Surgical considerations for syndactyly of the hand in harlequin ichthyosis: a 5-year experience of sequential surgical treatment in a single patient

Jaemin Choi, J. Hong
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Abstract

Harlequin ichthyosis (HI) is a rare congenital disease that primarily affects the skin. Its complications include syndactyly, which can cause severe constriction. Surgical release is thought to be helpful, but the details remain unclear due to a lack of data. We share our 5-year experience of a patient with HI with successful outcomes from five syndactyly release operations involving division, local flap interposition, and full-thickness skin grafts. A visible decrease in the ulnar deviation of the metacarpophalangeal joints and a wider range of motion were noted. There were no complications, except for one case of donor site dehiscence that was resolved after minor revision. The surgical design must conform to the anatomical structure, as it can be easily mistaken due to masking of the skin. A suitable donor site is a flat, non-folding surface, such as the lateral thigh.
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丑角鱼鳞病手部并指畸形的手术治疗:一名患者5年序贯手术治疗经验
小丑鱼鳞病(HI)是一种罕见的先天性疾病,主要影响皮肤。其并发症包括并指,可引起严重的收缩。手术释放被认为是有帮助的,但由于缺乏数据,细节尚不清楚。我们分享5年的治疗HI患者的经验,其中包括5次并指松解手术,包括分离、局部皮瓣插入和全层皮肤移植。掌指关节尺偏明显减少,活动范围更大。除一例供体部位裂开经轻微翻修后解决外,无其他并发症。手术设计必须符合解剖结构,因为它很容易被误认为是由于皮肤的掩盖。合适的供体部位是平坦的、不折叠的表面,如大腿外侧。
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