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.

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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. This debris includes bacterial colonies, exfoliated duc- tal epithelial cells, mucus plugs, foreign bodies or other cellular debris. Factors like stagnation of salivary flow, dehydration, commercial and change in salivary pH associated with oropharyngeal sepsis, impaired crystalloid solubility, high alkalinity, and increased cal- A B cium content, and physical traumaNon to sali- vary duct or gland may predispose to calcu- lus formation. The definite etiology of our cases still remains unknown. Two stages of sialolith formation can be found in the literature: i) central core forma- tion and ii) layered periphery formation.4 Firstly, mineral salts bound by certain organic substances precipitate to form the central core. Then, in the second phase, some organic and inorganic materials deposit around the central core in layers. Parotid and submandibular stones are thought to frequently form around a nidus of inflammatory cells or foreign body and a C D nidus of mucous respectively. Boynton and Lieblich in 2014 reported an unusual case in Figure 1. (A) Patient’s profile; (B) swelling in floor of the mouth; (C) mandibular occlusal radiograph showing sialolith in the submandibular region; (D) sialolithotomy and sutur- which a facial hair of the patient got ing of the defect. entrapped in the Wharton’s duct and acted

[Clinics and Practice 2019; 9:1119] [page 33] Case Report patients with isolated aplasia of a unilateral superior gland or proximal Wharton’s duct our stones. It is very rare to find a patient submandibular gland.9 may get superimposed on the teeth or with both radiopaque and radiolucent mandible, which can make visualization stones. So, finding one radiopaque stone is Clinical features difficult in extraoral radiographs. In the enough to rule out the possibility of finding Submandibular gland sialolithiasis is study of Lustmann et al., intraoral radi- any additional radiolucent stones. Other cal- generally asymptomatic in nature. The ographs detected 94.7% of the cases. cifications in the area, which may confuse symptoms include pain and swelling of the However, in extraoral radiographs stones the diagnoses, are: i) phleboliths or calcifi- involved gland caused by the accumulation were superimposed on bony structures and cations of intravascular thrombi either in a teeth so, their identification was difficult, hemangioma or in the lingual veins; ii) cal- of saliva due to blockage of the lumen of 2 Wharton’s duct by a salivary calculus. especially when they were small. cified cervical lymphadenopathy, which Recurrent infections may occur due to the Supporting this, intraoral mandibular may have occurred in relation to tubercu- ascent of bacteria into the parenchyma of occlusal radiographs precisely revealed all lous infection; iii) arterial atherosclerosis of the gland. All our patients suffered from pain and infection for 1 week to as long as 2-3 years. Table 1. Details of the sialoliths. Diagnosis Case report #1 Case report #2 Case report #3 Sialoliths are traditionally diagnosed by Sex Male Male Male clinical features coupled with radiographs, Age (years) 40 25 75 though newer more sophisticated tech- niques are now available. Gland involved Submandibular Submandibular Submandibular Side involved Left Left Right Investigation and palpation Location of the sialolith Inside the canal In the canal orifice Inside the canal The diagnosis of salivary calculi is Size of the sialolith 1.5×0.5 cm only 5×8.5mm 1×3cm based on the patient’s history and clinical Weight (in grams) 0.05g 0.01g 0.1g examination which are supplemented by radiographic findings. The patient may present with pain and swelling of the con- cerned gland at meal times and in response use to other salivary stimuli, pus may be seen draining from the duct and signs of sys- temic infection. Siddiqui found that small and sometimes even large sialoliths can be asymptomatic. A posterior to anterior bimanual palpa- tion of the floor of the mouth reveals sub- mandibular stones in a large number of cases. As all our stones were inside the Wharton’s duct, they were palpable on the floor of the mouth. Intraoral palpation around Stenson’s duct orifice may reveal a commercial parotid stone, though deeper stones are often not palpable. When minor salivary glands are involved, they are usually in the A B buccal mucosa or upper lip, formingNon a firm nodule that may mimic tumor. In all our cases, a diffuse hard swelling was found on the floor of the mouth of the affected side and the first and third cases were associated with pus discharge.

Radiograph When no stone can be seen or palpated, imaging studies can be a great help in diag- nosing sialolithiasis. Occlusal radiographs are extremely useful in showing radiopaque stones unless otherwise there are radiolu- cent stones. Submandibular gland calculi are mostly C D radiopaque (80% to 94.7%). According to Williams, standard extraoral radiographs Figure 2. (A) Patient profile; (B) intraoral swelling in submandibular region; (C) mandibular occlusal radiograph showing sialolith in the submandibular region; (D) are less diagnostic than intraoral radi- sialolithotomy and suturing of the defect. ographs or occlusal views. Stones in the

[page 34] [Clinics and Practice 2019; 9:1119] Case Report the lingual artery demonstrating calcifica- which leads to fluid extravasation in the Usually, a CT scan is done both with or tions along the floor of mouth. floor of the mouth. Temporary injury of the without contrast enhancement to differenti- lingual nerve has been also reported in the ate between calculi and vascular structures. Sialography literature. Magnetic resonance imaging is another very helpful tool for the identification of Sialography, which was previously used smaller stones and to differentiate acute from widely for detection of duct stricture, steno- Ultrasonography chronic obstruction. Sialo-Magnetic sis or sialoliths, is a method of injecting Katz et al. and various other authors Resonance Imaging is a non-invasive contrast material into Stenson’s or have reported that ultrasonography is a very method of diagnosing sialoliths. It does not Wharton’s duct for visualization. useful diagnostic tool for submandibular use any contrast vehicle and ionizing radia- Sialographic studies may reveal fea- sialolithiasis with a sensitivity and speci- 15 tion overcoming, the major disadvantages of tures of the involved salivary glands, having ficity between 90% and 95% respectively. both computed tomography and contrast important diagnostic value. The anatomy of However, a major drawback of this proce- sialography. Acute inflammation is not a the duct can be displayed, revealing its form dure is that smaller calculi (>2mm) remain contradiction to this technique, which is a as narrow or large, the presence of second- undetected most of the times. great advantage. The detailed morphology of ary branches leaving the main duct, and the the salivary ducts, up to second and third presence of accessory glands or sialolithia- Newer techniques order branches can be seen with this tech- sis, including their dimensions, number, and Newer techniques such as Computed nique, without the need for duct cannulation. positions. Sialography is a very helpful tool tomography (CT) and Magnetic resonance in cases when signs of sialadenitis are relat- imaging (MRI) have revolutionized the Management ed to a radiolucent or deeply impacted sub- diagnostic aspect of sialolithiasis. mandibular/parotid stone. Sialography was Computed tomography is very helpful Preservation of gland function in con- chosen over the other tools by Katz,10 in cases where other techniques provide junction with low-level risk and discomfort Hasson and Nahlieli,11 for salivary gland ambiguous results, especially in parotid for the patient should be the primary objec- assessment. On the other hand, Marchal and duct calculi. An associated abscess or ranula tive in the treatment of sialolithiasis. The Dulguerov12 have said that there is always a can be easily detected by CT scan in conju- treatmentonly of choice varies according to the risk of pushing the sialoliths deeper into the gation with soft tissue and bone algorithm. size, location and the number of stones. duct while injecting the contrast media, which may further complicate their removal. This was supported by Varghese et use al.,13 who claimed that the major disadvan- tage of sialography is its invasiveness. They studied 49 patients and concluded that mag- netic resonance sialography is more sensi- tive to tight strictures when compared to conventional sialography but not when sali- vary stones are present. Patients with acute infection and con- trast allergy is an absolute contradiction of sialography.4

Sialendoscopy commercial Introduction of sialendoscope has taken the diagnosis of these pathologies to the A B next level. Katz and Fritsch,10 used a flexi- ble endoscope for the first time Nonin the 1990s for evaluation of the salivary gland ducts. Sialendoscopy was upgraded through the ages by the advancements in technology resulting in enhancement of optical resolu- tion and miniaturization of the instruments. In addition to the radiolucent stones and those not shown in the imaging techniques, any possible duct stricture or other kinds of obstructions can also be found with this technique. In the case of a small sialolith and favorable conditions, a definitive sialolithotomy can be performed with sial- endoscopy. Kondo et al. recently proved its diagnostic value in patients with swelling of C D the parotid gland when sialoliths are unde- tected with computed tomography.14 Figure 3. (A) Patient profile showing no facial asymmetry; (B) sialolith protruding out of the duct orifice; (C) mandibular occlusal radiograph showing sialolith in the second Duct perforation is the most usual and molar region; (D) excised sialolith. common complication of the procedure,

[Clinics and Practice 2019; 9:1119] [page 35] Case Report

Conservative management ment of sialolithiasis as Gallo et al. found Intervention can be carried out during acute When stones are small, moist warm that nearly 40% of submandibular stones stages without spreading of infection, and are distal.23 with minimal post-operative pain and heat application with the administration of 2 sialogogues and gland massage help in edema. flushing the stone out of the duct. Small Surgical techniques A number of complications are associat sialoliths can be removed through the duct Upon failure of non-invasive techniques ed with an open surgical approach. The pri- orifice using bimanual palpation. The infec- mainly due to the size of the stones and their mary concern is Postoperative neurologic tion should be treated with antibiotics and location, the Sialoliths are treated by open damage. A major cause of patient morbidity these cases should be combined with simple surgical approaches. These include trans- includes various other complications, sialolithotomy when required. oral duct incision, purely external approach- including sialoceles, salivary fistula, facial es, or a combination of approaches. An scarring and Frey’s Syndrome. There is a Sialendoscopy Endoscopy assisted trans-oral removal tech- greater risk of facial palsy as in parotid or Stenons sialolithiasis, there are branches of Sialendoscopy, which is a minimally nique for larger or impacted stones can also be of utility. the facial nerve that cross over the duct. invasive technique, was first introduced by There might be instances of nonclosure of 10 Zenk et al. concluded in their study that Katz in 1991 and has since been developed the duct because of an external incision, 16 17 transoral removal should be the treatment of by Marchael et al. and Nahlieli et al. which might lead to salivary fistula. There have been many reports on choice in patients with submandibular stones that can be palpated bimanually and Extracorporeal and Endoscopic Sialendoscopic surgery for submandibular Intracorporeal Shockwave Lithotripsy: gland sialolithiasis which discussed the localized by ultrasound within the perihilar 24 There have been alternative techniques region of the gland. The minimum diame- usage of sialendoscopy for the removal of that have been found out which includes the 18 ter of our stones being 8.5mm and localiz- sialoliths. Klein and Ardekian mentioned usage of Extracorporeal Shock Wave able by palpation and radiographs, we chose that sialoliths upto the range of 4-5mm of Lithotripsy and the most recent being using the intraoral submandibular duct approach diameter are the ideal for sialendoscopic Endoscopic Intracorporeal Shock Wave over the other techniques. To minimize the removal while it is a challenge for the sur- Lithotripsy, where direct shockwaves are risk of injury to the sublingual glands, Park only geons to perform sialendoscopy of the delivered to the surface of the stone lodg- et al. have described an approach which has sialoliths located deep within the hilum. ment inside the duct without damaging an added advantage of accessing the hilum There are speculations regarding the endo- adjacent tissue (piezoelectric principle). A scopic approach since a general consensus is of the submandibular duct without full dis- 25 better option than surgical removal of the section of the duct. The surgical landmark yet to arrive on either the maximum diameter useaffected gland is Salivary Lithotripsy which is assessed by the triangle formed by the of the stones that could be removed without is more useful and beneficial, as this pre- posterior edge of the mylohyoid muscle, the fragmentation or whether impacted or hard vents the risks of General Anaesthesia, lingual nerve (retracted medially) and the stones could be managed effectively by Damage of the facial nerve, surgical scar, 19 medial margin of the mandible. endoscopy alone. The general indication Frey’s syndrome, and a minimum of dis- General Anaesthesia is preferred when for the approach is the combination of sialen- comfort to the patient, but also with the the stones are approached using the external doscopy with extra-corporeal lithotripsy preservation of the gland. (Storz SL1 Minilith) using Thullium laser open method or difficult intra-oral (Revolix) for the fragmentation of Lithiases approaches when the stone is located at or between the range of 4 and 8mm. proximal to the hilum of the submandibular Interventional sialendoscopy, particu- duct. General Anaesthesia is also preferred Conclusions larly in procedures involving large stones or for combined endoscopic/external stenotic ducts did fail even in the hands of approaches.commercial In cases requiring a trans-cervi- Sialoliths should be always considered experienced surgeons (about 20% failure cal approach, a shoulder roll is generally in submandibular and facial pain particular- rate). Marchal devised a technique to pre- used to hyperextend the neck slightly. ly when it is related to mealtime. A careful serve the glands by a procedure involving Although advances in sialoendoscopy history and correct imaging techniques are combined transoral Sialendoscopy and an and minimally invasive surgery have mini- required to confirm the clinical diagnosis external approach to the Sialoliths.Non While mized the need for salivary gland excision, and to define the precise location of the cal- involving the combined open and still there are some situations where exci- cification. Sialendoscopic approach, Marchal made sion is inevitable. This takes into account Though advanced and more efficient sure to stress on the exploration of the the inclusion of symptomatic or recurrent methods are available occlusal radiographs remaining of Wharton’s duct sialadenitis which is caused by multiple are still useful in diagnosing sialoliths. Sialendoscopically to rule out any remain- intraparenchymal stones or due to the pres- Small stones can be treated conservatively ing fragments.20 ence of a very large stone, intraoperative but for larger stones, sialolithotomy com- The measurement of the minor axes of complications of sialoendoscopy which bined with antibiotics is the treatment of the sialoliths with a soft tissue CT scan was requires gland removal, or inability in choice if newer technologies are not avail- correlated with treatment outcome of sial- extracting the stone when minimally inva- able. According to literature, the success endoscopy alone by Kondo et al.21 and the sive procedures listed above are used and/or rate of the intraoral submandibular duct measurement of the major axis showed no residual symptoms which are present approach is between 85% and 100%.24,25 correlation with outcomes of sialendoscopy despite the removal of stones such as recur- This submandibular duct technique is sim- alone. rent inflammation. The excision of sub- ple, adaptable to different anatomic situa- Retrograde sialendoscopy, described by mandibular gland for chronic sialadenitis is tions and, can be performed under local Potash and Hoffman,22 is a novel technique more frequent and is secondary to lithiasis. anesthesia. Immediate and permanent post- that is useful as an adjunct to standard sub- The CO2 laser enables bloodless sur- surgery complications, such as lingual mandibular gland resection in the manage- gery and a clear vision of the operating site. nerve injury, are exceptional.

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Non

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