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Choriocarcinoma presenting as a breast lump
European Journal of Suryical Oncology 1995; 2 1 : 5 7 6 - 5 7 8
CASE
REPORTS
Choriocarcinoma presenting as a breast lump
C. A. Fowler, S. Nicholson, M. Lott* and V. Barleyt
Departments of Surgery and *Histopathology, Weston General Hospital, Grange Road, Weston-Super-Mare and ~Department qf Oncology, Bristol Royal InfirmatT, Bristol, UK A case of ehoriocarcinoma in a previously well 32-year-old woman presenting as a tender breast lump whilst she is breastfeeding.
Key words:choriocarcinoma; breast.
Introduction
Discussion
Choriocarcinoma is an uncommon malignancy o f which 70% are preceded by benign trophoblastic disease. It occurs in I in 50,000 viable pregnancies--approximately 10% of the total cases of choriocarcinoma. Usually it presents with persistent uterine bleeding or with symptoms from pulmonary or cerebral secondary deposits postpartum. We report on an unusual case presenting as a breast lump.
The clinically observed incidence of extramammary metastatic tumours to the breast is 0.5-1.3% ~'" which is outweighed by primary tumours in the breast by I : 220. Most reports of metastatic disease in the breast are in patients who have had prior therapy for a known primary and present on average 1.9 years after the primary has been diagnosed? The primary sites are varied (Table I) with malignant melanoma and bronchial carcinoma the most reported? -9 Metastatic choriocarcinoma in the breast has been recorded only a few times in the literature (Table 2). All of the cases reviewed were young women and three were pregnant at the time of presentation.
Case report A 32-year-old woman presented to outpatients in December 1992 with a painful right breast lump, 6 weeks postpartum. Past obstetric history--menarche age 1 I, para 2 + 0 , first pregnancy age 26. She had first noticed a small lump in the third trimester of the pregnancy and she was now breast-feeding. On examination there was a red, hot, tender mass in the lower outer quadrant of her right breast. • The lump was aspirated and 30 ml of pus was sent for culture, she was already on antibiotics and the culture was negative. A week later another 35 ml of bloodstained serous fluid was aspirated, but two weeks later it had not settled and she was admitted for incision and drainage via the infra-mammary approach. Initially she improved but soon became pyrexial with a swinging fever which was unresponsive to antibiotics; cultures front daily aspirations were negative. She underwent a further examination under anaesthetic and biopsy of a large well circumscribed mass with a necrotic centre. Histology showed choriocarcinoma and a chest X-ray revealed multiple opacities in both lung fields. Her serum beta-HCG at this time was 9920 IU/1, with normal alpha-fetoprotein and carcinoembryonic antigen. She was referred to Charing Cross Hospital where she commenced 10 courses of etoposide, methotrexate, Adriamycin, vincristine and cyclophosphamide. She showed an excellent response with a dramatic decrease in the serum beta-HCG level (Fig. I ) and a > 50% reduction ofall measurable lesions on the chest X-ray with a complete response in the breast. Six weeks after finishing the chemotherapy she was admitted with right sided focal seizures and a CT scan revealed a left parietal metastasis, treated with dexamethasone, phenytoin and radiotherapy. In the months following this her urinary beta-HCG levels slowly rose again and in September 1993 she had further courses of chemotherapy and again had a reasonable response. Despite this the disease progressed rapidly and she died in December 1993, a year after her initial presentation.
50000
50000 Urinary 40000 6-HCG 30000
IU/L 20000
10000
~
0 ....... Jan
I I : ', , June
I I I ......... Sept
Dec Months
A -- Chemotherapy begun B -- Chemotherapy ended Correspondence to: Dr C. A. Fowler, Weston General Hospital, Grange Road, Weston-Super-Mare'BS23 4TQ, U K . 0748-7983/95/050576 + 03 $12.00/0
Fig. I. B e t a - - h u m a n chorionic gonadotrophin levels in response to chemotherapy © 1995 W.B. Saunders Company Limited
577
Case reports Table I. Origin of malignant tumours metastatic to the breast
Turnout
Total
No. presenting with breast lump
56 43 40 36 18 15 13 9 8 7 6 6
3 20 I 8 I 3 2 0 2 I 0 0
Malignant mehmoma Lung Prostate Lymphoma Sarcoma Ovary Stomach Cervix Carcinoid of gut Kidney Colon Thyroid
Table 2. Review of all cases of metastatic choriocarcinoma in the breast
Age
Presentation
Previous pregnancy
Beta-HCG at diagnosis
Resnick ( 1945)"
27
Breast lump in pregnancy
Spontaneous abortion
?
H reshchyshyn et al. (1961) II
25
Haematuria in pregnancy
T
Tsukamoto et al. (1980) I:
29
Breast lump in pregnancy
2 spontaneous abortions--one a proven hydatidiform mole Hydatidiform mole
Alvarez et al. (1991} t3
29
Breast lump
Kumar el a]. (1991) 14
22
Present case
32
Breast lump - 1.5 years after known choriocarcinoma Breast lump postpartum noticed in pregnancy
Other metastases at presentation
Treatment
Survival
Pulmonary, liver, spleen, kidney, adrenals Breast, pulmonary + widespread
Supportive
2 weeks
Amethopterin
2 weeks
T
Pulmonary, cerebral later
I month
Elective abortion
"[
Pulmonary, cerebral later
Term pregnancy with proven choriocarcinoma Term pregnancy
1"
Pulmonary, subcutaneous
None as beta-HCG levels fell on delivery of child Combination chemotherapy with breast and pulmonary surgery Unknown
T
Pulmonary, cerebral later
In all five cases the antecedent pregnancy had been a hydatidiform mole, a termination of pregnancy or choriocarcinoma following a normal pregnancy. Four of them presented with a breast lump and on further examination the woman with haematuria also had breast lumps. In all cases metastases to other organs were noted at presentation or soon afterwards, most commonly pulmonary and cerebral. The diagnosis can be delayed in these cases because of its rarity, particular if there is no past history of choriocarcinoma. Histology usually confirms the diagnosis either from breast biopsy or at post mortem. The present case showed the typical histological features of clusters of cytotrophoblast cells separated by syncytiotrophoblast with positive immunohistochemistry for betaHCG. The diagnosis was substantiated in all cases (except Resnick, 1945) I° by a high serum beta-HCG level. These findings are very similar to our case as her breast lump first appeared in pregnancy and at presentation she had markedly elevated beta-Ht~G levels and pulmonary metastases. In all cases presenting with breast metastases the prognosis is poor with a maximum survival of two years but most succumbing quickly. The longest survivor had been given alternating chemotherapy with etoposide, methotrexate and actinomycin D, then 5-fiuorouracil and platinol? 3 Our patient twice showed a good response to chemotherapy but as with previous cases
Combination chemotherapy
2 years
Unknown 1 year
eventually died of disseminated disease. It is important in these cases to distinguish a metastasis from a primary breast carcinoma in order to avoid unnecessary surgery and instigate appropriate treatment? .3.6.9-
References
1. Bohman LG, Bassett LW, Gold RH, Voet R. Breast metastases from extramammary malignancies. Diagn Rad 1982; 144: 30911. 2. Hajdu SI, Urban JA. Cancers metastatic to the breast, Cancer 1972; 29: 1691-6. 3. McCrea ES, Johnston C, Haney PJ. Metastases to the breast. Am J Radiol 1983; 141: 685-90. 4. Sandison AR. Metastatic tumours in the breast. B r J S u r g 1959; 47: 54-8. 5~ Deeley TJ. Secondary deposits in the breast. Br J Cancer 1965; 19: 738-43. 6. Mclntosh IH, Hooper AA, Millis RR, Greening WP. Metastatic carcinoma within the breast. Clin Oncol 1976; 2: 393-401.
578
Case reports
7. Kelly C, Henderson D, Corris P. Breast lumps: rare presentation of oat cell carcinoma of lung. J Clin Pathol 1988; 41:17 I-2. 8. Nielson M, Anderson JA, Henrikson PW, et al. Metastases to the breast from extramammary carcinomas. Acta Pathol Microbiol Scand (Section A) 1981; 89:25 I-6. 9. Paulus DD, Libshitz HI. Metastasis to the breast. Radiol Clin North Am 1982; 20: 561-7. 10. Resnick L. Extragenital chorionepithelioma, report of a ease with chorionepithelioma of the breast occurring during the course of pregnancy. J Obstet Gynaecol Br Emp 1945; 52: 1808. I1. Hreshchyshyn MM, Graham JB, Holland JF. Treatment of malignant trophoblastic growth in women, with special ref-
erence to amethopterin. Am J Obstet Gynaecol 1961; 81: 688705. 12. Tsukamoto N, Kashimura Y, Masatoshi S, et al. Choriocarcinoma occurring within the normal placenta with breast metastasis. Gynaecol Oncol 1981; 11: 348-63. 13, Alvarez RD, Gleason BP, Gore H, Partridge EE. Coexisting intraductal breast carcinoma and metastatic choriocarcinoma presenting as a breast mass. Gynaecol Oncol 1991; 43: 295-9. 14. Kumar PV, Esfahani FN, Salimi A. Choriocarcinoma metastatic to the breast diagnosed by fine needle aspiration. Acta C)'tol 1991; 35: 239--41. Accepted.for publication 22 September 1995
CASE
REPORTS
Choriocarcinoma presenting as a breast lump
C. A. Fowler, S. Nicholson, M. Lott* and V. Barleyt
Departments of Surgery and *Histopathology, Weston General Hospital, Grange Road, Weston-Super-Mare and ~Department qf Oncology, Bristol Royal InfirmatT, Bristol, UK A case of ehoriocarcinoma in a previously well 32-year-old woman presenting as a tender breast lump whilst she is breastfeeding.
Key words:choriocarcinoma; breast.
Introduction
Discussion
Choriocarcinoma is an uncommon malignancy o f which 70% are preceded by benign trophoblastic disease. It occurs in I in 50,000 viable pregnancies--approximately 10% of the total cases of choriocarcinoma. Usually it presents with persistent uterine bleeding or with symptoms from pulmonary or cerebral secondary deposits postpartum. We report on an unusual case presenting as a breast lump.
The clinically observed incidence of extramammary metastatic tumours to the breast is 0.5-1.3% ~'" which is outweighed by primary tumours in the breast by I : 220. Most reports of metastatic disease in the breast are in patients who have had prior therapy for a known primary and present on average 1.9 years after the primary has been diagnosed? The primary sites are varied (Table I) with malignant melanoma and bronchial carcinoma the most reported? -9 Metastatic choriocarcinoma in the breast has been recorded only a few times in the literature (Table 2). All of the cases reviewed were young women and three were pregnant at the time of presentation.
Case report A 32-year-old woman presented to outpatients in December 1992 with a painful right breast lump, 6 weeks postpartum. Past obstetric history--menarche age 1 I, para 2 + 0 , first pregnancy age 26. She had first noticed a small lump in the third trimester of the pregnancy and she was now breast-feeding. On examination there was a red, hot, tender mass in the lower outer quadrant of her right breast. • The lump was aspirated and 30 ml of pus was sent for culture, she was already on antibiotics and the culture was negative. A week later another 35 ml of bloodstained serous fluid was aspirated, but two weeks later it had not settled and she was admitted for incision and drainage via the infra-mammary approach. Initially she improved but soon became pyrexial with a swinging fever which was unresponsive to antibiotics; cultures front daily aspirations were negative. She underwent a further examination under anaesthetic and biopsy of a large well circumscribed mass with a necrotic centre. Histology showed choriocarcinoma and a chest X-ray revealed multiple opacities in both lung fields. Her serum beta-HCG at this time was 9920 IU/1, with normal alpha-fetoprotein and carcinoembryonic antigen. She was referred to Charing Cross Hospital where she commenced 10 courses of etoposide, methotrexate, Adriamycin, vincristine and cyclophosphamide. She showed an excellent response with a dramatic decrease in the serum beta-HCG level (Fig. I ) and a > 50% reduction ofall measurable lesions on the chest X-ray with a complete response in the breast. Six weeks after finishing the chemotherapy she was admitted with right sided focal seizures and a CT scan revealed a left parietal metastasis, treated with dexamethasone, phenytoin and radiotherapy. In the months following this her urinary beta-HCG levels slowly rose again and in September 1993 she had further courses of chemotherapy and again had a reasonable response. Despite this the disease progressed rapidly and she died in December 1993, a year after her initial presentation.
50000
50000 Urinary 40000 6-HCG 30000
IU/L 20000
10000
~
0 ....... Jan
I I : ', , June
I I I ......... Sept
Dec Months
A -- Chemotherapy begun B -- Chemotherapy ended Correspondence to: Dr C. A. Fowler, Weston General Hospital, Grange Road, Weston-Super-Mare'BS23 4TQ, U K . 0748-7983/95/050576 + 03 $12.00/0
Fig. I. B e t a - - h u m a n chorionic gonadotrophin levels in response to chemotherapy © 1995 W.B. Saunders Company Limited
577
Case reports Table I. Origin of malignant tumours metastatic to the breast
Turnout
Total
No. presenting with breast lump
56 43 40 36 18 15 13 9 8 7 6 6
3 20 I 8 I 3 2 0 2 I 0 0
Malignant mehmoma Lung Prostate Lymphoma Sarcoma Ovary Stomach Cervix Carcinoid of gut Kidney Colon Thyroid
Table 2. Review of all cases of metastatic choriocarcinoma in the breast
Age
Presentation
Previous pregnancy
Beta-HCG at diagnosis
Resnick ( 1945)"
27
Breast lump in pregnancy
Spontaneous abortion
?
H reshchyshyn et al. (1961) II
25
Haematuria in pregnancy
T
Tsukamoto et al. (1980) I:
29
Breast lump in pregnancy
2 spontaneous abortions--one a proven hydatidiform mole Hydatidiform mole
Alvarez et al. (1991} t3
29
Breast lump
Kumar el a]. (1991) 14
22
Present case
32
Breast lump - 1.5 years after known choriocarcinoma Breast lump postpartum noticed in pregnancy
Other metastases at presentation
Treatment
Survival
Pulmonary, liver, spleen, kidney, adrenals Breast, pulmonary + widespread
Supportive
2 weeks
Amethopterin
2 weeks
T
Pulmonary, cerebral later
I month
Elective abortion
"[
Pulmonary, cerebral later
Term pregnancy with proven choriocarcinoma Term pregnancy
1"
Pulmonary, subcutaneous
None as beta-HCG levels fell on delivery of child Combination chemotherapy with breast and pulmonary surgery Unknown
T
Pulmonary, cerebral later
In all five cases the antecedent pregnancy had been a hydatidiform mole, a termination of pregnancy or choriocarcinoma following a normal pregnancy. Four of them presented with a breast lump and on further examination the woman with haematuria also had breast lumps. In all cases metastases to other organs were noted at presentation or soon afterwards, most commonly pulmonary and cerebral. The diagnosis can be delayed in these cases because of its rarity, particular if there is no past history of choriocarcinoma. Histology usually confirms the diagnosis either from breast biopsy or at post mortem. The present case showed the typical histological features of clusters of cytotrophoblast cells separated by syncytiotrophoblast with positive immunohistochemistry for betaHCG. The diagnosis was substantiated in all cases (except Resnick, 1945) I° by a high serum beta-HCG level. These findings are very similar to our case as her breast lump first appeared in pregnancy and at presentation she had markedly elevated beta-Ht~G levels and pulmonary metastases. In all cases presenting with breast metastases the prognosis is poor with a maximum survival of two years but most succumbing quickly. The longest survivor had been given alternating chemotherapy with etoposide, methotrexate and actinomycin D, then 5-fiuorouracil and platinol? 3 Our patient twice showed a good response to chemotherapy but as with previous cases
Combination chemotherapy
2 years
Unknown 1 year
eventually died of disseminated disease. It is important in these cases to distinguish a metastasis from a primary breast carcinoma in order to avoid unnecessary surgery and instigate appropriate treatment? .3.6.9-
References
1. Bohman LG, Bassett LW, Gold RH, Voet R. Breast metastases from extramammary malignancies. Diagn Rad 1982; 144: 30911. 2. Hajdu SI, Urban JA. Cancers metastatic to the breast, Cancer 1972; 29: 1691-6. 3. McCrea ES, Johnston C, Haney PJ. Metastases to the breast. Am J Radiol 1983; 141: 685-90. 4. Sandison AR. Metastatic tumours in the breast. B r J S u r g 1959; 47: 54-8. 5~ Deeley TJ. Secondary deposits in the breast. Br J Cancer 1965; 19: 738-43. 6. Mclntosh IH, Hooper AA, Millis RR, Greening WP. Metastatic carcinoma within the breast. Clin Oncol 1976; 2: 393-401.
578
Case reports
7. Kelly C, Henderson D, Corris P. Breast lumps: rare presentation of oat cell carcinoma of lung. J Clin Pathol 1988; 41:17 I-2. 8. Nielson M, Anderson JA, Henrikson PW, et al. Metastases to the breast from extramammary carcinomas. Acta Pathol Microbiol Scand (Section A) 1981; 89:25 I-6. 9. Paulus DD, Libshitz HI. Metastasis to the breast. Radiol Clin North Am 1982; 20: 561-7. 10. Resnick L. Extragenital chorionepithelioma, report of a ease with chorionepithelioma of the breast occurring during the course of pregnancy. J Obstet Gynaecol Br Emp 1945; 52: 1808. I1. Hreshchyshyn MM, Graham JB, Holland JF. Treatment of malignant trophoblastic growth in women, with special ref-
erence to amethopterin. Am J Obstet Gynaecol 1961; 81: 688705. 12. Tsukamoto N, Kashimura Y, Masatoshi S, et al. Choriocarcinoma occurring within the normal placenta with breast metastasis. Gynaecol Oncol 1981; 11: 348-63. 13, Alvarez RD, Gleason BP, Gore H, Partridge EE. Coexisting intraductal breast carcinoma and metastatic choriocarcinoma presenting as a breast mass. Gynaecol Oncol 1991; 43: 295-9. 14. Kumar PV, Esfahani FN, Salimi A. Choriocarcinoma metastatic to the breast diagnosed by fine needle aspiration. Acta C)'tol 1991; 35: 239--41. Accepted.for publication 22 September 1995