Supraglottic immunoglobulin-G4 related plasma cell granuloma: Case report and literature review

Khoo, Jeremy F.; Batt, Michael; Stimpson, Paul; Safdar, Adnan

doi:10.1002/hed.23519

Cited by 22 publications

(27 citation statements)

References 11 publications

(21 reference statements)

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“…Upper respiratory tract presentation of the disease is rare (Table I) [3,8,9,11,12,13,14,15,16]. A review of the available literature showed only single cases of IgG4-RD of the oral cavity with presentation in the palate, tongue, floor of the mouth, and upper and lower gingiva [3,8,9].…”

Section: Discussionmentioning

confidence: 99%

IgG4-related disease in the head and neck region: report of two cases and review of the literature

Gontarz¹,

Wyszyńska-Pawelec²,

Zapała³

et al. 2016

pjp

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IgG4-related disease (IgG4-RD) is a rare immune-mediated condition characterized by extensive tissue fibrosis and infiltration by immunoglobulin G4 positive plasma cells in a single organ or systemic appearance. Two cases are presented including an unusual case of a 30-year-old man with IgG4-RD appearing simultaneously in the cervical lymph nodes, ethmoid, maxillary sinuses, and upper gingiva, with spontaneous loss of teeth. According to the literature, this is the first case with loss of teeth occurring in the course of the disease. The second case is a 46-year-old man suffering from IgG4-related chronic sclerosing sialadenitis of the right submandibular gland.

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Section: Discussionmentioning

confidence: 99%

IgG4-related disease in the head and neck region: report of two cases and review of the literature

Gontarz¹,

Wyszyńska-Pawelec²,

Zapała³

et al. 2016

pjp

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“…13 IgG4-related disease has been observed in nearly every organ, including the biliary system, thyroid, salivary glands, kidneys, lungs, skin, lymph nodes, and orbital tissues. 14 Airway involvement has been described in the nasal cavity, trachea, and lungs 8,10,15,16 and seldom in the larynx, 7,17 with no reports of isolated pharyngeal involvement. In addition, previously named entities such as Mikulicz's syndrome, Küttner's tumor, Riedel's thyroiditis, and inflammatory pseudotumor (including orbital pseudotumor) have now been recognized to be a part of the IgG4-RD spectrum.…”

Section: Discussionmentioning

confidence: 99%

Clinical Manifestations of IgG4-Related Disease in the Pharynx

Reder

Della‐Torre

Stone

et al. 2014

Ann Otol Rhinol Laryngol

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“…We identified 11 reports describing cases of laryngotracheal involvement due to IgG4-RD [16][17][18][19][20][21][22][23][24][25][26], 2 of them written in a non-English language [22,25]. Table 1 includes an update of the published cases written in English [16-21, 23, 24, 26].…”

Section: Resultsmentioning

confidence: 99%

“…We found a total of 12 patients with laryngopharyngeal involvement, most of them adult men. The most common sites of IgG4 + plasma cell infiltration were the pharynx [16,19,21] and the supraglottic region of the larynx [17,20,21,24]. Treatment strategies included local surgery and immunosuppressive drugs.…”

Section: Resultsmentioning

confidence: 99%

Atypical presentation of immunoglobulin G4-related disease as subglottic stenosis: a case-based review

et al. 2021

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Background Immunoglobulin G4-related disease (IgG4-RD) is a recently recognized fibro-inflammatory pathology that has been reported to affect principally the retroperitoneum, hepatobiliary system, salivary glands, orbital structures or lymph nodes. However, IgG4-RD with laryngeal involvement is a very rare entity. Our aims were to describe a case of subglottic stenosis as first and only manifestation of IgG4-RD and review the literature. A patient with IgG4-RD affecting the larynx that presented as subglottic stenosis is described. A MEDLINE database search of IgG4-RD cases with laryngopharyngeal manifestations was also conducted. A 30-year-old Caucasian woman was referred to a tertiary care hospital for dyspnea on exertion, which had been increasing for the last 4 months. Medical and surgical procedures revealed a subglottic stenosis, with a histological finding of IgG4 positive plasma cell infiltration. There was no evidence of other organ involvement. She was successfully treated with oral glucocorticoids and rituximab infusions. Glucocorticoids were rapidly tapered and the rituximab regimen was optimized, with no evidence of relapses. In the literature review, we found a total of 12 reported cases with laryngopharyngeal involvement, two of them with subglottic stenosis. IgG4-RD of the larynx is rare but should be considered after excluding more common disorders.

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Supraglottic immunoglobulin-G4 related plasma cell granuloma: Case report and literature review

Cited by 22 publications

References 11 publications

IgG4-related disease in the head and neck region: report of two cases and review of the literature

IgG4-related disease in the head and neck region: report of two cases and review of the literature

Clinical Manifestations of IgG4-Related Disease in the Pharynx

Atypical presentation of immunoglobulin G4-related disease as subglottic stenosis: a case-based review

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