Changing Clinical Picture of Mammary Hamartoma Carol E. H. Scott-Conner, MD, PhD, Cynthia Powers, MD, Charu Subramony, MD, Ralph H. Didlake, MD, Jackson,Mississippi
During a 6-year period, we identified 12 patients (age range: 16 to 7 2 years) with a histologie or mammographic diagnosis of mammary hamartoma. The lesion was found in 9 of 4 4 1 open breast biopsy specimens ( 2 % ) and was identified radiograpbically in 5 of 8 , 1 2 2 mammographie examinations (less than 1%). Two groups of patients were identified. Three patients under 3 0 years of age underwent the excision of small palpable lesions found on pathologic examination to be mammary hamartomas (group I ) . In nine patients over age 30, masses were identified or confirmed on mammography (group II). Five lesions showed the classic mammographic appearance of a mammary hamartoma (a circumscribed tumor of mixed soft tissue and fatty density), and the other four were indeterminate. Presentation in these older women who had a relatively high incidence of atypical mammographic findings mandates that biopsy be performed.
ammary hamartoma is usually described as a rare, benign tumor with a characteristic mammographic appearance. This lesion can be diagnosed mammographically with a high degree of assurance when a circumscribed mass consisting of both soft tissue and fatty elements is found. This abnormality was unexpectedly encountered in a postmenopausal woman with a mammographically indeterminate lesion when a biopsy was performed. This case prompted the present review.
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PATIENTS AND M E T H O D S The charts of all patients with mammary hamartoma diagnosed by histologic examination and/or mammography between January 1, 1986, and December 31, 1991, at the University of Mississippi Medical Center were reviewed. Pathologic reports and histologic sections of lesions classified as either mammary hamartoma or adenolipoma were reviewed by a pathologist (CS) to confirm that the lesions met the criteria for mammary hamartoma. Mammograms were reviewed by a radiologist (CP) and evaluated to determine lesion size, the presence or absence of fat within the lesion, margin characteristics, and correlation with physical findings.
RESULTS A total of 441 open breast biopsies and 8,122 mammographic examinations were performed during the 6year study period. Thirteen patients with the pathologic or mammographic diagnosis of mammary hamartoma were identified initially, 12 of whom had unilateral, solitary lesions. One patient had two masses in the right breast, one of which was related to a previous biopsy. Seven women presented with a palpable mass. Six lesions were detected by mammography, three of which were nonpalpable. The other three lesions were soft masses that were missed on initial physical examination but which could be palpated after mammographic correlation. Ten patients had mammography, 3 underwent breast ultrasonography, and 3 had no imaging study. The radiographs of one patient with a mammographic diagnosis of mammary hamartoma could not be located for review, and this patient was excluded from the study group. An open biopsy was performed in nine women. The final study group consisted of 12 women, ranging in age from 16 to 72 years (mean: 42 years). The patients were divided into two groups based on age and clinical presentation. group I: Three young women (ages 16, 25, and 27 From the Departmentsof Surgery (CEHSC, RHD), Radiology(CP), and Pathology (CS), Universityof Mississippi School of Medicine, years) presented with small palpable tumors that were Jackson, Mississippi. located relatively close to the surface and characterized Requests for reprints should be addressed to Carol E. H. Scott- as firm, ovoid, mobile, well-demarcated masses. The dinConner, MD, PhD, Departmentof Surgery,2500 North State Street, ical impression in all three cases was fibroadenoma. No Jackson, Mississippi39216-4505. Manuscript submitted January 21, 1992,and acceptedin revised imaging procedures were performed. Excisional biopsy form April 15, 1992. revealed well-circumscribed lesions ranging in size from 2 208
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Figure 1. Typical mammographic appearance of mammary hamartoma. This craniocaudal m a m m o g r a m
shows a subareolar mass consisting of soft tissue and lipomatous elements.
Figure 2. A mammographically indeterminate lesion histologically proven to be mammary hamartoma. The specimen radiograph shows a homogeneous soft tissue density.
• 2 • 3 cm to 2 • 3.5 • 4 cm. Histologically, these lesions consisted of fibrous and ductal tissue (with a predominance of fibrous tissue). Minimal fatty tissue was noted within these specimens. Group II: Nine older women (ages: 31 to 72 years; mean: 48.3 years) had abnormal mammographic results and/or palpable masses. Six women underwent a combination of mammography and excisional biopsy. Four patients had solitary, circumscribed masses found by mammography, which ranged in size from 1 • 2 cm to 4 • 4 cm. One lesion had mixed fatty and soft tissue elements typical of hamartoma (Figure 1); the other three lesions had relatively homogeneous soft tissue density and were considered mammographically indeterminate (Figure 2).
A fifth patient who underwent mammography was found to have a questionable lipomatous lesion in the area of a palpable mass. The sixth patient in this group had two lesions in one breast. One of these was a homogeneously dense mass at the site of a previous biopsy, which was subsequently proven to be related to the prior surgery; the second lesion was a 1.5 • 3.5-cm mixed fatty and soft tissue mass considered typical of hamartoma. The results of ultrasonography demonstrated solid lesions with uniform low-level echogenicity in two patients. Four masses were palpable upon careful examination. Three were soft, mobile, well demarcated, and similar in texture to the surrounding breast tissue. The fourth lession was firm and had a texture suggestive of malignancy
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Figure 3. Ultrasound scan demonstrated mixed hyperechoic, hypoechoic, and anechoic zones within the mass.
Figure 4. Gross appearance of mammary hamartoma. The well-circumscribed yellowish tumor is lensshaped or ovoid and soft.
on physical examination. Preoperative radiographic needle localization was used for the two nonpalpable masses. Surgical specimens ranged in size from 1.5 • 1.5 X 1.5 cm to 2.7 • 4.0 • 4.5 cm. All lesions were grossly ovoid or lens shaped, well encapsulated, and soft. A significant amount of adipose tissue was identified in only three cases. Biopsy specimens were not obtained in the other three women. All three met the radiographic criteria for mammary hamartoma (the lesion had to be well circumscribed and consist of an admixture of lipomatous and soft tissue elements). A surrounding capsule was seen in one of these lesions. All masses were solitary and ranged in size from 3 • 4.5 cm to 4.5 • 6.5 cm. Ultrasound was performed in one case and demonstrated a solid mass of mixed echogenicity (Figure 3). Two masses were palpable and were soft and well demarcated. Two patients, who were offered surgical excision but declined, have been lost to subsequent follow-up. One lesion has been mammographically stable for 53 months.
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COMMENTS In 1971, Arrigoni et al [1] defined mammary hamartomas as "a small group of lesions characterized microscopically by a mixture of epithelial and mesenchymal components" that did not correspond histologically to any of the recognized benign or malignant tumors of the breast. They noted that histologically these lesions resemble normal female breast tissue. This lesion has been referred to variously as an adenolipoma, a fibroadenolipoma, and a postlactational breast tumor [1-11]. Hamartomas of the breast are typically soft and well circumscribed (Figure 4). Because they are similar in firmness and texture to surrounding normal breast tissue, they are frequently difficult to palpate. The impression at surgery is that of a well-encapsulated lesion that readily shells out of the breast. Histologically (Figure 5), variable amounts of fat, fibrous tissue, ductal tissue, and, occasionally, smooth muscle and cartilage are seen within the tumor [1,2,5,12,I3]. Although some reports state that these lesions are encapsulated [1-2,14-16], other investi-
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Figure 5. Histologic examination of a mammary hamartoma demonstrates a breast Iobule with dense fibrous tissue and adipose tissue, (hematoxylin and eosin stain, 40;< magnification).
gators believe that the tumor lacks a true capsule [4,5,12]. Incomplete excision or fragmentation of the mass may cause the diagnosis to be missed microscopically because of the histologic similarity to normal breast tissue [13,15]. Mammary hamartomas are true hamartomas and thus are not premalignant lesions. However, they contain glandular elements of breast tissue and may undergo malignant changes just as normal breast tissue does. Lobular carcinoma in situ has been reported in one mammary hamartoma in which microcalcifications were noted mammographically [17]. These tumors may present with the asymmetric enlargement of one breast, noted at cessation of lactation or at menopause [6-11]. It has been postulated that the normal involution of the surrounding breast tissue makes the tumor more prominent [4-13]. Enucleation of large tumors can be accomplished with surprising ease, and typically there is little deformity of the breast after removal of even a very large mass. Early reports of this lesion describe predominately massive tumors [7-9]. The phrase "breast within breast" aptly describes the soft texture, common pattern of histology, and distortion of remaining normal breast tissue classically associated with these large tumors [18]. In contrast to these large tumors, our series had a predominance of small lesions. The younger subgroup of our patients appeared clinically to have fibroadenomas. In our older subgroup, lesions were commonly demonstrated on mammography but the typical mammographic appearance was not always seen. Most reports of the mammographic appearance of hamartoma describe a characteristic picture [4,5,16-20]. The lesion is seen as a circumscribed mass composed of both soft tissue and lipomatous elements, which is said to
resemble cut salami or cauliflower. The lesion may appear to be surrounded by a thin, fibrous capsule. These masses often reach several centimeters in size and may appear to fill the breast or deform the adjacent parenchymal elements. Recognition of the mixed density of the lesion is the key to mammographic diagnosis and enables one to predict that the lesion is benign with a high degree of assurance [5,13]. When the mammographic appearance is classic, many researchers believe that biopsy examination can be avoided [13,19]. One patient in this series with the characteristic appearance of hamartoma has been mammographically stable for 53 months. A second patient was stable for 13 months before a biopsy specimen was obtained. If biopsy specimens of a tumor with the typical appearance of mammary hamartoma are obtained, good communication between the radiologist, surgeon, and pathologist can help to insure that the diagnosis is not missed. Of the six women in our series who underwent both mammography and biopsy, only two had radiographic features typical for mammary hamartoma (three additional patients with classic mammographic findings did not undergo biopsy). This is similar to the experience of Helvie et al [19] who found only 2 of 17 pathologically proven hamartomas had a classic radiographic appearance. In summary, small mammary hamartomas may be noted with increasing frequency through the use of screening mammography and careful breast physical examination. In our recent experience, 9 of 441 (2%) open breast biopsies and 5 of 8,122 (less than 1%) mammograms performed over the past 6 years showed this lesion. Although mammary hamartoma is a benign lesion, the frequent indeterminate mammographic appearance and
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presentation in an older age group m a k e biopsy necessary. Excisional biopsy is the procedure of choice. Because these tumors m a y be difficult to palpate, prebiopsy needle localization m a y be required.
Brustdrfisengewebes durch echte Adenome und Fibroadenome. Beitr Pathol Anat 1928; 81: 1-44. 8. LeGal Y. Adenomas of the breast: relationship of adenofibromas to pregnancy and lactation. Am Surg 1961; 27: 14-22. 9. Menges V, Wuster K. Fibro-Adeno-Lipom der Mamma. Fortschr Rontgenstr 1976; 125: 192-3. The authors have focused on an entity not widely 10. Linell F, Ostberg G, Soderstom J, et al. Breast hamartomas: an recognized and therefore probably underdiagnosed. important entity of mammary pathology. Virchows Arch 1979; With the increased utilization of mammography, sur- 383: 253-64. geons need to be alerted to the typical appearance of 11. Duchatelle V, Auberger E, Amouroux J. Hamartomes du sein: breast hamartomas and adjust their clinical judgment a propose de 14 observations histologiquement contr01e~. Ann Pathol (Paris) 1986; 6: 335-9. accordingly. 12. Ljungqvist V, Andersson I, Hildell J. Mammary hamartoma, a benign breast lesion. Acta Chir Stand 1979; 145: 227-30. 13. Hessler CH, Schnyder P, OzzeUo L. Hamartoma of the breast: REFERENCES 1. Arrigoni MG, Dockerty MB, Judd ES. The identification and diagnostic observation of 16 cases. Radiology 1978; 126: 95-8. treatment of mammary hamartoma. Surg Gynecol Obstet 1971; 14. Jones MW, Norris HJ, Wargotz ES. Hamartomas of the breast. Surg Gynecol Obstet 1991; 173: 54-6. 133: 577-82. 2. Spalding JE. Adenolipoma and lipoma of the breast. Guys Hosp 15. Petrik PK. Mammary hamartoma [letter]. Am J Surg Pathol 1987; 11: 234-5. Rep 1945; 94: 80-4. 16. Vas W, Gagic N, Davidson JW. Xeromammographic diagnosis 3. Durso EA. Mammographic findings in adenolipoma. JAMA of mammary hamartoma. J Can Assoc Radiol 1980; 31: 208-9. 1971; 218: 886. 4. Andersson I, Hildell J, Linell F, Ljungvist U. Mammary hamar- 17. Mendiola H, Henrik-Nielsen R, Dyreborg U, Blichert-Toft M, toms. Acta Radiol (Diagn) 1979; 20: 712-20. A1-Hariri JA. Lobular carcinoma in situ occurring in adenolipoma 5. Crothers JG, Butler NF, Fortt RW, Gravelle IH. Fibroadenoli- of the breast. Report of a case. Acta Radiol Diag 1982; 23: 503-5. 18. Kronsbein VH, B~issler R, Daniels HV. Das Hamartom der poma of the breast. Br J Radiol 1985; 58: 191-202. 6. Hogeman KE, Ostberg G. Three cases of post-lactational breast Mamma. Fortschr Rfntgenstr 1983; 138: 613-9. tumour of a peculiar type. Acta Pathol Microbiol Scand 1968; 73: 19. Helvie MA, Adler DD, Rebner M, Oberman HA. Breast hamartomas: variable mammographic appearance. Radiology 169-76. 7. Prym P. Pseudoadenome, Adenome und Mastome der weibli- 1989; 170: 417-21. then Brustdrfise; Studien fiber die Entstehung umschriebener aden- 20. Riveros M, Cubillo LA, Perotta F, Solalinde V. Hamartoma of om~hnlicher Herde in der Mamma und fiber die Nachahmung der the breast. J Surg Oncol 1989; 42: 197-200.
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