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| CASE REPORT |
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| Year : 2015 | Volume
: 7
| Issue : 2 | Page : 129-131 |
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Lymphangioma of the buccal mucosa: A case report with a literature review
Chandrashekhar Pammar, Vijayalakshmi S Kotrashetti, Ramakant Nayak, Jagadish Hosmani
Department of Oral Pathology and Microbiology, Maratha Mandal's NGH Institute of Dental Sciences and Research Centre, Belgaum, Karnataka, India
| Date of Web Publication | 17-Nov-2015 |
Correspondence Address:
Dr. Vijayalakshmi S Kotrashetti
Department of Oral Pathology and Microbiology, Maratha Mandal's NGH Institute of Dental Sciences and Research Centre, Belgaum - 590 010, Karnataka
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0975-8844.164311
Lymphangioma is a benign, hamartomatous tumor of the lymphatic system. It is usually found in the head and neck region and is widely regarded as a developmental lesion rather than a true neoplasia. Most lymphangiomas are present at birth. In head and neck area, the most common location is the submandibular region, followed by the parotid gland. When lymphangioma occurs in the oral cavity, the anterior two third of the tongue is most commonly affected. The occurrence of lymphangioma in other parts of the oral cavity is very uncommon. Here, we report an unusual case of lymphangioma of buccal mucosa in a 45-year-old female. Keywords: Buccal mucosa, lymphangioma, lymphatic vessels, oral cavity
How to cite this article:
Pammar C, Kotrashetti VS, Nayak R, Hosmani J. Lymphangioma of the buccal mucosa: A case report with a literature review. J Orofac Sci 2015;7:129-31 |
How to cite this URL:
Pammar C, Kotrashetti VS, Nayak R, Hosmani J. Lymphangioma of the buccal mucosa: A case report with a literature review. J Orofac Sci [serial online] 2015 [cited 2023 Jun 9];7:129-31. Available from: https://www.jofs.in/text.asp?2015/7/2/129/164311 |
| Introduction | |  |
Lymphangioma is benign congenital or hamartomatous malformation of lymphatic vessels, first described by Redenbacher in 1828. [1] They arise from sequestrations of lymphatic tissue and have marked predilection for head and neck region. 50-70% of them occur in head and neck region. [2] They are usually found at birth and around 90% of them develop by 2 years of age. [3] lymphangiomas are known to occur in the oral cavity with a marked predilection for anterior two-third of the tongue. The occurrence of this lesion in the other sites of the oral cavity has also been reported.
Histologically they show multiple intertwining lymph vessels in a loose fibrovascular stroma. [4] Even though, lymphangiomas are benign lesions the involvement of vital structures or esthetic and functional requirements may necessitate the treatment. [3] Various treatments have been reported for lymphangioma, but the most accepted treatment is complete surgical excision. Prognosis is good for most cases of lymphangioma, except for large tumors of the tongue, which may result in airway obstruction and death. [5] Literature survey showed only 14 cases reported on lymphangioma of buccal mucosa. We here, report an additional case of lymphangioma of buccal mucosa and detail review of the literature on lymphangioma of buccal mucosa.
| Case Report | | |
A 45-year-old female patient reported to outpatient department of the institute with the chief complaint of growth in the left buccal mucosa since 2 years; she gave a history of cheek bite after which she developed small, asymptomatic growth, which gradually increased in size. The lesion was interfering with mastication and experienced pain when the growth is impinged between teeth. Patient gave a history of bleeding only if there was trauma. On examination, the single lobulated bluish red translucent lesion was observed on the left buccal mucosa, at the level of occlusal plane measuring about 0.8 cm × 0.9 cm. On palpation, the lesion was sessile, non-tender and soft in consistency [Figure 1]a. | Figure 1: (a) Intra-oral photograph showing growth in left buccal mucosa, (b) photograph showing gross appearance of excised specimen, (c) photomicrograph showing stratified squamous epithelium with juxta epithelial stroma showing large thin endothelial lined lymphatic vessels (×4 H and E), (d) photomicrograph showing eosinophilic material resembling lymph in lymphatic vessels with few lymphocytes (×4 H and E)
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Provisional clinical diagnosis of traumatic fibroma, pyogenic granuloma was made. Routine blood examination was performed, which showed all values in the normal range.
Excisional biopsy of the lesion was done and submitted for histopathology. The soft tissue was whitish red in color, soft in consistency, and measured 0.5 cm × 1 cm. The specimen was routinely fixed and processed [Figure 1]b. Hematoxylin and eosin stained section showed stratified squamous epithelium with juxta epithelial stroma showing large thin endothelial lined vessels, few vessels showed eosinophilic material resembling lymph and few lymphocytes [Figure 1]c and [Figure 1]d. Based on this a final diagnosis of lymphangioma was made. The healing of the lesion was uneventful. One year follow-up of the case showed no evidence of recurrence [Figure 2]. | Figure 2: Photograph showing postoperative follow-up after 1-year with good healing and no recurrence noticed
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| Review of Literature | | |
On English language literature search, we found total 14 cases of lymphangioma of buccal mucosa being reported until date to which we add the additional case of lymphangioma [Table 1]. | Table 1: Details of total number of lymphangioma of buccal mucosa cases reported in the literature
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Of 14 cases reported complete details were available only for four cases, which was individual case report, [6],[8],[9],[10] whereas eight cases were reported in review on lymphangioma of the oral cavity by Brennan et al. [3] Two cases were reported by Domingo et al. in a review on benign tumors of the oral mucosa from the archives of General University Hospital. [7] Details of all 14 cases have been mentioned in [Table 1].
Of 14 cases, only one case had a history of trauma and showed recurrence after 2 months of treatment. Hence, treatment for recurrence was done by cryosurgery. [8] In two cases, the lesion was present since birth. [6],[10]
| Discussion | | |
Lymphangiomas develop as a result of sequestration of portion of the primitive embryonic lymphatic anlage. These sequestrated areas never have efficient anastomoses with larger lymphatic channels. Hence they present as localized areas of lymphatic blockage which results at times to form lymphangiomas or cystic hygromas. [11],[12]
Lymphangiomas are classified morphologically macrocystic, microcystic, and mixed. Our case was a microcystic variant. [9] Usually, lymphangiomas are present since birth (60%). Adult lymphangiomas are very infrequent. A thorough review by Brennan et al. on oral lymphangiomas reported 22.7 years as the mean age of occurrence and both sexes being equally affected. Oral lymphangiomas occur more commonly in the anterior two-third of the tongue, resulting in macroglossia. Various other sites such as lip, palate, and buccal mucosa have been reported. He reported buccal mucosa as a second most common site for oral lymphangioma. Clinically, oral lymphangiomas present as superficial lesion, demonstrating pebbly surface, resembling a cluster of the translucent vesicle. The surface appears like frog eggs or tapioca pudding, deeper lesions present as soft ill-defined masses. [4] Trauma is said to be one of the cause for superficial lymphangioma. One case had a history of trauma. [8] Even though it is a benign tumor it may lead to the complicated case, due to its infiltrating nature, indefinite demarcation, and involvement of vital structures. Clinically, the lesion may appear like many other oral lesions such as hemangioma, teratoma, lingual thyroid nodule, granular cell tumor, and heterotrophic gastric mucosal cyst. Lymphangiomas neither become malignant nor have a familial tendency. Histologically they show proliferation of lymphatic vessels with lymph in the vessels with erythrocytes and lymphocytes. The superficial lesion consists of dilated lymph vessels lined by flat endothelial cells in a discontinuous layer immediately subjacent to the oral epithelium. Deeper lesion shows irregular, dilated, and interconnected lymphatic vessels.
Various treatment modalities have been reported. The main option for treatment includes surgical excision, radiation therapy, cryotherapy, electrocautery, sclerotherapy, steroid administration, ligation, and laser surgery. [11],[13],[14]
Most adult lymphangiomas are encapsulated or partially circumscribed and hence treatment of choice remains surgical excision. Recurrence after surgery is mainly due to inadequate tumor removal.
| Conclusion | | |
Occurrence of adult lymphangioma is very rare, especially lymphangioma of buccal mucosa. Here, we report an additional case of buccal mucosal lymphangioma with a history of trauma and no recurrence observed on 1-year follow-up after surgical excision. Therefore, early recognition and proper initiation of the treatment will prevent the occurrence of complications associated with lymphangioma.
| References | | |
| 1. | Souza RJ, Tone LG. Treatment of lymphangioma with alpha-2a-interferon. J Pediatr (Rio J) 2001;77:139-42.  |
| 2. | Neville BW, Damm DD, Allen CM. Oral and Maxillofacial Pathology. 3 rd ed. Philadelphia: Saunders Elsevier; 2009. p. 547-49. |
| 3. | Brennan TD, Miller AS, Chen SY. Lymphangiomas of the oral cavity: A clinicopathologic, immunohistochemical, and electron-microscopic study. J Oral Maxillofac Surg 1997;55:932-5. |
| 4. | Sunil S, Gopakumar D, Sreenivasan BS. Oral lymphangioma - Case reports and review of literature. Contemp Clin Dent 2012;3:116-8. [ PUBMED]  |
| 5. | Rajendra R, Sivapathasundaram B. Shafer′s Textbook of Oral Pathology. 6 th ed. Elsevier India: Elsevier Publications; 2009. p. 146-8. |
| 6. | Bozkaya S, Ugar D, Karaca I, Ceylan A, Uslu S, Baris E, et al. The treatment of lymphangioma in the buccal mucosa by radiofrequency ablation: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:e28-31. |
| 7. | Torres-Domingo S, Bagan JV, Jiménez Y, Poveda R, Murillo J, Díaz JM, et al. Benign tumors of the oral mucosa: A study of 300 patients. Med Oral Patol Oral Cir Bucal 2008;13:E161-6. |
| 8. | Dogan N, Durmaz CE, Sencimen M, Ucok O, Okcu KM, Gunhan O, et al. The treatment of recurrent lymphangioma in the oral buccal mucosa by cryosurgery: A case report. OHDMBSC 2010;9:7-10. |
| 9. | Coskunses F, Tugcu F, Koçyigit ID, Ozgul O, Karadeniz SN. Lymphangioma of buccal mucosa - A case report. Int J Dent Clin 2012;4:50-1. |
| 10. | Haranal S, Naresh N, Vinuth DP, Agarwal P, Rohit M. Lymphangioma of the buccal mucosa. Maxillofac Surg 2013;4:322-4. |
| 11. | Balakrishnan A, Bailey CM. Lymphangioma of the tongue. A review of pathogenesis, treatment and the use of surface laser photocoagulation. J Laryngol Otol 1991;105:924-9. |
| 12. | Bill AH Jr, Sumner DS. A unified concept of lymphangioma and cystic hygroma. Surg Gynecol Obstet 1965;120:79-86. |
| 13. | Suen J, Waner M. Treatment of oral cavity vascular malformations using the Meodymium: YAG laser. Arch Otolaryngol Head Neck Surg 1989;9:115-32. |
| 14. | Hellmann JR, Myer CM 3 rd , Prenger EC. Therapeutic alternatives in the treatment of life-threatening vasoformative tumors. Am J Otolaryngol 1992;13:48-53. |
[Figure 1], [Figure 2]
[Table 1]
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