Kryczka Karolina E, Dzielińska Zofia, Franaszczyk Maria, Wojtkowska Izabela, Henzel Jan, Śpiewak Mateusz, Stępińska Janina, Bilińska Zofia T, Płoski Rafał, Demkow Marcin
Department of Coronary and Structural Heart Diseases, Institute of Cardiology, Warsaw, Poland.
Department of Medical Biology, Molecular Biology Laboratory, Institute of Cardiology, Warsaw, Poland.
Am J Case Rep. 2018 Jul 12;19:820-824. doi: 10.12659/AJCR.909601.
BACKGROUND Peripartum cardiomyopathy (PPCM) is a potentially life-threatening, pregnancy-associated cause of heart failure affecting previously healthy women. Recent research suggests a possible role of 16-kDa prolactin in promoting cardiomyocyte damage. However, the genetic predisposition is not well recognized. CASE REPORT We report the case of a 25-year-old woman with a severe course of PPCM with left ventricle ejection fraction of 25-30%, complicated by ventricular arrhythmia and postpartum thyroiditis. As no traditional risk factors of PPCM were identified, the patient was referred for genetic testing. Next-generation sequencing revealed a novel titin gene-truncating mutation NM_001267550: p.Leu23499fs/c.70497_40498insT in the proband as well as in her mother. In the patient, a very late recovery >12 months postpartum was observed, which required long-term medical treatment with bromocriptine. CONCLUSIONS PPCM may occur in women with the genetic predisposition, being modified by an interaction of biological factors, such as a high prolactin level, a ventricular arrhythmia, and an autoimmune disorder. Recovery from severe heart failure due to an inherited cardiomyopathy is possible with careful and appropriate medical management.
围产期心肌病(PPCM)是一种潜在危及生命的、与妊娠相关的心力衰竭病因,影响既往健康的女性。近期研究表明16-kDa催乳素在促进心肌细胞损伤方面可能起作用。然而,遗传易感性尚未得到充分认识。病例报告:我们报告一例25岁女性,患有严重的PPCM,左心室射血分数为25%-30%,并发室性心律失常和产后甲状腺炎。由于未发现PPCM的传统危险因素,该患者被转诊进行基因检测。下一代测序在先证者及其母亲中发现了一种新的肌联蛋白基因截短突变NM_001267550:p.Leu23499fs/c.70497_40498insT。在该患者中,观察到产后12个月以上的非常晚期恢复,这需要用溴隐亭进行长期药物治疗。结论:PPCM可能发生在具有遗传易感性的女性中,受生物因素如高催乳素水平、室性心律失常和自身免疫性疾病相互作用的影响。通过仔细和适当的医疗管理,因遗传性心肌病导致的严重心力衰竭有可能恢复。