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The clinical spectrum of a nonsense mutation in KAT6A: a case report

KAT6A syndrome is an autosomal dominant genetic disorder associated with intellectual disability due to mutations in the lysine acetyltransferase 6A (KAT6A) gene. There are some differences in phenotype between KAT6A gene variants. This current case report describes a 1-month-old male infant that ha...

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Autores principales: Wang, Dongbo, He, Jun, Li, Xueyi, Yan, Shuyuan, Pan, Linglin, Wang, Tuanmei, Zhou, Liangrong, Liu, Jiyang, Peng, Xiangwen
Formato: Online Artículo Texto
Lenguaje:English
Publicado: SAGE Publications 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9806384/
https://www.ncbi.nlm.nih.gov/pubmed/36573038
http://dx.doi.org/10.1177/03000605221140304
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author Wang, Dongbo
He, Jun
Li, Xueyi
Yan, Shuyuan
Pan, Linglin
Wang, Tuanmei
Zhou, Liangrong
Liu, Jiyang
Peng, Xiangwen
author_facet Wang, Dongbo
He, Jun
Li, Xueyi
Yan, Shuyuan
Pan, Linglin
Wang, Tuanmei
Zhou, Liangrong
Liu, Jiyang
Peng, Xiangwen
author_sort Wang, Dongbo
collection PubMed
description KAT6A syndrome is an autosomal dominant genetic disorder associated with intellectual disability due to mutations in the lysine acetyltransferase 6A (KAT6A) gene. There are some differences in phenotype between KAT6A gene variants. This current case report describes a 1-month-old male infant that had a nonsense mutation in the KAT6A gene. Neither of his parents had the mutation. The proband had feeding difficulties and a physical examination revealed the following: moderate dysphagia, hypoplastic laryngeal cartilage, poor audio-visual response, poor head-up ability, no active grasping awareness, microcephaly, high arched palate and he was significantly behind other children of the same age. Echocardiography showed that the foramen ovale was not closed. He was diagnosed with atrial septal defect (ASD) when 2 years old. The patient received ASD repair at 32 months of age. Head colour Doppler ultrasonography and brain magnetic resonance imaging showed cysts in the right ventricle and choroid plexus, which returned to normal at 2 years of age. This current case demonstrates that immediate surgery should be considered in newborns with KAT6A syndrome presenting with a heart malformation. A new KAT6A syndrome phenotype is described in this current case report, which requires early diagnosis and treatment.
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spelling pubmed-98063842023-01-03 The clinical spectrum of a nonsense mutation in KAT6A: a case report Wang, Dongbo He, Jun Li, Xueyi Yan, Shuyuan Pan, Linglin Wang, Tuanmei Zhou, Liangrong Liu, Jiyang Peng, Xiangwen J Int Med Res Case Reports KAT6A syndrome is an autosomal dominant genetic disorder associated with intellectual disability due to mutations in the lysine acetyltransferase 6A (KAT6A) gene. There are some differences in phenotype between KAT6A gene variants. This current case report describes a 1-month-old male infant that had a nonsense mutation in the KAT6A gene. Neither of his parents had the mutation. The proband had feeding difficulties and a physical examination revealed the following: moderate dysphagia, hypoplastic laryngeal cartilage, poor audio-visual response, poor head-up ability, no active grasping awareness, microcephaly, high arched palate and he was significantly behind other children of the same age. Echocardiography showed that the foramen ovale was not closed. He was diagnosed with atrial septal defect (ASD) when 2 years old. The patient received ASD repair at 32 months of age. Head colour Doppler ultrasonography and brain magnetic resonance imaging showed cysts in the right ventricle and choroid plexus, which returned to normal at 2 years of age. This current case demonstrates that immediate surgery should be considered in newborns with KAT6A syndrome presenting with a heart malformation. A new KAT6A syndrome phenotype is described in this current case report, which requires early diagnosis and treatment. SAGE Publications 2022-12-26 /pmc/articles/PMC9806384/ /pubmed/36573038 http://dx.doi.org/10.1177/03000605221140304 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by-nc/4.0/Creative Commons Non Commercial CC BY-NC: This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 License (https://creativecommons.org/licenses/by-nc/4.0/) which permits non-commercial use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access pages (https://us.sagepub.com/en-us/nam/open-access-at-sage).
spellingShingle Case Reports
Wang, Dongbo
He, Jun
Li, Xueyi
Yan, Shuyuan
Pan, Linglin
Wang, Tuanmei
Zhou, Liangrong
Liu, Jiyang
Peng, Xiangwen
The clinical spectrum of a nonsense mutation in KAT6A: a case report
title The clinical spectrum of a nonsense mutation in KAT6A: a case report
title_full The clinical spectrum of a nonsense mutation in KAT6A: a case report
title_fullStr The clinical spectrum of a nonsense mutation in KAT6A: a case report
title_full_unstemmed The clinical spectrum of a nonsense mutation in KAT6A: a case report
title_short The clinical spectrum of a nonsense mutation in KAT6A: a case report
title_sort clinical spectrum of a nonsense mutation in kat6a: a case report
topic Case Reports
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9806384/
https://www.ncbi.nlm.nih.gov/pubmed/36573038
http://dx.doi.org/10.1177/03000605221140304
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