Long-term clinical follow-up of a family with Becker muscular dystrophy associated with a large deletion in the DMD gene.

Neuromuscul Disord

West Midlands Regional Genetics Service, Birmingham Women's and Children's Hospital, Birmingham B15 2TG, United Kingdom. Electronic address:

Published: June 2024

Duchenne muscular dystrophy is a neuromuscular disease caused by DMD gene mutations that result in an absence of functional dystrophin protein. Patients with Duchenne experience progressive muscle weakness, are typically wheelchair dependent by their early teens, and develop respiratory and cardiac complications that lead to death in their twenties or thirties. Becker muscular dystrophy is also caused by DMD gene mutations, but symptoms are less severe and progression is slower compared with Duchenne. We describe a case study of a patient with Becker muscular dystrophy who was still ambulant at age 61 years and had a milder phenotype than Duchenne, despite 46% of his DMD gene being missing. His affected relatives had similarly mild phenotypes and clinical courses. These data guided the understanding of the criticality of various regions of dystrophin and informed the development of micro-dystrophin constructs to compensate for the absence of functional dystrophin in Duchenne.

Download full-text PDF

Source
http://dx.doi.org/10.1016/j.nmd.2024.04.004DOI Listing

Publication Analysis

Top Keywords

muscular dystrophy
16
dmd gene
16
becker muscular
12
caused dmd
8
gene mutations
8
absence functional
8
functional dystrophin
8
duchenne
5
long-term clinical
4
clinical follow-up
4

Similar Publications

Most patients with Duchenne muscular dystrophy (DMD) are non-ambulant. Preserving proximal motor function is crucial, rarely studied. Tamoxifen, a selective oestrogen receptor modulator, reduced signs of muscular pathology in a DMD mouse model.

View Article and Find Full Text PDF

Pediatric Sleep Quality and Parental Stress in Neuromuscular Disorders: Descriptive Analytical Study.

Asian Pac Isl Nurs J

January 2025

Nursing Care Research Center, Clinical Sciences Institute, Nursing Faculty, Baqiyatallah University of Medical Sciences, Vanak Square, Tehran, Iran, 98 9127297199.

Background: Neuromuscular disorders (NMDs) constitute a heterogeneous group of disorders that affect motor neurons, neuromuscular junctions, and muscle fibers, resulting in symptoms such as muscle weakness, fatigue, and reduced mobility. These conditions significantly affect patients' quality of life and impose a substantial burden on caregivers. Spinal muscular atrophy (SMA) is a relatively common NMD in children that presents in various types with varying degrees of severity.

View Article and Find Full Text PDF

A man in his late 50s was referred by a speech and language therapist for consideration of a palatal lift prosthesis (PLP) to improve his speech intelligibility. He presented with hypokinetic dysarthria characterised by reduced loudness, breathy voice and hypernasality. The patient had a diagnosis of progressive muscular dystrophy and mobilised in a motorised wheelchair.

View Article and Find Full Text PDF

Skeletal muscle regeneration in adults is predominantly driven by satellite cells. Loss of satellite cell pool and function leads to skeletal muscle wasting in many conditions and disease states. Here, we demonstrate that the levels of fibroblast growth factor-inducible 14 (Fn14) were increased in satellite cells after muscle injury.

View Article and Find Full Text PDF

Transcriptome analysis of muscle atrophy in Leizhou black goats: identification of key genes and insights into limb-girdle muscular dystrophy.

BMC Genomics

January 2025

Zhanjiang Experimental Station, Chinese Academy of Tropical Agricultural Sciences, Zhanjiang, 524013, China.

Background: The Leizhou Black Goat (LZBG), a prominent breed in tropical China's meat goat industry, frequently exhibits inherent muscle atrophy and malnutrition-related traits. Particularly, muscles critical for support, such as the legs, often display severe symptoms. This study aimed to investigate the differential genes and signaling pathways influencing muscle development and atrophy across various muscle locations in LZBG from a muscular atrophy-affected family.

View Article and Find Full Text PDF

Want AI Summaries of new PubMed Abstracts delivered to your In-box?

Enter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!