MRI Features in Submandibular Gland Chronic Sclerosing Sialadenitis: A Report of Three Cases and Imaging Findings

Document Type : Case Report

Authors

1 Department of Radiology and Diagnostic Imaging, IRCCS Regina Elena National Cancer Institute, Rome, Italy.

2 Department of Pathology, IRCCS Regina Elena National Cancer Institute, Rome, Italy.

3 Department of Otolaryngology & Head and Neck Surgery, IRCCS Regina Elena National Cancer Institute, Rome, Italy.

Abstract

Introduction:
Chronic sclerosing sialadenitis (Küttner tumor) is a relatively uncommon and often under-recognized cause of salivary gland enlargement, characterised by sclerosing IgG4-related inflammation, producing a hard swelling of the gland that mimics malignancy. The name tumor is tricky and misleading, in fact the disease has no histological features of malignancy, but still it cannot easily be distinguished from cancer because of its hard consistency to touch.
 
Case Reports:
We aim to report three cases of Küttner tumor and to review morphological MRI features (homogeneous T1- and T2-hypointensity, homogeneous contrast enhancement) and diffusion weighted imaging findings (low ADC values) which can help radiologists to reach the correct diagnosis.
 
Conclusion:
Definite diagnosis of Küttner tumor is histopathological. However imaging features are straightforward and can address radiologists toward the correct diagnosis.

Keywords

References

  1. Mulholland GB, Jeffery CC, Satija P, Côté DW. Immunoglobulin G4-related diseases in the head and neck: a systematic review. Journal of Otolaryngology-Head & Neck Surgery 2015; 44(1): 24.
  2. Ochoa ML, López BG, Cabello RR, Feregrino RR. IgG4-related multiorgan disease: report of the first autopsy case. BMJ case reports 2013.
  3. Geyer JT, Deshpande V. IgG4-associated sialadenitis. Current opinion in rheumatology 2011; 23(1):95-101.
  4. Paul R, Shekar K, Singh M. Küttner tumour: an unusual cause of enlargement of a minor salivary gland in the lip. British Journal of Oral and Maxillofacial Surgery 2010;48(2):152-153.
  5. Beriat GK, Akmansu SH, Kocatürk S, Ataoğlu Ö. Chronic sclerosing sialadenitis (Küttner’s tumour) of the parotid gland. The Malaysian journal of medical sciences 2010;17(4):57-61.
  6. Sahoo PK, Rout SS, Naidu AA, Reddy KU, Muddu VT. Kuttner’s tumour: chronic sclerosing sialadenitis- a mimicker of malignancy (2020); 7(2):514-519.
  7. Harrison JD, Epivatianos A. Immunoprofile of Küttner Tumor (Chronic Sclerosing Sialadenitis). International journal of surgical pathology 2010;18(5):443.
  8. Adachi M, Fujita Y, Murata T, Majima Y. A case of Küttner tumor of the submandibular gland. AurisNasus Larynx 2004;31(3):309-312.
  9. Kamisawa T, Funata N, Hayashi Y, Eishi Y, Koike M, Tsuruta K, et al. A new clinicopathological entity of IgG4-related autoimmune disease. Journal of gastroenterology 2003; 38(10):982-984.
  10. Takano K, Yamamoto M, Takahashi H, Shinomura Y, Imai K, Himi T. Clinicopathologic similarities between Mikulicz disease and Küttner tumor. American journal of otolaryngology 2010; 31(6):429-434.
  11. Shin YU, Oh YH, Lee YJ. Unusual involvement of IgG4-related sclerosing disease in lacrimal and submandibular glands and extraocular muscles. Korean Journal of Ophthalmology 2012; 26(3):216-221.
  12. Sangha Brar JS, Gupta S, Haja Mohideen SM, Liauw L, Lath N. The pancreatic and extrapancreatic manifestations of IgG4-related disease. Diagnostic and Interventional Radiology 2018; 24(2):83-88.
  13. Maehara T, Moriyama M, Nakamura S. Review of a novel disease entity, immunoglobulin G4-related disease. Journal of the Korean Association of Oral and Maxillofacial Surgeons 2020; 46(1), 3-11.
  14. Narayan AK, Baer A, Fradin J. Sonographic findings of IgG4‐related disease of the salivary glands: Case report and review of the literature. Journal of Clinical Ultrasound 2018;46(1):73-77.
  15. Taylor MJ, Serpell JW, Thomson P. Preoperative fine needle cytology and imaging facilitates the management of submandibular salivary gland lesions. ANZ J Surg 2011;81(1-2):
    70-74.
  16. De Cocker LJ, D’Arco F, De Beule T, Tousseyn T, Blockmans D, Hermans R. IgG4-related systemic disease affecting the parotid and submandibular glands: magnetic resonance imaging features of IgG4-related chronic sclerosing sialadenitis and concomitant lymphadenitis. Clinical imaging 2014; 38(2), 195-198.
  17. Abu A, Motoori K, Yamamoyo S, Hanazawa T, Nagal Y, Kaneoya K, et al. MRI of chronic sclerosing sialoadenitis. Br J Radiol 2008; 81(967):531-536.
  18. Fujita A, Sakai O, Chapman MN, Sugimoto H. IgG4-related Disease of the Head and Neck: CT and MR Imaging Manifestations. Radiographics 2012; 32(7):1945-1958.
  19. Poghosyan A, Misakyan M, Sargsyan A, Khachatryan P, Hakobyan G. Chronic sclerosing sialadenitis (Küttner's tumor) of the submandibular salivary gland: Our experience of one case report. Clinical case reports 2019; 7(8), 1600-1604.
  20. Matsusue E, Fujihara Y, Matsuda E, Tokuyasu Y, Nakamoto S, Nakamura K, et al. Differentiating parotid tumors by quantitative signal intensity evaluation on MR imaging. Clinical imaging 2017; 46:37-43.

 

 

 

 

Iranian Journal of Otorhinolaryngology
Volume 32, Issue 6
November and December 2020
Pages 397-401

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