Immunoglobulin G4-Related Ophthalmic Disease Involving the Sclera Misdiagnosed as Intraocular Tumor: Report of One Case

Gotô, Hiroshi; Ueda, Shunichiro

doi:10.1159/000447405

Cited by 8 publications

(3 citation statements)

References 9 publications

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“…Immunoglobulin G4-related ophthalmic disease (IgG4-ROD) is a relatively common entity of Immunoglobulin G4 (IgG4)related disease (IgG4-RD). [1] Although the involved tissues include the lacrimal gland, orbital fat, extraocular muscles, trigeminal nerve, eyelid, sclera, lacrimal sac, and optic nerve, [1][2][3][4] the lacrimal gland involvement (dacryoadenitis) is most commonly encountered. [1] Concomitant extra-ophthalmic IgG4-RD is sometimes seen.…”

Section: Introductionmentioning

confidence: 99%

Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report

et al. 2023

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Purpose: Immunoglobulin G4 (IgG4)-related dacryoadenitis is rarely resolved spontaneously without steroids. Here, we report a case of IgG4-related dacryoadenitis and extra-ophthalmic lesions with spontaneous regression.Methods: This is a clinical case report. A 56-years-old man had a 1-year and 7-month-old histories of neck and eyelid swelling, respectively. On the first examination, the lacrimal and submandibular glands were palpable bilaterally. Computed tomographic images showed enlargement of the lacrimal gland on both sides, right pulmonary hilar lymph node, and pancreas, and thickening of the abdominal aortic wall. Blood tests demonstrated elevated serum IgG4 level and positive hepatitis B surface antibody. Pathological examination of the biopsied lacrimal gland specimens revealed marked IgG4-positive plasma cell infiltration.Results: The patient was monitored carefully without steroid administration. Serum IgG4 level had gradually decreased during follow-up period and reached the normal range 3 years after the biopsy. At 4-year follow-up, the lacrimal and submandibular glands were not palpable on either side. Computed tomographic images demonstrated no enlargement of the lacrimal gland, submandibular gland, or lymph nodes, and improvement of the enlarged pancreas and thickened abdominal aortic wall. Conclusion:Our case indicates that careful observation can be an option in selected cases with risks of steroid treatment or silent clinical course.

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Section: Introductionmentioning

confidence: 99%

Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report

et al. 2023

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“…The positive rates of T-cell markers CD2, CD3, CD4, CD5, CD7, and CD8 along with other cell surface markers, such as CD10, CD23, CD38, and CD138, are generally high in IgG4-ROD, while there is low expression of CD25 and CD19 [31, 32]. In addition, their B-cell marker (CD79) is also positive [33].…”

Section: Introductionmentioning

confidence: 99%

Novel Advances in the Study of IgG4-Related Disease in the Eye and Ocular Adnexa

Wang²,

Tian

et al. 2022

Ophthalmic Res

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Immunoglobin (IgG4)-related disease in the eye and ocular adnexa (IgG4-ROD) is a newly discovered autoimmune disease that histologically exhibits extensive lymphocyte and plasma cell infiltration, occlusive phlebitis and mat or whorled fibrosis. The disease can affect multiple ocular tissues and organs, such as the lacrimal gland, extraocular muscles, orbital fat and trigeminal nerve. The main clinical manifestations are chronic, painless swelling of the orbit or unilateral orbit and proptosis, which may be accompanied by peripheral lymphadenopathy. Usually, visual impairment is not apparent, but in severe cases, it can cause a loss of function of the tissues and organs involved and affect the daily lives of patients. The pathogenesis of IgG4-ROD is not clear. Based on existing literature, it is speculated that it may be related to factors such as autoantibody production, microbial infection and genetic inheritance. For the treatment of IgG4-ROD, glucocorticoids, immunosuppressive agents, biological agents and surgery are mainly used in clinical practice. Although these treatment methods can achieve a particular effect, they have limitations, such as high recurrence rates, serious side effects and postoperative complications. With the increase in IgG4-ROD-related reports, some progress has been made in the current understanding and research of the disease.

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“…The current approach to the diagnosis of IgG4-RD is based on a combination of comprehensive and organspecific diagnostic criteria. Concerning the kidney, the Mayo clinic classification and the guidelines of the Japanese Nephrology Society are applied [7][8][9].…”

Section: Introductionmentioning

confidence: 99%

A case report of IgG4-related disease: an insidious path to the diagnosis through kidney, heart and brain

et al. 2019

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Background: IgG4-related disease, described around the years 2000 as a form of autoimmune pancreatitis, is now increasingly accepted as a systemic syndrome. The diagnosis is based on both comprehensive and organ-specific criteria. For the kidney, Mayo clinic classification and the guidelines of the Japanese Nephrology Society are used. Ultimately, together with parameters that characterize every organ or apparatus involved, the key element is the confirmation of growing levels of IgG4 in blood or in tissues. Case presentation: We describe a male patient with chronic renal failure associated to hypertension without proteinuria. IgG4-related disease was diagnosed through renal biopsy. After an initial positive response to steroids, he presented tinnitus, and histological assessment showed cerebral and subsequently cardiac damage, both IgG4related. This case appears unique for the type of histologically documented cardiac and neurological parenchymal involvement, and at the same time, exemplifies the subtle and pernicious course of the disease. Frequently, blurred and non-specific signs prevail. Here, kidney damage was associated with minimal urinary findings, slowly progressive renal dysfunction and other factors that can be equivocated in the differential diagnosis. Neurological involvement was represented by tinnitus alone, while cardiac alterations were completely asymptomatic. Conclusions: This report is representative of the neurological and cardiac changes described in the literature for IgG4-related disease, which may be correlated or not with the renal form and highlights the need, in some cases, of targeted therapeutic approaches. In addition to glucocorticoids, as in this case, rituximab may be necessary.

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Immunoglobulin G4-Related Ophthalmic Disease Involving the Sclera Misdiagnosed as Intraocular Tumor: Report of One Case

Cited by 8 publications

References 9 publications

Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report

Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report

Novel Advances in the Study of IgG4-Related Disease in the Eye and Ocular Adnexa

A case report of IgG4-related disease: an insidious path to the diagnosis through kidney, heart and brain

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