Congenital myopathies are a heterogeneous group of rare inherited muscle disorders. Despite the good sensitivity of whole-exome sequencing in detecting pathogenic variants, many cases remain molecularly unsolved. Here, we present the case of a woman with congenital myopathy that remained unsolved for many years, in which the application of whole-genome sequencing enabled the identification of a novel deep intronic mutation in the MYH7 gene. A 22-year-old woman developed muscle weakness since infancy, with frequent falls, toe-walking, and difficulty climbing stairs. Muscle biopsy revealed atrophy of type 1 fibers relative to type 2, consistent with fiber-type disproportion. After a long “molecular odyssey,” whole-genome sequencing performed on the patient–parents trio identified a de novo deep intronic variant in MYH7. This case further underscores the importance of pursuing the search for the causative gene to enable more accurate clinical monitoring and tailored health care.

A novel deep intronic mutation expands the genotype spectrum of MYH7-related myopathies

Neri, Luca Maria;Gualandi, Francesca
2025

Abstract

Congenital myopathies are a heterogeneous group of rare inherited muscle disorders. Despite the good sensitivity of whole-exome sequencing in detecting pathogenic variants, many cases remain molecularly unsolved. Here, we present the case of a woman with congenital myopathy that remained unsolved for many years, in which the application of whole-genome sequencing enabled the identification of a novel deep intronic mutation in the MYH7 gene. A 22-year-old woman developed muscle weakness since infancy, with frequent falls, toe-walking, and difficulty climbing stairs. Muscle biopsy revealed atrophy of type 1 fibers relative to type 2, consistent with fiber-type disproportion. After a long “molecular odyssey,” whole-genome sequencing performed on the patient–parents trio identified a de novo deep intronic variant in MYH7. This case further underscores the importance of pursuing the search for the causative gene to enable more accurate clinical monitoring and tailored health care.
2025
Barp, Andrea; Neri, Luca Maria; Maggi, Lorenzo; Iascone, Maria; Gualandi, Francesca
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Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/11392/2623192
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