Episodic headache with spontaneous hypothermia reveal Shapiro's syndrome variant with effectiveness of clonidine therapy

J Headache Pain. 2021 Apr 26;22(1):30. doi: 10.1186/s10194-021-01245-3.

Abstract

Background: Episodic headache with spontaneous hypothermia constitute an uncommon association and is not well recognized in the International Classification of Headache Disorders (ICHD-3). Spontaneous periodic hypothermia, also called Shapiro's syndrome, is a rare disease characterized by hypothermia attacks associated or not with hyperhidrosis without any triggering factor.

Case presentation: We report a rare case of Shapiro's syndrome variantrevealed by episodes of headache with spontaneous hypothermia witheffectiveness of clonidine therapy in a 76-year-old Parkinson's disease woman.

Conclusions: In the literature, apart from Shapiro's syndrome, headache withhypothermia seem to occur very rarely. In our case,these symptoms may be considered as a very rare non-motor fluctuation ofParkinson's disease.

Keywords: Clonidine; Headache; Hyperhidrosis; Hypothermia; Parkinson’s disease; Shapiro’s syndrome.

Publication types

  • Case Reports

MeSH terms

  • Aged
  • Agenesis of Corpus Callosum
  • Clonidine / therapeutic use
  • Female
  • Headache
  • Humans
  • Hyperhidrosis*
  • Hypothermia* / complications
  • Syndrome

Substances

  • Clonidine

Supplementary concepts

  • Shapiro syndrome