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PMID: 8816708 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Clinical phenotypes of different MPZ (P0) mutations may include Charcot-Marie-Tooth type 1B, Dejerine-Sottas, and congenital hypomyelination.

Neuron ·Vol. 17 ·No. 3 ·1996-09-00 ·Pages 451-60

Warner LE, Hilz MJ, Appel SH, Killian JM, Kolodry EH, Karpati G, Carpenter S, Watters GV, Wheeler C, Witt D, Bodell A, Nelis E, Van Broeckhoven C, Lupski JR

Abstract

Hereditary demyelinating peripheral neuropathies consist of a heterogeneous group of genetic disorders that includes hereditary neuropathy with liability to pressure palsies (HNPP), Charcot-Marie-Tooth disease (CMT), Dejerine-Sottas syndrome (DSS), and congenital hypomyelination (CH). The clinical classification of these neuropathies into discrete categories can sometimes be difficult because there can be both clinical and pathologic variation and overlap between these disorders. We have identified five novel mutations in the myelin protein zero (MPZ) gene, encoding the major structural protein (P0) of peripheral nerve myelin, in patients with either CMT1B, DSS, or CH. This finding suggests that these disorders may not be distinct pathophysiologic entities, but rather represent a spectrum of related "myelinopathies" due to an underlying defect in myelination. Furthermore, we hypothesize the differences in clinical severity seen with mutations in MPZ are related to the type of mutation and its subsequent effect on protein function (i.e., loss of function versus dominant negative).

MeSH Terms
Adult Charcot-Marie-Tooth Disease/diagnosis,genetics Cloning, Molecular Cohort Studies Crystallography DNA Mutational Analysis Demyelinating Diseases/congenital,diagnosis,genetics Female Genotype Hereditary Sensory and Motor Neuropathy/diagnosis,genetics Humans Male Microscopy, Electron Myelin P0 Protein/chemistry,genetics Phenotype Point Mutation/physiology Protein Conformation Sural Nerve/ultrastructure
Chemicals
Myelin P0 Protein
Authors & Affiliations
14 authors, click to expand affiliations / ORCID
Warner L E
Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas 77030, USA.
Hilz M J
Appel S H
Killian J M
Kolodry E H
Karpati G
Carpenter S
Watters G V
Wheeler C
Witt D
Bodell A
Nelis E
Van Broeckhoven C
Lupski J R
Article Info
Journal
Neuron
Abbr.
Neuron
ISSN
0896-6273
Published
1996-09-00
Pages
451-60
Language
English
Region
United States
NLM ID
8809320
Subset
IM
Grants
NINDS NIH HHS · R01-NS27042 · United States
NIGMS NIH HHS · T32-GM08307 · United States
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