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Journal of Digestive Diseases 2014; 15; 154–157
doi: 10.1111/1751-2980.12121
Case report
Angiolipoma of the colon: A case report and literature review Ying HANG & Chang Qing ZHU Department of Emergency, Ren Ji Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
INTRODUCTION Angiolipoma is generally categorized to be a benign tumor that usually occurs in subcutaneous tissues. Histologically, the tumor is composed of adipose tissues and proliferative vascular components.1–3 The final diagnosis of angiolipoma depends on histopathological evaluation. Although endoscopic and radiological techniques such as computed tomography (CT) are helpful,4–7 preoperative diagnosis of the tumor is still difficult. Angiolipoma in the colon is extremely rare. Here, we report a case of angiolipoma of the colon and perform a literature review. CASE REPORT A 48-year-old woman was admitted to our hospital in November 2011, complaining of altered bowel habit for more than two years that had been worsening for one month, in the absence of abdominal pain, melena or hematochezia. The patient’s physical examination was unremarkable. During her hospitalization, routine hematological and biochemical results were found to be within the normal range. Tumor markers including α-fetoprotein, carcinoembryonic antigen, carbohydrate antigen 19-9, cancer antigen 15-3 and cancer antigen 125 were negative. Colonoscopy showed a cyst-like mass with congestive Correspondence to: Chang Qing ZHU, Department of Emergency, Ren Ji Hospital, School of Medicine, Shanghai Jiao Tong University, 160 Pujian Road, Shanghai 200127, China. Email: [email protected] Conflict of interest: None. © 2013 Chinese Medical Association Shanghai Branch, Chinese Society of Gastroenterology, Renji Hospital Affiliated to Shanghai Jiaotong University School of Medicine and Wiley Publishing Asia Pty Ltd
154
smooth surface protruding into the enteric cavity (Fig. 1) at approximately 50 cm from the anus. In light of a high risk for hemorrhage, endoscopic biopsy of the lesion was not performed. Therefore, preoperative pathological diagnosis could not be obtained. Enhanced CT (Fig. 2) revealed a mixed low and isodense mass with fatty attenuation and slight edge enhancement that was close to the splenic flexure of the colon. Given the large size of the mass, which occupied almost the entire enteric cavity as well as the potential high risk of hemorrhage, surgical exploration was considered as the best choice for this patient. Partial intestinal resection was successfully performed at the splenic flexure of the colon and the mass was completely removed. Macroscopically, the mass (5 cm × 3 cm × 4 cm) was pedicled and cyst-like with a yellow fatty base and a red congestive surface at the top (Fig. 3). Pathohistological diagnosis based on frozen section suggested angiolipoma. Due to the benign characteristics of angiolipoma colocolic anastomosis was subsequently performed and the final diagnosis of colonic angiolipoma was confirmed by postoperative pathology (Fig. 4). Immunohistochemistry analysis showed negative smooth muscle actin and melanoma-specific antibody tests. The patient was discharged on the eighth day postoperation without complications. During her outpatient follow-up, she had no abdominal pain or changes of bowel habit. Whole blood analysis, tumor markers and the routine stool examination plus occult blood test were repeatedly normal. The suggestion of repeated colonoscopy and CT scan were turned aside by the patient. No evidence of tumor recurrence was detected after oneyear follow-up.
Journal of Digestive Diseases 2014; 15; 154–157
Figure 1. The colonoscopy shows a pedicled congestive mass with smooth surface at approximately 50 cm from the anal verge.
Figure 2. Enhanced computed tomography shows a mixed low and isodense mass (white arrow) with fatty attenuation and slight edge enhancement close to the splenic flexure of the colon.
DISCUSSION Angiolipoma was first reported in 1912 by Bowen.8 In 1960 Howard and Helwig9 further described clinicopathological differences between angiolipomas and lipomas. Angiolipoma usually presents as an encapsulated tumor that is yellow below and has a variable red surface along the periphery. Histologically, angiolipomas are composed of mature adipose
Angiolipoma of the colon
155
Figure 3. The resected mass is pedicled and cyst-like, with yellow adipose tissue below and red vascular tissue at the top.
Figure 4. Pathological examination demonstrating that the tumor is composed of adipose cells and proliferative blood vessels (HE stain, ×100).
tissues and proliferative vascular components.1-3 Fibrin thrombi in the capillaries are characteristic. Angiolipomas can be classified as predominantly lipomatous or predominantly angiomatous by the composition of adipose and vascular tissues.9 The lesions generally occur in the subcutaneous adipose tissue.1,10 Patients with such tumors usually present with tender and/or pain as well as multiple masses on the arms and trunk in young adults. Gastrointestinal angiolipomas are rare and almost all occur as a solitary lesion. So far, only few literatures have identified
© 2013 Chinese Medical Association Shanghai Branch, Chinese Society of Gastroenterology, Renji Hospital Affiliated to Shanghai Jiaotong University School of Medicine and Wiley Publishing Asia Pty Ltd
156
Y Hang and CQ Zhu
the location of gastrointestinal angiolipomas, including the esophagus,11 stomach,3,6,12,13 duodenum,4,14 small intestine,15–20 colon5,7,21–24 and rectum25,26. Angiolipomas can be divided into noninfiltrating and infiltrating types.27 Most gastrointestinal angiolipomas are asymptomatic. Some patients may present with intestinal obstruction, gastrointestinal bleeding or intussusception. Hemorrhage in angiolipomas is generally attributed to their prominent proliferative capillary components. Angiolipoma, especially the predominantly angiomatous type, often presents as a low-density mass with variable edge enhancement on enhanced CT, which correlates well with its histological constitution by adipose and vascular structure.17 Preoperative diagnosis of gastrointestinal angiolipoma is extremely difficult due to its lack of characteristic clinical presentations and imaging findings as well as the difficulty of endoscopic biopsy. The final diagnosis of gastrointestinal angiolipomas is usually confirmed by surgical pathology. The differential diagnosis should consider colonic carcinoma, lipoma, well-differentiated liposarcoma and angiomyolipoma. So far, the optimal treatment for angiolipoma of the colon remained controversial. Surgical resection remains the recommended treatment, especially for broad-based or obstructive lesions, due to the increased risks of perforation and bleeding compared with endoscopic polypectomy.16 Intraoperative frozen section may provide an accurate diagnosis to guide surgical resection. Because angiolipomas are benign tumors, a minimally invasive laparoscopic resection may also be worth considering.21 Endoscopic polypectomy is still acceptable for small pedunculated polyps. A preinjection of epinephrine and the use of nylon loop or metal hemostatic clips before polypectomy may effectively decrease intraoperative bleeding.5,14 The prognosis of angiolipoma is excellent if the tumor can be removed completely, while an inadequate resection could result in a high recurrence rate of the tumor.15,26 REFERENCES 1 Lin 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. 2 Punia RS, Jain P, Amanjit, Mohan H, Singh R. Subcutaneous angiolipomas: a clinicopathological study of 12 cases. Indian J Pathol Microbiol 2005; 48: 197–8. 3 McGregor DH, Kerley SW, McGregor MS. Case report: gastric angiolipoma with chronic hemorrhage and severe anemia. Am J Med Sci 1993; 305: 229–35.
Journal of Digestive Diseases 2014; 15; 154–157 4 Jung IS, Jang JY, Ryu CB et al. Angiolipoma of the duodenum diagnosed after endoscopic resection. Endoscopy 2004; 36: 375. 5 Okuyama T, Yoshida M, Watanabe M, Kinoshita Y, Harada Y. Angiolipoma of the colon diagnosed after endoscopic resection. Gastrointest Endosc 2002; 55: 748–50. 6 Ferrozzi F, Tognini G, Bova D, Pavone P. Lipomatous tumors of the stomach: CT findings and differential diagnosis. J Comput Assist Tomogr 2000; 24: 854–8. 7 Chen YY, Soon MS. Preoperative diagnosis of colonic angiolipoma: a case report. World J Gastroenterol 2005; 11: 5087–9. 8 Bowen JT. Multiple subcutaneous hemangiomas, together with multiple lipomas, occurring in enormous numbers in an otherwise healthy, muscular subject. Am J Med Sci 1912; 144: 189–92. 9 Howard WR, Helwig EB. Angiolipoma. Arch Dermatol 1960; 82: 924–31. 10 Dixon AY, McGregor DH, Lee SH. Angiolipomas: an ultrastructural and clinicopathological study. Hum Pathol 1981; 12: 739–47. 11 Jensen EH, Klapman JB, Kelley ST. Angiolipoma of the esophagus: a rare clinical dilemma. Dis Esophagus 2006; 19: 203–7. 12 DeRidder PH, Levine AJ, Katta JJ, Catto JA. Angiolipoma of the stomach as a cause of chronic upper gastrointestinal bleeding. Surg Endosc 1989; 3: 106–8. 13 Hunt J, Tindal D. Solitary gastric Peutz-Jeghers polyp and angiolipoma presenting as acute haemorrhage. Aust N Z J Surg 1996; 66: 713–5. 14 Mohl W, Fischinger J, Moser C, Remberger K, Zeuzem S, Stallmach A. Duodenal angiolipoma – endoscopic diagnosis and therapy. Z Gastroenterol 2004; 42: 1381–3. 15 Della Volpe N, Bianco L, Bonuso C, Annecchiarico M, Di Silverio P, Caiazza A. Rare ileal localisation of angiolipoma presenting as chronic haemorrhage and severe anaemia: a case report. J Med Case Rep 2008; 2: 129. 16 Aminian A, Noaparast M, Mirsharifi R et al. Ileal intussusception secondary to both lipoma and angiolipoma: a case report. Cases J 2009; 2: 7099. 17 Ferrozzi F, Rubino S, Uccelli M. Ileal angiolipoma in a patient with multisystemic manifestations of tuberous sclerosis: diagnosis with computerized tomography. Radiol Med 1998; 96: 403–5 (in Italian). 18 Kaneko T, Karasawa Y, Inada H et al. An adult case of intussusception due to inverted Meckel’s diverticulum accompanied by angiolipoma. Nihon Shokakibyo Gakkai Zasshi 1996; 93: 260–5 (in Japanese). 19 Manner M, Scholz E, Wehrmann M, Stickel W. Invagination caused by angiolipoma of the small intestine – a rare cause of occult gastrointestinal hemorrhage. Chirurg 2001; 72: 305–7 (in German). 20 Kwak HS, Kim CS, Lee JM. Small intestinal angiolipoma: MR imaging appearance. Abdom Imaging 2003; 28: 515–7. 21 Kato K, Matsuda M, Onodera K, Sakata H, Kobayashi T, Kasai S. Angiolipoma of the colon with right lower quadrant abdominal pain. Dig Surg 1999; 16: 441–4. 22 Vandamme J. Angiolipoma of the colon. Acta Gastroenterol Belg 1964; 27: 750–7 (in French). 23 Maesawa C, Tamura G, Sawada H, Kamioki S, Nakajima Y, Satodate R. Angiomyolipoma arising in the colon. Am J Gastroenterol 1996; 91: 1852–4. 24 Molinares B, Goldstein A, Varela GJ, Mesa S. Colonic angiolipoma – a rare finding in the gastrointestinal tract.
© 2013 Chinese Medical Association Shanghai Branch, Chinese Society of Gastroenterology, Renji Hospital Affiliated to Shanghai Jiaotong University School of Medicine and Wiley Publishing Asia Pty Ltd
Journal of Digestive Diseases 2014; 15; 154–157 Case report and review of literature. J Radiol Case Rep 2012; 6: 23–8. 25 Kacar S, Kuran S, Temucin T, Odemis B, Karadeniz N, Sasmaz N. Rectal angiolipoma: a case report and review of literature. World J Gastroenterol 2007; 13: 1460–5.
Angiolipoma of the colon
157
26 Ishizuka M, Nagata H, Takagi K, Horie T, Abe A, Kubota K. Rectal angiolipoma diagnosed after surgical resection: a case report. World J Gastroenterol 2007; 13: 467–9. 27 Dionne GP, Seemayer TA. Infiltrating lipomas and angiolipomas revisited. Cancer 1974; 33: 732–8.
© 2013 Chinese Medical Association Shanghai Branch, Chinese Society of Gastroenterology, Renji Hospital Affiliated to Shanghai Jiaotong University School of Medicine and Wiley Publishing Asia Pty Ltd
Journal of Digestive Diseases 2014; 15; 154–157
doi: 10.1111/1751-2980.12121
Case report
Angiolipoma of the colon: A case report and literature review Ying HANG & Chang Qing ZHU Department of Emergency, Ren Ji Hospital, School of Medicine, Shanghai Jiao Tong University, Shanghai, China
INTRODUCTION Angiolipoma is generally categorized to be a benign tumor that usually occurs in subcutaneous tissues. Histologically, the tumor is composed of adipose tissues and proliferative vascular components.1–3 The final diagnosis of angiolipoma depends on histopathological evaluation. Although endoscopic and radiological techniques such as computed tomography (CT) are helpful,4–7 preoperative diagnosis of the tumor is still difficult. Angiolipoma in the colon is extremely rare. Here, we report a case of angiolipoma of the colon and perform a literature review. CASE REPORT A 48-year-old woman was admitted to our hospital in November 2011, complaining of altered bowel habit for more than two years that had been worsening for one month, in the absence of abdominal pain, melena or hematochezia. The patient’s physical examination was unremarkable. During her hospitalization, routine hematological and biochemical results were found to be within the normal range. Tumor markers including α-fetoprotein, carcinoembryonic antigen, carbohydrate antigen 19-9, cancer antigen 15-3 and cancer antigen 125 were negative. Colonoscopy showed a cyst-like mass with congestive Correspondence to: Chang Qing ZHU, Department of Emergency, Ren Ji Hospital, School of Medicine, Shanghai Jiao Tong University, 160 Pujian Road, Shanghai 200127, China. Email: [email protected] Conflict of interest: None. © 2013 Chinese Medical Association Shanghai Branch, Chinese Society of Gastroenterology, Renji Hospital Affiliated to Shanghai Jiaotong University School of Medicine and Wiley Publishing Asia Pty Ltd
154
smooth surface protruding into the enteric cavity (Fig. 1) at approximately 50 cm from the anus. In light of a high risk for hemorrhage, endoscopic biopsy of the lesion was not performed. Therefore, preoperative pathological diagnosis could not be obtained. Enhanced CT (Fig. 2) revealed a mixed low and isodense mass with fatty attenuation and slight edge enhancement that was close to the splenic flexure of the colon. Given the large size of the mass, which occupied almost the entire enteric cavity as well as the potential high risk of hemorrhage, surgical exploration was considered as the best choice for this patient. Partial intestinal resection was successfully performed at the splenic flexure of the colon and the mass was completely removed. Macroscopically, the mass (5 cm × 3 cm × 4 cm) was pedicled and cyst-like with a yellow fatty base and a red congestive surface at the top (Fig. 3). Pathohistological diagnosis based on frozen section suggested angiolipoma. Due to the benign characteristics of angiolipoma colocolic anastomosis was subsequently performed and the final diagnosis of colonic angiolipoma was confirmed by postoperative pathology (Fig. 4). Immunohistochemistry analysis showed negative smooth muscle actin and melanoma-specific antibody tests. The patient was discharged on the eighth day postoperation without complications. During her outpatient follow-up, she had no abdominal pain or changes of bowel habit. Whole blood analysis, tumor markers and the routine stool examination plus occult blood test were repeatedly normal. The suggestion of repeated colonoscopy and CT scan were turned aside by the patient. No evidence of tumor recurrence was detected after oneyear follow-up.
Journal of Digestive Diseases 2014; 15; 154–157
Figure 1. The colonoscopy shows a pedicled congestive mass with smooth surface at approximately 50 cm from the anal verge.
Figure 2. Enhanced computed tomography shows a mixed low and isodense mass (white arrow) with fatty attenuation and slight edge enhancement close to the splenic flexure of the colon.
DISCUSSION Angiolipoma was first reported in 1912 by Bowen.8 In 1960 Howard and Helwig9 further described clinicopathological differences between angiolipomas and lipomas. Angiolipoma usually presents as an encapsulated tumor that is yellow below and has a variable red surface along the periphery. Histologically, angiolipomas are composed of mature adipose
Angiolipoma of the colon
155
Figure 3. The resected mass is pedicled and cyst-like, with yellow adipose tissue below and red vascular tissue at the top.
Figure 4. Pathological examination demonstrating that the tumor is composed of adipose cells and proliferative blood vessels (HE stain, ×100).
tissues and proliferative vascular components.1-3 Fibrin thrombi in the capillaries are characteristic. Angiolipomas can be classified as predominantly lipomatous or predominantly angiomatous by the composition of adipose and vascular tissues.9 The lesions generally occur in the subcutaneous adipose tissue.1,10 Patients with such tumors usually present with tender and/or pain as well as multiple masses on the arms and trunk in young adults. Gastrointestinal angiolipomas are rare and almost all occur as a solitary lesion. So far, only few literatures have identified
© 2013 Chinese Medical Association Shanghai Branch, Chinese Society of Gastroenterology, Renji Hospital Affiliated to Shanghai Jiaotong University School of Medicine and Wiley Publishing Asia Pty Ltd
156
Y Hang and CQ Zhu
the location of gastrointestinal angiolipomas, including the esophagus,11 stomach,3,6,12,13 duodenum,4,14 small intestine,15–20 colon5,7,21–24 and rectum25,26. Angiolipomas can be divided into noninfiltrating and infiltrating types.27 Most gastrointestinal angiolipomas are asymptomatic. Some patients may present with intestinal obstruction, gastrointestinal bleeding or intussusception. Hemorrhage in angiolipomas is generally attributed to their prominent proliferative capillary components. Angiolipoma, especially the predominantly angiomatous type, often presents as a low-density mass with variable edge enhancement on enhanced CT, which correlates well with its histological constitution by adipose and vascular structure.17 Preoperative diagnosis of gastrointestinal angiolipoma is extremely difficult due to its lack of characteristic clinical presentations and imaging findings as well as the difficulty of endoscopic biopsy. The final diagnosis of gastrointestinal angiolipomas is usually confirmed by surgical pathology. The differential diagnosis should consider colonic carcinoma, lipoma, well-differentiated liposarcoma and angiomyolipoma. So far, the optimal treatment for angiolipoma of the colon remained controversial. Surgical resection remains the recommended treatment, especially for broad-based or obstructive lesions, due to the increased risks of perforation and bleeding compared with endoscopic polypectomy.16 Intraoperative frozen section may provide an accurate diagnosis to guide surgical resection. Because angiolipomas are benign tumors, a minimally invasive laparoscopic resection may also be worth considering.21 Endoscopic polypectomy is still acceptable for small pedunculated polyps. A preinjection of epinephrine and the use of nylon loop or metal hemostatic clips before polypectomy may effectively decrease intraoperative bleeding.5,14 The prognosis of angiolipoma is excellent if the tumor can be removed completely, while an inadequate resection could result in a high recurrence rate of the tumor.15,26 REFERENCES 1 Lin 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. 2 Punia RS, Jain P, Amanjit, Mohan H, Singh R. Subcutaneous angiolipomas: a clinicopathological study of 12 cases. Indian J Pathol Microbiol 2005; 48: 197–8. 3 McGregor DH, Kerley SW, McGregor MS. Case report: gastric angiolipoma with chronic hemorrhage and severe anemia. Am J Med Sci 1993; 305: 229–35.
Journal of Digestive Diseases 2014; 15; 154–157 4 Jung IS, Jang JY, Ryu CB et al. Angiolipoma of the duodenum diagnosed after endoscopic resection. Endoscopy 2004; 36: 375. 5 Okuyama T, Yoshida M, Watanabe M, Kinoshita Y, Harada Y. Angiolipoma of the colon diagnosed after endoscopic resection. Gastrointest Endosc 2002; 55: 748–50. 6 Ferrozzi F, Tognini G, Bova D, Pavone P. Lipomatous tumors of the stomach: CT findings and differential diagnosis. J Comput Assist Tomogr 2000; 24: 854–8. 7 Chen YY, Soon MS. Preoperative diagnosis of colonic angiolipoma: a case report. World J Gastroenterol 2005; 11: 5087–9. 8 Bowen JT. Multiple subcutaneous hemangiomas, together with multiple lipomas, occurring in enormous numbers in an otherwise healthy, muscular subject. Am J Med Sci 1912; 144: 189–92. 9 Howard WR, Helwig EB. Angiolipoma. Arch Dermatol 1960; 82: 924–31. 10 Dixon AY, McGregor DH, Lee SH. Angiolipomas: an ultrastructural and clinicopathological study. Hum Pathol 1981; 12: 739–47. 11 Jensen EH, Klapman JB, Kelley ST. Angiolipoma of the esophagus: a rare clinical dilemma. Dis Esophagus 2006; 19: 203–7. 12 DeRidder PH, Levine AJ, Katta JJ, Catto JA. Angiolipoma of the stomach as a cause of chronic upper gastrointestinal bleeding. Surg Endosc 1989; 3: 106–8. 13 Hunt J, Tindal D. Solitary gastric Peutz-Jeghers polyp and angiolipoma presenting as acute haemorrhage. Aust N Z J Surg 1996; 66: 713–5. 14 Mohl W, Fischinger J, Moser C, Remberger K, Zeuzem S, Stallmach A. Duodenal angiolipoma – endoscopic diagnosis and therapy. Z Gastroenterol 2004; 42: 1381–3. 15 Della Volpe N, Bianco L, Bonuso C, Annecchiarico M, Di Silverio P, Caiazza A. Rare ileal localisation of angiolipoma presenting as chronic haemorrhage and severe anaemia: a case report. J Med Case Rep 2008; 2: 129. 16 Aminian A, Noaparast M, Mirsharifi R et al. Ileal intussusception secondary to both lipoma and angiolipoma: a case report. Cases J 2009; 2: 7099. 17 Ferrozzi F, Rubino S, Uccelli M. Ileal angiolipoma in a patient with multisystemic manifestations of tuberous sclerosis: diagnosis with computerized tomography. Radiol Med 1998; 96: 403–5 (in Italian). 18 Kaneko T, Karasawa Y, Inada H et al. An adult case of intussusception due to inverted Meckel’s diverticulum accompanied by angiolipoma. Nihon Shokakibyo Gakkai Zasshi 1996; 93: 260–5 (in Japanese). 19 Manner M, Scholz E, Wehrmann M, Stickel W. Invagination caused by angiolipoma of the small intestine – a rare cause of occult gastrointestinal hemorrhage. Chirurg 2001; 72: 305–7 (in German). 20 Kwak HS, Kim CS, Lee JM. Small intestinal angiolipoma: MR imaging appearance. Abdom Imaging 2003; 28: 515–7. 21 Kato K, Matsuda M, Onodera K, Sakata H, Kobayashi T, Kasai S. Angiolipoma of the colon with right lower quadrant abdominal pain. Dig Surg 1999; 16: 441–4. 22 Vandamme J. Angiolipoma of the colon. Acta Gastroenterol Belg 1964; 27: 750–7 (in French). 23 Maesawa C, Tamura G, Sawada H, Kamioki S, Nakajima Y, Satodate R. Angiomyolipoma arising in the colon. Am J Gastroenterol 1996; 91: 1852–4. 24 Molinares B, Goldstein A, Varela GJ, Mesa S. Colonic angiolipoma – a rare finding in the gastrointestinal tract.
© 2013 Chinese Medical Association Shanghai Branch, Chinese Society of Gastroenterology, Renji Hospital Affiliated to Shanghai Jiaotong University School of Medicine and Wiley Publishing Asia Pty Ltd
Journal of Digestive Diseases 2014; 15; 154–157 Case report and review of literature. J Radiol Case Rep 2012; 6: 23–8. 25 Kacar S, Kuran S, Temucin T, Odemis B, Karadeniz N, Sasmaz N. Rectal angiolipoma: a case report and review of literature. World J Gastroenterol 2007; 13: 1460–5.
Angiolipoma of the colon
157
26 Ishizuka M, Nagata H, Takagi K, Horie T, Abe A, Kubota K. Rectal angiolipoma diagnosed after surgical resection: a case report. World J Gastroenterol 2007; 13: 467–9. 27 Dionne GP, Seemayer TA. Infiltrating lipomas and angiolipomas revisited. Cancer 1974; 33: 732–8.
© 2013 Chinese Medical Association Shanghai Branch, Chinese Society of Gastroenterology, Renji Hospital Affiliated to Shanghai Jiaotong University School of Medicine and Wiley Publishing Asia Pty Ltd
