Cargando…

Central Precocious Puberty in an Infant with Sotos Syndrome and Response to Treatment

Sotos syndrome (SS) is characterized by overgrowth, distinctive facial appearance, and learning disability. It is caused by heterozygous mutations, including deletions of NSD1 located at chromosome 5q35. While advanced bone age can occur in some cases, precocious puberty (PP) has only been reported...

Descripción completa

Detalles Bibliográficos
Autores principales: Kontbay, Tuğba, Şıklar, Zeynep, Ceylaner, Serdar, Berberoğlu, Merih
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Galenos Publishing 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9422921/
https://www.ncbi.nlm.nih.gov/pubmed/34013836
http://dx.doi.org/10.4274/jcrpe.galenos.2021.2020.0273
_version_ 1784777917521199104
author Kontbay, Tuğba
Şıklar, Zeynep
Ceylaner, Serdar
Berberoğlu, Merih
author_facet Kontbay, Tuğba
Şıklar, Zeynep
Ceylaner, Serdar
Berberoğlu, Merih
author_sort Kontbay, Tuğba
collection PubMed
description Sotos syndrome (SS) is characterized by overgrowth, distinctive facial appearance, and learning disability. It is caused by heterozygous mutations, including deletions of NSD1 located at chromosome 5q35. While advanced bone age can occur in some cases, precocious puberty (PP) has only been reported in three cases previously. Here, we reported a case of SS diagnosed in the infancy period with central PP. The discovery of potential factors that trigger puberty is one of the central mysteries of pubertal biology. Depot gonadotropin-releasing hormone analogs constitute the first-line therapy in central PP (CPP), which has proven to be both effective and safe. In our cases, leuprolide acetate at maximum dose was not successful in controlling pubertal progression, and cyproterone acetate (CPA) was added to therapy, with successful control of pubertal progression. In some specific syndromes with PP, such as SS, treatment can be challenging. CPA may be an asset for effective treatment.
format Online
Article
Text
id pubmed-9422921
institution National Center for Biotechnology Information
language English
publishDate 2022
publisher Galenos Publishing
record_format MEDLINE/PubMed
spelling pubmed-94229212022-09-07 Central Precocious Puberty in an Infant with Sotos Syndrome and Response to Treatment Kontbay, Tuğba Şıklar, Zeynep Ceylaner, Serdar Berberoğlu, Merih J Clin Res Pediatr Endocrinol Case Report Sotos syndrome (SS) is characterized by overgrowth, distinctive facial appearance, and learning disability. It is caused by heterozygous mutations, including deletions of NSD1 located at chromosome 5q35. While advanced bone age can occur in some cases, precocious puberty (PP) has only been reported in three cases previously. Here, we reported a case of SS diagnosed in the infancy period with central PP. The discovery of potential factors that trigger puberty is one of the central mysteries of pubertal biology. Depot gonadotropin-releasing hormone analogs constitute the first-line therapy in central PP (CPP), which has proven to be both effective and safe. In our cases, leuprolide acetate at maximum dose was not successful in controlling pubertal progression, and cyproterone acetate (CPA) was added to therapy, with successful control of pubertal progression. In some specific syndromes with PP, such as SS, treatment can be challenging. CPA may be an asset for effective treatment. Galenos Publishing 2022-09 2022-08-25 /pmc/articles/PMC9422921/ /pubmed/34013836 http://dx.doi.org/10.4274/jcrpe.galenos.2021.2020.0273 Text en ©Copyright 2022 by Turkish Pediatric Endocrinology and Diabetes Society | The Journal of Clinical Research in Pediatric Endocrinology published by Galenos Publishing House. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Kontbay, Tuğba
Şıklar, Zeynep
Ceylaner, Serdar
Berberoğlu, Merih
Central Precocious Puberty in an Infant with Sotos Syndrome and Response to Treatment
title Central Precocious Puberty in an Infant with Sotos Syndrome and Response to Treatment
title_full Central Precocious Puberty in an Infant with Sotos Syndrome and Response to Treatment
title_fullStr Central Precocious Puberty in an Infant with Sotos Syndrome and Response to Treatment
title_full_unstemmed Central Precocious Puberty in an Infant with Sotos Syndrome and Response to Treatment
title_short Central Precocious Puberty in an Infant with Sotos Syndrome and Response to Treatment
title_sort central precocious puberty in an infant with sotos syndrome and response to treatment
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9422921/
https://www.ncbi.nlm.nih.gov/pubmed/34013836
http://dx.doi.org/10.4274/jcrpe.galenos.2021.2020.0273
work_keys_str_mv AT kontbaytugba centralprecociouspubertyinaninfantwithsotossyndromeandresponsetotreatment
AT sıklarzeynep centralprecociouspubertyinaninfantwithsotossyndromeandresponsetotreatment
AT ceylanerserdar centralprecociouspubertyinaninfantwithsotossyndromeandresponsetotreatment
AT berberoglumerih centralprecociouspubertyinaninfantwithsotossyndromeandresponsetotreatment