HMSN-4

PHYH | 1997

What Is HMSN-4?

HMSN-4 is a type of CMT caused by autosomal recessive mutations in the PHYH gene. This gene provides instructions for making the enzyme phytanoyl-CoA hydroxylase, which breaks down phytanic acid, a fatty acid obtained from the diet. Mutations in the PHYH gene disrupt this function, leading to impaired nerve signal transmission.

HMSN-4 is autosomal recessive, meaning that both copies of the gene must have a mutation to cause this subtype.

Clinical Features

HMSN-4, also known as Refsum disease, is a metabolic disease caused by the buildup of phytanic acid, a fatty acid the body normally breaks down. Symptoms usually begin in childhood or adolescence and affect multiple systems in addition to the peripheral nerves. Nerve conduction studies reflect an axonal and demyelinating process.

HMSN-4 symptoms may include:

  • Progressive vision loss from retinitis pigmentosa
  • Reduced sense of smell (anosmia)
  • Peripheral neuropathy with weakness and reduced sensation
  • Poor balance and coordination (ataxia)
  • Hearing loss
  • Dry, scaly skin
  • Foot drop
  • Additional symptoms not listed here

Disease Course

HMSN-4 shows variability in severity and progression. Because the disease is driven by dietary phytanic acid, dietary management can slow or stabilize progression in some individuals. Disease progression is generally slow, though episodes may occur.

Clinical Basics

Subtype
HMSN-4

Classification
HMSN

Subtype Alias
Refsum Disease

Neuropathy Type
Axonal

Inheritance Pattern
autosomal recessive

Genetic Context

HGNC-Approved Gene Symbol
PHYH

Gene Full Name
Phytanoyl-CoA 2-Hydroxylase

HGNC Gene Alias(es)
PAHX

Chromosome
10p13

Zygosity of Responsible Variant
Homozygous

Variant Mechanism
Loss of Function (LoF)

ClinVar Pathogenic Variants

View HMSN-4 ClinVar Variants

HMSN-4 OMIM Entry

HMSN-4 OMIM

PHYH OMIM Entry

PHYH OMIM

More Info

HMSN-4 Research Opportunity

CMT Natural History Study

Original Discovery Publication

Publication Title

Identification of PAHX, a Refsum Disease Gene.

Authors

Mihalik, S. J., Morrell, J. C., Kim, D., Sacksteder, K. A., Watkins, P. A., & Gould, S. J.

Publication Date
October 1, 1997

Updated: July 18, 2026 | By: K. Raymond

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