Case Presentation: A 28-year-old female with a history of ulcerative colitis (UC) was hospitalized for surgical evaluation in the setting of refractory colitis. Seven weeks prior to presentation, she developed diffuse proctocolitis (Mayo score 2-3) that progressed despite treatment escalation from oral to intravenous steroids, followed by infliximab induction and mercaptopurine initiation three weeks prior to presentation.On the second night of her hospitalization, she developed acute onset, rapidly progressive bilateral swelling and pain of the lower face and neck with associated phagophobia. She had no respiratory distress or airway compromise. Her temperature was 36.6o C, blood pressure was 132/80 mmHg, and pulse was 95 bpm. Physical exam revealed symmetric submandibular swelling without fluctuance or purulence from the parotid or submandibular ducts (Figure 1C). Initial labs were notable only for a WBC of 12.6 g/dL. Head and neck computed tomography showed bilateral submandibular sialadenitis, inflammatory changes in the floor of the mouth, and pharyngeal and laryngeal edema (Figure 1A, 1B). Flexible fiberoptic nasopharyngolaryngoscopy showed thick secretions and mild left lateral pharyngeal wall edema. She was started on cefepime and intravenous dexamethasone and encouraged to suck sour hard candies with complete resolution of symptoms within 48 hours. Infliximab and mercaptopurine were discontinued. At 9-month follow up, she has had no reoccurrence of sialadenitis.
Discussion: Sialadenitis, or inflammation of a salivary gland, most often affects the parotid, submandibular, and sublingual glands. The majority of cases are unilateral and caused by sialolithiasis; other common causes include infection (bacterial or viral), ductal stricture, malignancy, autoimmune conditions, or infiltrative diseases. Each of these etiologies has a clearly delineated pathophysiologic mechanism for the development of acute sialadenitis (1,2). The bilateral ductal involvement and lack of a clear underlying cause make this particular case unique. Acute infection was unlikely because of the rapid resolution, bilateral distribution, and lack of fever. Stone, stricture, and malignancy were ruled out by imaging and direct visualization of patent ducts. Iodinated-contrast induced sialadenitis (“Iodide Mumps”) was unlikely because of the lack of renal disease or preceding exposure to a high contrast load (as described in cases of this rare phenomenon) (3). There has been no clear link between sialadenitis and inflammatory bowel disease. Sialadenitis is often considered idiopathic in etiology – many of these cases implicate adverse drug effect as the cause. Both infliximab and mercaptopurine have been associated with acute onset sialadenitis; these were considered the most likely culprits of this patient’s acute sialadenitis (5–7). Potent immunosuppression therapy may lead to reactivation of latent sialatropic viruses, direct toxic medication effect, or a coincidental consequence of a dysregulate immune system in the setting of systemic autoimmune or autoinflammatory disease.
Conclusions: The general internist should be familiar with the presentation of acute sialadenitis as well as the common causes. In patients who develop acute onset, non-purulent, non-obstructive, and bilateral sialadenitis, one should consider medication effect as an underlying etiology. Early recognition of this as the cause can lead to discontinuation of the inciting drug and the avoidance of unnecessary therapeutics.
