Immunoglobulin G4 related systemic sclerosing disease involving the temporal bone

Masterson, Liam; Pero, Marcos Martinez Del; Donnelly, Neil; Moffat, David A.; Rytina, E.

doi:10.1017/s0022215110001143

Cited by 31 publications

(50 citation statements)

References 26 publications

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“…7 However, there are few reports on the otologic manifestations of IgG4-related disease, and the clinical features of IgG4-related disease with ear involvement remain unknown. [8][9][10] Difficulty in diagnosis often delays the initiation of treatment, and the disease occasionally progresses to the irreversible phase. Here, we report cases of IgG4-related disease that presented with mixed hearing loss and discuss the clinical features of the otologic manifestations associated with IgG4-related disease.…”

Section: Introductionmentioning

confidence: 99%

Otologic Manifestations of Immunoglobulin G4-Related Disease

Takagi

Nakamaru

Fukuda

2014

Ann Otol Rhinol Laryngol

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Objective: Immunoglobulin (Ig) G4-related disease is systemic, and it has been reported that patients with IgG4-related disease complain of symptoms involving numerous organs. However, there are few reports concerning the otologic manifestations of IgG4-related disease. The purpose of this study is to investigate the clinical features of the otologic manifestations in IgG4-related disease. Methods: We recruited 39 consecutive patients diagnosed with IgG4-related disease. Otologic symptoms, laboratory data, and audiogram findings were retrospectively examined. Mucosal tissues from the inferior turbinate were obtained from subjects before treatment. The serum IgG4 and eosinophil levels together with clinical features were analyzed. Results: Five of the 39 cases had some otologic symptoms. Otitis media with effusion was present in 2 patients. Sensorineural hearing loss was also present in 1 patient. Eosinophilic otitis media was present in 2 patients with bilateral rhinosinusitis and bronchial asthma, and elevated serum eosinophil levels. Oral prednisolone was effective in the treatment of IgG4-related disease. Conclusion: We revealed a new clinical entity associated with the otologic manifestations of IgG4-related disease.

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Section: Introductionmentioning

confidence: 99%

Otologic Manifestations of Immunoglobulin G4-Related Disease

Takagi

Nakamaru

Fukuda

2014

Ann Otol Rhinol Laryngol

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“…The pathogenesis of IP is still obscure but immunological and infectious causes have been postulated [10, 27]. Recently, some IPs have been found to be associated with IgG4-related sclerosing diseases [28, 29], and Masterson et al [28] recommended a full review of all histological specimens in patients with a diagnosis of IP or inflammatory myofibroblastic tumor. Consideration should be given to staining for IgG4, together with measurement of serum levels.…”

Section: Discussionmentioning

confidence: 99%

“…Clinical manifestations vary widely among cases as indicated by our and previous reports. Laboratory abnormalities include anemia, thrombocytosis, polyclonal hypergammaglobulinemia, and elevated ESR, CRP, or IgG4 [1, 28]. CT scan usually shows erosive soft-tissue mass in the middle ear and/or mastoid with occasional extensions into adjacent structures.…”

Section: Discussionmentioning

confidence: 99%

Inflammatory Pseudotumor of the Temporal Bone: Three Cases and a Review of the Literature

Tian

Liu

Tang

et al. 2013

Case Reports in Medicine

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Inflammatory pseudotumor (IP) is a clinically aggressive but histologically benign condition of unknown cause. Its appearance in the temporal bone is uncommon. We present clinical, radiological, and histopathologic findings of three cases originating in the temporal bone. In the first case, a simultaneous IP of the temporal bone and parotid gland was found with histopathologic confirmation. In the second case, an enlarged cervical node, which was also believed to be related to IP, was observed accompanied with the temporal lesion. While the third case presented with chronic suppurative otitis media. Two of them were treated by surgery alone with complete resolve of the diseases. Another one underwent tympanomastoidectomy in combination with oral steroids, radiation, and chemotherapy, but the IP still recurred. A comprehensive review of the literature on clinical features of the temporal pseudotumor was conducted.

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“…2 Although IgG4related sialadenitis was thought to be a localized chronic inflammatory disease of unknown etiology for much of the past century, more recent studies have shown that it may be a feature of IgG4-related disease. 1,6,7 IgG4-related sialadenitis was recently recognized as part of the spectrum of IgG4related disease in both Asian and Western populations. [1][2][3][4] IgG4related disease involvement of the head and neck has been much less commonly reported, with only 3 prior publications in the otolaryngology head and neck surgery literature (to our knowledge).…”

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confidence: 99%

“…3 Salivary gland dysfunction, if any, is usually very mild in IgG4-related sialadenitis. In the head and neck, IgG4-related disease most frequently affects the salivary gland, but it may also affect the lacrimal gland (commonly in association with salivary gland involvement), 6,7 thyroid, or macula and uvea of the eye. In the head and neck, IgG4-related disease most frequently affects the salivary gland, but it may also affect the lacrimal gland (commonly in association with salivary gland involvement), 6,7 thyroid, or macula and uvea of the eye.…”

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confidence: 99%