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Sialadenitis of Unknown Etiology

  • Salivary Gland Disorders (HT Hoffman, Section Editor)
  • Published:
Current Otorhinolaryngology Reports Aims and scope Submit manuscript

Abstract

Purpose of Review

Most sialadenitis is attributed to infection, obstruction, or underlying autoimmunity; however, there are several rare processes affecting the salivary glands without clear etiology. We review the available literature, specifically addressing presentation, evaluation, and treatment.

Recent findings

Juvenile recurrent parotitis is a typically self-limiting entity occurring in school-age children and may be benefitted by sialendoscopy. Sclerosing polycystic adenosis is a rare cystic disorder of major salivary glands, diagnosed, and treated through surgery. Inflammatory pseudotumor is thought to be an abnormal focal immune response, mimicking a neoplasm. Rosai-Dorfman and Kimura diseases are considered lymphoproliferative disorders, and amyloidosis is a rare protein deposition disorder; all of which can affect the salivary glands.

Summary

Unusual clinical entities should be considered for atypical or persistent sialadenitis of unknown etiology. Work-up generally includes biopsy for histologic diagnosis. Treatment is typically supportive and/or related to treating associated systemic disease. Surgical excision is reserved to establish diagnosis, for severe/refractory cases, or when malignancy is suspected.

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References

Papers of particular interest, published recently, have been highlighted as: • Of importance

  1. Mahalakshmi S, Kandula S, Shilpa P, Kokila G. Chronic recurrent non-specific parotitis: a case report and review. Ethiop J Health Sci. 2017;27:95–100.

    Article  Google Scholar 

  2. • Garavello W, Redaelli M, Galluzzi F, Pignataro L. Juvenile recurrent parotitis: a systematic review of treatment studies. Int J Pediatr Otorhinolaryngol. 2018;112 July:151–7. https://doi.org/10.1016/j.ijporl.2018.07.002This systematic review addresses the available literature regarding juvenile recurrent parotitis and the available treatment strategies. The available evidence to support any particular intervention, including sialendsocopy, is weak.

    Article  Google Scholar 

  3. Canzi P, Occhini A, Pagella F, Marchal F, Benazzo M. Sialendoscopy in juvenile recurrent parotitis: a review of the literature. Acta Otorhinolaryngol Ital. 2013;33:367–73 http://www.ncbi.nlm.nih.gov/pubmed/24376291. http://www.pubmedcentral.nih.gov/articlerender.fcgi?artid=PMC3870450.

    CAS  PubMed  PubMed Central  Google Scholar 

  4. Isaacs D. Recurrent parotitis. J Paediatr Child Health. 2002;38:92–4. https://doi.org/10.1046/j.1440-1754.2002.00707.x.

    Article  CAS  PubMed  Google Scholar 

  5. Iro H, Zenk J. Salivary gland diseases in children. GMS Curr Top Otorhinolaryngol Head Neck Surg. 2014;13:1–30.

    Google Scholar 

  6. Reid E, Douglas F, Crow Y, Hollman A, Gibson J. Autosomal dominant juvenile recurrent parotitis. J Med Genet. 1998;35:417–9.

    Article  CAS  Google Scholar 

  7. Ericson S, Sjöbäck I. Salivary factors in children with recurrent parotitis. Part 2: Protein, albumin, amylase, IgA, lactoferrin lysozyme and kallikrein concentrations. Swed Dent J. 1996;20:199–207 http://www.ncbi.nlm.nih.gov/pubmed/9000329.

    CAS  PubMed  Google Scholar 

  8. Ramakrishna J, Strychowsky J, Gupta M, Sommer DD. Sialendoscopy for the management of juvenile recurrent parotitis: a systematic review and meta-analysis. Laryngoscope. 2015;125:1472–9.

    Article  Google Scholar 

  9. Schneider H, Koch M, Künzel J, Gillespie MB, Grundtner P, Iro H, et al. Juvenile recurrent parotitis: a retrospective comparison of sialendoscopy versus conservative therapy. Laryngoscope. 2014;124:451–5.

    Article  Google Scholar 

  10. Tucci FM, Roma R, Bianchi A, De Vincentiis GC, Bianchi PM. Juvenile recurrent parotitis: diagnostic and therapeutic effectiveness of sialography. Retrospective study on 110 children. Int J Pediatr Otorhinolaryngol. 2019;124:179–84. https://doi.org/10.1016/j.ijporl.2019.06.007.

    Article  PubMed  Google Scholar 

  11. Smith BC, Ellis GL, Slater LJ, Foss RD. Sclerosing polycystic adenosis of major salivary glands. a clinicopathologic analysis of nine cases. Am J Surg Pathol. 1996;20:161–70. https://doi.org/10.1097/00000478-199602000-00004.

    Article  CAS  PubMed  Google Scholar 

  12. Tang CG, Fong JB, Axelsson KL, Gurushanthaiah D. Sclerosing polycystic adenosis: a rare tumor of the salivary glands. Perm J. 2016;20:e113–4.

    PubMed  PubMed Central  Google Scholar 

  13. Gnepp DR. Salivary gland tumor “wishes” to add to the next WHO tumor classification: sclerosing polycystic adenosis, mammary analogue secretory carcinoma, cribriform adenocarcinoma of the tongue and other sites, and mucinous variant of myoepithelioma. Head Neck Pathol. 2014;8:42–9.

    Article  Google Scholar 

  14. Espinosa CA, Rua L, Torres HE, Fernández del Valle Á, Fernandes RP, Devicente JC. Sclerosing polycystic adenosis of the parotid gland: a systematic review and report of 2 new cases. J Oral Maxillofac Surg. 2017;75:984–93.

    Article  Google Scholar 

  15. Skálová A, Gnepp DR, Simpson RHW, Lewis JE, Janssen D, Sima R, et al. Clonal nature of sclerosing polycystic adenosis of salivary glands demonstrated by using the polymorphism of the human androgen receptor (HUMARA) locus as a marker. Am J Surg Pathol. 2006;30:939–44.

    Article  Google Scholar 

  16. Bishop JA, Gagan J, Baumhoer D, McLean-Holden AL, Oliai BR, Couce M, et al. Sclerosing polycystic “adenosis” of salivary glands: a neoplasm characterized by PI3K pathway alterations more correctly named sclerosing polycystic adenoma. Head Neck Pathol. 2020;14:630–6.

    Article  Google Scholar 

  17. Swelam WM. The pathogenic role of Epstein-Barr virus (EBV) in sclerosing polycystic adenosis. Pathol Res Pract. 2010;206:565–71.

    Article  CAS  Google Scholar 

  18. Eliot CA, Smith AB, Foss RD. Sclerosing polycystic adenosis. Head Neck Pathol. 2012;6:247–9.

    Article  Google Scholar 

  19. Fulciniti F, Losito NS, Ionna F, Longo F, Aversa C, Botti G, et al. Sclerosing polycystic adenosis of the parotid gland: report of one case diagnosed by fine-needle cytology with in situ malignant transformation. Diagn Cytopathol. 2010;38:368–73. https://doi.org/10.1002/dc.21228.

    Article  PubMed  Google Scholar 

  20. Matsumoto NM, Umezawa H, Ohashi R, Peng WX, Naito Z, Ogawa R. Surgical treatment of rare sclerosing polycystic adenosis of the deep parotid gland. Plast Reconstr Surg Glob Open. 2016;4:1–4.

    Google Scholar 

  21. Canas Marques R, Félix A. Invasive carcinoma arising from sclerosing polycystic adenosis of the salivary gland. Virchows Arch. 2014;464:621–5.

    Article  Google Scholar 

  22. Gorevic PD. Overview of Amyloidosis. In: Post TW, editor. UpToDate. Waltham, Massachusetts: UpToDate; 2020. https://www.uptodate.com/contents/overview-of-amyloidosis.

  23. Mercan R, Bıtık B, Tezcan ME, Kaya A, Tufan A, Özturk MA, et al. Minimally invasive minor salivary gland biopsy for the diagnosis of amyloidosis in a rheumatology clinic. ISRN Rheumatol. 2014;2014:1–3.

    Article  Google Scholar 

  24. Nandapalan V, Jones TM, Morar P, Clark AH, Jones AS. Localized amyloidosis of the parotid gland: a case report and review of the localized amyloidosis of the head and neck. Head Neck. 1998;20:73–8.

    Article  CAS  Google Scholar 

  25. Perera E, Revington P, Sheffield E. Low grade marginal zone B-cell lymphoma presenting as local amyloidosis in a submandibular salivary gland. Int J Oral Maxillofac Surg. 2010;39:1136–8. https://doi.org/10.1016/j.ijom.2010.05.001.

    Article  CAS  PubMed  Google Scholar 

  26. Vavrina J, Müller W, Gebbers JO. Recurrent amyloid tumor of the parotid gland. Eur Arch Otorhinolaryngol. 1995;252:53–6. https://doi.org/10.1007/BF00171441.

    Article  CAS  PubMed  Google Scholar 

  27. Kilic E, Ibrahimov M, Aslan M, Yener HM, Karaman E. Inflammatory myofibroblastic tumor of the parotid gland. J Craniofac Surg. 2012;23:557–8.

    Article  Google Scholar 

  28. Devaney KO, LaFeir DJ, Triantafyllou A, Mendenhall WM, Woolgar JA, Rinaldo A, et al. Inflammatory myofibroblastic tumors of the head and neck: evaluation of clinicopathologic and prognostic features. Eur Arch Otorhinolaryngol. 2012;269:2461–5.

    Article  Google Scholar 

  29. Escobar Sanz-Dranguet P, Márquez Dorsch FJ, Sanabria Brassart J, Gutiérrez Fonseca R, Villacampa Aubá JM, Pastormerlo G, et al. Inflammatory pseudotumor of paranasal sinuses. Acta Otorrinolaringol Esp. 2002;53:135–8. https://doi.org/10.1016/s0001-6519(02)78292-7.

    Article  CAS  PubMed  Google Scholar 

  30. Kansara S, Bell D, Johnson J, Zafereo M. Head and neck inflammatory pseudotumor: Case series and review of the literature. Neuroradiol J. 2016;29:440–6. https://doi.org/10.1177/1971400916665377.

    Article  PubMed  PubMed Central  Google Scholar 

  31. Dhua AK, Garg M, Sen A, Chauhan DS. Inflammatory myofibroblastic tumor of parotid in infancy--a new entity. Int J Pediatr Otorhinolaryngol. 2013;77:866–8. https://doi.org/10.1016/j.ijporl.2013.02.020.

    Article  PubMed  Google Scholar 

  32. Chen H, Thompson LDR, Aguilera NSI, Abbondanzo SL. Kimura disease: a clinicopathologic study of 21 cases. Am J Surg Pathol. 2004;28:505–13. https://doi.org/10.1097/00000478-200404000-00010.

    Article  PubMed  Google Scholar 

  33. Dhingra H, Nagpal R, Baliyan A, Alva SR. Kimura disease : case report and brief review of literature. Med Pharm Rep. 2018;92:195–9.

    Google Scholar 

  34. Chun S II, Ji HG. Kimura’s disease and angiolymphoid hyperplasia with eosinophilia: clinical and histopathologic differences. J Am Acad Dermatol. 1992;27:954–8.

    Article  CAS  Google Scholar 

  35. Sah P, Kamath A, Aramanadka C, Radhakrishnan R. Kimura’s disease - an unusual presentation involving subcutaneous tissue, parotid gland and lymph node. J Oral Maxillofac Pathol. 2013;17:455–9. https://doi.org/10.4103/0973-029X.125220.

    Article  PubMed  PubMed Central  Google Scholar 

  36. Jordan JW, Oxford LE, Adair CF. Infiltrative right parotid mass with lymphadenopathy. JAMA Otolaryngol Head Neck Surg. 2016;142:1015–6.

    Article  Google Scholar 

  37. Chang AR, Kim K, Kim HJ, Kim IH, Park C II, Jun YK. Outcomes of Kimura’s disease after radiotherapy or nonradiotherapeutic treatment modalities. Int J Radiat Oncol Biol Phys. 2006;65:1233–9. https://doi.org/10.1016/j.ijrobp.2006.02.024.

    Article  PubMed  Google Scholar 

  38. Rosai J, Dorfman RF. Sinus histiocytosis with massive lymphadenopathy. A newly recognized benign clinicopathological entity. Arch Pathol. 1969;87:63–70 http://www.ncbi.nlm.nih.gov/pubmed/5782438.

    CAS  PubMed  Google Scholar 

  39. Carbone A, Passannante A, Gloghini A, Devaney KO, Rinaldo A, Ferlito A. Review of sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease) of head and neck. Ann Otol Rhinol Laryngol. 1999;108(11 Pt 1):1095–104. https://doi.org/10.1177/000348949910801113.

    Article  CAS  PubMed  Google Scholar 

  40. Panikar N, Agarwal S. Salivary gland manifestations of sinus histiocytosis with massive lymphadenopathy: fine-needle aspiration cytology findings. A case report. Diagn Cytopathol. 2005;33:187–90. https://doi.org/10.1002/dc.20321.

    Article  PubMed  Google Scholar 

  41. Deshpande AH, Nayak S, Munshi MM. Cytology of sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease). Diagn Cytopathol. 2000;22:181–5. https://doi.org/10.1002/(SICI)1097-0339(20000301)22:3<181::AID-DC10>3.0.CO;2-6.

    Article  CAS  PubMed  Google Scholar 

  42. Gaitonde S. Multifocal, extranodal sinus histiocytosis with massive lymphadenopathy: an overview. Arch Pathol Lab Med. 2007;131:1117–21. https://doi.org/10.1043/1543-2165(2007)131[1117:MESHWM]2.0.CO;2.

    Article  PubMed  Google Scholar 

  43. Juskevicius R, Finley JL. Rosai-Dorfman disease of the parotid gland: cytologic and histopathologic findings with immunohistochemical correlation. Arch Pathol Lab Med. 2001;125:1348–50. https://doi.org/10.1043/0003-9985(2001)125<1348:RDDOTP>2.0.CO;2.

    Article  CAS  PubMed  Google Scholar 

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Author information

Authors and Affiliations

  1. Department of Otolaryngology, Washington University School of Medicine, 660 S. Euclid Ave, Box 8115, St. Louis, MO, 63110, USA

    Miranda Lindburg & Allison Ogden

  2. Department of Otolaryngology Head & Neck Surgery, Louisiana State University Health Sciences Center, 533 Bolivar Street, Suite 566, New Olreans, LA, 70112, USA

    Rohan R. Walvekar

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  1. Miranda Lindburg
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Correspondence to Allison Ogden.

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Lindburg, M., Walvekar, R.R. & Ogden, A. Sialadenitis of Unknown Etiology. Curr Otorhinolaryngol Rep 9, 378–382 (2021). https://doi.org/10.1007/s40136-021-00361-7

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