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.
Authors
- E. G. Kondratyeva
- Andrey V. Marakhonov (ORCID: https://orcid.org/0000-0002-0972-5118)
- Kristina Mikhalchuk (ORCID: https://orcid.org/0000-0002-0394-7801)
- O.V. Karpova (ORCID: https://orcid.org/0000-0002-9466-219X)
- Ekaterina Yu. Selina (ORCID: https://orcid.org/0009-0003-6545-1435)
- N. Kashirskaya (ORCID: https://orcid.org/0000-0003-0503-6371)
- Anastasia M. Bobreshova (ORCID: https://orcid.org/0009-0001-3476-053X)
- Yuliya L. Melyanovskaya (ORCID: https://orcid.org/0000-0002-8814-5532)
- T. N. Vasilyeva
- Rena Zinchenko
- D. E. Pershin
- A. R. Shakirova
- Anna Mukhina
- Farida Sibgatullina
- Zulfia Vafina
- Anna Shcherbina
- Dmitriy Kazakov
Institutions
- 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)
Publication Details
- 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
Funders
- Johns Hopkins University