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Year : 2015  |  Volume : 11  |  Issue : 3  |  Page : 650

Recurrent angio-fibroma of breast masquerading as phyllodes tumor

Department of Pathology, Jawaharlal Nehru Medical College (JNMC), Aligarh Muslim University (AMU), Aligarh, Uttar Pradesh, India

Date of Web Publication9-Oct-2015

Correspondence Address:
Jai K Chaurasia
Department of Pathology, Jawaharlal Nehru Medical College (JNMC), Aligarh Muslim University (AMU), Aligarh, U.P.,202 002
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0973-1482.138132

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 > Abstract 

A young Indian female presented with a recurring tumor in the right breast masquerading as phyllodes tumor. Patient had history of five times excision and recurrences of the tumor, diagnosed as fibrous phyllodes of the breast. Presently, a well-circumscribed tumor of about 10 cm size, comprising of benign fibrous-angiomatous tissue with evidence of foci of pyogenic vasculitis was observed. Immuno-histochemical markers for the myo-epithelial and epithelial elements excluded the possibility of fibrous phyllodes, inflammatory myofibroblastic tumor, desmoid fibromatosis, and metaplastic carcinoma. The present findings were diagnostic of an inflammatory angio-fibroma of the right breast, not reported in the earlier literature. The observations indicated that the female breast may be susceptible to spontaneous productive and common-antibiotic-resistant focal septic vascular inflammation giving rise to angio-fibromatous proliferation producing a well-defined tumor mass in the breast, distinguishable from the other breast lesions by the connective tissue stains and immuno-histochemical markers.

Keywords: Angio-fibroma, breast, phyllodes, vasculitis

How to cite this article:
Chaurasia JK, Alam F, Shadan M, Naim M. Recurrent angio-fibroma of breast masquerading as phyllodes tumor. J Can Res Ther 2015;11:650

How to cite this URL:
Chaurasia JK, Alam F, Shadan M, Naim M. Recurrent angio-fibroma of breast masquerading as phyllodes tumor. J Can Res Ther [serial online] 2015 [cited 2020 Jul 10];11:650. Available from: http://www.cancerjournal.net/text.asp?2015/11/3/650/138132

 > Introduction Top

Usually, the recurring benign fibromatous tumors reported earlier in the human breast were either a desmoid tumor, pseudo angiomatous stromal hyperplasia, or stromal phyllodes tumor. [1],[2],[3] Here, we report a case of a recurring breast tumor repeatedly misdiagnosed as fibrous phyllodes before being correctly diagnosed as an inflammatory angio-fibromatous tumor, not reported earlier in the female breast.

 > Case report Top

A 17-year-old unmarried Indian female presented with the complaint of a large painless lump in her right breast, which recurred after being operated and excised five times in the past three years. There was no family history. Routine hematological and urine examinations of the patient were normal. The HIV screening tests were negative. On clinical examination, no other sites except the right breast presented any lump or nodule. Patient's serum calcium was normal (9.2 mg %), and there was no evidence of hyper-thyroidism, hyper-parathyroidism, or any endocrinopathy. The surgeon excised the tumor and submitted for the final diagnosis with mention of earlier histopathological diagnosis of recurring phyllodes tumor. On gross examination, tumor was round to oval, circumscribed, well-encapsulated, measuring 12 × 10 × 6 cm. Cut surface was fleshy, cream-brown with multiple brown chocolate and mucinous cysts [Figure 1]a.
Figure 1: (a) Showing well-circumscribed breast tumor with cream-brown fleshy cut-surface presenting multiple small cysts (arrow), (b) showing angio-fibromatous morphology of the tumor (H and E x50), (c) showing fibro-collagenous stroma comprising of spindle cells (H and E x125), (d) showing focal pyogenic vasculitis (H and E x125)

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Hematoxylin and Eosin (HandE)-stained paraffin sections from the tumor showed benign angio-fibromatous tissues [Figure 1]b. The fibrous component comprised of benign spindle cells with scant light eosinophilic cytoplasm, poorly defined cell borders, and oval to fusiform nuclei [Figure 1]c. The fibrous spindle cells in their appearance closely resembled to the stromal cells of fibrous phyllodes tumor of the breast. The spindle cells lacked cytological atypia. Mitosis was not appreciable. The vessel walls showed foci of pyogenic vasculitis with thrombosis and neutrophil, eosinophil, and occasional plasma cell infiltration [Figure 1]d. No breast duct epithelial cells were found even on extensive search. Van-gieson (VG) stain showed collagen-producing spindle cells [Figure 2]a. Silver impregnation showed positivity around spindle cells and highlighted the blood vessels [Figure 2]b. Phospho-tungstic acid hematoxylin (PTAH) stain, and immuno-stains for cytokeratin, desmin, smooth muscle actin (SMA), and vimentin were negative [Figure 2]c and d, excluding myo-epithelioid and phyllodes origin of the fibrous element of the tumor. Further, immuno-staining with S-100, P-16 were negative excluding the possibility of sarcomatous component in the tumor.
Figure 2: (a) Showing the collagen-producing spindle tumor cells (VG x 50), (b) showing angio-fibromatous tumor components (silver impregnation, x125), (c and d) showing SMA and vimentin-negative tumor spindle cells, respectively (x125)

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The patient was kept on extended antibiotic (ZOSYN 3.375 gm I/V, 30 minutes infusion, every six hour) therapy. Post-operative recovery of the patient was satisfactory. The patient did not have any recurrence during follow-up over the last 11 months.

 > Discussion Top

The findings in the present case were diagnostic of a recurring inflammatory angio-fibroma of the breast. Angio-fibroma is a tumor of benign appearance with distinctive features of high recurrence rate, and the rapidity with which this tumor recurs, probably due to high tumor growth rate coupled with incomplete surgical excision. [4] About 11% of the angio-fibroma recur within 9-120 months after removal of the tumor. [5] Angio-fibroma of the breast, in the present case, recurred five times in the period of three years, probably due to persistent inflammation due to antibiotic resistance.

Desmoid fibromatosis may also present as a recurring, solitary, painless tumor in the breast. [1] Histopathologically, it consists of proliferating spindled fibroblasts and myofibroblasts forming interlacing fascicles, resembling an angio-fibroma. However, the presence of angiomatous component with vasculitis differentiates the inflammatory angio-fibromatous tumor from desmoid fibromatosis of the breast. Besides, the desmoid tumor presents with characteristic infiltrating finger-like projections at the periphery of the lesion entrapping the mammary ducts and lobules. [6] Immuno-histochemically, desmoid tumor is vimentin-positive with variable SMA positivity.

Pseudo angiomatous stromal hyperplasia (PASH) of the breast may sometimes present with nodular, circumscribed, painless breast mass and simulate the inflammatory angio-fibroma. [2] Histopathologically, PASH consists of complex inter-anastomosing, slit-like pseudo-vascular spaces with intervening stroma consisting of dense, hyalinized collagen and spindle cells. Silver impregnation fails to identify the basement membrane around the pseudo-vascular spaces. [7] Immuno-histochemically, the spindle cells of PASH are SMA and vimentin-positive. These findings distinguish between an angio-fibroma and PASH.

Fibrous variant of the phyllodes tumor sometimes recurs and presents fibro-angiomatous morphology. [3] The recurring inflammatory angio-fibroma in the present case was five times diagnosed on HandE-stained sections as recurring fibrous phyllodes tumor of the breast. The connective tissue and immuno-histochemical stains like vimentin and SMA, presently, excluded the possibility of fibrous phyllodes and distinguished the tumor to be inflammatory angio-fibroma of the breast. The fibrous phyllodes usually have ductal epithelial elements, while in present case, tumor showed absence of ductal epithelial component.

A low-grade sarcoma may sometimes give a deceptively benign appearance of an angio-fibroma. However, extensive search always reveals, in the former case, areas in which the cells display malignant features and infiltrative tendency. P-16 immuno-stain is usually positive in sarcomatous lesion, distinguishing it from an angio-fibroma. [8] Similarly, a hemangio-pericytoma can present as a well-circumscribed breast mass. It is distinguishable by distinctively plump ovoid spindle cells arranged around thin and irregularly branched vascular channels. Vascular lining cells and thrombo-vasculitis differentiated the presently inflammatory angio-fibroma from hemangio-pericytoma of the breast.

Occasionally, metaplastic carcinoma (spindle cell subtype) may present as a benign spindle cell tumor. [9] However, typical carcinomatous foci are always present and demonstrable immuno-histochemically by epithelial markers. [10]

Inflammatory myofibroblastic tumor (IMT) of the breast is also a well-circumscribed tumor comprising of proliferating spindle cells and liable to recur. [11] IMT presents with inflammatory foci showing lymphocytes, plasma cells, eosinophils, and histiocytes. The myo-fibroblasts show vimentin positivity. Vimentin negativity differentiated the inflammatory angio-fibromatous tumor presently from the IMT.

 > Conclusion Top

Case findings in this young Indian patient indicated that female breast is susceptible to spontaneous productive focal septic vascular inflammation giving rise to angio-fibromatous proliferation producing a well-defined tumor mass in the breast. Recurrences of such inflammatory angio-fibroma of the breast could indicate resistance to the commonly used post-operative antibiotic therapy. In HandE-stained sections, inflammatory angio-fibroma of the breast may masquerade as phyllodes tumor. Connective tissue staining with immune-histochemical markers for myo-epithelial elements, presently helped exclusion of fibrous phyllodes tumor, PASH, desmoid tumor, IMA, metaplastic carcinoma, and other neoplastic lesions for the final diagnosis of inflammatory angio-fibroma of the breast and need routine application in the breast diagnosis.

 > References Top

Rosen PP, Ernsberger D. Mammary fibromatosis. A benign spindle-cell tumor with significant risk of local recurrence. Cancer 1989;63:1363-9.  Back to cited text no. 1
Vuitch MF, Rosen PP, Erlandson RA. Pseudoangiomatous hyperplasia of mammary stroma. Hum Pathol 1986;17:185-91.  Back to cited text no. 2
Moffat CJ, Pinder SE, Dixon AR, Elston CW, Blamey RW, Ellis IO. Phyllodes tumours of the breast: A clinicopathological review of thirty-two cases. Histopathology 1995;27:205-18.  Back to cited text no. 3
Lloyd G, Howard D, Phelps P, Cheesman A. Juvenile angiofibroma: The lessons of 20 years of modern imaging. J Laryngol Otol 1999;113:127-34.  Back to cited text no. 4
Mariño-Enríquez A, Fletcher CD. Angiofibroma of soft tissue: Clinicopathologic characterization of a distinctive benign fibrovascular neoplasm in a series of 37 cases. Am J Surg Pathol 2012;36:500-8.  Back to cited text no. 5
Wargotz ES, Norris HJ, Austin RM, Enzinger FM. Fibromatosis of the breast. A clinical and pathological study of 28 cases. Am J Surg Pathol 1987;11:38-45.  Back to cited text no. 6
Fisher CJ, Hanby AM, Robinson L, Millis RR. Mammary hamartoma: A review of 35 cases. Histopathology 1992;20:99-106.  Back to cited text no. 7
Chen E, Fletcher CD. Cellular angiofibroma with atypia or sarcomatous transformation: Clinicopathologic analysis of 13 cases. Am J Surg Pathol 2010;34:707-14.  Back to cited text no. 8
Gobbi H, Simpson JF, Borowsky A, Jensen RA, Page DL. Metaplastic breast tumors with a dominant fibromatosis-like phenotype have a high risk of local recurrence. Cancer 1999;85:2170-82.  Back to cited text no. 9
Kurian KM, Al-Nafussi A. Sarcomatoid/metaplastic carcinoma of the breast: A clinicopathological study of 12 cases. Histopathology 2002;40:58-64.  Back to cited text no. 10
Yip CH, Wong KT, Samuel D. Bilateral plasma cell granuloma (inflammatory pseudotumour) of the breast. Aust N Z J Surg 1997;67:300-2.  Back to cited text no. 11


  [Figure 1], [Figure 2]


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