Cited by 122Thyroid Disorders and TreatmentsDown syndrome and intellectual disability researchThyroid and Parathyroid Surgery

Intellectual and motor development of young adults with congenital hypothyroidism diagnosed by neonatal screening

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Kempers, M. J. E., van der Sluijs Veer, L., Nijhuis-van der Sanden, M. W. G., Kooistra, L., Wiedijk, B. M., Faber, I., Last, B. F., de Vijlder, J. J. M. +2 more·2006·The Journal of Clinical Endocrinology & Metabolism

Abstract

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: The objective of this study was to examine cognitive and motor functioning in young adults with congenital hypothyroidism, born in the first 2 yr after the introduction of the Dutch neonatal screening program. DESIGN/SETTING/PATIENTS: Seventy patients were tested (mean age, 21.5 yr); 49 of them were previously tested at 9.5 yr. The median age at the start of treatment was 28 d (range, 4-293 d). Congenital hypothyroidism was classified as severe, moderate, or mild, according to pretreatment T(4) concentrations. MAIN OUTCOME MEASUREMENT: The main outcome measurement was the influence of the severity of congenital hypothyroidism and age at which T(4) 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 (total score, 3.2; ball skills, 0.7; balance, 1.1), and lower full-scale (95.8), verbal (96.4), and performance (95.6) intelligence quotient (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. The severity of congenital hypothyroidism, but not the starting day of treatment, was correlated with IQ and motor scores. CONCLUSIONS: It is concluded that the severity of congenital hypothyroidism, but not the 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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  1. [11]11. Vulsma T 1991 Etiology and pathogenesis of congenital hypothyroidism. Evaluation and examination of patients detected by neonatal screening in The Netherlands, academic thesis, University of Amsterdam
  2. [16]16. Haasen PP, de Bruyn EEJ, Pij YJ, Poortinga YH, Lutje Spelberg HC 1986 Wechsler intelligence scale for children-revised; Dutch edition, Lisse, The Netherlands. Lisse: Swets, Zeitlinger

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Authors
Kempers, M. J. E., van der Sluijs Veer, L., Nijhuis-van der Sanden, M. W. G., Kooistra, L., Wiedijk, B. M., Faber, I., Last, B. F., de Vijlder, J. J. M., Grootenhuis, M. A., & Vulsma, T.
Affiliations
  • Amsterdam UMC Location University of Amsterdam (NL)
  • Alberta Children's Hospital (CA)
  • Radboud University Nijmegen (NL)
Venue
The Journal of Clinical Endocrinology & Metabolism
Published
2006-02
Volume
91
Issue
2
Pages
418-424
ISSN
  • 0021-972X (ISSN-L)
  • 1945-7197
OpenAlex
W2120705054
Cited by
122
References
28
Citations by year
2026: 2 · 2025: 6 · 2024: 4 · 2023: 1 · 2022: 5 · 2021: 5 · 2020: 6 · 2019: 3 · 2018: 6 · 2017: 4 · 2016: 7 · 2015: 4 · 2014: 12 · 2013: 11 · 2012: 9
FWCI
5.91
Type
journal article
Language
en
Open access
Licence
other-oa
Locations
  • The Journal of Clinical Endocrinology & Metabolism (publishedVersion)
  • PubMed (publishedVersion)
  • Pure Amsterdam UMC (publishedVersion)
  • VU Research Portal (submittedVersion, other-oa)
  • Data Archiving and Networked Services (DANS) (submittedVersion)
  • Digital Academic REpository of VU University Amsterdam (Vrije Universiteit Amsterdam) (submittedVersion)
  • Radboud Repository (Radboud University) (submittedVersion)
  • Radboud Repository (Radboud University) (submittedVersion, other-oa)
  • Data Archiving and Networked Services (DANS) (submittedVersion, other-oa)
Topics
  • Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Thyroid Disorders and Treatments
  • Health Sciences › Medicine › Public Health, Environmental and Occupational Health › Down syndrome and intellectual disability research
  • Health Sciences › Medicine › Surgery › Thyroid and Parathyroid Surgery
Keywords
congenital hypothyroidism, newborn screening, young adults, cognitive function, motor function, IQ, motor skills, balance, long-term outcomes, developmental outcomes
MeSH terms
Adult, Congenital Hypothyroidism, Female, Follow-Up Studies, Humans, Intelligence, Longitudinal Studies, Male, Motor Skills, Thyroxine, Statistics, Nonparametric, Hormone Replacement Therapy

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