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DOI 10.1002/ajmg.a.63458

Медицинская библиотека · American journal of medical genetics. Part A · 2023

Aortic dissection in a young male with persistent ductus arteriosus and a novel variant in MYLK.

Boelman Maria Bejerholm, Hansen Thomas van Overeem, Smith Matthias Nybro, Hammer-Hansen Sophia, Christensen Alex Hørby, Diness Birgitte Rode

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Аннотация

Pathogenic variants in several genes involved in the function or regulation of smooth muscle cells (SMC) are known to predispose to congenital heart disease and thoracic aortic aneurysm and dissection (TAAD). Variants in MYLK are primarily known to predispose to TAAD, but a growing body of evidence points toward MYLK also playing an essential role in the regulation of SMC contraction outside the aorta. In this case report, we present a patient with co-occurrence of persistent ductus arteriosus (PDA) and thoracic aortic dissection. Genetic analyses revealed a novel splice acceptor variant (c.3986-1G > A) in MYLK, which segregated with disease in the family. RNA-analyses on fibroblasts showed that the variant induced skipping of exon 24, which resulted in an in-frame deletion of 101 amino acids. These findings suggest that MYLK-associated disease could include a broader phenotypic spectrum than isolated TAAD, including PDA and obstructive pulmonary disease. Genetic analyses could be considered in families with TAAD and PDA or obstructive pulmonary disease.

Для цитирования: Boelman Maria Bejerholm, Hansen Thomas van Overeem, Smith Matthias Nybro, Hammer-Hansen Sophia, Christensen Alex Hørby, Diness Birgitte Rode. Aortic dissection in a young male with persistent ductus arteriosus and a novel variant in MYLK.. American journal of medical genetics. Part A. 2023. DOI: 10.1002/ajmg.a.63458.

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