ArticleBritish journal of haematology2026
Phenotype-specific immune profiles and outcomes in childhood autoimmune neutropenia: A 20-year cohort study.
Article in British journal of haematology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
Childhood autoimmune neutropenia (AIN) encompasses heterogeneous entities; phenotype-specific immunological profiles and their relationship to infection outcomes remain incompletely defined. To characterise clinical, immunological and long-term outcomes across distinct phenotypes of childhood AIN. We conducted a retrospective cohort study of 112 children with antibody-confirmed AIN at a tertiary centre over 20 years. Patients were classified into three phenotypes: primary autoimmune neutropenia (pAIN), long-lasting neutropenia (LL-Np) and late-onset neutropenia (LO-Np). Outcomes included lymphocyte subsets, immunoglobulin levels, infection-related hospitalisation rates and time to resolution. The cohort comprised 90 pAIN (80.4%), 10 LL-Np (8.9%) and 12 LO-Np (10.7%) patients. LO-Np was characterised by higher neutrophil counts, marked leucopenia, multi-lineage lymphopenia (CD3+, CD4+, CD19+), frequent antinuclear antibodies (ANA) positivity (50%) and associated autoimmune disease (25%). No infection-related hospitalisations occurred in LO-Np (0.00/100 person-years [PY]; 95% confidence interval [CI] 0.00-2.30) versus 1.38/100 PY in pAIN and 5.58/100 PY in LL-Np. Immunoglobulin levels were normal across all patients. Median time to resolution was 19, 67 and 59 months in pAIN, LL-Np and LO-Np respectively (both p < 0.001 vs. pAIN). Late-onset AIN represents a distinct phenotype with systemic immune dysregulation and multi-lineage lymphopenia, yet paradoxically absence of infection-related hospitalisations, reflecting preserved humoral immunity. Phenotype-based stratification may guide clinical monitoring and therapeutic decisions.
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