[Skip to Content]
Access to paid content on this site is currently suspended due to excessive activity being detected from your IP address 18.204.55.168. Please contact the publisher to request reinstatement.
[Skip to Content Landing]

Solitary Mucus Cast at the Wharton Duct Orifice

Educational Objective
Based on this clinical scenario and the accompanying image, understand how to arrive at a correct diagnosis.
1 Credit CME

A woman presented with a 2-year history of recurring left submandibular gland swelling and pain. She reported no associated fevers, chills, dryness of mouth, nausea, vomiting, dysphagia or odynophagia. Her medical history was unremarkable except for allergic rhinitis. On physical examination, she had slight enlargement of the left submandibular gland, which was not tender on palpation. Ultrasonography results revealed normal submandibular and parotid glands bilaterally without any mass or calcifications. Blood examinations and basic metabolic panel results were all within normal limits. Sialoendoscopy of the left submandibular duct was performed under general anesthesia. The patient was found to have a cast at the opening of the Wharton duct, which was removed and sent for pathological evaluation (Figure). Using sialoendoscopy with saline irrigation, the entire duct was examined up to the hilum of the gland. No stones were noted. There were areas of mild partial webbing without complete obstruction. The areas of webbing were broken up with irrigation, and at the end of the procedure steroid instillation and irrigation were performed. There were no complications during or after the procedure.

Please finish quiz first before checking answer.

You answered correctly!

Read the answer below and download your certificate.

You answered incorrectly.

Read the discussion below and retake the quiz.

C. Eosinophilic sialodochitis

The histopathological examination of the removed cast revealed exudates with extensive eosinophilic infiltrates without any glandular tissue (Figure). The histological finding of eosinophil-rich cast combined with the clinical feature of recurrent left submandibular gland swelling confirmed a diagnosis of eosinophilic sialodochitis.

Baer et al1 first provided a proper case definition of eosinophilic sialodochitis, which is characterized by recurrent major salivary gland swelling associated with an eosinophil-rich mucus plug or histological evidence of sialodochitis with a periductal eosinophilic infiltration. The majority of these cases have been reported in Japan as sialodochitis fibrinosa.1,2 Eosinophilic sialodochitis is not well known in western countries, and only a few cases have been reported in English-language medical literature under identical nomenclature.1,3 Other published cases from the English-language literature, which exclusively fit in the Baer et al1 definition criteria of eosinophilic sialodochitis, have been reported under different names; for example, Kussmaul disease,2 sialodochitis fibrinosa,2,4,5 chronic sialodochitis with eosinophilia,6 allergic parotitis,7 and idiopathic eosinophilic parotitis.8

Survey Complete!

Sign in to take quiz and track your certificates

Buy This Activity

JN Learning™ is the home for CME and MOC from the JAMA Network. Search by specialty or US state and earn AMA PRA Category 1 CME Credit™ from articles, audio, Clinical Challenges and more. Learn more about CME/MOC

Article Information

Corresponding Author: Vivek Dokania, MBBS, MS, ENT, Krishna Institute of Medical Sciences, Karad, Maharashtra, India, 415110 (drvivekdokania@gmail.com).

Published Online: August 29, 2019. doi:10.1001/jamaoto.2019.2252

Conflict of Interest Disclosures: None reported.

References
1.
Baer  AN, Okuhama  A, Eisele  DW, Tversky  JR, Gniadek  TJ.  Eosinophilic sialodochitis: redefinition of allergic parotitis and sialodochitis fibrinosa.  Oral Dis. 2017;23(7):840-848. doi:10.1111/odi.12595PubMedGoogle ScholarCrossref
2.
Robles  BJF, Álvarez  BB, Sanchinel  AAS,  et al.  Sialodochitis fibrinosa (Kussmaul disease): report of 3 cases and literature review.  Medicine (Baltimore). 2016;95(42):e5132. doi:10.1097/MD.0000000000005132Google ScholarCrossref
3.
Pollak  N, Templer  JW, Esebua  M, Diaz-Arias  AA, Zitsch  RP.  Episodic painful parotid swelling caused by sialodochitis with eosinophilic inflammation: a new entity.  Otolaryngol Head Neck Surg. 2009;140(1):132-133. doi:10.1016/j.otohns.2008.10.004PubMedGoogle ScholarCrossref
4.
Chikamatsu  K, Shino  M, Fukuda  Y, Sakakura  K, Furuya  N.  Recurring bilateral parotid gland swelling: two cases of sialodochitis fibrinosa.  J Laryngol Otol. 2006;120(4):330-333. doi:10.1017/S0022215106000296PubMedGoogle ScholarCrossref
5.
Shimada  T, Okano  H, Hisa  Y.  A case of severe dilatation of the parotid duct due to fibrinous sialodochitis.  Acta Otolaryngol. 2006;126(10):1112-1114. doi:10.1080/00016480600621680PubMedGoogle ScholarCrossref
6.
Darling  MR, Phillips  VM, Erasmus  JH.  Bilateral submandibular salivary gland swelling: a report of chronic sialodochitis with eosinophilia.  SADJ. 2002;57(3):104-106.PubMedGoogle Scholar
7.
Waldbott  GL, Shea  JJ.  Allergic parotitis.  J Allergy. 1947;18(1):51-54. doi:10.1016/0021-8707(47)90067-1PubMedGoogle ScholarCrossref
8.
Frati  F, Boccardo  R, Scurati  S, Gelardi  M, Incorvaia  C.  Idiopathic eosinophilic parotitis in an eight-year-old boy: a case report.  J Med Case Rep. 2011;5:385. doi:10.1186/1752-1947-5-385PubMedGoogle ScholarCrossref
9.
Swinny  B.  Allergic parotitis.  Ann Allergy. 1953;11(4):473-474.PubMedGoogle Scholar
10.
Ishida  M, Fushiki  H, Watanabe  Y.  An IgG4-related salivary gland disorder: a case series presenting with a different clinical setting.  Case Reports Immunol. 2011;2011:236079. doi:10.1155/2011/236079PubMedGoogle ScholarCrossref
If you are not a JN Learning subscriber, you can either:
Subscribe to JN Learning for one year
Buy this activity
jn-learning_Modal_LoginSubscribe_Purchase
Close
If you are not a JN Learning subscriber, you can either:
Subscribe to JN Learning for one year
Buy this activity
jn-learning_Modal_LoginSubscribe_Purchase
Close
With a personal account, you can:
  • Access free activities and track your credits
  • Personalize content alerts
  • Customize your interests
  • Fully personalize your learning experience
Education Center Collection Sign In Modal Right
Close

Name Your Search

Save Search
Close
With a personal account, you can:
  • Track your credits
  • Personalize content alerts
  • Customize your interests
  • Fully personalize your learning experience
jn-learning_Modal_SaveSearch_NoAccess_Purchase
Close

Lookup An Activity

or

Close

My Saved Searches

You currently have no searches saved.

Close
With a personal account, you can:
  • Access free activities and track your credits
  • Personalize content alerts
  • Customize your interests
  • Fully personalize your learning experience
Education Center Collection Sign In Modal Right
Close