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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.
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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
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Corresponding Author: Vivek Dokania, MBBS, MS, ENT, Krishna Institute of Medical Sciences, Karad, Maharashtra, India, 415110 (firstname.lastname@example.org).
Published Online: August 29, 2019. doi:10.1001/jamaoto.2019.2252
Conflict of Interest Disclosures: None reported.
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