Distinctive Pulmonary Histopathology With Increased IgG4-positive Plasma Cells in Patients With Autoimmune Pancreatitis Report of 6 and 12 Cases With Similar Histopathology

被引:128
作者
Shrestha, Bijayee [1 ]
Sekiguchi, Hiroshi
Colby, Thomas V. [1 ]
Graziano, Paolo [4 ]
Aubry, Marie-Christine [1 ]
Smyrk, Thomas C. [1 ]
Feldman, Andrew L. [1 ]
Cornell, Lynn D. [1 ]
Ryu, Jay H.
Chari, Suresh T. [2 ]
Dueck, Amylou C. [3 ]
Yi, Eunhee S. [1 ]
机构
[1] Mayo Clin, Dept Lab Med & Pathol, Rochester, MN 55905 USA
[2] Mayo Clin, Div Gastroenterol & Hepatol, Dept Internal Med, Rochester, MN 55905 USA
[3] Mayo Clin, Biostat Sect, Scottsdale, AZ USA
[4] C Forlanini Hosp, Unit Pathol, Rome, Italy
关键词
IgG4; inflammatory and interstitial lung disease; autoimmune pancreatitis; Sjogren syndrome; Erdheim-Chester disease; Rosai-Dorfman disease; inflammatory myofibroblastic tumor; SERUM IGG4 LEVEL; INFLAMMATORY PSEUDOTUMORS; INTERSTITIAL PNEUMONIA; INFILTRATION; INVOLVEMENT; FIBROSIS; PROSTATITIS; DIAGNOSIS; LUNG;
D O I
10.1097/PAS.0b013e3181ac43b6
中图分类号
R36 [病理学];
学科分类号
100104 ;
摘要
Autoimmune pancreatitis (AP) is one manifestation of a systemic, steroid-responsive disease with elevated serum IgG4 and characteristic histopathology, including increased IgG4-positive (+) plasma cells in the tissue. The histopathology of pulmonary IgG4 disease has not been well established. Six lung biopsies from patients with documented AP were studied, along with 12 additional cases showing similar pulmonary histopathology. For comparison, we examined Erdheim-Chester disease (n = 3), pulmonary Sjogren syndrome (n = 19), inflammatory myofibroblastic tumor (n = 10), various inflammatory and interstitial lung disease (n = 61), and nodal or extranodal Rosai-Dorfman disease (RD) in adults (n = 8). All cases were stained for IgG4 and scored as 1, 2, and 3 as described in AP according to the following criteria: 0, < 5 (per high power field); 1, 5 to 10; 2, 11 to 30; and 3, > 30. Five lung biopsies from AP patients showed IgG4 score of 3, and I had a score of 2. Consistent findings in lung biopsies of AP patients included endothelialitis of pulmonary vessels, active fibrosis, lymphangitic inflammatory infiltrates rich in plasma cells and histiocytes with or Without nodule formation, and fibrinous pleuriti's. Prominent lymphatic dilatation with histiocytes showing emperipolesis of lymphocytes was also seen. All 12 additional cases showing these histologic features also had the IgG4 score of 2 or 3. Among other conditions, an IgG4 score of 2 or 3 was seen in 6 of 8 RD, 4 of 10 inflammatory myofibroblastic tumors, and 8 of 61 inflammatory and interstitial lung disease, but in none of the rest. In conclusion, distinctive pulmonary histopathology was associated with increased IgG4+ cells in both AP patients and those unknown for AP status. The significance of increased IgG4 + cells in high proportion of RD cases merits further study as does overlap of RD and IgG4 disease.
引用
收藏
页码:1450 / 1462
页数:13
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