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_~ H
.......
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Adenoid Cystic Carcinoma of Breast With Metastasis: A Case Report and Review of the Literature Sook K. Lim, MD, Joseph Kovi, MD, and Oswald G. Warner, MD Washington, DC
Adenoid cystic carcinoma of the breast is a rare tumor having a favorable prognosis. At this writing, less than 100 cases have been reported. A review of the literature discloses only six welldocumented cases with associated distant metastases. The characteristic biological behavior of this tumor appears to be the development of distant metastasis without prior detectable invasion of axillary lymph nodes. Adenoid cystic carcinoma of the breast is a rare form of breast cancer and is considered to be of low-grade malignancy.1 To date, less than 100 documented cases have appeared in the literature. Of these, distant metastases were reported in only six cases, five in females and one in a male.2-6 The authors present a patient with adenoid cystic carcinoma of the breast which showed no regional node metastasis at the time of radical mastectomy nine years before.
Case Report A 58-year-old black woman was admitted to the hospital on May 24, Requests for reprints should be addressed to Dr. Sook K. Lim, Department of Pathology, Howard University Hospital, 2041 Georgia Avenue, NW, Washington, DC 20060.
1978, with a 12-month history of severe discomfort and pain in the left chest, dyspnea on exertion, and productive cough with occasional bouts of hemoptysis. She also complained of a weight loss of 30 lb during the past three months. Nine years earlier (December 1969), she had undergone a radical mastectomy for an adenoid cystic carcinoma of the left breast. None of the ten axillary lymph nodes examined showed evidence of metastatic disease. During the current admission, a chest x-ray revealed a round, solitary mass in the upper lobe of the left lung. On June 8, 1978, the patient had a thoracotomy and the left upper lobe was resected. On gross examination, the tumor was a firm, yellow-gray, ovoid, circumscribed mass measuring 2 cm in diameter. On microscopic examination, the tumor consisted of well-defined, rounded, or lobulated groups of small dark epithelial cells that enclosed acinar structures which were distended and filled with myxomatous material.
JOURNAL OF THE NATIONAL MEDICAL ASSOCIATION, VOL. 71, NO. 4, 1979
The nests of tumor cells were arranged in a trabecular pattern and were surrounded by fibrous connective tissue stroma. These histologic findings are characteristic of adenoid cystic carcinoma of the breast (Figure 1). Complete work-up of the patient, including scans of liver, spleen, and brain and a thoracospine series, revealed no additional sites of metastasis.
Discussion The biologic behavior of adenoid cystic carcinoma of the breast has not been totally elucidated because of a paucity of reported cases. Among the seven documented cases with distant metastasis (including the present one), only one showed axillary lymph node spread at surgery.2 The mean time span between the initial mastectomy and dis329
Figure 1. Adenoid cystic carcinoma infiltrating lung parenchyma (H & E, x 100).
Table 1. Review of Cases of Adenoid Cystic Carcinoma of the Breast with Distant Metastasis Authors
Verani et aI2
Age/Sex at Initial Diagnosis 63, F
Site of Neoplasm and Treatment Left, Radical
Detection of Distant Metastases Following Surgery
(years) 14
78, M
Nayer3
39, F
O'Kell4
73, F
Wilson, et a15
54, F
Elsner6
44, F
Lim, et al
48, F
Right, Modified Radical Mastectomy Left, Radical Mastectomy Left, Radical Mastectomy
Right, Simple Mastectomy Right, Radical Mastectomy Left, Modified Radical Mastectomy
Follow-up (years)
1
Lung, hilar lymph nodes, vertebra Lung, liver
11/**
8
Lung, pleura
4**
3
1**
7
Thoracic wall, lung, Inferior vena cava Ribs, axillary lymph nodes Lung
9
Lung
Mastectomy* "0
Sites of Metastases
6
3**
10 6 1
*Axillary node metastases noted at time of surgery. metastatic disease.
**Died from
tant metastasis was 6.8 years. A characteristic feature of adenoid cystic carcinoma of the breast seems to be the appearance of distant metastases in about six percent of cases, without microscopically detectable involvement of axillary lymph nodes at the time of radical mastectomy. 330
Literature Cited 1. Qizibash AH, Patterson MC, Oliveira KF: Adenoid cystic carcinoma of the breast: Light and electron microscopy and a brief review of the literature. Arch Pathol 101:302-306, 1977 2. Verani RR, Van der Bel-Kahn J: Mammary adenoid cystic carcinoma with unusual features. Am J Clin Pathol 59:653-658, 1973 3. Nayer HR: Cylindroma of the breast with
pulmonary metastases. Dis Chest 31:324-327, 1957 4. O'Kell RT: Adenoid cystic carcinoma of the breast. Mo Med 61:855-858, 1964 5. Wilson WB, Spell JP: Adenoid cystic carcinoma of breast. A case with recurrence and regional metastases. Ann Surg 166:861864, 1967 6. Eisner B: Adenoid cystic carcinoma of the breast. Review of the literature and clinicopathologic study of seven patients. Pathol Eur 5:357-364, 1970
JOURNAL OF THE NATIONAL MEDICAL ASSOCIATION, VOL. 71, NO. 4, 1979
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_~ H
.......
.- . .
Adenoid Cystic Carcinoma of Breast With Metastasis: A Case Report and Review of the Literature Sook K. Lim, MD, Joseph Kovi, MD, and Oswald G. Warner, MD Washington, DC
Adenoid cystic carcinoma of the breast is a rare tumor having a favorable prognosis. At this writing, less than 100 cases have been reported. A review of the literature discloses only six welldocumented cases with associated distant metastases. The characteristic biological behavior of this tumor appears to be the development of distant metastasis without prior detectable invasion of axillary lymph nodes. Adenoid cystic carcinoma of the breast is a rare form of breast cancer and is considered to be of low-grade malignancy.1 To date, less than 100 documented cases have appeared in the literature. Of these, distant metastases were reported in only six cases, five in females and one in a male.2-6 The authors present a patient with adenoid cystic carcinoma of the breast which showed no regional node metastasis at the time of radical mastectomy nine years before.
Case Report A 58-year-old black woman was admitted to the hospital on May 24, Requests for reprints should be addressed to Dr. Sook K. Lim, Department of Pathology, Howard University Hospital, 2041 Georgia Avenue, NW, Washington, DC 20060.
1978, with a 12-month history of severe discomfort and pain in the left chest, dyspnea on exertion, and productive cough with occasional bouts of hemoptysis. She also complained of a weight loss of 30 lb during the past three months. Nine years earlier (December 1969), she had undergone a radical mastectomy for an adenoid cystic carcinoma of the left breast. None of the ten axillary lymph nodes examined showed evidence of metastatic disease. During the current admission, a chest x-ray revealed a round, solitary mass in the upper lobe of the left lung. On June 8, 1978, the patient had a thoracotomy and the left upper lobe was resected. On gross examination, the tumor was a firm, yellow-gray, ovoid, circumscribed mass measuring 2 cm in diameter. On microscopic examination, the tumor consisted of well-defined, rounded, or lobulated groups of small dark epithelial cells that enclosed acinar structures which were distended and filled with myxomatous material.
JOURNAL OF THE NATIONAL MEDICAL ASSOCIATION, VOL. 71, NO. 4, 1979
The nests of tumor cells were arranged in a trabecular pattern and were surrounded by fibrous connective tissue stroma. These histologic findings are characteristic of adenoid cystic carcinoma of the breast (Figure 1). Complete work-up of the patient, including scans of liver, spleen, and brain and a thoracospine series, revealed no additional sites of metastasis.
Discussion The biologic behavior of adenoid cystic carcinoma of the breast has not been totally elucidated because of a paucity of reported cases. Among the seven documented cases with distant metastasis (including the present one), only one showed axillary lymph node spread at surgery.2 The mean time span between the initial mastectomy and dis329
Figure 1. Adenoid cystic carcinoma infiltrating lung parenchyma (H & E, x 100).
Table 1. Review of Cases of Adenoid Cystic Carcinoma of the Breast with Distant Metastasis Authors
Verani et aI2
Age/Sex at Initial Diagnosis 63, F
Site of Neoplasm and Treatment Left, Radical
Detection of Distant Metastases Following Surgery
(years) 14
78, M
Nayer3
39, F
O'Kell4
73, F
Wilson, et a15
54, F
Elsner6
44, F
Lim, et al
48, F
Right, Modified Radical Mastectomy Left, Radical Mastectomy Left, Radical Mastectomy
Right, Simple Mastectomy Right, Radical Mastectomy Left, Modified Radical Mastectomy
Follow-up (years)
1
Lung, hilar lymph nodes, vertebra Lung, liver
11/**
8
Lung, pleura
4**
3
1**
7
Thoracic wall, lung, Inferior vena cava Ribs, axillary lymph nodes Lung
9
Lung
Mastectomy* "0
Sites of Metastases
6
3**
10 6 1
*Axillary node metastases noted at time of surgery. metastatic disease.
**Died from
tant metastasis was 6.8 years. A characteristic feature of adenoid cystic carcinoma of the breast seems to be the appearance of distant metastases in about six percent of cases, without microscopically detectable involvement of axillary lymph nodes at the time of radical mastectomy. 330
Literature Cited 1. Qizibash AH, Patterson MC, Oliveira KF: Adenoid cystic carcinoma of the breast: Light and electron microscopy and a brief review of the literature. Arch Pathol 101:302-306, 1977 2. Verani RR, Van der Bel-Kahn J: Mammary adenoid cystic carcinoma with unusual features. Am J Clin Pathol 59:653-658, 1973 3. Nayer HR: Cylindroma of the breast with
pulmonary metastases. Dis Chest 31:324-327, 1957 4. O'Kell RT: Adenoid cystic carcinoma of the breast. Mo Med 61:855-858, 1964 5. Wilson WB, Spell JP: Adenoid cystic carcinoma of breast. A case with recurrence and regional metastases. Ann Surg 166:861864, 1967 6. Eisner B: Adenoid cystic carcinoma of the breast. Review of the literature and clinicopathologic study of seven patients. Pathol Eur 5:357-364, 1970
JOURNAL OF THE NATIONAL MEDICAL ASSOCIATION, VOL. 71, NO. 4, 1979
