Neurological Presentations in Children with Inborn Errors of Immunity: Patient-Level Etiological Classification in a Retrospective Cohort

Background/Objectives: Neurological findings in children with inborn errors of immunity (IEIs) may reflect shared syndromic phenotypes, central nervous system (CNS) complications, or unrelated conditions. We assessed their likely etiology through patient-level review. Methods: We screened 1036 children evaluated for suspected IEI between October 2023 and May 2026. This single-center retrospective cohort included 55 children aged 0–18 years with an established or probable IEI and a neurological concern. Consensus review assigned each child to one of five etiological categories: likely IEI-related; independent coexisting diagnosis; acquired, provoked, or coincidental process; no significant neurological involvement; and undetermined. Exploratory Firth regression examined associations of prematurity and consanguinity with likely IEI-related classification. Results: Among children with ascertainable data, persistent neurodevelopmental delay occurred in 29/50 (58.0%), epilepsy in 14/53 (26.4%), and brain magnetic resonance imaging abnormalities in 28/40 (70.0%). Neurological findings preceded IEI recognition in 27/53 (50.9%); 24/54 (44.4%) had a neurological first presentation. Sixteen presentations (29.1%) were classified as likely IEI-related: 13 within monogenic, syndromic, or genetically mediated phenotypes and three as CNS complications. Other categories comprised independent diagnoses (n = 10), acquired, provoked, or coincidental processes (n = 9), no significant neurological involvement (n = 6), and undetermined etiology (n = 14). Adjusted odds ratios were 3.30 (95% CI, 0.97–12.09) for prematurity and 3.06 (95% CI, 0.87–11.49) for consanguinity, with wide confidence intervals. Conclusions: Neurological presentations in children with IEI warrant careful etiological interpretation. Multidisciplinary assessment may help distinguish IEI-associated manifestations from coexisting conditions. Further studies could inform diagnostic evaluation and clinical follow-up.

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Journal
Children
Published
2026-10-28
DOI
https://doi.org/10.3390/children13111492
Primary Topic
Immunodeficiency and Autoimmune Disorders
Type
article
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article

Neurological Presentations in Children with Inborn Errors of Immunity: Patient-Level Etiological Classification in a Retrospective Cohort

Pınar Gençpınar, Sanem Eren Akarcan, Nihal Olgaç Dündar, Seda Tunca et al.
2 links
Children
Immunodeficiency and Autoimmune Disorders
article

Neurological Presentations in Children with Inborn Errors of Immunity: Patient-Level Etiological Classification in a Retrospective Cohort

Pınar Gençpınar, Sanem Eren Akarcan, Nihal Olgaç Dündar, Seda Tunca, İdil Akay Hacı, Tuba Tunçel, Özlem Özsoy, Elif Didinmez Taşkırdı, Müge Baykan, İlke Taşkırdı, İpek Burcu Parlak İbiş, Ayşe Aygün, Özlem Yılmaz
article en

Abstract

Background/Objectives: Neurological findings in children with inborn errors of immunity (IEIs) may reflect shared syndromic phenotypes, central nervous system (CNS) complications, or unrelated conditions. We assessed their likely etiology through patient-level review. Methods: We screened 1036 children evaluated for suspected IEI between October 2023 and May 2026. This single-center retrospective cohort included 55 children aged 0–18 years with an established or probable IEI and a neurological concern. Consensus review assigned each child to one of five etiological categories: likely IEI-related; independent coexisting diagnosis; acquired, provoked, or coincidental process; no significant neurological involvement; and undetermined. Exploratory Firth regression examined associations of prematurity and consanguinity with likely IEI-related classification. Results: Among children with ascertainable data, persistent neurodevelopmental delay occurred in 29/50 (58.0%), epilepsy in 14/53 (26.4%), and brain magnetic resonance imaging abnormalities in 28/40 (70.0%). Neurological findings preceded IEI recognition in 27/53 (50.9%); 24/54 (44.4%) had a neurological first presentation. Sixteen presentations (29.1%) were classified as likely IEI-related: 13 within monogenic, syndromic, or genetically mediated phenotypes and three as CNS complications. Other categories comprised independent diagnoses (n = 10), acquired, provoked, or coincidental processes (n = 9), no significant neurological involvement (n = 6), and undetermined etiology (n = 14). Adjusted odds ratios were 3.30 (95% CI, 0.97–12.09) for prematurity and 3.06 (95% CI, 0.87–11.49) for consanguinity, with wide confidence intervals. Conclusions: Neurological presentations in children with IEI warrant careful etiological interpretation. Multidisciplinary assessment may help distinguish IEI-associated manifestations from coexisting conditions. Further studies could inform diagnostic evaluation and clinical follow-up.

ChildrenVol. 13(11)
Izmir University (TR), Recep Tayyip Erdoğan University (TR), Izmir Kâtip Çelebi University (TR), İzmir Şehir Hastanesi (TR)
Good health and well-being
Openalex Percentile: Top 19%
Immunodeficiency and Autoimmune Disorders
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