Unraveling Novel Pathogenic CAPN3 Variants in Limb-Girdle Muscular Dystrophy R1

Limb-Girdle Muscular Dystrophy R1 (LGMDR1) is an autosomal recessive neuromuscular disorder caused by mutations in the calpain-3 (CAPN3) gene, which encodes a calcium-dependent intracellular protease. Various pathogenic mutations have been reported to date, typically leading to weakness of the pelvic and shoulder girdle muscles. However, the clinical and pathological features of LGMDR1 are often heterogeneous and overlap with those of other LGMD subtypes, making a definitive molecular diagnosis essential for understanding the disease pathogenesis. Immunoblot analysis of calpain-3 protein revealed either complete or partial loss of expression in patients with LGMD. Additionally, multiple CAPN3 variants were identified and evaluated using in silico pathogenicity prediction tools. Alongside previously known mutations, Sanger sequencing also revealed novel pathogenic variants in the CAPN3 gene. Identification of genetic variants in LGMDR1 patients helps explore the mechanisms of disease pathogenesis and may support more accurate diagnosis and prognostication.

Publication Details

Journal
Journal of Rare Diseases Research & Treatment
Published
2025-05-07
DOI
https://doi.org/10.29245/2572-9411/2025/1.1218
Primary Topic
Vascular Tumors and Angiosarcomas
Type
article
Field-Weighted Citation Impact
0.00
Controls
|||
ALL TIME
JAN
FEB
MAR
APR
MAY
JUN
JUL
AUG
SEP
OCT
article

Unraveling Novel Pathogenic CAPN3 Variants in Limb-Girdle Muscular Dystrophy R1

Journal of Rare Diseases Research & Treatment
Vascular Tumors and Angiosarcomas
article

Unraveling Novel Pathogenic CAPN3 Variants in Limb-Girdle Muscular Dystrophy R1

article en

Abstract

Limb-Girdle Muscular Dystrophy R1 (LGMDR1) is an autosomal recessive neuromuscular disorder caused by mutations in the calpain-3 (CAPN3) gene, which encodes a calcium-dependent intracellular protease. Various pathogenic mutations have been reported to date, typically leading to weakness of the pelvic and shoulder girdle muscles. However, the clinical and pathological features of LGMDR1 are often heterogeneous and overlap with those of other LGMD subtypes, making a definitive molecular diagnosis essential for understanding the disease pathogenesis. Immunoblot analysis of calpain-3 protein revealed either complete or partial loss of expression in patients with LGMD. Additionally, multiple CAPN3 variants were identified and evaluated using in silico pathogenicity prediction tools. Alongside previously known mutations, Sanger sequencing also revealed novel pathogenic variants in the CAPN3 gene. Identification of genetic variants in LGMDR1 patients helps explore the mechanisms of disease pathogenesis and may support more accurate diagnosis and prognostication.

Journal of Rare Diseases Research & TreatmentVol. 9(1)
Openalex Percentile: Top 85%
Vascular Tumors and Angiosarcomas
AI Navigator

Ask Laika to Summarize, Analyze, and Connect papers live on the map.

Summarize Papers & Methodologies

Extract key findings, datasets, and comparative methods across publications.

Benchmark Rankings & Visual Analytics

Rank top research institutions, authors, funders, topics, and journals by Field-Weighted Citation Impact (FWCI) and paper volume with instant charts.

Connect Distant Disciplines

Bridge topological clusters on the map to find hidden collaborative intersections.

Unraveling Novel Pathogenic CAPN3 Variants in Limb-Girdle Muscular Dystrophy R1 · Journal of Rare Diseases Research & Treatment (2025) | TGRS Research Map | TGRS