Document Detail


Severe vincristine neuropathy in Charcot-Marie-Tooth disease type 1A.
MedLine Citation:
PMID:  8608515     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
BACKGROUND: A general predisposition for vincristine-related neuropathy has been observed in persons with a family history of hereditary neuropathies. METHODS: In a retrospective case series, we investigated the possible association between the DNA rearrangement found in patients with Charcot-Marie-Tooth Disease Type 1A (CMT1A) and susceptibility to the neurotoxicity of vincristine. In selected patients and family members, we performed electrodiagnostic studies and analyzed DNA samples for 17p11.1-12 duplication associated with CMT1A. RESULTS: We describe three families with autosomal dominant CMT1, among whom a family member with a neoplastic disease suffered rapid onset, severe neuropathy after receiving initial doses of vincristine as a part of a routine chemotherapy protocol. All three families had at least one affected family member with 17p11.2-12 duplication. CONCLUSIONS: These cases show that 17p11.2-12 duplication predisposes patients to severe neurotoxicity from vincristine and that this drug should be avoided with patients with CMT1A. It is therefore essential to obtain a detailed family history for all oncology patients to screen for possible hereditary neuropathies. In patients with unexplained or preexisting familial neuropathy, testing for 17p11.2-12 duplication should be carried out prior to initiating vincristine therapy. Patients with other hereditary neuropathies may also be at risk for severe neurotoxic reactions.
Authors:
W D Graf; P F Chance; M W Lensch; L J Eng; H P Lipe; T D Bird
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Publication Detail:
Type:  Case Reports; Journal Article; Research Support, U.S. Gov't, P.H.S.    
Journal Detail:
Title:  Cancer     Volume:  77     ISSN:  0008-543X     ISO Abbreviation:  Cancer     Publication Date:  1996 Apr 
Date Detail:
Created Date:  1996-05-28     Completed Date:  1996-05-28     Revised Date:  2010-03-24    
Medline Journal Info:
Nlm Unique ID:  0374236     Medline TA:  Cancer     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  1356-62     Citation Subset:  AIM; IM    
Affiliation:
Department of Pediatrics, University of Washington School of Medicine, Seattle, USA.
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Adult
Charcot-Marie-Tooth Disease / genetics*,  physiopathology*
Child
Chromosomes, Human, Pair 17
DNA / genetics
Disease Susceptibility
Electrophysiology
Female
Humans
Male
Middle Aged
Nervous System Diseases / chemically induced*,  diagnosis
Pedigree
Pregnancy
Retrospective Studies
Vincristine / adverse effects*
Grant Support
ID/Acronym/Agency:
R01-NS30804/NS/NINDS NIH HHS
Chemical
Reg. No./Substance:
57-22-7/Vincristine; 9007-49-2/DNA

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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