Isolated Conjunctival Inflammation Suggestive of IgG4-Related Disease

Aziz, Hassan A.; Villa‐Forte, Alexandra; Plesec, Thomas; Singh, Arun D.

doi:10.1159/000437308

Cited by 7 publications

(5 citation statements)

References 13 publications

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“…IgG4-ROD mainly involves the lacrimal gland, extraocular muscles and trigeminal nerve; the optic nerve, conjunctiva, sclera, uvea, and choroid are rarely involved [4][5][6][7][8]. Despite the rarity of conjunctival involvement in IgG4-ROD [6][7][8], this paper reports a another very rare case of LC with strong positive expression of IgG4 in histopathological results with only one published case in literature.…”

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confidence: 79%

“…According to Japanese studies, the incidence of IgG4-RD is 0.28-1.08/100,000, and that of IgG4-related ophthalmic disease (IgG4-ROD) accounts for 4-34% of total IgG4-RD cases [3]. IgG4-ROD mainly involves the lacrimal gland, extraocular muscles and trigeminal nerve; the optic nerve, conjunctiva, sclera, uvea, and choroid are rarely involved [4][5][6][7][8]. Despite the rarity of conjunctival involvement in IgG4-ROD [6][7][8], this paper reports a another very rare case of LC with strong positive expression of IgG4 in histopathological results with only one published case in literature.…”

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confidence: 99%

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A case of IgG4-positive ligneous conjunctivitis mistaken for a conjunctival mass

Li,

Liu,

Ren

et al. 2023

Diagn Pathol

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Background Ligneous conjunctivitis (LC) is a rare inflammatory lesion of the conjunctiva with an unknown etiology. It is easily confused with conjunctiva lymphoma or other diseases in clinical diagnosis, and the lesion is very difficult to treat. Case presentation We presented a 41-year-old female patient presented with bilateral conjunctival masses for more than six months. The patient had no contributory history of ocular trauma, family history of tumor and drug allergy. Taking the patient’s clinical and pathological features together, we considered this was a case of IgG4 + LC. Completely surgical resection combined with local glucocorticoid treatment might be effective. Conclusions This is a very rare case report of immunoglobulin G4 positive LC with only one published case in literature. The typical manifestations of LC are with the appearance of a hard, fibrin-rich, woody pseudomembranous lesion. A large number of lymphocyte and plasma cell are infiltrated in the pathological tissue. Inflammation of LC may cause immune abnormalities, resulting in IgG4 increasing.

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confidence: 79%

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confidence: 99%

A case of IgG4-positive ligneous conjunctivitis mistaken for a conjunctival mass

Li,

Liu,

Ren

et al. 2023

Diagn Pathol

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“…The most involved sites of IgG4-ROD were well established, including lacrimal glands, extra-ocular muscles, orbital soft tissue, and infraorbital nerves [ 12 ]. There were few reports of IgG4-ROD suspected in the isolated lesions of the eyelid and conjunctiva [ 9 , 13 , 14 , 15 ]. Nevertheless, there was no previous report of IgG4-ROD speculated in sweat gland cysts or lesions of other folliculosebaceous unit origins like ours.…”

Section: Discussionmentioning

confidence: 99%

Apocrine Hidrocystoma with IgG4 Plasma Cell Infiltration Presenting as Recurrent Chalazion: A Case Report

Huang

Lee

et al. 2022

Medicina

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Apocrine hidrocystomas are benign cystic tumors resulting from apocrine sweat glands’ proliferation. They typically present as solitary, slow-growing nodules at the head and neck, especially in the periorbital cutaneous region. We present a case of periorbital apocrine hidrocystoma in a 22-year-old woman that was treated as chalazion previously. Besides the hallmark histopathological findings of apocrine hidrocystoma, IgG4 plasma cell infiltration of the cystic wall was also found. The ratio of IgG4-to-IgG-positive plasma cells was high, whereas serum IgG4 was within the standard limit. This is, to date, the only probable IgG4-related ophthalmic disease associated with apocrine hidrocystoma.

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“…It is well-established that IgG4 can be involved in more than one-third of idiopathic inflammation of orbital tissue. It has been shown that higher systemic manifestations of IgG4-RD can be observed in impaired adnexal involvement [11]. These can include changes in immunological and biochemical parameters.…”

Section: Clinical Observationsmentioning

confidence: 99%

“…Additional IgG4-RDs have been identified, based on systemic fibroinflammatory disease characterized by the presence of lesions with lymphoplasmacytic infiltrate with high levels of IgG4-positive plasma cells. In addition,a correlation between higher levels of IgG4 and good response to therapy with corticoids has been observed [2,[6][7][8][9][11][12][13][14][15][16][17][18]. Hamano et al, [19,20] observed not only elevated levels of IgG4 in autoimmune pancreatitis, but also found characteristic histopathological manifestations accompanying retroperitoneal fibrosis.…”

Section: Introductionmentioning

confidence: 99%

Orbital IgG4-associated diseases

Závorková¹,

Richter²,

Větvička³

et al. 2017

Pathol Discov

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IgG4-related disease (IgG4-RD) is a distinct entity that frequently occurs in an ophthalmic location. IgG4-RD is not limited to the orbit, but may also involve other anatomical structures in and around the eye. Careful clinicoradiologic examination and the use of immunohistological examination are key diagnostic methods. Serum IgG4 levels are neither sensitive nor specific enough for the diagnosis of IgG4-RD and should not be relied upon solely. Careful evaluation of histologic and immunophenotypic features and clinical correlation are required to distinguish orbital IgG4-RD from other inflammatory lesions in the orbit. Glucocorticoids are the primary therapeutic choice for treatment of IgG4-RD. Azathioprine or Mofetil can be used as a second possibility. Rituximab can be effective in the patients with relapse IgG4-RD.

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Isolated Conjunctival Inflammation Suggestive of IgG4-Related Disease

Cited by 7 publications

References 13 publications

A case of IgG4-positive ligneous conjunctivitis mistaken for a conjunctival mass

A case of IgG4-positive ligneous conjunctivitis mistaken for a conjunctival mass

Apocrine Hidrocystoma with IgG4 Plasma Cell Infiltration Presenting as Recurrent Chalazion: A Case Report

Orbital IgG4-associated diseases

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