Lipoid proteinosis: A case with distinct histopathological and radiological findings

J Cutan Pathol. 2017 Oct;44(10):887-891. doi: 10.1111/cup.13002. Epub 2017 Jul 25.

Abstract

Lipoid proteinosis is a rare inherited genodermatosis characterized by hyaline deposits in various tissues. Clinically, it manifests with cutaneous as well as extracutaneous features. Periodic acid-Schiff (PAS)-reactive hyaline deposits in the upper dermis, with localization around blood vessels and eccrine sweat glands, in particular, is the histopathological hallmark finding. On brain imaging, bilateral symmetrical temporal lobe calcifications are considered to be pathognomonic of this disorder. We report a case of lipoid proteinosis in which hyaline deposits were present in the papillary and reticular dermis, without being seen at the periphery of eccrine sweat glands, along with dystrophic calcification. Magnetic resonance imaging (MRI) of brain revealed hydrocephalus, subependymal heterotropia and absent splenium of corpus callosum with no evidence of temporal lobe calcification. Thus, our case highlights the inherent diverse nature of lipoid proteinosis.

Keywords: dystrophic calcification; eccrine sweat glands; hydrocephalus; lipoid proteinosis.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Calcinosis* / diagnostic imaging
  • Calcinosis* / metabolism
  • Calcinosis* / pathology
  • Corpus Callosum* / diagnostic imaging
  • Corpus Callosum* / metabolism
  • Corpus Callosum* / pathology
  • Dermis / metabolism
  • Dermis / pathology*
  • Eccrine Glands / metabolism
  • Eccrine Glands / pathology*
  • Humans
  • Hyalin / metabolism
  • Hydrocephalus* / diagnostic imaging
  • Hydrocephalus* / metabolism
  • Hydrocephalus* / pathology
  • Lipoid Proteinosis of Urbach and Wiethe* / diagnostic imaging
  • Lipoid Proteinosis of Urbach and Wiethe* / metabolism
  • Lipoid Proteinosis of Urbach and Wiethe* / pathology
  • Magnetic Resonance Imaging*
  • Male
  • Temporal Lobe* / diagnostic imaging
  • Temporal Lobe* / metabolism
  • Temporal Lobe* / physiology