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© 1996 The Journal of Bone and Joint Surgery, Inc. Leiomyoma of the Hand in a Child Who Has the Human Immunodeficiency Virus. A Case Report*  , NEW YORK N.Y.Investigation performed at The New York Hospital-Cornell Medical Center, New York City
Leiomyomas, which are benign tumors of smooth-muscle origin, rarely occur in the upper extremity6,16 and are very uncommon in children15. We describe the case of a painful leiomyoma of the hand in a child who was infected with the human immunodeficiency virus. Although the association of the human immunodeficiency virus with malignant neoplasms such as B-cell lymphomas and Kaposi sarcoma has been well documented11, a non-random association between smooth-muscle tumors and infection with the virus has been identified only recently1,3,10,17,18. Leiomyoma and leiomyosarcoma involving the lung and the gastrointestinal tract have been found in children infected with the human immunodeficiency virus1,3,10,17,18; to our knowledge, there have been no reports of a leiomyoma involving the upper extremity of such a child.
A ten-year-old boy, who was known to be infected with the human immunodeficiency virus, was seen because of a painful mass in the right (dominant) hand. The child had been born prematurely after twenty-six weeks of gestation. He had received multiple transfusions while in the neonatal intensive-care unit and was presumed to have contracted the virus through a transfusion with contaminated blood. The patient had numerous manifestations of the disease, including lymphocytic interstitial pneumonitis, oral candidiasis, lymphadenopathy, recurrent fevers, and a chronic cough. At the time, the patient was receiving INH (isoniazid), prednisone, AZT (azidothymidine), and griseofulvin. Physical examination revealed a reddish, well circumscribed, nodular lesion, seven millimeters in diameter, in the center of the right palm (Fig. 1). The lesion was tender to palpation; it had increased in size over the previous nine months and had become symptomatic within the past two months. No other masses were detected in the hand or the upper extremity. No epitrochlear or axillary adenopathy was evident. The findings of a neurovascular examination were normal. A radiograph of the chest demonstrated lymphocytic interstitial pneumonitis. The histological analysis of a biopsy specimen that had been obtained with a three-millimeter-diameter punch showed benign spindle-cell proliferation, and the patient was referred to us for excision of the lesion.
With use of general anesthesia and tourniquet hemostasis, a marginal excision of the lesion was performed. An elliptical incision was made transversely in line with the flexion creases of the palm. A thin cuff of subcutaneous fat was resected with the specimen. All bleeding vessels were meticulously cauterized, and the wound was closed primarily with use of non-absorbable monofilament suture. Pathological analysis of the specimen revealed a smooth, discrete nodule in the dermis. The nodule was blue-pink and had a firm, rubbery consistency. It was seven millimeters in diameter. Histological examination demonstrated a storiform architecture, composed of spindle cells with elliptical nuclei (Fig. 2 and 3). The specimen had the typical microscopic appearance of a leiomyoma. Intertwining bundles of smooth-muscle cells surrounded numerous vascular channels, and mitotic figures were scarce. No tumor was evident below the deep dermal tissue or in the margins of the specimen.
At the three-month follow-up examination, there was no evidence of local recurrence and there was no pain at the site of the excision. The patient had a full range of motion of the wrist and the fingers. There was no evidence of other masses elsewhere in the body.
Leiomyomas are benign tumors of smooth-muscle origin. Uterine leiomyomas are the most common tumor in women, being found in approximately one in four women during the reproductive years15. Leiomyomas of the hand, however, are extremely rare and arise from the only non-striated muscles in the upper extremity—the erector pill, the sweat glands, and the vascular walls12. Most of the reports on leiomyomas of the hand have included only one or two patients each2,5-8,14,16,21. Uchida et al. reported on eleven patients who had a leiomyoma in the upper extremity. Neviaser and Newman reported twenty-four leiomyomas of the hand and forearm in their review of eighty-five vascular leiomyomas13, which we believe to have been the largest series to date. In general, a leiomyoma occurs in the third and fourth decades of life15. The average age of patients who have had a leiomyoma in the hand has been reported to be thirty-nine to forty-six years13,19. The youngest patient to have had a leiomyoma reportedly was a fourteen-year-old child who had a leiomyoma in the proximal part of the arm19. The lesion is twice as common in women and girls as it is in men and boys; when it occurs in the extremities, it is more common in the leg, ankle, and foot than it is in the upper extremity16. The most characteristic subjective symptom of a leiomyoma is tenderness that evolves to pain, which is often paroxysmal6. However, Uchida et al. reported that leiomyomas in the hand usually are not painful but that lesions in the arm and the forearm commonly are. Operative excision is the treatment of choice and, if an adequate margin is obtained, recurrence is rare6. The association between neoplasms and infection with the human immunodeficiency virus is well known. In particular, there is a high prevalence of Kaposi sarcoma and aggressive B-cell lymphomas in adults who have the virus10. Hodgkin lymphoma; colon cancer; anorectal carcinoma; and squamous-cell carcinoma of the head, neck, and oropharynx also have been reported to occur in these patients9. However, neoplasms have been documented in fewer than 2 per cent of children who have acquired immunodeficiency syndrome3. B-cell lymphomas account for most malignant lesions related to acquired immunodeficiency syndrome in children4. Chadwick et al., in 1990, reported the cases of three children who had the human immunodeficiency virus in whom a leiomyoma or a leiomyosarcoma developed in either the lung or the gastrointestinal tract. On the basis of the findings in their patients, as well as those described in a report of another patient11, the authors proposed that, in children, there is a specific and non-random relationship between infection with the human immunodeficiency virus and spindle-cell tumors of smooth-muscle origin3. Subsequent reports of smooth-muscle tumors in children who have the human immunodeficiency virus have confirmed this association. McLoughlin et al., in 1991, described the case of a four-year-old girl who was infected with the virus and who had disseminated leiomyosarcoma of the small intestine, with nodular metastases in the lung and the brain. Sabatino et al. reported on a child who had the virus in whom pulmonary leiomyosarcoma and leiomyoma developed simultaneously. Balsam and Segal found multiple bronchial leiomyomas in a seven-year-old girl who had the human immunodeficiency virus, and Ross et al. found primary hepatic leiomyosarcoma in a child who had acquired immunodeficiency syndrome. Recently, smooth-muscle tumors also have been found in adults who have the syndrome20. Our findings were exceedingly unusual not only because the leiomyoma developed in the hand but also because the patient was only ten years old. To our knowledge, there has been no previous report of the occurrence of a leiomyoma in an extremity of a child who has the human immunodeficiency virus. Patients who have acquired immunodeficiency syndrome are at an increased risk for the development of certain neoplasms, especially lymphomas and Kaposi sarcoma. In addition, smooth-muscle tumors have been shown to be disproportionately represented among children who have this syndrome. Leiomyomas and leiomyosarcomas have been found in the gastrointestinal tract and the pulmonary system of children infected with the human immunodeficiency virus1,3,10,17,18. We have shown that these tumors also may develop in the hand.
*No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. No funds were received in support of this study.
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