A Case of VACTERL Association With Thumb Polydactyly Requiring Surgical Treatment

We report a case of a rare type of polydactyly associated with VACTERL association (verebral defects, anal atresia, cardiac defects, tracheo-esophageal fistula,renal anomalies, and limb abnormalities) that was treated surgically in infancy with good results. In this case, the patient had radial and ulnar thumbs of almost equal size, and mild thumb hypoplasia. This thumb is as classified Blauth type Ⅰ thumb hypoplasia and as a type IV triphalangeal thumb in the Rotterdam classification. The radial thumb had one phalanx with no distal phalanx or nail plate, whereas the ulnar thumb had three phalanges, including a distal phalanx and nail plate. On-top plasty was performed, joining the ulnar thumb top to the radial thumb base. One year after surgery, the appearance of thumb is good, and the child can use the affected digit well in daily activities. However, flexion and extension of the interphalangeal joint of the thumb is weak. Follow-up will be continued to monitor growth of the reconstructed thumb.

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Publication Details

Journal
Journal of Hand Surgery Global Online
Published
2025-05-22
DOI
https://doi.org/10.1016/j.jhsg.2024.10.009
Primary Topic
Congenital limb and hand anomalies
Type
article
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article

A Case of VACTERL Association With Thumb Polydactyly Requiring Surgical Treatment

Sohachi Toriyabe, Hajime Matsumura, Takako Komiya, Kunio Aoki et al.
Journal of Hand Surgery Global Online
Congenital limb and hand anomalies
article

A Case of VACTERL Association With Thumb Polydactyly Requiring Surgical Treatment

Sohachi Toriyabe, Hajime Matsumura, Takako Komiya, Kunio Aoki, Kazuki Shimada, Rei Omura
article en

Abstract

We report a case of a rare type of polydactyly associated with VACTERL association (verebral defects, anal atresia, cardiac defects, tracheo-esophageal fistula,renal anomalies, and limb abnormalities) that was treated surgically in infancy with good results. In this case, the patient had radial and ulnar thumbs of almost equal size, and mild thumb hypoplasia. This thumb is as classified Blauth type Ⅰ thumb hypoplasia and as a type IV triphalangeal thumb in the Rotterdam classification. The radial thumb had one phalanx with no distal phalanx or nail plate, whereas the ulnar thumb had three phalanges, including a distal phalanx and nail plate. On-top plasty was performed, joining the ulnar thumb top to the radial thumb base. One year after surgery, the appearance of thumb is good, and the child can use the affected digit well in daily activities. However, flexion and extension of the interphalangeal joint of the thumb is weak. Follow-up will be continued to monitor growth of the reconstructed thumb.

Journal of Hand Surgery Global OnlineVol. 7(4)
Tokyo Medical University (JP), Sendai Medical Center (JP)
Good health and well-being
Openalex Percentile: Top 89%
Congenital limb and hand anomalies
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