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LETTER TO EDITOR  
Year : 2020  |  Volume : 63  |  Issue : 4  |  Page : 670-672
CD10 positive benign stromal spindle cell tumor (not otherwise specified) of the male breast


Department of Pathology, Smt. Kashibai Navale Medical College and General Hospital, Pune, Maharashtra, India

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Date of Submission31-Mar-2020
Date of Decision25-Jun-2020
Date of Acceptance30-Jun-2020
Date of Web Publication28-Oct-2020
 

How to cite this article:
Sinai Khandeparkar SG, Bharti N, Saragade P, Pathade S, Gogate B. CD10 positive benign stromal spindle cell tumor (not otherwise specified) of the male breast. Indian J Pathol Microbiol 2020;63:670-2

How to cite this URL:
Sinai Khandeparkar SG, Bharti N, Saragade P, Pathade S, Gogate B. CD10 positive benign stromal spindle cell tumor (not otherwise specified) of the male breast. Indian J Pathol Microbiol [serial online] 2020 [cited 2020 Nov 28];63:670-2. Available from: https://www.ijpmonline.org/text.asp?2020/63/4/670/299305




Dear Editor,

A 57-year-old male presented to the surgery outpatient department with gynecomastia of the right breast since one year. Local examination revealed 3 × 2 cm mobile, firm to hard mass in the retro areolar region of the right breast. There was no skin involvement or palpable axillary lymphadenopathy. Systemic examination findings were non-contributory. On enquiry no other significant past history was obtained. Mammography showed 4 × 1.9 cm hypoechoic lesion of BIRADS 3. In view of elderly age and firm to hard slowly enlarging tumor, trucut biopsy was done.

Trucut biopsy from the breast mass yielded fibrofatty and fibrocollagenous tissue with no ductulo-lobular units. There was seen focal area of vascular proliferation with mild lymphocytic infiltrate. In view of clinico-radiological and histopathological findings, excisional biopsy was suggested.

Lumpectomy specimen received was serially sectioned. On gross examination, a solid growth measuring 4 cm in diameter was identified. It was tan brown with glistening areas having pushing borders. [Figure 1]a Microscopic examination showed circumscribed tumor mass composed of hypo and hyper cellular areas. Hypercellular areas showed spindle cells with round to oval nucleus and moderate amount of eosinophilic cytoplasm, which at places had epithelioid morphology arranged in short whorls separated by collagen fibers. Occasional mitotic figure was noted. A few of the spindle cells showed atypical features in the form of nuclear pleomorphism. Occasional spindle cell showed floret like giant cell appearance. Hypocellular areas showed edematous and at places myxoid stroma. Small caliber vascular proliferation was noticed. At places ectatic blood vessels were observed showing hyalinization with perivascular lymphocytic infiltrate. Occasional vessel showed presence of a thrombus. Areas of hemorrhage, pigment laden macrophages and mast cells were also seen. [Figure 1]b, [Figure 1]c, [Figure 1]d, [Figure 1]e Based on these findings, diagnosis of the benign spindle cell tumors (BSCT) was suggested. Representative sections were subjected to immunohistochemical (IHC) studies.
Figure 1: (a) Gross photograph showing circumscribed tumor having tan brown color with glistening areas, (b) Photomicrograph showing hypo and hypercellular areas with presence of collagen fibers, (H&E, ×400) (c) ectatic blood vessels showing hyalinization with perivascular lymphocytic infiltrate, multinucleated floret like giant cell, (d) hypocellular areas showing vascular proliferation, (e) pseudoinclusions in tumor cells (H&E, ×400), (f) vimentin showing cytoplasmic immunoreactivity (×40), (g) ER and (h) PR showing weak nuclear immunoreactivity, (i) CD10 showing membrane immunoreactivity, (j) S100 negativity and (k) CD34 negativity (×400)

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IHC was performed with the following panel of antibodies utilised for predominant d/d of myofibroblastoma, schwannoma and vascular tumor. Panel of IHC markers performed included PANCK, vimentin, smooth muscle actin (SMA), desmin, S100, CD34, CD31, bcl2, ER, PR, CD10, EMA (epithelial membrane antigen), CD99 and Ki67. The tumor cells showed strong cytoplasmic immunoreactivity for vimentin and strong membrane immunoreactivity for CD10. The tumor cells showed weak nuclear immunoreactivity for ER and PR. The tumor cells were non-immunoreactive for PANCK, SMA, desmin, S100, CD34, CD31, bcl2, CD99 and EMA. [Figure 1]f-k] Ki67 labelling index (Ki67LI) was found to be less than 1%. Diagnosis of BSSCT (NOS) was made. The patient is free of disease on follow-up for 4 months.

Until puberty, the male breast is composed of fibrofatty tissue and ducts are lined by a single layer of epithelial cells with an underlying layer of myoepithelium. During puberty, testosterone levels increase in males, causing involution and atrophy of the ducts. As a result, the normal adult male breast is primarily composed of subcutaneous fat, stromal elements, a small nipple-areolar complex and an underlying, poorly developed ductal system that ends blindly. The presence of terminal duct lobular units is rare.[1] Gynecomastia is the most common benign condition of the male as seen in present case and the most common cause of a palpable mass. Lipoma is the most common benign tumor of the male breast. Male breast cancer accounts for only 0.7% of all breast cancers.[2]

Spindle cell lesions of the breast are rare and cover a wide spectrum of diseases ranging from reactive tumor-like lesions to high-grade malignant tumors. The recognition of the benign spindle cell tumor-like lesions such as nodular fasciitis; reactive spindle cell nodule after biopsy, inflammatory myofibroblastic tumor; pseudoangiomatous stromal hyperplasia and tumors like myofibroblastoma, benign fibroblastic spindle cell tumor, leiomyoma, schwannoma, spindle cell lipoma, solitary fibrous tumor and myxoma is crucial to avoid confusion with morphologically similar but more aggressive bland-appearing spindle cell tumors, such as desmoid-type fibromatosis, low-grade (fibromatosis-like) spindle cell carcinoma, low-grade fibrosarcoma/myofibroblastic sarcoma and dermatofibrosarcoma protuberans and malignant tumors such as pure sarcoma, phyllodes tumor and metaplastic carcinoma.[3],[4] Meticulous histopathological examination together with IHC are extremely crucial in arriving at an accurate diagnosis from the wide array of differential diagnosis to rule out possibility of malignant SCTs and then arrive at a specific diagnosis in benign category. Generally, the lack of marked cytological atypia, along with an absence of necrosis, high mitotic activity, atypical mitoses and proliferative IHC markers such as Ki67, are helpful to exclude malignancy.[5],[6]

Purely benign mesenchymal spindle cell neoplasms of the breast are currently labeled under various terms in the literature like BSCT, fibroma, spindle cell lipoma, myofibroblastoma, solitary fibrous tumor and myogenic stromal tumor. The lack of strict diagnostic criteria to clearly specify such mesenchymal neoplasms is the main reason which generated the risk of terming the same lesion under different names or, conversely, of collecting different types under the same term. Although such neoplasms exhibit morphological and immunophenotypical heterogeneity, they actually represent variations of the same tumor entity, likely arising from the uncommitted vimentin+/CD34+ fibroblasts of the mammary stroma, capable of multidirectional mesenchymal differentiation. To cover the entire spectrum of such lesions, the term “BSCT of the mammary stroma” is advocated. Benign spindle cell tumors (BSCT) are typically located in the subareolar region. These tumors are circumscribed but not encapsulated. BSCT can be subtyped into four main groups by light microscopy and immunocytochemistry as fibroblastic, myofibroblastic, fibrohistiocytic, and mixed forms.[7],[8]

However, in the present case, the benign spindle cells, identified by the lack of hyperchromasia, mitosis and Ki67LI less than 1%, though vimentin positive did not show CD34 expression. The histomorphology showed hypercellular areas such as spindle cells arranged in whorls separated by few collagen fibres with no atypical mitosis or necrosis resembling areas of myofibroblastoma. There were epithelioid cells in present case which in case if predominant i.e., more than 50% warrants a diagnosis of epithelioid variant of myofibroblastoma as described in literature.[9] Hyalinised collagen bundles of varying size are usually detected among neoplastic cells which helps suggest the diagnosis. Few atypical bizarre cells were observed in the present case. These are believed to be degenerative in nature. Epithelioid variant of myofibroblastoma also is known to show presence of multinucleated floret like cells resembling lipoblast as seen in present case.[9] Myofibroblastoma uncommonly show absent/focal expression of CD34, a potential diagnostic pitfall.[10] In such cases, characteristic staining with other IHC markers such as desmin is observed which can aid in confirming the diagnosis. Immunoreactivity for SMA, bcl2 and CD99 is frequently observed but with different extension in different tumors and also different areas of same tumor. ER, PR is seen in most cases.[9] In the present case, the tumor cells showed weak ER and PR positivity and did not express SMA, desmin, bcl2 and CD99. Hence, diagnosis of BSSCT (NOS) was offered. Recently CD10 expression is studied in benign and malignant spindle cell tumors of the breast.[9] Strong CD10 expression was seen in this case. Present case satisfied essential histomorphological features of myofibroblastoma[9] however with a few of the positive IHC markers such as vimentin, ER, PR and CD10 and absence of other markers such as SMA, desmin, bcl2 and CD99 we found the evidence insufficient to render the diagnosis of epithelioid variant of myofibroblastoma. The positive and negative controls added to the IHC study were found to be satisfactory.

There were focal areas showing nuclear palisading together with ectatic, hyalinised blood vessels which resembled schwannoma however as S100 was negative this diagnosis was ruled out.[11] Extensive vascular proliferation in hypocellular areas raised a possibility of vascular tumor. However, CD31 negativity and CD34 positivity in these vessels with weak hormone receptor expression ruled out the possibility of epithelioid hemangioendothelioma and low grade angiosarcoma.[5]

Although histopathology remains preeminent in the diagnosis of SCT of the breast, IHC is crucial in some cases for confirming the diagnosis and its subsequent subtyping. While confusing a benign lesion during its further subtyping may not affect the treatment modality, however, misinterpreting it for malignancy may have grave clinical implications. This case report adds to the spectrum of the benign BSCTs and delineates the nature of different types of these lesions in order to carefully select optimal therapeutic regimes.[7]

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Javed A, Lteif A. Development of the human breast. Semin Plast Surg 2013;27:5-12.  Back to cited text no. 1
    
2.
Yuan WH, Li AF, Chou YH, Hsu HC, Chen YY. Clinical and ultrasonographic features of male breast tumors: A retrospective analysis. PLoS One 2018;13:e0194651.  Back to cited text no. 2
    
3.
Magro G. Differential diagnosis of benign spindle cell lesions. Surg Pathol Clin 2018;11:91-121.  Back to cited text no. 3
    
4.
Rakha EA, Aleskandarany MA, Lee AH, Ellis IO. An approach to the diagnosis of spindle cell lesions of the breast. Histopathology 2016;68:33-44.  Back to cited text no. 4
    
5.
Ünal B, Erdoǧan G, Karaveli FŞ. Step by step approach to rare breast lesions containing spindle cells. Springerplus 2015;4:678.  Back to cited text no. 5
    
6.
Nakayama Y, Iwasaki H, Iwanaga S, Nakamura H, Shiroshita T, Kikuchi M, et al. Spindle cell carcinoma of the breast: A case report and an immunohistochemical study including p53 and Ki-67 expression. Pathol Int 1997;47:404-11.  Back to cited text no. 6
    
7.
Dragoumis D, Atmatzidis S, Chatzimavroudis G, Lakis S, Panagiotopoulou K, Atmatzidis K. Benign spindle cell tumor not otherwise specified (NOS) in a male breast. Int J Surg Pathol 2010;18:575-9.  Back to cited text no. 7
    
8.
Magro G, Michal M, Bisceglia M. Benign spindle cell tumors of the mammary stroma: Diagnostic criteria, classification, and histogenesis. Pathol Res Pract 2001;197:453-66.  Back to cited text no. 8
    
9.
Magro G. Mammary myofibroblastoma: A tumor with a wide morphologic spectrum. Arch Pathol Lab Med 2008;132:1813-20.  Back to cited text no. 9
    
10.
D'Alfonso TM, Subramaniyam S, Ginter PS, Mosquera JM, Croyle J, Liu YF, et al. Characterization of CD34-deficient myofibroblastomas of the breast. Breast J 2018;24:55-61.  Back to cited text no. 10
    
11.
Khatib Y, Pandey V, Khade AL, Pandey R. Myofibroblastoma of the breast: A rare cause of breast lump in a postmenopausal woman. J Midlife Health 2018;9:47-9.  Back to cited text no. 11
    

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Correspondence Address:
Siddhi Gaurish Sinai Khandeparkar
E-517, The Island, Wakad, Pune - 411 057, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_303_20

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