Key result
Late initiation of ataluren at age 12 years and 9 months in a boy with Duchenne muscular dystrophy was associated with stabilization of motor function and a decrease in creatine kinase levels over 2 years.
Why the study?
Earlier introduction of ataluren in DMD seems to give better results, but clinical experience with late initiation of treatment remains limited.
Does late treatment with ataluren stabilize functional status in a boy with Duchenne muscular dystrophy?
Case Report (n=1)
No
Does late treatment with ataluren stabilize functional status in a boy with Duchenne muscular dystrophy?
Late initiation of ataluren at age 12 in a boy with DMD was associated with functional stabilization and decreased creatine kinase levels over 2 years.
Case suggests benefit of earlier ataluren in nonsense-mutation DMD; leaves open need for controlled trials.
Duchenne muscular dystrophy (DMD) is a severe, progressive X-linked recessive disorder, caused by the absence of the dystrophin protein. A resolutive therapy for DMD is not yet available. The first approved drug for DMD patients with nonsense mutations is ataluren, approved for the treatment of children aged ≥ 2 yrs, that seems effective in slowing the disease progression. An earlier introduction of ataluren seems to give better results. We report the case of a 14-year-old DMD patient with a nonsense mutation in exon 70, still ambulant, who started taking ataluren at 12 years and remained stable for the following two years. The patient was on steroid since the age of 6, with beneficial effects. At two-years follow-up, an optimal disease evolution was observed, associated with a constant decrease of creatine kinase blood levels. Despite the late start of the treatment, ataluren seems to have significantly contributed to the stabilization of the functional status in this patient though it cannot be excluded that the result may have been influenced by the previous favorable course of the disease. However, further studies should be planned in patients with similar age treated with ataluren to better evaluate the treatment's results compared to the natural course of the disease.
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Pasca et al. (2022) conducted a case report in Duchenne muscular dystrophy (n=1). Ataluren was evaluated on Motor function and creatine kinase levels. Late initiation of ataluren at age 12 years and 9 months in a boy with Duchenne muscular dystrophy was associated with stabilization of motor function and a decrease in creatine kinase levels over 2 years.
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