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Unilateral dermatomal superficial telangiectasia.

M Okinaga1, K Go, M Mizoguchi

  • 1Department of Dermatology, Teikyo University School of Medicine, Tokyo, Japan.

The Journal of Dermatology
|October 1, 1990
PubMed
Summary

A rare case of unilateral dermatomal superficial telangiectasia (UDST) presented in two non-adjacent areas in a young child. This congenital condition, appearing early in life, suggests a possible link to developmental factors rather than surgical history.

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Area of Science:

  • Dermatology
  • Pediatrics
  • Medical Genetics

Background:

  • Unilateral dermatomal superficial telangiectasia (UDST) is a rare vascular malformation.
  • Congenital onset is typical, but presentation can vary.
  • Previous cases have not detailed involvement of non-adjacent dermatomes.

Observation:

  • A three-year-old boy presented with skin lesions characteristic of UDST.
  • Lesions were observed in two distinct, non-adjacent dermatomes: Trigeminal 2 and L4-5.
  • The patient had a history of a right hepatectomy at four months old for a liver hamartoma.

Findings:

  • This represents the first reported case of UDST affecting two non-adjacent dermatomes.
  • Onset at an early age (eight months post-operation) with normal liver function supports a congenital etiology.

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  • The extensive hepatectomy did not appear to influence the development or presentation of the UDST.

    • Implications:

    • This case expands the known clinical spectrum of unilateral dermatomal superficial telangiectasia.
    • It highlights the importance of considering congenital factors in early-onset vascular malformations, even with a history of surgery.
    • Further research may elucidate the genetic or developmental pathways involved in UDST affecting multiple, non-contiguous dermatomes.