A case of Kabuki syndrome with congenital pulmonary airway malformation

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Authors: Lan Yang, Qinghui Chen, Weifang Zhou, Jianping Deng, Jianmei Tian
Cited by: 0
References: 23

Abstract

This study reports one case of Kabuki syndrome (KS) accompanied by a new mutation in the KMT2D gene and concurrent congenital pulmonary airway malformation (CPAM). After birth, the child demonstrated delayed physical growth and neurological, psychological, and intellectual development. Notable facial features included arched eyebrows and elongated eyelids toward the lower side. Chest computed tomography revealed pulmonary airway malformation. Whole-exome high-throughput sequencing revealed a novel heterozygous missense mutation in the KMT2D gene, Exon38c.10481C>T (p.Pro3494Leu), a possible pathogenic mutation that has not been previously reported. Both parents were wild-type. The clinical manifestations of KS are complex; however, cases with concomitant CPAM have rarely been reported. This study elucidates the mutation spectrum of the KS gene and the clinical characteristics of KS patients.

Fields of Study & Subjects

Genomics and Rare Diseases Genomic variations and chromosomal abnormalities Congenital Diaphragmatic Hernia Studies

Publication details

Journal / source
Journal of Investigative Medicine
Volume
74
Issue
7
Pages
721–726
Publication date
17 February 2026
Language
EN