Pulmonary hemorrhage associated with celiac disease
Idiopathic pulmonary hemosiderosis is a severe and potentially fatal disease characterized by recurrent episodes of alveolar hemorrhage, hemoptysis, and anemia. His association with celiac disease, described as Lane- Hamilton syndrome, could be due to the fact that both entities share a common patho...
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Veröffentlicht in: | Archivos argentinos de pediatría 2012-08, Vol.110 (4), p.e72-e76 |
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description | Idiopathic pulmonary hemosiderosis is a severe and potentially fatal disease characterized by recurrent episodes of alveolar hemorrhage, hemoptysis, and anemia. His association with celiac disease, described as Lane- Hamilton syndrome, could be due to the fact that both entities share a common pathogenic immune pathway. We report two patients of 13 years who consulted for hemoptysis and severe anemia that had not responded to immunosuppressive treatment with pulses of methyl prednisolone, oral meprednisone and hydroxychloroquine. Although both children highlight the absence of gastrointestinal symptoms at the time of consultation, the dosage of anti-endomysial and anti-transglutaminase antibodies was positive and biopsy confirmed the presence of intestinal enteropathy. It is emphasized that in patients with diffuse alveolar hemorrhage, even in the absence of gastrointestinal symptoms, the concomitant presence of celiac disease should be evaluated. If celiac disease is present, the incorporation of a gluten-free diet helps to control the symptoms, allows reducing the immunosuppressive treatment and improves the clinical course of both entities. |
doi_str_mv | 10.1590/S0325-00752012000400016 |
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His association with celiac disease, described as Lane- Hamilton syndrome, could be due to the fact that both entities share a common pathogenic immune pathway. We report two patients of 13 years who consulted for hemoptysis and severe anemia that had not responded to immunosuppressive treatment with pulses of methyl prednisolone, oral meprednisone and hydroxychloroquine. Although both children highlight the absence of gastrointestinal symptoms at the time of consultation, the dosage of anti-endomysial and anti-transglutaminase antibodies was positive and biopsy confirmed the presence of intestinal enteropathy. It is emphasized that in patients with diffuse alveolar hemorrhage, even in the absence of gastrointestinal symptoms, the concomitant presence of celiac disease should be evaluated. If celiac disease is present, the incorporation of a gluten-free diet helps to control the symptoms, allows reducing the immunosuppressive treatment and improves the clinical course of both entities.</description><identifier>EISSN: 1668-3501</identifier><identifier>DOI: 10.1590/S0325-00752012000400016</identifier><identifier>PMID: 22859336</identifier><language>spa</language><publisher>Argentina</publisher><subject>Adolescent ; Anemia, Iron-Deficiency - etiology ; Celiac Disease - diagnosis ; Hemoptysis - etiology ; Hemorrhage - diagnosis ; Hemosiderosis - diagnosis ; Hemosiderosis, Pulmonary ; Humans ; Lung Diseases - diagnosis ; Male ; Syndrome</subject><ispartof>Archivos argentinos de pediatría, 2012-08, Vol.110 (4), p.e72-e76</ispartof><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,776,780,27903,27904</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/22859336$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Testa, María Eugenia</creatorcontrib><creatorcontrib>Maffey, Alberto</creatorcontrib><creatorcontrib>Colom, Alejandro</creatorcontrib><creatorcontrib>Agüero, Luis</creatorcontrib><creatorcontrib>Rogé, Ignacio</creatorcontrib><creatorcontrib>Andrewartha, María Sol</creatorcontrib><creatorcontrib>Teper, Alejandro</creatorcontrib><title>Pulmonary hemorrhage associated with celiac disease</title><title>Archivos argentinos de pediatría</title><addtitle>Arch Argent Pediatr</addtitle><description>Idiopathic pulmonary hemosiderosis is a severe and potentially fatal disease characterized by recurrent episodes of alveolar hemorrhage, hemoptysis, and anemia. His association with celiac disease, described as Lane- Hamilton syndrome, could be due to the fact that both entities share a common pathogenic immune pathway. We report two patients of 13 years who consulted for hemoptysis and severe anemia that had not responded to immunosuppressive treatment with pulses of methyl prednisolone, oral meprednisone and hydroxychloroquine. Although both children highlight the absence of gastrointestinal symptoms at the time of consultation, the dosage of anti-endomysial and anti-transglutaminase antibodies was positive and biopsy confirmed the presence of intestinal enteropathy. It is emphasized that in patients with diffuse alveolar hemorrhage, even in the absence of gastrointestinal symptoms, the concomitant presence of celiac disease should be evaluated. If celiac disease is present, the incorporation of a gluten-free diet helps to control the symptoms, allows reducing the immunosuppressive treatment and improves the clinical course of both entities.</description><subject>Adolescent</subject><subject>Anemia, Iron-Deficiency - etiology</subject><subject>Celiac Disease - diagnosis</subject><subject>Hemoptysis - etiology</subject><subject>Hemorrhage - diagnosis</subject><subject>Hemosiderosis - diagnosis</subject><subject>Hemosiderosis, Pulmonary</subject><subject>Humans</subject><subject>Lung Diseases - diagnosis</subject><subject>Male</subject><subject>Syndrome</subject><issn>1668-3501</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2012</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNo1j0FLxDAQhYMg7rr6F7RHL9WZpEnboyy6CgsK7r0kk9FG2m1ttoj_3oDr4THw3sfwnhDXCLeoa7h7AyV1DlBqCSgBoEhCcyKWaEyVKw24EOcxfqZEqbo8EwspK10rZZZCvc5dP-zt9JO13A_T1NoPzmyMAwV7YJ99h0ObEXfBUuZDZBv5Qpy-2y7y5fGuxO7xYbd-yrcvm-f1_TYftTG5gULL0pFnrSr0DooSK6a6IEucmhCaZHgpnbFYG--IiClB7EyhS1YrcfP3dpyGr5njoelDTE06u-dhjg2CQjQIUib06ojOrmffjFPo06Tmf6f6BWGsU5Y</recordid><startdate>201208</startdate><enddate>201208</enddate><creator>Testa, María Eugenia</creator><creator>Maffey, Alberto</creator><creator>Colom, Alejandro</creator><creator>Agüero, Luis</creator><creator>Rogé, Ignacio</creator><creator>Andrewartha, María Sol</creator><creator>Teper, Alejandro</creator><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>7X8</scope></search><sort><creationdate>201208</creationdate><title>Pulmonary hemorrhage associated with celiac disease</title><author>Testa, María Eugenia ; Maffey, Alberto ; Colom, Alejandro ; Agüero, Luis ; Rogé, Ignacio ; Andrewartha, María Sol ; Teper, Alejandro</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-p566-604527bcde5381db04718ec94cace043c16718d22b6a196dbcccec471eb6457e3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>spa</language><creationdate>2012</creationdate><topic>Adolescent</topic><topic>Anemia, Iron-Deficiency - etiology</topic><topic>Celiac Disease - diagnosis</topic><topic>Hemoptysis - etiology</topic><topic>Hemorrhage - diagnosis</topic><topic>Hemosiderosis - diagnosis</topic><topic>Hemosiderosis, Pulmonary</topic><topic>Humans</topic><topic>Lung Diseases - diagnosis</topic><topic>Male</topic><topic>Syndrome</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Testa, María Eugenia</creatorcontrib><creatorcontrib>Maffey, Alberto</creatorcontrib><creatorcontrib>Colom, Alejandro</creatorcontrib><creatorcontrib>Agüero, Luis</creatorcontrib><creatorcontrib>Rogé, Ignacio</creatorcontrib><creatorcontrib>Andrewartha, María Sol</creatorcontrib><creatorcontrib>Teper, Alejandro</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>MEDLINE - Academic</collection><jtitle>Archivos argentinos de pediatría</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Testa, María Eugenia</au><au>Maffey, Alberto</au><au>Colom, Alejandro</au><au>Agüero, Luis</au><au>Rogé, Ignacio</au><au>Andrewartha, María Sol</au><au>Teper, Alejandro</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Pulmonary hemorrhage associated with celiac disease</atitle><jtitle>Archivos argentinos de pediatría</jtitle><addtitle>Arch Argent Pediatr</addtitle><date>2012-08</date><risdate>2012</risdate><volume>110</volume><issue>4</issue><spage>e72</spage><epage>e76</epage><pages>e72-e76</pages><eissn>1668-3501</eissn><abstract>Idiopathic pulmonary hemosiderosis is a severe and potentially fatal disease characterized by recurrent episodes of alveolar hemorrhage, hemoptysis, and anemia. His association with celiac disease, described as Lane- Hamilton syndrome, could be due to the fact that both entities share a common pathogenic immune pathway. We report two patients of 13 years who consulted for hemoptysis and severe anemia that had not responded to immunosuppressive treatment with pulses of methyl prednisolone, oral meprednisone and hydroxychloroquine. Although both children highlight the absence of gastrointestinal symptoms at the time of consultation, the dosage of anti-endomysial and anti-transglutaminase antibodies was positive and biopsy confirmed the presence of intestinal enteropathy. It is emphasized that in patients with diffuse alveolar hemorrhage, even in the absence of gastrointestinal symptoms, the concomitant presence of celiac disease should be evaluated. If celiac disease is present, the incorporation of a gluten-free diet helps to control the symptoms, allows reducing the immunosuppressive treatment and improves the clinical course of both entities.</abstract><cop>Argentina</cop><pmid>22859336</pmid><doi>10.1590/S0325-00752012000400016</doi></addata></record> |
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subjects | Adolescent Anemia, Iron-Deficiency - etiology Celiac Disease - diagnosis Hemoptysis - etiology Hemorrhage - diagnosis Hemosiderosis - diagnosis Hemosiderosis, Pulmonary Humans Lung Diseases - diagnosis Male Syndrome |
title | Pulmonary hemorrhage associated with celiac disease |
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