Ocular adnexal IgG4‐related disease has uniform clinicopathology

Sato, Yasuharu; Ohshima, Koh Ichi; Ichimura, Kouichi; Sato, Masakazu; Yamadori, Ichiro; Tanaka, Takehiro; Takata, Katsuyoshi; Morito, Toshiaki; Kondo, Eisaku; Yoshino, Tadashi

doi:10.1111/j.1440-1827.2008.02257.x

Cited by 270 publications

(291 citation statements)

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“…[1][2][3][4][5][6][7][8][9][10] Clinical manifestations are apparent in the pancreas, bile duct, gallbladder, lacrimal gland, salivary gland, retroperitoneum, kidney, lung, and prostate, in which tissue fibrosis is pathologically induced. [1][2][3][4][5][6][7][8][9][10] Recently, many cases have been reported in Western countries, as well as in Japan. 3 However, the precise pathogenesis and pathophysiology of IgG4-related disease remain unclear.…”

Section: Discussionmentioning

confidence: 99%

“…As patients presenting with prominent fibrosis are frequently suspected of having malignant tumors, IgG4-related disease should be considered in the differential diagnosis to avoid unnecessary surgery. [1][2][3][4][5][6][7][8][9][10] Recent studies have reported that IgG4-related disease sometimes involves the systemic lymph nodes but clinicopathological characteristics have not been well documented yet. 3,4,11 In this study, we sought to clarify the clinicopathologic features of systemic IgG4-related lymphadenopathy.…”

Section: Discussionmentioning

confidence: 99%

“…We recently reported the first case of marginal zone B-cell lymphoma arising from ocular adnexal IgG4-related disease 7 and IgG4-producing marginal zone B-cell lymphoma. 32 However, the present series showed no immunoglobulin light-chain restriction and no immunoglobulin heavy chain gene rearrangement.…”

Section: Discussionmentioning

confidence: 99%

“…Such abnormalities include an elevated serum IgG4 level and numerous IgG4-positive plasma cells infiltrating the affected tissue, and these disorders are classified as IgG4-related diseases. [1][2][3][4][5][6][7][8][9][10][11] Castleman's disease is a rather rare atypical lymphoproliferative disorder 12 classified according to the histopathologic findings of the affected lymph nodes as plasma cell type, hyaline-vascular type or a mixed-type variant of the two. 13,14 Patients with the plasma cell or the mixed-type variant frequently have systemic manifestations (so-called multicentric Castleman's disease), such as low-grade fever, fatigue, loss of appetite, and weight loss.…”

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confidence: 99%

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Systemic IgG4-related lymphadenopathy: a clinical and pathologic comparison to multicentric Castleman's disease

Kojima²,

et al. 2009

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 99%

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Systemic IgG4-related lymphadenopathy: a clinical and pathologic comparison to multicentric Castleman's disease

Kojima²,

et al. 2009

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“…Some case reports have showed effectiveness of methotrexate (105), cyclosporine (106), and cyclophosphamide (106,107).…”

Section: Steroid-sparing Immunomodulatorsmentioning

confidence: 99%

IgG4-related disease: A relatively new concept for clinicians

Vasaitis

2016

European Journal of Internal Medicine

105

129

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IgG4-related disease (IgG4-RD) is a recently recognized chronic fibrotic inflammation, which can affect almost every organ, and may come to clinical attention first due to visible organ swelling or organ dysfunction, or is identified incidentally by imaging and specific biopsy.The disorder has an allergic background and is immune-mediated. Up-regulated responses of T helper 2 and T regulatory cells and their cytokines play a major role in disease progression.About 30-50% of patients are atopic or have mild eosinophilia. IgG4-RD predominantly affects middle-aged male patients. The cornerstones of diagnosis of the disease are compatible clinical features and typical histopathology. Swelling of salivary and lacrimal glands, lymphadenopathy, and type 1 autoimmune pancreatitis (AIP) are the most common manifestations of the disease. However, other tissues and organs, such as retroperitoneum, lung, kidney, aorta, upper airways, thyroid gland, meninges, heart, mesenterium and skin may be involved. Typical histopathology is lymphoplasmacytic infiltration abundant in IgG4-positive plasma cells, storiform-type fibrosis, and obliterative phlebitis. Elevated serum IgG4 concentration supports the diagnosis. Characteristic imaging features such as a "capsule-like rim" surrounding the pancreatic lesions is highly specific to type 1 AIP. 18F-fluorodeoxyglucose positron emission tomography/computed tomography enables mapping the sites of inflammation, permits evaluation of the extent of the disease, helps in guiding biopsy decision, and may be used in monitoring response to treatment. Glucocorticoids alone or in combination with B-cell depletion with rituximab induces prompt clinical response to IgG4-RD.This article reviews the current understanding, different clinical manifestations, and approaches to diagnosis and treatment of IgG4-RD. 3Highlights Ø IgG4-related disease is a chronic fibrotic inflammation.Ø The disease affects multiple organs, is progressive, and impairs function.Ø The disorder has an allergic background and is immune-mediated.Ø Compatible clinical signs and typical pathology are the basis for the diagnosis.Ø Glucocorticoids and/or B-cell depletion induces prompt clinical response.Immunoglobulin G4-related disease (IgG4-RD) is a chronic fibrotic inflammation, characterized by tissue infiltration by lymphocytes, IgG4-positive plasma cells, simultaneous development of fibrosis, and often, but not always, elevated serum levels of IgG4 (1, 2). The disease can affect multiple organs. Early diagnosis of IgG4-RD is not easy because of the indolent course of the disease with vague, nonspecific symptoms. It may first come to clinical attention as visible organ swelling or detected organ dysfunction due to fibrosis, or is identified incidentally by imaging and specific biopsy when the patient undergoes investigation of suspected tumor disease. Most forms of organ involvement in IgG4-RD, even in the advanced fibrotic stages, respond well to glucocorticoids (3, 4). 4A comprehensive search for all articles in E...

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IgG4-Positive Dacryoadenitis and Küttner Submandibular Sclerosing Inflammatory Tumor

Jakobiec

2010

Arch Ophthalmol

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topical caspofungin was progressively decreased over 3 further weeks. Following the completion of treatment, complete healing of the corneal epithelium and resolution of the corneal infiltrate were observed, although the corneal opacity persisted (Figure, D). There was no evidence of ocular toxic effects and no recurrence of fungal keratitis over a follow-up period of 6 months. Comment. C albicans is the most frequent cause of fungal keratitis in temperate regions and is an opportunistic organism that can complicate chronic keratopathy and corneal grafting. Persistent epithelial defects and suture-related problems, along with immunosuppression, have been found to be the major predisposing risk factors. Caspofungin is a first-in-class echinocandin with potent activity against Candida and Aspergillus, the dominant human fungal pathogens. In contrast to all other antifungal drugs, echinocandins have a selective action on a target present only in fungal cell walls (not in mammalian cells): they inhibit the synthesis of an essential component, (1,3)-D-glucan. Caspofungin is fungicidal in vivo and in vitro against all Candida species, including fluconazole-resistant strains. Its activity differs from that of the azole antifungal group, which is fungistatic. 4 In our case, we think that voriconazole stopped progression of the infiltrate but did not kill the microorganism. The presence of the fungus after 1 month could be due to poor drug penetration, fungal resistance, or both. We therefore suggest that an ideal treatment protocol should include antifungal agents chosen on the basis of in vitro susceptibility of the fungus with a duration assessed by in vivo monitoring of fungal filaments or yeasts. To our knowledge, the topical ocular use of caspofungin has been reported in rabbits. 5 There has been only 1 report of its use in humans, although it was in association with other antifungal drugs. 4 In conclusion, topical caspofungin, 0.5%, is a new, promising option in the treatment of refractory fungalrelated corneal ulcers with no evidence of ocular toxic effects. However, future studies with larger samples are indicated to further evaluate its efficacy and tolerance.

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Ocular adnexal IgG4‐related disease has uniform clinicopathology

Cited by 270 publications

References 23 publications

Systemic IgG4-related lymphadenopathy: a clinical and pathologic comparison to multicentric Castleman's disease

Systemic IgG4-related lymphadenopathy: a clinical and pathologic comparison to multicentric Castleman's disease

IgG4-related disease: A relatively new concept for clinicians

IgG4-Positive Dacryoadenitis and Küttner Submandibular Sclerosing Inflammatory Tumor

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