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A Case of Oral Lymphangiom Mimicking Mucocele

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A Case of Oral Lymphangiom Mimicking Mucocele

Letter To The Editor DOI: 10.6003/jtad.17114l1

Published: J Turk Acad Dermatol 2017; 11 (4): 17114l1.This article is available from: http://www.jtad.org/2017/4/jtad17114l1.pdf Key Words: Oral Lymphangiom, Micocele

To the editor - Lymphangiomas are benign hemar- tomaoustumours of lymphatic channels [1]. They are mostly seen at birth or early childhood. There are capillary, cavernous and cystic types of lymphangiomas.Head and neck are the mostly en- countered regions for lymphangiomas but they can be seen in all parts of the body including mucosal areas. However,oral cavity localizations are extre- mely rare, if seen, they involve dorsum of tongue as the most frequent, palate, buccal mucosa, gin- giva and lips [2]. Mucocele is a common cystic mu- cous-filled lesion of oral cavity originated from minor salivary glands.

23 yrs male patient admitted to dermatology out- patient department complaining from a lump on his lower lip emerged six months ago (Figure 1).

He had no subjective symptom except cosmetic concern and the lesion did not grow during this time. In examination, an edematous and erythe- matous slightly verrucous papule measuring 6x9 mm was observed over inner surface of lower lip.The patient was send to be excised as a preli- minary diagnosis of mucocele to ENT surgeon. Ex- cisional biopsy was done followed by primary suturing. Pathological examination revealed der- mal thinning, dilatation of lymphatic channels fi lled with lymph fluid compatible with lymphan- gioma (Figure 2). There was no recurrence in fol- low-up of the patient during one year.

Lymphangiomas are benign hemartomaous tumo- urs of lymphatic channels. They are caused by broken central lymphatic drain finally resulting to accumulation of lymphatic secretions and channel dilatation. Half of all lymphangiomas are observed at birth while 90% of them seen first 2 years of life [1]. Lymphangioma during adulthood is extremely rare [2]. Although every part of the body may be involved, 75% of them are seen in head and neck region [1]. Proximal extremities, hip and trunk are other involved areas respectively. Oral lymphan- giomas are so rare. Dorsum of tongue, palates

buccal mucosa, gingiva and lips are the areas of oral localizations of lymphangiomas [3]. Lower lip lymphangiomas constitute 6 % of oral lymphan- giomas [3].

In addition to cosmetic disfigurement, oral lypmhangiomas may cause to speech and feeding problems as well as macroglossia in terms of in- volved area in oral cavity. Sometimes fatal cases had been reported due to secondary infection and trauma [1,2,4]. Our patient had admitted to hos- pital only for cosmetic concern. Solitary lesion re- sembling mucocele and its common location on inner surface of lower surface in adult age did not make us think of a lymphangioma diagnosis at presentation. Acinic cell carcinoma was also repor- ted which has an initial diagnosis of mucocele [5].

Additionally,lymphoepitelial cysts should be put as a differential diagnosis of lymphangioma [6].

The clinical appearance of lymphangioma ma inly depends on depth of the lesion. Superficial lymphangiomas are generally seen as smooth sur- faced lumps containing clear and rarely pink or yellowish fluid [2]. However a superficial solitary lump like our case may be easily confused with mucocele.

Histopathological features of lymphangioma are very suggestive, which has dilated lymphatic channels containing eryhtrocyes, lymph fluid, lymphocytes, neutrophils and macrophages [2], whereas mucocele has minor salivary gland tissue filled with mucus in the specimen. Treatment of lymphangiomas is optional according to their lo- calization status: surgical excision, electrocauteri- zation, cryosurgery, radotherpy, sclerosing agent injection, ablation, embolization, carbondioxide and argon laser use along with frequent recur- rence [2,4,7]. Small and clear-margin lymphangi omas can be succesfully excised with low recur- rence rate like our case who has no reappearing after one year.

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Consequently, lymphangiomas should be kept in mind in the differential diagnosis of solitary cystic lesions of lower lip mucosa despite mucocele is a primary diagnosis on that location. Surgical exci- sion is an efficent way of removing.Histopathologi- cal examination must be done in every mucosal lesion even a definite prediagnosed benign cyst.

Suha Ertugrul,1MD Gökşen Ertugrul,2MD Habibullah Aktaş,2MD Alpay Aktumen,³ MD

Harun Erol,³ MD

1Karabuk University Karabuk Tranining and Research Hospital ENT Department

2Karabuk University Traniningand Research Hospital Dermatology ENT Department

3Karabuk University Training and research hospital Pathology Department

E-mail: aktashabib@hotmail.com

References

1. Neville BW, Damm DD, Allen CM, Bouquot JE. Soft tissue tumors. In: Neville BW, editor. Oral and Ma- xillofacial Pathology. 2nd ed. New Delhi, Philadelphia:

Saunders Publishers 2002; p. 475-477.

2. Sunil S, Gopakumar D, Sreenivasan BS. Oral lymphangioma - Case reports and review of litera- ture. Contemporary Clinical Dentistry 2012; 3: 116–

118. PMID: 22557912

3. Bernnan TD, Miller AS, Chen S.L ymphangiomas of the oral cavity:A Clinicopathologic, Immunohistoche- mical and Electron microscopic study. J Oral Maxil- lofac Surg 1997; 55: 932-935. PMID: 9294501 4. Goswami M, Singh S, Gokkulakrishnan S, Singh A.

Lymphangioma of the tongue. Natl J Maxillofac Surg 2011; 2: 86-88 PMID: 22442618

5. Cho JH, Yoon SY, Bae EY, Lee CN, Lee JD, Cho SH.

Acinic cell carcinoma on the lower lip resembling a mucocele Clin Exp Dermatol 2005; 30: 490-493.

PMID: 16045674

6. Baurmash HD. Mucoceles and ranulas. J Oral Maxil- lofac Surg 2003; 61: 369-378. PMID: 12618979 7. Usha V, Sivasankari T, Jeelani S, Asokan GS, Part-

hiban J. Lymphangioma of the Tongue - A Case Re- port and Review of Literature J Clin Diagn Res 2014;

8: 12-14. PMID: 25386537

J Turk Acad Dermatol 2017; 11 (4): 17114l1. http://www.jtad.org/2017/4/jtad17114l1.pdf

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(page number not for citation purposes) Figure 2. Dermal thinning, dilatation of lymphatic channels filled with

lymph fluid Figure 1. An edematous papule on

his lower lip

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