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Table of Contents   
CASE REPORT  
Year : 2012  |  Volume : 23  |  Issue : 3  |  Page : 411-414
Down's syndrome patient with multiple sialoliths in Stenson's duct


1 Department of Prosthodontics, Sinhgad Dental College and Hospital, Pune - 411 041, Maharashtra, India
2 Department of Oral Pathology, Sinhgad Dental College and Hospital, Pune - 411 041, Maharashtra, India

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Date of Submission14-May-2011
Date of Decision23-Aug-2011
Date of Acceptance15-Feb-2012
Date of Web Publication11-Oct-2012
 

   Abstract 

The presence of multiple sialoliths (calculi) in parotid duct is considered extremely rare. Parotid duct sialoliths are usually small and single. Multiple calculi formation, in any case, is an uncommon finding in the salivary glands. Salivary lithiasis is comparatively common in the Wharton's duct due to its tortuous ascending course and chemical composition of saliva of submandibular gland. The mechanism of sialolith formation in Down's syndrome patients is not completely known, and there are seldom cases reported in the literature available. The present case report is a very rare combination of a Down's syndrome with multiple sialoliths in Stenson's duct of left parotid gland in a 21year old male patient.

Keywords: Calculi, Down′s syndrome, parotid duct, sialolith, stenson′s duct

How to cite this article:
Palaskar J, Palaskar S, Joshi N. Down's syndrome patient with multiple sialoliths in Stenson's duct. Indian J Dent Res 2012;23:411-4

How to cite this URL:
Palaskar J, Palaskar S, Joshi N. Down's syndrome patient with multiple sialoliths in Stenson's duct. Indian J Dent Res [serial online] 2012 [cited 2020 Jul 8];23:411-4. Available from: http://www.ijdr.in/text.asp?2012/23/3/411/102242
Down's syndrome is the most prevalent chromosomal abnormality found in humans; according to Buxton and Hunter, every year, 1 of every 800 children in the U.S. is born with this syndrome. [1] This condition is caused by a trisomy of chromosome 21, that results in mental retardation, short stature and phenotypic abnormalities. [1] Sialolithiasis is blockage of salivary gland or its ducts. The symptoms associated with this condition are swelling, pain, infection of the affected gland. [2] The clinical symptoms include an increase in the size of the affected gland and increased salivary secretion that results in pain during eating. [1] Since the blockage rarely causes complete obstruction, the retained saliva seeps past the obstruction, and gland swelling and discomfort subsides gradually once the patient stops eating. [1]


   Case Report Top


A 21-year-old male patient with Down's syndrome was brought to the private clinic by his parents with a complaint of recurrent painless swelling of left side of the face during meal time since last 2 months [Figure 1]. This swelling disappears within a relatively short time after meals, never lasting for more than 2 hours. This swelling was not associated with rise in temperature. On physical examination, patient presented with features.
Figure 1: Swelling of Stenson's duct (left side) just after an intake of food

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Down's syndrome such as head and face with flattened back of head, with one eye lid a little droopy, slightly flattened bridge across nose, smallish ears, smallish mouth, and slightly protruding tongue. Hand showing slightly shortened fingers. Foot with slightly enlarged gap between the big and second toes; and in addition, he was unable to speak. Patient was co-operative as far as clinical examination was concerned, but his parents were apprehensive that he would not be the same for any invasive procedure. He had a history of some dental fillings in deciduous dentition under general anesthesia.

On intra-oral examination, patient presented a good oral hygiene having no carious/filled teeth with healthy gingiva. On thorough examination of buccal mucosa, opening of Stenson's duct was visible on the right side, but it was not obviously visible on the left side. On palpation through the course of the Stenson's duct, no calculi or sialoliths were palpable. No significant changes appeared on an examination of left parotid gland.

The clinical symptoms were characteristically clear and allowed for an easy, probable diagnosis of sialolithiasis of the left parotid gland. In order to confirm the probable diagnosis, radiographs were used by keeping the occlusal X-ray film on the left buccal mucosa, but no sialoliths could be located. Patient was then referred for ultrasonography, as a first line of investigation, which revealed multiple sialoliths of <1 cm size around the right angle bend of the Stenson's duct of left parotid gland [Figure 2]. The patient was then referred for non-contrast helical computerized tomography (CT) to confirm the location, number and size of the sialoliths. After confirmation on CT scan and giving due consideration that the patient had Down's syndrome, an early subsiding recurrent swelling (within 2 hrs), no pain or signs of an infection, the relatively inaccessible anatomical location, the small size and multiple number of the sialoliths, it was decided to treat the patient non-invasively. Accordingly, the patient was referred for shockwave lithotripsy as it is one of the better non-invasive treatment options.
Figure 2: Ultrasonograph of left Stenson's duct showing multiple sialoliths, one of which measures 3.06 mm

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   Discussion Top


Diseases that obstruct the ducts can also cause sialoadenitis, the most common condition being sialolithiasis. [1] An incomplete obstruction by a sialolith may be associated with secondary infection of the gland; bacterial infections commonly ascend from an oral cavity because of the decreased salivary flow. When a complete obstruction occurs, glandular atrophy eventually ensues. [2]

Parotid duct stones are usually small in size compared to submandibular gland stones, and are usually single. [1] The presence of multiple calculi in the parotid gland is considered extremely rare. [3] Sialolithiasis most commonly involves the submandibular gland (83% to 94%), less frequently the parotid gland (4% to 10%), and sublingual glands (1% to 7%). [4] In Down's syndrome, Sialolithiasis is uncommon with limited documented epidemiological data. The present case is a unique case of Down's syndrome with multiple sialoliths in left Stenson's duct of parotid gland.

The mechanism of calculi (sialolith) formation in Down's syndrome is not yet completely known; as such this is rarely reported as per the dental literature available. Probably high concentration of calcium salts may be possible etiological factor. Calculi generally consist of a mixture of various calcium phosphates (hydroxyl apatite, carbonate apatite) together with an organic matrix. An initial organic nidus progressively grows by deposition of the layers of organic and inorganic substances. A more inorganic calculus is more radio-opaque and can be more easily detected in plain x-rays. The use of an ultrasound in the diagnostic workup solved these problems as calculi >2 mm can be identified, independently of their composition. [3]

Most salivary calculi are small; usually less than 1 cm, but megaliths or giant calculi have been reported. [5] They are composed of mineralized debris that accumulates within the duct lumen including calcium phosphate, carbon, and trace amounts of magnesium, potassium, and ammonium. [5] Salivary calculi grow by deposition at an estimated rate of 1 mm to 1.5 mm per year. [5] In the parotid duct, multiple salivary calculi are rare. [5]

In this present case, plain x-rays were taken by keeping the occlusal film between the cheek and teeth on the left side of the face, but it could not locate tiny calculi, therefore, the patient was referred for an ultrasound examination, which revealed multiple calculi (<1 cm each) in Stenson's duct of left parotid gland. The Stenson's duct is approximately 5 cm in length and follows a slightly uphill course as it exits from the gland on its path toward its intra-oral orifice. The duct traverses the lateral surface of the masseter. At the muscle's anterior border, it makes a right angle bend, perforates the buccinator muscle, and exits on the buccal mucosa adjacent to the maxillary second molar. [6] Location of multiple sialoliths in the present case was around the right angle bend of the Stenson's duct through the buccinator muscle, which is rather difficult to approach surgically.

Diagnostic imaging to identify presumed salivary calculi include conventional radiography, sialography and ultrasonography. But currently, high-resolution non-contrast computerized tomography (CT) scanning is an imaging modality of choice for an evaluation of salivary stones. This is because many sialoliths are not detected by the conventional radiography until they are 60% to 70% calcified, [5] with at least 20% of submandibular and 50% of parotid stones not identifiable on an intra-oral and panoramic radiography. Non-contrast helical CT with multiplanar reconstructions seems to be the gold standard for the diagnosis of lithiasis, especially when the small and poorly calcified sialoliths may not be visible on standard radiographs. CT allows an accurate characterization of the number and position of the lithiasis. [7]

In sialography, dye is injected into the duct, and it can demonstrate an obstruction as a filling defect in the duct and duct stenosis. It cannot, however, demonstrate the small secretion plugs or secretion plaques, and it is contraindicated in an acute infection, or in the patients with a significant contrast allergy. Ultrasonography (US) identifies calculus as white echogenic structures with glandular inflammatory changes of the salivary gland. In spite of its limitations, US represent an excellent first line imaging technique, because it is non-invasive and widely available. [7]

Sialography was the most preferred diagnostic tool in the recent past to confirm and locate the sialoliths. It was avoided in this patient because it has inherent disadvantages; it is an invasive method that is associated with complications such as bleeding, traumatic perforation or rupture of the duct and side effects from contrast material. [8] Indications for sialography have become rare, and radiologists are less and less experienced for the salivary gland cannulation. [8] Also, parents of the patient were apprehensive of this invasive procedure and hence it was ruled out. Computed Tomography and nuclear magnetic resonance can also be used for the detection of sialoliths. Although these techniques are expensive than the sialography, they are non-invasive. Crystallographic studies reveal the compositional difference between the parotid and submandibular calculi. The different chemical properties of the saliva secreted by both the glands explains why parotid calculi have about 70% more organic component, 40% more proteins and 54% more lipid as compared to the submandibular calculi. [9]

Around 20% of the submandibular gland sialoliths and 40% of the parotid ones are radiolucent due to the low mineral component of the secretion, especially in the case of parotid calculi. [9] Parotid gland salivary calculi are usually unilateral and are located in the duct. Their size is smaller than the submandibular sialoliths, most of them are <1 cm. [4],[10] Small caliber endoscopy was developed to treat obstructive disorders of the salivary gland duct system. [5] It is both diagnostic and therapeutic, and has the benefit of differentiating between obstructive inflammatory conditions and calculi. Despite most sialoliths being composed of calcium elements, they are not associated with systemic calcium abnormalities; therefore, a serum calcium level evaluation is not always needed. [5] Sialoendoscopy, fluoroscopy-guided wire basket extraction, lithotripsy, and surgical removal are the other options. The decision about which technique to utilize depends on the stone size, location, and procedure availability. The stone will stay in the gland until it is removed. Typically stones, which are <1 cm in diameter can be treated without surgical intervention. A conservative approach, including an oral analgesia, hydration, local warm heat therapy, massage to milk out the stone, sialogogues (i.e.tart hard candies) to promote the ductal secretions, and discontinuation of anticholenergic medications when possible, are recommended. In most cases, removing the stone will relieve pain except when an associated infection exists. Antibiotics covering an oral flora for gland super infection are recommended.

Surgical removal in cases with small calculi is best avoided; it has the disadvantage of compromising the facial nerve, depending on the location of the sialoliths. Extra oral surgical techniques are not indicated because of the risk of leaving anti esthetic scars, and an intraoral surgery proved to be very difficult at that location. However, severe obstruction usually requires surgical intervention, especially when the obstruction is close to the gland. [11]


   Conclusion Top


The reported cases of multiple sialoliths of Stenson's duct in combination with Down's syndrome are rare and need to be investigated further in terms of occurrence, prevalence, co-relating factors (if any), and etiology. This case report adds to the scant literature on this subject, and it is hoped that this may help in future research.

 
   References Top

1.Estela K, Paulo RF, Staell F, Ronaldo C, Paulo RF. Parotid duct sialolithiasis in a patient with Down syndrome - Case report. Gen Dent 2005;53:421-2.  Back to cited text no. 1
    
2.Sumi M, Izumi M, Yonetsu K, Nakamura T. The M R Imaging assessment of submandibular gland sialoadenitis secondary to sialolithoiasis: Correction with CT and histopathologic findings. AJNR Am J Neuroradiol 1999;20:1737-43.  Back to cited text no. 2
[PUBMED]    
3.Konstantinidis I, Paschaloudi S, Triaridis S, Fyrmpas G, Sectilidis S. Bilateral multiple sialolithiasis of the parotid gland in a patient with Sjogran's syndrome. Acta Otorhinolaryngol Ital 2007;27:41-4.  Back to cited text no. 3
    
4.Ottiviani F, Galli A, Lucia MB, Ventura G. Bilateral parotid Sialolithiasis in an acquired immunodeficiency syndrome and immunoglobulin G multiple myloma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;83:552-4.  Back to cited text no. 4
    
5.Wiler JL. Diagnosis: Salivary gland stone/sialolith. Emerg Med News 2006;30:6-8.  Back to cited text no. 5
    
6.Moore KL, editor. Clinically oriented anatomy. 3 rd ed. Baltimore: Williams and Wilkins; 1992. p. 670.  Back to cited text no. 6
    
7.Faye N, Tassart M, Périé S, Deux JF, Kadi N, Marsault C. Imaging of salivary lithiasis. J Radiol 2006;87:9-15.  Back to cited text no. 7
    
8.Tassart M, Zeitoun D, Iffenecker C, Bahlouli F, Bigot JM, Boudghène F. MR Sialography. J Radiol 2003;84:15-26.  Back to cited text no. 8
    
9.Bodner L. Parotid Sialolithiasis. J Laryngol Otol 1999;113:266-7.  Back to cited text no. 9
[PUBMED]    
10.Lagares DT, Piedra SB, Fiallo MA, Iglesias PH, Sahuquillo MA, Perez JL. Parotid sialolithiasis in Stenson's duct. Med Oral Patol Oral Cir Bucal 2006;11:E80-4.  Back to cited text no. 10
    
11.Pinto A. Pediatric soft tissue lesions. Dent Clin North Am 2005;49:241-58.  Back to cited text no. 11
[PUBMED]    

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Correspondence Address:
Jayant Palaskar
Department of Prosthodontics, Sinhgad Dental College and Hospital, Pune - 411 041, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.102242

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