Central Annals of Otolaryngology and Rhinology Case Report *Corresponding author Danielle Gill, Department of Oral and Maxillofacial Surgery, Meharry Medical College, Nashville, TN, USA, A Giant Submandibular Sialolith Tel: 1 (615) 327-6844; Fax: 1 (615) 327-5722; Email: in the Setting of Chronic Submitted: 11 June 2016 Accepted: 04 July 2016 Sialodenitis: A Case Report and Published: 19 July 2016 ISSN: 2379-948X Copyright Literature Review © 2016 Gill et al. Danielle Gill*, Robin Daniel, Leslie Halpern, and Janet OPEN ACCESS Southerland Keywords Department of Oral and Maxillofacial Surgery, Meharry Medical College, USA • Giant sialolith • Megalith Abstract • Chronic Sialadenitis • Treatment of giant sialoliths Sialolithiasis affects about 1% of the population and represents over 50% of disease that are associated with major salivary glands. Although the etiologies of sialolithiasis have been heavily debated, most agree that there is a multifactorial causation. Most authors conclude that salivary stones are formed from the deposition of calcium salts within the ductal system of salivary glands usually originating from desquamated epithelial cells, foreign bodies, microorganisms, and/or mucous plugs. Sialolith size varies from 6mm to 8cm. Those larger than 1.5cm have been deemed “giant sialoliths”, or megaliths. There are only 13 reported cases of sialoliths greater than 55mm. These appear to occur largely in the submandibular glands, and have a male predilection. We report a case of a giant sialolith in a 48-year-old African- American male presenting with a chronic sialadenitis, followed by a literature review of giant sialolith pathology and options for treatment. ABBREVIATIONS management options followed by a review of the literature. cm: Centimeters; mm: Millimeters; CT: Computed CASE PRESENTATION Tomography HPI/Clinical Presentation INTRODUCTION A 48-year-old African American male presented to the oral and Sialolithiasis is considered to be the most common non- maxillofacial surgery clinic at Meharry Medical College, Nashville, neoplastic salivary disorder and represents about 50% of all TN with a complaint of facial pain and swelling associated with major salivary gland disease [1]. This benign disorder has a the left submandibular region. The patient reported three relatively low prevalence at roughly 1% of the population with months of progressive swelling, and recent persistent pain during symptomatic sialolithiasis occurring at a rate of 0.45% [1-3]. The mealtime. The patient recorded a 10-pound weight loss, however etiology of the sialolith is both controversial and multifactorial denied nausea, vomiting, fever, chills, paresthesias and dyspnea. with some suggesting that sialolith formation is precipitated/ exacerbated by the presence of desquamated epithelial cells, Past medical and surgical histories included obesity and left foreign bodies, microorganisms, and/ or mucous plugs within the foot 5th digit osteomyelitis secondary to nail injury treated by ductal canal, potentially creating a nidus for calcium deposition, incision and drainage and followed by amputation of left foot as well as other etiologic agents [1]. For example, stasis of digit. The patient denied any allergies. Social history included saliva due to the course of the ductal system or the nidus itself, smoking one pack of cigarettes every other day for over 20 years can enhance the development of a salivary stone [2]. Other and drinking beer or wine weekly. Family history was non- precipitating factors that predispose sialolithiasis are metabolic; contributory. i.e. Gout [1]. Head and neck examination revealed moderate asymmetry of We present a case report that describes a giant sialolith in the the left neck predominately in the submandibular region (Figure 1A). The area was solid and tender to palpation. No extra-oral as well as discernable facial asymmetry. Clinical presentation, erythema or purulence was noted. In addition, there was no radiographic,left submandibular and histologic region with features perforation are discussed, of floor of oralas well cavity, as associated lymphadenopathy or temporomandibular associated Cite this article: Gill D, Daniel R, Halpern L, Southerland J (2016) A Giant Submandibular Sialolith in the Setting of Chronic Sialodenitis: A Case Report and Literature Review. Ann Otolaryngol Rhinol 3(8): 1128. Gill et al. (2016) Email: Central pathologies. Maximum incisal opening was approximately 30mm, with guarding due to pain. Intraoral exam revealed an oropharynx that was clear, a uvula that was midline with no exhibited a perforation of approximately 2cm as well as a large, plaque-covered,associated palatal movable draping. calculus The left (Figure posterior 1B). floor In ofaddition, the mouth the tender to palpation. entire left floor of mouth was mildly elevated, indurated and Panoramic imaging revealed a large, oblong radiopaque mass superimposed on the left mandibular angle (Figure 2). Computed tomography (CT) scan of the region showed a heterogeneous Figure 1 Preoperative extraoral and intraoral photographs. (A) enhancement and mild generalized enlargement of the left – Moderate left-sided lower facial swelling present (B) - Calculus submandibular gland. Centered within the posterior aspect perforated through floor of mouth. Limited opening. stoneof the measuringgland, and 2.1 extending x 4.3 x 3.1 to cmthe (Figures floor of 3A,3B).the mouth along the course of Wharton’s duct, there appeared to be a densely calcified Differential Diagnosis A working differential diagnosis was developed based on the patient’s medical history, symptomatology, clinical presentation, possible differential diagnoses: Sialolithiasis with a concomitant and radiographic findings. The following were suggested as left submandibular chronic sialadenitis, foreign body, calcified lymph node, vascular calcification, osteomas, myositis ossificans, and/or a calcified neoplasm [4]. Sialolithiasis was deemed to be Figure 2 Hospital Course left posterior mandible. the most definitive diagnosis based upon the work-up presented. Panoramic View: Large radiopaque mass superimposed on The patient was consented and taken to the operating room with a diagnosis of chronic sialolithiasis/chronic sialadenitis of the left submandibular gland. The treatment plan was to explore submandibular gland sialadenectomy, and left sialolithectomy. the ductal pathway along the left floor of the mouth, as well as a left Wharton’sThe gland ductwas revealedaccessed a viagrossly a modified dilated duct.Risdon The approach distal portion and offound the duct to be was fibrotic then ligated upon andevaluation. the gland Further was then dissection removed. along With further manipulation the proximal duct, there was no success with retrieving the sialolith and it was subsequently dislodged Figure 3 thethrough mouth. the Thefloor proximal of the mouth duct was into then the oralligated. cavity, The via perforation a tear in oblong radiopaque mass associated with left submandibular gland. CT Maxillofacial w/ Contrast (A) – Coronal View: Large wasWharton’s closed ductintraorally leaving with a large 3-0 resorbable,perforation synthetic through thesuture. floor The of submandibular gland, attached duct, and sialolith were sent for Gland is markedly enlarged. Left facial asymmetry appreciated. (B) - Sagittal View: Large radiopaque mass roughly 3cm in height. finalHistology gross pathologic/histological of specimen evaluation. dysplastic changes were identified during examination. giant sialolith arising from an associated chronic sialadenitis of that measured, 5.7cm x 2.3cm x 2.2cm and the left submandibular the Theleft submandibularabove findings gland.characterized The patient a definitive returned diagnosis to our clinic of a A gross examination of the specimen identified a giant sialolith gland specimen (Figure 4). The submandibular gland presented at 2 weeks for a surgical follow-up. Healing was uneventful and as a rubbery tan nodule weighing 20 grams, measuring 4.5cm x 3.0cm x 2.3cm. Histological examination of the submandibular theDISCUSSION patient was satisfied with treatment. tissue. The lobules contained glands composed of both serous Sialolithiasis affects about 1% of the population and gland showed lobules separated by dense fibrous connective represents over 50% of diseases that are associated with the major salivary glands [1]. The average age at diagnosis is 30- and mucus cells and small ductiles. A dense chronic inflammatory plastic or 70 years [5]. Although all major and minor salivary glands can infiltrate extended to the lobules and also in the surrounding dense fibrotic stroma (Figures 5A, 5B, 5C). No neo Ann Otolaryngol Rhinol 3(8): 1128 (2016) 2/7 Gill et al. (2016) Email: Central salivary stones [1]. This anatomy also lends to the increased viscosityof saliva ofand the is saliva the most and relativelysignificant high factor content in the of calciumformation salts, of submandibular gland more prone to stone formation. Most of the giantspecifically sialoliths phosphates, reported carbonates, in the article and were oxalates associated which makewith the submandibular gland and were within the ductal system, with only 26% found in the gland parenchyma [2, 9, 10]. Sialoliths are thought to enlarge at the rate of approximately 1–1.5 mm per year, although some reports show a rate of 3.5mm per year [11]. Stones larger than 3 cm are extremely rare (Table 1). Mean size is roughly 7.3mm, but stones greater than 8cm have been documented [6, 66]. There are only