A Diagnostic Dilemma: Concurrent Diagnosis of Cystic Fibrosis and Definitive Kabuki Syndrome Type 1

The article presents a clinical case involving a patient with presumptive coexistence of two hereditary disorders, confirmed by molecular genetic analyses. Clinical evaluation of the proband, a 9-year-old girl, revealed features characteristic of Kabuki syndrome, including a typical "Kabuki makeup" facial phenotype, short stature, intracranial hypertension, and diffuse muscular hypotonia. Additional clinical findings included chronic right-sided otitis media, conjunctivitis, recurrent pneumonia, bilateral conductive hearing loss, astigmatism, and primary adenitis. Clinical assessment and molecular genetic testing were performed. High-throughput sequencing identified a previously reported pathogenic heterozygous variant in the <i>KMT2D</i> gene, NM_003482.4:c.15142C>T p.Arg5048Cys, and two known heterozygous variants in the <i>CFTR</i> gene: NM_000492.4:c.1521_1523delCTT p.Phe508del and c.3454G>C p.Asp1152His, classified as pathogenic and of variable clinical significance, respectively. Segregation analysis demonstrated that the <i>KMT2D</i> variant most likely arose in the proband de novo, whereas the <i>CFTR</i> variants were inherited from each of the parents. Notably, the proband's clinically unaffected elder sister carried the same <i>CFTR</i> genotype. Based on the clinical presentation and molecular genetic findings, the diagnosis of Kabuki syndrome type 1 was conclusively established in the patient. Functional assessment of CFTR demonstrated its preserved function, which did not support a diagnosis of CF or CFTR-related disorders.

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Journal
International Journal of Molecular Sciences
Published
2026-03-09
DOI
https://doi.org/10.3390/ijms27052510
Primary Topic
Genomics and Rare Diseases
Type
article
Field-Weighted Citation Impact
0.00

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article

A Diagnostic Dilemma: Concurrent Diagnosis of Cystic Fibrosis and Definitive Kabuki Syndrome Type 1

E. G. Kondratyeva, Andrey V. Marakhonov, Kristina Mikhalchuk, O.V. Karpova et al.
International Journal of Molecular Sciences
Genomics and Rare Diseases
article

A Diagnostic Dilemma: Concurrent Diagnosis of Cystic Fibrosis and Definitive Kabuki Syndrome Type 1

E. G. Kondratyeva, Andrey V. Marakhonov, Kristina Mikhalchuk, O.V. Karpova, Ekaterina Yu. Selina, N. Kashirskaya, Anastasia M. Bobreshova, Yuliya L. Melyanovskaya, T. N. Vasilyeva, Rena Zinchenko, D. E. Pershin, A. R. Shakirova, Anna Mukhina, Farida Sibgatullina, Zulfia Vafina, Anna Shcherbina, Dmitriy Kazakov
article en

Abstract

The article presents a clinical case involving a patient with presumptive coexistence of two hereditary disorders, confirmed by molecular genetic analyses. Clinical evaluation of the proband, a 9-year-old girl, revealed features characteristic of Kabuki syndrome, including a typical "Kabuki makeup" facial phenotype, short stature, intracranial hypertension, and diffuse muscular hypotonia. Additional clinical findings included chronic right-sided otitis media, conjunctivitis, recurrent pneumonia, bilateral conductive hearing loss, astigmatism, and primary adenitis. Clinical assessment and molecular genetic testing were performed. High-throughput sequencing identified a previously reported pathogenic heterozygous variant in the <i>KMT2D</i> gene, NM_003482.4:c.15142C>T p.Arg5048Cys, and two known heterozygous variants in the <i>CFTR</i> gene: NM_000492.4:c.1521_1523delCTT p.Phe508del and c.3454G>C p.Asp1152His, classified as pathogenic and of variable clinical significance, respectively. Segregation analysis demonstrated that the <i>KMT2D</i> variant most likely arose in the proband de novo, whereas the <i>CFTR</i> variants were inherited from each of the parents. Notably, the proband's clinically unaffected elder sister carried the same <i>CFTR</i> genotype. Based on the clinical presentation and molecular genetic findings, the diagnosis of Kabuki syndrome type 1 was conclusively established in the patient. Functional assessment of CFTR demonstrated its preserved function, which did not support a diagnosis of CF or CFTR-related disorders.

International Journal of Molecular SciencesVol. 27(5)
Pirogov Russian National Research Medical University (RU), Republican Clinical Hospital of the Ministry of Health of the Republic of Tatarstan (RU), Research Centre for Medical Genetics (RU), Dmitry Rogachev National Research Center of Pediatric Hematology, Oncology and Immunology (RU)
Johns Hopkins University
Good health and well-being
Openalex Percentile: Top 29%
Genomics and Rare Diseases
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