Atypical keratosis pilaris-like lesions in a patient with Bethlem myopathy

Pediatr Dermatol. 2022 Mar;39(2):309-311. doi: 10.1111/pde.14862. Epub 2021 Dec 9.

Abstract

Bethlem myopathy is a collagen VI-related myopathy. Collagen VI is primarily not only associated with the extracellular matrix of skeletal muscle, but is also found in the skin, blood vessels, and other organs. Dermatologic findings described for Bethlem myopathy include follicular hyperkeratosis and abnormal scar formation, although clinical and histopathologic photographs remain elusive in the literature. We present a case of atypical keratosis pilaris-like follicular lesions in a patient with Bethlem myopathy and provide histopathologic correlation to better characterize the development of skin lesions in this rare neuromuscular disease.

Keywords: Bethlem myopathy; collagen diseases; connective tissue disorders; muscular dystrophies; skin signs of systemic disease.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple
  • Collagen Type VI* / genetics
  • Contracture* / genetics
  • Darier Disease
  • Eyebrows / abnormalities
  • Humans
  • Muscle, Skeletal / pathology
  • Muscular Dystrophies / congenital
  • Mutation

Substances

  • Collagen Type VI

Supplementary concepts

  • Bethlem myopathy
  • Burnett Schwartz Berberian syndrome