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Bhat M, Rai R, Vaidyanathan V. A Rare Radiopaque

JOURNAL OF CLINICAL AND DIAGNOSTIC RESEARCH

JCDR doi:350-411 (published online first 1st Dec 2008)

JCDR doi:350-411 (published online first 1st Dec 2008) Bhat M, Rai R, Vaidyanathan V. A Rare Radiopaque Parotid Duct Calculus

Case Report

A Rare Radiopaque Parotid Duct Calculus. A Case Report

Bhat M*, Rai R**, Vaidyanathan V***

ABSTRACT

Salivary duct lithiasis is a condition characterized by the obstruction of a or its excretory duct due to the formation of calcareous concretions or sialoliths, resulting in salivary ectasia, and even provoking the subsequent dilation of the salivary gland.Sialolithiasis accounts for 30% of salivary diseases, and most commonly involves the submaxillary gland (83 to 94%), and less frequently, the parotid (4 to 10%) and sublingual glands (1 to 7%). The present study reports the case of a 50-year-old female patient who attended our clinic, complaining of a painful swelling over the cheek which aggravated with chewing movements, bad breath and foul-tasting mouth at meal times and presenting with a salivary calculus in the right Stensen´s duct. Once the patient was diagnosed with a radiopaque stone, the sialolith was surgically removed using general anaesthesia. In this paper, we have also updated a series of concepts related to the aetiology, diagnosis and treatment of sialolithiasis.

Key Words: Lithiasis, parotid diseases, radiopaque salivary duct calculi. we take into account that pain is only *Asst.Prof., Dept.of ENT, ** Asst. Prof.,Dept of one of the symptoms and that it does General Surgery,***Junior Resident,Dept. of not occur in 17% of the cases[2]. ENT, Fr. Muller’s Medical College Mangalore, Karnataka, (India). Sialolithiasis accounts for 30% of Corresponding Author: Dr. Mahesh Bhat MS (ENT), salivary diseases, and it most commonly Asst.Prof., Dept.of ENT,Fr. Muller’s Medical involves the submaxillary glands (83 to College, Mangalore, Karnataka, (India). 94%) and less frequently the parotid (4 Email- [email protected] to 10%) and sublingual glands (1 to Phone- +91- 9886734374 7%). Sialolithiasis usually appears around the age of 40, though it can have an Introduction early onset in teenagers, and it can also Salivary duct lithiasis is a condition affect old patients. It has a predilection which is characterized by the obstruction for male patients, particularly in the case of a salivary gland or its excretory duct, of lithiasis[1]. due to the formation of calcareous concretions or sialoliths, resulting in Several hypotheses have been put salivary ectasia, and even provoking the forward to explain the aetiology of these subsequent dilation of the salivary gland. calculi: mechanical, inflammatory, A further effect may be the infection of chemical, neurogenic, infectious, strange the salivary gland, which may result in bodies, etc. However, it appears that the chronic [1]. combination of a variety of these factors usually provokes the precipitation of the The clinical symptoms are clear, and amorphous tricalcic phosphate, which, allow for an easy diagnosis, whenever JCDR doi:350-411 (published online first 1st Dec 2008) Bhat M, Rai R, Vaidyanathan V. A Rare Radiopaque Parotid Duct Calculus once crystallized and transformed into the temporomandibular articulation and hydroxyapatite, becomes the initial focus. osteomyelitis affecting the ascending From this moment on, it acts as a maxillary branch. It is also important to catalyst that attracts and supports the differentiate sialoliths from other soft proliferation of new deposits of different tissue calcifications. While the former are substances[2] . Salivary calculi affecting characterized by pain and swelling of the the parotid gland, are usually unilateral, salivary gland, other calcifications such and are located in the duct. Their size is as those of the lymphatic ganglia, are smaller than submaxillary sialoliths, most symptom free. of them < 1 cm. [3], [4]. In the case of small calculi, it is Different conditions should be considered advisable to try a non-surgical treatment when carrying out the differential (spasmolitics, diet, antibiotics, etc) [6]. diagnosis of salivary duct lithiasis. The Otolaryngologists and odontologists are in unilateral enlargement of the parotid charge, together with other sanitary region is characterized by the presence professionals, of the diagnosis of salivary of a discreet, palpable mass, or either a glands diseases. They must be aware of diffuse swelling. Sialodenitis may be them and must be able to apply modern considered in the absence of this mass. A imaging techniques for their diagnosis, superficial mass in the salivary gland and if necessary, manage and treat these may suggest either a case of diseases. lymphadenitis, a preauricular cyst, a sebaceous cyst, benign lymphoid Case Study hyperplasia or extraparotid tumour. We report the case of a 50-year-old female patient, complaining about acute A mass inside the salivary gland may pain, suppuration and unilateral swelling suggest either a neoplasia (benign or in the parotid region. She also reported malignant), an intraparotid adenopathy or bad breath and a foul-tasting mouth, both a hamartoma [5]. Malignancy involving salty and sour at the same time, most the parotid gland would present with frequently at meal times. These rapid growth, facial nerve palsy, petrous symptoms disappear within a relatively texture, pain and a higher incidence rate short period, never lasting for more than among elderly patients. 2 hrs. The patient had been suffering from these symptoms for 9 days, and The differential diagnosis of the she had not noticed high temperature or asymptomatic bilateral enlargement of the any further symptoms. parotid region includes benign lymphoepithelial lesions (Mikulicz Intra-oral examination revealed a swelling syndrome), Sjogren’s syndrome and near the right parotid duct opening, sialadenitis secondary to alcoholism, which was fibrous to touch and was not long- term treatment with different drugs adhered to any deeper structure. There (iodine and heavy metals) and Whartin´s was mucopus at the duct opening. A simple tumour. Painful bilateral enlargements showed a radiopaque sialolith may result from radiotherapy, or may be located in the excretory duct [Table/Fig secondary to viral sialadenitis (including 1]. To perform the radiological diagnosis, ), whenever they co-occur with a radiographic film was placed at the other systemic symptoms. Among the level of the swelling and the beam was conditions presenting with diffuse facial kept perpendicular to the film; this way, swelling of the parotid region, but the whole calcification located in the unrelated to the glands, we must mention cheek was reflected in the film. masseter muscle hypertrophy, lesions in JCDR doi:350-411 (published online first 1st Dec 2008) Bhat M, Rai R, Vaidyanathan V. A Rare Radiopaque Parotid Duct Calculus

and satisfactory results, as regards the preservation of glandular function.

Our first decision was to treat the symptoms. Pain was treated with analgesic-anti- inflammatory drugs (diclofenac sodium, 50 mg every 8 hrs for a period of 7 days), and the bacterial infection was treated with antibiotics (cefixime 200 mg, every 12 hrs for 7 days). The patient should follow a diet rich in proteins and liquids, including acid food and drinks to stimulate the production of . Once the symptoms were controlled, we planned the treatment of the disease. Due to the location and size of the calculus, medical therapy was discarded, and the spontaneous discharge of the sialolith was aimed at. We decided on the surgical removal of the calculus. The first step of our treatment, once the sialolith had been located, was to achieve its immobilization by means of suture, to prevent it from moving along the duct during the surgical procedure.

Then, we performed an incision on the swollen region [Table/Fig 2], and a small pressure exerted at this level of the cheek, provoked the discharge of the sialolith through the incision [Table/Fig 3]. The size of the sialolith coincided with the radiographic image [Table/Fig 4] measuring 0.8 cm long.

Once the sialolith was out, the duct had The margins of the lesion were separated to be repaired and cicatrized. Two using dissecting scissors [Table/Fig 5]. possible solutions were considered in this Thus, the cicatrization of the duct was sense, anastomosis of the duct by means hindered, preventing its obliteration and of microsurgery, or diversion of salivary favouring the formation of a salivary flow by creation of an oral fistula. The fistula, creating a new access to the oral second possibility was the technique of cavity. In successive follow-up visits, we choice because of its simplicity, efficacy JCDR doi:350-411 (published online first 1st Dec 2008) Bhat M, Rai R, Vaidyanathan V. A Rare Radiopaque Parotid Duct Calculus observed the complete remission of the with areas of fibrous swelling in the symptoms, the effectiveness of the duct[1]. salivary drainage and the normal functioning of the parotid gland. The epidemiological features of our patient also coincide with those reported in the bibliography (predilection for pa- tients ≥ 40 yrs). It is seen more commonly in males, but we’ve reported in a female patient. Although the parotid gland is less frequently involved (4 to 10% of cases), it can not be considered strange or rare[2],[15]. Submaxillary duct calculi are quite common in South India, but parotid duct calculi are very uncommon in this region, and hence we consider it as a rare case presentation owing to the geographical distribution. Sialoliths are usually more or less organized hard concretions with a pale Discussion yellow colour and a porous texture. They Salivary duct lithiasis, ie: the obstruction usually have an oval or long shape, of a salivary gland or its excretory duct although we may also find some in the due to the presence of a sialolith, is form of a cast [2]. Crystallographic studies characterized by a series of symptoms. revealed the differences between parotid and The first one is salivary duct swelling, submaxillary calculi. With respect to the either without any obvious reason or at composition of parotid calculi, we must meal times. This symptom lasts for a mention the study conducted by relatively short period, not for more than Slomiany et al., who reported a total 2 hrs, and it disappears throughout the lipidic component of 8.5% and a mineral day. On some occasions, the swelling is component of 20.2% [7]. accompanied by pain, and then the patient presents with an episode of The different chemical properties of the salivary colic, an acute, lacerating pain saliva secreted by both glands explain which does not last for long and why parotid calculi have about 70% disappears after 15 or 20 mins[17]. In more organic component, 40% more this case, the patient did not have proteins and 54% more lipids than classical pain, but swelling used to submaxillary calculi [2]. change in size with chewing and was occasionally associated with pain. She The composition and size of salivary was diagnosed to have recurrent , calculi has some diagnostic implications. and was managed conservatively for the Around 20% of submaxillary gland same in the past. sialoliths and 40% of parotid ones are radiolucent due to the low mineral The evolution of this condition is component of the secretion, especially in characterized by the repetition of any of the case of parotid calculi[2]. But, in our these two clinical stages during case, we’ve reported a radiopaque parotid successive episodes. However, the duct calculi which is not so common. swelling of the gland tends to persist, it becomes indurated, and does not recover The knowledge of the clinical symptoms its normal size. Our patient presented is vital for the diagnosis of this condition, and as mentioned before, it is JCDR doi:350-411 (published online first 1st Dec 2008) Bhat M, Rai R, Vaidyanathan V. A Rare Radiopaque Parotid Duct Calculus possible that the calculi can go of the duct, as this technique could undetected despite being present, as in move the calculus to the most proximal the present case. It was treated as portion of the duct, thereby complicating recurrent parotitis, and a simple bidigital its removal [9]. is also useful palpation of stenson’s duct revealed the to locate obstructions that cannot be possibility of a calculi. However, the detected by means of bidimensional ideal approach is to be able to identify radiography, especially whenever them, and several techniques are sialoliths are radiolucid, or whenever available for this purpose. they are not present (as is the case with stenosis) [10]. Conventional intra-oral radiography may be useful, although parotid gland Computerized tomography and nuclear sialoliths are more difficult to detect than magnetic resonance can also be used for submaxillary ones, because of the the detection of sialoliths. Although these winding course of Stensen´s duct around techniques are more complex and the anterior portion of the masseter expensive than sialography, they are not muscle and through the buccinator. In invasive. According to some authors, CT general, only the sialoliths located in the scan is the technique of choice to detect anterior part of the duct, in front of the calculi inside, or near the salivary masseter muscle, can be visualized by glands. Its sensitivity makes it possible means of intra-oral radiography [8]. to detect recently calcified calculi, which go undetected through conventional Conventional extra-oral radiography is of radiography [9]. limited use, because most of the images of parotid gland sialoliths are superposed Though scyntigraphy is not clearly on the body and on the maxillary branch. indicated for the diagnosis of The sialoliths located in the distal part sialolithiasis, on some occasions, it may of Stensen´s duct or within the parotid be useful as a complementary exploratory gland, are difficult to see by means of technique. A functional study of salivary lateral intra-oral or extra-oral glands is accomplished by means of radiographies. Nevertheless, a scyntigraphy. When the Tc 99m- posteroanterior image of the swollen pertechnetate is intravenously cheek may make the sialolith become administered, it concentrates and is detached from the bony area, thus excreted through such glandular structures making it visible. Even so, the sialoliths as the salivary gland, thyroid and with low mineral content may become mammary glands. After a few minutes, it darkened by the shadows cast by dense is possible to detect the radioisotope in soft tissues in posteroanterior images [8]. the salivary gland ducts, and it achieves its higher concentration level 30-45 Sialography is the most adequate minutes after infusion [8]. technique to detect salivary gland calculi, as it allows for the visualization of the Scyntigraphy allows for the analysis of whole duct system. Submaxillary and all salivary glands at the same time. In parotid glands are more easily studied by the event of a suspected sialolithiasis, means of this technique, than sublingual this technique is mainly applied when glands. However, sialography is not sialography is not indicated, and in indicated in the case of acute infections patients with non permeable glandular or in patients who are sensitive to ducts. Functional salivary pathology substances containing iodine. It should (either lithiasic or not) can be detected not be used either, if a radiopaque by the increase, reduction or absence of calculus is observed in the distal portion radioisotope uptake areas [8]. JCDR doi:350-411 (published online first 1st Dec 2008) Bhat M, Rai R, Vaidyanathan V. A Rare Radiopaque Parotid Duct Calculus

been used as an alternative to medical Recent studies show that ultrasonography therapy, whenever the latter was not may also be useful for the diagnosis of possible or when it proved ineffective. duct sialoliths [11]. is a The case reported here, exemplifies this relatively new technique to detect causes therapeutic management. Surgical removal of obstruction in the parotid duct and of the calculus has the disadvantage of gland directly, and to manage the chronic compromising the facial nerve, depending obstructive parotitis, efficiently combined on the location of the sialolith. Extra- with continuous lavage and perfusion oral surgical techniques are not indicated, simultaneously [18]. Methylene blue has because of the risk of leaving been used to identify the duct papilla as antiaesthetic scars, and also because sialoendoscopy can be problematic, intra-oral surgery has proved more wherein the dye is tipped around the effective [6]. A recently published technique papilla site, and as the saliva is squeezed prevents these complications. It consists of out, the opening becomes prominent, and the use of ultrasound expansive waves to 1 or 2 minutes, the saliva will lead to provoke the fragmentation of the some washout effect of methylene blue, calculus. Moreover, it does not require leaving a circle of brighter tissue amid anaesthesia, sedation or analgesia. The the deep blue of the orifice [19]. procedure lasts for about 30 minutes, and is administered in a series of In short, there is not a single technique successive weekly sessions, until all the for the diagnosis of salivary gland fragments of the calculus are eliminated, sialolithiasis, and we must select the using sialogogues as coadjuvant most adequate technique according to the therapy[12]. Some authors treat circumstances and pathology to be sialolithiasis by means of intraductal treated. We must obtain an accurate instillation of penicillin or saline. The diagnosis and at the same time, we low recurrence rate proves, according to should minimize the risk and these authors, the efficacy of this method inconvenience for the patient. in comparison with the systemic administration of drugs. Moreover, this A diet rich in proteins and liquids therapy offers the advantage of acting at including acid food and drinks, is different levels : it dilates ducts, dislodges advisable in order to avoid the formation sialoliths adherent to the walls of ducts of further calculi in the salivary gland and flushes out the obstructing [1]. coagulated albumin[13]. As regards the surgical removal of calculi located in In the case of small calculi, the Stensen´s duct, there are several possible treatment of choice should be medical, solutions. The most conservative instead of surgical. The patient can be technique is the anastomosis of stensen´s administered natural sialogogues such as duct by means of microsurgery. Another small slices of lemon or sialogogue feasible option is the creation of a medication. Drugs stimulating ductal salivary fistula, which is an easier contraction such as pilocarpine can be technique yielding equally positive prescribed, as also the application of results, as far as glandular function is short-wave infrared heating. However, if concerned. In the case of this latter the calculus is of a medium or large technique, once the sialolith has been size, a salivary colic may occur and the removed, the margins of the lesion are calculus may not be discharged. separated, thus avoiding duct collapse in the cicatrisation process. This way, we Surgical removal of the calculus (or even favour the formation of a salivary fistula of the whole gland) has traditionally JCDR doi:350-411 (published online first 1st Dec 2008) Bhat M, Rai R, Vaidyanathan V. A Rare Radiopaque Parotid Duct Calculus

which acts as a new access to the oral [4].Ottaviani F, Galli A, Lucia MB, Ventura G. cavity [14],[16] . Bilateral parotid sialolithiasis in a patient with acquired immunodeficiency syndrome Inhibition of salivary gland function is and immunoglobulin G multiple myeloma. Oral Surg Oral Med Oral Pathol Oral Radiol hardly employed, with the exception of Endod. 1997; 83: 552-4. cases of sialorrhoea, due to the possible [5].Marchal F, Becker M, Vavrina J, Dulgerov P, complications associated with this Lehmann W. Diagnostic et traitement des technique, and the reduction of salivary sialolithiases. Bull Med Suisses 1998; flow it provokes. The technique consists 79:1023-8. in the closure of the duct with suture, [6].Dulguerov P, Marchal F, Lehmann W. once the calculus has been removed. Postparotidectomy facial nerve paralysis: This provokes the collapse and possible etiologic factors and results with routine facial nerve inflammation of the gland. Atrophy is monitoring.Laryngoscope 1999;109: 754-62. achieved by means of pressure and the [7].Slomiany BL, Murty VL, Aono M, Slomiany A, administration of successive drugs to Mandel ID. Lipid composition of human attain this goal [14]. All the therapies parotid salivary gland stones. J Dent Res. described here, require the previous 1983; 62: 866-9. treatment of symptoms, and all of them [8]. Goaz PW, White SC, eds. Radiología oral principios e interpretación. Barcelona: confirm that there is not a single Mosby; 1995. p. 127-229. therapeutic approach to the treatment of [9].Hong KH, Yang YS. Sialolithiasis in the obstructive sialolithiasis, as it can be . J Laryngol Otol 2003; successfully treated by using different 117:905-7. techniques and even by a combination of [10]. Becker M, Marchal F, Becker CD, some of them. The use of one or Dulguerov P, Georgakopoulos G, Lehmann W another technique depends to a large et al. Sialolithiasis and salivary ductal extent on the sialolith size, location and stenosis: diagnostic accuracy of MR sialography with a three-dimensional composition. extended-phase conjugate-symmetry rapid spin-echo sequence. Radiology 2000; Conclusion 217:347-58. In conclusion, parotid duct calculus is a [11]. Nguyen BD. Demonstration of renal rare entity, treated conservatively by lithiasis on technetium-99m MDP bone many physicians as recurrent parotitis, scintigraphy. Clin Nucl Med 2000; 25:380-2 which is very disabling to the patient. [12]. Schlegel N, Brette MD, Cussenot I, Monteil JP. Extracorporeal lithotripsy in the The need for a clinical intraoral treatment of salivary lithiasis. A prospective examination and a plain radiograph study apropos of 27 cases. Ann Otolaryngol cannot be overemphasized. With advent Chir Cervicofac 2001; 118:373-7. of newer diagnostic modalities, the [13]. Antoniades D, Harrison JD, Epivatianos smallest duct calculus can be detected. A, Papanayotou P. Treatment of chronic sialadenitis by intraductal penicillin or saline. J Oral Maxillofac Surg 2004; 62:431- References 4. [1].Lustran J, Regev E, Melamed Y. [14]. Steinberg MJ, Herrera AF. Management Sioalolithiasis: a survey on 245 patients and of parotid duct injuries Oral Surg Oral Med review of the literatura. Int J Oral Oral Pathol Oral Radiol Endod 2005; 99:136- Maxillofac Surg. 1990;19: 135-8. 41. [2].Bodner L. Parotid sialolithiasis. J Laryngol [15]. Torres LD, Barranco PS, Serrera FMA, Otol 1999;113: 266-7. Hita IP, Martinez SMA, Gutierrez PJL. [3]. Seifert G, Miehlke A, Hanbrich J, Chilla R, Parotid sialolithiasis in Stenson’s duct. Med eds. Diseases of the salivary glands: Oral Patol Oral Cir Bucal. 2006; Jan 1; pathology, diagnosis, treatment, facial 11(1): E80-4 nerve surgery. Stuttgart:George Thime [16]. Park MJ, Mandel L. Parotid stone Verlag; 1986. p. 85-90. removal. N Y State Dent J. 2007; Jan; 73(1): 54-6 JCDR doi:350-411 (published online first 1st Dec 2008) Bhat M, Rai R, Vaidyanathan V. A Rare Radiopaque Parotid Duct Calculus

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JCDR doi:350-411 (published online first 1st Dec 2008)