Journal of Otology & RhinologyISSN: 2324-8785

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Case Report, J Otol Rhinol Vol: 5 Issue: 1

A Case of Large Pseudocyst Originating from the Submandibular Gland Presented with a Systematic Review

Saida K1, Yokoyama J2,5*, Fukumura Y3, Katsuta H4 and Ishibasi K1
1Department of Oral maxillofacial Surgery, Moriyama Memorial Hospital, Tokyo,Japan
2Department of Head and Neck Cancer Center, Moriyama Memorial Hospital,Tokyo, Japan
3Department of Human Pathology School of Medicine, Juntendo University, Tokyo,Japan
4Division of Oral Oncology, Department of Oral and Maxillofacial Surgery, School of Dentistry, Showa University, Tokyo, Japan
5Department of Otorhinolaryngology-Head and Neck Surgery, Kyorin University School of Medicine, Tokyo, Japan
Corresponding author :Yokoyama J
MD, DDS, PhD, Department of Head and Neck Cancer Center, Moriyama Memorial Hospital, 7-12-7 Nishikasai, Edogawaku, Tokyo, Japan 134-0088
Tel: +81-3-5679-1211; Fax: +81-5679-1212
E-Mail: [email protected]
Received: January 18, 2016 Accepted: February 18, 2016 Published: February 23, 2016
Citation: Saida K, Yokoyama J, Fukumura Y, Katsuta H Ishibasi K (2016) A Case of Large Pseudocyst Originating from the Submandibular Gland Presented with a Systematic Review. J Otol Rhinol 5:1.. doi:10.4172/2324-8785.1000267

Abstract

The majority of mucous cysts originate from the sublingual gland. Mucoceles originating from the submandibular glands are extremely rare. We report the case of a 24-year-old girl with submandibular gland mucocele. Magnetic resonance imaging (MRI) revealed cystic lesions in close proximity to the submandibular gland without tail sign. The cyst with submandibular gland was successfully removed through a cervical approach without complications. The pathologic and intraoperative examination confirmed the pseudocyst originating from the submandibular gland. The patient has no evidence of recurrence. We review previous cases of mucoceles originating from the submandibular gland.

Keywords: Pseudocyst; Submandibular gland; Mucocele; Ranula

Keywords

Pseudocyst; Submandibular gland; Mucocele; Ranula

List of Abbreviation

MRI: Magnetic resonance imaging, CT: Computed tomography

Introduction

Among mucoceles originating from major salivary glands, most cases develop from a sublingual gland referred to as “ranula”. The ranula can be further divided into sublingual type and submandibular type termed plunging ranula.While mucoceles originating from the submandibular are extremely rare, the differential diagnosis of submandibular gland mucocele and plunging ranula is significantly important for treatment1. In the plunging ranula, what is typically referred to as the “tail sign” is recognized as a key characteristic of the sublingual mucocele with MRI.

Case Presentation

A 24-year-old woman was diagnosed with unilateral cervical ranula six years ago. Due to the fact that the lesion had increased slowly and had been repeatedly infected, the patient was introduced to our hospital for radical treatment.
The patient had no traumatic history or surgical history regarding the oral and left submandibular region. Palpation of the neck revealed that the submandibular gland was hard, with two soft swollen and non-tender masses (Figure 1).
Figure 1: Cervical findings: Preoperative photograph shows left submandibular swelling.
There were no abnormal findings on the floor of the mouth. MRI scan revealed large, smooth-bordered cystic lesions with T2-weighted image in close proximity to the left submandibular gland and extending through the submental space to left parapharyngeal space without “tail sign”(Figure 2). There were no abnormalities detected in the blood examination.
Figure 2: Preoperative MRI: a) T2-weighted MRI (axial section), b) T1-weighted MRI(axial section), c) T2-weighted MRI(axial section) d) T2-weighted MRI(coronal section) There is no tail sign in preoperative MRI(a-d).
Under general anesthesia, the cysts were surgically excised with the left submandibular gland via an external cervical incision. Intraoperative findings showed that the cysts did not originate from the sublingual gland (Figure 3).
Figure 3: Operative findings: a) Intraoperative findings showed that the cysts did not originate from the sublingual gland. 1: pseudocyst, 2: submandibular gland, b) Resected specimen. The cysts excised with the left submandibular gland demonstrated originating from the submandibular gland. 1: pseudocyst, 2: submandibular gland.
The marginal mandibular branch of the facial nerve, the lingual nerve and the hypoglossal nerve were preserved. Histopathological examination of the specimen showed a non-epithelial lined cyst filled with foam cell, erythrocyte and neutrophil. There was atrophy of the glandular lobe and dilatation of the duct (Figure 4).
Figure 4: Pathological findings: a) Low power magnification (Hematoxylin -eosin stain ×5). The enlarged cyst wall has no epithelial lining and replaced by granulation tissue close to submandibular gland. b) High power magnification (×40). The non-epithelial lined psuedocyst was filled with foam cell, neutrophil, and mucin-laden Macrophage.
The enlarged pseudocyst wall contained mucin-laden Macrophage and the final pathological diagnosis was mucoceles originating from the submandibular gland. The patient experienced no postoperative complications and no evidence of recurrence.

Discussion

The majority of mucoceles arising from major salivary glands develop from the sublingual gland, which is known as the ranula. A specific type of ranula causing neck swelling is called plunging ranula. They extend inferiorly beyond the mylohyoid muscle into the submandibular space and adjacent structures. In such cases, it is difficult to distinguish whether psuedocysts originate from either sublingual or submandibular glands. It is critical to identify the origin of mucocele in order to determine effective treatment.
There have only been 13 cases of submandibular gland mucocele reported in English literature [1-10]. We show a list of these cases originating from the submandibular gland including oue case (Table 1).
Table 1: A list of literatures originating from submandibular gland.
The table shows 10 males and 4 females, which is a slight preponderance of 2.5:1 (M:F). The average age was 23 years. There was no history of trauma, surgery or infection on the ipsilateral side of patients necks in all cases.
In regard to modalities of diagnostic imaging, 8 cases were diagnosed by CT (Computed tomography) and 5 cases were diagnosed by MRI. Both CT and MRI were used for 3 cases. When the lack of “tail sign” is shown, almost all authors clinically diagnosed mucocele as originating from the submandibular gland. Fine needle aspiration was performed in 4 cases.
In regard to treatments, it has been proposed that the optimal treatment of mucocele is to resect the origin of salivary glands together. No recurrences were observed.
Other treatments such as aspiration, fenestration and injection of sclerosing agents have been reported. However, recurrences have sometimes been reported with these procedures. Non-surgical treatments are sometimes performed in young females and children who are concerned about postoperative cervical scaring.
In order to avoid the postoperative cervical scaring, some surgeons [11,12] reported excision of the submandibular gland via the transoral approach with endoscopic assistance. The indication of this surgical procedure is submandibular gland diseases with the exclusion of malignancy. Assisted by endoscope, the risk of damage to the lingual nerve, hypoglossal nerve, and marginal mandibular branch of facial nerve is lower than the transcervical approach. However, this procedure has some disadvantages; First, an assistant is needed to hold the endoscope during surgery. Second, all surgeons must understand anatomical relationships around the intraoral and the submandibular gland. This is necessary given that the unusual view which surgeons have is not always familiar and can cause confusion. Third, surgeons are required to employ more sophisticated techniques than required in the standard transcervical approach in order to avoid lingual paresthesia. Fourth, large size or severely scared submandibular cannot be resected by this procedure. In our case, the large size pseudocyst and adhesions led us to expect not to be able to completely resect without postoperative complications such as delayed healing and infection associated with the remaining large dead space. As a result, we selected to resect the submandibular gland with psuedocysts through the transcervical approach. In the future, the transoral approach should be considered as the first-line procedure for similar cases.

Conclusion

This manuscript outlines on a rare case of a submandibular gland mucocele and provides a systematic review. The lesion was resected with the submandibular gland.

Consent

Written informed consent was obtained from the patient for publication of this case report.

Authors contributions

JY conceived of the study. KS prepared and edited the manuscript. HK contributed to the acquisition of data. YF contributed to the pathological analysis. KI performed the statistical analysis. JY revised the final version of the manuscript. All authors read and approved the final manuscript

Acknowledgments

This study was supported in part by Grants-in Aid for Scientific Research from the Ministry of Education, Culture, Sports, and Technology (22591920) of Japan.

References

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