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IgG4-related lung disease showing high standardized uptake values on FDG-PET: report of two cases.

Kitada M, Matuda Y, Hayashi S, Ishibashi K, Oikawa K, Miyokawa N, Ohsaki Y - J Cardiothorac Surg (2013)

Bottom Line: Case 1: A 75-year-old man under treatment for autoimmune pancreatitis and diabetes mellitus was noted to have multiple nodular opacities in both lungs and a mass density in the right paravertebral region on computed tomography (CT).Case 2: A 48-year-old woman consulted our clinic with a chief complaint of bloody sputum.Chest CT revealed a mass density with 12-, 13-, and 16-mm spiculations in the S2 segment of the right upper lobe and irregular thickening of the pleura including the paravertebral region.

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Affiliation: Department of Respiratory Center, Asahikawa Medical University, Midorigaoka-Higashi 2-1-1-1, Asahikawa, Hokkaido 078-8510, Japan. k1111@asahikawa-med.ac.jp

ABSTRACT
Immunoglobulin G4 (IgG4)-related lung disease is a disease in which IgG4-positive plasma cells and lymphocytes infiltrate lung tissues along with immunohistochemically evident fibrous interstitial proliferation in the background, in addition to hyper-IgG4 disease. The diagnosis of this disease can be difficult. Here, we report 2 cases with IgG4-related lung disease that was difficult to differentiate from malignant tumors because both cases had pulmonary lesions showing high standardized uptake values (SUV) on positron emission tomography (PET). Case 1: A 75-year-old man under treatment for autoimmune pancreatitis and diabetes mellitus was noted to have multiple nodular opacities in both lungs and a mass density in the right paravertebral region on computed tomography (CT). As high SUVmax was noted for both lesions on exploration by fluorodeoxyglucose (FDG)-PET/CT, an advanced malignant tumor was diagnosed and a video-assisted thoracoscopic (VATS) biopsy was performed and diagnosed IgG4-related lung disease. Case 2: A 48-year-old woman consulted our clinic with a chief complaint of bloody sputum. Chest CT revealed a mass density with 12-, 13-, and 16-mm spiculations in the S2 segment of the right upper lobe and irregular thickening of the pleura including the paravertebral region. The lesion was a mass showing high SUV in the S2 segment on FDG-PET. Malignancy was suspected from the imaging findings, and a VATS biopsy was performed and diagnosed IgG4-related lung disease. Actively undertaking VATS biopsy in cases with this disease is valuable for making the differential diagnosis between malignant tumors and IgG4-related lung disease, since the diagnosis can be difficult in some patients showing high SUV.

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Microscopic examination. a) Histopathological examination revealed pronounced inflammatory cell infiltration consisting largely of plasma cells, macrophages and lymphocytes on a background comprised of fibrous interstices with fibrosis and fibroblast proliferation. b) Immunohistochemical examination revealed for IgG4 showed a high IgG4/IgG ratio (×400).
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Figure 4: Microscopic examination. a) Histopathological examination revealed pronounced inflammatory cell infiltration consisting largely of plasma cells, macrophages and lymphocytes on a background comprised of fibrous interstices with fibrosis and fibroblast proliferation. b) Immunohistochemical examination revealed for IgG4 showed a high IgG4/IgG ratio (×400).

Mentions: A 75-year-old Japanese man presenting with no respiratory symptoms, who was being treated elsewhere for autoimmune pancreatitis and diabetes, was referred to our clinic because of abnormal opacities noted on computed tomography (CT) of the chest. Tumor markers were not elevated. Chest CT demonstrated multiple nodular densities in both lungs, hilar adenopathy, and a right paravertebral mass lesion (Figure 1). FDG-PET/CT scans disclosed a nodular lesion measuring 35 × 13 mm in size in the right S7 segment with a maximum standardized uptake value (SUV max) of 8.4, multiple lesions in both lungs, and high-SUV areas in the hilar lymph nodes (Figure 2). Furthermore, masses were noted not only in the right paravertebral region but also in part of the pleura; therefore, lung cancer, multiple lung metastasis, and pleural dissemination were diagnosed. Blood chemical laboratory data showed no abnormal value and no elevations of tumor markers. No malignant cells were noted on endoscopic right S7 transbronchial lung biopsy or bronchoalveolar lavage (BAL) examination. Based on the above, a lung biopsy was performed under VATS with the aim of determining a treatment policy. A partial resection including the mass in the S7 segment was carried out along with rapid pathological diagnosis, which led to a diagnosis of plasma cell tumor or inflammatory mass. The tumor was well-demarcated and elastic hard, and the cut surface was almost uniformly milky white (Figure 3). Histopathological examination revealed pronounced inflammatory cell infiltration consisting largely of plasma cells, macrophages and lymphocytes on a background comprised of fibrous interstices with fibrosis and fibroblast proliferation. Plasma cell infiltrates were particularly conspicuous, with a portion showing atypia such as polynuclear cells, and represented reactive growth. Slides stained for IgG4 showed 90 IgG4-positive cells per HPF and the IgG4/IgG ratio was 35%-46% (Figure 4). There were findings consistent with obliterating phlebitis, and a diagnosis of IgG4-related inflammatory pseudotumor was thus made. The serum IgG4 level, as determined postoperatively, was elevated at 520 mg/dL. The densities noted on chest scans disappeared following oral corticosteroid administration.


IgG4-related lung disease showing high standardized uptake values on FDG-PET: report of two cases.

Kitada M, Matuda Y, Hayashi S, Ishibashi K, Oikawa K, Miyokawa N, Ohsaki Y - J Cardiothorac Surg (2013)

Microscopic examination. a) Histopathological examination revealed pronounced inflammatory cell infiltration consisting largely of plasma cells, macrophages and lymphocytes on a background comprised of fibrous interstices with fibrosis and fibroblast proliferation. b) Immunohistochemical examination revealed for IgG4 showed a high IgG4/IgG ratio (×400).
© Copyright Policy - open-access
Related In: Results  -  Collection

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Show All Figures
getmorefigures.php?uid=PMC3717047&req=5

Figure 4: Microscopic examination. a) Histopathological examination revealed pronounced inflammatory cell infiltration consisting largely of plasma cells, macrophages and lymphocytes on a background comprised of fibrous interstices with fibrosis and fibroblast proliferation. b) Immunohistochemical examination revealed for IgG4 showed a high IgG4/IgG ratio (×400).
Mentions: A 75-year-old Japanese man presenting with no respiratory symptoms, who was being treated elsewhere for autoimmune pancreatitis and diabetes, was referred to our clinic because of abnormal opacities noted on computed tomography (CT) of the chest. Tumor markers were not elevated. Chest CT demonstrated multiple nodular densities in both lungs, hilar adenopathy, and a right paravertebral mass lesion (Figure 1). FDG-PET/CT scans disclosed a nodular lesion measuring 35 × 13 mm in size in the right S7 segment with a maximum standardized uptake value (SUV max) of 8.4, multiple lesions in both lungs, and high-SUV areas in the hilar lymph nodes (Figure 2). Furthermore, masses were noted not only in the right paravertebral region but also in part of the pleura; therefore, lung cancer, multiple lung metastasis, and pleural dissemination were diagnosed. Blood chemical laboratory data showed no abnormal value and no elevations of tumor markers. No malignant cells were noted on endoscopic right S7 transbronchial lung biopsy or bronchoalveolar lavage (BAL) examination. Based on the above, a lung biopsy was performed under VATS with the aim of determining a treatment policy. A partial resection including the mass in the S7 segment was carried out along with rapid pathological diagnosis, which led to a diagnosis of plasma cell tumor or inflammatory mass. The tumor was well-demarcated and elastic hard, and the cut surface was almost uniformly milky white (Figure 3). Histopathological examination revealed pronounced inflammatory cell infiltration consisting largely of plasma cells, macrophages and lymphocytes on a background comprised of fibrous interstices with fibrosis and fibroblast proliferation. Plasma cell infiltrates were particularly conspicuous, with a portion showing atypia such as polynuclear cells, and represented reactive growth. Slides stained for IgG4 showed 90 IgG4-positive cells per HPF and the IgG4/IgG ratio was 35%-46% (Figure 4). There were findings consistent with obliterating phlebitis, and a diagnosis of IgG4-related inflammatory pseudotumor was thus made. The serum IgG4 level, as determined postoperatively, was elevated at 520 mg/dL. The densities noted on chest scans disappeared following oral corticosteroid administration.

Bottom Line: Case 1: A 75-year-old man under treatment for autoimmune pancreatitis and diabetes mellitus was noted to have multiple nodular opacities in both lungs and a mass density in the right paravertebral region on computed tomography (CT).Case 2: A 48-year-old woman consulted our clinic with a chief complaint of bloody sputum.Chest CT revealed a mass density with 12-, 13-, and 16-mm spiculations in the S2 segment of the right upper lobe and irregular thickening of the pleura including the paravertebral region.

View Article: PubMed Central - HTML - PubMed

Affiliation: Department of Respiratory Center, Asahikawa Medical University, Midorigaoka-Higashi 2-1-1-1, Asahikawa, Hokkaido 078-8510, Japan. k1111@asahikawa-med.ac.jp

ABSTRACT
Immunoglobulin G4 (IgG4)-related lung disease is a disease in which IgG4-positive plasma cells and lymphocytes infiltrate lung tissues along with immunohistochemically evident fibrous interstitial proliferation in the background, in addition to hyper-IgG4 disease. The diagnosis of this disease can be difficult. Here, we report 2 cases with IgG4-related lung disease that was difficult to differentiate from malignant tumors because both cases had pulmonary lesions showing high standardized uptake values (SUV) on positron emission tomography (PET). Case 1: A 75-year-old man under treatment for autoimmune pancreatitis and diabetes mellitus was noted to have multiple nodular opacities in both lungs and a mass density in the right paravertebral region on computed tomography (CT). As high SUVmax was noted for both lesions on exploration by fluorodeoxyglucose (FDG)-PET/CT, an advanced malignant tumor was diagnosed and a video-assisted thoracoscopic (VATS) biopsy was performed and diagnosed IgG4-related lung disease. Case 2: A 48-year-old woman consulted our clinic with a chief complaint of bloody sputum. Chest CT revealed a mass density with 12-, 13-, and 16-mm spiculations in the S2 segment of the right upper lobe and irregular thickening of the pleura including the paravertebral region. The lesion was a mass showing high SUV in the S2 segment on FDG-PET. Malignancy was suspected from the imaging findings, and a VATS biopsy was performed and diagnosed IgG4-related lung disease. Actively undertaking VATS biopsy in cases with this disease is valuable for making the differential diagnosis between malignant tumors and IgG4-related lung disease, since the diagnosis can be difficult in some patients showing high SUV.

Show MeSH
Related in: MedlinePlus