Abstract
OBJECTIVES:
Breast hamartoma is an uncommon breast tumor that accounts for approximately 4.8% of all benign breast masses. The pathogenesis is still poorly understood and breast hamartoma is not a well-known disorder, so its diagnosis is underestimated by clinicians and pathologists. This study was designed to present our experience with breast hamartoma, along with a literature review.
METHOD:
We reviewed the demographic data, pathologic analyses and imaging and results of patients diagnosed with breast hamartoma between December 2003 and September 2013.
RESULTS:
In total, 27 cases of breast hamartoma operated in the Ankara University Medicine Faculty's Department of General Surgery were included in the study. All patients were female and the mean age was 41.8±10.8 years. The mean tumor size was 3.9±2.7 cm. Breast ultrasound was performed on all patients before surgery. The most common additional lesion was epithelial hyperplasia (22.2%). Furthermore, lobular carcinoma in situ was identified in one case and invasive ductal carcinoma was observed in another case. Immunohistochemical staining revealed myoid hamartoma in one case (3.7%).
CONCLUSION:
Breast hamartomas are rare benign lesions that may be underdiagnosed because of the categorization of hamartomas as fibroadenomas by pathologists. Pathologic examinations can show variability from one case to another. Thus, the true incidence may be higher than the literature indicates.
Breast; Hamartoma; Carcinoma
INTRODUCTION
Breast hamartoma is an uncommon breast tumor that accounts for approximately 4.8% of
all benign breast masses (11. Charpin C, Mathoulin MP, Andrac L, Barberis J, Boulat J,
Sarradour B, et al. Reappraisal of breast hamartomas. A morphological study
of 41 cases. Pathol Res Pract. 1994;190(4):362-71,
http://dx.doi.org/10.1016/S0344-0338(11)80408-5.
http://dx.doi.org/10.1016/S0344-0338(11)...
) and that
contains lobular breast tissue involving various fibrous, fibrocystic and adipose
tissues (22. Fisher CJ, Hanby AM, Robinson L, Millis RR. Mammary
hamartoma—a review of 35 cases. Histopathology. 1992;20(2):99-106,
http://dx.doi.org/10.1111/j.1365-2559.1992.tb00938.x.
http://dx.doi.org/10.1111/j.1365-2559.19...
). However, with increasing social
awareness and widespread breast cancer screening, hamartomas are being routinely
diagnosed with greater frequency. The pathogenesis of hamartomas remains unclear and
its diagnosis is underestimated by clinicians and pathologists. In the literature,
breast hamartoma is presented only in case reports and reviews of wide series are
very rare. Thus, this study was designed to present the clinicopathologic findings
of twenty-seven patients with breast hamartoma.
METHODS
Between December 2003 and September 2013, 27 cases of breast hamartoma operated in the Ankara University Medicine Faculty's Department of General Surgery were included in this study. The histopathology records, clinical follow-up data and previous breast imaging results of the patients were obtained. These data were again analyzed and annotated by clinicians and pathologists.
RESULTS
Demographic features, tumor sizes, preoperative imaging procedures, pathologic results and treatment modalities are shown in Table 1. Clinically, most (2424. Kuroda N, Goishi K, Ohara M, Hirouchi T, Mizumo K, Nakagawa K. Bilateral hamartoma arising in axillary accessory mammary glands. Case report. APMIS. 2006;114(1):77-8.) patients had painless, soft-to-firm, palpable breast lumps and three patients had breast asymmetry. Breast ultrasonography (US) was performed on all of the patients as the first preoperative imaging procedure. In addition to US, mammography was performed in 6 cases and magnetic resonance imaging (MRI) was conducted in one case. In total, 23 cases showed features characteristic of fibroadenoma or hamartoma. In 4 patients (14.8%), US revealed a partial irregular border, and fine-needle aspiration cytology (FNAC) was performed and revealed benign lesions. Pathologic examination usually revealed a well-circumscribed mass with normal breast components, such as a terminal ductal lobular unit, fat and a hyalinized stroma (Figures 1A-B). The lesions usually showed fibrocystic changes, columnar cell changes and adenosis. No proliferative lesions were observed in 14 patients (51.9%). The most common additional proliferative lesion was ductal epithelial hyperplasia, which was diagnosed in 6 patients (22.2%) (Figure 2). We identified lobular carcinoma in situ (LCIS) in one case and invasive ductal carcinoma (IDC) in another case, near the breast hamartoma (3.7%). Pseudoangiomatous hyperplasia was observed in 7 cases (25.9%) (Figure 3). One case (3.7%) was pathologically reported as breast myoid hamartoma and this hamartoma stained positive for desmin, smooth muscle actin (SMA) and CD34 (Figures 4A-C). Mastectomy and sentinel lymph node biopsy were performed on the patient with IDC and lumpectomies were performed on the other patients.
(A) and (B) Well-circumscribed mass with normal terminal ductal lobuler unit, fat and hyalinized stroma, Hematoksilen Eosin (H&E), ×40.
DISCUSSION
Breast hamartomas are poorly defined, rare, benign breast neoplasms. Hamartomas were
initially defined as mastomas in 1928 by Prym (33. Prym P. Pseudoadenome, Adenome und Mastome der weiblithen
Brustdrfise; Studien fiber die Entstehung umschriebener adenom∼ hnlicher
Herde in der Mamma und fiber die Nachahmung der Brustdrfisengewebes durch echte
Adenome und Fibroadenome. Beitr Pathol Anat. 1928;81:1-44.). Afterward, several cases were reported and classified as
adenolipomas, fibroadenolipomas or lipofibroadenomas (44. Altermatt HJ, Gebbers JO, Laissue JA. Multiple hamartomas of the
breast. Appl Pathol. 1989;7(2):145-8.). Arrigoni et al. (55. Arrigoni MG, Dockerty MB, Judd ES. The identification and
treatment of mammary hamartoma. Surg Gynecol Obstet.
1971;133(4):577-82.)
first used the term hamartoma in 1971. Hamartoma is referred to as myoid hamartoma,
a rarer form, when it shows a significant smooth muscle component. This term was
first used by Davies and Riddell (66. Davies JD, Riddell RH. Muscular hamartomas of the breast.
J Pathol. 1973;111(3):209-11,
http://dx.doi.org/10.1002/path.1711110309.
http://dx.doi.org/10.1002/path.171111030...
) in
1973.
Articles related to breast hamartoma in the literature are mostly case reports and
rarely case series; therefore, they are limited in number. Thus, there are few data
about these tumors. We compiled the published case reports and case series in PubMed
from between 1993 and 2013 in terms of demographic features, tumor size, applied
imaging procedures, pathology reports and the results of additional staining, which
are shown in Table 2 (77. Nasit JG, Parikh B, Trivedi P, Shah M. Myoid (muscular) hamartoma
of the breast with chondroid metaplasia. Indian J Pathol Microbiol.
2012;55(1):121-2.
8. Mizuta N, Sakaguchi K, Mizuta M, Imai A, Nakatsukasa K, Morita M,
et al. Myoid hamartoma of the breast that proved difficult to diagnose: a case
report. World J Surg Oncol. 2012;10:12.
9. Kai M, Tada K, Tamura M, Gomi N, Horii R, Akiyama F, et al.
Breast cancer associated with mammary hamartoma. Breast Cancer.
2012;19(2):183-6, http://dx.doi.org/10.1007/s12282-009-0147-3.
http://dx.doi.org/10.1007/s12282-009-014...
10. Uchoa DM, Cruz DB, Schaefer PG, Pegas KL, Cambruzzi E.
Myofibroblastoma arising in mammary hamartoma: a case report. Patholog Res Int.
2010;2010: 4 pages. Article ID726829.
11. Choi N, Ko ES. Invasive ductal carcinoma in a mammary hamartoma:
case report and review of the literature. Korean J Radiol.
2010;11(6):687-91.
12. Ko MS, Jung WS, Cha ES, Choi HJ. A rare case of recurrent myoid
hamartoma mimicking malignancy: imaging appearances. Korean J Radiol.
2010;11(6):683-6.
13. Gupta SS, Singh O, Hastir A, Arora G, Sabharwal G, Mishra H.
Breast hamartoma with intrathoracic extension in a 13-year-old boy.
J Cancer Res Ther. 2010;6(1):86-8,
http://dx.doi.org/10.4103/0973-1482.63559.
http://dx.doi.org/10.4103/0973-1482.6355...
14. Kajo K, Zubor P, Danko J. Myoid (Muscular) Hamartoma of the
Breast: Case Report and Review of the Literature. Breast Care (Basel).
2010;5(5):331-4, http://dx.doi.org/10.1159/000321341.
http://dx.doi.org/10.1159/000321341...
15. Khoo JJ, Alwi RI, Abd-Rahman I. Myoid hamartoma of breast with
chondroid metaplasia: a case report. Malays J Pathol.
2009;31(1):77-80.
16. Pervatikar SK, Rao R, Dinesh US, Parameswaraiah S. Large mammary
hamartoma with focal invasive ductal carcinoma. Indian J Pathol
Microbiol. 2009;52(2):249-51.
17. Lee WF, Sheen-Chen SM, Chi SY, Huang HY, Ko SF. Hamartoma of the
breast: an underrecognized disease? Tumori.
2008;94(1):114-5.
18. Hernanz F, Vega A, Palacios A, Fleitas MG. Giant hamartoma of
the breast treated by the mammaplasty approach. ANZ J Surg.
2008;78(3):216-7.
19. Stafyla V, Kotsifopoulos N, Grigoriadis K, Bakoyiannis CN, Peros
G, Sakorafas GH. Myoid hamartoma of the breast: a case report and review of the
literature. Breast J. 2007;13(1):85-7,
http://dx.doi.org/10.1111/j.1524-4741.2006.00369.x.
http://dx.doi.org/10.1111/j.1524-4741.20...
20. Murat A, Ozdemir H, Yildirim H, Poyraz AK, Ozercan R. Hamartoma
of the breast. Australas Radiol. 2007;51 Spec No.:B37-9,
http://dx.doi.org/10.1111/j.1440-1673.2007.01818.x.
http://dx.doi.org/10.1111/j.1440-1673.20...
21. Murugesan JR, Joglekar S, Valerio D, Bradley S, Clark D, Jibril
JA. Myoid hamartoma of the breast: case report and review of the literature.
Clin Breast Cancer. 2006;7(4):345-6,
http://dx.doi.org/10.3816/CBC.2006.n.050.
http://dx.doi.org/10.3816/CBC.2006.n.050...
22. da Silva BB, Rodrigues JS, Borges US, Pires CG, Pereira da Silva
RF. Large mammary hamartoma of axillary supernumerary breast tissue. Breast.
2006;15(1):135-6,
http://dx.doi.org/10.1016/j.breast.2005.03.004.
http://dx.doi.org/10.1016/j.breast.2005....
23. Ruiz-Tovar J, Reguero-Callejas ME, Arano-Bermejo JI,
Gonzalez-Palacios F, Cabanas-Navarro L. [Mammary hamartoma]. Cir Esp.
2006;79(3):186-8,
http://dx.doi.org/10.1016/S0009-739X(06)70848-X.
http://dx.doi.org/10.1016/S0009-739X(06)...
24. Kuroda N, Goishi K, Ohara M, Hirouchi T, Mizumo K, Nakagawa K.
Bilateral hamartoma arising in axillary accessory mammary glands. Case report.
APMIS. 2006;114(1):77-8.
25. Breucq C, Verfaillie G, Perdaens C, Vermeiren B, Stadnik T.
Lobular carcinoma located in a breast hamartoma. Breast J.
2005;11(6):508-9,
http://dx.doi.org/10.1111/j.1075-122X.2005.00144.x.
http://dx.doi.org/10.1111/j.1075-122X.20...
26. Barbaros U, Deveci U, Erbil Y, Budak D. Breast hamartoma: a case
report. Acta Chir Belg. 2005;105(6):658-9.
27. Gatti G, Mazzarol G, Simsek S, Viale G. Breast hamartoma: a case
report. Breast Cancer Res Treat. 2005;89(2):145-7,
http://dx.doi.org/10.1007/s10549-004-1656-6.
http://dx.doi.org/10.1007/s10549-004-165...
28. Filho OG, Gordan AN, Mello Rde A, Neto CS, Heinke T. Myoid
hamartomas of the breast: report of 3 cases and review of the literature.
Int J Surg Pathol. 2004;12(2):151-3.
29. Lee EH, Wylie EJ, Bourke AG, Bastiaan De Boer W. Invasive ductal
carcinoma arising in a breast hamartoma: two case reports and a review of the
literature. Clin Radiol. 2003;58(1):80-3,
http://dx.doi.org/10.1053/crad.2003.1133.
http://dx.doi.org/10.1053/crad.2003.1133...
30. Baron M, Ladonne JM, Gravier A, Picquenot JM, Berry M. Invasive
lobular carcinoma in a breast hamartoma. Breast J. 2003;9(3):246-8,
http://dx.doi.org/10.1046/j.1524-4741.2003.09313.x.
http://dx.doi.org/10.1046/j.1524-4741.20...
31. Tse GM, Law BK, Pang LM, Cheung HS. Ductal carcinoma in situ
arising in mammary hamartoma. J Clin Pathol. 2002;55(7):541-2,
http://dx.doi.org/10.1136/jcp.55.7.541.
http://dx.doi.org/10.1136/jcp.55.7.541...
32. Kuroda N, Sugimoto T, Numoto S, Enzan H. Microinvasive lobular
carcinoma associated with intraductal spread arising in a mammary hamartoma.
J Clin Pathol. 2002;55(1):76-7,
http://dx.doi.org/10.1136/jcp.55.1.76.
http://dx.doi.org/10.1136/jcp.55.1.76...
33. Herbert M, Sandbank J, Liokumovich P, Yanai O, Pappo I, Karni T,
et al. Breast hamartomas: clinicopathological and immunohistochemical studies of
24 cases. Histopathology. 2002;41(1):30-4,
http://dx.doi.org/10.1046/j.1365-2559.2002.01429.x.
http://dx.doi.org/10.1046/j.1365-2559.20...
34. Tse GM, Law BK, Ma TK, Chan AB, Pang LM, Chu WC, et al.
Hamartoma of the breast: a clinicopathological review. J Clin Pathol.
2002;55(12):951-4, http://dx.doi.org/10.1136/jcp.55.12.951.
http://dx.doi.org/10.1136/jcp.55.12.951...
35. Ravakhah K, Javadi N, Simms R. Hamartoma of the breast in a man:
first case report. Breast J. 2001;7(4):266-8,
http://dx.doi.org/10.1046/j.1524-4741.2001.20079.x.
http://dx.doi.org/10.1046/j.1524-4741.20...
36. Weinzweig N, Botts J, Marcus E. Giant hamartoma of the breast.
Plast Reconstr Surg. 2001;107(5):1216-20,
http://dx.doi.org/10.1097/00006534-200104150-00019.
http://dx.doi.org/10.1097/00006534-20010...
37. Wahner-Roedler DL, Sebo TJ, Gisvold JJ. Hamartomas of the
breast: clinical, radiologic, and pathologic manifestations. Breast J.
2001;7(2):101-5,
http://dx.doi.org/10.1046/j.1524-4741.2001.007002101.x.
http://dx.doi.org/10.1046/j.1524-4741.20...
38. Takeuchi M, Kashiki Y, Shibuya C, Yamamoto S, Kitamura F, Nagao
Y, et al. A case of muscular hamartoma of the breast. Breast Cancer.
2001;8(3):243-5, http://dx.doi.org/10.1007/BF02967516.
http://dx.doi.org/10.1007/BF02967516...
39. Mester J, Simmons RM, Vazquez MF, Rosenblatt R. In situ and
infiltrating ductal carcinoma arising in a breast hamartoma. AJR
Am J Roentgenol. 2000;175(1):64-6,
http://dx.doi.org/10.2214/ajr.175.1.1750064.
http://dx.doi.org/10.2214/ajr.175.1.1750...
40. Chiacchio R, Panico L, D'Antonio A, Delrio P, Bifano D,
Avallone M, et al. Mammary hamartomas: an immunohistochemical study of ten
cases. Pathol Res Pract. 1999;195(4):231-6,
http://dx.doi.org/10.1016/S0344-0338(99)80039-9.
http://dx.doi.org/10.1016/S0344-0338(99)...
41. Blomqvist L, Malm M, Fernstad R. Hamartoma of the breast:
surgical treatment and reconstruction. Case report. Scand J Plast
Reconstr Surg Hand Surg. 1997;31(4):365-9,
http://dx.doi.org/10.3109/02844319709008985.
http://dx.doi.org/10.3109/02844319709008...
42. Anani PA, Hessler C. Breast hamartoma with invasive ductal
carcinoma. Report of two cases and review of the literature. Pathol Res Pract.
1996;192(12):1187-94,
http://dx.doi.org/10.1016/S0344-0338(96)80149-X.
http://dx.doi.org/10.1016/S0344-0338(96)...
-4343. Garfein CF, Aulicino MR, Leytin A, Drossman S, Hermann G,
Bleiweiss IJ. Epithelioid cells in myoid hamartoma of the breast: a potential
diagnostic pitfall for core biopsies. Arch Pathol Lab Med.
1996;120(7):676-80.).
The incidence of breast hamartoma among benign breast tumors was reported to be 4.8%
by Charpin et al. (11. Charpin C, Mathoulin MP, Andrac L, Barberis J, Boulat J,
Sarradour B, et al. Reappraisal of breast hamartomas. A morphological study
of 41 cases. Pathol Res Pract. 1994;190(4):362-71,
http://dx.doi.org/10.1016/S0344-0338(11)80408-5.
http://dx.doi.org/10.1016/S0344-0338(11)...
) in a large case series
in 1994. In total, 27 hamartomas (3.1%) were diagnosed among 864 benign breast
tumors at our center over the last ten years. In the literature (2323. Ruiz-Tovar J, Reguero-Callejas ME, Arano-Bermejo JI,
Gonzalez-Palacios F, Cabanas-Navarro L. [Mammary hamartoma]. Cir Esp.
2006;79(3):186-8,
http://dx.doi.org/10.1016/S0009-739X(06)70848-X.
http://dx.doi.org/10.1016/S0009-739X(06)...
,2828. Filho OG, Gordan AN, Mello Rde A, Neto CS, Heinke T. Myoid
hamartomas of the breast: report of 3 cases and review of the literature.
Int J Surg Pathol. 2004;12(2):151-3.,2929. Lee EH, Wylie EJ, Bourke AG, Bastiaan De Boer W. Invasive ductal
carcinoma arising in a breast hamartoma: two case reports and a review of the
literature. Clin Radiol. 2003;58(1):80-3,
http://dx.doi.org/10.1053/crad.2003.1133.
http://dx.doi.org/10.1053/crad.2003.1133...
,3131. Tse GM, Law BK, Pang LM, Cheung HS. Ductal carcinoma in situ
arising in mammary hamartoma. J Clin Pathol. 2002;55(7):541-2,
http://dx.doi.org/10.1136/jcp.55.7.541.
http://dx.doi.org/10.1136/jcp.55.7.541...
,3333. Herbert M, Sandbank J, Liokumovich P, Yanai O, Pappo I, Karni T,
et al. Breast hamartomas: clinicopathological and immunohistochemical studies of
24 cases. Histopathology. 2002;41(1):30-4,
http://dx.doi.org/10.1046/j.1365-2559.2002.01429.x.
http://dx.doi.org/10.1046/j.1365-2559.20...
,3434. Tse GM, Law BK, Ma TK, Chan AB, Pang LM, Chu WC, et al.
Hamartoma of the breast: a clinicopathological review. J Clin Pathol.
2002;55(12):951-4, http://dx.doi.org/10.1136/jcp.55.12.951.
http://dx.doi.org/10.1136/jcp.55.12.951...
,3737. Wahner-Roedler DL, Sebo TJ, Gisvold JJ. Hamartomas of the
breast: clinical, radiologic, and pathologic manifestations. Breast J.
2001;7(2):101-5,
http://dx.doi.org/10.1046/j.1524-4741.2001.007002101.x.
http://dx.doi.org/10.1046/j.1524-4741.20...
,4040. Chiacchio R, Panico L, D'Antonio A, Delrio P, Bifano D,
Avallone M, et al. Mammary hamartomas: an immunohistochemical study of ten
cases. Pathol Res Pract. 1999;195(4):231-6,
http://dx.doi.org/10.1016/S0344-0338(99)80039-9.
http://dx.doi.org/10.1016/S0344-0338(99)...
41. Blomqvist L, Malm M, Fernstad R. Hamartoma of the breast:
surgical treatment and reconstruction. Case report. Scand J Plast
Reconstr Surg Hand Surg. 1997;31(4):365-9,
http://dx.doi.org/10.3109/02844319709008985.
http://dx.doi.org/10.3109/02844319709008...
-4242. Anani PA, Hessler C. Breast hamartoma with invasive ductal
carcinoma. Report of two cases and review of the literature. Pathol Res Pract.
1996;192(12):1187-94,
http://dx.doi.org/10.1016/S0344-0338(96)80149-X.
http://dx.doi.org/10.1016/S0344-0338(96)...
) the average age of the patients with breast hamartoma
ranged between 33.5 and 66.5 years. In our study, the age range was from 19-56 years
and the mean age was 41.8 years. There was only one patient under 20 years of age
and 7 patients were over 50 years of age.
The etiopathogenesis of breast hamartomas is not clear but they are thought to result
from dysgenesis (4444. Guray M, Sahin AA. Benign breast diseases: classification,
diagnosis, and management. Oncologist. 2006;11(5):435-49,
http://dx.doi.org/10.1634/theoncologist.11-5-435.
http://dx.doi.org/10.1634/theoncologist....
) rather than a true
tumorous process. However, female sex steroid hormones (3434. Tse GM, Law BK, Ma TK, Chan AB, Pang LM, Chu WC, et al.
Hamartoma of the breast: a clinicopathological review. J Clin Pathol.
2002;55(12):951-4, http://dx.doi.org/10.1136/jcp.55.12.951.
http://dx.doi.org/10.1136/jcp.55.12.951...
) have been implicated in the development of breast
hamartomas. In one study, Herbert et al. (3333. Herbert M, Sandbank J, Liokumovich P, Yanai O, Pappo I, Karni T,
et al. Breast hamartomas: clinicopathological and immunohistochemical studies of
24 cases. Histopathology. 2002;41(1):30-4,
http://dx.doi.org/10.1046/j.1365-2559.2002.01429.x.
http://dx.doi.org/10.1046/j.1365-2559.20...
)
reported estrogen receptor (ER) and progesterone receptor (PR) positivity in
epithelial cells and stromal cells in all 24 cases with breast hamartomas.
Additionally, there are no clear data on the source of smooth muscle for myoid
hamartomas, but this muscle could derive from vessels, the nipples, undifferentiated
breast stromal tissue or myoepithelial cells (44. Altermatt HJ, Gebbers JO, Laissue JA. Multiple hamartomas of the
breast. Appl Pathol. 1989;7(2):145-8.,1414. Kajo K, Zubor P, Danko J. Myoid (Muscular) Hamartoma of the
Breast: Case Report and Review of the Literature. Breast Care (Basel).
2010;5(5):331-4, http://dx.doi.org/10.1159/000321341.
http://dx.doi.org/10.1159/000321341...
). Another hypothesized
smooth muscle source is the metaplasia of breast stromal cells (1414. Kajo K, Zubor P, Danko J. Myoid (Muscular) Hamartoma of the
Breast: Case Report and Review of the Literature. Breast Care (Basel).
2010;5(5):331-4, http://dx.doi.org/10.1159/000321341.
http://dx.doi.org/10.1159/000321341...
) into smooth muscle cells. The existence of
CD34 on smooth muscles (2121. Murugesan JR, Joglekar S, Valerio D, Bradley S, Clark D, Jibril
JA. Myoid hamartoma of the breast: case report and review of the literature.
Clin Breast Cancer. 2006;7(4):345-6,
http://dx.doi.org/10.3816/CBC.2006.n.050.
http://dx.doi.org/10.3816/CBC.2006.n.050...
,3434. Tse GM, Law BK, Ma TK, Chan AB, Pang LM, Chu WC, et al.
Hamartoma of the breast: a clinicopathological review. J Clin Pathol.
2002;55(12):951-4, http://dx.doi.org/10.1136/jcp.55.12.951.
http://dx.doi.org/10.1136/jcp.55.12.951...
) is an important sign of the metaplasia of
stromal cells into smooth muscle cells, but there are no clear data on this subject.
Myoid hamartomas stain strongly positive for SMA, desmin and vimentin (88. Mizuta N, Sakaguchi K, Mizuta M, Imai A, Nakatsukasa K, Morita M,
et al. Myoid hamartoma of the breast that proved difficult to diagnose: a case
report. World J Surg Oncol. 2012;10:12.) by immunohistochemical staining. In our
study, only 1 patient of 27 was diagnosed with myoid hamartoma and
immunohistochemical staining for desmin, SMA and CD34 was positive, consistent with
the literature.
With increasing social awareness and the use of numerous diagnostic procedures on
breast lumps, including mammography, ultrasound, and MRI, it is expected that more
hamartomas will be reported. Breast hamartomas have a typical mammographic
appearance. The radiolucent lesions include fat; various amounts of fibrous and
adenomatous tissues, with smooth rims; and occasionally, a thin capsule (44. Altermatt HJ, Gebbers JO, Laissue JA. Multiple hamartomas of the
breast. Appl Pathol. 1989;7(2):145-8.). US evaluates breast hamartomas as having
sonolucent fat and a heterogeneous internal echo pattern with echogenic fibrous
components (4545. Kopans DB. Pathologic, mammographic and sonographic correlation.
In: Kopans DB. Breast Imaging, 2nd ed. Boston, Massachusetts: Lippincott-Raven,
1998:558-560.). Alongside US, MRI
characteristically shows a smooth and well-defined hypointense rim, internal
heterogeneous enhancement and the presence of fat density (3434. Tse GM, Law BK, Ma TK, Chan AB, Pang LM, Chu WC, et al.
Hamartoma of the breast: a clinicopathological review. J Clin Pathol.
2002;55(12):951-4, http://dx.doi.org/10.1136/jcp.55.12.951.
http://dx.doi.org/10.1136/jcp.55.12.951...
). In the present study, US was used for all patients as the
first imaging procedure and in addition to US, mammography was utilized for 6
patients. MRI was used only for 1 patient with IDC.
The pathologic features of hamartomas are not well known. The original definition
consisted of a fibrous fatty stroma including various amounts of epithelial
elements, as well as nodular lesions. The fibrous and fatty tissues were used for
the early classification of hamartomas: McGuire et al. (4646. McGuire LI, Cohn D. Hamartoma of the breast.
Aust N Z J Surg. 1991;61(9):713-6.) classified breast hamartomas as fibrous, fatty or
fibrous-fatty. Jones et al. (4747. Jones MW, Norris HJ, Wargotz ES. Hamartomas of the breast. Surg
Gynecol Obstet. 1991;173(1):54-6.) suggested 4
classification groups for breast hamartomas: encapsulated fibrocystic changes,
fibroadenoma with a fibrous stroma, fibroadenoma-like and surrounded adenolipoma.
These classifications were not widely accepted. No criteria with detailed
explanations of breast hamartomas are used by pathologists. Fechner et al. (4848. Fechner RE. Fibroadenoma and related lesions. In:Page DL,
Anderson TJ, eds. Diagnostic Histopathology of the Breast: Edinburgh: Churchill
Livingstone; 1987:72-85.) described the lobular distribution and the
presence of fat in breast hamartomas as distinguishing features compared with
fibroadenomas. A fibrotic stroma surrounding the lobules and extending into the
interlobular areas, causing obliteration, is the most frequently observed feature;
most authors (22. Fisher CJ, Hanby AM, Robinson L, Millis RR. Mammary
hamartoma—a review of 35 cases. Histopathology. 1992;20(2):99-106,
http://dx.doi.org/10.1111/j.1365-2559.1992.tb00938.x.
http://dx.doi.org/10.1111/j.1365-2559.19...
,4747. Jones MW, Norris HJ, Wargotz ES. Hamartomas of the breast. Surg
Gynecol Obstet. 1991;173(1):54-6.,4848. Fechner RE. Fibroadenoma and related lesions. In:Page DL,
Anderson TJ, eds. Diagnostic Histopathology of the Breast: Edinburgh: Churchill
Livingstone; 1987:72-85.) refer to this
feature as interlobular fibrosis. However, interlobular fibrosis is not specific to
breast hamartomas. In sclerosing hyperplasia, an increasing number of interlobular
glands are observed as round masses with interlobular fibrosis, which can mimic
breast hamartomas. The absence of fatty tissue in the stromal structure is often
associated with fibroadenomas, so this condition can be distinguished from
sclerosing lobular hyperplasia and breast hamartomas (3333. Herbert M, Sandbank J, Liokumovich P, Yanai O, Pappo I, Karni T,
et al. Breast hamartomas: clinicopathological and immunohistochemical studies of
24 cases. Histopathology. 2002;41(1):30-4,
http://dx.doi.org/10.1046/j.1365-2559.2002.01429.x.
http://dx.doi.org/10.1046/j.1365-2559.20...
,3434. Tse GM, Law BK, Ma TK, Chan AB, Pang LM, Chu WC, et al.
Hamartoma of the breast: a clinicopathological review. J Clin Pathol.
2002;55(12):951-4, http://dx.doi.org/10.1136/jcp.55.12.951.
http://dx.doi.org/10.1136/jcp.55.12.951...
,4545. Kopans DB. Pathologic, mammographic and sonographic correlation.
In: Kopans DB. Breast Imaging, 2nd ed. Boston, Massachusetts: Lippincott-Raven,
1998:558-560.). Fischer et al. (22. Fisher CJ, Hanby AM, Robinson L, Millis RR. Mammary
hamartoma—a review of 35 cases. Histopathology. 1992;20(2):99-106,
http://dx.doi.org/10.1111/j.1365-2559.1992.tb00938.x.
http://dx.doi.org/10.1111/j.1365-2559.19...
) defined the presence of a pseudoangiomatous stroma
particularly in breast hamartomas. This condition was revealed to be a consistent
observation in subsequent reports and the incidence rate (3434. Tse GM, Law BK, Ma TK, Chan AB, Pang LM, Chu WC, et al.
Hamartoma of the breast: a clinicopathological review. J Clin Pathol.
2002;55(12):951-4, http://dx.doi.org/10.1136/jcp.55.12.951.
http://dx.doi.org/10.1136/jcp.55.12.951...
,4949. Daya D, Trus T, D'Souza TJ, Minuk T, Yemen B. Hamartoma of
the breast, an underrecognized breast lesion. A clinicopathologic and
radiographic study of 25 cases. Am J Clin Pathol.
1995;103(6):685-9.) varied between 16
and 71% (3434. Tse GM, Law BK, Ma TK, Chan AB, Pang LM, Chu WC, et al.
Hamartoma of the breast: a clinicopathological review. J Clin Pathol.
2002;55(12):951-4, http://dx.doi.org/10.1136/jcp.55.12.951.
http://dx.doi.org/10.1136/jcp.55.12.951...
). We also found the rate to be
25.9% in our series, in line with the literature.
Malignancies associated with breast hamartomas (99. Kai M, Tada K, Tamura M, Gomi N, Horii R, Akiyama F, et al.
Breast cancer associated with mammary hamartoma. Breast Cancer.
2012;19(2):183-6, http://dx.doi.org/10.1007/s12282-009-0147-3.
http://dx.doi.org/10.1007/s12282-009-014...
,1111. Choi N, Ko ES. Invasive ductal carcinoma in a mammary hamartoma:
case report and review of the literature. Korean J Radiol.
2010;11(6):687-91.,1616. Pervatikar SK, Rao R, Dinesh US, Parameswaraiah S. Large mammary
hamartoma with focal invasive ductal carcinoma. Indian J Pathol
Microbiol. 2009;52(2):249-51.,2929. Lee EH, Wylie EJ, Bourke AG, Bastiaan De Boer W. Invasive ductal
carcinoma arising in a breast hamartoma: two case reports and a review of the
literature. Clin Radiol. 2003;58(1):80-3,
http://dx.doi.org/10.1053/crad.2003.1133.
http://dx.doi.org/10.1053/crad.2003.1133...
,3131. Tse GM, Law BK, Pang LM, Cheung HS. Ductal carcinoma in situ
arising in mammary hamartoma. J Clin Pathol. 2002;55(7):541-2,
http://dx.doi.org/10.1136/jcp.55.7.541.
http://dx.doi.org/10.1136/jcp.55.7.541...
,4242. Anani PA, Hessler C. Breast hamartoma with invasive ductal
carcinoma. Report of two cases and review of the literature. Pathol Res Pract.
1996;192(12):1187-94,
http://dx.doi.org/10.1016/S0344-0338(96)80149-X.
http://dx.doi.org/10.1016/S0344-0338(96)...
,5050. Kronsbein H, Bassler R. [Metaplasias and malignant
transformations in hamartomas of the breast]. Verh Dtsch Ges Pathol.
1985;69:310-5.) are rarely observed, and
only a few studies have reported invasive breast cancer associated with breast
hamartoma. We identified IDC in only 1 case and LCIS in 1 other case. Although
epithelial hyperplasia is not a characteristic feature of breast hamartomas, ductal
epithelial hyperplasia was identified in 6 cases (22.2%). Wahner-Rodler et al.
(3737. Wahner-Roedler DL, Sebo TJ, Gisvold JJ. Hamartomas of the
breast: clinical, radiologic, and pathologic manifestations. Breast J.
2001;7(2):101-5,
http://dx.doi.org/10.1046/j.1524-4741.2001.007002101.x.
http://dx.doi.org/10.1046/j.1524-4741.20...
) reported this incidence rate as 26%
in their large case series. These high coincidence rates are very important.
Patients with a certain or suspected diagnosis of hamartoma must be operated and
these patients should never be followed, even if their tumors are small.
In conclusion, we present our institutional experience with breast hamartomas in 27 patients. Although breast hamartomas are rare, benign lesions, these lesions can reach large sizes. A diagnosis can be made by core needle biopsy, along with an appropriate correlation of clinical and radiologic features. Breast hamartomas may be underdiagnosed because pathologists may categorize these lesions as fibroadenomas instead of hamartomas. Thus, the true incidence may be higher than the literature indicates.
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-
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-
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-
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-
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-
No potential conflict of interest was reported.
Publication Dates
-
Publication in this collection
Aug 2014
History
-
Received
25 Nov 2013 -
Reviewed
16 Jan 2013 -
Accepted
4 Feb 2014