Testicular Adrenal Rest Tumors in Adult Males with Congenital Adrenal Hyperplasia: Evaluation of Pituitary-Gonadal Function before and after Successful Testis-Sparing Surgery in Eight Patients
Context: In male patients with congenital adrenal hyperplasia (CAH), testicular adrenal rest tumors (TART) are frequently present. These tumors can interfere with testicular function. Intensifying glucocorticoid therapy does not always lead to tumor regression and improvement of testicular function....
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| Veröffentlicht in: | The journal of clinical endocrinology and metabolism 2007-02, Vol.92 (2), p.612-615 |
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| creator | Claahsen-van der Grinten, Hedi L. Otten, Barto J. Takahashi, Satoru Meuleman, Eric J. H. Hulsbergen-van de Kaa, Christina Sweep, Fred C. G. J. Hermus, Ad R. M. M. |
| description | Context: In male patients with congenital adrenal hyperplasia (CAH), testicular adrenal rest tumors (TART) are frequently present. These tumors can interfere with testicular function. Intensifying glucocorticoid therapy does not always lead to tumor regression and improvement of testicular function. Recently, testis-sparing surgery was introduced for treatment of TART.
Objective: The aim of this study was to evaluate tumor volume, symptoms, and pituitary-gonadal function in male patients with CAH caused by 21-hydroxylase deficiency and bilateral TART before and after testis-sparing surgery.
Setting: This study was conducted at Radboud University Nijmegen Medical Centre in The Netherlands.
Patients: Eight adult male CAH patients with bilateral TART and infertility were included.
Interventions: Evaluation of testicular magnetic resonance imaging, symptoms, fasting serum concentrations of ACTH, LH, FSH, inhibin B, 17-OH progesterone, androstenedione, testosterone, and estrone, and semen analysis (six of eight patients) was performed before and 6 and 22 months after testis-sparing surgery.
Main Outcome Measures: The main outcome measures were absence of residual tumor and improvement of symptoms and pituitary-gonadal function.
Results: Residual tumors were not found on any of the patients’ magnetic resonance imaging after surgery. Two patients reported testicular pain and discomfort that disappeared after surgery. Parameters of pituitary-gonadal function did not improve after surgery: semen analysis showed azoospermia (five patients) or oligospermia (one patient) without improvement, and all patients had persistently low inhibin B concentrations.
Conclusion: Testis-sparing surgery did not improve pituitary-gonadal function despite successful removal of the tumors. Further studies are needed to investigate whether surgery at an earlier stage in the natural history of TART can prevent permanent testicular damage. |
| doi_str_mv | 10.1210/jc.2006-1311 |
| format | Article |
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Objective: The aim of this study was to evaluate tumor volume, symptoms, and pituitary-gonadal function in male patients with CAH caused by 21-hydroxylase deficiency and bilateral TART before and after testis-sparing surgery.
Setting: This study was conducted at Radboud University Nijmegen Medical Centre in The Netherlands.
Patients: Eight adult male CAH patients with bilateral TART and infertility were included.
Interventions: Evaluation of testicular magnetic resonance imaging, symptoms, fasting serum concentrations of ACTH, LH, FSH, inhibin B, 17-OH progesterone, androstenedione, testosterone, and estrone, and semen analysis (six of eight patients) was performed before and 6 and 22 months after testis-sparing surgery.
Main Outcome Measures: The main outcome measures were absence of residual tumor and improvement of symptoms and pituitary-gonadal function.
Results: Residual tumors were not found on any of the patients’ magnetic resonance imaging after surgery. Two patients reported testicular pain and discomfort that disappeared after surgery. Parameters of pituitary-gonadal function did not improve after surgery: semen analysis showed azoospermia (five patients) or oligospermia (one patient) without improvement, and all patients had persistently low inhibin B concentrations.
Conclusion: Testis-sparing surgery did not improve pituitary-gonadal function despite successful removal of the tumors. Further studies are needed to investigate whether surgery at an earlier stage in the natural history of TART can prevent permanent testicular damage.</description><identifier>ISSN: 0021-972X</identifier><identifier>EISSN: 1945-7197</identifier><identifier>DOI: 10.1210/jc.2006-1311</identifier><identifier>PMID: 17090637</identifier><identifier>CODEN: JCEMAZ</identifier><language>eng</language><publisher>Bethesda, MD: Endocrine Society</publisher><subject>17-alpha-Hydroxyprogesterone - blood ; Adrenal Hyperplasia, Congenital - pathology ; Adrenal Hyperplasia, Congenital - physiopathology ; Adrenal Hyperplasia, Congenital - surgery ; Adrenal Rest Tumor - pathology ; Adrenal Rest Tumor - physiopathology ; Adrenal Rest Tumor - surgery ; Adrenals. Adrenal axis. Renin-angiotensin system (diseases) ; Adrenocorticotropic Hormone - blood ; Adult ; Androstenedione - blood ; Azoospermia - pathology ; Azoospermia - physiopathology ; Biological and medical sciences ; Biopsy ; Endocrinopathies ; Estrone - blood ; Follicle Stimulating Hormone - blood ; Fundamental and applied biological sciences. Psychology ; Humans ; Inhibins - blood ; Luteinizing Hormone - blood ; Magnetic Resonance Imaging ; Male ; Medical sciences ; Non tumoral diseases. Target tissue resistance. Benign neoplasms ; Pituitary Gland - pathology ; Pituitary Gland - physiology ; Postoperative Complications - physiopathology ; Preoperative Care ; Testicular Neoplasms - pathology ; Testicular Neoplasms - physiopathology ; Testicular Neoplasms - surgery ; Testis - pathology ; Testis - physiology ; Testis - surgery ; Testosterone - blood ; Treatment Outcome ; Urologic Surgical Procedures, Male - methods ; Vertebrates: endocrinology</subject><ispartof>The journal of clinical endocrinology and metabolism, 2007-02, Vol.92 (2), p.612-615</ispartof><rights>2007 INIST-CNRS</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c401t-1d96e15570faf70718b39946a4164377e2d1ed7fe399ec4147b4ee3042f0d52c3</citedby><cites>FETCH-LOGICAL-c401t-1d96e15570faf70718b39946a4164377e2d1ed7fe399ec4147b4ee3042f0d52c3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,778,782,27911,27912</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=18533906$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/17090637$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Claahsen-van der Grinten, Hedi L.</creatorcontrib><creatorcontrib>Otten, Barto J.</creatorcontrib><creatorcontrib>Takahashi, Satoru</creatorcontrib><creatorcontrib>Meuleman, Eric J. H.</creatorcontrib><creatorcontrib>Hulsbergen-van de Kaa, Christina</creatorcontrib><creatorcontrib>Sweep, Fred C. G. J.</creatorcontrib><creatorcontrib>Hermus, Ad R. M. M.</creatorcontrib><title>Testicular Adrenal Rest Tumors in Adult Males with Congenital Adrenal Hyperplasia: Evaluation of Pituitary-Gonadal Function before and after Successful Testis-Sparing Surgery in Eight Patients</title><title>The journal of clinical endocrinology and metabolism</title><addtitle>J Clin Endocrinol Metab</addtitle><description>Context: In male patients with congenital adrenal hyperplasia (CAH), testicular adrenal rest tumors (TART) are frequently present. These tumors can interfere with testicular function. Intensifying glucocorticoid therapy does not always lead to tumor regression and improvement of testicular function. Recently, testis-sparing surgery was introduced for treatment of TART.
Objective: The aim of this study was to evaluate tumor volume, symptoms, and pituitary-gonadal function in male patients with CAH caused by 21-hydroxylase deficiency and bilateral TART before and after testis-sparing surgery.
Setting: This study was conducted at Radboud University Nijmegen Medical Centre in The Netherlands.
Patients: Eight adult male CAH patients with bilateral TART and infertility were included.
Interventions: Evaluation of testicular magnetic resonance imaging, symptoms, fasting serum concentrations of ACTH, LH, FSH, inhibin B, 17-OH progesterone, androstenedione, testosterone, and estrone, and semen analysis (six of eight patients) was performed before and 6 and 22 months after testis-sparing surgery.
Main Outcome Measures: The main outcome measures were absence of residual tumor and improvement of symptoms and pituitary-gonadal function.
Results: Residual tumors were not found on any of the patients’ magnetic resonance imaging after surgery. Two patients reported testicular pain and discomfort that disappeared after surgery. Parameters of pituitary-gonadal function did not improve after surgery: semen analysis showed azoospermia (five patients) or oligospermia (one patient) without improvement, and all patients had persistently low inhibin B concentrations.
Conclusion: Testis-sparing surgery did not improve pituitary-gonadal function despite successful removal of the tumors. Further studies are needed to investigate whether surgery at an earlier stage in the natural history of TART can prevent permanent testicular damage.</description><subject>17-alpha-Hydroxyprogesterone - blood</subject><subject>Adrenal Hyperplasia, Congenital - pathology</subject><subject>Adrenal Hyperplasia, Congenital - physiopathology</subject><subject>Adrenal Hyperplasia, Congenital - surgery</subject><subject>Adrenal Rest Tumor - pathology</subject><subject>Adrenal Rest Tumor - physiopathology</subject><subject>Adrenal Rest Tumor - surgery</subject><subject>Adrenals. Adrenal axis. Renin-angiotensin system (diseases)</subject><subject>Adrenocorticotropic Hormone - blood</subject><subject>Adult</subject><subject>Androstenedione - blood</subject><subject>Azoospermia - pathology</subject><subject>Azoospermia - physiopathology</subject><subject>Biological and medical sciences</subject><subject>Biopsy</subject><subject>Endocrinopathies</subject><subject>Estrone - blood</subject><subject>Follicle Stimulating Hormone - blood</subject><subject>Fundamental and applied biological sciences. Psychology</subject><subject>Humans</subject><subject>Inhibins - blood</subject><subject>Luteinizing Hormone - blood</subject><subject>Magnetic Resonance Imaging</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Non tumoral diseases. Target tissue resistance. Benign neoplasms</subject><subject>Pituitary Gland - pathology</subject><subject>Pituitary Gland - physiology</subject><subject>Postoperative Complications - physiopathology</subject><subject>Preoperative Care</subject><subject>Testicular Neoplasms - pathology</subject><subject>Testicular Neoplasms - physiopathology</subject><subject>Testicular Neoplasms - surgery</subject><subject>Testis - pathology</subject><subject>Testis - physiology</subject><subject>Testis - surgery</subject><subject>Testosterone - blood</subject><subject>Treatment Outcome</subject><subject>Urologic Surgical Procedures, Male - methods</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>eNptkUFvEzEQhS0EoiFw44x8gRNb7LV3HXOrorRFKqKiQeK2crzj1JFjb-01KP-On4a3CeqFk6U3n2fezEPoLSXntKbk006f14S0FWWUPkMzKnlTCSrFczQjpKaVFPXPM_QqpR0hlPOGvURnVBBJWiZm6M8a0mh1diriiz6CVw5_LxJe532ICVtf5OxG_FU5SPi3He_xMvgteDsW9N-X68MAcXAqWfUZr34pl9Vog8fB4Fs75sLGQ3UVvOoLfJm9fqxuwIQIWPkeKzNCxHdZa0jJZIcffaXqblDR-m2pxC3Ew-RnZbf3I74tA8CP6TV6YZRL8Ob0ztGPy9V6eV3dfLv6sry4qTQndKxoL1ugTSOIUUYQQRcbJiVvFactZ0JA3VPohYGiguaUiw0HYITXhvRNrdkcfTj2HWJ4yMVct7dJg3PKQ8ipaxdS1qKVBfx4BHUMKUUw3RDtvuzfUdJNiXU73U2JdVNiBX936ps3e-if4FNEBXh_AlTSypmovLbpiVs0jE3kHLEjB74PuhwNhlhu2e1CjiWi9P_xfwGNPLLD</recordid><startdate>20070201</startdate><enddate>20070201</enddate><creator>Claahsen-van der Grinten, Hedi L.</creator><creator>Otten, Barto J.</creator><creator>Takahashi, Satoru</creator><creator>Meuleman, Eric J. H.</creator><creator>Hulsbergen-van de Kaa, Christina</creator><creator>Sweep, Fred C. G. J.</creator><creator>Hermus, Ad R. M. M.</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>20070201</creationdate><title>Testicular Adrenal Rest Tumors in Adult Males with Congenital Adrenal Hyperplasia: Evaluation of Pituitary-Gonadal Function before and after Successful Testis-Sparing Surgery in Eight Patients</title><author>Claahsen-van der Grinten, Hedi L. ; Otten, Barto J. ; Takahashi, Satoru ; Meuleman, Eric J. H. ; Hulsbergen-van de Kaa, Christina ; Sweep, Fred C. G. J. ; Hermus, Ad R. M. M.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c401t-1d96e15570faf70718b39946a4164377e2d1ed7fe399ec4147b4ee3042f0d52c3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2007</creationdate><topic>17-alpha-Hydroxyprogesterone - blood</topic><topic>Adrenal Hyperplasia, Congenital - pathology</topic><topic>Adrenal Hyperplasia, Congenital - physiopathology</topic><topic>Adrenal Hyperplasia, Congenital - surgery</topic><topic>Adrenal Rest Tumor - pathology</topic><topic>Adrenal Rest Tumor - physiopathology</topic><topic>Adrenal Rest Tumor - surgery</topic><topic>Adrenals. Adrenal axis. Renin-angiotensin system (diseases)</topic><topic>Adrenocorticotropic Hormone - blood</topic><topic>Adult</topic><topic>Androstenedione - blood</topic><topic>Azoospermia - pathology</topic><topic>Azoospermia - physiopathology</topic><topic>Biological and medical sciences</topic><topic>Biopsy</topic><topic>Endocrinopathies</topic><topic>Estrone - blood</topic><topic>Follicle Stimulating Hormone - blood</topic><topic>Fundamental and applied biological sciences. Psychology</topic><topic>Humans</topic><topic>Inhibins - blood</topic><topic>Luteinizing Hormone - blood</topic><topic>Magnetic Resonance Imaging</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Non tumoral diseases. Target tissue resistance. Benign neoplasms</topic><topic>Pituitary Gland - pathology</topic><topic>Pituitary Gland - physiology</topic><topic>Postoperative Complications - physiopathology</topic><topic>Preoperative Care</topic><topic>Testicular Neoplasms - pathology</topic><topic>Testicular Neoplasms - physiopathology</topic><topic>Testicular Neoplasms - surgery</topic><topic>Testis - pathology</topic><topic>Testis - physiology</topic><topic>Testis - surgery</topic><topic>Testosterone - blood</topic><topic>Treatment Outcome</topic><topic>Urologic Surgical Procedures, Male - methods</topic><topic>Vertebrates: endocrinology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Claahsen-van der Grinten, Hedi L.</creatorcontrib><creatorcontrib>Otten, Barto J.</creatorcontrib><creatorcontrib>Takahashi, Satoru</creatorcontrib><creatorcontrib>Meuleman, Eric J. H.</creatorcontrib><creatorcontrib>Hulsbergen-van de Kaa, Christina</creatorcontrib><creatorcontrib>Sweep, Fred C. G. J.</creatorcontrib><creatorcontrib>Hermus, Ad R. M. M.</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>Claahsen-van der Grinten, Hedi L.</au><au>Otten, Barto J.</au><au>Takahashi, Satoru</au><au>Meuleman, Eric J. H.</au><au>Hulsbergen-van de Kaa, Christina</au><au>Sweep, Fred C. G. J.</au><au>Hermus, Ad R. M. M.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Testicular Adrenal Rest Tumors in Adult Males with Congenital Adrenal Hyperplasia: Evaluation of Pituitary-Gonadal Function before and after Successful Testis-Sparing Surgery in Eight Patients</atitle><jtitle>The journal of clinical endocrinology and metabolism</jtitle><addtitle>J Clin Endocrinol Metab</addtitle><date>2007-02-01</date><risdate>2007</risdate><volume>92</volume><issue>2</issue><spage>612</spage><epage>615</epage><pages>612-615</pages><issn>0021-972X</issn><eissn>1945-7197</eissn><coden>JCEMAZ</coden><abstract>Context: In male patients with congenital adrenal hyperplasia (CAH), testicular adrenal rest tumors (TART) are frequently present. These tumors can interfere with testicular function. Intensifying glucocorticoid therapy does not always lead to tumor regression and improvement of testicular function. Recently, testis-sparing surgery was introduced for treatment of TART.
Objective: The aim of this study was to evaluate tumor volume, symptoms, and pituitary-gonadal function in male patients with CAH caused by 21-hydroxylase deficiency and bilateral TART before and after testis-sparing surgery.
Setting: This study was conducted at Radboud University Nijmegen Medical Centre in The Netherlands.
Patients: Eight adult male CAH patients with bilateral TART and infertility were included.
Interventions: Evaluation of testicular magnetic resonance imaging, symptoms, fasting serum concentrations of ACTH, LH, FSH, inhibin B, 17-OH progesterone, androstenedione, testosterone, and estrone, and semen analysis (six of eight patients) was performed before and 6 and 22 months after testis-sparing surgery.
Main Outcome Measures: The main outcome measures were absence of residual tumor and improvement of symptoms and pituitary-gonadal function.
Results: Residual tumors were not found on any of the patients’ magnetic resonance imaging after surgery. Two patients reported testicular pain and discomfort that disappeared after surgery. Parameters of pituitary-gonadal function did not improve after surgery: semen analysis showed azoospermia (five patients) or oligospermia (one patient) without improvement, and all patients had persistently low inhibin B concentrations.
Conclusion: Testis-sparing surgery did not improve pituitary-gonadal function despite successful removal of the tumors. Further studies are needed to investigate whether surgery at an earlier stage in the natural history of TART can prevent permanent testicular damage.</abstract><cop>Bethesda, MD</cop><pub>Endocrine Society</pub><pmid>17090637</pmid><doi>10.1210/jc.2006-1311</doi><tpages>4</tpages><oa>free_for_read</oa></addata></record> |
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| ispartof | The journal of clinical endocrinology and metabolism, 2007-02, Vol.92 (2), p.612-615 |
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| source | MEDLINE; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; Oxford University Press Journals All Titles (1996-Current) |
| subjects | 17-alpha-Hydroxyprogesterone - blood Adrenal Hyperplasia, Congenital - pathology Adrenal Hyperplasia, Congenital - physiopathology Adrenal Hyperplasia, Congenital - surgery Adrenal Rest Tumor - pathology Adrenal Rest Tumor - physiopathology Adrenal Rest Tumor - surgery Adrenals. Adrenal axis. Renin-angiotensin system (diseases) Adrenocorticotropic Hormone - blood Adult Androstenedione - blood Azoospermia - pathology Azoospermia - physiopathology Biological and medical sciences Biopsy Endocrinopathies Estrone - blood Follicle Stimulating Hormone - blood Fundamental and applied biological sciences. Psychology Humans Inhibins - blood Luteinizing Hormone - blood Magnetic Resonance Imaging Male Medical sciences Non tumoral diseases. Target tissue resistance. Benign neoplasms Pituitary Gland - pathology Pituitary Gland - physiology Postoperative Complications - physiopathology Preoperative Care Testicular Neoplasms - pathology Testicular Neoplasms - physiopathology Testicular Neoplasms - surgery Testis - pathology Testis - physiology Testis - surgery Testosterone - blood Treatment Outcome Urologic Surgical Procedures, Male - methods Vertebrates: endocrinology |
| title | Testicular Adrenal Rest Tumors in Adult Males with Congenital Adrenal Hyperplasia: Evaluation of Pituitary-Gonadal Function before and after Successful Testis-Sparing Surgery in Eight Patients |
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