Literature DB >> 32235152

GLILD Revisited: Pulmonary Pathology of Common Variable and Selective IgA Immunodeficiency.

Brandon T Larsen1, Maxwell L Smith1, Henry D Tazelaar1, Eunhee S Yi2, Jay H Ryu3, Andrew Churg4.   

Abstract

Common variable immunodeficiency (CVID) and selective immunoglobulin A deficiency (IgAD) often cause chronic lung disease, but the pulmonary pathologic features of these systemic diseases are poorly recognized by pathologists. It has been claimed that CVID cases show a characteristic combination of noncaseating granulomas-lymphoid proliferations termed granulomatous-lymphocytic interstitial lung disease (GLILD). We present 34 surgical lung biopsy cases of CVID and 4 of IgAD. Noncaseating granulomas were seen in 23/34 (68%) CVID and 2/4 (50%) IgAD cases. A statistically identical pattern of benign lymphoid proliferation was found in CVID and IgAD whether or not granulomas were present. Organizing pneumonia, sometimes considered a part of GLILD, was seen in 25/34 (74%) CVID and 2/4 (50%) IgAD cases and did not correlate with the presence of granulomas. On follow-up, 3 CVID patients died (only 1 of pulmonary disease), while 21 others are alive at 1 to 300 months with no difference by presence or absence of granulomas. Three IgAD patients with follow-up are alive. We conclude that CVID and IgAD are indistinguishable in surgical lung biopsies and a subset of both show patterns that would qualify as GLILD, while other cases lack granulomas but have identical patterns of lymphoid infiltration and organizing pneumonia. We suggest that GLILD is neither a specific nor a useful entity, and biopsies from CVID and IgAD patients should be diagnosed simply by microscopic pattern(s) observed. The prognosis of CVID with lymphoid infiltrates with or without granulomas in this series was good, contrary to claims in the literature about GLILD.

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Year:  2020        PMID: 32235152     DOI: 10.1097/PAS.0000000000001479

Source DB:  PubMed          Journal:  Am J Surg Pathol        ISSN: 0147-5185            Impact factor:   6.394


  4 in total

Review 1.  Treatment of immune-mediated cytopenias in patients with primary immunodeficiencies and immune regulatory disorders (PIRDs).

Authors:  Markus G Seidel
Journal:  Hematology Am Soc Hematol Educ Program       Date:  2020-12-04

2.  Raised Serum Markers of T Cell Activation and Exhaustion in Granulomatous-Lymphocytic Interstitial Lung Disease in Common Variable Immunodeficiency.

Authors:  Mai Sasaki Aanensen Fraz; Annika Elisabet Michelsen; Natasha Moe; Trond Mogens Aaløkken; Magnhild Eide Macpherson; Ingvild Nordøy; Pål Aukrust; Eli Taraldsrud; Are Martin Holm; Thor Ueland; Silje Fjellgård Jørgensen; Børre Fevang
Journal:  J Clin Immunol       Date:  2022-07-05       Impact factor: 8.542

3.  Granulomatous-Lymphocytic Interstitial Lung Disease in Common Variable Immunodeficiency-Features of CT and 18F-FDG Positron Emission Tomography/CT in Clinically Progressive Disease.

Authors:  Mai Sasaki Aanensen Fraz; Natasha Moe; Mona-Elisabeth Revheim; Maria L Stavrinou; Michael T Durheim; Ingvild Nordøy; Magnhild Eide Macpherson; Pål Aukrust; Silje Fjellgård Jørgensen; Trond Mogens Aaløkken; Børre Fevang
Journal:  Front Immunol       Date:  2021-01-26       Impact factor: 7.561

Review 4.  Histology of Interstitial Lung Disease in Common Variable Immune Deficiency.

Authors:  Fatima Dhalla; Dylan J Mac Lochlainn; Helen Chapel; Smita Y Patel
Journal:  Front Immunol       Date:  2020-11-20       Impact factor: 7.561

  4 in total

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