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肺泡毛细血管发育不良伴肺静脉异位连接:一例足月新生儿的外科肺活检和尸检。

Alveolar capillary dysplasia with misalignment of the pulmonary veins: A surgical lung biopsy and autopsy in a full-term newborn.

机构信息

Servicio de Anatomía Patológica, Hospital Universitari i Politècnic La Fe, València, Spain.

Servicio de Anatomía Patológica, Hospital Universitari i Politècnic La Fe, València, Spain.

出版信息

Rev Esp Patol. 2024 Oct-Dec;57(4):305-308. doi: 10.1016/j.patol.2024.06.005. Epub 2024 Jul 23.

Abstract

Alveolar capillary dysplasia with misalignment of the pulmonary veins (ACD/MPV) is a rare and lethal interstitial lung disorder, caused by a congenital abnormality affecting the development of the parenchyma and pulmonary vessels. We report the case of a newborn at the end of 40 weeks of pregnancy, who showed no cardiopulmonary anomalies in prenatal control ultrasounds. However, after delivery, pulmonary hypertension and hypoxemic respiratory failure became apparent. She died after 12 days from refractory hemodynamic and respiratory failure despite intensive therapy. A surgical lung biopsy and clinical autopsy were performed, both revealing the same histopathological signs consistent with this disorder. In our case, the findings of digestive and genital malformations, together with the genetic result of the alteration in the FOXF1 gene, led us to conclude the definitive diagnosis of alveolar capillary dysplasia.

摘要

肺静脉异位连接性肺泡毛细血管发育不良(ACD/MPV)是一种罕见且致命的间质性肺疾病,由影响实质和肺血管发育的先天性异常引起。我们报告了一例妊娠 40 周末期的新生儿,其产前超声检查未显示心肺异常。然而,分娩后出现肺动脉高压和低氧性呼吸衰竭。尽管进行了强化治疗,她仍在 12 天后因难治性血流动力学和呼吸衰竭而死亡。进行了外科肺活检和临床尸检,均显示出相同的组织病理学特征,符合这种疾病。在我们的病例中,消化道和生殖器畸形的发现,以及 FOXF1 基因改变的基因结果,使我们得出了肺泡毛细血管发育不良的明确诊断。

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