Plast Surg 1992;45:547-9.

[7.] Goodman AH, Br.ggs RC. Deep le.omyoma of an extrem.ty. J Bone Joint Surg Am 1965;47:529-32.

[8.] Stout AP. Solitary cutaneous and subcutaneous leiomyoma. Am J Cancer 1937;29:435-69.

[9.] Billings SD, Folpe AL, Weiss SW. Do leiomyomas of deep soft tissue exist? An analysis of highly differentiated smooth muscle tumors of deep soft tissue supporting two distinct subtypes. Am J Surg Pathol 2001;25:1134-42.

[10.] Weiss SW, Goldblum JR, Folpe AL. Benign tumors of smooth muscle. In: Enzinger and Weiss's Soft Tissue Tumors. 5th ed. St Louis, MO: Mosby; 2008. p. 517-44.

[11.] Badeloe S, Frank J. Clinical and molecular genetic aspects of hereditary multiple cutaneous leiomyomatosis. Eur J Dermatol 2009;19:545-51.

Alper Ural, Fatma Bilgen, Mehmet Bekerecioglu, Sevgi Bakaris (1)

Departments of Plastic, Reconstructive and Aesthetic Surgery and (1) Pathology, Faculty of Medicine, Kahramanmaras Sutcu Imam University, Kahramanmaras, Turkey

Address for correspondence: Dr. Alper Ural, Department of Plastic, Reconstructive and Aesthetic Surgery, Faculty of Medicine, Kahramanmaras Sutcu Imam University, Kahramanmaras, Turkey.

E.mail: alper_ural@yahoo.com

How to cite this article: Ural A, Bilgen F, Bekerecioglu M, Bakaris S. A rare location and unusual development: Antecubital leiomyoma. Turk J Plast Surg 2019;27:84-6.

DOI: 10.4103/tjps.tjps_72_18
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Title Annotation:Case Report
Author:Ural, Alper; Bilgen, Fatma; Bekerecioglu, Mehmet; Bakaris, Sevgi
Publication:Turkish Journal of Plastic Surgery
Article Type:Clinical report
Date:Apr 1, 2019
Words:1344
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