Journal of Investigative and Clinical Dentistry (2010), 1, 47–49
CASE REPORT
Angiofibrolipoma of the oral cavity Apostolos Epivatianos1, Alexandros Kolokotronis1, Apostolos Matiakis2 & Athanasios Poulopoulos1 1 Department of Oral Medicine and Oral Pathology, Dental School, Aristotle University of Thessaloniki, Thessaloniki, Greece 2 Private Practice, Thessaloniki, Greece
Keywords CD-34, histological, immunohistological, lipoma oral angiofibrolipoma. Correspondence Assistant Prof. Athanasios Poulopoulos, Department of Oral Medicine and Oral Pathology, Dental School, Aristotle University of Thessaloniki, Thessaloniki 54124, Greece. Tel: +302310-999528 Fax: +302310-999455 Email: [email protected] Received 24 November 2009; accepted 1 February 2010.
Abstract We describe an unusual and rare case of a lipoma variant known as angiofibrolipoma. A literature search revealed only a few cases of angiofibrolipoma which were located in different anatomical sites; no case of angiofibrolipoma in the oral cavity has been previously reported. We present and discuss the histopatholological–immunohistological findings and features. Angiofibrolipoma histologically consists of mature adipocytes, blood vessels, and bundles of collagenous connective tissue with low to moderate cellularity. It is not encapsulated, but is histologically distinct from surrounding tissue. Positive immunohistological staining with CD-34 presented a large number of blood vessels of different sizes located between collagen fibers and adipocytes. The treatment of choice for a patient with angiofibrolipoma is surgical excision, which is expected to be curative.
doi: 10.1111/j.2041-1626.2010.00003.x
Introduction Lipomas are benign mesenchymal neoplasms composed of mature adipocytes, usually encapsulated. Although they are the most common soft tissue tumor, only 1–4% of cases occur in the oral cavity.1 Based on their histopathological features, lipomas can be classified in different variants.2 A rare histological variant, angiofibrolipoma, has been described in the English literature.3 A literature search revealed only seven references of angiofibrolipoma,3–9 most of which are not English-language publications. All these cases of angiofibrolipoma were located in different anatomical sites, and no case in the oral cavity has been previously reported. Therefore, in the present study, we present a case of angiofibrolipoma affecting the alveolar mucosa of the oral cavity. Materials and methods A 42-year-old man was referred by his dentist in April 2008 to the Department of Oral Medicine and Oral Pathology at the dental school of the University of Thessaloniki, Thessaloniki, Greece for evaluation of asymptomatic swelling in the alveolar mucosa adjacent to the ª 2010 Blackwell Publishing Asia Pty Ltd
left upper canine and first left upper premolar. The swelling was noticed 2 weeks earlier. The medical and family histories of the patient were unremarkable. Clinical examination showed a firm, sessile, well-circumscribed swelling with a diameter of 0.8 cm, covered by normal mucosa (Figure 1a). No pathological radiographic findings were observed, and the teeth adjacent to the swelling were unaffected. Under local anesthesia, excisional biopsy was carried out with clear margins of healthy tissue. The wound was then closed, and the healing was uneventful. One year of follow up showed no evidence of recurrence. The patient gave informed consent, and the whole study was performed according to the Declaration of Helsinski. Results An histological examination of the lesion showed it to be composed of mature adipocytes, collagen fibers, and blood vessels of different sizes. Some blood vessels contained fibrin thrombi, whereas many small capillaries were compressed by collagen fibers and adipocytes (Figure 1b). The lesion was not encapsulated, but was histologically distinct from surrounding connective tissue. Mitotic figures and atypia were not observed. Additional sections 47
Oral angiofibrolipoma
A. Epivatianos et al.
(a)
(b)
Figure 1. (a) Clinical appearance of angiofibrolipoma presenting as asymptomatic swelling in the alveolar mucosa. (b) High-power view showing the presence of thin capillaries (arrows), collagen fibers, adipocytes, and fibrin thrombi in a blood vessel (arrowhead). (Hematoxylin–eosin. Original magnification ·100.)
were stained immunohistochemically with CD-34 using the streptavidin–biotin–peroxidase method. CD-34 staining showed the presence of a large number of capillaries. An histological diagnosis of angiofibrolipoma was made based on histological and immunohistochemical findings. Discussion Angiofibrolipoma histologically consists of mature adipocytes, blood vessels, and bundles of collagenous connec-
References 1 Bande´ca MC, de Pa´dua JM, Nadalin MR, Ozo´rio JE, Silva-Sousa YT, da Cruz Perez DE. Oral soft tissue lipomas: a case series. J Can Dent Assoc 2007; 73: 431–4. 2 Furlong MA, Fanburg-Smith JC, Childers EL. Lipoma of the oral and
48
tive tissue with low to moderate cellularity. It is not encapsulated, but is histologically distinct from surrounding tissue.3,8,10 Furthermore, because angiofibrolipoma shares the histological features of angiolipoma, fibrin thrombi could be present in its blood vessels. Our case fulfills the above-mentioned histological criteria3,8,10 for the diagnosis of angiofibrolipoma, and to our knowledge, is the first report of angiofibrolipoma in the oral cavity presented in the literature. The differential diagnosis of angiofibrolipomas from the neoplasms of adipose tissue is based strictly on the histological findings, and especially on the presence or absence of their specific tissue components. Angiofibrolipoma, in addition to adipose and vascular tissue, also contains bands of collagenous connective tissue that are absent in angiolipomas. Fibrolipomas are not characterized by numerous blood vessels, which is a characteristic feature of angiofibrolipomas. Positive staining of Masson’s trichrome method and negative immunohistochemical staining for smooth muscle actin allow the differentiation of angiofibrolipoma from angiomyolipoma. The presence of fibrin thrombi in blood vessels and the lack of gaping vessels differentiate angiofibrolipoma from intramuscular hemangioma.2 The most important clinical implication is that angiofibrolipoma must be distinguished early from liposarcoma, for which the diagnosis requires a deep incisional biopsy from the lesion’s center, and a sufficiently large specimen to be removed so that the pathologist can study enough representative tissue to evaluate the differentiation of the lesion. Multipotential stellate precursor blast cells of mesodermal origin can differentiate so as to produce circulatory, fat, and fibrous connective tissues. Therefore, tumors consisting of these tissues, as angiofibrolipoma, can arise in any body structure with a mesodermal origin.3 Ligget et al.10 suggested that angiofibrolipomas are hamartomas (polyclonal), as opposed to true neoplasms (monoclonal). According to English-language studies,3,8,10 the treatment of choice for patients with angiofibrolipoma is surgical excision, which is expected to be curative, and recurrence is rare.
maxillofacial region: site and subclassification of 125 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004; 98: 441–50. 3 Kershisnik W, McCarthy DJ, O’Donnel E. Angiofibrolipoma. A histologic variant of the lipoma. J Am Podiatr Med Assoc 1986; 76: 67–70.
4 Toth C. Kidney angiofibrolipoma. Z Urol Nephrol 1975; 68: 279– 82. 5 Ogorodnikova LS, Zubkova TV, Tiurbeeva ML. Benign non-epithelial tumors of the stomach. Arkh Patol 1979; 416: 17–20. 6 Krausen C, Becker K, Hamann KF. Angiofibrolipoma of the tonsil.
ª 2010 Blackwell Publishing Asia Pty Ltd
A. Epivatianos et al.
Laryngol Rhinol Otol (Stuttg) 1986; 65: 355–6. 7 Pazarola P, Bosso P, Ercolani S. Mammography and echography in male breast pathology. Radiol Med (Torino) 1992; 84: 32–5.
ª 2010 Blackwell Publishing Asia Pty Ltd
Oral angiofibrolipoma
8 Jacob A, Kneile J, Welling B. Angiofibrolipoma of the ear canal. Laryngoscope 2005; 115: 1461–2. 9 Novozhilov VN, Dolidze UR, Degterev DB, Il’ina VA, Bakhtybaev DO. Angiofibrolipoma of the trans-
verse colon. Vestn Khir Im I I Grek 2006; 165: 102–3. 10 Ligget AD, Frazier KS, Styer EL. Angiolipomatous tumors in dogs and a cat. Vet Pathol 2002; 39: 286–9.
49
CASE REPORT
Angiofibrolipoma of the oral cavity Apostolos Epivatianos1, Alexandros Kolokotronis1, Apostolos Matiakis2 & Athanasios Poulopoulos1 1 Department of Oral Medicine and Oral Pathology, Dental School, Aristotle University of Thessaloniki, Thessaloniki, Greece 2 Private Practice, Thessaloniki, Greece
Keywords CD-34, histological, immunohistological, lipoma oral angiofibrolipoma. Correspondence Assistant Prof. Athanasios Poulopoulos, Department of Oral Medicine and Oral Pathology, Dental School, Aristotle University of Thessaloniki, Thessaloniki 54124, Greece. Tel: +302310-999528 Fax: +302310-999455 Email: [email protected] Received 24 November 2009; accepted 1 February 2010.
Abstract We describe an unusual and rare case of a lipoma variant known as angiofibrolipoma. A literature search revealed only a few cases of angiofibrolipoma which were located in different anatomical sites; no case of angiofibrolipoma in the oral cavity has been previously reported. We present and discuss the histopatholological–immunohistological findings and features. Angiofibrolipoma histologically consists of mature adipocytes, blood vessels, and bundles of collagenous connective tissue with low to moderate cellularity. It is not encapsulated, but is histologically distinct from surrounding tissue. Positive immunohistological staining with CD-34 presented a large number of blood vessels of different sizes located between collagen fibers and adipocytes. The treatment of choice for a patient with angiofibrolipoma is surgical excision, which is expected to be curative.
doi: 10.1111/j.2041-1626.2010.00003.x
Introduction Lipomas are benign mesenchymal neoplasms composed of mature adipocytes, usually encapsulated. Although they are the most common soft tissue tumor, only 1–4% of cases occur in the oral cavity.1 Based on their histopathological features, lipomas can be classified in different variants.2 A rare histological variant, angiofibrolipoma, has been described in the English literature.3 A literature search revealed only seven references of angiofibrolipoma,3–9 most of which are not English-language publications. All these cases of angiofibrolipoma were located in different anatomical sites, and no case in the oral cavity has been previously reported. Therefore, in the present study, we present a case of angiofibrolipoma affecting the alveolar mucosa of the oral cavity. Materials and methods A 42-year-old man was referred by his dentist in April 2008 to the Department of Oral Medicine and Oral Pathology at the dental school of the University of Thessaloniki, Thessaloniki, Greece for evaluation of asymptomatic swelling in the alveolar mucosa adjacent to the ª 2010 Blackwell Publishing Asia Pty Ltd
left upper canine and first left upper premolar. The swelling was noticed 2 weeks earlier. The medical and family histories of the patient were unremarkable. Clinical examination showed a firm, sessile, well-circumscribed swelling with a diameter of 0.8 cm, covered by normal mucosa (Figure 1a). No pathological radiographic findings were observed, and the teeth adjacent to the swelling were unaffected. Under local anesthesia, excisional biopsy was carried out with clear margins of healthy tissue. The wound was then closed, and the healing was uneventful. One year of follow up showed no evidence of recurrence. The patient gave informed consent, and the whole study was performed according to the Declaration of Helsinski. Results An histological examination of the lesion showed it to be composed of mature adipocytes, collagen fibers, and blood vessels of different sizes. Some blood vessels contained fibrin thrombi, whereas many small capillaries were compressed by collagen fibers and adipocytes (Figure 1b). The lesion was not encapsulated, but was histologically distinct from surrounding connective tissue. Mitotic figures and atypia were not observed. Additional sections 47
Oral angiofibrolipoma
A. Epivatianos et al.
(a)
(b)
Figure 1. (a) Clinical appearance of angiofibrolipoma presenting as asymptomatic swelling in the alveolar mucosa. (b) High-power view showing the presence of thin capillaries (arrows), collagen fibers, adipocytes, and fibrin thrombi in a blood vessel (arrowhead). (Hematoxylin–eosin. Original magnification ·100.)
were stained immunohistochemically with CD-34 using the streptavidin–biotin–peroxidase method. CD-34 staining showed the presence of a large number of capillaries. An histological diagnosis of angiofibrolipoma was made based on histological and immunohistochemical findings. Discussion Angiofibrolipoma histologically consists of mature adipocytes, blood vessels, and bundles of collagenous connec-
References 1 Bande´ca MC, de Pa´dua JM, Nadalin MR, Ozo´rio JE, Silva-Sousa YT, da Cruz Perez DE. Oral soft tissue lipomas: a case series. J Can Dent Assoc 2007; 73: 431–4. 2 Furlong MA, Fanburg-Smith JC, Childers EL. Lipoma of the oral and
48
tive tissue with low to moderate cellularity. It is not encapsulated, but is histologically distinct from surrounding tissue.3,8,10 Furthermore, because angiofibrolipoma shares the histological features of angiolipoma, fibrin thrombi could be present in its blood vessels. Our case fulfills the above-mentioned histological criteria3,8,10 for the diagnosis of angiofibrolipoma, and to our knowledge, is the first report of angiofibrolipoma in the oral cavity presented in the literature. The differential diagnosis of angiofibrolipomas from the neoplasms of adipose tissue is based strictly on the histological findings, and especially on the presence or absence of their specific tissue components. Angiofibrolipoma, in addition to adipose and vascular tissue, also contains bands of collagenous connective tissue that are absent in angiolipomas. Fibrolipomas are not characterized by numerous blood vessels, which is a characteristic feature of angiofibrolipomas. Positive staining of Masson’s trichrome method and negative immunohistochemical staining for smooth muscle actin allow the differentiation of angiofibrolipoma from angiomyolipoma. The presence of fibrin thrombi in blood vessels and the lack of gaping vessels differentiate angiofibrolipoma from intramuscular hemangioma.2 The most important clinical implication is that angiofibrolipoma must be distinguished early from liposarcoma, for which the diagnosis requires a deep incisional biopsy from the lesion’s center, and a sufficiently large specimen to be removed so that the pathologist can study enough representative tissue to evaluate the differentiation of the lesion. Multipotential stellate precursor blast cells of mesodermal origin can differentiate so as to produce circulatory, fat, and fibrous connective tissues. Therefore, tumors consisting of these tissues, as angiofibrolipoma, can arise in any body structure with a mesodermal origin.3 Ligget et al.10 suggested that angiofibrolipomas are hamartomas (polyclonal), as opposed to true neoplasms (monoclonal). According to English-language studies,3,8,10 the treatment of choice for patients with angiofibrolipoma is surgical excision, which is expected to be curative, and recurrence is rare.
maxillofacial region: site and subclassification of 125 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004; 98: 441–50. 3 Kershisnik W, McCarthy DJ, O’Donnel E. Angiofibrolipoma. A histologic variant of the lipoma. J Am Podiatr Med Assoc 1986; 76: 67–70.
4 Toth C. Kidney angiofibrolipoma. Z Urol Nephrol 1975; 68: 279– 82. 5 Ogorodnikova LS, Zubkova TV, Tiurbeeva ML. Benign non-epithelial tumors of the stomach. Arkh Patol 1979; 416: 17–20. 6 Krausen C, Becker K, Hamann KF. Angiofibrolipoma of the tonsil.
ª 2010 Blackwell Publishing Asia Pty Ltd
A. Epivatianos et al.
Laryngol Rhinol Otol (Stuttg) 1986; 65: 355–6. 7 Pazarola P, Bosso P, Ercolani S. Mammography and echography in male breast pathology. Radiol Med (Torino) 1992; 84: 32–5.
ª 2010 Blackwell Publishing Asia Pty Ltd
Oral angiofibrolipoma
8 Jacob A, Kneile J, Welling B. Angiofibrolipoma of the ear canal. Laryngoscope 2005; 115: 1461–2. 9 Novozhilov VN, Dolidze UR, Degterev DB, Il’ina VA, Bakhtybaev DO. Angiofibrolipoma of the trans-
verse colon. Vestn Khir Im I I Grek 2006; 165: 102–3. 10 Ligget AD, Frazier KS, Styer EL. Angiolipomatous tumors in dogs and a cat. Vet Pathol 2002; 39: 286–9.
49
