Genotype and Ultrastructure Correlation in Primary Ciliary Dyskinesia Among Saudi Children: A Case Series

Background: Primary ciliary dyskinesia (PCD) is a rare inherited disorder of motile cilia causing chronic oto-sinopulmonary disease. Transmission electron microscopy (TEM) remains essential for confirming ultrastructural defects, especially in resource-limited settings. This study characterizes the clinicopathologic and molecular spectrum of Saudi children with PCD. Methods: Ten Saudi children with confirmed PCD (2018–2025) were retrospectively reviewed. Clinical, radiological, ultrastructural, and whole exome sequencing (WES) data were analyzed to assess genotype–phenotype correlation. Results: The cohort included seven females and three males (median age 9 years). Consanguinity was present in 80%, and five had situs inversus. TEM showed class 1 defects in 3 patients, class 2 in 2, and abnormalities of uncertain significance in 3. WES identified variants in DNAAF3, DNAAF4, RSPH9, GAS2L2, and NME5. Conclusions: This study expands the molecular and ultrastructural profile of PCD in Saudi Arabia. TEM remains essential for diagnosis and variant interpretation.

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Journal
Fetal and Pediatric Pathology
Published
2026-07-30
DOI
https://doi.org/10.1080/15513815.2026.2699115
Primary Topic
Cystic Fibrosis Research Advances
Type
article
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article

Genotype and Ultrastructure Correlation in Primary Ciliary Dyskinesia Among Saudi Children: A Case Series

Wajd A. Althakfi
Fetal and Pediatric Pathology
Cystic Fibrosis Research Advances
article

Genotype and Ultrastructure Correlation in Primary Ciliary Dyskinesia Among Saudi Children: A Case Series

Wajd A. Althakfi
article en

Abstract

Background: Primary ciliary dyskinesia (PCD) is a rare inherited disorder of motile cilia causing chronic oto-sinopulmonary disease. Transmission electron microscopy (TEM) remains essential for confirming ultrastructural defects, especially in resource-limited settings. This study characterizes the clinicopathologic and molecular spectrum of Saudi children with PCD. Methods: Ten Saudi children with confirmed PCD (2018–2025) were retrospectively reviewed. Clinical, radiological, ultrastructural, and whole exome sequencing (WES) data were analyzed to assess genotype–phenotype correlation. Results: The cohort included seven females and three males (median age 9 years). Consanguinity was present in 80%, and five had situs inversus. TEM showed class 1 defects in 3 patients, class 2 in 2, and abnormalities of uncertain significance in 3. WES identified variants in DNAAF3, DNAAF4, RSPH9, GAS2L2, and NME5. Conclusions: This study expands the molecular and ultrastructural profile of PCD in Saudi Arabia. TEM remains essential for diagnosis and variant interpretation.

Fetal and Pediatric Pathology
King Saud Medical City (SA), King Saud University (SA)
Zero hunger
Openalex Percentile: Top 10%
Cystic Fibrosis Research Advances
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