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CASE REPORT |
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| Year : 2023 | Volume
: 28
| Issue : 3 | Page : 253-255 |
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Mucocele arising from the submandibular gland presenting as neck swelling: A rare case report
Probal Chatterji1, Rajul Rastogi2, Akshay Jain1, Somya Choudhary1, Nitanshi Jain2
1 Department of Otorhinolaryngology and Head Neck Surgery, Teerthanker Mahaveer Medical College and Research Center, Moradabad, Uttar Pradesh, India
2 Department of Radiodiagnosis, Teerthanker Mahaveer Medical College and Research Center, Moradabad, Uttar Pradesh, India
| Date of Submission | 09-Nov-2022 |
| Date of Decision | 23-Dec-2022 |
| Date of Acceptance | 02-Jan-2023 |
| Date of Web Publication | 21-Mar-2023 |
Correspondence Address:
Rajul Rastogi
Department of Radiodiagnosis, Teerthanker Mahaveer Medical College and Research Center, Delhi Road, Moradabad - 244 001, Uttar Pradesh
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/jiaps.jiaps_163_22
 Abstract | | |
Mucoceles rarely involve major salivary glands. Very few cases involving the submandibular gland have been reported to date. A young male child presented with diffuse, soft, and painless swelling of the left submandibular region. Investigations suggested mucocele of the submandibular salivary gland. The mucocele was excised along with the left submandibular gland. Recovery was uneventful.
Keywords: Mucocele, neck swelling, submandibular gland
How to cite this article:
Chatterji P, Rastogi R, Jain A, Choudhary S, Jain N. Mucocele arising from the submandibular gland presenting as neck swelling: A rare case report. J Indian Assoc Pediatr Surg 2023;28:253-5 |
How to cite this URL:
Chatterji P, Rastogi R, Jain A, Choudhary S, Jain N. Mucocele arising from the submandibular gland presenting as neck swelling: A rare case report. J Indian Assoc Pediatr Surg [serial online] 2023 [cited 2023 Jun 2];28:253-5. Available from: https://www.jiaps.com/text.asp?2023/28/3/253/372254 |
| Introduction | |  |
Mucoceles are benign cystic lesions that arise due to extravasation or retention of mucus, usually in relation to the minor salivary glands. About 60%–70% of them involve the lower lip.[1] Among the major salivary glands, most often the sublingual salivary gland is associated with the development of ranula. However, mucocele arising in relation to the submandibular salivary gland is extremely rare. The first such case was reported in the literature in 1985 and only a handful of such cases have been reported to date.[2]
Mucoceles present as a slow-growing swelling with nonspecific signs and symptoms. Etiology is often unclear. Clinically, it is mostly confused with plunging ranula, but proper radiological imaging can differentiate between the two.[3] This condition is primarily managed by surgery and final confirmatory diagnosis is obtained by histological examination of excised specimen.[2]
We report an unusual case of a mucocele of the submandibular salivary gland which was managed by surgery. The patient had an uneventful recovery.
| Case Report | | |
A 5-year-old male child presented with a complaint of gradual swelling below the left jaw for 1-year duration. According to his mother, a similar swelling had occurred 2 years ago at the same location but regressed spontaneously after taking antibiotics from a local practitioner. There was no other complaint such as local pain, tenderness, and fever. There was no difficulty in chewing of food and no change in the size of the swelling during meals.
On examination, there was facial asymmetry with a diffuse swelling (3 × 2 inches) just below the jaw in the left submandibular region [Figure 1]a. The skin over the swelling was normal. On palpation, the swelling was soft, cystic, nontender, nonpulsatile, and compressible. The margins of the swelling were indistinct. Oral cavity examination revealed a slight fullness on the floor of the mouth on the left side. The rest of the examination was unremarkable. A clinical diagnosis of plunging ranula was made. | Figure 1: (a) Patient presenting with diffuse swelling of the left submandibular region and (b) intraoperative photo showing mucocele arising from the left submandibular gland
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High-resolution ultrasonography (USG) examination revealed a cystic lesion without obvious internal septations or debris or nodules located in close apposition to the left submandibular gland, but the exact relationship of the lesion with the gland could not be discretely established. Therefore, contrast-enhanced magnetic resonance imaging (CEMRI) of the neck was advised. A fine-needle aspiration cytology (FNAC) test under USG guidance yielded 4–5 ml of straw-colored fluid on aspiration. Cytology showed the presence of nonspecific inflammatory cells in the background. CEMRI neck revealed fluid-intensity lesion with peripheral rim enhancement epicentered within the left submandibular gland without obvious internal septations or calcification. The lesion measured approximately 50*35*25mm corresponding approximately to 20–25 ml in volume. The right submandibular and both sublingual glands were normally visualized [Figure 2]. | Figure 2: MRI images (a-e) showing mucocele (asterisk) arising from the anterior part of the left submandibular gland (arrows and arrowheads) with normal-appearing right submandibular gland and sublingual glands. (a) Sagittal T2WI, (b) axial T1WI, (c) axial DWI, (d) fat-suppressed, contrast-enhanced, axial T1WI, (e) fat-suppressed, contrast-enhanced, sagittal T1WI. DWI: Diffusion-weighted imaging, T2WI: T2-weighted image, T1WI: T1-weighted image
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Thus, a diagnosis of mucocele of the left submandibular gland was made and the patient was counseled for surgery. A submandibular incision was given about 1 inch below and parallel to the margin of the left mandible, and the mass was adequately exposed [Figure 1]b. The whole mass along with the ipsilateral submandibular salivary gland was excised as a single specimen and sent for histopathological examination. A negative suction drain was inserted, and the wound was closed in layers. Histopathological report confirmed the diagnosis. The patient had an uneventful recovery, and no recurrence of the swelling has been seen up to 6 months of follow-up.
| Discussion | | |
As already mentioned, mucocele arising from the submandibular salivary gland is extremely rare. The first such case was described by Surkin et al.[4] in 1985. According to Salissou et al., who published a case report of such a condition in a 9-year-old female child in 2021, only 13 such cases have been reported in the literature till 2017.[2] The age of presentation varies from childhood to middle age and most cases have unilateral involvement. Choi et al. have, however, reported a case of bilateral submandibular gland mucocele in a 16-month-old child.[1] There appears to be a strong male preponderance in the few cases reported to date.[2]
The etiology of mucocele is not clear. Studies suggest some form of trauma leading to obstruction or rupture of the duct of the involved salivary gland leading to extravasation and/or retention of secretions.[2]
The swelling usually presents as a diffuse mass in the submandibular region and upper neck with or without swelling in the floor of the mouth.[1],[2],[5] Clinical presentation is mostly nonspecific and as the swelling enlarges in size, vague complaints such as change in the quality of speech along with difficulty in chewing and swallowing of food may occur. Most authors agree that considering the rarity of this condition and nonspecific nature of the presentation, it may be difficult to suspect this condition initially and it is most likely to be confused with a plunging ranula.[3] Radiological investigation such as computed tomography (CT) scan or MRI can help in differentiating between the two conditions. It has been suggested that the specific “tail sign” on CT scan due to the longitudinal extension is characteristic of plunging ranula and is typically absent in case of mucocele arising from the submandibular salivary gland. USG may also be a useful investigation to demonstrate dehiscence of the mylohyoid muscle in case of the former.[3]
Other important differential diagnoses that must be kept in mind include congenital malformations or various acquired conditions such as those with infective, inflammatory, and neoplastic etiology that may affect this anatomical region.[5] FNAC will usually be conclusive in most cases along with radiological evaluation.
Various techniques have been employed to treat this condition including simple aspiration and injection of sclerosants with a high failure rate. The best option is to remove the mass along with the involved submandibular gland by an external approach.[5] A minimally invasive technique using intraoral endoscopic approach for the removal of the submandibular gland mucocele has also been employed in one case by a different author as reported by Salissou et al.[2] The advantage of this approach is its minimally invasive nature, absence of external scar, and reduced risk of damage to the branches of the facial nerve.
| Conclusion | | |
Mucocele of the submandibular gland is extremely rare but mostly presents as a diffuse swelling of the submandibular region. Most cases are seen in males, unilateral in presentation, and etiology is unknown. It can be clinically confused with a plunging ranula and certain other conditions of this anatomical region. FNAC and radiological imaging are useful to identify it. The treatment of choice includes the removal of the mucocele along with the involved submandibular gland to prevent the chance of recurrence.
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
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Choi HJ, Kim SG, Kim JD, Kim JH, Kim JH, Kim SM. A case of bilateral submandibular gland mucoceles in a 16-month-old child. Korean J Pediatr 2012;55:215-8.  |
| 2. | Salissou I, Savadogo S, Aloua R, Belem O, Opoko U, Ayoub S, et al. Mucocele of the submandibular gland extended to the oral floor in a child. J Pediatr Surg Case Rep 2021;72:101952. |
| 3. | Anastassov GE, Haiavy J, Solodnik P, Lee H, Lumerman H. Submandibular gland mucocele: Diagnosis and management. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;89:159-63. |
| 4. | Surkin M, Remsen K, Lawson W, Som P, Biller HF. A mucocele of the submandibular gland. Arch Otolaryngol 1985;111:623-5. |
| 5. | Felstead A, Patel P, Revington P. Submandibular gland mucocele presenting as a lateral neck swelling. J Surg Case Rep 2012;2012:8. |
[Figure 1], [Figure 2]
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