An unusual presentation of chronic sclerosing sialadenitis of submandibular gland (Kuttner’s tumour)

Lakshmanan, Somu; Manimaran, Vinoth; Valliappan, Valliammai; Arumugam, Vasugi Gramani

doi:10.1136/bcr-2019-231189

Cited by 6 publications

(4 citation statements)

References 9 publications

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“…However, we detected IgG4-positivity in samples from patients with sialoliths as well. Findings somewhat similar to those in our series have, however, also been reported in a small cohort of six patients 4 , and in a case report 17 , where a sialolith was reported in patients with IgG4-related chronic sclerosing sialadenitis. Elevated numbers of IgG4-positive plasma cells have also been reported in other nonspecific chronic inflammatory conditions of the oral cavity, such as apical granulomas, plasma cell gingivitis, and oral lichen ruber planus 18 .…”

Section: Discussionsupporting

confidence: 92%

IgG4-positive plasma cells in nonspecific sialadenitis and sialolithiasis

et al. 2022

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Chronic sclerosing sialadenitis is commonly regarded as a manifestation of IgG4-related disease. We previously found that a high IgG4 expression or IgG4-related disease could accompany nonspecific sialadenitis, whereas chronic sclerosing sialadenitis was not directly associated with IgG4-related disease. Our previous findings lead us to hypothesize that these inflammatory conditions of the submandibular gland signify a continuous progression of disease rather than different disease entities. We, therefore, aimed to determine the presence of IgG4-positivity and genuine IgG4-related disease in a cohort of 165 submandibular gland specimens from patients who underwent surgery due to chronic nonspecific sialadenitis or sialolithiasis. To do so, we re-evaluated histopathological features and divided samples into three groups: (A) nonspecific sialadenitis without known sialolithiasis, (B) sialadenitis with sialolithiasis, and (C) sialolithiasis without sialadenitis. We performed immunohistochemical staining for IgG4, IgG, and CD31, and assessed the Boston consensus statement criteria for IgG4-related disease in IgG4-positive samples. We also reviewed patient records and supplemented follow-up data with a questionnaire among patients with IgG4-positive samples. IgG4-positive plasma cells (range 1–344) were found in 131 samples. Among these, 19 samples were classified as IgG4-positive (≥70 IgG4-positive plasma cells/high-power field). Two IgG4-positive samples were histologically highly suggestive of IgG4-related disease, but only one had a clinically confirmed diagnosis of IgG4-related disease. Our results indicate that patients with sialadenitis and sialolithiasis often present with IgG4-positive lymphoplasmacytic infiltrates, but exceedingly rarely present with genuine IgG4-related disease. In sialolithiasis without sialadenitis, IgG4-positive plasma cells are often absent or appear in small numbers. These results support our hypothesis of a continuum of disease, and indicate that progressive inflammation of the submandibular gland leads to the development of more specific pathological features over time.

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Section: Discussionsupporting

confidence: 92%

IgG4-positive plasma cells in nonspecific sialadenitis and sialolithiasis

et al. 2022

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“…The total search revealed 6366 articles on WPWS. The search yielded 24 cohorts with a preoperative or perioperative diagnosis of the WPWS in association with procedures of the head and neck/oral and maxillofacial region and have been summarized in Table 1 8–31 …”

Section: Resultsmentioning

confidence: 99%

Wolff–Parkinson–White syndrome: Review of published cases associated with head and neck/oral and maxillofacial procedures

et al. 2022

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Aim The objective of this article is to review the demographics, diagnostic criteria, etiopathogenesis, comorbidities and management of Wolff–Parkinson–White syndrome (WPWS). In addition, the relevant cardiovascular outcomes of published cases involving head and neck/oral and maxillofacial procedures were summarized. Materials and Methods A search of the PubMed database, restricted to human studies published in the English language literature, was performed from 1960 through July 2021 for articles. Results The search strategy yielded 24 patients, totalling 25 dates of treatment, with WPWS concordant with head and neck/oral and maxillofacial procedures, predominately involving biopsy, tumour resection and extractions; significantly, 5 of whom, experienced their first episode of WPWS while undergoing treatment of the head and neck. None of the procedures resulted in patient death. Conclusions Practitioners should be familiar with the clinical features of WPWS and be prepared to manage emergent cardiovascular deterioration. Electrocardiogram monitoring of this cohort is advised during head and neck/oral and maxillofacial surgical procedures, particularly involving general anaesthesia. Breakthrough episodes of WPWS may still occur in patients who have previously undergone cardioconversion.

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“…La TAC muestra un agrandamiento de la glándula comprometida así como una o múltiples zonas de hiperdensidad a consecuencia de la calcificación de la glándula, por lo general cerca del conducto salival como en la tomografía de nuestro caso, que revelaba un área hiperdensa a tejido óseo sugerente de un sialolito de gran tamaño, aunado a la importante xerostomía que refería la paciente, coincidiendo con un probable diagnóstico de sialolitiasis crónica. 3,21 El tumor de Küttner o SCE se presenta en adultos de mediana edad, los cuales suelen tener antecedentes de inflamación unilateral en el cuello y en ocasiones dolor localizado dentro de un periodo de dos a nueve meses, se manifiesta con más frecuencia en la glándula submandibular izquierda, similar a nuestro caso en cuanto a edad y tiempo de evolución. 22 Se han reportado casos de relación con enfermedad asociada con la IgG4 en estudios realizados principalmente en Asia.…”

Section: Wwwmedigraphicorgmxunclassified

“…Puede ser asintomática o mostrar odinofagia. 3 Se ha reportado baja incidencia en la glándula parótida, mientras que en la mayoría de los casos se presentan en la glándula submandibular, una teoría indica que esto se debe a que la secreción de la glándula submandibular es más mucoide y menos serosa, lo que aumenta la probabilidad de formación de cálculos. 4 Existen teorías sobre la etiología de esta enfermedad como la obstrucción del conducto salival, estasis salival, sialolitiasis, disfunción secretora y respuesta autoinmunológica.…”

Section: Introductionunclassified

Tumor de Küttner sin relación con enfermedad de IgG4: reporte de un caso

Dorantes¹,

Castañeda²,

Hernández³

et al. 2022

Revista Mexicana De Cirugía Bucal Y Maxilofacial

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Introducción: el tumor de Küttner o sialoadenitis crónica esclerosante (SCE) es una enfermedad poco común de las glándulas salivales mayores. Se caracteriza por un aumento de volumen, usualmente de manera unilateral, aunque también se ha reportado su presencia en glándulas salivales menores con o sin una relación con la enfermedad de IgG4 produciendo una infiltración fibroinflamatoria en los tabiques interlobulillares, pero se conserva su arquitectura y en algunos casos presencia de cálculos y células IgG4 positivas. Material y métodos: paciente femenino de 44 años de edad con hallazgos tomográficos de una calcificación de glándula submandibular izquierda, por lo que se realiza exéresis quirúrgica de la glándula obteniendo un diagnóstico histopatológico de tumor de Küttner. Resultados: se continúa con un seguimiento bimestral de la paciente, actualmente sin presencia de alteraciones en relación con la enfermedad IgG4. Conclusiones: el tumor de Küttner o sialoadenitis crónica esclerosante (SCE) en ocasiones puede ser el hallazgo inicial o único de la enfermedad de IgG4. El diagnóstico

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An unusual presentation of chronic sclerosing sialadenitis of submandibular gland (Kuttner’s tumour)

Cited by 6 publications

References 9 publications

IgG4-positive plasma cells in nonspecific sialadenitis and sialolithiasis

IgG4-positive plasma cells in nonspecific sialadenitis and sialolithiasis

Wolff–Parkinson–White syndrome: Review of published cases associated with head and neck/oral and maxillofacial procedures

Tumor de Küttner sin relación con enfermedad de IgG4: reporte de un caso

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