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Yoh Zen - One of the best experts on this subject based on the ideXlab platform.

  • A case of immunoglobulin G4-related Chronic Sclerosing Sialadenitis and dacryoadenitis associated with tuberculosis
    Modern Rheumatology, 2008
    Co-Authors: Mitsuhiro Kawano, Kazunori Yamada, Yasushi Kakuchi, Kiyoaki Ito, Ryoko Hamano, Hiroshi Fujii, Ryo Inoue, Masami Matsumura, Masayuki Takahira, Yoh Zen
    Abstract:

    We describe a 64-year-old woman with Chronic Sclerosing Sialadenitis and dacryoadenitis, which developed during treatment for cervical lymph node tuberculosis. Anti-tuberculosis treatment did not improve the swelling in the lacrimal and submandibular glands, and a biopsy specimen of the lacrimal gland showed inflammation, with abundant lymphoid follicles with fibrosis and granuloma without caseous necrosis. Immunohistological examination of a repeat biopsy specimen showed abundant immunoglobulin (Ig) G4-positive plasma cell infiltration. Corticosteroid therapy improved the salivary gland swelling without reactivation of the tuberculosis. This case suggests that an abnormal immunological reaction to tuberculosis may be one of the etiological candidates for IgG4-related disease.

  • pathology and immunopathology of immunoglobulin g4 related Sclerosing cholangitis the latest addition to the Sclerosing cholangitis family
    Hepatology Research, 2007
    Co-Authors: Yasuni Nakanuma, Yoh Zen
    Abstract:

    Sclerosing cholangitis is heterogeneous in its etiopathogenesis. Recently, Sclerosing cholangitis showing abundant immunoglobulin (Ig)G4+ plasma cell infiltration was added to the Sclerosing cholangitis group. This form was frequently associated with Sclerosing pancreatitis (autoimmune pancreatitis) and also occasionally with other diseases such as Chronic Sclerosing Sialadenitis, all of which falls within IgG4-related Sclerosing disease. Herein, this new member, called IgG4-related Sclerosing cholangitis (IgG4-SC), is reviewed. IgG4-SC shows grossly medullary and fleshy lesions along the biliary tree, and histologically marked lymphoplasmacytic infiltration with extensive fibrosis, and obliterative phlebitis, sharing histopathological features with Sclerosing pancreatitis. Peribiliary glands are also severely affected. Interestingly, hepatic inflammatory pseudotumor (HIP) is not infrequently associated with IgG4-SC, and is thought as a local exaggeration of IgG4-SC. Immunohistochemically, many IgG4+ plasma cells and CD4+/CD25+ regulatory T cells are found around the affected bile ducts and portal tracts. Incontrast, these cells are scarce in the affected bile ducts of primary Sclerosing cholangitis (PSC), a prototype of Sclerosing cholangitis. Biliary lining epithelia are relatively spared in IgG4-SC in comparison with those of PSC showing degeneration and ulceration. In some cases of IgG4-SC, IgG4+ plasma cells are also found considerably in small portal tracts, so needle liver biopsy is useful for the diagnosis of IgG4-SC. Therapeutically, IgG4-SC responds well to steroid therapy, while such character is not reported in PSC. Taken together, IgG4-SC may be etiologically different from PSC, and immunopathological processes relating to IgG4 and regulatory T cells may be involved in the pathogenesis of IgG4-SC. Further studies are needed to clarify the etiopathogenesis of IgG4-SC and its related disorders.

  • igg4 positive plasma cells in inflammatory pseudotumor plasma cell granuloma of the lung
    Human Pathology, 2005
    Co-Authors: Yoh Zen, Satoshi Kitagawa, Hiroshi Minato, Hiroshi Kurumaya, Kazuyoshi Katayanagi, Shinji Masuda, Hideki Niwa, Masaki Fujimura, Yasuni Nakanuma
    Abstract:

    The association between IgG4 dysregulation and inflammatory pseudotumor (IPT) was first reported in Sclerosing pancreatitis. Recently, we described IPTs of the liver and breast, into both of which many IgG4-positive plasma cells had infiltrated. In this study, we examined the clinical and histological features of 9 cases of IPT (histologically corresponding to plasma cell granuloma) of the lung with an emphasis on IgG4-positive plasma cell infiltration. The lesions were characterized histologically by dense lymphoplasmacytic infiltrates intermixed with fibrosis and, in some cases, prominent eosinophilic infiltration, irregular narrowing of bronchioles entrapped in nodules, and an interstitial pneumonia pattern at the boundaries of nodules. Obliterative phlebitis was easily found in all cases, and 5 lesions also had obliterative arteritis. Immunostaining revealed many IgG4-positive plasma cells diffusely distributed within nodules, and the ratios of IgG4-positive to other plasma cells were extraordinarily high. Of the 9 patients, 8 underwent surgical treatment and in 1 patient, lesion was diagnosed on transbronchial biopsy and effectively treated with corticosteroid. Two cases were associated with Chronic Sclerosing Sialadenitis or lymphadenopathy, in which many IgG4-positive plasma cells were also identified by immunostaining. The clinicopathologic similarities between IPT of the lung and Sclerosing pancreatitis suggest that IgG4-related immunopathologic processes might be involved in the pathogenesis of the pulmonary lesions.

Yasuni Nakanuma - One of the best experts on this subject based on the ideXlab platform.

  • pathology and immunopathology of immunoglobulin g4 related Sclerosing cholangitis the latest addition to the Sclerosing cholangitis family
    Hepatology Research, 2007
    Co-Authors: Yasuni Nakanuma, Yoh Zen
    Abstract:

    Sclerosing cholangitis is heterogeneous in its etiopathogenesis. Recently, Sclerosing cholangitis showing abundant immunoglobulin (Ig)G4+ plasma cell infiltration was added to the Sclerosing cholangitis group. This form was frequently associated with Sclerosing pancreatitis (autoimmune pancreatitis) and also occasionally with other diseases such as Chronic Sclerosing Sialadenitis, all of which falls within IgG4-related Sclerosing disease. Herein, this new member, called IgG4-related Sclerosing cholangitis (IgG4-SC), is reviewed. IgG4-SC shows grossly medullary and fleshy lesions along the biliary tree, and histologically marked lymphoplasmacytic infiltration with extensive fibrosis, and obliterative phlebitis, sharing histopathological features with Sclerosing pancreatitis. Peribiliary glands are also severely affected. Interestingly, hepatic inflammatory pseudotumor (HIP) is not infrequently associated with IgG4-SC, and is thought as a local exaggeration of IgG4-SC. Immunohistochemically, many IgG4+ plasma cells and CD4+/CD25+ regulatory T cells are found around the affected bile ducts and portal tracts. Incontrast, these cells are scarce in the affected bile ducts of primary Sclerosing cholangitis (PSC), a prototype of Sclerosing cholangitis. Biliary lining epithelia are relatively spared in IgG4-SC in comparison with those of PSC showing degeneration and ulceration. In some cases of IgG4-SC, IgG4+ plasma cells are also found considerably in small portal tracts, so needle liver biopsy is useful for the diagnosis of IgG4-SC. Therapeutically, IgG4-SC responds well to steroid therapy, while such character is not reported in PSC. Taken together, IgG4-SC may be etiologically different from PSC, and immunopathological processes relating to IgG4 and regulatory T cells may be involved in the pathogenesis of IgG4-SC. Further studies are needed to clarify the etiopathogenesis of IgG4-SC and its related disorders.

  • igg4 positive plasma cells in inflammatory pseudotumor plasma cell granuloma of the lung
    Human Pathology, 2005
    Co-Authors: Yoh Zen, Satoshi Kitagawa, Hiroshi Minato, Hiroshi Kurumaya, Kazuyoshi Katayanagi, Shinji Masuda, Hideki Niwa, Masaki Fujimura, Yasuni Nakanuma
    Abstract:

    The association between IgG4 dysregulation and inflammatory pseudotumor (IPT) was first reported in Sclerosing pancreatitis. Recently, we described IPTs of the liver and breast, into both of which many IgG4-positive plasma cells had infiltrated. In this study, we examined the clinical and histological features of 9 cases of IPT (histologically corresponding to plasma cell granuloma) of the lung with an emphasis on IgG4-positive plasma cell infiltration. The lesions were characterized histologically by dense lymphoplasmacytic infiltrates intermixed with fibrosis and, in some cases, prominent eosinophilic infiltration, irregular narrowing of bronchioles entrapped in nodules, and an interstitial pneumonia pattern at the boundaries of nodules. Obliterative phlebitis was easily found in all cases, and 5 lesions also had obliterative arteritis. Immunostaining revealed many IgG4-positive plasma cells diffusely distributed within nodules, and the ratios of IgG4-positive to other plasma cells were extraordinarily high. Of the 9 patients, 8 underwent surgical treatment and in 1 patient, lesion was diagnosed on transbronchial biopsy and effectively treated with corticosteroid. Two cases were associated with Chronic Sclerosing Sialadenitis or lymphadenopathy, in which many IgG4-positive plasma cells were also identified by immunostaining. The clinicopathologic similarities between IPT of the lung and Sclerosing pancreatitis suggest that IgG4-related immunopathologic processes might be involved in the pathogenesis of the pulmonary lesions.

A S C Ching - One of the best experts on this subject based on the ideXlab platform.

  • kuttner tumour Chronic Sclerosing Sialadenitis of the submandibular gland sonographic appearances
    Ultrasound in Medicine and Biology, 2003
    Co-Authors: Anil T Ahuja, Polly S Richards, K T Wong, Ann D King, H Y Yuen, A S C Ching
    Abstract:

    Patients with Kuttner tumours present with clinical features simulating a submandibular tumour. This article aims to familiarise sonologists with the sonographic appearances of these tumours, to help in their differentiation from other salivary lesions. In 13 patients with histologically or cytologically proven Kuttner tumours, the features evaluated on sonography included: shape and border of the gland, internal architecture, duct dilatation, presence/absence of calculi and presence and distribution of vascularity. There was diffuse involvement of the submandibular glands in 11 patients and focal involvement in 2. The majority (9 of 11) of the patients with diffuse involvement showed sonographic appearances simulating a "cirrhotic" liver; 2 showed diffuse heterogeneous involvement with duct dilatation and calculus. Doppler showed prominent intraglandular vessels, with no evidence of displacement. Focal lesions (2 of 11) were seen as hypoechoic, heterogeneous "masses," with a radial branching vascular pattern within, on Doppler. In conclusion, sonographic features may help in identifying Kuttner tumours of the submandibular glands.

Hideki Niwa - One of the best experts on this subject based on the ideXlab platform.

  • igg4 positive plasma cells in inflammatory pseudotumor plasma cell granuloma of the lung
    Human Pathology, 2005
    Co-Authors: Yoh Zen, Satoshi Kitagawa, Hiroshi Minato, Hiroshi Kurumaya, Kazuyoshi Katayanagi, Shinji Masuda, Hideki Niwa, Masaki Fujimura, Yasuni Nakanuma
    Abstract:

    The association between IgG4 dysregulation and inflammatory pseudotumor (IPT) was first reported in Sclerosing pancreatitis. Recently, we described IPTs of the liver and breast, into both of which many IgG4-positive plasma cells had infiltrated. In this study, we examined the clinical and histological features of 9 cases of IPT (histologically corresponding to plasma cell granuloma) of the lung with an emphasis on IgG4-positive plasma cell infiltration. The lesions were characterized histologically by dense lymphoplasmacytic infiltrates intermixed with fibrosis and, in some cases, prominent eosinophilic infiltration, irregular narrowing of bronchioles entrapped in nodules, and an interstitial pneumonia pattern at the boundaries of nodules. Obliterative phlebitis was easily found in all cases, and 5 lesions also had obliterative arteritis. Immunostaining revealed many IgG4-positive plasma cells diffusely distributed within nodules, and the ratios of IgG4-positive to other plasma cells were extraordinarily high. Of the 9 patients, 8 underwent surgical treatment and in 1 patient, lesion was diagnosed on transbronchial biopsy and effectively treated with corticosteroid. Two cases were associated with Chronic Sclerosing Sialadenitis or lymphadenopathy, in which many IgG4-positive plasma cells were also identified by immunostaining. The clinicopathologic similarities between IPT of the lung and Sclerosing pancreatitis suggest that IgG4-related immunopathologic processes might be involved in the pathogenesis of the pulmonary lesions.

  • abundant igg4 positive plasma cell infiltration characterizes Chronic Sclerosing Sialadenitis kuttner s tumor
    The American Journal of Surgical Pathology, 2005
    Co-Authors: Satoshi Kitagawa, Kenichi Harada, Motoko Sasaki, Yasunori Sato, Hiroshi Minato, Kishichiro Watanabe, Hiroshi Kurumaya, Kazuyoshi Katayanagi, Shinji Masuda, Hideki Niwa
    Abstract:

    Chronic Sclerosing Sialadenitis (CSS) is a cryptogenic tumor-like condition of the salivary gland(s). While immunemediated processes are suspected in its pathogenesis, and CSS is occasionally reported to be associated with Sclerosing pancreatitis, an IgG4-related disease, the exact immunopathologic processes of CSS remain speculative. In this study, we examined the clinicopathologic findings of CSS (12 cases) in comparison with sialolithiasis (8 cases) and Sjogren's syndrome (13 cases), and tried to clarify whether CSS is an IgG4-related disease or not. Submandibular gland(s) were affected in all cases of CSS. CSS cases could be divided into two types: 5 cases were associated with Sclerosing lesions in extrasalivary glandular tissue (systemic type), while only salivary gland(s) were affected in the remaining 7 cases (localized type). In the former type, which showed male predominance, bilateral salivary glands were frequently affected, and eosinophilia and elevations of γ-globulin and IgG in serum were frequently found. Histologically, all cases of CSS showed marked lymphoplasmacytic infiltration admixed with fibrosis and the destruction of glandular lobules. Obliterative phlebitis was found in the affected salivary glands in all cases of CSS. Immunohistochemically, the proportion of IgG4/IgG-positive plasma cells was more than 45% in CSS, while it was less than 5% in controls. The resemblance of the clinicopathologic features of CSS with those of Sclerosing pancreatitis suggests the participation of a similar immunopathologic process with IgG4 disturbance in CSS. The abundance of IgG4-positive plasma cells in the lesions would be useful for distinguishing CSS from other forms of Sialadenitis.

Tunyen Hsu - One of the best experts on this subject based on the ideXlab platform.