title: Evaluation of autoimmune phenomena in patients with pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections (PANDAS).
abstract: The pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections (PANDAS) are basically characterized by obsessive-compulsive symptoms and/or tics triggered by group-A beta-hemolytic Streptococcus infections. Poor data are available about the clear definition of PANDAS's autoimmune origin. The aim of our study was to evaluate the prevalence of autoimmune phenomena, including thyroid function abnormalities, specific celiac disease antibodies, and positivity of organ- or nonorgan-specific autoantibodies in a large cohort of Caucasian children and adolescents with PANDAS. Seventy-seven consecutive patients (59 males, 18 females; mean age 6.3±2.5 years, range 2.0-14.5 years) strictly fulfilling the clinical criteria for PANDAS diagnosis were recruited. In all subjects we evaluated serum concentrations of free-T3, free-T4, thyrotropin, and the following auto-antibodies: anti-thyroperoxidase, anti-thyroglobulin, anti-thyrotropin receptor, anti-gliadin, anti-endomysium, anti-tissue transglutaminase, anti-nuclear, anti-smooth muscle, anti-extractable nuclear antigens, anti-phospholipid, plus lupus-like anticoagulant. The results were compared with those obtained from 197 age- and sex-matched healthy controls (130 males, 67 females; mean age 6.8±2.9 years, range 2.3-14.8 years). The frequencies of subclinical (3.8% vs 3.6%) and overt hypothyroidism (1.2% vs 0%), autoimmune thyroiditis (2.46% vs 1.14%), celiac disease (1.2% vs 0.05%), and positivity of organ- and nonorgan-specific autoantibodies (5.1% vs 4.8%) were not statistically significant between patients with PANDAS and controls. Evaluating the overall disease duration, we did not observe any significant difference between patients with (3.4±2.15 years) and without (3.4±2.89 years) autoimmune abnormalities. However, PANDAS patients with autoimmune diseases or positivity for any organ- and nonorgan-specific antibodies showed significantly higher anti-streptolysin O and anti-DNAse B titers, as well as a history of more frequent throat infections than controls (p<0.0001). Abnormalities of thyroid function and thyroid autoimmune diseases, as well as the association with celiac disease or organ- and nonorgan-specific autoimmunity seem not more frequent in children and adolescents with PANDAS than in healthy controls. A potential relationship between autoimmunity and PANDAS should be assessed further in larger studies. Children and adolescents with PANDAS should not be actually screened for thyroid function, celiac disease and/or autoimmune diseases.
authors.affiliation: Health Sciences Department, University of Florence, Anna Meyer Childrens University Hospital, Florence, Italy. Electronic address: stefano.stagi@yahoo.it.
authors.forename: Stefano
authors.initials: S
authors.lastname: Stagi
authors.affiliation: Institute of Pediatrics, Università Cattolica Sacro Cuore, Rome, Italy.
authors.forename: Donato
authors.initials: D
authors.lastname: Rigante
authors.affiliation: Department of Internal Medicine, Section of Rheumatology, Transition Clinic, University of Florence, Florence, Italy.
authors.forename: Gemma
authors.initials: G
authors.lastname: Lepri
authors.affiliation: Department of Internal Medicine, Section of Rheumatology, Transition Clinic, University of Florence, Florence, Italy.
authors.forename: Federico
authors.initials: F
authors.lastname: Bertini
authors.affiliation: Department of Internal Medicine, Section of Rheumatology, Transition Clinic, University of Florence, Florence, Italy.
authors.forename: Marco
authors.initials: M
authors.lastname: Matucci-Cerinic
authors.affiliation: Department of Internal Medicine, Section of Rheumatology, Transition Clinic, University of Florence, Florence, Italy.
authors.forename: Fernanda
authors.initials: F
authors.lastname: Falcini
mesh_terms: D001327:Autoimmune Diseases / Q000453:epidemiology / Q000276:immunology*; D015551:Autoimmunity / Q000276:immunology; D015331:Cohort Studies; D006801:Humans; D009771:Obsessive-Compulsive Disorder; D015995:Prevalence; D013290:Streptococcal Infections / Q000453:epidemiology / Q000276:immunology*
keywords: Autoimmunity; Child; Group-A beta-hemolytic streptococcal infection; PANDAS