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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 30  |  Issue : 3  |  Page : 318-320

Angiofibrolipoma of the lower lip: A rare case report


1 Department of Oral Medicine and Radiology, Pushpagiri College of Dental Sciences, Thiruvalla, Kerala, India
2 Department of Oral and Maxillofacial Pathology, Pushpagiri College of Dental Sciences, Thiruvalla, Kerala, India
3 Department of Oral and Maxillofacial Surgery, Pushpagiri College of Dental Sciences, Thiruvalla, Kerala, India

Date of Submission28-May-2018
Date of Acceptance20-Jul-2018
Date of Web Publication18-Oct-2018

Correspondence Address:
Dr. Lisa Elizabeth Jacob
Department of Oral Medicine and Radiology, Pushpagiri College of Dental Sciences, Thiruvalla, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_94_18

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   Abstract 


Lipomas are commonly occurring benign tumors consisting of mature white adipocytes surrounded by a thin fibrous capsule. It is the most common type of soft tissue mesenchymal neoplasm. Angiofibrolipoma is a rare subtype of lipoma. The most common site of angiofibrolipoma is in the back, the neck, or the shoulders. Only a few cases have been reported to occur in the oral region. This article reports a case of a 34-year-old male patient with a chief complaint of a swelling in the lower lip for the past 2 years and was diagnosed as an angiofibrolipoma. A thorough search of the literature was carried out using Pubmed, Sciencedirect, Google Scholar, and Medline until 2018 using angiofibrolipoma as a keyword. To the best of our knowledge, this is the first case reported in the literature involving the lower lip.

Keywords: Angiofibrolipoma, lower lip, lipoma, neoplasm


How to cite this article:
Jacob LE, Babu SS, Nair SS, Koshy S. Angiofibrolipoma of the lower lip: A rare case report. J Indian Acad Oral Med Radiol 2018;30:318-20

How to cite this URL:
Jacob LE, Babu SS, Nair SS, Koshy S. Angiofibrolipoma of the lower lip: A rare case report. J Indian Acad Oral Med Radiol [serial online] 2018 [cited 2019 Jun 19];30:318-20. Available from: http://www.jiaomr.in/text.asp?2018/30/3/318/243672




   Introduction Top


Lipomas are benign tumors consisting of mature white adipocytes with a thin fibrous capsule. It is the most common type of soft tissue mesenchymal neoplasm.[1] The incidence in the head and neck is between 15% and 20% and in the oral region around 1%–4%.[2] The most common sites include buccal mucosa, labial mucosa, tongue, floor of the mouth, and rarely intraosseous.[3],[4]

The histopathological variants of lipoma include fibrolipoma, angiolipoma, angiomyolipoma, angiofibrolipoma, myolipoma, pleomorphic lipoma, myxolipoma, osteolipoma, and adenolipoma. Fibrolipoma is the most common variant. Angiofibrolipoma is a rare subtype.[5] Angiofibrolipoma is a neoplasm containing capillaries and fibrocytes with mature adipose tissue.[2],[6]


   Case Report Top


A 34-year-old male patient reported to the Department of Oral Medicine and Radiology with a chief complaint of a swelling in the lower lip for the past 2 years. No history of pain, trauma, color change, or bleeding was present and there was no history of similar lesion in the past occurring on the lip or elsewhere in the body. The patient reported that the swelling had gradually increased to attain the present size. On inspection, a single swelling was found on the lower lip, left of the midline, measuring 6 × 6 mm in diameter, well-circumscribed margins, the skin over the swelling was normal. On palpation, the swelling was found to be slightly firm in consistency and nontender with absence of any kind of discharge [Figure 1] and [Figure 2]. A provisional diagnosis of a fibroma was made with the differential diagnosis including mucocoele and lipoma. The patient was advised to undergo surgical excision of the lesion followed by histopathological examination of the specimen. [Figure 3] shows the immediate postoperative photograph of the patient. Excisional biopsy was carried out and the sample was stained with hematoxylin and eosin. Histopathological analysis revealed the presence of fibrous connective tissue with vascular channels and lipid cells [Figure 4] and [Figure 5]. Based on these features, a diagnosis of angiofibrolipoma was made. The patient has been followed up for a period of 6 months and no complications or recurrences have been reported.
Figure 1: Clinical appearance of the swelling on the lower lip, left of the midline

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Figure 2: Clinical appearance of the swelling on the lower lip, lateral view

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Figure 3: Immediate postoperative image

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Figure 4: Photomicrographs of angiofibrolipoma: ×10

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Figure 5: Photomicrograph of angiofibrolipoma: ×40

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   Discussion Top


The etiology of lipoma is uncertain; however, various factors such as hormonal factors, infarction, infection, chronic irritation, obesity, trauma, and familial link contribute to the etiology.[6]

Two theories have been proposed to explain the formation of lipomas. The hypertrophy theory states that obesity and the associated increase of adipose tissue may together play a role in the formation or lipomas.[7] However, this theory fails to explain how lipomas also occur in areas which do not contain any pre-existing adipose tissue. The theory of metaplasia suggests that the mesenchymal cells undergo aberrant differentiation in situ leading to the formation of lipoblasts.[7]

The clinical appearance of lipomas depends on the location in which it occurs. Lipomas commonly occur in middle-aged individuals with a male predilection. They are usually slow growing, painless, well circumscribed, mobile, fluctuant and have a yellow hue. They are clinically nonaggressive. If the yellow colour does not show through the overlying mucosa, the lesion can be easily misdiagnosed.[6],[8] Angiofibrolipomas in particular usually occur as a subcutaneous, solitary, lesions on the neck, back, and shoulders, and only a few cases have been reported to occur orally.

Lipomas usually occur as a single entity. However, multiple lipomas occur in Cowden's syndrome, Gardener's syndrome, neurofibromatosis, Decrum's disease, Proteus syndrome, encephalocraniocutaneous lipomatosis, Pai syndrome, and multiple familial lipomatosis.[6],[9]

Agha-Hosseini et al. reported a case of angiofibrolipoma of the retromolar pad.[2] Saddik et al. reported a case of a liposarcoma involving the base of the tongue as well as the tonsillar fossa.[9] The patient had a history of a recurrent mass, which was surgically resected several times and was at one point diagnosed as an angiofibrolipoma. Brkić et al. reported a case of angiofibrolipoma of the buccal mucosa.[10]

The differential diagnosis of lipoma depends on the site of occurrence and includes fibroma, salivary gland pathology, epidermoid cyst, dermoid cyst, neurofibroma, granular cell tumour, schwannoma, and lymphoma.[9]

The management of angiofibrolipomas is similar to that for lipomas. Conservative surgery is advised. Angiofibrolipoma has a good prognosis. The rate of recurrence is extremely low. Infiltrating lesions, however, have a higher rate of recurrence. Regular follow-up is required.

The clinical significance of this lesion lies in the fact that angiofibrolipomas can easily be misdiagnosed and serves as a diagnostic challenge. Although the incidence of these lesions are very rare, they must be included in the differential diagnosis of soft tissue swellings, which occur in the oral region and they must also be identified and distinguished early from other neoplasms. An extensive search of the literature was carried out using Pubmed, Sciencedirect, Google Scholar, and Medline until 2018 using angiofibrolipoma as a keyword. To the best of our knowledge, this is the first case reported in the literature involving the lower lip.


   Conclusion Top


Angiofibrolipomas are a rare variant of lipoma. To the best of our knowledge, this is the only case of an angiofibrolipoma to be reported in the lip.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Mariappan N, Singh R, Gayathri T. Angiofibrolipoma of the Tendon Sheath. J Adv Plast Surg Res 2016;2:5-8.  Back to cited text no. 1
    
2.
Agha-Hosseini F, Moslemi E. Angiofibrolipoma of the retromolar pad region: Case report. N Y State Dent J 2014;80:33-7.  Back to cited text no. 2
    
3.
Sachdeva SK, Rout P, Dutta S, Verma P. Oral lipoma: An uncommon clinical entity. J Oral Maxillofac Radiol 2013;1:118-21.  Back to cited text no. 3
  [Full text]  
4.
Johnson EC. Intraosseous lipoma: Report of case. J Oral Surg 1969;27:868-70.  Back to cited text no. 4
    
5.
Kucuker I, Keles MK, Yosma E, Polat V. Angiofibrolipoma of the tendon sheath. Case Reports Plast Surg Hand Surg 2014;1:11-2.  Back to cited text no. 5
    
6.
Uwale Eyesan S, Christopher Ayeni S, Adesope Adesina S, Tolulope Kehinde G, Olawepo A, Kazeem Ibrahim O, et al. Angiofibrolipoma of the calf. Rare Tumors 2013;5:e48.  Back to cited text no. 6
    
7.
Surej Kumar LK, Kurien NM, Raghavan VB, Menon PV, Khalam SA. Intraoral Lipoma: A case report. Case Reports in Medicine 2014;2014:4.  Back to cited text no. 7
    
8.
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 2nd ed. Philadelphia: W. B. Saunders; 2002. p. 398-404.  Back to cited text no. 8
    
9.
Saddik M, Oldring DJ, Mourad WA. Liposarcoma of the base of tongue and tonsillar fossa: A possibly underdiagnosed neoplasm. Arch Pathol Lab Med 1996;120:292-5.  Back to cited text no. 9
    
10.
Brkić A, Ozçamur C, Gürkan-Köseoğlu B, Olgac V. Angiofibrolipoma of the buccal mucosa: A case report. J Oral Sci 2010;52:173-6.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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