Clinics and Practice 2019; volume 9:1119 Submandibular sialolithiasis: ology, usual and unusual locations, clinical A series of three case reports features, diagnostic and treatment modali- Correspondence: Sucharu Ghosh, Department ties, along with their indications and con- of Oral and Maxillofacial Surgery, Haldia with review of literature traindications. Institute of Dental Sciences and Research, West Bengal University of Health Sciences, Haldia, West Bengal, India. Sandeep Pachisia, Gaurav Mandal, Tel.: +919062792661. Sudipto Sahu, Sucharu Ghosh E-mail: [email protected] Case Reports Department of Oral and Maxillofacial Surgery, Haldia Institute of Dental Key words: Sialolithiasis; Submandibular Case #1 gland; Sialolithotomy. Sciences and Research, West Bengal A 40-year-old male patient reported to University of Health Sciences, Haldia, our institute with a chief complaint of recur- Contributions: the authors contributed equally. West Bengal, India rent episodes of pain, difficulty in swallow- ing and swelling in the neck (Figure 1A), Conflict of interest: the authors declare no potential conflict of interest. for the last 2-3 years. The last episode occurred 1-2 months prior to the visit and Funding: none. Abstract the pain has been persistent since. On extraoral examination, a diffuse Received for publication: 28 November 2018. One of the most common disorders of swelling over the left submandibular region Revision received: 20 February 2019. the salivary glands is sialolithiasis. A histo- was found. On bimanual palpation left sub- Accepted for publication: 20 February 2019. ry of pain or/and swelling in the salivary mandibular gland was firm and tender and a This work is licensed under a Creative glands, especially during meal suggests this single tender left submandibular lymph diagnosis. For small and accessible stones Commons Attribution NonCommercial 4.0 node was palpated. License (CC BY-NC 4.0). conservative therapies like milking of ducts Intraorally left submandibular duct with palliative therapy can produce satisfac- opening was inflamed and erythematous ©Copyright S. Pachisia et al., 2019 tory results. Surgical management should along with a diffuse swelling and discharge Licenseeonly PAGEPress, Italy be considered when the stone/stones are of pus (Figure 1B). The swelling was firm Clinics and Practice 2019; 9:1119 inaccessible or large in size as conservative to hard on palpation, running from 1 cm doi:10.4081/cp.2019.1119 therapies turned out to be unsatisfactory. In posterior to the ductal opening to the base of this paper, we present three cases of the tongue posteriorly. Also, mild elevationuse sialolithiasis in the submandibular gland of the tongue was seen. along with a review of existing literature. The mandibular occlusal radiograph exposed and retrieved. The sialolith was The purpose of this paper is to add three revealed a single ovoid radiopacity extend- about 5×8.5mm in size and weighted only more cases to the literature and review the ing from 36 tooth to the distal aspect 37 0.03g. Sutures were placed to close the sur- theories of etiology, clinical features, avail- tooth (Figure 1C). On the basis of clinical gical area (Figure 2D). able diagnostic and treatment procedures. and radiological findings, we diagnosed the case as a left submandibular sialolithiasis. Case #3 All preoperative investigations were A 75-year-old man visited us with a under normal limits and the patient was chief complaint of swelling and pain during Introduction under antibiotic coverage. Under local anes- swallowing on the right side of the face Sialolithiasis is considered to be the thesia,commercial an intraoral incision was made in the (Figure 3A). most common salivary gland disorder and it floor of the mouth. The duct was opened Extraoral examination revealed no facial accounts for about 1.2% of unilateral major and the sialolith was removed in a single asymmetry but firm and tender right sub- salivary gland swellings. Submandibular piece. It was 1.5 cm in length and 1 cm in mandibular lymph nodes. Intraoral examina- gland has the highest predilection for diameter. It weighed 0.07g on an electronic tion showed mild elevation of the tongue sialolithiasis with 80% occurrence rate,Non fol- weighing machine. Sutures were placed to with swelling and the stone was visible at the lowed by the parotid (19%) and the sublin- close the area (Figure 1D). right Wharton’s duct opening (Figure 3B). A gual (1%) glands. Sialolithiasis is usually diffuse swelling was palpable on the floor of seen between the age of 30 and 60 years. It Case #2 the mouth on the right side of the lingual is uncommon in children as only 3% of all A 26-year-old male patient reported with frenum. On the basis of the above findings, sialolithiasis cases has been reported in the swelling below the tongue, which was asso- we came to a provisional diagnosis of right pediatric population. Males are affected ciated with pain for 1 week (Figure 2A). submandibular sialolithiasis. A diffused twice as much as females. Clinical examination revealed a superfi- radiopacity was seen from the mesial surface The clinical symptoms include swelling cial, 5 mm hard swelling situated near the of the first molar to the distal surface of the and pain in the affected gland. If the block- lingual frenum, which was extremely tender second molar in the mandibular occlusal age of the duct is complete, the symptoms on palpation. There was no associated dis- radiograph, which confirmed our diagnosis will be severe. Pain and swelling, may he charge or bleeding reported from the area (Figure 3C). recurrent and most pronounced during (Figure 2B). The mandibular occlusal radi- Sialolithotomy was done under local meals. In this paper we present three cases ograph revealed a radiopacity extending anesthesia and sutures were placed (Figure of large (>8mm) sialoliths of the sub- from the lateral incisor to the second pre- 3D). The retrieved sialolith was approxi- mandibular gland, treated with transoral molar area (Figure 2C). mately 1×3cm in size and weighted almost sialolithotomy and a review of existing lit- Under local anesthesia, an incision was 0.1g. Post-operative healing was smooth erature, emphasizing on the theories of eti- made at the ductal orifice and calculi was and uneventful. [page 32] [Clinics and Practice 2019; 9:1119] Case Report as a nidus for the formation of a sialolith.5 Location Discussion Another theory has proposed that an Salivary calculi related to the sub- unknown metabolic phenomenon can lead Sialoliths are condensations of calcium mandibular gland are more common than to precipitation of salivary calcium and salt primarily calcium phosphate in the the parotid gland due to some factors like phosphate ions by increasing the salivary the direction of salivary flow against gravi- form of hydroxyapatite with small amounts bicarbonate content, which in turn alters the of magnesium carbonate and ammonium. ty, a longer and more tortuous structure of calcium phosphate solubility. Wharton duct and the higher calcium and Wakely reported the distribution of A retrograde theory suggested that any sialoliths: 64% in submandibular gland and mucin content of saliva produced in the substance or bacteria of the oral cavity, that 7 duct, 20 % in the parotid gland and duct and submandibular gland. Calculi are more had migrated into the salivary ducts, can act often found within the Wharton’s duct, than 16% in the sublingual gland and duct. as a nidus for further calcification.4 Marchal Most of the sialolith are usually of 5 4 at the hilum of the duct or inside the gland. et al. further suggested that easier retro- 8 mm in maximum diameter and all the According to Pizzirani et al., they are more grade migration of materials can occur due frequently found in the left submandibular stones over 10 mm should be reported as a to variation in the sphincter-like mechanism sialolith of unusual size. Furthermore, they gland as in our case. However, few studies in the first 3 cm of the Wharton’s duct. reported a higher incidence in the right site. are classified as giant in case any dimension Recently, Sherman and McGurk6 the exceeds 15 mm. One of the largest sialoliths Locations of the stones found in our cases incidence of salivary calculi is not signifi- are listed in Table 1. of 72 mm size was reported by Rai and cantly associated with water hardness. 1 Sialoliths usually remain within the Burman. All the stones found by us were Long-standing obstruction by a sialolith between 8.5mm to 30mm size, the third gland and enlarge but they rarely migrate to may severely damage the acini of the gland, other locations; Drage et al.7 reported three case had the highest and the second case resulting in a permanent decrease, or even had the lowest dimensions. cases of migratory sialoliths in 2005. absence of salivary secretion. This reduced Sialoliths of submandibular gland detected Males are affected twice as much as or absent salivary secretion may give rise to females. All our cases were in male patients in the oral cavity or fistulized to neck have recurrent infections, which can lead to atro- been reported in the literature. Koo et al. which supports the data found by most phy of the gland with loss of secretory func- 2 reported two cases of sialolithiasis within other studies. While Seldin et al. and tion and ultimately fibrosis. only 3 the ipsilateral remaining Wharton’s duct in Lustmann et al. found the M:F ratio to be 1:1 in their studies. Etiology use Though definite etiology is still ambiguous, sialoliths are thought to occur as a result of deposition of mineral salts around an initial nidus consisting of sali- vary mucin, bacteria or desquamated epithelial cells. They form as a result of mineralization of debris that has accumulat- ed in the lumen of the duct.