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Juvenile Fibroadenoma with Features of Phyllodes Tumor

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Juvenile Fibroadenoma with Features of Phyllodes Tumor Human Pathology: Case Reports (2015) xx, xxx–xxx http://www.humanpathologycasereports.com Juvenile fibroadenoma with features of phyllodes tumor showing intraductal growth and prominent epithelial hyperplasia in an 11-year-old girl☆ Jun Miyauchi MD a,b,⁎, Fumiko Yoshida MD c, Seiya Akatsuka MD b, Miwako Nakano MD c aDepartment of Pathology and Laboratory Medicine, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba-ken, Japan 272-8513 bDepartment of Clinical Laboratory, Saitama City Hospital, Saitama, Saitama-ken, Japan 336-8522 cDepartment of Pediatric Surgery, Saitama City Hospital, Saitama, Saitama-ken, Japan 336-8522 Received 3 March 2015; revised 26 June 2015; accepted 7 July 2015 Keywords: Abstract Breast tumors in children are uncommon, with the majority of them being adult-type fibroadenoma Juvenile fibroadenoma; (FA). We report a case of juvenile FA (JFA) with features of a benign phyllodes tumor (PT) in an 11-year-old Phyllodes tumor; girl, showing very unusual intraductal/intracystic growth. The tumor was located at the outer peripheral Intraductal papilloma; portion of the right breast apart from the nipple. Histologically, the tumor showed extensive leaf-like Intraductal growth; papillary structures with a broad fibrous stroma, protruding into multiple contiguous cystic spaces lined by Pediatric breast tumor flat ductal epithelium, and closely resembled PT but the stroma of the tumor was only slightly cellular, showing no nuclear atypia and very few mitotic figures. In contrast, epithelial cells covering the fronds exhibited marked hyperplasia, forming a thick multilayered epithelium. The histology of the tumor with intracystic papillary structures and epithelial hyperplasia showed some similarities with intraductal papilloma (IDP). The mechanism of such unusual intraductal growth of fibroepithelial tumors, including FA/ JFA and PT, and their possible common histogenesis with IDP are discussed. © 2015 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). 1. Introduction (FA), which comprises 44–70% of all breast lesions [1,2]. Although adult-type FA accounts for the large majority (93% Breast tumors are uncommon in children and adolescents. [3]) of FA cases in childhood, a small proportion of The tumor most often seen in these ages is fibroadenoma fibroepithelial tumors clinically show rapid growth, often giving rise to large-sized mass lesions, and pathologically ☆ Conflict of interest: None. exhibit somewhat different histology, including cellular ⁎ Corresponding author at: Department of Pathology and Laboratory stroma and epithelial hyperplasia, which is often florid. Medicine, Tokyo Dental College Ichikawa General Hospital, 5-11-13 Sugano, Ichikawa, Chiba-ken, Japan 272-8513. Tel.: +81 47 322 0151; fax: These tumors are called juvenile fibroadenoma (JFA) [3]. +81 47 325 4456. Phyllodes tumor (PT) is another fibroepithelial tumor, E-mail address: [email protected] (J. Miyauchi). rarely seen (about 1% [1,2,4]) in children. PT is distinguished http://dx.doi.org/10.1016/j.ehpc.2015.07.001 2214-3300/© 2015 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by- nc-nd/4.0/). 2 J. Miyauchi et al. from FA by higher degree of stromal cell growth and peculiar and her general health condition was good. Physical intracanalicular growth pattern, producing characteristic examination confirmed a well-defined movable mass at the leaf-like structures with very broad cellular fibrous stroma. boundary portion between upper and lower outer quadrants PT is classified into three categories, namely, benign, of her right breast, apart from the nipple. Ultrasonography borderline and malignant, depending on the degree of stromal revealed a slightly lobulated oval tumor with sharply hypercellularity, cytological atypia and mitoses, stromal demarcated smooth margins, measuring 19 × 15 × 10 mm overgrowth, and nature of tumor borders/margins [5]. in diameter (Fig. 1). The tumor showed slightly hypoechoic Although JFA typically lacks an intracanalicular growth homogeneous internal structure with slight posterior echo pattern and leaf-like structures, which characterize PT, JFA may enhancement. No signs of echogenic spots indicative of exhibit such a growth pattern and show considerable overlap- calcification were present. FA was clinically suspected and ping features with benign PT, both clinically as well as the tumor was surgically enucleated. The resected tumor was pathologically [2,4]. Some authors have included benign PT in pathologically diagnosed as JFA with features of benign PT childhood and adolescents into the entity and concept of JFA [4]. as described below and the patient underwent postoperative Here, we report a case of JFA with an extensive phyllodes follow-up with physical examination and ultrasonography at pattern in an 11-year-old girl, which showed extremely unusual 3- to 6-month intervals. She has been well without any signs intraductal growth in multiple contiguous cystic spaces along of recurrence for 3 years since the surgery. with coincident, prominent epithelial hyperplasia, showing some similarities with intraductal papilloma (IDP). Only a limited number of cases of fibroepithelial tumors, including FA 3. Results and PT, showing intraductal/intracystic growth have been reported in the literature, with some of them described as having foci of IDP. These cases may indicate a possible causative link On gross examination, the surgically resected specimen and common histogenesis between intraductal fibroepithelial was a fairly circumscribed tumor, the cut surface of which was tumors and IDP. solid, yellowish white in color, and 17 × 14 mm in diameter. Histologically, the tumor consisted of fibroepithelial cells forming large leaf-like structures with a broad fibrous stroma covered by thick columnar epithelial layers, protruding into 2. Case report multiple contiguous cystic cavities (Fig. 2A and B). This phyllodes pattern was extensively present throughout the An 11-year-old girl visited the hospital since she had tumor, with only a small part showing an intracanalicular FA noticed a palpable lump in her right breast for 5 months, pattern. The inner surface of the cyst wall was covered with a during which it gradually increased the size. She had no thin monolayer of flat ductal epithelium (Fig. 2CandD).The particular past history and family history for breast cancer tumor contained a mildly cellular stroma, with some Fig. 1 Ultrasonography. The tumor exhibits a slightly lobulated oval mass lesion with smooth margins (arrow heads), slightly hypoechoic homogeneous internal structure and slight enhancement of posterior echoes. 3 A B C D E F Fig. 2 Histology of the tumor. (A) and (B) Low-power view of the tumor. Leaf-like structures with a broad fibrous stroma are present in multiple cystic cavities (A). The tumor is connected to the cyst wall through a stalk-like structure (arrowhead) (B). H-E stain. (C) and (D) Higher magnification shows mildly cellular fibrous stroma covered by thick epithelial layers. Inner surface of the cyst wall is covered by flat epithelium (arrowhead) (D). H-E stain. (E) High-power micrograph depicts bland-looking, plump stromal cells and prominent hyperplasia of multilayered epithelial cells with frequent mitotic figures (arrowheads). H-E stain. (F) Immunohistochemistry (p63). Myoepithelial cells positively stained for p63 are orderly arranged at the basal portion of the epithelium. Scale bars: 1 mm (A and B), 500 μm(C),200μm (D and F) and 100 μm (E). fibroblasts being plump but showing no nuclear atypia and papillary carcinoma. Immunostaining for p63 clearly very few mitotic figures (b1 per 10 high-power fields demonstrated the presence of myoepithelial cells orderly [HPF]), and loose or dense collagenous matrix (Fig. 2C–E). arranged at the basal layer of the epithelium (Fig. 2F). Based In contrast, hyperplasia of epithelial cells covering the upon these findings, a pathological diagnosis of “JFA with stromal projections was striking, constituting a thick, features of PT” was made. pseudostratified epithelium with slit-like structures and frequent mitotic figures (7 per 10 HPF) (Fig. 2C–E). On Ki-67 immunostaining, approximately 20% of epithelial 4. Discussion cells were positive. However, the epithelial cells were bland-looking with variable nuclear configurations and no Cellular FA, including JFA, and benign PT, which fall increase of chromatin, being inconsistent with the features of into the same spectrum of benign fibroepithelial lesions, 4 J. Miyauchi et al. Table 1 Summary of patients with intraductal PT or FA reported in the literature. Case Ref. no. Age Location of Pathology Therapy Follow-up Recurrence no. (year) (year/sex) tumor and period nipple discharge 1 [6] (1998) 62/F Rt. U/O Borderline PT Wide excision 2 y None 2 [7] (2001) 13/F Rt. subareolar, bloody Benign PT Excision through 15 mo None discharge (+) circumareolar incision 3 [8] (2007) 45/F Lt., behind the nipple, Benign PT + ADH (2 Microdochectomy + wide 12 mo None clear discharge (+) lesions) local excision 4 [9] (2008) 33/F Lt., L/O Intraductal Excision through ND ND fibroadenomatosis with circumareolar incision overlapping features of FA, benign PT, IDP and ductal adenoma 5 [10] (2009) 13/F Rt. FA + IDP + areas Wide local excision ND ND resembling PT 6 [10] (2009) 60/F Rt., U/O FA + IDP + low-grade Wide local excision
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