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家族性 LEOPARD 综合征伴肥厚型心肌病。

Familial LEOPARD Syndrome With Hypertrophic Cardiomyopathy.

机构信息

Aurora Cardiovascular and Thoracic Services, Aurora Sinai/Aurora St. Luke's Medical Centers, University of Wisconsin School of Medicine and Public Health, Milwaukee, Wisconsin.

Aurora Cardiovascular and Thoracic Services, Aurora Sinai/Aurora St. Luke's Medical Centers, University of Wisconsin School of Medicine and Public Health, Milwaukee, Wisconsin.

出版信息

Am J Cardiol. 2020 Nov 15;135:168-173. doi: 10.1016/j.amjcard.2020.08.027. Epub 2020 Aug 28.

DOI:10.1016/j.amjcard.2020.08.027
PMID:32866449
Abstract

Multiple lentigines syndrome is an autosomal dominant inherited condition with variable expressivity that is also known as LEOPARD syndrome. LEOPARD stands for lentigines, electrocardiographic conduction defects, ocular hypertelorism, pulmonary valve stenosis, abnormalities of genitalia, retardation of growth, and deafness. LEOPARD syndrome most frequently develops secondary to a missense mutation of protein-tyrosine phosphatase nonreceptor type 11 gene, which encodes tyrosine phosphatase. The missense mutation p.Tyr279Cys can either occur as a de novo mutation or affect multiple family members. Although hypertrophic cardiomyopathy is not part of the LEOPARD acronym, it is the most frequent cardiac anomaly observed in this syndrome. The recognition of increased left or right ventricular wall thickness in patients with LEOPARD syndrome may have significant impact on their clinical course similar to classic hypertrophic cardiomyopathy, which may require septal reduction procedures for relief of left or right ventricular outflow tract obstruction or implantable cardioverter-defibrillator placement for sudden cardiac death prevention. We describe a case series of a family with diffuse lentigines and hypertrophic cardiomyopathy in which the son carries the protein-tyrosine phosphatase nonreceptor type 11 (p.Tyr279Cys) gene mutation and both the son and daughter underwent left ventricular myectomy at an early age. In conclusion, our case series of a family with LEOPARD syndrome illustrates the importance of recognizing hypertrophic cardiomyopathy as part of this syndrome.

摘要

多发性黑子综合征是一种常染色体显性遗传疾病,具有可变的外显率,也被称为 LEOPARD 综合征。LEOPARD 代表黑子、心电图传导缺陷、眼距过宽、肺动脉瓣狭窄、生殖器异常、生长迟缓以及耳聋。LEOPARD 综合征最常继发于蛋白酪氨酸磷酸酶非受体型 11 基因的错义突变,该基因编码酪氨酸磷酸酶。错义突变 p.Tyr279Cys 既可以是新生突变,也可以影响多个家族成员。尽管肥厚型心肌病不是 LEOPARD 缩写的一部分,但它是该综合征中最常见的心脏异常。在 LEOPARD 综合征患者中,识别出左心室或右心室壁增厚可能对其临床病程有重大影响,类似于经典肥厚型心肌病,这可能需要进行室间隔切除术以缓解左心室或右心室流出道梗阻,或植入心脏复律除颤器以预防心源性猝死。我们描述了一个家族的病例系列,该家族有弥漫性黑子和肥厚型心肌病,其儿子携带蛋白酪氨酸磷酸酶非受体型 11 基因(p.Tyr279Cys)突变,儿子和女儿都在早期接受了左心室心肌切除术。总之,我们对一个 LEOPARD 综合征家族的病例系列研究说明了认识到肥厚型心肌病是该综合征的一部分的重要性。

相似文献

1
Familial LEOPARD Syndrome With Hypertrophic Cardiomyopathy.家族性 LEOPARD 综合征伴肥厚型心肌病。
Am J Cardiol. 2020 Nov 15;135:168-173. doi: 10.1016/j.amjcard.2020.08.027. Epub 2020 Aug 28.
2
Importance of cardiovascular examination in patients with multiple lentigines: two cases of LEOPARD syndrome with hypertrophic cardiomyopathy.多发性雀斑样痣患者心血管检查的重要性:两例伴有肥厚型心肌病的豹斑综合征病例
Acta Clin Belg. 2019 Apr;74(2):82-85. doi: 10.1080/17843286.2018.1467531. Epub 2018 May 2.
3
LEOPARD syndrome: a variant of Noonan syndrome strongly associated with hypertrophic cardiomyopathy.豹斑综合征:一种与肥厚型心肌病密切相关的努南综合征变异型。
Rev Esp Cardiol (Engl Ed). 2013 May;66(5):350-6. doi: 10.1016/j.rec.2012.09.015. Epub 2013 Jan 11.
4
Leopard syndrome.豹皮综合征
Orphanet J Rare Dis. 2008 May 27;3:13. doi: 10.1186/1750-1172-3-13.
5
Familial aggregation of genetically heterogeneous hypertrophic cardiomyopathy: a boy with LEOPARD syndrome due to PTPN11 mutation and his nonsyndromic father lacking PTPN11 mutations.基因异质性肥厚型心肌病的家族聚集性:一名因PTPN11突变患有豹皮综合征的男孩及其无PTPN11突变的非综合征型父亲。
Birth Defects Res A Clin Mol Teratol. 2004 Feb;70(2):95-8. doi: 10.1002/bdra.10148.
6
LEOPARD syndrome and hypertrophic obstructive cardiomyopathy: a case report.
Turk J Pediatr. 2004 Oct-Dec;46(4):373-6.
7
Phenotypical diversity of patients with LEOPARD syndrome carrying the worldwide recurrent p.Tyr279Cys PTPN11 mutation.携带全球复发性p.Tyr279Cys PTPN11突变的豹皮综合征患者的表型多样性。
Arch Dermatol Res. 2015 Dec;307(10):891-5. doi: 10.1007/s00403-015-1597-4. Epub 2015 Sep 16.
8
LEOPARD syndrome without hearing loss or pulmonary stenosis: a report of 2 cases.无听力丧失或肺动脉狭窄的豹综合征:2例报告
Actas Dermosifiliogr. 2015 May;106(4):e19-22. doi: 10.1016/j.ad.2014.11.004. Epub 2014 Dec 24.
9
LEOPARD Syndrome Caused by Tyr279Cys Mutation in the PTPN11 Gene.由PTPN11基因中Tyr279Cys突变引起的豹皮综合征
Mol Syndromol. 2012 Apr;2(6):251-253. doi: 10.1159/000335995. Epub 2012 Jan 31.
10
Atypical left-ventricular hypertrophy with apical aneurysm in leopard syndrome.豹综合征患者出现非典型性左心室肥厚伴心尖部瘤样扩张。
Int J Cardiovasc Imaging. 2024 Aug;40(8):1809-1811. doi: 10.1007/s10554-024-03185-w. Epub 2024 Jul 8.

引用本文的文献

1
Case Report: Left ventricular apical hypertrophy in a patient with Leopard syndrome mimicking a cardiac tumor: a diagnostic challenge resolved by multimodality imaging.病例报告:豹皮综合征患者的左心室心尖肥厚酷似心脏肿瘤:多模态成像解决的诊断难题
Front Cardiovasc Med. 2024 Jul 22;11:1378078. doi: 10.3389/fcvm.2024.1378078. eCollection 2024.