Clinical Predictors of a Diagnosis of Common Variable Immunodeficiency-related Granulomatous-Lymphocytic Interstitial Lung Disease

被引:48
作者
Mannina, Amar [1 ]
Chung, Jonathan H. [5 ]
Swigris, Jeffrey J. [1 ]
Solomon, Joshua J. [1 ]
Huie, Tristan J. [1 ]
Yunt, Zulma X. [1 ]
Truong, Tho Q. [2 ]
Brown, Kevin K. [1 ]
Achcar, Rosane Duarte [3 ]
Olson, Amy L. [1 ]
Cox, Christian W. [6 ]
Kligerman, Seth J. [7 ]
Curran-Everett, Douglas [4 ]
Perez, Evans R. Fernandez [1 ]
机构
[1] Natl Jewish Hlth, Dept Pulm & Crit Care Med, Southside Bldg,Off G012,1400 Jackson St, Denver, CO 80206 USA
[2] Natl Jewish Hlth, Dept Allergy & Immunol, Denver, CO USA
[3] Natl Jewish Hlth, Dept Pathol, Denver, CO USA
[4] Natl Jewish Hlth, Dept Biostat & Bioinformat, Denver, CO USA
[5] Univ Chicago, Dept Thorac Radiol, Chicago, IL 60637 USA
[6] Mayo Clin, Dept Thorac Radiol, Rochester, MN USA
[7] Univ Maryland, Sch Med, Dept Diagnost Radiol & Nucl Med, Baltimore, MD 21201 USA
关键词
common variable immunodeficiency; granulomatous lymphocytic interstitial lung disease; splenomegaly; polyarthritis; cytopenia; MEMORY B-CELLS; IMMUNE-DEFICIENCY; DISORDERS; SARCOIDOSIS; MANAGEMENT; MUTATIONS; SUBGROUPS; CRITERIA;
D O I
10.1513/AnnalsATS.201511-728OC
中图分类号
R56 [呼吸系及胸部疾病];
学科分类号
摘要
Rationale: Granulomatous-lymphocytic interstitial lung disease (GLILD) has emerged as a major cause of morbidity in patients with common variable immunodeficiency (CVID). While GLILD is among the most serious noninfectious pulmonary complications of CVID, risk factors for this condition have not been reported. Objectives: To identify clinical, physiologic, and serologic risk factors for GLILD in adults with CVID. Methods: Of 345 consecutive adult patients with CVID, we identified 34 in the National Jewish Health research database who had a radiographic-pathologic diagnosis of GLILD evaluated between 2002 and 2014. Each case was age and sex matched to 52 CVID control subjects. We used logistic regression to determine independent predictors of GLILD. A mixed effects model was used to estimate the longitudinal change in percent predicted FVC. Measurements and Main Results: The mean time from CVID diagnosis to GLILD detection was 7.8 years. Compared with matched control subjects, cases were more likely to have a history of autoimmune cytopenia, hypersplenism, polyarthritis, lower marginal zone and switched memory B cells, and restrictive lung function. Multivariate analysis revealed that hypersplenism (odds ratio [OR], 24; 95% confidence interval [CI], 4.5-179.1), polyarthritis (OR, 19; 95% CI, 2.3-206.8), and percent predicted FVC(OR, 0.93; 95% CI, 0.87-0.98) were independently associated with the development of GLILD. The rate of change of percent predicted FVC (slope, P = 0.48) did not vary significantly in patients with GLILD over a mean follow-up of 7 years after diagnosis. Conclusions: Hypersplenism and polyarthritis are strong risk factors for GLILD in patients with CVID. Percent predicted FVC remained stable over time in patients with GLILD.
引用
收藏
页码:1042 / 1049
页数:8
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