Langerhans cell histiocytosis with multifocal bone lesions: comparative clinical features between single and multi-systems
Langerhans cell histiocytosis (LCH) can be a single system or multi-system disease. Both disease types can be associated with multi-focal bone lesions, but their bone involvement patterns have not been compared systematically. Of the new pediatric LCH cases enrolled into the JLSG-02 study during 200...
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| Veröffentlicht in: | International journal of hematology 2009-11, Vol.90 (4), p.506-512 |
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| creator | Imashuku, Shinsaku Kinugawa, Naoko Matsuzaki, Akinobu Kitoh, Toshiyuki Ohki, Kentaro Shioda, Yoko Tsunematsu, Yukiko Imamura, Toshihiko Morimoto, Akira |
| description | Langerhans cell histiocytosis (LCH) can be a single system or multi-system disease. Both disease types can be associated with multi-focal bone lesions, but their bone involvement patterns have not been compared systematically. Of the new pediatric LCH cases enrolled into the JLSG-02 study during 2002–2007, 67 cases of single system multifocal bone (SMFB) LCH and 97 cases of multi-system bone (MSB) LCH were analyzed to determine if the bone involvement patterns differ in these two types, and whether these differences correlate with outcome. Statistical analysis was performed with Mann–Whitney
U
test, Fisher’s exact test, and other measures. Onset ages were higher for SMFB (
P
< 0.001), but the two types did not differ in the number of bone lesions per patient. The skull was most frequently affected in both types, followed by the spine. Lesions in the temporal bone (
P
= 0.002), ear-petrous bone (
P
< 0.001), orbita (
P
= 0.003), and zygomatic bone (
P
= 0.016) were significantly more common in MSB. The two types did not differ in response to treatment, but MSB was associated with a significantly higher incidence of diabetes insipidus (DI) (
P
< 0.001). Novel measures are required in preventing the development of DI in MSB-type LCH patients with “risk” bone lesions. |
| doi_str_mv | 10.1007/s12185-009-0420-4 |
| format | Article |
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U
test, Fisher’s exact test, and other measures. Onset ages were higher for SMFB (
P
< 0.001), but the two types did not differ in the number of bone lesions per patient. The skull was most frequently affected in both types, followed by the spine. Lesions in the temporal bone (
P
= 0.002), ear-petrous bone (
P
< 0.001), orbita (
P
= 0.003), and zygomatic bone (
P
= 0.016) were significantly more common in MSB. The two types did not differ in response to treatment, but MSB was associated with a significantly higher incidence of diabetes insipidus (DI) (
P
< 0.001). Novel measures are required in preventing the development of DI in MSB-type LCH patients with “risk” bone lesions.</description><identifier>ISSN: 0925-5710</identifier><identifier>EISSN: 1865-3774</identifier><identifier>DOI: 10.1007/s12185-009-0420-4</identifier><identifier>PMID: 19779766</identifier><language>eng</language><publisher>Tokyo: Springer Japan</publisher><subject>Adolescent ; Age of Onset ; Biological and medical sciences ; Bone Diseases - drug therapy ; Bone Diseases - physiopathology ; Child ; Child, Preschool ; Cohort Studies ; Diabetes Insipidus - complications ; Drug Therapy, Combination ; Endocrinopathies ; Female ; Hematologic and hematopoietic diseases ; Hematology ; Histiocytosis, Langerhans-Cell - classification ; Histiocytosis, Langerhans-Cell - complications ; Histiocytosis, Langerhans-Cell - drug therapy ; Histiocytosis, Langerhans-Cell - physiopathology ; Humans ; Hypothalamus. Hypophysis. Epiphysis (diseases) ; Infant ; Male ; Medical sciences ; Medicine ; Medicine & Public Health ; Non tumoral diseases. Target tissue resistance. Benign neoplasms ; Oncology ; Original Article ; Other diseases. Hematologic involvement in other diseases ; Survival Analysis</subject><ispartof>International journal of hematology, 2009-11, Vol.90 (4), p.506-512</ispartof><rights>The Japanese Society of Hematology 2009</rights><rights>2015 INIST-CNRS</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c549t-9812a98ff94379a89b31ce1945f4f75c0e627c2188e462c84b404216765ce6363</citedby><cites>FETCH-LOGICAL-c549t-9812a98ff94379a89b31ce1945f4f75c0e627c2188e462c84b404216765ce6363</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s12185-009-0420-4$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s12185-009-0420-4$$EHTML$$P50$$Gspringer$$H</linktohtml><link.rule.ids>314,776,780,27901,27902,41464,42533,51294</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=22235339$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/19779766$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Imashuku, Shinsaku</creatorcontrib><creatorcontrib>Kinugawa, Naoko</creatorcontrib><creatorcontrib>Matsuzaki, Akinobu</creatorcontrib><creatorcontrib>Kitoh, Toshiyuki</creatorcontrib><creatorcontrib>Ohki, Kentaro</creatorcontrib><creatorcontrib>Shioda, Yoko</creatorcontrib><creatorcontrib>Tsunematsu, Yukiko</creatorcontrib><creatorcontrib>Imamura, Toshihiko</creatorcontrib><creatorcontrib>Morimoto, Akira</creatorcontrib><creatorcontrib>Japan LCH Study Group</creatorcontrib><title>Langerhans cell histiocytosis with multifocal bone lesions: comparative clinical features between single and multi-systems</title><title>International journal of hematology</title><addtitle>Int J Hematol</addtitle><addtitle>Int J Hematol</addtitle><description>Langerhans cell histiocytosis (LCH) can be a single system or multi-system disease. Both disease types can be associated with multi-focal bone lesions, but their bone involvement patterns have not been compared systematically. Of the new pediatric LCH cases enrolled into the JLSG-02 study during 2002–2007, 67 cases of single system multifocal bone (SMFB) LCH and 97 cases of multi-system bone (MSB) LCH were analyzed to determine if the bone involvement patterns differ in these two types, and whether these differences correlate with outcome. Statistical analysis was performed with Mann–Whitney
U
test, Fisher’s exact test, and other measures. Onset ages were higher for SMFB (
P
< 0.001), but the two types did not differ in the number of bone lesions per patient. The skull was most frequently affected in both types, followed by the spine. Lesions in the temporal bone (
P
= 0.002), ear-petrous bone (
P
< 0.001), orbita (
P
= 0.003), and zygomatic bone (
P
= 0.016) were significantly more common in MSB. The two types did not differ in response to treatment, but MSB was associated with a significantly higher incidence of diabetes insipidus (DI) (
P
< 0.001). Novel measures are required in preventing the development of DI in MSB-type LCH patients with “risk” bone lesions.</description><subject>Adolescent</subject><subject>Age of Onset</subject><subject>Biological and medical sciences</subject><subject>Bone Diseases - drug therapy</subject><subject>Bone Diseases - physiopathology</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Cohort Studies</subject><subject>Diabetes Insipidus - complications</subject><subject>Drug Therapy, Combination</subject><subject>Endocrinopathies</subject><subject>Female</subject><subject>Hematologic and hematopoietic diseases</subject><subject>Hematology</subject><subject>Histiocytosis, Langerhans-Cell - classification</subject><subject>Histiocytosis, Langerhans-Cell - complications</subject><subject>Histiocytosis, Langerhans-Cell - drug therapy</subject><subject>Histiocytosis, Langerhans-Cell - physiopathology</subject><subject>Humans</subject><subject>Hypothalamus. Hypophysis. Epiphysis (diseases)</subject><subject>Infant</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Non tumoral diseases. Target tissue resistance. Benign neoplasms</subject><subject>Oncology</subject><subject>Original Article</subject><subject>Other diseases. 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Hypophysis. Epiphysis (diseases)</topic><topic>Infant</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Non tumoral diseases. Target tissue resistance. Benign neoplasms</topic><topic>Oncology</topic><topic>Original Article</topic><topic>Other diseases. Hematologic involvement in other diseases</topic><topic>Survival Analysis</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Imashuku, Shinsaku</creatorcontrib><creatorcontrib>Kinugawa, Naoko</creatorcontrib><creatorcontrib>Matsuzaki, Akinobu</creatorcontrib><creatorcontrib>Kitoh, Toshiyuki</creatorcontrib><creatorcontrib>Ohki, Kentaro</creatorcontrib><creatorcontrib>Shioda, Yoko</creatorcontrib><creatorcontrib>Tsunematsu, Yukiko</creatorcontrib><creatorcontrib>Imamura, Toshihiko</creatorcontrib><creatorcontrib>Morimoto, Akira</creatorcontrib><creatorcontrib>Japan 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>ProQuest Central (Corporate)</collection><collection>Nursing & Allied Health Database</collection><collection>Immunology Abstracts</collection><collection>Industrial and Applied Microbiology Abstracts (Microbiology A)</collection><collection>Nucleic Acids Abstracts</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Technology Research Database</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central</collection><collection>Environmental Sciences and Pollution Management</collection><collection>ProQuest One Community College</collection><collection>Engineering Research Database</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Nursing & Allied Health Database (Alumni Edition)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Nursing & Allied Health Premium</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><collection>Calcium & Calcified Tissue Abstracts</collection><jtitle>International journal of hematology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Imashuku, Shinsaku</au><au>Kinugawa, Naoko</au><au>Matsuzaki, Akinobu</au><au>Kitoh, Toshiyuki</au><au>Ohki, Kentaro</au><au>Shioda, Yoko</au><au>Tsunematsu, Yukiko</au><au>Imamura, Toshihiko</au><au>Morimoto, Akira</au><aucorp>Japan LCH Study Group</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Langerhans cell histiocytosis with multifocal bone lesions: comparative clinical features between single and multi-systems</atitle><jtitle>International journal of hematology</jtitle><stitle>Int J Hematol</stitle><addtitle>Int J Hematol</addtitle><date>2009-11-01</date><risdate>2009</risdate><volume>90</volume><issue>4</issue><spage>506</spage><epage>512</epage><pages>506-512</pages><issn>0925-5710</issn><eissn>1865-3774</eissn><abstract>Langerhans cell histiocytosis (LCH) can be a single system or multi-system disease. Both disease types can be associated with multi-focal bone lesions, but their bone involvement patterns have not been compared systematically. Of the new pediatric LCH cases enrolled into the JLSG-02 study during 2002–2007, 67 cases of single system multifocal bone (SMFB) LCH and 97 cases of multi-system bone (MSB) LCH were analyzed to determine if the bone involvement patterns differ in these two types, and whether these differences correlate with outcome. Statistical analysis was performed with Mann–Whitney
U
test, Fisher’s exact test, and other measures. Onset ages were higher for SMFB (
P
< 0.001), but the two types did not differ in the number of bone lesions per patient. The skull was most frequently affected in both types, followed by the spine. Lesions in the temporal bone (
P
= 0.002), ear-petrous bone (
P
< 0.001), orbita (
P
= 0.003), and zygomatic bone (
P
= 0.016) were significantly more common in MSB. The two types did not differ in response to treatment, but MSB was associated with a significantly higher incidence of diabetes insipidus (DI) (
P
< 0.001). Novel measures are required in preventing the development of DI in MSB-type LCH patients with “risk” bone lesions.</abstract><cop>Tokyo</cop><pub>Springer Japan</pub><pmid>19779766</pmid><doi>10.1007/s12185-009-0420-4</doi><tpages>7</tpages></addata></record> |
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| ispartof | International journal of hematology, 2009-11, Vol.90 (4), p.506-512 |
| issn | 0925-5710 1865-3774 |
| language | eng |
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| subjects | Adolescent Age of Onset Biological and medical sciences Bone Diseases - drug therapy Bone Diseases - physiopathology Child Child, Preschool Cohort Studies Diabetes Insipidus - complications Drug Therapy, Combination Endocrinopathies Female Hematologic and hematopoietic diseases Hematology Histiocytosis, Langerhans-Cell - classification Histiocytosis, Langerhans-Cell - complications Histiocytosis, Langerhans-Cell - drug therapy Histiocytosis, Langerhans-Cell - physiopathology Humans Hypothalamus. Hypophysis. Epiphysis (diseases) Infant Male Medical sciences Medicine Medicine & Public Health Non tumoral diseases. Target tissue resistance. Benign neoplasms Oncology Original Article Other diseases. Hematologic involvement in other diseases Survival Analysis |
| title | Langerhans cell histiocytosis with multifocal bone lesions: comparative clinical features between single and multi-systems |
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