Immunoglobulin g subclass and specific polysaccharide antibody deficiency : a clinical and immunological profile in a tertiary cohort
Abstract
Background and Aims: Immunoglobulin(Ig)G-subclass deficiency (IgGSD) and specific polysaccharide antibody deficiency (SPAD) are two common antibody deficiencies, mainly associated with recurrent respiratory tract infections (RTI). The clinical variability and effect of therapy in a large cohort is not well studied. Methods: A retrospective observational study of children and adults with IgG2SD and/or IgG3SD and/or SPAD and normal total IgG. Results: In total, 105 patients (age 2y-79y (mean 24y), 53% pediatric, 44% male) were included. To our knowledge, this is the largest cohort of IgGSD and/or SPAD patients studied. Isolated IgG3SD was both in children (16/56;29%) and adults (30/49;61%) most prevalent. Of all IgG2SD(+/-SPAD) cases, 85% were found in children (22/26). In total, isolated SPAD was less frequent (16/105;15%) than IgGSD+SPAD (21/105;20%). Recurrent upper and lower RTI (76-82% and 38-57% respectively) were most frequently observed, similar in all groups. Gastrointestinal infections (42%;P=0.017) and fatigue (45%;P=0.005) were associated with IgG3SD. Bacterial skin infections (38%;P=0.024) were frequently observed in SPAD. Autoimmunity (25%;P=0.001), lymphadenopathy (71%;P=0.002) and fatigue (59%;P<0.001) were significantly common in adults compared to children. Autoimmunity was not associated with aberrant B-cell maturation, only present in 2.8%. IgG2SD patients (69%;P=0.046) and children (73%;P<0.001) were hospitalized more. Monocytopenia was repeatedly observed…
How strong is the evidence?
Evidence Hub reads this as a cohort study, done in people. Its evidence level is 3 of 5 (Moderate · trial / cohort / secondary analysis). Trials without a large randomized design, cohorts and secondary analyses give moderate evidence. Reported size: 105 participants or records.
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