title: Eosinophilic sialodochitis: redefinition of 'allergic parotitis' and 'sialodochitis fibrinosa'.
abstract: Sialodochitis fibrinosa and allergic parotitis have described rare patients with recurrent salivary gland swelling and mucus plugs, often with atopy. We have evaluated three patients with atopic disease, recurrent salivary gland swelling, and an eosinophilic sialodochitis. Two had eosinophil-rich mucus plugs. Fifty-six additional cases were identified in a medical literature database search, each defined by recurrent salivary gland swelling associated with eosinophil-rich mucus plugs or sialodochitis with periductal eosinophilic infiltration. The majority (78%) were reported from Japan. Females were predominantly affected (F:M = 2.3) with a median age of 47 years at evaluation. The parotid and submandibular glands were involved, respectively, in 71% and 46%. Allergic symptoms were present in 66%, atopic disease in 63% of those with reported allergy testing, and blood eosinophilia in 71%. Contrast sialography and other imaging modalities documented ductal dilatation in 82%. Treatments included anti-allergic medications (58%), systemic glucocorticoids (25%), duct cannulation with irrigation, steroid injection, and/or duct dilatation (36%), and glandular resection (19%). We recommend the diagnosis 'eosinophilic sialodochitis' be applied to patients who meet this case definition. The disease is a unique cause of chronic recurrent salivary gland swelling. Its likely allergic etiology may be amenable to current or future biologic therapies.
authors.affiliation: Division of Rheumatology, Department of Medicine, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
authors.forename: A N
authors.initials: AN
authors.lastname: Baer
authors.affiliation: Faculty of Medicine, The University of Tokyo, Tokyo, Japan.
authors.forename: A
authors.initials: A
authors.lastname: Okuhama
authors.affiliation: Department of Otolaryngology-Head and Neck Surgery, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
authors.forename: D W
authors.initials: DW
authors.lastname: Eisele
authors.affiliation: Division of Allergy and Clinical Immunology, Department of Medicine, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
authors.forename: J R
authors.initials: JR
authors.lastname: Tversky
authors.affiliation: Department of Pathology, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
authors.forename: T J
authors.initials: TJ
authors.lastname: Gniadek
mesh_terms: D001327:Autoimmune Diseases / Q000150:complications; D003937:Diagnosis, Differential; D004802:Eosinophilia / Q000150:complications / Q000000981:diagnostic imaging* / Q000473:pathology*; D006801:Humans; D007249:Inflammation / Q000150:complications / Q000000981:diagnostic imaging / Q000473:pathology; D010309:Parotitis / Q000276:immunology; D018987:Salivary Ducts; D012466:Salivary Gland Diseases / Q000150:complications / Q000000981:diagnostic imaging* / Q000473:pathology*
keywords: Kussmaul disease; allergic parotitis; eosinophils; salivary gland; sialodochitis fibrinosa