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Year : 2020  |  Volume : 15  |  Issue : 2  |  Page : 292-294

Submandibular gland wharton's duct phlegomous calculus sialoadenitis: A rare case report

Department of General Surgery, Jawaharlal Nehru Medical College, Datta Meghe Institute of Medical Sciences (Deemed to be University), Sawangi (Meghe), Wardha, Maharashtra, India

Date of Submission05-Mar-2020
Date of Decision10-Mar-2020
Date of Acceptance25-Mar-2020
Date of Web Publication21-Dec-2020

Correspondence Address:
Dr. Suresh R Chandak
Department of General Surgery, JNMC, DMIMS, Sawangi, Wardha, Maharashtra
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jdmimsu.jdmimsu_56_20

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Background: Many conditions affect the salivary glands. Acute sialadenitis is infectious or inflammatory disorders of the salivary glands. The exact frequency of submandibular sialadenitis is unclear. The acute conditions more typically involve the parotid and submandibular glands. Submandibular gland and its duct calculi are known entities, but their typical presentation is rare. Material and methods: During an acute inflammatory process, there is swelling of the affected gland, overlying pain, gland tenderness, fever, and on occasion, difficulty in opening the mouth. Results and Conclusion: Here, we present a case of 23-year-old male with unilateral left submandibular gland and Wharton's duct calculi expelling calculus and pus and impacted calculus in the proximal duct and gland from its opening under surface of the tongue and left submandibular sialoadenectomy done through the submandibular external approach.

Keywords: Sialoadenectomy, submandibular gland, Wharton's duct calculi

How to cite this article:
Chandak SR, Chandu R. Submandibular gland wharton's duct phlegomous calculus sialoadenitis: A rare case report. J Datta Meghe Inst Med Sci Univ 2020;15:292-4

How to cite this URL:
Chandak SR, Chandu R. Submandibular gland wharton's duct phlegomous calculus sialoadenitis: A rare case report. J Datta Meghe Inst Med Sci Univ [serial online] 2020 [cited 2021 Jan 20];15:292-4. Available from: http://www.journaldmims.com/text.asp?2020/15/2/292/304266

  Introduction Top

Many conditions affect the salivary glands. They affect all of the salivary tissues, but all conditions affect the parotid and the submandibular glands preferentially because of their size and location. Adults and children are commonly affected. Sialadenitis of the submandibular gland is a relatively commonly encountered yet infrequently discussed topic. Causes range from a simple infection to autoimmune etiologies although not as frequent as sialadenitis of the parotid gland.

Acute sialadenitis is infectious or inflammatory disorders of the salivary glands. The exact frequency of submandibular sialadenitis is unclear. The incidence of acute suppurative parotitis has been reported at 0.01%–0.02% of all hospital admissions. The submandibular gland is suggested to account for approximately 10% of all cases of sialadenitis of the major salivary glands. No race, age, and sex predilection per se exists. Sialadenitis as a whole tends to occur in older, debilitated, or dehydrated patient.

The acute conditions more typically involve the parotid and submandibular glands. During an acute inflammatory process, there is swelling of the affected gland, overlying pain, gland tenderness, fever, and, on occasion, difficulty in opening the mouth. Often, the pain is intensified with eating in that food ingestion stimulates saliva flow, which will typically cause the gland to swell and thus exacerbate the preexisting symptoms. Acute inflammatory processes largely fall into bacterial, viral, and autoimmune states. In chronic gland disorder, the symptoms are similar although much less intense. In the inflammatory conditions, the gland is not so much a target of bacterial or viral processes but is inflamed by antibodies directed against the salivary gland tissues.[1],[2]

  Etiology Top

Although definite etiology is still ambiguous, sialoliths are thought to occur as a result of deposition of mineral salts around an initial nidus consisting of salivary mucin, bacteria, or desquamated epithelial cells. They form as a result of mineralization of debris that has accumulated in the lumen of the duct. This debris includes bacterial colonies, exfoliated ductal epithelial cells, mucus plugs, foreign bodies, or other cellular debris. Factors like stagnation of salivary flow, dehydration, and change in salivary pH associated with oropharyngeal sepsis, impaired crystalloid solubility, high alkalinity, and increased calcium content, and physical trauma to salivary duct or gland may predispose to calculus formation. The definite etiology of our cases still remains unknown. Two stages of sialolith formation can be found in the literature: (i) central core formation and (ii) layered periphery formation. First, 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 bodies and a nidus of mucous, respectively. Boynton and Lieblich in 2014 reported an unusual case in which the facial hair of the patient got entrapped in the Wharton's duct and acted as a nidus for the formation of a sialolith.[5] Another theory has proposed that an unknown metabolic phenomenon can lead to precipitation of salivary calcium and phosphate ions by increasing the salivary bicarbonate content, which in turn alters the calcium phosphate solubility. A retrograde theory suggested that any substance or bacteria of the oral cavity that had migrated into the salivary ducts can act as a nidus for further calcification.[4] Marchal et al. further suggested that easier retrograde migration of materials can occur due to variation in the sphincter-like mechanism in the first 3 cm of the Wharton's duct. Recently, Sherman and McGurk6 the incidence of salivary calculi is not significantly associated with water hardness. Longstanding obstruction by a sialolith may severely damage the acini of the gland, resulting in a permanent decrease or even absence of salivary secretion. This reduced or absent salivary secretion may give rise to recurrent infections, which can lead to atrophy of the gland with loss of secretory function and ultimately fibrosis. Submandibular sialolithiasis is common when compared to other salivary glands due to thick mucus discharge and nondependent drainage and obliquity of the gland.

  Case Report Top

A 23-year-old man visited us with a chief complaint of swelling and pain in the oral cavity with left submandibular swelling which was tender and soft. Extraoral examination revealed no facial asymmetry but firm and tender left submandibular lymph nodes. Intraoral examination showed mild elevation of the tongue with swelling, and the stone was visible with pus discharge at the left Wharton's duct opening. A diffuse swelling was palpable on the floor of the mouth on the left side of the lingual frenulum. On the basis of the above findings, we came to a provisional diagnosis of left submandibular sialoadenitis. X-ray confirmed the diagnosis of calculi in the oral cavity. Left sialoadenectomy was done through an external approach. The left gland and duct with calculi were extracted. Duct probing was done from the proximal end to the distal end, i.e., from the submandibular gland side to the oral opening. There were pus and calculus which extracted from the oral cavity. Oral opening was dilated, duct transacted near oral opening and extracted. Postoperative healing was smooth and uneventful [Figure 1], [Figure 2], [Figure 3].
Figure 1: Preoperative image

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Figure 2: Intraoperative image

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Figure 3: Postoperative image

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Ethical Clearance

Ref no: Date- 22/2/2018. Ref No-DMIMS(DU)/IEC/2018-19/7215.

  Discussion Top

A variety of factors affect the susceptibility of the different salivary glands to bacterial infection, but among the most important are their rates of salivary flow, the composition of their saliva, and variations in or damage to their duct systems.[4] Raad et al. have drawn attention to and reviewed reports of this entity, of which there were 12 cases among their 29 patients with acute bacterial sialadenitis. Unlike suppurative parotitis, sialolithiasis was an important predisposing factor, but xerostomia was also common.[4]

Clinically, acute submandibular sialoadenitis differs from parotitis mainly in the site of the swelling and discharge of pus from the Wharton's duct. A wide variety of bacteria have been incriminated, but Staphylococcus aureus has been the most frequently reported isolate.[5] The other isolated organisms have included streptococci, Pseudomonas aeruginosa,  Escherichia More Details coli, and  Moraxella More Details catarrhalis.

The diagnosis of submandibular sialoadenitis can be made on clinical grounds, and submandibular sialoadenitis takes several forms. The diagnostic workup of any submandibular enlargement begins with a thorough history. However, systemic manifestations may be minimal.

Examination with ultrasound is noninvasive, cheap, and useful for diagnosis, differential diagnosis and excluding the other predisposing factors such as anatomical abnormalities of Wharton's duct, mechanical salivary duct obstruction secondary to a sialolith, and infection related to a submandibular gland neoplasm; however, in our case, the patient had a bacterial infection of the submandibular salivary gland secondary to sialolith.[5]

The administration of antimicrobial therapy is an essential part of the management of patients with suppurative sialoadenitis. Most cases respond to antimicrobial therapy; however, sometimes, abscess formation requires surgical drainage.[6]

In the acute viral and the vast majority of acute bacterial infections, the gland returns to an asymptomatic state. Certain individuals with chronic bacterial infections not responding to appropriate conservative and antibiotic measures may require either radiation or removal of the affected gland to control its symptoms. The prognosis of acute sialoadenitis is very good. Most cases are easily treated with conservative medical management, and admission is the exception, not the rule. Acute symptoms resolve within 1 week; however, edema in the area may last for several weeks.[3]

  Conclusion Top

Patients with any form of sialoadenitis should be educated as to the value of hydration and excellent oral hygiene. This lessens the severity of the attacks and prevents dental complications. Patients with sialoadenosis should be educated regarding the mechanism of their underlying pathology and methods of maintaining control over them.[7],[8],[9],[10],[11],[12]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship


Conflicts of interest

There are no conflicts of interest.

  References Top

Bradley PJ. Pathology and treatment of salivary gland conditions. Surgery 2006;24:304-11.  Back to cited text no. 1
Isacsson G, Isberg A, Haverling M, Lundquist PG. Salivary calculi and chronic sialoadenitis of the submandibular gland: A radiographic and histologic study. Oral Surg Oral Med Oral Pathol 1984;58:622-7.  Back to cited text no. 2
Loury MC. Salivary gland disorder. Adv Otolaryngol 2006.  Back to cited text no. 3
Raad II, Sabbagh MF, Caranasos GJ. Acute bacterial sialadenitis: A study of 29 cases and review. Rev Infect Dis 1990;12:591-601.  Back to cited text no. 4
Cawson RA, Gleeson MJ, Eveson JW. Sialadenitis. In: The Pathology and Surgery of the Salivary Glands. 1st ed., Ch. 4. 1997. p. 1-34.  Back to cited text no. 5
Tapisiz A, Belet N, Ciftçi E, Fitöz S, Ince E, Doğru U. Neonatal suppurative submandibular sialadenitis. Turk J Pediatr 2009;51:180-2.  Back to cited text no. 6
Silvers AR, Som PM. Salivary glands. Radiol Clin North Am 1998;36:941-66, vi.  Back to cited text no. 7
Bradley PJ. Benign salivary gland disease. Hosp Med 2001;62:392-5.  Back to cited text no. 8
Panchbhai A, Bhowate R. MRI Evaluation of Involvement of Parotid and Submandibular Glands by Tongue Squamous Cell Carcinoma. Oral Oncol 2020;102. Available from: https://doi.org/10.1016/j.oraloncology.2019.104557. [Last accessed on 2020 Jan 28].  Back to cited text no. 9
Heramot SO, Singh NS, Sarkar R, Singh TH. Association between gall bladder diameter and calculus in the cystic duct in gall stone diseases and its importance in the current surgical practice- a cross-sectional study in jnims, porompat, manipur. J Evol Med Dent Sci 2018;7:856-9. Available from: https://doi.org/10.14260/jemds/2018/195. [Last accessed on 2020 Jan 28].  Back to cited text no. 10
Bhushan PM, Surana S, Bhola N, Oswal S, Dakshinkar P. Multiple Recurrent Simultaneous Salivary Calculi. J Clin Diagn Res 2018;12:ZJ1-2. Available from: https://doi.org/10.7860/JCDR/2018/34546.11462. [Last accessed on 2020 Jan 28].  Back to cited text no. 11
Harish AH, Sonone A, Porwar R, Lohe V, Dangore S, Meshram M. Evaluation of Oral Microbial Flora in Saliva of Patients of Oral Submucous Fibrosis. J Evol Med Dent Sci 2020;9:409-12. Available from: https://doi.org/10.14260/jemds/2020/93. [Last accessed on 2020 Jan 28].  Back to cited text no. 12


  [Figure 1], [Figure 2], [Figure 3]


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