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Granulomatous-Lymphocytic Interstitial Lung Disease in 22q11.2 Deletion Syndrome: a Case Report and Literature Review
Purpose of Review Granulomatous-lymphocytic interstitial lung disease (GLILD) has classically been associated with common variable immune deficiency (CVID), but is increasingly being reported in other immunodeficiencies. We describe the second reported case of GLILD in a patient with 22q11.2 deletio...
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| Published in: | Current allergy and asthma reports 2018-03, Vol.18 (3), p.14-10, Article 14 |
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| container_title | Current allergy and asthma reports |
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| creator | Sood, Amika K. Funkhouser, William Handly, Brian Weston, Brent Wu, Eveline Y. |
| description | Purpose of Review
Granulomatous-lymphocytic interstitial lung disease (GLILD) has classically been associated with common variable immune deficiency (CVID), but is increasingly being reported in other immunodeficiencies. We describe the second reported case of GLILD in a patient with 22q11.2 deletion syndrome (22q11.2DS) and review the recent literature surrounding GLILD.
Recent Findings
GLILD is characterized by granulomata and lymphoproliferation. Consensus statements and retrospective and case-control studies have better elucidated the clinicopathological and radiographic manifestations of GLILD, allowing for its differentiation from similar conditions like sarcoidosis. Gaps of knowledge remain, however, particularly regarding optimal management strategies. Combination therapies targeting T and B cell populations have recently shown favorable results.
Summary
GLILD is associated with poorer outcomes in CVID. Its recognition as a rare complication of 22q11.2DS and other immunodeficiencies therefore has important therapeutic and prognostic implications. Additional research is needed to better understand the natural history and pathogenesis of GLILD and to develop evidence-based practice guidelines. |
| doi_str_mv | 10.1007/s11882-018-0769-7 |
| format | article |
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Granulomatous-lymphocytic interstitial lung disease (GLILD) has classically been associated with common variable immune deficiency (CVID), but is increasingly being reported in other immunodeficiencies. We describe the second reported case of GLILD in a patient with 22q11.2 deletion syndrome (22q11.2DS) and review the recent literature surrounding GLILD.
Recent Findings
GLILD is characterized by granulomata and lymphoproliferation. Consensus statements and retrospective and case-control studies have better elucidated the clinicopathological and radiographic manifestations of GLILD, allowing for its differentiation from similar conditions like sarcoidosis. Gaps of knowledge remain, however, particularly regarding optimal management strategies. Combination therapies targeting T and B cell populations have recently shown favorable results.
Summary
GLILD is associated with poorer outcomes in CVID. Its recognition as a rare complication of 22q11.2DS and other immunodeficiencies therefore has important therapeutic and prognostic implications. Additional research is needed to better understand the natural history and pathogenesis of GLILD and to develop evidence-based practice guidelines.</description><identifier>ISSN: 1529-7322</identifier><identifier>EISSN: 1534-6315</identifier><identifier>DOI: 10.1007/s11882-018-0769-7</identifier><identifier>PMID: 29470661</identifier><language>eng</language><publisher>New York: Springer US</publisher><subject>Adolescent ; Allergology ; Autoimmunity (TK Tarrant ; Case reports ; Common Variable Immunodeficiency - complications ; Common Variable Immunodeficiency - pathology ; DiGeorge Syndrome - complications ; DiGeorge Syndrome - pathology ; Female ; Humans ; Lung diseases ; Lung Diseases, Interstitial - diagnosis ; Lung Diseases, Interstitial - pathology ; Male ; Medicine ; Medicine & Public Health ; Retrospective Studies ; Section Editor ; Topical Collection on Autoimmunity</subject><ispartof>Current allergy and asthma reports, 2018-03, Vol.18 (3), p.14-10, Article 14</ispartof><rights>Springer Science+Business Media, LLC, part of Springer Nature 2018</rights><rights>Current Allergy and Asthma Reports is a copyright of Springer, (2018). All Rights Reserved.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c536t-81c8eb697cf8e3c4595fb33156c628185d0b9dc3641a7a88c63a25555000e5ba3</citedby><cites>FETCH-LOGICAL-c536t-81c8eb697cf8e3c4595fb33156c628185d0b9dc3641a7a88c63a25555000e5ba3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>230,314,780,784,885,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/29470661$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Sood, Amika K.</creatorcontrib><creatorcontrib>Funkhouser, William</creatorcontrib><creatorcontrib>Handly, Brian</creatorcontrib><creatorcontrib>Weston, Brent</creatorcontrib><creatorcontrib>Wu, Eveline Y.</creatorcontrib><title>Granulomatous-Lymphocytic Interstitial Lung Disease in 22q11.2 Deletion Syndrome: a Case Report and Literature Review</title><title>Current allergy and asthma reports</title><addtitle>Curr Allergy Asthma Rep</addtitle><addtitle>Curr Allergy Asthma Rep</addtitle><description>Purpose of Review
Granulomatous-lymphocytic interstitial lung disease (GLILD) has classically been associated with common variable immune deficiency (CVID), but is increasingly being reported in other immunodeficiencies. We describe the second reported case of GLILD in a patient with 22q11.2 deletion syndrome (22q11.2DS) and review the recent literature surrounding GLILD.
Recent Findings
GLILD is characterized by granulomata and lymphoproliferation. Consensus statements and retrospective and case-control studies have better elucidated the clinicopathological and radiographic manifestations of GLILD, allowing for its differentiation from similar conditions like sarcoidosis. Gaps of knowledge remain, however, particularly regarding optimal management strategies. Combination therapies targeting T and B cell populations have recently shown favorable results.
Summary
GLILD is associated with poorer outcomes in CVID. Its recognition as a rare complication of 22q11.2DS and other immunodeficiencies therefore has important therapeutic and prognostic implications. Additional research is needed to better understand the natural history and pathogenesis of GLILD and to develop evidence-based practice guidelines.</description><subject>Adolescent</subject><subject>Allergology</subject><subject>Autoimmunity (TK Tarrant</subject><subject>Case reports</subject><subject>Common Variable Immunodeficiency - complications</subject><subject>Common Variable Immunodeficiency - pathology</subject><subject>DiGeorge Syndrome - complications</subject><subject>DiGeorge Syndrome - pathology</subject><subject>Female</subject><subject>Humans</subject><subject>Lung diseases</subject><subject>Lung Diseases, Interstitial - diagnosis</subject><subject>Lung Diseases, Interstitial - pathology</subject><subject>Male</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Retrospective Studies</subject><subject>Section Editor</subject><subject>Topical Collection on Autoimmunity</subject><issn>1529-7322</issn><issn>1534-6315</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2018</creationdate><recordtype>article</recordtype><recordid>eNp1kU1v1DAQhi0Eoh_wA7ggS1y4pPVH7DgckNC2lEqRkPg4W44zu3WV2FvbabX_vg5bSkHCF1szz7zjmRehN5ScUEKa00SpUqwiVFWkkW3VPEOHVPC6kpyK58ublSBn7AAdpXRNCCtV7CU6YG3dECnpIZovovHzGCaTw5yqbjdtr4LdZWfxpc8QU3bZmRF3s9_gM5fAJMDOY8ZuKD1h-AxGyC54_H3nhxgm-IANXi3QN9iGmLHxA-5cUTJ5jkv01sHdK_RibcYErx_uY_Tz8_mP1Zeq-3pxufrUVVZwmStFrYJeto1dK-C2Fq1Y97zMJq1kiioxkL4dLJc1NY1RykpumCiHEAKiN_wYfdzrbud-gsGCz9GMehvdZOJOB-P03xnvrvQm3GrR8qJCi8D7B4EYbmZIWU8uWRhH46HsSy8LbRWpJS_ou3_Q6zBHX8b7RdWKN2yh6J6yMaQUYf34GUr0Yqrem6qLqXoxVTel5u3TKR4rfrtYALYHUkn5DcQ_rf-veg9rJq0K</recordid><startdate>20180301</startdate><enddate>20180301</enddate><creator>Sood, Amika K.</creator><creator>Funkhouser, William</creator><creator>Handly, Brian</creator><creator>Weston, Brent</creator><creator>Wu, Eveline Y.</creator><general>Springer US</general><general>Springer Nature B.V</general><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>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>20180301</creationdate><title>Granulomatous-Lymphocytic Interstitial Lung Disease in 22q11.2 Deletion Syndrome: a Case Report and Literature Review</title><author>Sood, Amika K. ; Funkhouser, William ; Handly, Brian ; Weston, Brent ; Wu, Eveline Y.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c536t-81c8eb697cf8e3c4595fb33156c628185d0b9dc3641a7a88c63a25555000e5ba3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2018</creationdate><topic>Adolescent</topic><topic>Allergology</topic><topic>Autoimmunity (TK Tarrant</topic><topic>Case reports</topic><topic>Common Variable Immunodeficiency - complications</topic><topic>Common Variable Immunodeficiency - pathology</topic><topic>DiGeorge Syndrome - complications</topic><topic>DiGeorge Syndrome - pathology</topic><topic>Female</topic><topic>Humans</topic><topic>Lung diseases</topic><topic>Lung Diseases, Interstitial - diagnosis</topic><topic>Lung Diseases, Interstitial - pathology</topic><topic>Male</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Retrospective Studies</topic><topic>Section Editor</topic><topic>Topical Collection on Autoimmunity</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Sood, Amika K.</creatorcontrib><creatorcontrib>Funkhouser, William</creatorcontrib><creatorcontrib>Handly, Brian</creatorcontrib><creatorcontrib>Weston, Brent</creatorcontrib><creatorcontrib>Wu, Eveline Y.</creatorcontrib><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>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</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)</collection><collection>ProQuest Central</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</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>PubMed Central (Full Participant titles)</collection><jtitle>Current allergy and asthma reports</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Sood, Amika K.</au><au>Funkhouser, William</au><au>Handly, Brian</au><au>Weston, Brent</au><au>Wu, Eveline Y.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Granulomatous-Lymphocytic Interstitial Lung Disease in 22q11.2 Deletion Syndrome: a Case Report and Literature Review</atitle><jtitle>Current allergy and asthma reports</jtitle><stitle>Curr Allergy Asthma Rep</stitle><addtitle>Curr Allergy Asthma Rep</addtitle><date>2018-03-01</date><risdate>2018</risdate><volume>18</volume><issue>3</issue><spage>14</spage><epage>10</epage><pages>14-10</pages><artnum>14</artnum><issn>1529-7322</issn><eissn>1534-6315</eissn><abstract>Purpose of Review
Granulomatous-lymphocytic interstitial lung disease (GLILD) has classically been associated with common variable immune deficiency (CVID), but is increasingly being reported in other immunodeficiencies. We describe the second reported case of GLILD in a patient with 22q11.2 deletion syndrome (22q11.2DS) and review the recent literature surrounding GLILD.
Recent Findings
GLILD is characterized by granulomata and lymphoproliferation. Consensus statements and retrospective and case-control studies have better elucidated the clinicopathological and radiographic manifestations of GLILD, allowing for its differentiation from similar conditions like sarcoidosis. Gaps of knowledge remain, however, particularly regarding optimal management strategies. Combination therapies targeting T and B cell populations have recently shown favorable results.
Summary
GLILD is associated with poorer outcomes in CVID. Its recognition as a rare complication of 22q11.2DS and other immunodeficiencies therefore has important therapeutic and prognostic implications. Additional research is needed to better understand the natural history and pathogenesis of GLILD and to develop evidence-based practice guidelines.</abstract><cop>New York</cop><pub>Springer US</pub><pmid>29470661</pmid><doi>10.1007/s11882-018-0769-7</doi><tpages>10</tpages><oa>free_for_read</oa></addata></record> |
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| subjects | Adolescent Allergology Autoimmunity (TK Tarrant Case reports Common Variable Immunodeficiency - complications Common Variable Immunodeficiency - pathology DiGeorge Syndrome - complications DiGeorge Syndrome - pathology Female Humans Lung diseases Lung Diseases, Interstitial - diagnosis Lung Diseases, Interstitial - pathology Male Medicine Medicine & Public Health Retrospective Studies Section Editor Topical Collection on Autoimmunity |
| title | Granulomatous-Lymphocytic Interstitial Lung Disease in 22q11.2 Deletion Syndrome: a Case Report and Literature Review |
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