Int J Clin Exp Med 2017;10(2):3868-3871

www.ijcem.com /ISSN:1940-5901/IJCEM0044758

Case Report
Submandibular gland mucocele:
a case report and literature review
Tzu-Hang Chi1,2, Rong-Feng Chen1, Chien-Han Yuan1, Shang-Tao Chien3

Departments of 1Otolaryngology, 3Pathology, Kaohsiung Armed Forces General Hospital, Kaohsiung, Taiwan,
Republic of China; 2Department of Otolaryngology, Taoyuan Armed Forces General Hospital, Taoyuan, Taiwan,
Republic of China
Received November 21, 2016; Accepted December 25, 2016; Epub February 15, 2017; Published February 28,
2017

Abstract: Mucoceles occur most commonly in the oral mucosa and minor salivary glands. The lower lip is the most
common site for the development of a mucocele, and occurrence in one of the major salivary glands is rare. A review
of literature written in English revealed 13 cases of submandibular gland mucocele. The etiology of mucoceles is
thought to involve partial obstruction or disruption of the salivary gland duct. Mucoceles are categorized as reten-
tion or extravasation mucoceles based on their histopathological appearance. Surgical removal is the preferable
treatment. Excision of a mucocele in continuity with the submandibular gland reduces the recurrence rate. A rare
case of a submandibular gland mucocele is reported.

Keywords: Mucocele, salivary gland, submandibular gland, retention, extravasation

Introduction right submandibular triangle of the neck (Fig-

ure 1). The neck mass was smooth and mo-
Mucoceles are common benign cystic lesions bile with no tenderness, and there was no ad-
that occur in the minor salivary glands. About ditional lymphadenopathy or palpable mass
60 to 70% of mucoceles occur in the lower lip, of the neck. The nose, ears, oral cavity, phar-
6 to 15% occur on the floor of the mouth, and ynx, and larynx were within normal limits in a
only a few cases have been reported to occur in series of examinations. The laboratory findings
the submandibular gland [1]. Factors such as showed a white blood cell count of 10.1×103/
trauma or obstruction of the salivary gland μL, neutrophil count of 75.5%, lymphocyte
ducts are thought to contribute to the develop- count of 14.0%, and C-reactive protein level of
ment of mucoceles [2]. Total excision of muco- 0.9 mg/dL. Computed tomography of the na-
celes reduces the recurrence rate. A case of a sopharynx to the neck with contrast showed
24-year-old man with a submandibular gland one 4.2×2.7-centimeter cystic-like, hypo-dense,
mucocele presenting as right-sided lateral neck and peripherally enhanced lesion involving the
swelling is reported. submandibular gland in the right submandi-
bular space (Figure 2). Fine needle aspiration
Case report
cytology of the cyst was performed, and the
The patient was a 24-year-old man with Down yellowish, serous, aspirated fluid was negative
syndrome. He was sent to the otolaryngology for malignancy. After physical, laboratory, radio-
outpatient department for treatment, and pain- logical, and cytological examinations, as well as
less swelling of the right lateral neck for one consideration of the patient’s history, a bran-
week was reported. There was no sore throat, chial cleft cyst was the tentative diagnosis.
toothache, dysphagia, otalgia, weight loss, or
fever. There was no family history of disease, Excisional surgery under general analgesia was
and there was no history of habitual cigarette performed. The incision was made about two
smoking or alcohol consumption. fingerbreadths below the jawline. The cystic-like
tumor closely adhered to the submandibular
The initial physical examination showed one gland, and no tract connecting the tumor to the
palpable 5.0×4.0-centimeter mass over the pharynx or hyoid bone could be identified. The
 Mucocele: a case report and literature review

Figure 1. One palpable 5.0×4.0 centimeter mass Figure 3. Macroscopic appearance of the specimen
over the right submandibular triangle of neck. shows the excised cyst (white arrow) and subman-
dibular gland (black arrow).

Figure 2. Computed tomography of the nasopharynx Figure 4. Histological examination of the cyst reveals
to the neck shows one hypo-dense lesion of about mucinous material covered by granulation tissue (he-
1.2×2.7 centimeters with peripheral enhancement matoxylin and eosin stain, ×40).
involving the submandibular gland in the right sub-
mandibular space.
glands. The occurrence of mucoceles in major
tumor was completely excised along with the salivary glands is uncommon, and mucoceles
submandibular gland (Figure 3). The operative in the submandibular gland are rare. Searching
procedure went smoothly with no immediate published literature written in English, we found
complications. On gross examination of the 13 cases of submandibular gland mucocele
specimen, the cyst had a thick wall with yellow- were reported since the first case published
ish serous fluid contents. Microscopic examina- by Surkin et al at 1985 (Table 1). This literature
tion revealed mucus covered with granulation review reveals uneven distribution between
tissue without a lining epithelium (Figure 4). genders that the incidence was higher in
males compared to females (11 males and 2
The patient then received timely follow-up in females). Moreover, submandibular gland mu-
the otolaryngology outpatient department for cocele occurred in patients ranged from 16
six months. The patient had no evidence of months to 39 years of age (mean age 20.7±
recurrence or additional problems. 11.4 years). The lesion was observed on the
right side in 8 patients, on the left side for 4
Discussion patients and 1 patient had bilateral lesions.

Mucoceles are benign, cystic-like lesions that Mucoceles are categorized as retention or
can present at various sites of the oral mucosa extravasation mucoceles based on their histo-
and most commonly develop in minor salivary pathological appearance. A retention mucocele

3869 Int J Clin Exp Med 2017;10(2):3868-3871

 Mucocele: a case report and literature review

Table 1. Demographic characteristics of 13 published cases of submandibular gland mucocele

Duration of
No. Auther Gender Age Side Treatment* Follow-up
symptoms
1 Anastassov et al [1] Male 18 years 3 months Left Excision with SMG, SLG 16 months
2 Anastassov et al [1] Male 30 years 10 years Right Excision with SMG, SLG 12 months
3 Surkin et al [4] Male 39 years 6 weeks Right Excision with SMG No evidence of disease
4 Choi et al [6] Male 16 months Several days Bilateral Excision with SMG, SLG 24 months
5 Van der Goten et al [7] Male 7 years 3 weeks Right Excision with SMG Not available
6 Van der Goten et al [7] Female 18 years 8 weeks Left Excision with SMG, SLG Not available
7 Boneu-Bonet et al [9] Male 25 years 6 months Right Excision with SMG No evidence of disease
8 Hze-Khoong et al [10] Male 21 years 1 week Right Excision with SMG No evidence of disease
9 Okumura et al [11] Male 7 years 2 years Right Excision with SMG, SLG No evidence of disease
10 Ozturk et al [12] Female 11 years 6 months Right Excision with SMG, SLG 8 months
11 Ozturk et al [12] Male 38 years 8 years Left Excision with SMG, SLG 34 months
12 Stranc et al [13] Male 29 years 1 month Right Excision Recurrence
13 Cholankeril et al [14] Male 25 years 6 months Left Excision with SMG 6 months
*
SMG: submandibular gland; SLG: sublingual gland.

is a mucus-containing cyst with an epithelial Surgical intervention is the preferable treat-

wall covering, while an extravasation mucocele ment for a submandibular gland mucocele.
is also a mucus-containing cyst but is covered Currently, the surgical methods include marsu-
by granulation tissue without an epithelium [3]. pialization, cystectomy, injection of sclerosing
agents, incision and drainage, excision of the
The mechanism of development is thought to mucocele in continuity with the sublingual
be partial obstruction of the salivary gland duct gland, and/or the submandibular gland [1].
in the case of retention mucoceles or disrup- Compared to other surgical methods that main-
tion of the salivary gland duct in the case of tain the submandibular gland, excision of a
extravasation mucoceles [4]. Retention muco- mucocele in continuity with the submandibu-
celes are more common at advanced age, while lar gland has the advantage of a lower recur-
extravasation mucoceles are commonly found rence rate. If the submandibular gland muco-
among young people [5]. cele affecting or closely next to the sublingual
gland, it is recommended to excise the sublin-
The clinical symptoms of submandibular gland
gual gland as well [9].
mucoceles include a painless, slow-growing
mass in the submandibular region. They usually In summary, even though mucoceles are rare
present as a soft, well-circumscribed, mobile and benign lesions that occur in the subman-
masses [6]. The differential diagnosis of a sub- dibular gland, they should always be included
mandibular cystic-like mass includes congeni- in the differential diagnosis of submandibu-
tal lesions such as branchial cleft cysts, der- lar cystic-like masses. The clinical symptoms
moid cysts, cystic hygroma, and thyroglossal include a painless, slow-growing mass in the
duct cysts, as well as acquired lesions such as submandibular region. Surgical intervention
ranula, abscesses, and cystic degeneration of with excision of the mucocele in continuity
neoplasms [7]. with the submandibular gland is the preferr-
ed treatment.
Computed tomography or magnetic resonance
imaging examinations are important for deter- Disclosure of conflict of interest
mining the accurate location of the lesion and
for distinguishing between benign and malig- None.
nant lesions. In addition, fine needle aspiration
cytology is useful for diagnosing cervical mass- Address correspondence to: Dr. Shang-Tao Chien,
es. The sensitivity rate for such masses is Department of Pathology, Kaohsiung Armed Forces
about 90 to 100%, but the false negative rate General Hospital, 2, Chung Cheng 1st Road, Kao-
for cervical cysts is as high as 50% [8]. hsiung 802, Taiwan, Republic of China. Tel: +886-7-

3870 Int J Clin Exp Med 2017;10(2):3868-3871

 Mucocele: a case report and literature review

7494965; Fax: +886-7-7495175; E-mail: tzuhang- [8] Gourin CG, Johnson JT. Incidence of unsus-
chi@gmail.com pected metastases in lateral cervical cysts.
Laryngoscope 2000; 110: 1637-1641.
References [9] Boneu-Bonet F, Vidal-Homs E, Maizcurrana-
Tornil A, González-Lagunas J. Submaxillary
[1] Anastassow GE, Haiavy J, Solodnik P, Lee H, gland mucocele: presentation of a case. Med
Lumerman H. Submandibular gland mucocele: Oral Patol Oral Cir Bucal 2005; 10: 180-184.
diagnosis and management. Oral Surg Oral [10] Hze-Khoong EP, Xu L, Shen S, Yin X, Wang L,
Med Oral Pathol Oral Radiol Endod 2000; 89: Zhang C. Submandibular gland mucocele as-
159-163. sociated with a mixed ranula. Oral Surg Oral
[2] Yamasoba T, Tayama N, Syoji M, Fukuta M. Med Oral Pathol Oral Radiol 2012; 113: e6-e9.
Clinicostatistical study of lower lip mucoceles. [11] Okumura K, Inui M, Nakase M, Nakamura S,
Head Neck 1990; 12: 316-320. Hiramoto K, Tagawa T. A case of submandibu-
[3] Baurmash HD. Mucoceles and ranulas. J Oral lar gland mucocel. J Clin Pediatr Dent 2007;
Maxillofac Surg 2003; 61: 369-378. 31: 207-209.
[4] Surkin M, Remsen K, Lawson W, Som P, Biller [12] Ozturk K, Yaman H, Arbag H, Koroglu D, Toy H.
HF. A mucocele of the submandibular gland. Submandibular gland mucocele: report of two
Arch Otolaryngol 1985; 111: 623-625. cases. Oral Surg Oral Med Oral Pathol Oral
[5] Nico MM, Park JH, Lourenco SV. Mucocele in Radiol Endod 2005; 100: 732-735.
pediatric patients: analysis of 36 children. [13] Stranc MF, Skoracki R. A complication of sub-
Pediatr Dermatol 2008; 25: 308-311. mandibular intubation in a panfacial fracture
[6] Choi HJ, Kim SG, Kim JD, Kim JH, Kim JH, Kim patient. J Craniomaxillofac Surg 2001; 29:
SM. A case of bilateral submandibular gland 174-176.
mucoceles in a 16-month-old child. Korean J [14] Cholankeril JV, Scioscia PA. Post-traumatic si-
Pediatr 2012; 55: 215-218. aloceles and mucoceles of the salivary glands.
[7] Van der Goten A, Hermans R, Smet MH, Baert Clin Imaging 1993; 17: 41-45.
AL. Submandibular gland mucocele of the ex-
travasation type. Report of two cases. Pediatr
Radiol 1995; 25: 366-368.

3871 Int J Clin Exp Med 2017;10(2):3868-3871