Bilateral presentation of fibroadenoma with digital fibroma-like inclusions in the male breast

Department of Pathology and Laboratory Medicine-Starr 1009, New York Presbyterian Hospital-Weill Medical College of Cornell University, 525 E 68th St, New York, NY 10021, USA.
Archives of pathology & laboratory medicine (Impact Factor: 2.84). 08/2007; 131(7):1126-9. DOI: 10.1043/1543-2165(2007)131[1126:BPOFWD]2.0.CO;2
Source: PubMed


Fibroepithelial lesions are uncommon in the male breast. Most published reports describe phyllodes tumors. Fibroadenomas are very common in female breasts, but are exceedingly rare in the male breast. Gynecomastia and/or lobular differentiation have been known to coexist in both types of fibroepithelial lesions in men. We report an exceptional case of recurrent, bilateral fibroadenomas in a man under treatment for prostatic carcinoma. Intracytoplasmic inclusion bodies in stromal cells identical to those seen in infantile digital fibromatosis were identified in one fibroadenoma. To the best of our knowledge, this is the first reported case of bilaterally occurring fibroadenomas in the male breast, one of which also contained digital fibroma-like inclusions, a second unreported phenomenon.

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    • "Both gynecomastia and male breast cancer share hormonal imbalance as a same factor. Exogenous estrogen use in the male patient is evident [5]. Because our case had gynecomastoid hyperplasia and tumor on his AGMLG, we were concerned that he may also have a hormone imbalance as well as gynecomastia or tumor on the breast. "
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    ABSTRACT: Lesions of anogenital mammary-like glands are rare, and only 44 female cases have been reported. Herein, we describe a particularly rare case of phyllodes tumor of anogenital mammary-like glands in a 41-year-old male presenting anal bleeding. Papillectomy was performed. The excised tumor was circumscribed in shape, and after it was sliced into sections, it was noted that there were leaf-like slits on the surface of cut side. Under the microscope, the tumor was found to be biphasic, with a bland glandular epithelium and low-to-intermediate cellular stroma, which together created the leaf-like slits. Gynecomastoid hyperplasia was evident at the periphery. The epithelium showed immuno-activity for ER, PR(focal), AR, and GCDFP-15. The stromal cells showed positive staining for CD34 and vimentin. The morphology and immunophenotype were similar to benign phyllodes tumors of breast. To the best of our knowledge, this case report represents the first case of phyllodes tumor of anogenital mammary-like glands with gynecomastoid hyperplasia at the periphery in a male patient. To make a diagnosis, we had to differentiate this lesion from hidradenoma papilliferum of skin appendage, phyllodes tumor of ectopic prostatic tissue, and other tumors of anogenital mammary-like glands analogous to the breast tumor (e.g., fibroadenoma phyllodes, periductal stromal sarcoma, and spindle cell carcinoma). While gynecomastia of male breast is usually a result of hormone imbalance, our patient’s tumor did not seem to be related to peripheral hormone status in the anogenital mammary-like glands. Nevertheless, because hormone imbalance has been strongly related to male breast cancer, hormone levels may need to be followed in male patients who have this rare malady. Virtual slides The virtual slide(s) for this article can be found here:
    Diagnostic Pathology 03/2013; 8(1):49. DOI:10.1186/1746-1596-8-49 · 2.60 Impact Factor
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    ABSTRACT: The clinicopathologic and immunohistochemical features of 69 pediatric examples of infantile digital fibroma/fibromatosis (IDF) were analyzed. Thirty males, 26 females, and 1 child (sex unstated) ranging from newborn to 120 months of age (median, 12 mo) manifested 74 lesions (5 identified in follow-up) involving the toe or finger (n=71) and the hand or foot (n=3). Tumors ranged in size from 3 to 35 (median, 10) mm. All but 4 study members presented with a solitary lesion. Metachronous IDFs developed in 7 patients within 17 to 82 months. Microscopically, a cytologically bland, fibroproliferative lesion was observed forming a dome-shaped/polypoid nodule directly beneath the epidermis and invading dermal adnexa. Mitotic figures per 20 high-powered fields ranged from 0 to 7 (median, 1). Paranuclear cytoplasmic inclusions were identified in 57 tumors. Tumor cells immunohistochemically expressed calponin (11 of 11 tumors), desmin (9/9), alpha-smooth muscle actin (11/11), CD99 (11/11), CD117 (6/8), heavy caldesmon (2/11 and scattered cytoplasmic inclusions in 4 tumors), CD10 (1/9), nuclear beta-catenin (2/11), and CD34 (1/11), but not muscle actin (HUC1-1), keratins, estrogen/progesterone receptor proteins, or activated caspase-3. Twenty-eight of 38 patients (74%) experienced recurrent/persistent disease (single in 22; multiple in 6) (median, 4 mo after surgery). One recurrent tumor spontaneously regressed and the size of another remained unchanged for almost 17 years before reexcision. All 23 patients with >5 years follow-up are currently disease free (median disease-free interval, 23 y). Minor postoperative functional/cosmetic complaints were reported in 47%. No patient with adequate clinical data developed the digitocutaneous dysplasia syndrome or a conventional fibromatosis, or relayed a family history of IDF/conventional fibromatosis. Our results indicate that IDF is a unique myofibroblastic process separable from conventional fibromatoses and from histologic mimics. Conservative excision or observation after biopsy (with additional surgery employed as necessary) are recommended treatment options.
    The American journal of surgical pathology 10/2008; 33(1):1-13. DOI:10.1097/PAS.0b013e3181788533 · 5.15 Impact Factor
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    ABSTRACT: SUMMARY: BACKGROUND: The purpose of this study was to define the diagnostic accuracy of mammography and ultrasound in the evaluation of male breast disease, and to suggest a diagnostic protocol for male breast disease. MATERIAL AND METHODS: We retrospectively reviewed clinical, radiographic, and pathologic records of 75 patients. Breast Imaging Reporting and Data System (BI-RADS) category 4-5 mammograms and ultrasonograms were suggested as suspicious for malignancy. RESULTS: Of the 75 patients, 23 (31%) were considered to have suspicious lesions by mammography and/or ultrasonography. 13 of the patients were shown to have breast cancer. The remaining 52 (69%) were referred for biopsy by clinicians; all of the biopsy specimens were benign (gynecomastia). The accuracy data of mammography and ultrasonography are: sensitivity, 69 and 100%; specificity, 87 and 97%; positive predictive value, 53 and 87%; negative predictive value, 93 and 100%; and accuracy, 84 and 97%, respectively. CONCLUSION: We suggest a new diagnostic algorithm for the evaluation of male breast disease in which ultrasonography may be used to evaluate palpable abnormalities as the first diagnostic tool of choice. To use and to trust imaging would decrease the number of false-positive biopsies that would be generated by physical examination alone.
    Breast Care 08/2009; 4(4):255-259. DOI:10.1159/000226284 · 0.63 Impact Factor
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