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| Year : 2018 | Volume
: 61
| Issue : 1 | Page : 120-122 |
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| Anastomosing hemangioma with extensive fatty stroma in the retroperitoneum |
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Ananthvikas Jayaram1, Marie Therese Manipadam1, Paul Mazhuvanchary Jacob2
1 Department of Pathology, Christian Medical College, Vellore, Tamil Nadu, India
2 Department of Endocrine Surgery, Christian Medical College, Vellore, Tamil Nadu, India
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| Date of Web Publication | 22-Mar-2018 |
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 Abstract | | |
Anastomosing hemangiomas are a recently recognized benign vascular neoplasm, first described by Montgomery and Epstein in 2009. A few cases have been described in the genitourinary tract, especially in the renal hilum. These are fairly well-demarcated lesions with lobules of sinusoidal-like capillaries lined by hobnail endothelial cells containing eosinophilic hyaline globules in the cytoplasm. Extramedullary hematopoiesis has been described in a few cases, along with large feeding vessels. A predominant adipocytic component has been described in only one case.[9] We describe a case of a retroperitoneal anastomosing hemangioma occurring in an extrarenal site in a 53-year-old female, followed by a review of the current literature.
Keywords: Anastomosing hemangioma, capillaries, hobnail, sinusoidal
How to cite this article:
Jayaram A, Manipadam MT, Jacob PM. Anastomosing hemangioma with extensive fatty stroma in the retroperitoneum. Indian J Pathol Microbiol 2018;61:120-2 |
How to cite this URL:
Jayaram A, Manipadam MT, Jacob PM. Anastomosing hemangioma with extensive fatty stroma in the retroperitoneum. Indian J Pathol Microbiol [serial online] 2018 [cited 2023 Sep 30];61:120-2. Available from: https://www.ijpmonline.org/text.asp?2018/61/1/120/228165 |
| Introduction | |  |
Retroperitoneal hemangiomas, and vascular tumors in general, are relatively rare tumors which must be differentiated from their potential mimics, both on radiologic as well as histologic examinations. When present, they usually arise from the kidney, or less commonly from the adrenal gland. In a case series of primary retroperitoneal neoplasms, hemangiomas accounted for 4.5% of all tumors.[1] Among the benign vascular tumors of the retroperitoneum, a few cases of vascular malformations and cavernous hemangiomas have been reported in literature.[2],[3] We present the case of a 53-year-old female with a primary anastomosing hemangioma with extensive fatty changes in the retroperitoneum, a recently described benign vascular neoplasm with sinusoidal-like features, and highlight the diagnostic pitfalls.
| Case Report | | |
Our patient was a 53-year-old female with a history of carcinoma of the left breast in 2001, cT3N1M0, for which she underwent mastectomy and axillary node clearance. Histopathology was then reported as an infiltrative ductal carcinoma grade III, estrogen receptor and progesterone receptor negative, and she received 4 cycles of chemotherapy followed by radiotherapy. She has had regular follow-up since then with chest roentgenography, ultrasonograms, and mammograms every year. On a routine ultrasonogram of the abdomen in early 2015, an oval hypoechoic mass measuring 2.8 cm × 1.8 cm was detected in the left iliac fossa, suggestive of a paraganglioma/metastatic lesion. Magnetic resonance imaging of the abdomen showed a brightly enhancing T2-hyperintense mass lesion measuring 4.5 cm × 3.7 cm × 3 cm in the left paravertebral region inferior to the kidney and abutting the left common iliac vessels with a maintained fat plane, and a diagnosis of paraganglioma was favored over a metastatic lesion. Positron emission tomography-computed tomography (CT) revealed abnormal fludeoxyglucose uptake and densely enhancing, well-defined pelvic lesion with focal necrotic/cystic areas medial to the left psoas muscle, abutting the left common iliac artery. There was no significant encroachment onto the surrounding structures or fat stranding. Metaiodobenzylguanidine scintigraphy was not done. A diagnosis of paraganglioma was suggested, and surgery was advised. She had no history of abdominal pain, hypertension, or backache, and was normotensive on examination. She did not have any syndromic features. Investigations for urine metanephrines and normetanephrines were well within the normal limits, and 24-h urine dopamine levels were 607 μg (reference range: 52–480 μg/24 h). Urine 5-hydroxyindoleacetic acid was negative.
At surgery, a 4-cm highly vascular, ovoid, encapsulated mass was noted, situated between the common iliac artery and vein, splaying them with a single-feeding artery direct from the left common iliac artery. The ureter was closely applied to the anteromedial aspect of the tumor with no evidence of obstruction. On examination, the mass was fairly circumscribed and on sectioning revealed a dark brown-to-tan colored, soft cut surface with areas of hemorrhage [Figure 1]a. On histopathology of the resected tumor tissue, low-power examination revealed a circumscribed, unencapsulated tumor composed of many dilated sinusoidal-like capillaries that were lined in foci by plump endothelial cells displaying very minimal atypia [Figure 1]b and [Figure 1]c. These vascular channels resembled splenic red pulp in many foci. Larger feeder vessels were identified within the tumor with congestion and variably hyalinized vessel walls. The intervening stroma showed varying degrees of edema and hyalinization [Figure 1]d. Also seen was a significant component of mature adipose tissue scattered within the tumor [Figure 2]a. Smooth muscle component was not identified. There were no hobnail cells, extramedullary hematopoiesis, or fibrin thrombi. The differential diagnoses we considered were an anastomosing hemangioma, angiomyolipoma, a variant of glomangioma, and less likely a renal cell carcinoma with extensive hemangioma-like features.[1] A panel of markers including HMB-45, synaptophysin, CD34, S-100, and pan cytokeratin was examined by immunohistochemistry. CD34 highlighted the vascular channels while the adipocytes stained for S100 [Figure 2]b and [Figure 2]c. HMB-45, synaptophysin, and pancytokeratin were negative. These features were consistent with a diagnosis of anastomosing hemangioma with extensive fatty changes. | Figure 1: (a) Gross appearance with a dark brown-colored cut surface; (b) low-magnification view with many sinusoidal-like capillaries; (c) capillaries lined by plump endothelial cells displaying very minimal atypia; (d) intervening stroma with varying degrees of edema and hyalinization
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 | Figure 2: (a) Mature adipocytes in the stroma; (b) CD34 immunohistochemistry highlighting the capillaries; (c) S100 immunohistochemistry highlighting adipocytes
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| Discussion | | |
Anastomosing hemangiomas are a recently recognized benign vascular neoplasm, first described by Montgomery and Epstein in 2009.[4] Since then, there have been a limited number of reported cases of anastomosing hemangioma, which mainly raised from the kidney, genitourinary tract, gastrointestinal tract, and liver.[5],[6],[7],[8] Most tumors were located in the renal hilum and had a male predominance. These tumors were originally described as displaying a loosely lobulated architecture with areas of intravascular extension. They may be sharply demarcated or have a focally infiltrative pattern.
Radiologically, anastomosing hemangiomas are generally detected incidentally, as in our case, where it was detected on a routine follow-up scan for a patient treated for carcinoma of the breast. They are generally small, have nonspecific imaging findings, and are seldom diagnosed radiologically. Our case was diagnosed as a paraganglioma on CT.
Macroscopically, anastomosing hemangiomas tend to be well demarcated and unencapsulated, and on sectioning reveal a mahogany brown cut surface, devoid of necrosis.[7]
The vascular pattern is reminiscent of splenic parenchyma with anastomosing capillary sized blood vessels within a framework of supporting cells and lined by endothelial cells displaying hobnail-like features. These endothelial cells are cytologically bland in appearance and infrequently contain eosinophilic hyaline globular inclusions which are positive for periodic acid–Schiff stain with diastase digestion. These were ultrastructurally interpreted as secondary lysosomes by Kryvenko et al.[6] Another feature that has been variably described is the presence of extramedullary hematopoiesis. A few case reports have described, in addition to the above, the presence of a striking adipocytic component in the intervening stroma.[9]
Immunohistochemically, these cases are positive for vascular endothelial markers such as CD34, CD31, and Factor VIII-related antigen, and are negative for HMB45, SMA, EMA, Melan A, HHV8, and AE1/AE3.
In a case series by Kryvenko et al.,[6] they described eight cases of genitourinary anastomosing hemangiomas, in which four cases showed a lobulated architecture, five cases showed large tortuous feeding vessels, and six cases did not show any evidence of extramedullary hematopoiesis, which were correlating with the findings in our case. They also described a hobnail appearance of the endothelial cells in only three cases and intracytoplasmic hyaline globules in five cases, both of which were not seen in our case.
These cases may lead to potential diagnostic pitfalls with other morphologically similar entities, such as angiomyolipoma and angiolipoma. The former comes into the differential diagnosis when there is a significant adipocytic component, as in our case. Angiomyolipomas are positive for HMB45, whereas anastomosing hemangiomas are negative. Angiolipomas usually tend to occur superficially and are only rarely seen in the deep soft tissue. The other entities that are considered in the differential diagnosis of these cases are pericytic tumors, angiosarcomas, and hemangioendotheliomas. Anastomosing hemangiomas and angiosarcomas have anastomosing vascular channels; however, anastomosing hemangiomas have very minimal or no cellular atypia, lack stratification, and are almost devoid of apoptotic and mitotic activity, and therefore can be differentiated from angiosarcomas.
Another rare possibility to be always considered in cases of anastomosing hemangiomas within or adjacent to the kidney is a clear cell renal cell carcinoma with a predominant sinusoidal architecture.[5] The sinusoidal pattern may be present in varying proportions of the tumor, and immunohistochemistry may be required in some cases to establish the diagnosis.
Anastomosing hemangiomas are benign neoplasms and are expected to have an indolent course, as described by Heidegger et al.[10] who followed up a case of renal anastomosing hemangioma in a 56-year-old male for 10 years. However, since the differential diagnosis includes frank malignancies such as angiosarcomas, diagnosis requires careful and thorough study. The reported cases so far have shown a benign course. However, selection of treatment modalities and disease prognostication is yet unclear due to the paucity of cases reported.
To conclude, this is the first case report of an anastomosing hemangioma from India, as per our knowledge. The tumor was found in the retroperitoneum adjacent to the ureter, unlike in the previous cases which were reported in the renal parenchyma. Anastomosing hemangiomas are a recently described entity with only a handful of cases reported in literature. The differential diagnosis ranges from seemingly benign and innocuous lesions such as vascular malformations to malignant neoplasms such as angiosarcomas and clear cell renal cell carcinoma.
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 | | |
| 1. | Lane RH, Stephens DH, Reiman HM. Primary retroperitoneal neoplasms: CT findings in 90 cases with clinical and pathologic correlation. AJR Am J Roentgenol 1989;152:83-9.  |
| 2. | He H, Du Z, Hao S, Yao L, Yang F, Di Y, et al. Adult primary retroperitoneal cavernous hemangioma: A case report. World J Surg Oncol 2012;10:261. |
| 3. | Powis SJ, Rushton DI. A case of retroperitoneal haemangioma. Br J Surg 1972;59:74-6. |
| 4. | Montgomery E, Epstein JI. Anastomosing hemangioma of the genitourinary tract: A lesion mimicking angiosarcoma. Am J Surg Pathol 2009;33:1364-9. |
| 5. | Kryvenko ON, Roquero L, Gupta NS, Lee MW, Epstein JI. Low-grade clear cell renal cell carcinoma mimicking hemangioma of the kidney: A series of 4 cases. Arch Pathol Lab Med 2013;137:251-4. |
| 6. | Kryvenko ON, Gupta NS, Meier FA, Lee MW, Epstein JI. Anastomosing hemangioma of the genitourinary system: Eight cases in the kidney and ovary with immunohistochemical and ultrastructural analysis. Am J Clin Pathol 2011;136:450-7. |
| 7. | Zhao M, Li C, Zheng J, Sun K. Anastomosing hemangioma of the kidney: A case report of a rare subtype of hemangioma mimicking angiosarcoma and review of the literature. Int J Clin Exp Pathol 2013;6:757-65. |
| 8. | Lin J, Bigge J, Ulbright TM, Montgomery E. Anastomosing hemangioma of the liver and gastrointestinal tract: An unusual variant histologically mimicking angiosarcoma. Am J Surg Pathol 2013;37:1761-5. |
| 9. | Tran TA, Pernicone P. Anastomosing hemangioma with fatty changes of the genitourinary tract: A lesion mimicking angiomyolipoma. Cent European J Urol 2012;65:40-2. |
| 10. | Heidegger I, Pichler R, Schäfer G, Zelger B, Zelger B, Aigner F, et al. Long-term follow up of renal anastomosing hemangioma mimicking renal angiosarcoma. Int J Urol 2014;21:836-8. |
Correspondence Address:
Ananthvikas Jayaram
Department of Pathology, Christian Medical College, Vellore - 632 004, Tamil Nadu
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/IJPM.IJPM_259_16
[Figure 1], [Figure 2] |
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