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Thyroid function from birth to adolescence in Prader-Willi syndrome.
J Pediatr 2013; 163(3):800-5JPed

Abstract

OBJECTIVES

To describe the response of thyroid-stimulating hormone (TSH) to thyroid-releasing hormone in children and adolescents with Prader-Willi syndrome (PWS), and to compare TSH and total thyroxine (TT4) concentrations measured on neonatal screening for congenital hypothyroidism in children with PWS and controls.

STUDY DESIGN

All participants had genetically confirmed PWS. The TSH responses to thyroid-releasing hormone, free thyroxine (fT4), and free triiodothyronine (fT3) were measured in 21 subjects (14 females and 7 males; mean age, 6.4 years). Capillary TT4 was measured on neonatal screening samples from 23 subjects with PWS (14 females and 9 males), each of whom was matched for birth weight and sex with 4 anonymized controls.

RESULTS

One subject with PWS had tertiary hypothyroidism. TSH level increased from 1.37 mU/L at baseline to 39.6 mU/L at 20 minutes, 47.2 mU/L at 40 minutes, 44.5 mU/L at 60 minutes, and 47.2 mU/L at 120 minutes. fT4 concentration was 6.3 pmol/L, and fT3 concentration was 4.6 pmol/L. In the other 20 subjects, mean TSH level was 1.9 mU/L (range, 0.8-4.2 mU/L) at baseline and 21.8 mU/L (range, 10.0-46.7 mU/L) at 20 minutes (peak). Mean fT4 concentration (10.4 pmol/L; range, 8.2-13.5 pmol/L) was in the lower one-third of the normal range in 18 subjects, and mean fT3 concentration (6.1 pmol/L; range, 4.8-8.4 pmol/L) was above the median in 13 subjects. In neonates, mean TSH level was 3.1 mU/L (range, 0.4-10.0 mU/L) in subjects with PWS versus 3.3 mU/L (range, 0.0-7.0 mU/L) in controls, and mean TT4 in subjects with PWS was 111% (range, 17%-203%) that of controls (P = not significant).

CONCLUSION

Thyroid function was normal in our newborn subjects. In older children, frank hypothyroidism was found in only 1 of our 21 subjects. Thus, levothyroxine treatment should not be routinely prescribed to youth with PWS.

Authors+Show Affiliations

Endocrinology and Diabetes Unit, British Columbia's Children's Hospital, University of British Columbia, Vancouver, British Columbia, Canada.No affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Journal Article

Language

eng

PubMed ID

23623517

Citation

Sharkia, Mohamad, et al. "Thyroid Function From Birth to Adolescence in Prader-Willi Syndrome." The Journal of Pediatrics, vol. 163, no. 3, 2013, pp. 800-5.
Sharkia M, Michaud S, Berthier MT, et al. Thyroid function from birth to adolescence in Prader-Willi syndrome. J Pediatr. 2013;163(3):800-5.
Sharkia, M., Michaud, S., Berthier, M. T., Giguère, Y., Stewart, L., Deladoëy, J., ... Chanoine, J. P. (2013). Thyroid function from birth to adolescence in Prader-Willi syndrome. The Journal of Pediatrics, 163(3), pp. 800-5. doi:10.1016/j.jpeds.2013.03.058.
Sharkia M, et al. Thyroid Function From Birth to Adolescence in Prader-Willi Syndrome. J Pediatr. 2013;163(3):800-5. PubMed PMID: 23623517.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Thyroid function from birth to adolescence in Prader-Willi syndrome. AU - Sharkia,Mohamad, AU - Michaud,Stéphanie, AU - Berthier,Marie-Thérèse, AU - Giguère,Yves, AU - Stewart,Laura, AU - Deladoëy,Johnny, AU - Deal,Cheri, AU - Van Vliet,Guy, AU - Chanoine,Jean-Pierre, Y1 - 2013/04/25/ PY - 2012/11/07/received PY - 2013/02/20/revised PY - 2013/03/20/accepted PY - 2013/4/30/entrez PY - 2013/4/30/pubmed PY - 2013/11/19/medline KW - BMI KW - Body mass index KW - CV KW - Coefficient of variation KW - Free thyroxine KW - Free triiodothyronine KW - HDL KW - High-density lipoprotein KW - LDL KW - LT4 KW - Levothyroxine KW - Low-density lipoprotein KW - PWS KW - Prader-Willi syndrome KW - TRH KW - TSH KW - TT4 KW - Thyroid-releasing hormone KW - Thyroid-stimulating hormone KW - Total thyroxine KW - fT3 KW - fT4 SP - 800 EP - 5 JF - The Journal of pediatrics JO - J. Pediatr. VL - 163 IS - 3 N2 - OBJECTIVES: To describe the response of thyroid-stimulating hormone (TSH) to thyroid-releasing hormone in children and adolescents with Prader-Willi syndrome (PWS), and to compare TSH and total thyroxine (TT4) concentrations measured on neonatal screening for congenital hypothyroidism in children with PWS and controls. STUDY DESIGN: All participants had genetically confirmed PWS. The TSH responses to thyroid-releasing hormone, free thyroxine (fT4), and free triiodothyronine (fT3) were measured in 21 subjects (14 females and 7 males; mean age, 6.4 years). Capillary TT4 was measured on neonatal screening samples from 23 subjects with PWS (14 females and 9 males), each of whom was matched for birth weight and sex with 4 anonymized controls. RESULTS: One subject with PWS had tertiary hypothyroidism. TSH level increased from 1.37 mU/L at baseline to 39.6 mU/L at 20 minutes, 47.2 mU/L at 40 minutes, 44.5 mU/L at 60 minutes, and 47.2 mU/L at 120 minutes. fT4 concentration was 6.3 pmol/L, and fT3 concentration was 4.6 pmol/L. In the other 20 subjects, mean TSH level was 1.9 mU/L (range, 0.8-4.2 mU/L) at baseline and 21.8 mU/L (range, 10.0-46.7 mU/L) at 20 minutes (peak). Mean fT4 concentration (10.4 pmol/L; range, 8.2-13.5 pmol/L) was in the lower one-third of the normal range in 18 subjects, and mean fT3 concentration (6.1 pmol/L; range, 4.8-8.4 pmol/L) was above the median in 13 subjects. In neonates, mean TSH level was 3.1 mU/L (range, 0.4-10.0 mU/L) in subjects with PWS versus 3.3 mU/L (range, 0.0-7.0 mU/L) in controls, and mean TT4 in subjects with PWS was 111% (range, 17%-203%) that of controls (P = not significant). CONCLUSION: Thyroid function was normal in our newborn subjects. In older children, frank hypothyroidism was found in only 1 of our 21 subjects. Thus, levothyroxine treatment should not be routinely prescribed to youth with PWS. SN - 1097-6833 UR - https://www.unboundmedicine.com/medline/citation/23623517/Thyroid_function_from_birth_to_adolescence_in_Prader_Willi_syndrome_ L2 - https://linkinghub.elsevier.com/retrieve/pii/S0022-3476(13)00377-6 DB - PRIME DP - Unbound Medicine ER -