Testicular Adrenal Rest Tumors and Leydig and Sertoli Cell Function in Boys with Classical Congenital Adrenal Hyperplasia
Context: Infertility observed in adult males with congenital adrenal hyperplasia (CAH) has been associated with testicular adrenal rest tumors (TART) that may originate during childhood. Objective: Our objective was to describe the prevalence of TART and Sertoli and Leydig cell function in a group o...
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| Veröffentlicht in: | The journal of clinical endocrinology and metabolism 2007-12, Vol.92 (12), p.4583-4589 |
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| creator | Martinez-Aguayo, A. Rocha, A. Rojas, N. García, C. Parra, R. Lagos, M. Valdivia, L. Poggi, H. Cattani, A. |
| description | Context: Infertility observed in adult males with congenital adrenal hyperplasia (CAH) has been associated with testicular adrenal rest tumors (TART) that may originate during childhood.
Objective: Our objective was to describe the prevalence of TART and Sertoli and Leydig cell function in a group of boys aged 2–10 yr with CAH and to compare prevalence with that of a control group.
Design: From August 2005 to January 2007, 19 patients with classical CAH (CAH group) were referred from seven endocrinology centers.
Methods: We studied 19 subjects in the CAH group and, as a control group, 13 boys from the community that did not have testicular diseases. A complete physical exam was performed. High-resolution ultrasound was used to determine TART prevalence. Inhibin B and anti-Müllerian hormone were used as Sertoli cell markers. The ratio between basal testosterone levels and testosterone levels 72 h after β-human chorionic gonadotropin (5000 U/m2) treatment [(T72− T0)/T0] was used to evaluate Leydig cell response.
Results: CAH and control groups were comparable in chronological age (5.9 vs. 5.6 yr; P = 0.67) and bone age/chronological age ratio (1.09 vs. 1.03; P = 0.09). TART prevalence was four of 19 (21%) in the CAH group. Lower values for inhibin B (49.2. vs. 65.2 pg/ml; P = 0.018), anti-Müllerian hormone (70.1 vs. 94.2 ng/ml; P = 0.002), and (T72− T0)/T0 (5.6 vs. 13.6; P < 0.01) were observed in the CAH group.
Conclusion: TART in prepubertal males with classic CAH could be found during childhood. We also report differences in markers of gonadal function in a subgroup of patients, especially in those with inadequate control. |
| doi_str_mv | 10.1210/jc.2007-0383 |
| format | Article |
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Objective: Our objective was to describe the prevalence of TART and Sertoli and Leydig cell function in a group of boys aged 2–10 yr with CAH and to compare prevalence with that of a control group.
Design: From August 2005 to January 2007, 19 patients with classical CAH (CAH group) were referred from seven endocrinology centers.
Methods: We studied 19 subjects in the CAH group and, as a control group, 13 boys from the community that did not have testicular diseases. A complete physical exam was performed. High-resolution ultrasound was used to determine TART prevalence. Inhibin B and anti-Müllerian hormone were used as Sertoli cell markers. The ratio between basal testosterone levels and testosterone levels 72 h after β-human chorionic gonadotropin (5000 U/m2) treatment [(T72− T0)/T0] was used to evaluate Leydig cell response.
Results: CAH and control groups were comparable in chronological age (5.9 vs. 5.6 yr; P = 0.67) and bone age/chronological age ratio (1.09 vs. 1.03; P = 0.09). TART prevalence was four of 19 (21%) in the CAH group. Lower values for inhibin B (49.2. vs. 65.2 pg/ml; P = 0.018), anti-Müllerian hormone (70.1 vs. 94.2 ng/ml; P = 0.002), and (T72− T0)/T0 (5.6 vs. 13.6; P < 0.01) were observed in the CAH group.
Conclusion: TART in prepubertal males with classic CAH could be found during childhood. We also report differences in markers of gonadal function in a subgroup of patients, especially in those with inadequate control.</description><identifier>ISSN: 0021-972X</identifier><identifier>EISSN: 1945-7197</identifier><identifier>DOI: 10.1210/jc.2007-0383</identifier><identifier>PMID: 17895312</identifier><identifier>CODEN: JCEMAZ</identifier><language>eng</language><publisher>Bethesda, MD: Endocrine Society</publisher><subject>Adrenal Hyperplasia, Congenital - complications ; Adrenal Hyperplasia, Congenital - genetics ; Adrenal Hyperplasia, Congenital - physiopathology ; Adrenal Rest Tumor - complications ; Adrenal Rest Tumor - genetics ; Adrenal Rest Tumor - physiopathology ; Adrenals. Adrenal axis. Renin-angiotensin system (diseases) ; Anthropometry ; Anti-Mullerian Hormone - metabolism ; Biological and medical sciences ; Child ; Child, Preschool ; DNA - genetics ; Endocrinopathies ; Feeding. Feeding behavior ; Fundamental and applied biological sciences. Psychology ; Hormones - blood ; Humans ; Inhibins - metabolism ; Leydig Cells - physiology ; Male ; Medical sciences ; Non tumoral diseases. Target tissue resistance. Benign neoplasms ; Sertoli Cells - physiology ; Testicular Neoplasms - complications ; Testicular Neoplasms - genetics ; Testicular Neoplasms - physiopathology ; Vertebrates: anatomy and physiology, studies on body, several organs or systems ; Vertebrates: endocrinology</subject><ispartof>The journal of clinical endocrinology and metabolism, 2007-12, Vol.92 (12), p.4583-4589</ispartof><rights>2008 INIST-CNRS</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c401t-2c2be9075bfae915a2094685a778545042072a49e13f3e269302e4c91bc2f17e3</citedby><cites>FETCH-LOGICAL-c401t-2c2be9075bfae915a2094685a778545042072a49e13f3e269302e4c91bc2f17e3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,776,780,27901,27902</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=19895831$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/17895312$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Martinez-Aguayo, A.</creatorcontrib><creatorcontrib>Rocha, A.</creatorcontrib><creatorcontrib>Rojas, N.</creatorcontrib><creatorcontrib>García, C.</creatorcontrib><creatorcontrib>Parra, R.</creatorcontrib><creatorcontrib>Lagos, M.</creatorcontrib><creatorcontrib>Valdivia, L.</creatorcontrib><creatorcontrib>Poggi, H.</creatorcontrib><creatorcontrib>Cattani, A.</creatorcontrib><creatorcontrib>Chilean Collaborative Testicular Adrenal Rest Tumor Study Group</creatorcontrib><title>Testicular Adrenal Rest Tumors and Leydig and Sertoli Cell Function in Boys with Classical Congenital Adrenal Hyperplasia</title><title>The journal of clinical endocrinology and metabolism</title><addtitle>J Clin Endocrinol Metab</addtitle><description>Context: Infertility observed in adult males with congenital adrenal hyperplasia (CAH) has been associated with testicular adrenal rest tumors (TART) that may originate during childhood.
Objective: Our objective was to describe the prevalence of TART and Sertoli and Leydig cell function in a group of boys aged 2–10 yr with CAH and to compare prevalence with that of a control group.
Design: From August 2005 to January 2007, 19 patients with classical CAH (CAH group) were referred from seven endocrinology centers.
Methods: We studied 19 subjects in the CAH group and, as a control group, 13 boys from the community that did not have testicular diseases. A complete physical exam was performed. High-resolution ultrasound was used to determine TART prevalence. Inhibin B and anti-Müllerian hormone were used as Sertoli cell markers. The ratio between basal testosterone levels and testosterone levels 72 h after β-human chorionic gonadotropin (5000 U/m2) treatment [(T72− T0)/T0] was used to evaluate Leydig cell response.
Results: CAH and control groups were comparable in chronological age (5.9 vs. 5.6 yr; P = 0.67) and bone age/chronological age ratio (1.09 vs. 1.03; P = 0.09). TART prevalence was four of 19 (21%) in the CAH group. Lower values for inhibin B (49.2. vs. 65.2 pg/ml; P = 0.018), anti-Müllerian hormone (70.1 vs. 94.2 ng/ml; P = 0.002), and (T72− T0)/T0 (5.6 vs. 13.6; P < 0.01) were observed in the CAH group.
Conclusion: TART in prepubertal males with classic CAH could be found during childhood. We also report differences in markers of gonadal function in a subgroup of patients, especially in those with inadequate control.</description><subject>Adrenal Hyperplasia, Congenital - complications</subject><subject>Adrenal Hyperplasia, Congenital - genetics</subject><subject>Adrenal Hyperplasia, Congenital - physiopathology</subject><subject>Adrenal Rest Tumor - complications</subject><subject>Adrenal Rest Tumor - genetics</subject><subject>Adrenal Rest Tumor - physiopathology</subject><subject>Adrenals. Adrenal axis. Renin-angiotensin system (diseases)</subject><subject>Anthropometry</subject><subject>Anti-Mullerian Hormone - metabolism</subject><subject>Biological and medical sciences</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>DNA - genetics</subject><subject>Endocrinopathies</subject><subject>Feeding. Feeding behavior</subject><subject>Fundamental and applied biological sciences. Psychology</subject><subject>Hormones - blood</subject><subject>Humans</subject><subject>Inhibins - metabolism</subject><subject>Leydig Cells - physiology</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Non tumoral diseases. Target tissue resistance. Benign neoplasms</subject><subject>Sertoli Cells - physiology</subject><subject>Testicular Neoplasms - complications</subject><subject>Testicular Neoplasms - genetics</subject><subject>Testicular Neoplasms - physiopathology</subject><subject>Vertebrates: anatomy and physiology, studies on body, several organs or systems</subject><subject>Vertebrates: endocrinology</subject><issn>0021-972X</issn><issn>1945-7197</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2007</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNptkE2P0zAURS0EYkphxxp5Aysy-Ct1vJyJGAapEhIUiZ3lOi-Dq9QOfolQ_j0uLZoNq3f1dHxtH0Jec3bNBWcfDv5aMKYrJhv5hKy4UXWludFPyYoxwSujxY8r8gLxwBhXqpbPyRXXjaklFyuy7ACn4OfBZXrTZYhuoF_Liu7mY8pIXezoFpYuPPyN3yBPaQi0hWGgd3P0U0iRhkhv04L0d5h-0nZwiMGXnjbFB4hhKvFf9f0yQh4LEdxL8qx3A8Kry1yT73cfd-19tf3y6XN7s628YnyqhBd7MEzX-96B4bUTzKhNUzutm1rVTAmmhVMGuOwliI2RTIDyhu-96LkGuSbvzr1jTr_m8jV7DOjL-12ENKPdGCaa07k1eX8GfU6IGXo75nB0ebGc2ZNqe_D2pNqeVBf8zaV33h-he4Qvbgvw9gI4LDr67KIP-MiZwjWSF06eOYhd8jlEGDMg2kOac3GG_7_-D6vIl4M</recordid><startdate>20071201</startdate><enddate>20071201</enddate><creator>Martinez-Aguayo, A.</creator><creator>Rocha, A.</creator><creator>Rojas, N.</creator><creator>García, C.</creator><creator>Parra, R.</creator><creator>Lagos, M.</creator><creator>Valdivia, L.</creator><creator>Poggi, H.</creator><creator>Cattani, A.</creator><general>Endocrine Society</general><scope>IQODW</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope></search><sort><creationdate>20071201</creationdate><title>Testicular Adrenal Rest Tumors and Leydig and Sertoli Cell Function in Boys with Classical Congenital Adrenal Hyperplasia</title><author>Martinez-Aguayo, A. ; Rocha, A. ; Rojas, N. ; García, C. ; Parra, R. ; Lagos, M. ; Valdivia, L. ; Poggi, H. ; Cattani, A.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c401t-2c2be9075bfae915a2094685a778545042072a49e13f3e269302e4c91bc2f17e3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2007</creationdate><topic>Adrenal Hyperplasia, Congenital - complications</topic><topic>Adrenal Hyperplasia, Congenital - genetics</topic><topic>Adrenal Hyperplasia, Congenital - physiopathology</topic><topic>Adrenal Rest Tumor - complications</topic><topic>Adrenal Rest Tumor - genetics</topic><topic>Adrenal Rest Tumor - physiopathology</topic><topic>Adrenals. Adrenal axis. Renin-angiotensin system (diseases)</topic><topic>Anthropometry</topic><topic>Anti-Mullerian Hormone - metabolism</topic><topic>Biological and medical sciences</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>DNA - genetics</topic><topic>Endocrinopathies</topic><topic>Feeding. Feeding behavior</topic><topic>Fundamental and applied biological sciences. Psychology</topic><topic>Hormones - blood</topic><topic>Humans</topic><topic>Inhibins - metabolism</topic><topic>Leydig Cells - physiology</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Non tumoral diseases. Target tissue resistance. Benign neoplasms</topic><topic>Sertoli Cells - physiology</topic><topic>Testicular Neoplasms - complications</topic><topic>Testicular Neoplasms - genetics</topic><topic>Testicular Neoplasms - physiopathology</topic><topic>Vertebrates: anatomy and physiology, studies on body, several organs or systems</topic><topic>Vertebrates: endocrinology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Martinez-Aguayo, A.</creatorcontrib><creatorcontrib>Rocha, A.</creatorcontrib><creatorcontrib>Rojas, N.</creatorcontrib><creatorcontrib>García, C.</creatorcontrib><creatorcontrib>Parra, R.</creatorcontrib><creatorcontrib>Lagos, M.</creatorcontrib><creatorcontrib>Valdivia, L.</creatorcontrib><creatorcontrib>Poggi, H.</creatorcontrib><creatorcontrib>Cattani, A.</creatorcontrib><creatorcontrib>Chilean Collaborative Testicular Adrenal Rest Tumor Study Group</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>The journal of clinical endocrinology and metabolism</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Martinez-Aguayo, A.</au><au>Rocha, A.</au><au>Rojas, N.</au><au>García, C.</au><au>Parra, R.</au><au>Lagos, M.</au><au>Valdivia, L.</au><au>Poggi, H.</au><au>Cattani, A.</au><aucorp>Chilean Collaborative Testicular Adrenal Rest Tumor Study Group</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Testicular Adrenal Rest Tumors and Leydig and Sertoli Cell Function in Boys with Classical Congenital Adrenal Hyperplasia</atitle><jtitle>The journal of clinical endocrinology and metabolism</jtitle><addtitle>J Clin Endocrinol Metab</addtitle><date>2007-12-01</date><risdate>2007</risdate><volume>92</volume><issue>12</issue><spage>4583</spage><epage>4589</epage><pages>4583-4589</pages><issn>0021-972X</issn><eissn>1945-7197</eissn><coden>JCEMAZ</coden><abstract>Context: Infertility observed in adult males with congenital adrenal hyperplasia (CAH) has been associated with testicular adrenal rest tumors (TART) that may originate during childhood.
Objective: Our objective was to describe the prevalence of TART and Sertoli and Leydig cell function in a group of boys aged 2–10 yr with CAH and to compare prevalence with that of a control group.
Design: From August 2005 to January 2007, 19 patients with classical CAH (CAH group) were referred from seven endocrinology centers.
Methods: We studied 19 subjects in the CAH group and, as a control group, 13 boys from the community that did not have testicular diseases. A complete physical exam was performed. High-resolution ultrasound was used to determine TART prevalence. Inhibin B and anti-Müllerian hormone were used as Sertoli cell markers. The ratio between basal testosterone levels and testosterone levels 72 h after β-human chorionic gonadotropin (5000 U/m2) treatment [(T72− T0)/T0] was used to evaluate Leydig cell response.
Results: CAH and control groups were comparable in chronological age (5.9 vs. 5.6 yr; P = 0.67) and bone age/chronological age ratio (1.09 vs. 1.03; P = 0.09). TART prevalence was four of 19 (21%) in the CAH group. Lower values for inhibin B (49.2. vs. 65.2 pg/ml; P = 0.018), anti-Müllerian hormone (70.1 vs. 94.2 ng/ml; P = 0.002), and (T72− T0)/T0 (5.6 vs. 13.6; P < 0.01) were observed in the CAH group.
Conclusion: TART in prepubertal males with classic CAH could be found during childhood. We also report differences in markers of gonadal function in a subgroup of patients, especially in those with inadequate control.</abstract><cop>Bethesda, MD</cop><pub>Endocrine Society</pub><pmid>17895312</pmid><doi>10.1210/jc.2007-0383</doi><tpages>7</tpages><oa>free_for_read</oa></addata></record> |
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| source | Oxford University Press Journals All Titles (1996-Current); MEDLINE; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals |
| subjects | Adrenal Hyperplasia, Congenital - complications Adrenal Hyperplasia, Congenital - genetics Adrenal Hyperplasia, Congenital - physiopathology Adrenal Rest Tumor - complications Adrenal Rest Tumor - genetics Adrenal Rest Tumor - physiopathology Adrenals. Adrenal axis. Renin-angiotensin system (diseases) Anthropometry Anti-Mullerian Hormone - metabolism Biological and medical sciences Child Child, Preschool DNA - genetics Endocrinopathies Feeding. Feeding behavior Fundamental and applied biological sciences. Psychology Hormones - blood Humans Inhibins - metabolism Leydig Cells - physiology Male Medical sciences Non tumoral diseases. Target tissue resistance. Benign neoplasms Sertoli Cells - physiology Testicular Neoplasms - complications Testicular Neoplasms - genetics Testicular Neoplasms - physiopathology Vertebrates: anatomy and physiology, studies on body, several organs or systems Vertebrates: endocrinology |
| title | Testicular Adrenal Rest Tumors and Leydig and Sertoli Cell Function in Boys with Classical Congenital Adrenal Hyperplasia |
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