Carrier detection of duchenne and becker muscular dystrophy using muscle dystrophin immunohistochemistry

To ascertain whether dystrophin immunohistochemistry could improve DMD/ BMD carrier detection, we analyzed 14 muscle biopsies from 13 DMD and one BMD probable and possible carriers. All women were also evaluated using conventional methods, including genetic analysis, clinical and neurological evalua...

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Main Authors: Acary S. Bulle Oliveira, Alberto A. Gabbai, Beny Schmidt, Beatriz Hitomi Kiyomoto, G. Camargo Lima, Carlo Minetti, Eduardo Bonilla
Format: Article
Language:English
Published: Thieme Revinter Publicações 1992-12-01
Series:Arquivos de Neuro-Psiquiatria
Subjects:
Online Access:http://www.scielo.br/scielo.php?script=sci_arttext&pid=S0004-282X1992000400010&lng=en&tlng=en
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author Acary S. Bulle Oliveira
Alberto A. Gabbai
Beny Schmidt
Beatriz Hitomi Kiyomoto
G. Camargo Lima
Carlo Minetti
Eduardo Bonilla
author_facet Acary S. Bulle Oliveira
Alberto A. Gabbai
Beny Schmidt
Beatriz Hitomi Kiyomoto
G. Camargo Lima
Carlo Minetti
Eduardo Bonilla
author_sort Acary S. Bulle Oliveira
collection DOAJ
description To ascertain whether dystrophin immunohistochemistry could improve DMD/ BMD carrier detection, we analyzed 14 muscle biopsies from 13 DMD and one BMD probable and possible carriers. All women were also evaluated using conventional methods, including genetic analysis, clinical and neurological evaluation, serum CK levels, KMG, and muscle biopsy. In 6 cases, there was a mosaic of dystrophin-positive and dystrophin-deficient fibers that allowed to make the diagnosis of a carrier state. Comparing dystrophin immunohistochemistry to the traditional methods, it was noted that this method is less sensitive than serum CK measuremens, but is more sensitive than EMG and muscle biopsy. The use of dystrophin immunohistochemistry in addition to CK, EMG and muscle biopsy improved the accuracy of carrier detection. This method is also helpful to distinguish manifesting DMD carriers from patients with other neuromuscular diseases like limb-girdle muscular dystrophy and spinal muscular atrophy.
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publisher Thieme Revinter Publicações
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series Arquivos de Neuro-Psiquiatria
spelling doaj-art-8ba5cfc81d894f97bff1cafd01d32ae72025-08-20T01:58:22ZengThieme Revinter PublicaçõesArquivos de Neuro-Psiquiatria1678-42271992-12-0150447848510.1590/S0004-282X1992000400010S0004-282X1992000400010Carrier detection of duchenne and becker muscular dystrophy using muscle dystrophin immunohistochemistryAcary S. Bulle OliveiraAlberto A. GabbaiBeny SchmidtBeatriz Hitomi KiyomotoG. Camargo LimaCarlo MinettiEduardo BonillaTo ascertain whether dystrophin immunohistochemistry could improve DMD/ BMD carrier detection, we analyzed 14 muscle biopsies from 13 DMD and one BMD probable and possible carriers. All women were also evaluated using conventional methods, including genetic analysis, clinical and neurological evaluation, serum CK levels, KMG, and muscle biopsy. In 6 cases, there was a mosaic of dystrophin-positive and dystrophin-deficient fibers that allowed to make the diagnosis of a carrier state. Comparing dystrophin immunohistochemistry to the traditional methods, it was noted that this method is less sensitive than serum CK measuremens, but is more sensitive than EMG and muscle biopsy. The use of dystrophin immunohistochemistry in addition to CK, EMG and muscle biopsy improved the accuracy of carrier detection. This method is also helpful to distinguish manifesting DMD carriers from patients with other neuromuscular diseases like limb-girdle muscular dystrophy and spinal muscular atrophy.http://www.scielo.br/scielo.php?script=sci_arttext&pid=S0004-282X1992000400010&lng=en&tlng=endistrofia muscular (Duchenne e Becker)detecção de portadorasdistrofina
spellingShingle Acary S. Bulle Oliveira
Alberto A. Gabbai
Beny Schmidt
Beatriz Hitomi Kiyomoto
G. Camargo Lima
Carlo Minetti
Eduardo Bonilla
Carrier detection of duchenne and becker muscular dystrophy using muscle dystrophin immunohistochemistry
Arquivos de Neuro-Psiquiatria
distrofia muscular (Duchenne e Becker)
detecção de portadoras
distrofina
title Carrier detection of duchenne and becker muscular dystrophy using muscle dystrophin immunohistochemistry
title_full Carrier detection of duchenne and becker muscular dystrophy using muscle dystrophin immunohistochemistry
title_fullStr Carrier detection of duchenne and becker muscular dystrophy using muscle dystrophin immunohistochemistry
title_full_unstemmed Carrier detection of duchenne and becker muscular dystrophy using muscle dystrophin immunohistochemistry
title_short Carrier detection of duchenne and becker muscular dystrophy using muscle dystrophin immunohistochemistry
title_sort carrier detection of duchenne and becker muscular dystrophy using muscle dystrophin immunohistochemistry
topic distrofia muscular (Duchenne e Becker)
detecção de portadoras
distrofina
url http://www.scielo.br/scielo.php?script=sci_arttext&pid=S0004-282X1992000400010&lng=en&tlng=en
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