title: Impaired neuromotor outcome in school-age children with congenital hypothyroidism receiving early high-dose substitution treatment.
abstract: Congenital hypothyroidism (CH) can lead to intellectual deficits despite early high-dose treatment. Our study aimed to determine whether motor impairments can occur despite early high-dose treatment. Sixty-three children with CH and early (median age of onset of treatment 9 d), high-dose treatment (median starting dose of levothyroxine 14.7 μg/kg/d) were tested with the Zurich Neuromotor Assessment (ZNA) at a median age of 13.8 y (range 7.0-14.2 y). Median z-scores in the children with CH were -0.95 in the pure and -0.56 in the adaptive fine motor component, significantly lower than in the ZNA test norms (p < 0.001 and p = 0.01, respectively). The 26 children with athyreosis were more affected than the 33 children with dysgenesis, particularly in the pure motor (-1.55 versus -0.76, p = 0.03), adaptive fine motor (-1.31 versus 0.13, p < 0.01), and static balance task (-0.47 versus 0.67, p = 0.01). Boys performed worse than girls. Older age at onset of treatment was related to poorer adaptive fine motor performance. Movement quality (assessed by associated movements) was not affected. We conclude that severe CH can cause neuromotor deficits persisting into adolescence. These deficits cannot completely be reversed by postnatal treatment, but earlier age at treatment may reduce the degree of impairment.
authors.affiliation: Division of Endocrinology and Diabetology, University Children's Hospital Zurich, Zurich 8032, Switzerland.
authors.forename: Annik
authors.initials: A
authors.lastname: Hauri-Hohl
authors.forename: Nicole
authors.initials: N
authors.lastname: Dusoczky
authors.forename: Anastasia
authors.initials: A
authors.lastname: Dimitropoulos
authors.forename: Russia Ha-Vinh
authors.initials: RH
authors.lastname: Leuchter
authors.forename: Luciano
authors.initials: L
authors.lastname: Molinari
authors.forename: Jon
authors.initials: J
authors.lastname: Caflisch
authors.forename: Oskar G
authors.initials: OG
authors.lastname: Jenni
authors.forename: Beatrice
authors.initials: B
authors.lastname: Latal
mesh_terms: D000293:Adolescent; D000704:Analysis of Variance; D002648:Child; D003409:Congenital Hypothyroidism / Q000188:drug therapy* / Q000503:physiopathology*; D005260:Female; D006801:Humans; D007231:Infant, Newborn; D008297:Male; D009048:Motor Skills / Q000187:drug effects / Q000502:physiology*; D015997:Neonatal Screening; D012737:Sex Factors; D013557:Switzerland; D013974:Thyroxine / Q000008:administration & dosage / Q000494:pharmacology* / Q000627:therapeutic use; D016896:Treatment Outcome
chemical_list: D013974:Thyroxine