Clinical Case Notes

Koppens, Joanna M; Pon, Jo-Anne M C; Allen, Jonathan P.; Sloan, Brian

doi:10.1046/j.1442-9071.2004.00771.x

Cited by 10 publications

(4 citation statements)

References 6 publications

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“…MCD is a systemic lymphoproliferative disorder that can lead to mass lesion formation in various parts of the body, including the lungs, kidneys, and extranodal sites. Although orbital tissue involvement is relatively rare due to the lack of lymphatic components, previous cases of MCD affecting the lacrimal gland have been reported [ 8 , 9 ]. Venizelos et al reported that orbital involvement might be the initial clinical presentation of CD and those with unicentric hyaline-vascular CD cases showed no association with systemic symptoms and were rarely recurrent [ 10 ].…”

Section: Discussionmentioning

confidence: 99%

A Rare Case of Orbital Castleman Disease with Overlapping IgG4-Related Disease

et al. 2023

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Multicentric Castleman disease (MCD) is a systemic lymphoproliferative disorder that can lead to mass lesions in various body parts, including the lungs, kidneys, and extranodal sites. Meanwhile, orbital Castleman disease is extremely rare. Immunoglobulin G4-related disease (IgG4-RD) is a recently recognized fibroinflammatory disorder and is characterized by the formation of tumor-like lesions with lymphoplasmacytic infiltrates, which are enriched in IgG4-positive plasma cells and may present with a characteristic storiform pattern of fibrosis to variable degrees. In this study, we report a case of a 67-year-old Taiwanese man with a 7-year history of bilateral eyelid swelling and proptosis. Orbital magnetic resonance imaging revealed soft tissue lesions in the bilateral intraconal region, demonstrating strong enhancement in the lacrimal glands, and extension into the bilateral infraorbital foramen, suggesting an orbital lymphoproliferative disease. The histopathological results of the intraorbital tumor excision were suggestive of a plasma-cell-predominant mixed-cell variant of MCD. However, the patient also showed definitive signs of IgG4-RD, including lacrimal gland enlargement and histopathological results of plasmacytosis, fibrosis, and germinal centers, with an increased ratio of IgG4 cells and elevated serum IgG4 levels. This case suggests a potential interacting pathway between these two disease entities that needs further studies.

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Section: Discussionmentioning

confidence: 99%

A Rare Case of Orbital Castleman Disease with Overlapping IgG4-Related Disease

et al. 2023

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“…Castleman disease of the lacrimal glands is extremely rare, but unfortunately the serum IgG4 levels have not been reported in the earlier cases. 7 – 10 Because the signs and the symptoms of both diseases may overlap, clinicians should examine the serum IgG4 levels in patients with Castleman disease routinely. 2 – 5…”

Section: Discussionmentioning

confidence: 99%

Relationship between chronic sclerosing dacryoadenitis with high level of IgG4 and Castleman disease

Oshitari¹,

Yotsukura²,

Asahagi³

et al. 2010

OPTH

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The purpose of this study is to present a case of chronic sclerosing dacryoadenitis with high level of IgG4 in a patient diagnosed earlier with Castleman disease. A 79-year-old man noticed a swelling of his lower left jaw that was first seen 8 years earlier. He was diagnosed with Castleman disease from the histopathological examination of a biopsy of the submandibular gland. Since then, the size of the gland had not changed, and he had no systemic inflammatory signs or symptoms. He developed diplopia a year earlier, and CT scans showed bilateral swelling of the lacrimal glands. He was referred to our hospital for further examinations. The patient underwent partial dacryoadenectomy. From the histopathological examinations, he was diagnosed with chronic sclerosing dacryoadenitis with high level of the serum IgG4. He underwent oral steroid therapy and the swollen lacrimal glands were significantly improved. The results suggest that there may be pathological links between IgG4-related dacryoadenitis and Castleman disease.

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“…Unicentric CaD confined to the orbits has been successfully treated with surgical excision in multiple cases [195][196][197][198][199]. Algorithms for the treatment of multicentric CaD have not been established.…”

Section: Castleman's Diseasementioning

confidence: 99%