Journal of Integrated Health Sciences

CASE REPORT
Year
: 2020  |  Volume : 8  |  Issue : 2  |  Page : 91--94

Oral Soft-Tissue Angiolipoma: Rarest Intraoral Lipomatous Lesion


Ankita Giri1, Ravleen Nagi2, Manish Kumar3, Stuti Shukla1,  
1 Department of Oral Medicine and Radiology, New Horizon Dental College and Research Institute, Bilaspur, Chhattisgarh, India
2 Department of Oral Medicine and Radiology, Swami Devi Dayal Hospital and Dental College, Panchkula, Haryana, India
3 Dental Department, Chitwan Medical College, Bharatpur, Nepal

Correspondence Address:
Dr. Ravleen Nagi
Department of Oral Medicine and Radiology, Swami Devi Dayal Hospital and Dental College, Panchkula, Haryana
India

Abstract

Intraoral angiolipoma (AL) is a rare clinical and histological variant of lipoma consisting of predominantly proliferating vascular channels interspersed with mature fat cells. The exact etiopathogenesis of this neoplasm still has to be established, and it clinically presents intraorally as a well-circumscribed, asymptomatic, or tender submucosal nodular mass that requires complete surgical excision. This report describes a rare case of AL located in the upper lip of a 65-year-old male patient and also focuses on its clinical and histological characteristic features for accurate diagnosis and better management.



How to cite this article:
Giri A, Nagi R, Kumar M, Shukla S. Oral Soft-Tissue Angiolipoma: Rarest Intraoral Lipomatous Lesion.J Integr Health Sci 2020;8:91-94


How to cite this URL:
Giri A, Nagi R, Kumar M, Shukla S. Oral Soft-Tissue Angiolipoma: Rarest Intraoral Lipomatous Lesion. J Integr Health Sci [serial online] 2020 [cited 2021 May 5 ];8:91-94
Available from: https://www.jihs.in/text.asp?2020/8/2/91/291881


Full Text



 Introduction



Angiolipoma (AL), a variant of lipoma, is a benign soft-tissue tumor in which mature adipocytes are intermingled with proliferating small vascular channels. It occurs more commonly in the forearm, trunk, and extremities of young adults; around 20% of cases have been reported to arise in the head-and-neck region and very rarely, it involves the oral cavity.[1] This tumor was first described as a separate clinical entity by Bowen in 1912. Intraoral ALs have higher predilection for males, and usually present as well-defined, encapsulated, painless, soft-tissue masses more commonly on the buccal mucosa, followed by labial mucosa.[2],[3] To the best of our knowledge, very limited cases have described the clinicopathological characteristics of this rare entity, so this report describes an additional case of AL located in the upper lip in a 65-year-old male patient and also discusses its literature review.

 Case Report



A 65-year-old Indian male presented to the department of oral medicine and radiology, with a chief complaint of painless swelling in the upper lip, approximately present for 6–7 years [Figure 1] with slight increase in the size for the past 6 months. There was no history of associated trauma, or any relevant family and medical history. Personal history revealed that he had the habit of chewing tobacco 4–5 times a day for 40 years. Physical examination showed a good physique and a good nutritional status, with no abnormalities in the trunk and extremities. On head-and-neck examination, the patient had symmetrical facial configuration, and no abnormal sensation or motor palsy of the cheek, and cervical lymph node enlargement was observed.{Figure 1}

Intraoral examination revealed an asymptomatic reddish pink, dome-shaped nodular mass at the right side of the upper labial mucosa. On palpation, the mass was well circumscribed; 1.5 cm × 1.5 cm in diameter, soft to firm in consistency, nontender, nonfluctuant, and no bruit was present. It showed no fixity to the underlying tissues, and the overlying mucosa was normal with no remarkable changes in color or thickness [Figure 1]. Based on the patient's history and clinical examination, the mass was provisionally diagnosed as chronic mucocele, and lipoma, hemangioma, AL, and angiosarcoma were considered under differential diagnosis. Hematological parameters were within the normal limits. After obtaining patient's consent, surgical excision was performed under local anesthesia. As the mass was surrounded by a thin capsule and was nonadherent to the surrounding tissue, detachment was easy and was removed as a lump. The excised specimen revealed a solid, soft-tissue mass, 12 mm × 10 mm in size, with a smooth yellowish surface [Figure 2].{Figure 2}

Microscopic examination under low magnification showed numerous adipocytes separated by fibrocellular connective tissue composed of medium-to-large-sized blood vessels at the periphery, and surrounded by a fibrous capsule. Under higher magnification, adipocytes showed a thin rim of cytoplasm with peripherally placed nucleus. The fibrocellular connective tissue was made up of numerous bundles of collagen fibers along with fibroblasts and fibrocytes; the vascular channels or blood vessels were lined by endothelial cells and contained numerous red blood and white blood corpuscles [Figure 3]. Few chronic inflammatory cells, mainly lymphocytes, were also observed [Figure 4]. Overall histological features were diagnostic of AL. The patient was regularly followed up and no signs of recurrence were observed.{Figure 3}{Figure 4}

 Discussion



Lipomas are most common soft-tissue tumor of adipocytes, accounting for 0.1% to 5% of all benign neoplasms of the oral cavity. They have been reported to occur in the head-and-neck region in 15%–20% of cases; only 1%–4% of lipomas have been found to involve the oral cavity, an unusual site for the occurrence of lipoma.[1],[4] They usually occur in the areas of fat accumulation, predominantly buccal mucosa, followed by tongue, floor of the mouth, buccal vestibule, lip, palate, and gingiva. Lipomas clinically manifest as soft nodular painless swellings covered by an intact mucosa. Oral infiltrating lipomas are larger in size and present clinically as deep-seated, slow-growing, painless mass.[1],[5],[6] Oral AL is an extremely rare histological subtype of lipoma, which accounts for 5%–17% of all lipomas, and consists predominately mature fat cells separated by branching network of small blood vessels.[3],[7]

The etiopathogenesis of AL is still unclear, but some authors suggested it to be of hamartomatous origin. History of trauma and hormonal lipomatous differentiation also contribute to the occurrence of this rare condition.[3],[7],[8] Ida-Yonemochi et al. revealed that mast cells around blood vessels expressed high levels of vascular endothelial growth factor, that may be responsible for enhanced vascularity of this tumor.[8] Reported cases from the existing literature found AL to occur predominately in males of middle age, mostly on the buccal mucosa, followed by lip as asymptomatic nodular well-circumscribed mass; similar clinical findings were seen in our patient. They may be tender or painful on palpation, particularly during the initial growth period. It may also occur as diffuse intramuscular form that is usually tender, infiltrates surrounding deep soft tissues and adjacent muscles, and poses difficulty in complete surgical excision.[8],[9],[10]

Histologically, these tumors present as yellow nodules, consisting of mature fat cells separated by a branching network of small blood vessels. The vessels commonly contain fibrin thrombi, without evidence of necrosis due to the extensive collateral circulation.[9],[10] Due to the presence of vascular component, it is essential to exclude other vascular pathologies such as lipoma with prominent blood vessels, hemangiomas, lymphangiomas, angiosarcoma, and Kaposi sarcoma (KS). Lipomas predominantly consist of mature fat cells in comparison to AL that mainly consist of small proliferating blood vessels interspersed with adipocytes. Hemangioma exhibit characteristic pulsations and fluctuations on clinical examination. KS occurs as exophytic reddish purple nodules on skin, and intraorally palate is the most common site of occurrence. Moreover, KS has a high incidence of malignant transformation.[10],[11]

Limited cases of AL have been reported to occur in the upper lip. Gerard and Schultz[12] examined and treated soft, nontender, nonpulsatile mass measuring approximately 2 cm × 3 cm in size, in the upper left lip in a 65-year-old male patient, which was diagnosed as AL due to the presence of proliferating blood vessels mixed with an adipose tissue microscopically, and after 1-year follow–up, no recurrence of the lesion was observed. Another case of AL was reported by Sah et al.,[13] in which an asymptomatic, well-circumscribed, soft, movable, nontender, nonfluctuant, swelling about 2 cm × 2 cm in diameter was observed in the midline of the upper lip in a 9-year-old young girl. Histopathological features of adipose tissue separated by branched vascular network, led to the final diagnosis of AL and the patient reported no signs or symptoms of recurrence 6 months after surgery.

Most of the intraoral ALs are well managed by simple surgical excision, with no evidence of recurrence during follow-up visits. Silva-Junior et al.[14] reported two cases of noninfiltrative intraoral ALs that presented as painless, well-defined nodular mass on the buccal mucosa and were successfully managed by conservative surgical excision under local anesthesia. Similarly, in our patient, the mass was nonadherent to the underlying tissues and was easily removed by surgical excision, thus eliciting the benign nature of these lesions. Contrary to the above findings, Hemavathy et al. and Shahi et al. reported a case of intramandibular AL and infiltrative AL of cheek extending into the buccal space, respectively, which were explored surgically through intraoral approach under general anesthesia.[7],[15] Carbon dioxide laser therapy and interferon-α are proposed alternative treatments of this condition. Effective surgical resection with wide margins is recommended to reduce its recurrence, which may occur due to incomplete resection and infiltrative margins.[14],[15]

 Conclusion



This case report focused on the clinical and histological characteristic features of AL which is an extremely rare benign variant of lipoma, occurring in the oral cavity. It may represent hyperplasia of adipocytes or vascular channels, and complete surgical excision is the primary treatment of choice for both circumscribed and diffuse, infiltrating forms. Studies focusing on the effectiveness of alternative treatment modalities such as lasers and to establish the exact nature of these tumors are being encouraged for appropriate diagnosis and management of this rare clinical entity.

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 conflict of interest.

References

1de Visscher JG. Lipomas and fibrolipomas of the oral cavity. J Maxillofac Surg 1982;10:177-81.
2Hatziotis JC. Lipoma of the oral cavity. Oral Surg Oral Med Oral Pathol 1971;31:511-24.
3Ohnishi Y, Watanabe M, Fujii T, Yasui H, Kubo H, Kakudo K. Infiltrating angiolipoma of the lower lip: A case report and literature review. Oncol Lett 2015;9:833-6.
4Fregnani ER, Pires FR, Falzoni R, Lopes MA, Vargas PA. Lipomas of the oral cavity: Clinical findings, histological classification and proliferative activity of 46 cases. Int J Oral Maxillofac Surg 2003;32:49-53.
5Lucas RB. Tumors of adipose tissue. In: Pathology of Tumors of the Oral Tissues. 4th ed.. London, UK: Churchill-Livingstone; 1984. p. 176-9.
6Ayasaka N, Chino T Jr., Chino T, Antoh M, Kawakami T. Infiltrating lipoma of the mental region: Report of a case. Br J Oral Maxillofac Surg 1993;31:388-90.
7Hemavathy S, Roy S, Kiresur A. Intraosseous angiolipoma of the mandible. J Oral Maxillofac Pathol 2012;16:283-7.
8Ida-Yonemochi H, Swelam W, Saito C, Saku T. Angiolipoma of the buccal mucosa: A possible role of mast cell-derived VEGF in its enhanced vascularity. J Oral Pathol Med 2005;34:59-61.
9Epivatianos A, Markopoulos AK, Papanayotou P. Benign tumors of adipose tissue of the oral cavity: A clinicopathologic study of 13 cases. J Oral Maxillofac Surg 2000;58:1113-7.
10Howard WR, Helwig EB. Angiolipoma. Arch Dermatol 1960;82:924-31.
11Lin JJ, Lin F. Two entities in angiolipoma. A study of 459 cases of lipoma with review of literature on infiltrating angiolipoma. Cancer 1974;34:720-7.
12Gerard N, Schultz DA. Angiolipoma of the upper lip: Report of a case. J Oral Maxillofac Surg 2009;67:1340-1.
13Sah K, Kadam A, Sunita J, Chandra S. Non-infiltrating angiolipoma of the upper lip: A rare entity. J Oral Maxillofac Pathol 2012;16:103-6.
14Silva-Junior GO, Picciani BL, Costa RC, Barbosa SM, Silvares MG, Souza RB, et al. Oral soft-tissue angiolipoma: Report of two cases of rare oral lipomatous lesion with emphasis on morphological and immunohistochemical features. J Oral Sci 2013;55:85-8.
15Shahi AK, Ash H, Chatterji K, Singh R. Cellular infiltrative angiolipoma of cheek in an infant. Natl J Maxillofac Surg 2014;5:202-5.