• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

一例罕见的1p36缺失综合征婴儿因严重扩张型心肌病继发收缩性心力衰竭。

A Rare Case of an Infant With 1p36 Deletion Syndrome Presenting With Systolic Heart Failure Secondary to Severe Dilated Cardiomyopathy.

作者信息

Ogbuji Chukwunonye O, Ortega Lucio E, Ward Haven, Ugochukwu Nzubechukwu, Donthula Rakesh, Alapati Srilatha

机构信息

Pediatrics, Texas Tech University Health Sciences Center, Amarillo, USA.

Pediatric Cardiology, Rady Children's Hospital, San Diego, USA.

出版信息

Cureus. 2023 Sep 21;15(9):e45746. doi: 10.7759/cureus.45746. eCollection 2023 Sep.

DOI:10.7759/cureus.45746
PMID:37872928
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10590472/
Abstract

1p36 deletion syndrome is a common terminal chromosomal deletion syndrome in humans. It is caused by the deletion of genetic material from a specific region in the short arm of chromosome 1. Symptoms range from seizure disorders, abnormalities of tone, visual and auditory disturbances. Cardiac abnormalities like left ventricular non-compaction (LVNC) and dilated cardiomyopathies (DCM) are commonly associated with this syndrome. This case report presents a 15-month-old female with dilated cardiomyopathy associated with 1p36 deletion syndrome, who has been followed from birth. Cardiac function was normal at birth with an ejection fraction of 65%. At three weeks of age, the patient presented with severe tachypnea, cyanosis, poor weight gain, and diaphoresis with feeding. Echocardiogram showed an ejection fraction of 22%. The patient was diagnosed with Modified Ross Heart Failure Class III. The patient was admitted to the cardiovascular intensive care unit where diuretics, phosphodiesterase inhibitors, and ionotropic agents were used to manage the heart failure. The patient relapsed two months later following a severe adenovirus infection. She was readmitted and heart failure medications were optimized. This patient has maintained a steady growth, meeting most milestones with no further relapse. The heterogeneity of 1p36 deletion syndrome presentation poses a diagnostic challenge for most clinicians. Cardiac involvements are very common and infants presenting with signs and symptoms of heart failure need to be screened for chromosomal abnormalities when other causes have been ruled out.

摘要

1p36缺失综合征是人类常见的末端染色体缺失综合征。它是由1号染色体短臂特定区域的遗传物质缺失引起的。症状包括癫痫障碍、肌张力异常、视觉和听觉障碍。心脏异常如左心室心肌致密化不全(LVNC)和扩张型心肌病(DCM)通常与该综合征相关。本病例报告介绍了一名15个月大患有与1p36缺失综合征相关的扩张型心肌病的女性,自出生起就接受随访。出生时心脏功能正常,射血分数为65%。三周大时,患者出现严重呼吸急促、紫绀、体重增加不佳以及喂食时出汗。超声心动图显示射血分数为22%。患者被诊断为改良罗斯心力衰竭III级。患者被收入心血管重症监护病房,使用利尿剂、磷酸二酯酶抑制剂和正性肌力药物来治疗心力衰竭。两个月后,患者在严重腺病毒感染后复发。她再次入院,心力衰竭药物治疗得到优化。该患者保持稳定生长,达到了大多数发育里程碑,未再复发。1p36缺失综合征表现的异质性给大多数临床医生带来了诊断挑战。心脏受累非常常见,当排除其他病因后,出现心力衰竭体征和症状的婴儿需要进行染色体异常筛查。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ed5/10590472/f5907138824e/cureus-0015-00000045746-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ed5/10590472/0101145f7c14/cureus-0015-00000045746-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ed5/10590472/7dc623633d43/cureus-0015-00000045746-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ed5/10590472/162101b53abf/cureus-0015-00000045746-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ed5/10590472/f5907138824e/cureus-0015-00000045746-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ed5/10590472/0101145f7c14/cureus-0015-00000045746-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ed5/10590472/7dc623633d43/cureus-0015-00000045746-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ed5/10590472/162101b53abf/cureus-0015-00000045746-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ed5/10590472/f5907138824e/cureus-0015-00000045746-i04.jpg

相似文献

1
A Rare Case of an Infant With 1p36 Deletion Syndrome Presenting With Systolic Heart Failure Secondary to Severe Dilated Cardiomyopathy.一例罕见的1p36缺失综合征婴儿因严重扩张型心肌病继发收缩性心力衰竭。
Cureus. 2023 Sep 21;15(9):e45746. doi: 10.7759/cureus.45746. eCollection 2023 Sep.
2
1p36 Deletion Syndrome and Left Ventricular Non-compaction Cardiomyopathy-Two Cases Report.1p36缺失综合征与左心室致密化不全心肌病——两例报告
Front Pediatr. 2021 Jun 7;9:653633. doi: 10.3389/fped.2021.653633. eCollection 2021.
3
Left-ventricular non-compaction in a patient with monosomy 1p36.1p36单体综合征患者的左心室心肌致密化不全
Eur J Med Genet. 2007 May-Jun;50(3):233-6. doi: 10.1016/j.ejmg.2007.01.002. Epub 2007 Jan 27.
4
Left-ventricular non-compaction (LVNC): a clinical feature more often observed in terminal deletion 1p36 than previously expected.左心室心肌致密化不全(LVNC):一种在1p36末端缺失中比先前预期更常观察到的临床特征。
Eur J Med Genet. 2008 Nov-Dec;51(6):685-8. doi: 10.1016/j.ejmg.2008.07.006. Epub 2008 Jul 31.
5
Dental anomalies as a possible clue of 1p36 deletion syndrome due to germline mosaicism: a case report.牙齿异常可能是 1p36 缺失综合征的线索:一例嵌合体病例报告。
BMC Pediatr. 2020 May 9;20(1):201. doi: 10.1186/s12887-020-02049-1.
6
Fine mapping of the 1p36 deletion syndrome identifies mutation of PRDM16 as a cause of cardiomyopathy.1p36 缺失综合征的精细定位确定 PRDM16 突变是心肌病的一个原因。
Am J Hum Genet. 2013 Jul 11;93(1):67-77. doi: 10.1016/j.ajhg.2013.05.015. Epub 2013 Jun 13.
7
Cutis laxa in a patient with 1p36 deletion syndrome.1p36 缺失综合征患者的皮肤松弛症。
J Dermatol. 2018 Jul;45(7):871-873. doi: 10.1111/1346-8138.14311. Epub 2018 Apr 3.
8
Cardiovascular Phenotypic Spectrum of 1p36 Deletion Syndrome.1p36缺失综合征的心血管表型谱
J Pediatr Genet. 2021 Jul 29;12(4):329-334. doi: 10.1055/s-0041-1732473. eCollection 2023 Dec.
9
Left Ventricular Noncompaction Cardiomyopathy in an Elderly Patient: A Case Report and Literature Review.老年患者左心室心肌致密化不全心肌病:一例报告及文献复习
Cureus. 2023 Apr 29;15(4):e38305. doi: 10.7759/cureus.38305. eCollection 2023 Apr.
10
Comparison of Clinical Course and Outcomes between Dilated and Hypokinetic Non-Dilated Cardiomyopathy.扩张型和非扩张性低动力型心肌病的临床过程和结局比较。
Cardiology. 2023;148(5):395-401. doi: 10.1159/000531534. Epub 2023 Jun 13.

本文引用的文献

1
Cardiomyopathies in Children and Systemic Disorders When Is It Useful to Look beyond the Heart?儿童心肌病与全身性疾病:何时超越心脏进行检查才有用?
J Cardiovasc Dev Dis. 2022 Jan 31;9(2):47. doi: 10.3390/jcdd9020047.
2
1p36 Deletion Syndrome and Left Ventricular Non-compaction Cardiomyopathy-Two Cases Report.1p36缺失综合征与左心室致密化不全心肌病——两例报告
Front Pediatr. 2021 Jun 7;9:653633. doi: 10.3389/fped.2021.653633. eCollection 2021.
3
1p36 deletion syndrome: an update.1p36缺失综合征:最新进展
Appl Clin Genet. 2015 Aug 27;8:189-200. doi: 10.2147/TACG.S65698. eCollection 2015.
4
Cardiomyopathy Phenotypes and Outcomes for Children With Left Ventricular Myocardial Noncompaction: Results From the Pediatric Cardiomyopathy Registry.左心室心肌致密化不全患儿的心肌病表型及预后:来自儿童心肌病注册研究的结果
J Card Fail. 2015 Nov;21(11):877-84. doi: 10.1016/j.cardfail.2015.06.381. Epub 2015 Jul 9.
5
GENETIC CAUSES OF DILATED CARDIOMYOPATHY.扩张型心肌病的遗传病因
Prog Pediatr Cardiol. 2014 Dec;37(1-2):13-18. doi: 10.1016/j.ppedcard.2014.10.003.
6
Identification of critical regions and candidate genes for cardiovascular malformations and cardiomyopathy associated with deletions of chromosome 1p36.鉴定与 1p36 缺失相关的心血管畸形和心肌病的关键区域和候选基因。
PLoS One. 2014 Jan 15;9(1):e85600. doi: 10.1371/journal.pone.0085600. eCollection 2014.
7
Further delineation of deletion 1p36 syndrome in 60 patients: a recognizable phenotype and common cause of developmental delay and mental retardation.60例1p36缺失综合征患者的进一步描述:一种可识别的表型及发育迟缓与智力障碍的常见病因
Pediatrics. 2008 Feb;121(2):404-10. doi: 10.1542/peds.2007-0929.
8
Identification of proximal 1p36 deletions using array-CGH: a possible new syndrome.使用阵列比较基因组杂交技术鉴定近端1p36缺失:一种可能的新综合征。
Clin Genet. 2007 Oct;72(4):329-38. doi: 10.1111/j.1399-0004.2007.00876.x.
9
The challenge of cardiomyopathy.心肌病的挑战。
J Am Coll Cardiol. 1989 May;13(6):1219-39. doi: 10.1016/0735-1097(89)90293-3.