When Recurrent Infections Don’t Always Reveal a Primary Immunodeficiency

Background Recurrent infections often suggest an underlying immunodeficiency, particularly when local (e.g., an overlooked intrabronchial foreign body) or systemic (e.g., retroviral infection) causes are excluded. Primary immunodeficiencies (PIDs) are a heterogeneous group of inherited disorders characterized by immune system defects, leading to increased susceptibility to infections, autoimmunity, and malignancies. Recurrent respiratory infections are a key clinical feature of PIDs and often prompt extensive immunological evaluations. However, even when immune workups and cystic fibrosis testing are inconclusive, conditions that mimic PID must be considered to prevent misdiagnosis and treatment delays. Objectives This study aims to discuss cases of patients with recurrent infections initially suspected of having a PID and to evaluate the role of genetic testing in establishing a definitive diagnosis. Methods Patients with unexplained recurrent respiratory infections underwent a systematic evaluation, including immunoglobulin level measurements (IgG, IgA, IgM, and IgE), lymphocyte subset analysis, a sweat test for cystic fibrosis, HIV screening, and whole-exome sequencing (WES) to detect inborn errors of immunity and ciliopathy-related mutations. Results Seven patients (five males and two females) with a median age of 12.7 years were evaluated. Immunological testing was normal in five patients, while two had abnormal immune profiles: one female was diagnosed with combined immunodeficiency and one male with Wiskott-Aldrich syndrome. Notably, WES identified, in all seven patients, a homozygous pathogenic mutation in primary ciliary dyskinesia (PCD)-related genes, a disorder caused by defective ciliary function. Conclusion This study underscores the importance of considering PCD in the differential diagnosis of patients with recurrent respiratory infections suspected of PID, particularly when immunological tests yield inconclusive results. Additionally, PCD may coexist with other immunodeficiencies, complicating the diagnostic process. In populations with high consanguinity, incorporating genetic testing into routine diagnostic workflows is critical for accurate diagnosis, ultimately improving patient outcomes and preventing complications related to delayed treatment.

Authors

Institutions

Publication Details

Journal
Journal of Human Immunity
Published
2025-08-22
DOI
https://doi.org/10.70962/asid2025abstract.16
Primary Topic
Immunodeficiency and Autoimmune Disorders
Type
article
Field-Weighted Citation Impact
0.00
Controls
|||
ALL TIME
JAN
FEB
MAR
APR
MAY
JUN
JUL
AUG
SEP
OCT
article

When Recurrent Infections Don’t Always Reveal a Primary Immunodeficiency

Fatima Ailal, Ahmed Aziz Bousfiha, Ibtihal Benhsaien, Naima Amenzoui et al.
Journal of Human Immunity
Immunodeficiency and Autoimmune Disorders
article

When Recurrent Infections Don’t Always Reveal a Primary Immunodeficiency

Fatima Ailal, Ahmed Aziz Bousfiha, Ibtihal Benhsaien, Naima Amenzoui, Zahra Aadam, F. Adnane, Ikbal Belkhou, I. Mansouri, Doha Benchaji, Siham Satlane
article en

Abstract

Background Recurrent infections often suggest an underlying immunodeficiency, particularly when local (e.g., an overlooked intrabronchial foreign body) or systemic (e.g., retroviral infection) causes are excluded. Primary immunodeficiencies (PIDs) are a heterogeneous group of inherited disorders characterized by immune system defects, leading to increased susceptibility to infections, autoimmunity, and malignancies. Recurrent respiratory infections are a key clinical feature of PIDs and often prompt extensive immunological evaluations. However, even when immune workups and cystic fibrosis testing are inconclusive, conditions that mimic PID must be considered to prevent misdiagnosis and treatment delays. Objectives This study aims to discuss cases of patients with recurrent infections initially suspected of having a PID and to evaluate the role of genetic testing in establishing a definitive diagnosis. Methods Patients with unexplained recurrent respiratory infections underwent a systematic evaluation, including immunoglobulin level measurements (IgG, IgA, IgM, and IgE), lymphocyte subset analysis, a sweat test for cystic fibrosis, HIV screening, and whole-exome sequencing (WES) to detect inborn errors of immunity and ciliopathy-related mutations. Results Seven patients (five males and two females) with a median age of 12.7 years were evaluated. Immunological testing was normal in five patients, while two had abnormal immune profiles: one female was diagnosed with combined immunodeficiency and one male with Wiskott-Aldrich syndrome. Notably, WES identified, in all seven patients, a homozygous pathogenic mutation in primary ciliary dyskinesia (PCD)-related genes, a disorder caused by defective ciliary function. Conclusion This study underscores the importance of considering PCD in the differential diagnosis of patients with recurrent respiratory infections suspected of PID, particularly when immunological tests yield inconclusive results. Additionally, PCD may coexist with other immunodeficiencies, complicating the diagnostic process. In populations with high consanguinity, incorporating genetic testing into routine diagnostic workflows is critical for accurate diagnosis, ultimately improving patient outcomes and preventing complications related to delayed treatment.

Journal of Human ImmunityVol. 1(ASID2025)
Centre Hospitalier Universitaire Ibn Rochd (MA), University of Hassan II Casablanca (MA)
Openalex Percentile: Top 77%
Immunodeficiency and Autoimmune Disorders
AI Navigator

Ask Laika to Summarize, Analyze, and Connect papers live on the map.

Summarize Papers & Methodologies

Extract key findings, datasets, and comparative methods across publications.

Benchmark Rankings & Visual Analytics

Rank top research institutions, authors, funders, topics, and journals by Field-Weighted Citation Impact (FWCI) and paper volume with instant charts.

Connect Distant Disciplines

Bridge topological clusters on the map to find hidden collaborative intersections.