Sialolipoma of the Lower Lip: Case Report and Literature Review

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208 The Open Dentistry Journal, 2012, 6, 208-211

1874-2106/12 2012 Bentham Open

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Sialolipoma of the Lower Lip: Case Report and Literature Review

Nada O. Binmadi

^1,*

, Risa Chaisuparat

²

, Bernard A. Levy

³

and Nikolaos G Nikitakis

⁴

1

Department of Oral, Basic, and Clinical Sciences, King Abdulaziz University, Jeddah, Saudi Arabia

2

Department of Oral Pathology, Faculty of Dentistry, Chulalongkorn University

3

Director of Global Operations, Oral and Maxillofacial Pathology, Department of Oncology and Diagnostic Sciences, Dental School, University of Maryland, Baltimore

4

Department of Oral Pathology and Medicine, Dental School, National and Kapodistrian University of Athens

Abstract: Sialolipoma is a relatively rare and fairly recently described as a variant of lipoma with salivary elements. Any site within the oral and maxillofacial region may be involved with the parotid gland being the most common location.

Herein, we present a case of silaolipoma in lower lip. The clinical and histological features and differential diagnosis are discussed.

Keywords: Lipoma, Salivary gland, Oral cavity.

INTRODUCTION

Sialolipoma is a new histological variant of salivary gland lipoma, which is composed of adipose and glandular tissues. It was first described by Nagao et al., in 2001 [1].

The etiology of sialolipoma is not completely understood. It typically arises within the major salivary glands and the mi- nor salivary gland of oral cavity. To the best of our knowl- edge only 35 cases of sialolipoma have been reported in English literature (Table1 [1-21]) including the present case.

CASE REPORT

A 54-year-old Caucasian female was seen by her general dentist for evaluation of a painless swelling in her lower lip.

There was no history of trauma or infection and the patient’s medical history was unremarkable. An intra-oral examina- tion revealed a 0.6x0.6cm soft tissue mass with normal over- lying mucosa in her left lower lip, while an extra-oral ex- amination revealed a normal facial morphology. The clinical differential diagnosis included mucocele, fibroma, lipoma, and salivary gland neoplasm. An excisional biopsy was per- formed and submitted to the Oral and Maxillofacial Pathol- ogy Department at University of Maryland, Baltimore. On gross examination, the mass was well-circumscribed, tan in color, soft in consistency and measured 0.6cm at its largest diameter. The histological examination revealed a mass of mature adipose tissue completely encapsulated by a fibrous band. Islands of salivary gland acini and ducts were located within the tumor. Neither atypia nor mitotic figures were observed in either the salivary glandular type tissue or the adipocytes. Mild lymphocytic infiltration and ductal dilation were seen (Fig. 1). Consequently, the lesion was diagnosed as sialolipoma and no further treatment was required. The

*Address correspondence to this author at the Department of Oral, Basic, and Clinical Sciences, King Abdulaziz University, Jeddah, Saudi Arabia;

Tel: 0505699092; E-mail: nmadi@kau.edu.sa

patient has been followed for 3 years without evidence of recurrence.

DISCUSSION

Sialolipoma, an uncommon variant of head and neck lipoma, is composed of proliferative adipocytes with en- trapped normal salivary gland islands [1]. Almost any site within the oral and maxillofacial region may be involved with the parotid gland being the most frequently reported location [1-3, 6, 7, 9, 14, 21]. To our knowledge, 34 cases of sialolipoma have previously been reported in the English literature and eighteen of them were found in minor salivary glands [seven on the palate [1, 8, 10, 16, 17, 19], three in buccal mucosa [4, 10, 20], three on floor of the mouth [5, 11, 20], two on the tongue [4, 20], two in lower lip [12,the pre- sent case], and one on retromolar pad [20].

Clinically, sialolipomas usually present as a solitary pain- less palpable mass with an average size of 2.74 cm in diame- ter. Females are affected slightly more than males (with ratio 1.1:2). Patient’s ages range from 6 weeks to 84 years, with average of 47.6 years. The duration of the lesion range from two months to ten years, with average of three years. In the present report, the lesion is in the lower lip and the diameter is 0.6 cm. Because lower lip is a preferable site of mucocele, it is probable that superficially located sialolipoma might be misdiagnosed clinically as mucocele. The other most com- mon preoperative diagnoses are fibroma and salivary gland tumor. There is no distinguishable radiographic sign for sia- lolipoma in either computed tomography scan (CT) or mag- netic resonance imaging (MRI) compared to a typical fatty lesion in the head and neck region [14].

Histological findings of haematoxylin and eosin staining

in previous studies include a well circumscribed mass sur-

rounded by a delicate fibrous tissue. The tumors are com-

posed of mature adipose elements mixed with salivary gland

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Sialolipoma of the Lower Lip: Case Report and Literature Review The Open Dentistry Journal, 2012, Volume 6 209

Fig. (1). (A) Photomicrograph showing islands of salivary gland tissue present within an adipose tissue tumor encapsulated by thin fibrous tissue (arrow) (hematoxylin and eosin, original magnification 4x); (B) Higher magnification revealing mild ductal dilatation with fibrosis within the tumor mass (hematoxylin and eosin, original magnification 10x).

Table 1. Clinical Features of 36 Cases of Sialolipoma

Author Age

(years) Sex Location Size in cm Duration Treatment Follow-up

Walts and Perzik, [2]

48 65

M M

Parotid gland Parotid gland

3.5x2.5x1 2.6 diameter

NA 2 months

Superficial parotidectomy Superficial parotidectomy

NED NED Baker et al., [3] 44 M Parotid gland 1.0 diameter 2 months Superficial parotidectomy 30 mo; NED Nagao et al., [1]

Nagao et al., [1]

20 45 67 66 42 66 75

M F M F M M M

Parotid gland Parotid gland Parotid gland Parotid gland Parotid gland

Soft palate Hard palate

3.5x3.0x2.2 6.0x3.0x2.0 1.7diameter 6.0 diameter 6.0 diameter 2.2x1.5x1.5 1.0 diameter

4 months 10 years 2 months 5 months 10 years 6 years 3 years

Superficial parotidectomy Superficial parotidectomy Superficial parotidectomy Superficial parotidectomy Superficial parotidectomy

Surgical excision Surgical excision

7 yr, 7 mo;NED 7 yr, 1 mo;NED 3 yr,1mo; NED 2 yr,11mo;NED 1 yr 8 mo; NED 11 mo; NED

NA Fregnani et al., [4]

Fregnani et al., [4]

NA NA

Tongue Buccal sulcus

NA NA

NED NED

Lin et al., [5] 67 F Floor of the mouth 3.0x2.0 1 year Surgical excision 2 yr; NED

Hornigold et al., [6] 7 wk F Parotid gland 2.0x1.7x1.1 10 weeks Surgical excision 2 yr; NED Michaelidis et al., [7] 44 M Parotid gland 3.5 diameter 1 .5 years Total parotidectomy 2 yr; NED

Sakai et al., [8] 60 F Hard palate 1.8x1.2x1.0 10 years Surgical excision NED

Kadivar et al., [9] 3 F Parotid gland 3.0 diameter 8 months Superficial parotidectomy NA Ramer et al., [10]

Ramer et al., [10]

84 43

F F

Buccal mucosa Soft palate

1.0x1.0 2.0x2.0

NA NA

11 mo; NED NA

Ponniah et al., [11] 70 M Floor of mouth 2.0 diameter NA Surgical excision 2 yr; NED

De Freitas et al., [12] 38 M Lower lip 1.0 diameter NA Surgical excision NA

Parente et al., [13] 77 F Submandibular gland 3.0x2.0x1.8 NA Surgical excision 22mo; NED

Dogan et al., [14] 33 M Parotid gland 2.0x2.0 1year Superficial parotidectomy NED

Jang et al., [15] 62 F Submandibular gland 5.0 diameter 2-3years Surgical excision 17mo,NED

Okada et al., [16] 66 F Hard palate 0.8 diameter 10 years NA NA

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210 The Open Dentistry Journal, 2012, Volume 6 Binmadi et al.

Table 1. contd…

Author Age

(years) Sex Location Size in cm Duration Treatment Follow-up

De Moraes et al., [17] 72 F Hard palate 2.0 diameter 2 weeks Surgical excision 8 mo; NED

Sato et al., [18] 3 M Submandibular gland 4.0x3.0 NA Surgical excision 3 yr; NED

Akrish et al., [19]

52 67

M F

Submandibular gland Hard Palate

3.5x2.0x1.5 5.0x4.0x4.0

NA NA

1yr; NED 1yr; NED Nonaka et al., [20]

Nonaka et al., [20]

27 73 65 68

F F F F

Tongue Floor of mouth Buccal Mucosa Retromolar pad

1.0x1.0 4.0x1.0 2.0 diameter 0.9 diameter

5 years NA 2 years

NA

Surgical excision Surgical excision Surgical excision Surgical excision

NA NA NA 14mo; NED Kidambi et al., [21] 6 wk M Parotid gland 4.7x4.5x3.0 4 wk Total parotidectomy with

facial nerve dissection

3 mo; NED

Case report*, 54 F Lower lip 0.6 diameter NA Surgical excision 3 yr; NED

Total number of cases=35

Avg:

47.6

M:F 15:18

Parotid gland:13, Hard palate:5, Soft palate:2, Tongue :2, Floor of mouth:3,

Buccal mu- cosa:3,Lower lip :2,submandibular gland:4, retromolar

pad: 1

Avg:2.74 Avg: 3.04 years

*Present case report; NA, not available; NED, no evidence of disease.

tissues. The glandular components, consisting of acinar cells and ductal components, may be scattered through out the tumor or located in the periphery of the tumor [1, 5]. The 80 % of sialolipomas in major salivary gland are composed of adipose tissue while in minor salivary gland the glandular elements are clustered and evenly distributed around fat tis- sue [10,11]. No mitosis is seen in adipocytes or acinar and ductal cells [5]. The glandular components may be showed ductal dilation, oncocytic changes and squamous ductal metaplasia [1, 5-10]. In some cases areas of fibrosis are seen while myxoid changes are reported only in one case [9, 15].

Additionally, lymphocyte infiltration and enlarged congested vessels are reported [1, 10, 15].

The pathogenesis of the sialolipoma is not completely understood. However, immunohistological and ultrastruc- tural studies confirmed that the glandular elements of the lesion could arise from entrapment of minor salivary gland during lipomatous proliferation rather than representing neo- plastic process [1, 4, 5].

The morphologic differential diagnosis includes a variety of entities. Adenolipoma has histologic characteristic similar to sialolipoma; but it is composed of adipocytes and duct elements without acinar cells. Adenolipoma also differs from sialolipoma by the lack of organoid arrangement of the duc- tal type tissue [1, 5, 8]. Lipomatosis which typically occurs in older patients can be excluded by the microscopic lack of the fibrous capsule in addition to the absence of any medical condition associated with lipomatosis, for instance diabetes mellitus, malnutrition, chronic alcoholism and liver cirrhosis

[1, 7, 9]. The distinction from pleomorphic adenoma is made by the presence of extensive fatty elements within the nor- mal salivary gland tissue and lack of ducts and strands of dark-staining myoepithelial cells in sialolipoma [1, 5, 10].

Sialolipoma in the minor salivary glands is treated by complete surgical excision. However, most of tumors in pa- rotid glands are treated with superficial parotidectomy. A complete parotidectomy with preservation of the facial nerve has been reported in two cases [7, 21] (Table 1). Malignant transformation of sialolipoma has not been reported yet in the literature [5]. The follow up period ranged from 2 months to 10 years and there is no evidence of recurrent sia- lolipoma.

CONFLICT OF INTEREST

The authors confirm that this article content has no con- flicts of interest.

ACKNOWLEDGEMENT Declared none.

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Received: August 16, 2012 Revised: September 17, 2012 Accepted: October 30, 2012

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