Amika K Sood1,2, William Funkhouser3, Brian Handly4, Brent Weston5, Eveline Y Wu6. 1. Department of Pediatrics, Division of Allergy, Immunology, and Rheumatology, University of North Carolina, Chapel Hill, NC, USA. amika_sood@med.unc.edu. 2. Center for Environmental Medicine, Asthma and Lung Biology, University of North Carolina at Chapel Hill, 104 Mason Farm Road, CB #7310, Chapel Hill, NC, 27599-7310, USA. amika_sood@med.unc.edu. 3. Deparment of Pathology and Laboratory Medicine, University of North Carolina, Chapel Hill, NC, USA. 4. Department of Radiology, University of North Carolina, Chapel Hill, NC, USA. 5. Department of Pediatrics, Division of Hematology-Oncology, University of North Carolina, Chapel Hill, NC, USA. 6. Department of Pediatrics, Division of Allergy, Immunology, and Rheumatology, University of North Carolina, Chapel Hill, NC, USA.
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. 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.
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. 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.
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