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A child with longitudinal cleavage of the upper extremity: treatment and etiology considerations

Chirag Shah1, Paul R Manske, Charles A Goldfarb

  • 1Shriners Hospital for Children and St. Louis Children’s Hospital, Department of OrthopaedicSurgery, Washington University School of Medicine, St. Louis, MO 63110, USA.

The Journal of Hand Surgery
|October 19, 2010
PubMed

Insights

This study details a rare case of a boy with one normal right arm and two left arms. Surgical intervention involved removing a nonfunctional limb and reconstructing the other for improved function.

Area of Science:

  • Developmental biology
  • Orthopedic surgery
  • Pediatric medicine

Background:

  • Congenital anomalies of the upper limb present unique challenges in diagnosis and treatment.
  • Understanding limb development is crucial for addressing complex malformations.

Observation:

  • A 4-year-old boy presented with a normal right upper extremity and two left upper extremities.
  • One left extremity had radial components and a rudimentary thumb, extending from the humerus.
  • The second left extremity had ulnar components attached inferiorly to the thorax, with limited finger function.

Findings:

  • The superior left extremity's radius and thumb were excised due to lack of function.
  • An "on-top-plasty" was performed, mobilizing the inferior left extremity and fusing its ulna to the humerus of the superior extremity.
  • This surgical approach aimed to improve the patient's limb functionality.

Implications:

  • This case highlights a potential proximal manifestation of central longitudinal deficiency.
  • Early limb bud cleavage affecting the apical ectodermal ridge and mesoderm could explain this anomaly.
  • The surgical technique offers a potential solution for similar complex limb malformations.

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