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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...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Galenos Publishing
2022
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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 |
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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 |
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