ArticleJournal of clinical medicine2023
The Evolutionary Scenario of Pediatric Unclassified Primary Antibody Deficiency to Adulthood.
Article in Journal of clinical medicine, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.
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Who cites it
5 citing papers in PubMed, 5 citations in OpenAlex.
- Behind the scenes: Primary immunodeficiencies in pediatric eosinophilic gastrointestinal diseases.Pediatric allergy and immunology : official publication of the European Society of Pediatric Allergy and Immunology · 2026Article
- Unclassified Inborn Errors of Immunity Patients without any Pathogenic Variant in Targeted Next-Generation Sequencing: Long-Term Follow-up and Whole Exome Sequencing Results.Journal of clinical immunology · 2026Article
- Selective IgM deficiency: evaluation of 75 patients according to different diagnostic criteria.Immunologic research · 2024Observational
- IPINeT Ped-unPAD Study: Goals, Design, and Preliminary Results.Journal of clinical medicine · 2024Article
- Primary Immunodeficiencies: Pathogenetic Advances, Diagnostic and Management Challenges.Journal of clinical medicine · 2023Article
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Authors and funding
10 authors at 4 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundUnclassified primary antibody deficiency (unPAD) is a relatively novel inborn error of immunity (IEI) condition that can vary with time to more defined entities. Since long-term follow-up (FU) studies are scarce, we aimed to provide insight into the evolutionary clinical and immunological scenario of unPAD children to adulthood and identification of biomarkers of primary immune deficiency (PID) persistence.
methodsA total of 23 pediatric unPAD patients underwent clinical and immunological FU for a mean time of 14 years (range 3-32 years, median 16 years).
resultsUnPAD diagnosis may change over time. At the last FU, 10/23 (44%) children matched the diagnosis of transient hypogammaglobulinemia of infancy and 13/23 (56%) suffered from a persistent PID. In detail, an unPAD condition was confirmed in 7/23 (30%) patients, whereas 3/23 (13%), 2/23 (9%), and 1/23 (4%) were reclassified as common variable immunodeficiency, selective IgA deficiency, and isolated IgM deficiency, respectively. Low IgA, low specific antibody response to pneumococcus, and lower respiratory tract infections at diagnosis were independently associated with IEI persistence.
conclusionsLong-term monitoring of unPAD patients is required to define their outcome and possible evolution towards a definitive IEI diagnosis.
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