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1 AVON Breast Comprehensive Center and Department of Radiology, Massachusetts
General Hospital, WACC 219R, 15 Parkman St., Boston, MA 02114.
2 Department of Pathology, Massachusetts General Hospital, Boston, MA.
3 Department of Surgical Oncology, Massachusetts General Hospital, Boston,
MA.
Received June 29, 2005;
accepted after revision September 28, 2005.
Address correspondence to D. Georgian-Smith
(dgeorgiansmith{at}partners.org).
Abstract
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We encountered a mammographically calcified breast mass in a 30-year-old man. It was initially thought to be comedo-type ductal carcinoma in situ because of the dense calcifications, but sonography and MRI suggested a highly vascular lesion. The final pathologic diagnosis was hemangioma.
Conclusion
Vascular tumors of the breast occur infrequently and are even more rare in males. The clinical and radiologic diagnosis of breast hemangioma is often difficult, but different imaging techniques, when used together, can provide important information for differential diagnosis and management. A biopsy is required.
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Male breast cancer was an important consideration in this case. It accounts for fewer than 1% of all cancers in men and has the same prognosis as breast cancer in women [3]. Most male breast cancers are invasive ductal cancer, with most of the remaining tumors being medullary and papillary tumors [4]. It generally manifests as a hard, painless subareolar mass eccentric to the nipple, with occasional nipple discharge or ulceration [5]. Breast cancer is usually diagnosed in men at or around the age of 60 years. The patient's young age, stability of breast mass size over 4 years, and absence of family history makes cancer unlikely, although this possibility cannot be entirely excluded.
On mammography, male breast cancer generally presents as a dense mass with variable border patterns [1, 6, 7]. Secondary features include skin thickening, nipple retraction, and axillary lymphadenopathy [1, 6]. The mammographic appearance in this case is significantly larger than the usual presentation of breast cancer, and if the mass had been malignant, it would have likely been associated with metastases because of its large size and alleged 4-year history. This patient was otherwise healthy, a point that argued against a clinical presentation of metastases.
It was initially thought that the calcifications might indicate an in situ process, such as comedo-type ductal carcinoma, despite the large mass. Calcifications are infrequent in male breast cancer, reported in 13-30% of cases [7]; they have been noted to appear coarser and less frequently linear compared with female breast cancer [2, 6, 7]. Therefore, the presence of calcifications did not rule out breast cancer.
Other breast masses associated with calcifications include vascular lesions (e.g., hemangioma), trauma with fat necrosis, tuberculosis, and parasitic infections.
Hemangiomas contain calcifications secondary to phlebolith formation. A review of the literature indicates that mammographic findings of a breast hemangioma are nonspecific and include a normal mammogram or a well-circumscribed mass with or without calcifications. Calcifications of hemangiomas are usually punctate, but coarse and bizarre calcifications have also been described [8-10]. The appearance of this patient's mammogram was consistent with, but not specific to, a hemangioma (Figs. 1A, and 1B). In retrospect, the round calcifications were most likely phleboliths.
Another vascular lesion of the breast, arteriovenous fistula (AVF), is exceedingly rare. It can be congenital or acquired through biopsy or chest trauma [11]. Only a few cases are reported in the literature, and the mammographic appearance of AVFs is not well documented. The lack of bruit or palpable thrill on physical examination made this diagnosis unlikely, although sonography is a better test for evaluating AVFs.
A breast mass with calcification can also be attributed to trauma and dystrophic calcium deposition due to fat necrosis. The predominant features on mammograms are radiolucent oil cysts and round or asymmetric opacities that may calcify, producing curvilinear, punctate, and heterogeneous calcifications [12]. Hematomas in the chronic phase may also contain dystrophic calcification. In this case, the diagnosis of an old hematoma was unlikely without the patient's knowledge of trauma, but was possible if based on the imaging only.
Breast tuberculosis is a rare form of tuberculosis typically found in women from endemic areas and has been reported in males. Patients may present with masses, mastalgia, nipple discharge, and skin sinus [13, 14]. In addition to mass lesions, duct ectasia, skin thickening, nipple retraction, and macrocalcifications are other features seen on mammography. Intramammary or axillary adenopathy are associated findings [13, 14]. Only a minority of patients have concomitant involvement of other organs, such as the lung [13]. The mammographic appearance of this patient's left breast mass was consistent with breast tuberculosis, even though the patient had a negative chest radiograph. However, the absence of any palpable adenopathy on examination or history of known tuberculosis made this diagnosis less likely.
Another rare cause of male breast mass is parasitic infection. There are reported cases of breast masses caused by schistosomiasis, paragonimiasis, and myiasis, which are all endemic to Central and South America [15-17]. In one series of five Brazilian patients with cutaneous myiasis, ill-defined masses on mammography were seen in all patients and linear microcalcifications were found in two (40%) [17]. It has not been documented in the literature whether calcifications are seen in other forms of parasitic infections. Inflammatory signs and pain often accompany infections. The lack of these symptoms in this patient argued against this diagnosis.
Other unusual breast tumors that have been reported in males include lymphoma, lipoma, hamartoma, osteosarcoma, and metastasis. Untreated lymphoma has not been reported with calcifications, to our knowledge. Lipomatous tumors, such as the fibrolipoma or liposarcoma, are commonly associated with fat-density tissue, which this mass did not have. Although hamartomas can be diagnosed without fat [18], the extent of the dense tissue is much larger than expected for a hamartoma, in our experience. In addition, hamartoma is considered an entity almost exclusively in females, and to our knowledge, only one case has been reported in a male [19].
Osteosarcoma of the breast is extremely rare. It often presents as a large mass with relatively well-defined margins and lobulated borders containing coarse, dense calcifications [20]. However, osteosarcoma is an aggressive tumor and therefore an unlikely diagnosis given the 4-year stability. The patient had no known primary tumor and was in good health, making metastatic disease unlikely. These entities were not seriously considered in our differential diagnosis.
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Hemangioma remained in the differential diagnosis. There are few published cases of the sonographic findings of hemangioma: It can have well-defined or ill-defined borders; can be hypoechoic or hyperechoic with distal shadowing [9, 10]; and can have internal bright echoes that likely represent calcifications within them [9, 10, 21], as was noted in our patient (Fig. 2).
The presence of a solid mass otherwise did not rule out the remaining diagnostic possibilities. Heterogeneous hypoechoic or complex cystic lesions can be seen in male breast cancer [22], but usually in smaller focal masses. As noted previously, the diffuse extent made an invasive male breast carcinoma unlikely, given there were no signs of metastatic disease. Based on the sonographic appearance alone, hamartoma is still a consideration. However, as previously discussed, the extensiveness of this lesion and rarity of hamartomas in males make it an unlikely diagnosis.
Tuberculosis and parasitic involvement of the breast often present as hypoechoic masses on sonography. Hyperechoic mass representing larvae surrounded by a hypoechoic cavity is described in myiasis and may be seen with other types of larvae [17].
A hypoechoic, homogeneous or heterogeneous well-defined mass is the most common sonographic finding in patients with malignant lymphoma [23]. A pseudocystic serpentine mass appearance has also been suggested for breast lymphoma [24]. Although lymphoma can involve the breast extensively and appear heterogeneous, as in our patient, the presence of calcifications made a diagnosis of lymphoma unlikely.
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Expert Discussion (Dr. Georgian-Smith)
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An important differential consideration is a malignant vascular tumornamely, angiosarcoma. On MRI, angiosarcomas are of variable intensity on T1-weighted images, are high intensity on T2-weighted images, and show contrast enhancement [28]. These imaging features make them difficult to distinguish from hemangiomas. Definitive diagnosis requires pathologic examination.
Intense contrast enhancement also occurs in breast carcinoma. Invasive ductal carcinoma is virtually always manifested as a focal, avidly enhancing mass with irregular, spiculated, or sometimes smooth borders. Rim enhancement and enhancing internal septations are particularly suspicious. T2 signal of carcinoma is similar to breast tissue and distinguishes it from hemangioma and angiosarcoma.
MRI features of tuberculosis or parasitic infections of the breast are not well documented in the literature. However, the smooth borders and relatively homogeneous signal characteristics of this lesion make infection less likely. Furthermore, numerous vascular channels seen on the gadolinium-enhanced high-resolution gradient-echo T1 images are not features of breast carcinoma or mastitis.
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At surgery, the left breast mass was shown to involve the tissue directly beneath the nipple, with a visually estimated diameter of approximately 7 cm; it was excised with grossly negative margins. While the fascia of the pectoralis muscle was being removed along with the lesion, three to four large vessels were encountered.
On gross pathologic examination, the specimen was 5.5 cm in greatest dimension (Fig. 4A). Sectioning revealed a heterogeneous surface consisting of gray to white indurated areas admixed with tan to yellow fibroadipose tissue. There were distinct areas of hemorrhage located throughout the specimen (Fig. 4B). Microscopically, the lesion showed irregular borders and consisted of lobulated groups of large dilated anastomosing vessels with thin to thick vessel walls and other groups of small-caliber vessels (Fig. 5A). The vessels contained RBCs, and hemosiderin deposition was seen in the surrounding fibrous tissue. Scattered small and large vessels within the lesion showed calcification (Fig. 5B). Scant lymphoid collections were present in the fibrous tissue. No breast epithelium was present.
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Histologically, a high-grade angiosarcoma shows a spindle cell proliferation with highly cellular areas, necrosis, and malignant nuclear features. A low- or intermediate-grade angiosarcoma, however, may be more difficult to distinguish from a hemangioma. Hemangiomas tend to have more lobulated borders rather than infiltrating, and at least grossly, hemangiomas appear better defined. Sarcomas invade and expand the preexisting normal structures, whereas hemangiomas surround or lie next to these structures. The pathologic findings in this case were most consistent with hemangioma.
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Hemangioma can be difficult to diagnose by mammography and sonography alone because its appearance is nonspecific and mimics many other entities. The presence of calcification on mammography helps limit the differential considerations. Sonography is useful for differentiating solid versus cystic lesions; it can also aid in lesion detection and identifying infiltration of adjacent tissues.
An interesting observation in this case is that the measurement on sonography was approximately half of the true size of the pathologic specimen. This may have been because the footprint of the transducer (3.8 cm) was smaller than the tumor (> 5 cm) and because the echotexture of the mass was similar to that of normal tissue. Our finding is similar to the experience of Glazebrook et al. [30], who also reported that hemangiomas were less conspicuous sonographically than they were clinically or mammographically. We find only two case reports in which the sonographic size measurement of breast hemangioma approximated the specimen size [9, 21].
MRI is the best technique in characterizing the vascular nature of the lesion and anatomically defining the extent of involvement. In our case, the knowledge of pectoralis muscle involvement was helpful at surgery. As such, MRI can be valuable for presurgical planning.
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This article has been cited by other articles:
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A. A. Shi, D. Georgian-Smith, and F. S. Chew Imaging of the Male Breast: Self-Assessment Module Am. J. Roentgenol., December 1, 2005; 185(6_Supplement): S211 - S213. [Abstract] [Full Text] [PDF] |
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