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DOI 10.1542/peds.2023-063054

Медицинская библиотека · Pediatrics · 2024

Molecular Autopsy With Banked Cord Blood Reveals Brugada Syndrome in Past Sudden Death Case.

Kume Eitaro, Yamakawa Masaru, Miyakoshi Chisato, Aota Chie, Tsuruta Satoru, Horie Minoru, Ohno Seiko

СтатьяКлинический случайПоддержка исследования: вне правительства СШАПеревод пока не подготовлен

Аннотация

Molecular autopsy has recently been gaining attention as a means of postmortem diagnosis; however, it is usually performed using the victim's blood sample at the time of death. Here, we report the first case of a deceased infant with Brugada syndrome whose diagnosis was made with banked cord blood. A seemingly healthy 1-year-old male infant collapsed while having a fever; this collapse was witnessed by his mother. Despite cardiopulmonary resuscitation, he died of ventricular fibrillation. No abnormalities of cardiac structure were identified on autopsy. Genomic samples were not stored at the time because of a lack of suspicion for familial arrhythmia. Five years later, his sister showed Brugada electrocardiogram pattern while febrile from Kawasaki disease. Their father showed a spontaneous type 1 Brugada electrocardiogram pattern. A heterozygous SCN5A p.R893C variant was found by genetic testing in the proband's father and sister. Furthermore, the proband's genetic testing was performed using his banked cord blood, which identified the same variant. Family history of Brugada syndrome with an SCN5A-R893C variant and clinical evidence led to a postmortem diagnosis of Brugada syndrome in the proband. Identification of this variant in this case later contributed to verifying SCN5A-R893C as a pathogenic variant through data accumulation. Banked cord blood may prove useful for conducting molecular autopsies in previously undiagnosed cases of sudden death in which genomic samples were not stored.

Для цитирования: Kume Eitaro, Yamakawa Masaru, Miyakoshi Chisato, Aota Chie, Tsuruta Satoru, Horie Minoru, Ohno Seiko. Molecular Autopsy With Banked Cord Blood Reveals Brugada Syndrome in Past Sudden Death Case.. Pediatrics. 2024. DOI: 10.1542/peds.2023-063054.

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