TOPIC: Pulmonary Manifestations of Systemic Disease
TYPE: Medical Student/Resident Case Reports
INTRODUCTION: Sialolithiasis is a relatively benign obstructive phenomenon frequently occurring in the submandibular salivary glands (1). The clinical sequelae includes unilateral, localized pain and swelling, which may progress to salivary duct dilatation and infection, giving rise to sialadenitis (2). We report a case of unilateral sialadenitis rapidly progressing to angioedema and airway obstruction requiring cricothyrotomy
CASE PRESENTATION: A 95 year old female with history of left mandibular non-obstructing sialolith presented with painful right jaw and neck swelling. Examination revealed tender unilateral right submandibular and submental swelling and good oral hygiene. She received epinephrine, antihistamine and glucocorticoids. Flexible laryngoscopy revealed extensive swelling of the tongue base compressing the epiglottis with arytenoid edema. She unexpectedly developed respiratory compromise with muffled speech, drooling and oxygen saturations of 32% unresponsive to bag-valve mask. Intubation was unsuccessful necessitating an emergent cricothyrotomy. There was no circulatory collapse and our patient did not suffer from gastrointestinal symptoms. Evaluation revealed leucocytes 16.7thou/uL, neutrophils 78.6%, lactate 2.3mmol/L, ESR 42mm/hr and CRP 16.22mg/dL. C4 level could not be interpreted due to advanced age. Tryptase and C1 levels were not performed. Blood cultures remained sterile. CT neck demonstrated an obstructive 0.3cm stone within the right submandibular duct associated with right-sided sialadenitis and intraglandular duct dilatation. She required tracheostomy which was later decannulated and made a full recovery.
DISCUSSION: Sialadenitis predominantly occurs in the setting of bacterial parotid gland infection although infrequently arises from sialolithiasis affecting thesubmandibular glands. Nonetheless, they are regarded as benign conditions with the latter typically managed conservatively. Sialoadenitis triggering life-threatening angioedema requiring cricothyrotomy is unreported. Our case was atypical for mast-cell mediated angioedema in the absence of circulatory collapse. Bradykinin-mediated angioedema usually involves gastrointestinal symptoms. Acquired C1-inhibitor deficiency presents in the 4th decade with lymphoproliferative or autoimmune disease and recurrent episodes (3). Angiotensin-converting enzyme inhibitors are frequent culprits, there are reports of angioedema secondary to certain angiotensin-II-receptor blockers. Our patient had been taking irbesartan for several decades without adverse effects. Ludwig's angina was excluded as CT findings demonstrated unilateral inflammation without involvement of submylohyoid andsublingual spaces.
CONCLUSIONS: The differential diagnoses for upper airway compromise are wide and prompt identification is crucial. It is paramount for physicians to remain unbiased, as one may be faced with an atypical life-threatening presentation.
REFERENCE #1: Gupta A, Rattan D, Gupta R. Giant sialoliths of submandibular gland duct: Report of two cases with unusual shape. Contemp Clin Dent. 2013;4(1):78- 80. doi:10.4103/0976-237X.111599
REFERENCE #2: Iqbal A, Gupta AK, Natu SS, Gupta AK. Unusually large sialolith of Wharton's duct. Ann Maxillofac Surg. 2012;2(1):70-73. doi:10.4103/2231-0746.95327
REFERENCE #3: Cugno M, Zanichelli A, Bellatorre AG, Griffini S, Cicardi M. Plasma biomarkers of acute attacks in patients with angioedema due to C1-inhibitor deficiency. Allergy. 2009;64(2):254-257. doi:10.1111/j.1398-9995.2008.01859.
DISCLOSURES: no disclosure on file for Samuel Pope;
No relevant relationships by Rafae Shaikh, source=Web Response
No relevant relationships by Asiya Tafader, source=Web Response
© 2021 American College of Chest Physicians. Published by Elsevier Inc. All rights reserved.