一例年轻患者主动脉夹层和先前未报道的ACTA2错义变异突变。

IF 1.3 4区 医学 Q3 PATHOLOGY
Pediatric and Developmental Pathology Pub Date : 2023-09-01 Epub Date: 2023-09-06 DOI:10.1177/10935266231194701
Satoshi Marutani, Takako Nishino, Osamu Shimokawa, Ritsuko K Pooh, Hiroko Morisaki, Noburu Inamura
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引用次数: 0

摘要

已知遗传性结缔组织病可在早期引起主动脉病变。家族性主动脉瘤/夹层是由于影响平滑肌结构的ACTA2突变引起的。我们提出一个15岁的男孩与轻度发育障碍的情况下,在以前的身体检查中没有发现异常。患者表现为严重的左心衰,从升主动脉到髂总动脉广泛剥离,心肌和脑梗死。他接受了紧急的本塔尔手术。6个月后,患者接受了从主动脉弓处重建腹主动脉的手术,恢复了正常的日常活动。病理检查显示没有弹性纤维,但从膜内膜到中膜有丰富的网状纤维和粘多糖。基因检测显示ACTA2基因的杂合错义变异。据我们所知,这是第一例散发性平滑肌组织结构异常导致临床症状的病例,以前没有报道过致病性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Aortic Dissection and a Previously Unreported ACTA2 Missense Variant Mutation in a Young Patient: A Case Report.

Hereditary connective tissue disease is known to cause aortic lesions at an early age. Familial aortic aneurysm/dissection is caused due to an ACTA2 mutation that affects smooth muscle structure. We present a case of a 15-year-old boy with a mild developmental disorder in whom no abnormalities were identified on previous physical examinations. The patient presented with severe left heart failure, extensive dissection from the ascending aorta to the common iliac artery, and myocardial and cerebral infarctions. He underwent an urgent Bentall surgery. Six months later, the patient underwent surgical reconstruction of the abdominal aorta from the aortic arch and returned to normal daily activities. Pathological examination demonstrated the absence of elastic fibers but presence of abundant reticular fibers and mucopolysaccharides from the tunica intima to the media. Genetic testing revealed a heterozygous missense variant of the ACTA2 gene. To the best of our knowledge, this is the first sporadic case of structurally abnormal smooth muscle organization resulting in clinical symptoms with no previously reported pathogenicity.

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来源期刊
CiteScore
3.70
自引率
5.30%
发文量
59
审稿时长
6-12 weeks
期刊介绍: The Journal covers the spectrum of disorders of early development (including embryology, placentology, and teratology), gestational and perinatal diseases, and all diseases of childhood. Studies may be in any field of experimental, anatomic, or clinical pathology, including molecular pathology. Case reports are published only if they provide new insights into disease mechanisms or new information.
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