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Submitted on May 31, 2005
Accepted on November 16, 2005
Emma Children's Hospital AMC, University of Amsterdam, Department of Pediatric Endocrinology, Department of Pediatric Psychology, Amsterdam, The Netherlands, Radboud University Nijmegen Medical Center, Department of Pediatric Physical Therapy, Nijmegen, The Netherlands, Alberta Children's Hospital, Department of Paediatrics, Behavioural Research Unit, Calgary, Canada
* To whom correspondence should be addressed. E-mail: m.j.kempers{at}amc.uva.nl.
Context: Long-term follow-up data on cognitive and motor functioning in adult patients with congenital hypothyroidism, diagnosed by neonatal screening, are scarce. Hence, it is still unclear whether the frequently reported cognitive and motor deficits observed during childhood persist in adulthood.
Objective: To study cognitive and motor functioning in young adults with congenital hypothyroidism, born in the first two years after the introduction of the Dutch neonatal screeningprogram.
Design/setting/patients: Seventy patients were tested (mean age 21.5 yr); 49 of them were previously tested at 9.5 yr. Median age at start of treatment was 28 days (range 4-293). Congenital hypothyroidism was classified as severe, moderate or mild, according to pre-treatment T4-concentration.
Main outcome measurement: The influence of severity of congenital hypothyroidism and age at which T4-supplementation was started on cognitive and motor outcome.
Results: Patients, particularly those with severe congenital hypothyroidism, had significantly higher (i.e. worse) motor scores (Total score 7.8, Ball Skills 2.0, Balance 4.1) compared with controls (3.2, 0.7, 1.1), and lower Full Scale (95.8), Verbal (96.4), and Performance (95.6) IQ scores than the normal population. No significant change in IQ from childhood to adulthood was found and for the majority of patients motor score classification remained the same. Severity of congenital hypothyroidism but not starting day of treatment was correlated with IQ and motor scores.
Conclusions: It is concluded that severity of congenital hypothyroidism but not timing of treatment initiation is an important factor determining long term cognitive and motor outcome. Clearly, detrimental effects on developmental outcome in patients with congenital hypothyroidism persist over time.
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