Literature DB >> 31416498

[Clinical features and COL4A1 genotype of a toddler with hereditary angiopathy with nephropathy, aneurysms and muscle cramps syndrome].

Li-Dan Shan1, Jing Peng, Hui Xiao, Li-Wen Wu, Hao-Lin Duan, Nan Pang, Kessi Miriam, Fei Yin.   

Abstract

Hereditary angiopathy with nephropathy, aneurysms and muscle cramps (HANAC) syndrome is an autosomal dominant genetic disease caused by COL4A1 gene mutation, with major clinical manifestations of white matter lesion, aneurysm, retinal artery tortuosity, polycystic kidney, microscopic hematuria and muscle cramps. This article reports the clinical features and genotype of one toddler with HANAC syndrome caused by COL4A1 gene mutation. The boy, aged 1 year and 8 months, had an insidious onset, with the clinical manifestations of pyrexia and convulsion, white matter lesions in the periventricular region and the centrum semiovale on both sides, softening lesions beside the left basal ganglia, retinal arteriosclerosis, microscopic hematuria and muscle cramps. Whole exome sequencing revealed a pathogenic de novo heterozygous mutation in the COL4A1 gene, (NM_001845) c.4150+1(IVS46)G>T, and therefore, the boy was diagnosed with HANAC syndrome. COL4A1 gene mutation detection should be performed for children with unexplained white matter lesion, stroke, hematuria, polycystic kidney, cataract and retinal artery tortuosity or families with related history.

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Year:  2019        PMID: 31416498      PMCID: PMC7389900     

Source DB:  PubMed          Journal:  Zhongguo Dang Dai Er Ke Za Zhi        ISSN: 1008-8830


  24 in total

1.  Intracranial sonographic features demonstrating in utero development of hemorrhagic brain damage leading to schizencephaly-associated COL4A1 mutation.

Authors:  Tadashi Matsumoto; Kei Miyakoshi; Marie Fukutake; Daigo Ochiai; Kazuhiro Minegishi; Mamoru Tanaka
Journal:  J Med Ultrason (2001)       Date:  2015-01-18       Impact factor: 1.314

2.  Ophthalmological features associated with COL4A1 mutations.

Authors:  Isabelle Coupry; Igor Sibon; Bruno Mortemousque; François Rouanet; Manuele Mine; Cyril Goizet
Journal:  Arch Ophthalmol       Date:  2010-04

3.  Severe Hemolytic Jaundice in a Neonate with a Novel COL4A1 Mutation.

Authors:  Seiichi Tomotaki; Hiroshi Mizumoto; Takayuki Hamabata; Akira Kumakura; Mitsutaka Shiota; Hiroshi Arai; Kazuhiro Haginoya; Daisuke Hata
Journal:  Pediatr Neonatol       Date:  2014-05-23       Impact factor: 2.083

4.  Hereditary endotheliopathy with retinopathy, nephropathy, and stroke (HERNS).

Authors:  J Jen; A H Cohen; Q Yue; J T Stout; H V Vinters; S Nelson; R W Baloh
Journal:  Neurology       Date:  1997-11       Impact factor: 9.910

5.  Novel COL4A1 mutation in an infant with severe dysmorphic syndrome with schizencephaly, periventricular calcifications, and cataract resembling congenital infection.

Authors:  Robert Smigiel; Magdalena Cabala; Aleksandra Jakubiak; Hirofumi Kodera; Marek J Sasiadek; Naomichi Matsumoto; Maria M Sasiadek; Hirotomo Saitsu
Journal:  Birth Defects Res A Clin Mol Teratol       Date:  2016-02-16

6.  Porencephaly in a fetus and HANAC in her father: variable expression of COL4A1 mutation.

Authors:  Toshiki Takenouchi; Masaki Ohyagi; Chiharu Torii; Rika Kosaki; Takao Takahashi; Kenjiro Kosaki
Journal:  Am J Med Genet A       Date:  2014-11-25       Impact factor: 2.802

7.  Novel mutations in three families confirm a major role of COL4A1 in hereditary porencephaly.

Authors:  G Breedveld; I F de Coo; M H Lequin; W F M Arts; P Heutink; D B Gould; S W M John; B Oostra; G M S Mancini
Journal:  J Med Genet       Date:  2005-08-17       Impact factor: 6.318

Review 8.  Cadasil.

Authors:  Hugues Chabriat; Anne Joutel; Martin Dichgans; Elizabeth Tournier-Lasserve; Marie-Germaine Bousser
Journal:  Lancet Neurol       Date:  2009-07       Impact factor: 44.182

9.  COL4A1 mutation in preterm intraventricular hemorrhage.

Authors:  Kaya Bilguvar; Michael L DiLuna; Matthew J Bizzarro; Yasar Bayri; Karen C Schneider; Richard P Lifton; Murat Gunel; Laura R Ment
Journal:  J Pediatr       Date:  2009-11       Impact factor: 4.406

10.  Use of sodium 4-phenylbutyrate to define therapeutic parameters for reducing intracerebral hemorrhage and myopathy in Col4a1 mutant mice.

Authors:  Genki Hayashi; Cassandre Labelle-Dumais; Douglas B Gould
Journal:  Dis Model Mech       Date:  2018-07-04       Impact factor: 5.758

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  1 in total

Review 1.  Reduced anogenital distance, hematuria and left renal hypoplasia in a patient with 13q33.1-34 deletion: case report and literature review.

Authors:  Xue He; Huijun Shen; Haidong Fu; Chunyue Feng; Zhixia Liu; Yanyan Jin; Jianhua Mao
Journal:  BMC Pediatr       Date:  2020-07-02       Impact factor: 2.125

  1 in total

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