Intellectual and Motor Development of Young Adults with Congenital Hypothyroidism Diagnosed by Neonatal Screening
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....
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| Veröffentlicht in: | The journal of clinical endocrinology and metabolism 2006-02, Vol.91 (2), p.418-424 |
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| creator | 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. |
| description | 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 T4 concentrations.
Main Outcome Measurement: The main outcome measurement was the influence of the 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 (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. |
| doi_str_mv | 10.1210/jc.2005-1209 |
| format | Article |
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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 T4 concentrations.
Main Outcome Measurement: The main outcome measurement was the influence of the 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 (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.</description><identifier>ISSN: 0021-972X</identifier><identifier>EISSN: 1945-7197</identifier><identifier>DOI: 10.1210/jc.2005-1209</identifier><identifier>PMID: 16303842</identifier><identifier>CODEN: JCEMAZ</identifier><language>eng</language><publisher>Bethesda, MD: Endocrine Society</publisher><subject>Adult ; Biological and medical sciences ; Congenital Hypothyroidism - physiopathology ; Congenital Hypothyroidism - therapy ; Endocrinopathies ; Female ; Follow-Up Studies ; Fundamental and applied biological sciences. Psychology ; Hormone Replacement Therapy ; Humans ; Intelligence ; Longitudinal Studies ; Male ; Medical sciences ; Motor Skills - physiology ; Non tumoral diseases. Target tissue resistance. Benign neoplasms ; Statistics, Nonparametric ; Thyroid. Thyroid axis (diseases) ; Thyroxine - therapeutic use ; Vertebrates: endocrinology</subject><ispartof>The journal of clinical endocrinology and metabolism, 2006-02, Vol.91 (2), p.418-424</ispartof><rights>Copyright © 2006 by The Endocrine Society</rights><rights>2006 INIST-CNRS</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4773-a43f226a274c1b9ff062cc77baa8ab726e4bcb808615bedb981ca6e7d82bc0533</citedby></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,777,781,27905,27906</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=17489963$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/16303842$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Kempers, M. J. E.</creatorcontrib><creatorcontrib>van der Sluijs Veer, L.</creatorcontrib><creatorcontrib>Nijhuis-van der Sanden, M. W. G.</creatorcontrib><creatorcontrib>Kooistra, L.</creatorcontrib><creatorcontrib>Wiedijk, B. M.</creatorcontrib><creatorcontrib>Faber, I.</creatorcontrib><creatorcontrib>Last, B. F.</creatorcontrib><creatorcontrib>de Vijlder, J. J. M.</creatorcontrib><creatorcontrib>Grootenhuis, M. A.</creatorcontrib><creatorcontrib>Vulsma, T.</creatorcontrib><title>Intellectual and Motor Development of Young Adults with Congenital Hypothyroidism Diagnosed by Neonatal Screening</title><title>The journal of clinical endocrinology and metabolism</title><addtitle>J Clin Endocrinol Metab</addtitle><description>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 T4 concentrations.
Main Outcome Measurement: The main outcome measurement was the influence of the 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 (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.</description><subject>Adult</subject><subject>Biological and medical sciences</subject><subject>Congenital Hypothyroidism - physiopathology</subject><subject>Congenital Hypothyroidism - therapy</subject><subject>Endocrinopathies</subject><subject>Female</subject><subject>Follow-Up Studies</subject><subject>Fundamental and applied biological sciences. Psychology</subject><subject>Hormone Replacement Therapy</subject><subject>Humans</subject><subject>Intelligence</subject><subject>Longitudinal Studies</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Motor Skills - physiology</subject><subject>Non tumoral diseases. Target tissue resistance. Benign neoplasms</subject><subject>Statistics, Nonparametric</subject><subject>Thyroid. Thyroid axis (diseases)</subject><subject>Thyroxine - therapeutic use</subject><subject>Vertebrates: endocrinology</subject><issn>0021-972X</issn><issn>1945-7197</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2006</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqFkUFv1DAQhS0EotvCjTPyhZ6aYjuOnRyrLdBKBQ6ABCfLdia7WRI7tR1W--9xtCv1goSl8Vy-N_a8h9AbSq4po-T9zl4zQqqCMtI8Qyva8KqQtJHP0YoQRotGsp9n6DzGHSGU86p8ic6oKElZc7ZCj_cuwTCATbMesHYt_uyTD_gW_sDgpxFcwr7Dv_zsNvimnYcU8b5PW7z2bgOuT1l1d5h82h6C79s-jvi21xvnI7TYHPAX8E4v0DcbIPNu8wq96PQQ4fWpX6AfHz98X98VD18_3a9vHgrLpSwLzcuOMaGZ5JaapuuIYNZKabSutZFMADfW1KQWtDLQmqamVguQbc2MJVVZXqDL49wp-McZYlJjH23eVTvwc1RCirIUlfgvSCXJVkmSwasjaIOPMUCnptCPOhwUJWrJQu2sWrJQSxYZf3uaO5sR2if4ZH4G3p0AHa0euqCd7eMTJ3ndNGLZhB-5vR8ShPh7mPcQ1Bb0kLaK5MOFrIv8siD5IkUuusjKowxc623oHUwBYlQ7PweXnf_3r_8CfWWzxQ</recordid><startdate>200602</startdate><enddate>200602</enddate><creator>Kempers, M. J. E.</creator><creator>van der Sluijs Veer, L.</creator><creator>Nijhuis-van der Sanden, M. W. G.</creator><creator>Kooistra, L.</creator><creator>Wiedijk, B. M.</creator><creator>Faber, I.</creator><creator>Last, B. F.</creator><creator>de Vijlder, J. J. M.</creator><creator>Grootenhuis, M. A.</creator><creator>Vulsma, T.</creator><general>Endocrine Society</general><general>Copyright by The Endocrine Society</general><scope>IQODW</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7TK</scope><scope>7X8</scope></search><sort><creationdate>200602</creationdate><title>Intellectual and Motor Development of Young Adults with Congenital Hypothyroidism Diagnosed by Neonatal Screening</title><author>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.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c4773-a43f226a274c1b9ff062cc77baa8ab726e4bcb808615bedb981ca6e7d82bc0533</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2006</creationdate><topic>Adult</topic><topic>Biological and medical sciences</topic><topic>Congenital Hypothyroidism - physiopathology</topic><topic>Congenital Hypothyroidism - therapy</topic><topic>Endocrinopathies</topic><topic>Female</topic><topic>Follow-Up Studies</topic><topic>Fundamental and applied biological sciences. Psychology</topic><topic>Hormone Replacement Therapy</topic><topic>Humans</topic><topic>Intelligence</topic><topic>Longitudinal Studies</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Motor Skills - physiology</topic><topic>Non tumoral diseases. Target tissue resistance. Benign neoplasms</topic><topic>Statistics, Nonparametric</topic><topic>Thyroid. Thyroid axis (diseases)</topic><topic>Thyroxine - therapeutic use</topic><topic>Vertebrates: endocrinology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Kempers, M. J. E.</creatorcontrib><creatorcontrib>van der Sluijs Veer, L.</creatorcontrib><creatorcontrib>Nijhuis-van der Sanden, M. W. G.</creatorcontrib><creatorcontrib>Kooistra, L.</creatorcontrib><creatorcontrib>Wiedijk, B. M.</creatorcontrib><creatorcontrib>Faber, I.</creatorcontrib><creatorcontrib>Last, B. F.</creatorcontrib><creatorcontrib>de Vijlder, J. J. M.</creatorcontrib><creatorcontrib>Grootenhuis, M. A.</creatorcontrib><creatorcontrib>Vulsma, T.</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Neurosciences Abstracts</collection><collection>MEDLINE - Academic</collection><jtitle>The journal of clinical endocrinology and metabolism</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Kempers, M. J. E.</au><au>van der Sluijs Veer, L.</au><au>Nijhuis-van der Sanden, M. W. G.</au><au>Kooistra, L.</au><au>Wiedijk, B. M.</au><au>Faber, I.</au><au>Last, B. F.</au><au>de Vijlder, J. J. M.</au><au>Grootenhuis, M. A.</au><au>Vulsma, T.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Intellectual and Motor Development of Young Adults with Congenital Hypothyroidism Diagnosed by Neonatal Screening</atitle><jtitle>The journal of clinical endocrinology and metabolism</jtitle><addtitle>J Clin Endocrinol Metab</addtitle><date>2006-02</date><risdate>2006</risdate><volume>91</volume><issue>2</issue><spage>418</spage><epage>424</epage><pages>418-424</pages><issn>0021-972X</issn><eissn>1945-7197</eissn><coden>JCEMAZ</coden><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 T4 concentrations.
Main Outcome Measurement: The main outcome measurement was the influence of the 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 (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.</abstract><cop>Bethesda, MD</cop><pub>Endocrine Society</pub><pmid>16303842</pmid><doi>10.1210/jc.2005-1209</doi><tpages>7</tpages><oa>free_for_read</oa></addata></record> |
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| source | MEDLINE; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; Oxford University Press Journals All Titles (1996-Current) |
| subjects | Adult Biological and medical sciences Congenital Hypothyroidism - physiopathology Congenital Hypothyroidism - therapy Endocrinopathies Female Follow-Up Studies Fundamental and applied biological sciences. Psychology Hormone Replacement Therapy Humans Intelligence Longitudinal Studies Male Medical sciences Motor Skills - physiology Non tumoral diseases. Target tissue resistance. Benign neoplasms Statistics, Nonparametric Thyroid. Thyroid axis (diseases) Thyroxine - therapeutic use Vertebrates: endocrinology |
| title | Intellectual and Motor Development of Young Adults with Congenital Hypothyroidism Diagnosed by Neonatal Screening |
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