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Infantile digital fibromatosis after web construction in syndactyly
Insights
Infantile digital fibromatosis presented unusually in a young girl after surgery for syndactyly. Recurrent nodules at the skin graft site led to diagnosis via characteristic inclusion bodies.
Area of Science:
- Pediatric Pathology
- Dermatology
- Surgical Oncology
Background:
- Infantile digital fibromatosis (IDF) is a rare benign soft tissue tumor affecting children.
- IDF typically presents as solitary or multiple nodules on the digits.
- Unusual presentations of IDF, particularly post-surgical recurrence, warrant detailed case reporting.
Observation:
- A case of a Japanese girl with simple syndactyly of the right ring and little fingers.
- Post-operative development of multiple nodules at the skin graft edge three months after surgical correction.
- Recurrence of nodules following initial excision, necessitating extensive tumor removal at age eight.
Findings:
- Histopathological examination revealed intracytoplasmic inclusion bodies.
- The presence of these inclusion bodies confirmed the diagnosis of infantile digital fibromatosis.
- The unusual onset and recurrence pattern highlight potential challenges in managing IDF.
Implications:
- This case underscores the importance of considering IDF in the differential diagnosis of digital nodules, even with atypical presentations.
- Understanding the behavior of IDF, including its potential for recurrence, is crucial for effective clinical management.
- Further research into the pathogenesis of IDF may elucidate mechanisms underlying its unusual onset and recurrence patterns.
Abstract:
A case of infantile digital fibromatosis with an unusual onset is reported. A Japanese girl, with a simple syndactyly of the right ring and little fingers, had an operation at the age of 2 1/2 years. Three months after the operation, multiple nodules appeared at the skin graft edge. The nodules were excised but soon recurred. When the patient was 8 years old, a large-scale excision of the tumors was performed. Intracytoplasmic inclusion bodies were shown by phosphotungstic acid-hematoxylin stain, and the case was diagnosed as infantile digital fibromatosis.