Growth hormone deficiency in a child with benign hereditary chorea caused by a


Journal

Journal of pediatric endocrinology & metabolism : JPEM
ISSN: 2191-0251
Titre abrégé: J Pediatr Endocrinol Metab
Pays: Germany
ID NLM: 9508900

Informations de publication

Date de publication:
28 Mar 2022
Historique:
received: 14 06 2021
accepted: 11 10 2021
pubmed: 29 10 2021
medline: 5 4 2022
entrez: 28 10 2021
Statut: epublish

Résumé

Benign Hereditary Chorea (BHC) (MIM 118700) is a rare childhood-onset movements disorder characterized by non-progressive chorea. It is usually caused by variants in the thyroid transcription factor 1 (TITF-1/NKX2-1) gene and it is associated with thyroid dysfunction and pulmonary symptoms in the brain-lung-thyroid syndrome. We reported the clinical case of a toddler presenting with neurological symptoms (hypotonia, delayed motor milestones, and axial dystonia) and subclinical hypothyroidism in which we found a ' The peculiarity of our case is that the mild alteration of thyroid-stimulating hormone (TSH) levels, hypotonia, and delayed motor milestones were associated with growth hormone deficiency.

Identifiants

pubmed: 34710315
pii: jpem-2021-0402
doi: 10.1515/jpem-2021-0402
doi:

Substances chimiques

NKX2-1 protein, human 0
Nuclear Proteins 0
Thyroid Nuclear Factor 1 0
Transcription Factors 0
Growth Hormone 9002-72-6

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Pagination

411-415

Informations de copyright

© 2021 Walter de Gruyter GmbH, Berlin/Boston.

Références

Haerer, AF, Currier, RD, Jackson, JF. Hereditary nonprogressive chorea of early onset. N Engl J Med 1967;276:1220–4. https://doi.org/10.1056/NEJM196706012762202.
de Vries, BB, Arts, WF, Breedveld, GJ, Hoogeboom, JJ, Niermeijer, MF, Heutink, P. Benign hereditary chorea of early onset maps to chromosome 14q. Am J Hum Genet 2000;66:136–42. https://doi.org/10.1086/302725.
Breedveld, GJ, Percy, AK, MacDonald, ME, de Vries, BB, Yapijakis, C, Dure, LS, et al.. Clinical and genetic heterogeneity in benign hereditary chorea. Neurology 2002;59:579–84. https://doi.org/10.1212/wnl.59.4.579.
Peall, KJ, Kurian, MA. Benign hereditary chorea: an update. Tremor Other Hyperkinet Mov (N Y) 2015;5:314. https://doi.org/10.7916/D8RJ4HM5.
Carré, A, Szinnai, G, Castanet, M, Sura-Trueba, S, Tron, E, Broutin-L’Hermite, I, et al.. Five new TTF1/NKX2.1 mutations in brain-lung-thyroid syndrome: rescue by PAX8 synergism in one case. Hum Mol Genet 2009;18:2266–76. https://doi.org/10.1093/hmg/ddp162.
Cacciari, E, Milani, S, Balsamo, A, Spada, E, Bona, G, Cavallo, L, et al.. Italian cross-sectional growth charts for height, weight and BMI (2 to 20 yr). J Endocrinol Invest 2006;29:581–93. https://doi.org/10.1007/BF03344156.
Liu, Z, Yanagisawa, K, Griesing, S, Iwai, M, Kano, K, Hotta, N, et al.. TTF-1/NKX2-1 binds to DDB1 and confers replication stress resistance to lung adenocarcinomas. Oncogene 2017;36:3740–8. https://doi.org/10.1038/onc.2016.524.
Peall, KJ, Lumsden, D, Kneen, R, Madhu, R, Peake, D, Gibbon, F, et al.. Benign hereditary chorea related to NKX2.1: expansion of the genotypic and phenotypic spectrum. Dev Med Child Neurol 2014;56:642–8. https://doi.org/10.1111/dmcn.12323.
Kimura, S, Hara, Y, Pineau, T, Fernandez-Salguero, P, Fox, CH, Ward, JM, et al.. The T/ebp null mouse: thyroid-specific enhancer-binding protein is essential for the organogenesis of the thyroid, lung, ventral forebrain, and pituitary. Genes Dev 1996;10:60–9. https://doi.org/10.1101/gad.10.1.60.
Glik, A, Vuillaume, I, Devos, D, Inzelberg, R. Psychosis, short stature in benign hereditary chorea: a novel thyroid transcription factor-1 mutation. Mov Disord 2008;23:1744–7. https://doi.org/10.1002/mds.22215.

Auteurs

Viola Trevisani (V)

Department of Medical and Surgical Sciences of the Mothers, Post Graduate School of Pediatrics, Children and Adults, University of Modena and Reggio Emilia, Modena, Italy.

Barbara Predieri (B)

Department of Medical and Surgical Sciences of the Mothers, Post Graduate School of Pediatrics, Children and Adults, University of Modena and Reggio Emilia, Modena, Italy.
Department of Medical and Surgical Sciences of the Mothers, Pediatric Unit, Children and Adults, University of Modena and Reggio Emilia, Modena, Italy.

Simona Filomena Madeo (SF)

Department of Medical and Surgical Sciences of the Mothers, Pediatric Unit, Children and Adults, University of Modena and Reggio Emilia, Modena, Italy.

Carlo Fusco (C)

Department of Pediatrics, Child Neurology Unit and Pediatric Neurophysiology Laboratory, Azienda USL-IRCCS, Reggio Emilia, Italy.

Livia Garavelli (L)

Mother and Child Health Department, Medical Genetics Unit, Azienda USL-IRCCS, Reggio Emilia, Italy.

Stefano Caraffi (S)

Mother and Child Health Department, Medical Genetics Unit, Azienda USL-IRCCS, Reggio Emilia, Italy.

Lorenzo Iughetti (L)

Department of Medical and Surgical Sciences of the Mothers, Post Graduate School of Pediatrics, Children and Adults, University of Modena and Reggio Emilia, Modena, Italy.
Department of Medical and Surgical Sciences of the Mothers, Pediatric Unit, Children and Adults, University of Modena and Reggio Emilia, Modena, Italy.

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