A patient with PMP22-related hereditary neuropathy and DBH-gene-related dysautonomia

J Neurol. 2015 Oct;262(10):2373-81. doi: 10.1007/s00415-015-7896-z. Epub 2015 Sep 26.

Abstract

Recurrent focal neuropathy with liability to pressure palsies is a relatively frequent autosomal-dominant demyelinating neuropathy linked to peripheral myelin protein 22 (PMP22) gene deletions. The combination of PMP22 gene mutations with other genetic variants is known to cause a more severe phenotype than expected. We present the case of a patient with severe orthostatic hypotension since 12 years of age, who inherited a PMP22 gene deletion from his father. Genetic double trouble was suspected because of selective sympathetic autonomic disturbances. Through exome-sequencing analysis, we identified two novel mutations in the dopamine beta hydroxylase gene. Moreover, with interactome analysis, we excluded a further influence on the origin of the disease by variants in other genes. This case increases the number of unique patients presenting with dopamine-β-hydroxylase deficiency and of cases with genetically proven double trouble. Finding the right, complete diagnosis is crucial to obtain adequate medical care and appropriate genetic counseling.

Keywords: Dopamine-β-hydroxylase deficiency; Exome sequencing, dysautonomia; Recurrent focal neuropathy with liability to pressure palsies.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Autonomic Nervous System Diseases / congenital*
  • Dopamine beta-Hydroxylase / deficiency*
  • Dysautonomia, Familial / genetics
  • Hereditary Sensory and Autonomic Neuropathies / genetics*
  • Humans
  • Myelin Proteins / genetics*
  • Norepinephrine / deficiency*

Substances

  • Myelin Proteins
  • PMP22 protein, human
  • Dopamine beta-Hydroxylase
  • Norepinephrine

Supplementary concepts

  • dopamine beta hydroxylase deficiency