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一名患有唐氏综合征和单纯继发孔型房间隔缺损的6个月大婴儿的重度早发型肺动脉高压

Severe Early-Onset Pulmonary Hypertension in a Six-Month-Old With Down Syndrome and Isolated Secundum Atrial Septal Defect.

作者信息

Abeer Fatima, Wani Aasim Ayaz, Javid Bisma, Mahmood Aisha, Andleeb Gazala

机构信息

Department of Internal Medicine, Government Medical College, Srinagar, Srinagar, IND.

Department of Engineering, National Institute of Technology Srinagar, Srinagar, IND.

出版信息

Cureus. 2025 May 13;17(5):e84019. doi: 10.7759/cureus.84019. eCollection 2025 May.

DOI:10.7759/cureus.84019
PMID:40510055
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12160662/
Abstract

Infants with Down syndrome (trisomy 21) commonly present with congenital heart defects and immune dysregulation, significantly increasing the risk of early-onset pulmonary arterial hypertension (PAH). Although secundum atrial septal defects (ASDs) are often considered hemodynamically mild in non-syndromic children, they can progress aggressively in the presence of trisomy 21. We describe a six-month-old male infant with karyotype-confirmed trisomy 21 who developed severe PAH secondary to a rapidly enlarging secundum ASD - a highly atypical presentation for an isolated lesion. The infant presented with fever, respiratory distress, vomiting, and diarrhea, alongside a clinical history of neonatal sepsis, recurrent infections, failure to thrive (weight below the 5th percentile), and subclinical hypothyroidism (TSH 8.12 μIU/mL). Echocardiography revealed that the ASD had enlarged from 6 mm at five months to 10 mm, creating a substantial left-to-right shunt (Qp:Qs >1.5:1). Management with IV ceftriaxone, sildenafil (2 mg twice daily), supplemental oxygen, and nutritional support stabilized the infant within five days (SpO₂ 93-94% on room air). He was discharged for deferred surgical ASD closure, highlighting the value of early pulmonary vasodilator therapy as a bridge to definitive repair. This case underscores the markedly increased susceptibility of infants with Down syndrome to severe PAH, even in the setting of a seemingly hemodynamically insignificant ASD. Early cardiac evaluation, prompt intervention, and multidisciplinary management are crucial to preventing irreversible pulmonary vascular disease in this high-risk population.

摘要

患有唐氏综合征(21三体综合征)的婴儿通常伴有先天性心脏缺陷和免疫失调,这显著增加了早发性肺动脉高压(PAH)的风险。虽然继发孔型房间隔缺损(ASD)在非综合征儿童中通常被认为血流动力学影响较轻,但在21三体综合征患儿中可能会迅速进展。我们描述了一名6个月大的男婴,其核型确诊为21三体综合征,因继发孔型ASD迅速增大而发展为严重PAH,这是一种孤立病变的高度非典型表现。该婴儿出现发热、呼吸窘迫、呕吐和腹泻,同时有新生儿败血症、反复感染、发育不良(体重低于第5百分位)和亚临床甲状腺功能减退(促甲状腺激素8.12 μIU/mL)的病史。超声心动图显示,ASD从5个月时的6毫米扩大到10毫米,形成了大量的左向右分流(肺循环血流量与体循环血流量之比>1.5:1)。通过静脉注射头孢曲松、西地那非(每日两次,每次2毫克)、补充氧气和营养支持,婴儿在5天内病情稳定(室内空气下血氧饱和度93 - 94%)。他出院等待延期进行ASD封堵手术,这凸显了早期肺血管扩张剂治疗作为确定性修复桥梁的价值。该病例强调了唐氏综合征婴儿即使在看似血流动力学影响不大的ASD情况下,对严重PAH的易感性也显著增加。早期心脏评估、及时干预和多学科管理对于预防这一高危人群中不可逆的肺血管疾病至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/35f1/12160662/c83d9fd77d6d/cureus-0017-00000084019-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/35f1/12160662/c83d9fd77d6d/cureus-0017-00000084019-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/35f1/12160662/c83d9fd77d6d/cureus-0017-00000084019-i01.jpg

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本文引用的文献

1
Common shunt lesions with pulmonary hypertension-who will benefit from surgery?伴有肺动脉高压的常见分流性病变——谁将从手术中获益?
Indian J Thorac Cardiovasc Surg. 2025 Jun;41(6):718-729. doi: 10.1007/s12055-024-01786-7. Epub 2024 Aug 13.
2
Pulmonary Hypertension Definition, Classification, and Epidemiology in Asia.亚洲肺动脉高压的定义、分类及流行病学
JACC Asia. 2022 Aug 2;2(5):538-546. doi: 10.1016/j.jacasi.2022.04.008. eCollection 2022 Oct.
3
Endocrinal dysfunction in children with Down syndrome.唐氏综合征患儿的内分泌功能障碍。
Ann Pediatr Endocrinol Metab. 2022 Mar;27(1):15-21. doi: 10.6065/apem.2142236.118. Epub 2022 Mar 31.
4
Congenital heart defects among Down's syndrome cases: an updated review from basic research to an emerging diagnostics technology and genetic counselling.唐氏综合征患者的先天性心脏缺陷:从基础研究到新兴诊断技术和遗传咨询的最新综述。
J Genet. 2021;100.
5
Adults with Down syndrome challenge another paradigm: When aging no longer entails arterial hypertension.唐氏综合征患者挑战了另一个范式:当衰老不再伴随着动脉高血压。
J Clin Hypertens (Greenwich). 2020 Jul;22(7):1127-1133. doi: 10.1111/jch.13930. Epub 2020 Jul 9.
6
The Link between Thyroid Autoimmunity, Depression and Bipolar Disorder.甲状腺自身免疫、抑郁症和双相情感障碍之间的联系。
Open Med (Wars). 2019 Jan 16;14:52-58. doi: 10.1515/med-2019-0008. eCollection 2019.
7
Current understanding and perioperative management of pediatric pulmonary hypertension.小儿肺动脉高压的当前认识与围手术期管理
Paediatr Anaesth. 2019 May;29(5):441-456. doi: 10.1111/pan.13542. Epub 2018 Dec 19.
8
Pulmonary arterial hypertension: pathogenesis and clinical management.肺动脉高压:发病机制与临床管理
BMJ. 2018 Mar 14;360:j5492. doi: 10.1136/bmj.j5492.
9
The regulator of calcineurin (RCAN1) an important factor involved in atherosclerosis and cardiovascular diseases development.钙调神经磷酸酶调节剂(RCAN1)是参与动脉粥样硬化和心血管疾病发展的一个重要因素。
J Med Life. 2014 Oct-Dec;7(4):481-7.
10
Infections and immunodeficiency in Down syndrome.唐氏综合征相关的感染与免疫缺陷。
Clin Exp Immunol. 2011 Apr;164(1):9-16. doi: 10.1111/j.1365-2249.2011.04335.x. Epub 2011 Feb 24.