Cognitive outcome in congenital central hypothyroidism: a systematic review with meta-analysis of individual patient data
Open accessAbstract
Objective: To provide an overview of cognitive and motor outcome, and quality of life (QoL) in patients with congenital central hypothyroidism (CH-C). Design: Systematic review with individual patient data (IPD) meta-analysis. Methods: OVID MEDLINE, EMBASE and PsycInfo were searched from inception to June 11th, 2019. Studies in patients with CH-C, either isolated or with multiple pituitary hormone deficiency (MPHD), were included if CH-C patients could be separated from any additional patient groups. Primary outcomes were full-scale intelligence quotient (FSIQ) and motor outcome; secondary outcome was QoL. Following data-extraction, one-stage IPD meta-analysis was performed, fitting a linear mixed model with FSIQ as dependent variable. Random intercepts were fitted for each study. Results: Six studies measuring FSIQ were eligible for meta-analysis, comprising 30 CH-C patients (20 males; 27 MPHD patients). FSIQ range was wide (64–123). Mean weighted FSIQ was 97 (95% CI: 88–105). Twenty-seven percent had an FSIQ below 85 (≥1 s.d. below norm score), and 10% below 70 (≥2 s.d. below norm score). There was no significant association between FSIQ and sex or age. Age at treatment initiation was available from three studies only, thus impeding a reliable analysis of this parameter. Motor outcome and QoL were each studied in one study; no quantitative analyses could be performed for these outcomes. Conclusion: A wide range in FSIQ scores was observed in CH-C patients. Results should be interpreted with caution, because included patients mainly had MPHD and age at treatment initiation was unknown for the majority of patients.
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In those studies, full-scale intelligence quotients were shifted towards lower values, with 10% of patients having an FSIQ below 70.
“FSIQs were shifted towards lower values with 10% of patients having an FSIQ <70 (below –2 SD in the general population)”
From Cognitive and Motor Outcome in Patients with Early-Detected Central Congenital Hypothyroidism Compared with Siblings (2021) · cites this paper as [14]
The systematic review identified 6 studies measuring full-scale intelligence quotient, including 30 patients in total (27/30 with multiple pituitary hormone deficiencies).
“We identified 6 studies that measured full-scale intelligence quotient (FSIQ), including 30 patients in total (27/30 MPHD).”
From Cognitive and Motor Outcome in Patients with Early-Detected Central Congenital Hypothyroidism Compared with Siblings (2021) · cites this paper as [14]
So far, FSIQ has only been reported in a few patients with isolated central congenital hypothyroidism; in the authors' previous systematic review, patients with isolated central congenital hypothyroidism were underrepresented.
“So far, FSIQ has only been reported in a few patients with isolated central CH; in our previous systematic review patients with isolated central CH were underrepresented, highlighting the need for further studies on cognitive outcome in this condition (14, 28).”
From Cognitive and Motor Outcome in Patients with Early-Detected Central Congenital Hypothyroidism Compared with Siblings (2021) · cites this paper as [14]
The included studies were small, heterogeneous, and possibly suffered from selection bias, so results should be interpreted with caution.
“but the included studies were small, heterogeneous, and possibly suffered from selection bias, so that results should be interpreted with caution (14).”
From Cognitive and Motor Outcome in Patients with Early-Detected Central Congenital Hypothyroidism Compared with Siblings (2021) · cites this paper as [14]
The indexed Abstract passage is a publication record, not a discussion of the included studies. Searches for small studies, heterogeneity, selection bias, and limitations found no passage addressing those points, so the claimed caution cannot be verified from our copy. (LLM, conf=0.72)
Experimental — accuracy not yet measuredIn a recent systematic review, sufficiently large studies on cognitive outcome in patients with central congenital hypothyroidism are lacking.
“In a recent systematic review, we showed that sufficiently large studies on cognitive outcome in patients with central CH are lacking (14).”
From Cognitive and Motor Outcome in Patients with Early-Detected Central Congenital Hypothyroidism Compared with Siblings (2021) · cites this paper as [14]
The passages available from our copy identify the paper as a systematic review of cognitive outcome in congenital central hypothyroidism, but contain no findings about the size of existing studies. Searches for the claimed lack of sufficiently large studies and for small sample sizes found no passage addressing that conclusion. (LLM, conf=0.70)
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- Authors
- Naafs, J. C., Vendrig, L. M., Limpens, J., van der Lee, H. J., Duijnhoven, R. G., Marchal, J. P., Trotsenburg, A. S. V., & Zwaveling-Soonawala, N.
- Affiliations
- Emma Kinderziekenhuis (NL)
- Amsterdam University Medical Centers (NL)
- 3Pediatric Clinical Research Office, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands
- 1Department of Pediatric Endocrinology, Emma Children’s Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands
- Venue
- European Journal of Endocrinology
- Publisher
- Oxford University Press (OUP)
- Published
- 2020-03
- Volume
- 182
- Issue
- 3
- Pages
- 351-361
- ISSN
- 0804-4643 (ISSN-L)
- 1479-683X
- OpenAlex
- W3000943509
- Cited by
- 24
- References
- 0
- Citations by year
- 2025: 1 · 2024: 5 · 2023: 7 · 2022: 6 · 2021: 4 · 2020: 1
- FWCI
- 0.69
- Type
- journal article
- Language
- en
- Open access
- green
- pure.amsterdamumc.nl
- Licence
- other-oa
- Locations
- European Journal of Endocrinology (publishedVersion)
- PubMed (publishedVersion)
- Data Archiving and Networked Services (DANS) (submittedVersion)
- Pure Amsterdam UMC (submittedVersion, other-oa)
- Topics
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Thyroid Disorders and Treatments
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Pituitary Gland Disorders and Treatments
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Growth Hormone and Insulin-like Growth Factors
- Keywords
- multiple pituitary hormone deficiency, intelligence quotient, cognitive outcomes, motor outcome, quality of life, age at treatment initiation, intellectual disability
- MeSH terms
- Adolescent, Child, Child, Preschool, Congenital Hypothyroidism, Female, Humans, Hypopituitarism, Intelligence Tests, Male, Quality of Life, Young Adult
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