Introduction
Dupuytren’s disease is uncommon in childhood, though scattered cases have been reported in the literature. Most have involved the palm and ulnar-sided digits, with a single report involving the thumb FPL tendon sheath1. We present a unique case of Dupuytren’s disease, in a child, arising from the lateral digital sheet of the thumb, causing a flexion contracture of the IPJ.
Case:
A 14-year-old female with a family history of Dupuytren’s disease presented to the clinic with a de-novo, progressive swelling on the radial-volar aspect of the thumb IPJ. This was associated with a flexion deformity of the IPJ. The swelling had progressively grown over several months, and was painful, tender and occasionally erythematous. Intra-operatively, a fibrous mass was identified arising from the lateral digital sheet, radial to the radial neurovascular bundle, and extending distally towards the insertion of the radial collateral ligament onto the distal phalanx. Histopathology revealed a nodular, soft tissue mass comprised of bland fibroblastic cells, without atypia or mitoses; findings consistent with Dupuytren’s disease.
Discussion:
We report the first documented case of paediatric Dupuytren’s disease involving the lateral fibrous structures of the thumb interphalangeal joint. Dupuytren’s disease in the adult population involves the ring and little fingers most commonly, followed by the thumb2. In the long digits, involvement of the lateral digital sheet, Grayson’s ligament and a spiral cord may all contribute to disease found lateral to the neurovascular bundle. In rare cases where contractures of the thumb IPJ have been reported, most were described as arising from central structures, in particular a pre-tendinous band or the FPL tendon sheath1, 3. This novel case exemplifies the breadth of structures that may be involved in Dupuytren’s disease, even in the paediatric population.