Cargando…
Testicular Adrenal Rest Tumors in Congenital Adrenal Hyperplasia: Study of a Cohort of Patients from a Single Italian Center
Testicular adrenal rest tumors (TARTs) are a common complication in male patients with congenital adrenal hyperplasia (CAH). The aim of our cross-sectional cohort study is to estimate the frequency of TARTs with the correlation of genotype and disease control on tumor development. Thirty-five male p...
Autores principales: | , , , , , , , , , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2023
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10528159/ https://www.ncbi.nlm.nih.gov/pubmed/37761418 http://dx.doi.org/10.3390/children10091457 |
_version_ | 1785111231298797568 |
---|---|
author | Ortolano, Rita Cassio, Alessandra Alqaisi, Randa S. Candela, Egidio Di Natale, Valeria Assirelli, Valentina Bernardini, Luca Bortolamedi, Elisa Cantarelli, Erika Corcioni, Beniamino Renzulli, Matteo Balsamo, Antonio Baronio, Federico |
author_facet | Ortolano, Rita Cassio, Alessandra Alqaisi, Randa S. Candela, Egidio Di Natale, Valeria Assirelli, Valentina Bernardini, Luca Bortolamedi, Elisa Cantarelli, Erika Corcioni, Beniamino Renzulli, Matteo Balsamo, Antonio Baronio, Federico |
author_sort | Ortolano, Rita |
collection | PubMed |
description | Testicular adrenal rest tumors (TARTs) are a common complication in male patients with congenital adrenal hyperplasia (CAH). The aim of our cross-sectional cohort study is to estimate the frequency of TARTs with the correlation of genotype and disease control on tumor development. Thirty-five male patients, aged 14–26 years, were included in the study, all followed by the same center of pediatric endocrinology in Bologna. We studied genotypes, hormonal profiles at different time intervals and testicular ultrasound. A logistic regression model with multivariant analysis was developed for the statistical analysis. TARTs were detected in 31.4% of the cases, 90.9% of them had a classic form with salt wasting, while 9.1% had a non-classic form. Additionally, a significant correlation between the incidence of TARTs and severity of genotype was detected. Patients with TARTs had markedly worse metabolic control on average (p = 0.027), reflected by high ACTH, 17OH progesterone, and overall delta4-androstenedione. In conclusion, a screening tool is mandatory, especially (but not exclusively) in patients with the most severe forms of CAH and poor endocrine control of the disease. |
format | Online Article Text |
id | pubmed-10528159 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-105281592023-09-28 Testicular Adrenal Rest Tumors in Congenital Adrenal Hyperplasia: Study of a Cohort of Patients from a Single Italian Center Ortolano, Rita Cassio, Alessandra Alqaisi, Randa S. Candela, Egidio Di Natale, Valeria Assirelli, Valentina Bernardini, Luca Bortolamedi, Elisa Cantarelli, Erika Corcioni, Beniamino Renzulli, Matteo Balsamo, Antonio Baronio, Federico Children (Basel) Article Testicular adrenal rest tumors (TARTs) are a common complication in male patients with congenital adrenal hyperplasia (CAH). The aim of our cross-sectional cohort study is to estimate the frequency of TARTs with the correlation of genotype and disease control on tumor development. Thirty-five male patients, aged 14–26 years, were included in the study, all followed by the same center of pediatric endocrinology in Bologna. We studied genotypes, hormonal profiles at different time intervals and testicular ultrasound. A logistic regression model with multivariant analysis was developed for the statistical analysis. TARTs were detected in 31.4% of the cases, 90.9% of them had a classic form with salt wasting, while 9.1% had a non-classic form. Additionally, a significant correlation between the incidence of TARTs and severity of genotype was detected. Patients with TARTs had markedly worse metabolic control on average (p = 0.027), reflected by high ACTH, 17OH progesterone, and overall delta4-androstenedione. In conclusion, a screening tool is mandatory, especially (but not exclusively) in patients with the most severe forms of CAH and poor endocrine control of the disease. MDPI 2023-08-26 /pmc/articles/PMC10528159/ /pubmed/37761418 http://dx.doi.org/10.3390/children10091457 Text en © 2023 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/). |
spellingShingle | Article Ortolano, Rita Cassio, Alessandra Alqaisi, Randa S. Candela, Egidio Di Natale, Valeria Assirelli, Valentina Bernardini, Luca Bortolamedi, Elisa Cantarelli, Erika Corcioni, Beniamino Renzulli, Matteo Balsamo, Antonio Baronio, Federico Testicular Adrenal Rest Tumors in Congenital Adrenal Hyperplasia: Study of a Cohort of Patients from a Single Italian Center |
title | Testicular Adrenal Rest Tumors in Congenital Adrenal Hyperplasia: Study of a Cohort of Patients from a Single Italian Center |
title_full | Testicular Adrenal Rest Tumors in Congenital Adrenal Hyperplasia: Study of a Cohort of Patients from a Single Italian Center |
title_fullStr | Testicular Adrenal Rest Tumors in Congenital Adrenal Hyperplasia: Study of a Cohort of Patients from a Single Italian Center |
title_full_unstemmed | Testicular Adrenal Rest Tumors in Congenital Adrenal Hyperplasia: Study of a Cohort of Patients from a Single Italian Center |
title_short | Testicular Adrenal Rest Tumors in Congenital Adrenal Hyperplasia: Study of a Cohort of Patients from a Single Italian Center |
title_sort | testicular adrenal rest tumors in congenital adrenal hyperplasia: study of a cohort of patients from a single italian center |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10528159/ https://www.ncbi.nlm.nih.gov/pubmed/37761418 http://dx.doi.org/10.3390/children10091457 |
work_keys_str_mv | AT ortolanorita testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT cassioalessandra testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT alqaisirandas testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT candelaegidio testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT dinatalevaleria testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT assirellivalentina testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT bernardiniluca testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT bortolamedielisa testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT cantarellierika testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT corcionibeniamino testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT renzullimatteo testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT balsamoantonio testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter AT baroniofederico testicularadrenalresttumorsincongenitaladrenalhyperplasiastudyofacohortofpatientsfromasingleitaliancenter |