The Journal of Foot & Ankle Surgery 51 (2012) 342–344
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Fibroma of the Flexor Hallucis Longus Tendon Sheath Sang Wha Kim, MD 1, So Young Lee, MD 2, Sung-No Jung, MD, PhD 3, Won Il Sohn, MD 4, Ho Kwon, MD, PhD 5 1
Fellow, Department of Plastic Surgery, College of Medicine, The Catholic University of Korea, Uijongbu, Korea Resident, Department of Plastic Surgery, College of Medicine, The Catholic University of Korea, Uijongbu, Korea 3 Associate Professor, Department of Plastic Surgery, College of Medicine, The Catholic University of Korea, Uijongbu, Korea 4 Clinical Assistant Professor, Department of Plastic Surgery, College of Medicine, The Catholic University of Korea, Uijongbu, Korea 5 Professor, Department of Plastic Surgery, College of Medicine, The Catholic University of Korea, Uijongbu, Korea 2
a r t i c l e i n f o
a b s t r a c t
Level of Clinical Evidence: 4 Keywords: collagen foot neoplasm spindle cells surgery tumor ultrasound
Fibroma of tendon sheath is a rare benign tumor that usually occurs in upper extremities. It is mostly asymptomatic and grows slowly within the tendons or tendon sheaths. Histopathologic findings show welldemarcated nodules consisting of haphazardly arranged fibroblast-like spindle cells, which are embedded in a dense, collagenous matrix. We present a patient with fibroma of the tendon sheath on the flexor hallucis longus tendon, which was in an unusual location and has never been reported. The lesion was completely excised and showed no evidence of recurrence after 2 years of follow-up. Ó 2012 by the American College of Foot and Ankle Surgeons. All rights reserved.
Fibroma of tendon sheath is a rare benign tumor that usually occurs in the upper extremity, particularly in fingers (49%), hands (21%), and wrists (12%) (1). It is observed as a slowly growing, nontender, protruding nodule closely adhered to tendons or tendon sheaths. Histologically, it is observed as a mass with a distinct boundary and is occasionally lobulated, consisting of hyalinized fibrous matrix and spindle cells. We present a patient with fibroma of tendon sheath that developed in the flexor hallucis longus tendon. We wish to emphasize its unusual location through this case report and review of the literature. Case Report A 42-year-old male patient visited our clinic with a nodule in the right plantar area that had been growing slowly for 12 months. Above the lesion, the skin was normal with no signs of inflammation. Physical examination revealed a 1 1 cm oval-shaped subcutaneous nodule protruding in the right plantar area, which was observed only when the patient fully extended his toes (Fig. 1). The nodule was not protruded when the foot was in a neutral position or when the patient flexed his toes. There was no restriction of movements. However, the patient felt some discomfort while palpating the mass Financial Disclosure: None reported. Conflict of Interest: None reported. Address correspondence to: Sung-No Jung, MD, PhD, Associate Professor, Department of Plastic Surgery, Uijongbu St. Mary’s Hospital, College of Medicine, Catholic University of Korea, 65-1 Kumoh-Dong, Uijongbu, 480-135, Korea. E-mail address:
[email protected] (S.-N. Jung).
and walking. Plain radiography of the right foot was normal. Ultrasonography examination showed a 0.7 0.9 cm hypoechoic mass on the flexor hallucis longus tendon (Fig. 2). While the patient was under general anesthesia, a zigzag incision was performed to reveal a well-circumscribed 0.8 0.7 0.6 cm mass, which was firmly adhered to the flexor hallucis longus tendon. The mass was excised without any injury to the tendon (Fig. 3). Histological examination demonstrated the mass to be fibroma of tendon sheath, which consisted of spindle cells distributed irregularly within the dense collagenous matrix and several slit-like vascular structures (Fig. 4). There was no recurrence during 2 years of follow-up. Discussion Fibroma of the tendon sheath was first described by Geschickter and Copeland in 1936 as “fibrotic neoplasm” or “reactive fibrosis” (2). In 1979, Chung and Enzinger (3) summarized the characteristics of their clinical and pathohistological findings, and afterward, many cases were reported in the literature. Fibroma of the tendon sheath can occur at any age, usually between the ages of 20 and 40 years. Men are more commonly affected than women, with a ratio of 1.5:1 to 3:1 (1). About 75% to 82% of the fibroma presents in the upper extremities, most commonly in the fingers, hands, and wrists (1). Symptoms of nerve compression of the distal forearm have been described, which presents as median nerve neuropathy (4). Its occurrence in the foot is rare and, as far as we know, its development in the flexor hallucis longus tendon has never been reported. Macroscopically, fibroma of tendon sheath is observed as a graywhite or pearl-colored, hard nodule with a distinct boundary,
1067-2516/$ - see front matter Ó 2012 by the American College of Foot and Ankle Surgeons. All rights reserved. doi:10.1053/j.jfas.2012.02.002
S.W. Kim et al. / The Journal of Foot & Ankle Surgery 51 (2012) 342–344
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Fig. 1. Subcutaneous nodule protruded (arrow) when patient extended his toes.
Fig. 3. A well-circumscribed 0.8 0.7 0.6 cm mass, which was firmly adhered to the flexor hallucis longus tendon.
closely adhered to tendon or tendon sheath. Diverse sizes from 0.5 to 5.5 cm have been reported; in most cases it is smaller than 2 cm. Most are asymptomatic, although tenderness and mild pain present in 31% of patients. Histopathologically, fibroma of tendon sheath is a subcutaneous nodule with small lobules with slitlike vascular channels consisting of matrix with hyalinized collagen fibers and spindle cells. As for the etiology of fibroma of tendon sheath, reactive fibrous proliferation or tumorous changes have been considered, but it is not clear. An association with trauma has been reported in approximately 10% of cases (1). Recently, it also has been reported that tumorous changes caused by chromosomal abnormality of 2:11 translocation may induce fibroma of tendon sheath (5).
The diagnosis of fibroma is based on the patient’s history and clinical examination; however, radiologic studies such as magnetic resonance imaging or ultrasonography examination may be useful. Plain radiography findings are usually negative, except when a large mass compresses the surrounding muscles or fat, or when erosive bony changes are present (1). Differential diagnoses include circumscribed fibromatosis, nodular fasciitis, neurofibroma, leiomyoma, scar tissue, giant cell tumor of tendon sheath, and fibrous histiocytoma. Distinction from nodular fasciitis and fibrous histiocytoma is relatively straightforward because fibroma is lobulated and not nodular. The distinctly circumscribed margins of the fibroma exclude fibromatosis. Clinically, differentiating from a giant cell tumor of tendon sheath is important. It has been reported that the age for onset
Fig. 2. Ultrasonography examination shows a 0.7 0.9 cm hypoechoic mass on flexor hallucis longus tendon.
Fig. 4. Hematoxylin and eosin stain, 200. Tumor cells appear to be spindle shaped and surrounded by abundant collagenic matrix.
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of fibroma of tendon sheath is younger than that of giant cell tumor of tendon sheath; it also occurs more frequently in men. However, it is difficult to differentiate the clinical findings only. Histopathologically, fibrosis of the matrix of fibroma is more severe than a giant cell tumor of tendon sheath. In addition, spindle cells are more abundant, the frequency of giant cells is low, and foam cells or siderophages are absent in fibroma. Thus, it could be differentiated from a giant cell tumor of tendon sheath (6). Interestingly, Satti (7) suggested that fibroma of tendon sheath is the end-stage or sclerosing stage of a giant cell tumor, which is probably the consequence of progressive vascular impairment. Treatment for fibroma of tendon sheath is simple resection; however, its recurrence rate is 18% to 24%, which is relatively high (6). It is considered that such a high recurrence rate is due to the difficulty of complete resection owing to its firm adherence to tendons, tendon sheaths, and nerve. Therefore, complete resection with caution should be performed (1). The malignant transformation has not been reported. In our case, we experienced fibroma of tendon sheath in an unusual location, namely the flexor hallucis longus tendon. Because
the development of fibroma of tendon sheath on the flexor hallucis longus tendon has not been reported, we report our case with a review of the literature. References 1. Ciatti R, Mariani PP. Fibroma of tendon sheath located within the ankle joint capsule. J Orthop Traumatol 10(3):147–150, 2009. 2. Copeland MM, Geschickter CF. Malignant bone tumors: primary and metastatic. CA Cancer J Clin 13:232–238, 1963. 3. Weiss SW, Goldblum JR. Enzinger and Weiss’s Soft Tissue Tumors, ed 5, Mosby, St. Louis, 2007. 4. Bertolotto M, Rosenberg I, Parodi RC, Perrone R, Gentile S, Rollandi GA, Succi S. Case report: fibroma of tendon sheath in the distal forearm with associated median nerve neuropathy: US, CT and MR appearances. Clin Radiol 51(5):370–372, 1996. 5. Dal Cin P, Sciot R, De Smet L, Van den Berghe H. Translocation 2; 11 in a fibroma of tendon sheath. Histopathology 32(5):433–435, 1998. 6. Maluf HM, DeYoung BR, Swanson PE, Wick MR. Fibroma and giant cell tumor of tendon sheath: a comparative histological and immunohistological study. Mod Pathol 8(2):155–159, 1995. 7. Satti MB. Tendon sheath tumours: a pathological study of the relationship between giant cell tumour and fibroma of tendon sheath. Histopathology 20: 213–220, 1992.