Malignant Phyllodes Tumour with Liposarcomatous Differentiation, Invasive Tubular Carcinoma, and Ductal and Lobular Carcinoma In Situ: Case Report and Review of the Literature
A 43-year-old woman presented with a right breast lump that had enlarged over 5 months. She had chemoradiotherapy for non-Hodgkin’s lymphoma in 1989. Histology revealed a malignant phyllodes tumour (PT) with liposarcomatous differentiation and ductal carcinoma in situ (DCIS) within the tumour with invasive tubular carcinoma, DCIS, and lobular carcinoma in situ in the surrounding breast. She had surgery and adjuvant radiotherapy. One year follow-up showed no recurrence or metastatic disease. Liposarcomatous differentiation is uncommon in PTs, and coexisting carcinoma is rare with 38 cases in 31 reports in the literature. Carcinoma is reported in malignant ( ), benign ( ) and in borderline PTs ( ) with invasive carcinoma ( ) and pure in situ carcinoma ( ) recorded in equal frequency. Carcinoma is more commonly found within the confines of benign PTs; whereas it is more often found surrounding the PT or in the contralateral breast in malignant PTs. Previous radiotherapy treatment is reported in only two cases. The aetiology of co-existing carcinoma is unclear but the rarity of previous radiotherapy treatment suggests that it is incidental. This case highlights the diverse pathology that can occur with PTs, which should be considered when evaluating pathology specimens as they may impact on patient management. 1. Introduction Phyllodes tumors (PTs) of the breast are uncommon biphasic fibroepithelial neoplasms that account for <1% of all breast tumours. Most PTs are benign and carry a risk of local recurrence whereas malignant PTs have a 13% risk of haematogenous metastasis [1].The distinction between benign, borderline and malignant PT is based on the assessment of a number of histological features including infiltrative margin, stromal overgrowth, stromal atypia, cellularity, and mitotic activity.However,while histological features are helpful, they are not accurate predictors of tumour behavior, and no single parameter is reliable in all cases [2]. PTs are believed to arise from intralobular or periductal stroma and may arise de novo or from pre-existing fibroadenomas [2]. Up to 30% of PTs show malignant transformation, most often in the form of malignant transformation of the stroma, which usually shows fibrosarcomatous differentiation and rarely heterologous sarcomatous elements. Malignant transformation of epithelial elements is very rare with only 38 cases reported in the literature. We present a case of a malignant PT that contained heterologous liposarcomatous stromal differentiation and exhibited a range of epithelial pathology. Ductal carcinoma in
References
[1]
J. R. Parfitt, C. Armstrong, F. O'Malley, J. Ross, and A. B. Tuck, “In-situ and invasive carcinoma within a phyllodes tumor associated with lymph node metastases,” World Journal of Surgical Oncology, vol. 2, article 46, 2004.
[2]
F. Tavassoli and P. Devilee, “Fibroepithelial tumours,” in World Health Organization Classification of Tumours of the Breast and Female Genital Organs, pp. 99–103, IARC Press, Lyon, France, 2003.
[3]
D. L. Page, I. O. Ellis, and C. W. Elston, “Histologic grading of breast cancer. Let's do it,” American Journal of Clinical Pathology, vol. 103, no. 2, pp. 123–124, 1995.
[4]
R. Yamaguchi, M. Tanaka, Y. Kishimoto, K. Ohkuma, M. Ishida, and M. Kojiro, “Ductal carcinoma in situ arising in a benign phyllodes tumor: report of a case,” Surgery Today, vol. 38, no. 1, pp. 42–45, 2008.
[5]
M. J. Ramdass and S. Dindyal, “Phyllodes breast tumour showing invasive squamous-cell carcinoma with invasive ductal, clear-cell, secretory, and squamous components,” Lancet Oncology, vol. 7, no. 10, p. 880, 2006.
[6]
T. Kodama, K. Kameyama, M. Mukai, H. Sugiura, T. Ikeda, and Y. Okada, “Invasive lobular carcinoma arising in phyllodes tumor of the breast,” Virchows Archiv, vol. 442, no. 6, pp. 614–616, 2003.
[7]
G. de Rosa, G. Ferrara, P. Goglia, C. Chicas, and P. Zeppa, “In situ and microinvasive carcinoma with squamoid differentiation arising in a phyllodes tumor: report of a case,” Tumori, vol. 75, no. 5, pp. 514–517, 1989.
[8]
T. Yasumura, S. Matsui, T. Hamajima, et al., “Infiltrating ductal carcinoma developing within cystosarcoma phyllodes—a case report,” Japanese Journal of Surgery, vol. 18, no. 3, pp. 326–329, 1988.
[9]
P. J. T. Knudsen and J. Ostergaard, “Cystosarcoma phylloides with lobular and ductal carcinoma in situ,” Archives of Pathology and Laboratory Medicine, vol. 111, no. 9, pp. 873–875, 1987.
[10]
A. Grove and L. Deibjerg Kristensen, “Intraductal carcinoma within a phyllodes tumor of the breast: a case report,” Tumori, vol. 72, no. 2, pp. 187–190, 1986.
[11]
L. Christensen, M. Nielsen, and P. M. Madsen, “Cystosarcoma phyllodes: a review of 19 cases with emphasis on the occurrence of associated breast carcinoma,” Acta Pathologica Microbiologica et Immunologica Scandinavica, Section A, vol. 94, no. 1, pp. 35–41, 1986.
[12]
T. Ishida, M. Izuo, and T. Kawai, “Breast carcinoma arising in cystosarcoma phyllodes: report of a case with a review of the literature,” Japanese Journal of Clinical Oncology, vol. 14, no. 1, pp. 99–106, 1984.
[13]
K. Stone-Tolin, E. W. Pollak, W. Dorzab, and J. Printz, “Recurring cystosarcoma phyllodes associated with breast carcinoma,” Southern Medical Journal, vol. 75, no. 7, pp. 881–884, 1982.
[14]
A. S. Y. Leong and D. J. Meredith, “Tubular carcinoma developing within a recurring cystosarcoma phyllodes of the breast,” Cancer, vol. 46, no. 8, pp. 1863–1867, 1980.
[15]
W. G. Richards and R. W. WAY, “Co-existent cystosarcoma phyllodes and schirrhous adenocarcinoma of the breast,” Wisconsin Medical Journal, vol. 62, pp. 425–428, 1963.
[16]
J. Lester and A. P. Stout, “Cystosarcoma phyllodes,” Cancer, vol. 7, no. 2, pp. 335–353, 1954.
[17]
P.-H. Tan, T. Jayabaskar, K.-L. Chuah, et al., “Phyllodes tumors of the breast: the role of pathologic parameters,” American Journal of Clinical Pathology, vol. 123, no. 4, pp. 529–540, 2005.
[18]
K. K. Deodhar, J. B. Baraniya, K. N. Naresh, S. R. Shinde, and R. F. Chinoy, “Cancerization of phyllodes tumour,” Histopathology, vol. 30, no. 1, pp. 98–99, 1997.
[19]
A. Korula, J. Varghese, M. Thomas, F. Vyas, and A. Korula, “Malignant phyllodes tumour with intraductal and invasive carcinoma and lymph node metastasis,” Singapore Medical Journal, vol. 49, no. 11, pp. e318–e321, 2008.
[20]
M. Kefeli, L. Yildiz, I. Akpolat, P. Balci, and N. Ozen, “The coexistence of invasive ductal carcinoma and malignant phyllodes tumor with liposarcomatous and chondrosarcomatous differentiation in the same breast in a post-osteosarcoma case,” Pathology Research and Practice, vol. 204, no. 12, pp. 919–923, 2008.
[21]
T. Sugie, E. Takeuchi, F. Kunishima, F. Yotsumoto, and Y. Kono, “A case of ductal carcinoma with squamous differentiation in malignant phyllodes tumor,” Breast Cancer, vol. 14, no. 3, pp. 327–332, 2007.
[22]
B. Merck, P. Cansado Martinez, M. Pérez Ramos, N. Martínez Banaclocha, F. J. Lacueva Gómez, and R. Calpena, “Infiltrating ductal carcinoma and synchronous malignant phyllodes tumour: diagnostic and therapeutic approaches,” Clinical and Translational Oncology, vol. 8, no. 11, pp. 830–832, 2006.
[23]
M. Nomura, Y. Inoue, S. Fujita, et al., “A case of noninvasive ductal carcinoma arising in malignant phyllodes tumor,” Breast Cancer, vol. 13, no. 1, pp. 89–94, 2006.
[24]
S. M. Lim and P. H. Tan, “Ductal carcinoma in situ within phyllodes tumour: a rare occurrence,” Pathology, vol. 37, no. 5, pp. 393–396, 2005.
[25]
L. Auerbach, “Concomitant phyllodes tumour and homolateral breast cancer,” Lancet Oncology, vol. 3, no. 5, p. 279, 2002.
[26]
L. H. Gebrim, J. R. Bernardes Júnior, A. C. Nazário, C. Kemp, and G. R. Lima, “Malignant phyllodes tumor in the right breast and invasive lobular carcinoma within fibroadenoma in the other: case report,” Sao Paulo Medical Journal, vol. 118, no. 2, pp. 46–48, 2000.
[27]
R. Nishimura, T. Hasebe, S. Imoto, and K. Mukai, “Malignant phyllodes tumour with a noninvasive ductal carcinoma component,” Virchows Archiv, vol. 432, no. 1, pp. 89–93, 1998.
[28]
V. Padmanabhan, J. E. Dahlstrom, G. C. Chong, and G. Bennett, “Phyllodes tumor with lobular carcinoma in situ and liposarcomatous stroma,” Pathology, vol. 29, no. 2, pp. 224–226, 1997.
[29]
M. Kasami, M. Yoshida, J. Isogaki, et al., “Bilateral breast tumors, malignant phyllodes tumor and invasive lobular carcinoma in a 46,XX/46,XY mosaic female with family history of breast cancer,” Pathology International, vol. 47, no. 2-3, pp. 147–154, 1997.
[30]
C. M. Powell and P. P. Rosen, “Adipose differentiation in cystosarcoma phyllodes: a study of 14 cases,” American Journal of Surgical Pathology, vol. 18, no. 7, pp. 720–727, 1994.
[31]
T. Morimoto, T. Tanaka, K. Komaki, et al., “The coexistence of lobular carcinoma in a fibroadenoma with a malignant phyllodes tumor in the opposite breast: report of a case,” Surgery Today, vol. 23, no. 7, pp. 656–660, 1993.
[32]
M. Huntrakoon, “Malignant cystosarcoma phyllodes with simultaneous carcinoma in the ipsilateral breast,” Southern Medical Journal, vol. 77, no. 9, pp. 1176–1178, 1984.
[33]
T. A. Seemayer, G. Tremblay, and H. Shibata, “The unique association of mammary stromal sarcoma with intraductal carcinoma,” Cancer, vol. 36, no. 2, pp. 599–605, 1975.
[34]
P. A. Isotalo, R. L. George, R. Walker, and S. K. SenGupta, “Malignant phyllodes tumor with liposarcomatous differentiation,” Archives of Pathology and Laboratory Medicine, vol. 129, no. 3, pp. 421–422, 2005.
[35]
R. M. Austin and W. B. Dupree, “Liposarcoma of the breast: a clinicopathologic study of 20 cases,” Human Pathology, vol. 17, no. 9, pp. 906–913, 1986.
[36]
P. P. Rosen, “Fibroepithelial neoplasms,” in Rosen's Breast Pathology, pp. 176–200, Lippincott Williams & Wilkins, Philadelphia, Pa, USA, 2nd edition, 2001.
[37]
A. M. Jones, R. Mitter, R. Springall, et al., “A comprehensive genetic profile of phyllodes tumours of the breast detects important mutations, intra-tumoral genetic heterogeneity and new genetic changes on recurrence,” Journal of Pathology, vol. 214, no. 5, pp. 533–544, 2008.
[38]
A. Kuijper, A. M. Snijders, E. M. J. J. Berns, et al., “Genomic profiling by array comparative genomic hybridization reveals novel DNA copy number changes in breast phyllodes tumours,” Cellular Oncology, vol. 31, no. 1, pp. 31–39, 2009.
[39]
M. Laé, A. Vincent-Salomon, A. Savignoni, et al., “Phyllodes tumors of the breast segregate in two groups according to genetic criteria,” Modern Pathology, vol. 20, no. 4, pp. 435–444, 2007.
[40]
E. J. Sawyer, A. M. Hanby, P. Ellis, et al., “Molecular analysis of phyllodes tumors reveals distinct changes in the epithelial and stromal components,” American Journal of Pathology, vol. 156, no. 3, pp. 1093–1098, 2000.
[41]
E. J. Sawyer, A. M. Hanby, A. J. Rowan, et al., “The Wnt pathway, epithelial-stromal interactions, and malignant progression in phyllodes tumours,” Journal of Pathology, vol. 196, no. 4, pp. 437–444, 2002.
