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Neuromuscular disorders : NMD · 2026

TTN-related limb-girdle muscular dystrophy caused by a homozygous c.107837A>C variant: first report in an African patient.

Клинический случайСтатьяНазвание и аннотация: Перевод пока не подготовлен
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Elseed Maha, Schiava Marianela, Harris Elizabeth, Parkhurst Yolande, Joshi Abhijit, Grover Emma, James Meredith K, Wong Karen, Sodhi Jassi, Salman Doaa, Töpf Ana, Bettolo Chiara Marini, Straub Volker

Аннотация

Tibial muscular dystrophy (TMD) is a late-onset autosomal dominant distal myopathy initially described in the Finnish population and caused by monoallelic, mostly missense, TTN variants. More recently, earlier-onset limb girdle muscular dystrophy phenotypes have been reported in patients carrying biallelic TTN variants. We describe the first African patient homozygous for a previously described TMD related TTN variant presenting with a limb-girdle muscular dystrophy (LGMD) phenotype. The proband, an ambulant 11-year-old boy originally from Libya, presented with progressive lower limb weakness that was first noted at the age of 9 months. He displayed a phenotype characterised by proximal and distal lower-limb weakness that evolved over 10 years into a clear limb-girdle pattern. T1-weighted muscle MRI images revealed fatty replacement of the gluteus maximus, vasti, adductor magnus, soleus, and tibialis anterior muscles. Formal cardiac and pulmonary evaluations were unremarkable, and electromyography depicted a myopathic pattern. Muscle biopsy revealed mild dystrophic changes with fibre size variation, internal nuclei, rimmed vacuoles, with absent calpain-3 expression on western blot. Whole-genome sequencing revealed that the proband is homozygous for the c.107837A>C (p.His35946Pro) pathogenic TTN variant. His first-degree consanguineous parents are heterozygous for the variant, yet they remain asymptomatic. This case expands the genetic spectrum of biallelic TTN variants associated with LGMD R10.

ИП Урсолов Владлен Сергеевич

ИНН 500118482352 · ОГРНИП 314500117700049

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