Bilateral Proximal Delta Phalanges: An Unusual Presentation of Familial Congenital Clinodactyly

Abstract

We report a unique case of bilateral proximal delta phalanges with secondary distal accessory ossification centers of the long fingers in a 16-month-old boy. The bony anomalies presented as severe ulnar deviation at the proximal interphalangeal joints bilaterally. This is a unique occurrence of congenital clinodactyly which has not been previously reported. Several family members show similar deformity, although to lesser degrees, suggesting an autosomal dominant mode of inheritance with incomplete penetrance and variable expressivity.

Surgical Management of a Rare Case of Clinodactyly with Delta Phalanx in a Pediatric Patient

“…Optimal results have been observed in children who have the surgery prior to 6 years of age, with long term follow-up required to demonstrate procedural efficacy [10]. There are studies that indicate that if correction is not obtained earlier in development, corrective osteotomy may be required later [1]. However, it has been noted that attempting repeat physiolysis is not recommended initially given the minimal benefit and risk of premature physical fusion; delayed wedge osteotomy can safely follow physiolysis to gain further correction [1].…”

“…There are studies that indicate that if correction is not obtained earlier in development, corrective osteotomy may be required later [1]. However, it has been noted that attempting repeat physiolysis is not recommended initially given the minimal benefit and risk of premature physical fusion; delayed wedge osteotomy can safely follow physiolysis to gain further correction [1]. Future retrospective studies should be conducted to examine older patients with correction of clinodactyly with delta phalanx, and their postoperative radiographic and functional outcomes.…”

“…One form of clinodactyly is a delta phalanx, which presents as a C-shaped epiphysis bracketing. Clinodactyly with delta phalanx is commonly inherited in an autosomal dominant manner and can have variable expressivity [1]. This defect can be observed most commonly in the middle phalanx, but also in the proximal phalanx or the metacarpal [2].…”

Phalangeal Intra-Articular Osteochondroma Caused a Rare Clinodactyly Deformity in Children: Case Series and Literature Review

“…Clinodactyly is defined as a congenital curvature of a digit caused by an interphalangeal joint in the coronal plane (1). This curvature was generated by malalignment of the interphalangeal joint which was due to an abnormal trapezoidal or triangular shape of one or more phalanges (2)(3)(4). The visible curvature of the digit arises due to the abnormal shape that leads to asymmetric longitudinal growth in a direction from the original longitudinal axis of the finger.…”

Congenital Familial Clinodactyly of Index Finger with Proximal Delta Phalanges and Ulnar Deviation

“…In this report, mild ulnar clinodactyly of the bilateral second digits also was presented. 5 Numerous surgical techniques have been described, with the majority involving wedge osteotomy (opening, closing, or reverse). In 1987, Vickers described physeal bar resection and interposing fat graft (physiolysis).…”