Severe pulmonary vascular occlusive disease following bone marrow transplantation in Omenn syndrome
A 5-month-old infant presented with severe combined immunodeficiency disease, reticuloendotheliosis, and hypereosinophilia (Omenn syndrome) resulting in recurrent infections and endomyocardial disease. Bone marrow transplantation from an HLA-identical donor after chemotherapeutic conditioning led to...
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| Veröffentlicht in: | European journal of pediatrics 1991-02, Vol.150 (4), p.242-245 |
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| container_end_page | 245 |
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| container_issue | 4 |
| container_start_page | 242 |
| container_title | European journal of pediatrics |
| container_volume | 150 |
| creator | Brückmann, C Lindner, W Roos, R Permanetter, W Haas, R J Haworth, S G Belohradsky, B H |
| description | A 5-month-old infant presented with severe combined immunodeficiency disease, reticuloendotheliosis, and hypereosinophilia (Omenn syndrome) resulting in recurrent infections and endomyocardial disease. Bone marrow transplantation from an HLA-identical donor after chemotherapeutic conditioning led to both immunological and clinical recovery. Bone marrow transplantation, however, was followed by severe pulmonary occlusive disease. The patient gradually recovered while on increased inspiratory oxygen and the calcium channel blocker nifedipine. |
| doi_str_mv | 10.1007/BF01955521 |
| format | Article |
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Bone marrow transplantation from an HLA-identical donor after chemotherapeutic conditioning led to both immunological and clinical recovery. Bone marrow transplantation, however, was followed by severe pulmonary occlusive disease. The patient gradually recovered while on increased inspiratory oxygen and the calcium channel blocker nifedipine.</description><identifier>ISSN: 0340-6199</identifier><identifier>EISSN: 1432-1076</identifier><identifier>DOI: 10.1007/BF01955521</identifier><identifier>PMID: 2029913</identifier><language>eng</language><publisher>Germany</publisher><subject>Arterial Occlusive Diseases - complications ; Arterial Occlusive Diseases - etiology ; Bone Marrow Transplantation - adverse effects ; Eosinophilia - immunology ; Eosinophilia - surgery ; Heart Neoplasms - immunology ; Heart Neoplasms - surgery ; Humans ; Hypertension, Pulmonary - etiology ; Immunologic Deficiency Syndromes - immunology ; Immunologic Deficiency Syndromes - surgery ; Infant ; Lymphatic Diseases - immunology ; Lymphatic Diseases - surgery ; Male ; Pulmonary Artery - pathology</subject><ispartof>European journal of pediatrics, 1991-02, Vol.150 (4), p.242-245</ispartof><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c282t-7d9e65cd35e32904405d256f0a60132003aacbfa9d284d18d5d72a7af5356ae93</citedby><cites>FETCH-LOGICAL-c282t-7d9e65cd35e32904405d256f0a60132003aacbfa9d284d18d5d72a7af5356ae93</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/2029913$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Brückmann, C</creatorcontrib><creatorcontrib>Lindner, W</creatorcontrib><creatorcontrib>Roos, R</creatorcontrib><creatorcontrib>Permanetter, W</creatorcontrib><creatorcontrib>Haas, R J</creatorcontrib><creatorcontrib>Haworth, S G</creatorcontrib><creatorcontrib>Belohradsky, B H</creatorcontrib><title>Severe pulmonary vascular occlusive disease following bone marrow transplantation in Omenn syndrome</title><title>European journal of pediatrics</title><addtitle>Eur J Pediatr</addtitle><description>A 5-month-old infant presented with severe combined immunodeficiency disease, reticuloendotheliosis, and hypereosinophilia (Omenn syndrome) resulting in recurrent infections and endomyocardial disease. Bone marrow transplantation from an HLA-identical donor after chemotherapeutic conditioning led to both immunological and clinical recovery. Bone marrow transplantation, however, was followed by severe pulmonary occlusive disease. The patient gradually recovered while on increased inspiratory oxygen and the calcium channel blocker nifedipine.</description><subject>Arterial Occlusive Diseases - complications</subject><subject>Arterial Occlusive Diseases - etiology</subject><subject>Bone Marrow Transplantation - adverse effects</subject><subject>Eosinophilia - immunology</subject><subject>Eosinophilia - surgery</subject><subject>Heart Neoplasms - immunology</subject><subject>Heart Neoplasms - surgery</subject><subject>Humans</subject><subject>Hypertension, Pulmonary - etiology</subject><subject>Immunologic Deficiency Syndromes - immunology</subject><subject>Immunologic Deficiency Syndromes - surgery</subject><subject>Infant</subject><subject>Lymphatic Diseases - immunology</subject><subject>Lymphatic Diseases - surgery</subject><subject>Male</subject><subject>Pulmonary Artery - pathology</subject><issn>0340-6199</issn><issn>1432-1076</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>1991</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpFkM9LwzAUx4Moc04v3oWcPAjVl6Rpm6MOf8FgB_VcsuRVKmkyk3Zj_72VDT29y4cP3_ch5JLBLQMo7x6egCkpJWdHZMpywTMGZXFMpiByyAqm1Ck5S-kLRlixakImHLhSTEyJecMNRqTrwXXB67ijG53M4HSkwRg3pHaD1LYJdULaBOfCtvWfdBU80k7HGLa0j9qntdO-130bPG09XXboPU07b2Po8JycNNolvDjcGfl4enyfv2SL5fPr_H6RGV7xPiutwkIaKyQKriDPQVouiwZ0AUxwAKG1WTVaWV7lllVW2pLrUjdSyEKjEjNyvfeuY_geMPV11yaDbpyGYUh1BVLmo2kEb_agiSGliE29ju34za5mUP8Wrf-LjvDVwTqsOrR_6CGh-AFornIT</recordid><startdate>19910201</startdate><enddate>19910201</enddate><creator>Brückmann, C</creator><creator>Lindner, W</creator><creator>Roos, R</creator><creator>Permanetter, W</creator><creator>Haas, R J</creator><creator>Haworth, S G</creator><creator>Belohradsky, B H</creator><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>19910201</creationdate><title>Severe pulmonary vascular occlusive disease following bone marrow transplantation in Omenn syndrome</title><author>Brückmann, C ; Lindner, W ; Roos, R ; Permanetter, W ; Haas, R J ; Haworth, S G ; Belohradsky, B H</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c282t-7d9e65cd35e32904405d256f0a60132003aacbfa9d284d18d5d72a7af5356ae93</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>1991</creationdate><topic>Arterial Occlusive Diseases - complications</topic><topic>Arterial Occlusive Diseases - etiology</topic><topic>Bone Marrow Transplantation - adverse effects</topic><topic>Eosinophilia - immunology</topic><topic>Eosinophilia - surgery</topic><topic>Heart Neoplasms - immunology</topic><topic>Heart Neoplasms - surgery</topic><topic>Humans</topic><topic>Hypertension, Pulmonary - etiology</topic><topic>Immunologic Deficiency Syndromes - immunology</topic><topic>Immunologic Deficiency Syndromes - surgery</topic><topic>Infant</topic><topic>Lymphatic Diseases - immunology</topic><topic>Lymphatic Diseases - surgery</topic><topic>Male</topic><topic>Pulmonary Artery - pathology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Brückmann, C</creatorcontrib><creatorcontrib>Lindner, W</creatorcontrib><creatorcontrib>Roos, R</creatorcontrib><creatorcontrib>Permanetter, W</creatorcontrib><creatorcontrib>Haas, R J</creatorcontrib><creatorcontrib>Haworth, S G</creatorcontrib><creatorcontrib>Belohradsky, B H</creatorcontrib><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>European journal of pediatrics</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Brückmann, C</au><au>Lindner, W</au><au>Roos, R</au><au>Permanetter, W</au><au>Haas, R J</au><au>Haworth, S G</au><au>Belohradsky, B H</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Severe pulmonary vascular occlusive disease following bone marrow transplantation in Omenn syndrome</atitle><jtitle>European journal of pediatrics</jtitle><addtitle>Eur J Pediatr</addtitle><date>1991-02-01</date><risdate>1991</risdate><volume>150</volume><issue>4</issue><spage>242</spage><epage>245</epage><pages>242-245</pages><issn>0340-6199</issn><eissn>1432-1076</eissn><abstract>A 5-month-old infant presented with severe combined immunodeficiency disease, reticuloendotheliosis, and hypereosinophilia (Omenn syndrome) resulting in recurrent infections and endomyocardial disease. Bone marrow transplantation from an HLA-identical donor after chemotherapeutic conditioning led to both immunological and clinical recovery. Bone marrow transplantation, however, was followed by severe pulmonary occlusive disease. The patient gradually recovered while on increased inspiratory oxygen and the calcium channel blocker nifedipine.</abstract><cop>Germany</cop><pmid>2029913</pmid><doi>10.1007/BF01955521</doi><tpages>4</tpages></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 0340-6199 |
| ispartof | European journal of pediatrics, 1991-02, Vol.150 (4), p.242-245 |
| issn | 0340-6199 1432-1076 |
| language | eng |
| recordid | cdi_proquest_miscellaneous_80554132 |
| source | MEDLINE; SpringerLink Journals - AutoHoldings |
| subjects | Arterial Occlusive Diseases - complications Arterial Occlusive Diseases - etiology Bone Marrow Transplantation - adverse effects Eosinophilia - immunology Eosinophilia - surgery Heart Neoplasms - immunology Heart Neoplasms - surgery Humans Hypertension, Pulmonary - etiology Immunologic Deficiency Syndromes - immunology Immunologic Deficiency Syndromes - surgery Infant Lymphatic Diseases - immunology Lymphatic Diseases - surgery Male Pulmonary Artery - pathology |
| title | Severe pulmonary vascular occlusive disease following bone marrow transplantation in Omenn syndrome |
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