Incidence of Growth Hormone Deficiency in Pediatric-Onset Langerhans Cell Histiocytosis: Efficacy and Safety of Growth Hormone Treatment
We retrospectively studied 61 patients with GH deficiency (GHD), identified among 589 patients with Langerhans cell histiocytosis (LCH) enrolled in a nationwide survey between 1993 and 2001. Overall, 141 patients in the survey developed diabetes insipidus. The median follow-up of the 61 patients wit...
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| Veröffentlicht in: | The journal of clinical endocrinology and metabolism 2004-02, Vol.89 (2), p.604-609 |
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| creator | Donadieu, Jean Rolon, Maria-Alejandra Pion, Isabelle Thomas, Caroline Doz, François Barkaoui, Mohamed Robert, Alain Deville, Anne Mazingue, Françoise David, Michel Brauner, Raja Cabrol, Sylvie Garel, Catherine Polak, Michel |
| description | We retrospectively studied 61 patients with GH deficiency (GHD), identified among 589 patients with Langerhans cell histiocytosis (LCH) enrolled in a nationwide survey between 1993 and 2001. Overall, 141 patients in the survey developed diabetes insipidus. The median follow-up of the 61 patients with GHD was 12 yr. The 5- and 10-yr risks of GHD among patients with diabetes insipidus were 34.7 ± 4.5% and 53.7 ± 5.2%, respectively. Growth velocity decreased soon after LCH diagnosis in patients who developed GHD, and anterior pituitary height, estimated by magnetic resonance imaging, was significantly reduced relative to patients who remained free of GHD. GH replacement therapy was administered to 47 of the 61 patients with GHD. Among GH-treated patients, median final height (−0.8 sd) was significantly greater than median height at GHD diagnosis (−1.6 sd) but remained below midparental (target) height. Among patients with pituitary involvement, the number of LCH disease episodes appeared not significantly influenced by GHD or GH administration, suggesting an absence of deleterious effect of GH therapy on LCH disease activity. |
| doi_str_mv | 10.1210/jc.2003-030907 |
| format | Article |
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Overall, 141 patients in the survey developed diabetes insipidus. The median follow-up of the 61 patients with GHD was 12 yr. The 5- and 10-yr risks of GHD among patients with diabetes insipidus were 34.7 ± 4.5% and 53.7 ± 5.2%, respectively. Growth velocity decreased soon after LCH diagnosis in patients who developed GHD, and anterior pituitary height, estimated by magnetic resonance imaging, was significantly reduced relative to patients who remained free of GHD. GH replacement therapy was administered to 47 of the 61 patients with GHD. Among GH-treated patients, median final height (−0.8 sd) was significantly greater than median height at GHD diagnosis (−1.6 sd) but remained below midparental (target) height. Among patients with pituitary involvement, the number of LCH disease episodes appeared not significantly influenced by GHD or GH administration, suggesting an absence of deleterious effect of GH therapy on LCH disease activity.</description><identifier>ISSN: 0021-972X</identifier><identifier>EISSN: 1945-7197</identifier><identifier>DOI: 10.1210/jc.2003-030907</identifier><identifier>PMID: 14764769</identifier><identifier>CODEN: JCEMAZ</identifier><language>eng</language><publisher>Bethesda, MD: Endocrine Society</publisher><subject>Adolescent ; Age of Onset ; Biological and medical sciences ; Child ; Endocrine System Diseases - complications ; Endocrinopathies ; Female ; Fundamental and applied biological sciences. 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Among patients with pituitary involvement, the number of LCH disease episodes appeared not significantly influenced by GHD or GH administration, suggesting an absence of deleterious effect of GH therapy on LCH disease activity.</description><subject>Adolescent</subject><subject>Age of Onset</subject><subject>Biological and medical sciences</subject><subject>Child</subject><subject>Endocrine System Diseases - complications</subject><subject>Endocrinopathies</subject><subject>Female</subject><subject>Fundamental and applied biological sciences. Psychology</subject><subject>Growth - drug effects</subject><subject>Histiocytosis, Langerhans-Cell - complications</subject><subject>Histiocytosis, Langerhans-Cell - epidemiology</subject><subject>Human Growth Hormone - adverse effects</subject><subject>Human Growth Hormone - deficiency</subject><subject>Human Growth Hormone - therapeutic use</subject><subject>Humans</subject><subject>Incidence</subject><subject>Magnetic Resonance Imaging</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Pituitary Diseases - drug therapy</subject><subject>Pituitary Diseases - epidemiology</subject><subject>Pituitary Diseases - etiology</subject><subject>Pituitary Gland, Anterior</subject><subject>Retrospective Studies</subject><subject>Risk Factors</subject><subject>Treatment Outcome</subject><subject>Vertebrates: endocrinology</subject><issn>0021-972X</issn><issn>1945-7197</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2004</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp1kM-LEzEUx4Mobl29epRc9Db1JZN0ZrxJd90uFFZwBW8hzbzY1GlSkwyl_4F_tilT2IuG_Djk833v8SHkLYM54ww-7sycA9QV1NBB84zMWCdk1bCueU5mAJxVXcN_XJFXKe0AmBCyfkmumGgWZXcz8ufeG9ejN0iDpXcxHPOWrkLcB4_0Bq0zrnyeqPP0K_ZO5-hM9eATZrrW_ifGrfaJLnEY6Mql7II55ZBc-kRvbQnrEtW-p9-0xXz6R4vHiDrv0efX5IXVQ8I3l_eafP9y-7hcVeuHu_vl53VlJAdRScnbjoFtpGEgkFnBYdPVNVgAzXtp-gZlzwXnC8Y7W7O2M4I1QlvN7QYW9TX5MNU9xPB7xJTV3iVT5tcew5hUC0xyIaGA8wk0MaQU0apDdHsdT4qBOrtXO6PO7tXkvgTeXSqPmz32T_hFdgHeXwCdjB5s1MV9euKkaDkTZ05M3DEMGWP6NYxHjGqLeshbBWWJRdNWpbeAckFVDhclJqcY-j6Y6DweIqakdmGMvij939x_AZwHrYs</recordid><startdate>200402</startdate><enddate>200402</enddate><creator>Donadieu, Jean</creator><creator>Rolon, Maria-Alejandra</creator><creator>Pion, Isabelle</creator><creator>Thomas, Caroline</creator><creator>Doz, François</creator><creator>Barkaoui, Mohamed</creator><creator>Robert, Alain</creator><creator>Deville, Anne</creator><creator>Mazingue, Françoise</creator><creator>David, Michel</creator><creator>Brauner, Raja</creator><creator>Cabrol, Sylvie</creator><creator>Garel, Catherine</creator><creator>Polak, Michel</creator><general>Endocrine Society</general><general>Copyright by The 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>200402</creationdate><title>Incidence of Growth Hormone Deficiency in Pediatric-Onset Langerhans Cell Histiocytosis: Efficacy and Safety of Growth Hormone Treatment</title><author>Donadieu, Jean ; Rolon, Maria-Alejandra ; Pion, Isabelle ; Thomas, Caroline ; Doz, François ; Barkaoui, Mohamed ; Robert, Alain ; Deville, Anne ; Mazingue, Françoise ; David, Michel ; Brauner, Raja ; Cabrol, Sylvie ; Garel, Catherine ; Polak, Michel</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c5204-5528910f75c104e1f420b9330f00a2d5cd7e5d24226129f3189c4174afa2fb063</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2004</creationdate><topic>Adolescent</topic><topic>Age of Onset</topic><topic>Biological and medical sciences</topic><topic>Child</topic><topic>Endocrine System Diseases - complications</topic><topic>Endocrinopathies</topic><topic>Female</topic><topic>Fundamental and applied biological sciences. Psychology</topic><topic>Growth - drug effects</topic><topic>Histiocytosis, Langerhans-Cell - complications</topic><topic>Histiocytosis, Langerhans-Cell - epidemiology</topic><topic>Human Growth Hormone - adverse effects</topic><topic>Human Growth Hormone - deficiency</topic><topic>Human Growth Hormone - therapeutic use</topic><topic>Humans</topic><topic>Incidence</topic><topic>Magnetic Resonance Imaging</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Pituitary Diseases - drug therapy</topic><topic>Pituitary Diseases - epidemiology</topic><topic>Pituitary Diseases - etiology</topic><topic>Pituitary Gland, Anterior</topic><topic>Retrospective Studies</topic><topic>Risk Factors</topic><topic>Treatment Outcome</topic><topic>Vertebrates: endocrinology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Donadieu, Jean</creatorcontrib><creatorcontrib>Rolon, Maria-Alejandra</creatorcontrib><creatorcontrib>Pion, Isabelle</creatorcontrib><creatorcontrib>Thomas, Caroline</creatorcontrib><creatorcontrib>Doz, François</creatorcontrib><creatorcontrib>Barkaoui, Mohamed</creatorcontrib><creatorcontrib>Robert, Alain</creatorcontrib><creatorcontrib>Deville, Anne</creatorcontrib><creatorcontrib>Mazingue, Françoise</creatorcontrib><creatorcontrib>David, Michel</creatorcontrib><creatorcontrib>Brauner, Raja</creatorcontrib><creatorcontrib>Cabrol, Sylvie</creatorcontrib><creatorcontrib>Garel, Catherine</creatorcontrib><creatorcontrib>Polak, Michel</creatorcontrib><creatorcontrib>French LCH 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>Donadieu, Jean</au><au>Rolon, Maria-Alejandra</au><au>Pion, Isabelle</au><au>Thomas, Caroline</au><au>Doz, François</au><au>Barkaoui, Mohamed</au><au>Robert, Alain</au><au>Deville, Anne</au><au>Mazingue, Françoise</au><au>David, Michel</au><au>Brauner, Raja</au><au>Cabrol, Sylvie</au><au>Garel, Catherine</au><au>Polak, Michel</au><aucorp>French LCH Study Group</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Incidence of Growth Hormone Deficiency in Pediatric-Onset Langerhans Cell Histiocytosis: Efficacy and Safety of Growth Hormone Treatment</atitle><jtitle>The journal of clinical endocrinology and metabolism</jtitle><addtitle>J Clin Endocrinol Metab</addtitle><date>2004-02</date><risdate>2004</risdate><volume>89</volume><issue>2</issue><spage>604</spage><epage>609</epage><pages>604-609</pages><issn>0021-972X</issn><eissn>1945-7197</eissn><coden>JCEMAZ</coden><abstract>We retrospectively studied 61 patients with GH deficiency (GHD), identified among 589 patients with Langerhans cell histiocytosis (LCH) enrolled in a nationwide survey between 1993 and 2001. Overall, 141 patients in the survey developed diabetes insipidus. The median follow-up of the 61 patients with GHD was 12 yr. The 5- and 10-yr risks of GHD among patients with diabetes insipidus were 34.7 ± 4.5% and 53.7 ± 5.2%, respectively. Growth velocity decreased soon after LCH diagnosis in patients who developed GHD, and anterior pituitary height, estimated by magnetic resonance imaging, was significantly reduced relative to patients who remained free of GHD. GH replacement therapy was administered to 47 of the 61 patients with GHD. Among GH-treated patients, median final height (−0.8 sd) was significantly greater than median height at GHD diagnosis (−1.6 sd) but remained below midparental (target) height. Among patients with pituitary involvement, the number of LCH disease episodes appeared not significantly influenced by GHD or GH administration, suggesting an absence of deleterious effect of GH therapy on LCH disease activity.</abstract><cop>Bethesda, MD</cop><pub>Endocrine Society</pub><pmid>14764769</pmid><doi>10.1210/jc.2003-030907</doi><tpages>6</tpages><oa>free_for_read</oa></addata></record> |
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| subjects | Adolescent Age of Onset Biological and medical sciences Child Endocrine System Diseases - complications Endocrinopathies Female Fundamental and applied biological sciences. Psychology Growth - drug effects Histiocytosis, Langerhans-Cell - complications Histiocytosis, Langerhans-Cell - epidemiology Human Growth Hormone - adverse effects Human Growth Hormone - deficiency Human Growth Hormone - therapeutic use Humans Incidence Magnetic Resonance Imaging Male Medical sciences Pituitary Diseases - drug therapy Pituitary Diseases - epidemiology Pituitary Diseases - etiology Pituitary Gland, Anterior Retrospective Studies Risk Factors Treatment Outcome Vertebrates: endocrinology |
| title | Incidence of Growth Hormone Deficiency in Pediatric-Onset Langerhans Cell Histiocytosis: Efficacy and Safety of Growth Hormone Treatment |
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