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CORRESPONDENCE
Year : 2017  |  Volume : 13  |  Issue : 6  |  Page : 1059-1061

Triple malignancy involving breast, ovary, and uterine vault: A case report and literature review


1 Department of Radiotherapy, J. N. Medical College and Hospital, Aligarh Muslim University, Aligarh, Uttar Pradesh, India
2 Department of Obstetrics and Gynaecology, J. N. Medical College and Hospital, Aligarh Muslim University, Aligarh, Uttar Pradesh, India

Date of Web Publication13-Dec-2017

Correspondence Address:
Dr. Mohammad Shadab Alam
Department of Radiotherapy, J. N. Medical College and Hospital, Aligarh Muslim University, Aligarh - 202 002, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.220419

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 > Abstract 

The occurrence of two or more primary malignant neoplasms in the same person is rare. We report a case report of a 45-year-old woman with triple malignancy involving breast, ovary, and uterine vault managed at our center for 5 years. Our patient presented as a postoperated case of two primary malignant neoplasms of carcinoma, breast and ovary. For carcinoma ovary, she underwent adjuvant chemotherapy and interval cytoreductive surgery. For carcinoma breast, she received adjuvant locoregional radiotherapy and chemotherapy. After 42 months, the patient was diagnosed with squamous cell carcinoma vault, for which she received pelvic radiotherapy. She is on regular follow-up. Our patient had two synchronous and one metachronous malignancy. She was diagnosed with carcinoma uterine vault when she was in regular follow-up, and the two previous primaries were controlled. This emphasizes the importance of a regular follow-up and the need of a meticulous workup for early diagnosis and prompt management of any metachronous malignancy.

Keywords: Infiltrating ductal carcinoma of breast, multiple primary malignancies, papillary serous cystadenocarcinoma of ovary, squamous cell carcinoma cervix


How to cite this article:
Alam MS, Perween R, Siddiqui SA. Triple malignancy involving breast, ovary, and uterine vault: A case report and literature review. J Can Res Ther 2017;13:1059-61

How to cite this URL:
Alam MS, Perween R, Siddiqui SA. Triple malignancy involving breast, ovary, and uterine vault: A case report and literature review. J Can Res Ther [serial online] 2017 [cited 2019 Dec 14];13:1059-61. Available from: http://www.cancerjournal.net/text.asp?2017/13/6/1059/220419


 > Introduction Top


Warren and Gates first described multiple primary malignancies (MPMs) in the same individual. It is defined as the occurrence of two or more malignancies in the same individual without any relationship between the tumors.[1] It is of two types: (a) synchronous in which all the malignancies occur at the same time or within 6 months of first malignancy, (b) metachronous in which the second or high order malignancies occur at least 6 months after the previous one.[2]

Multiple primary tumors mainly involve respiratory, gastrointestinal, and genitourinary tract.[3] According to various studies, their prevalence is in the range of 3%–5% among which triple tumors occur in only 0.5% of cases.[4] There is an increase in the incidence of MPM, and one of the most important causes of this increasing trend is the gain in survival of cancer patients nowadays due to advances in diagnostic modalities, treatment, and supportive care.[5] According to the National Cancer Institute's Surveillance, Epidemiology, and End Results Program report, about one in six cancer patients develops the second malignant neoplasm in their further lifetime.[6] After extensive search in literature, we have found that this is the first case report of three primary malignancies in a postmenopausal woman from India, in which two malignancies involving breast and ovary are synchronous type and one involving uterine vault is metachronous type.


 > Case Report Top


It is a case of a previously undescribed combination of coexistent triple primary malignant neoplasms. A 45-year-old Indian woman with triple primary malignancy of carcinoma of breast and ovary followed by carcinoma of uterine vault over the last 5 years is managed successfully at our center. In her medical history, she had no personal or family history of cancer or management related to it. She is an unmarried woman with age of menarche at 15 years and surgical menopause at 40 years. Our patient never received hormone replacement therapy and oral contraceptive pills in her lifetime. She is a known hypertensive since 10 years and is on antihypertensive drugs. In January 2010, the patient presented to our outpatient department as postoperated case of carcinoma breast and ovary for adjuvant treatment. She had a history of breast lump and ovarian cyst, for which she got operated outside in a private clinic. She had undergone modified radical mastectomy for breast lump and bilateral ovarian cystectomy for ovarian cyst. Histopathology of mastectomy specimen revealed infiltrating ductal carcinoma breast (5 cm × 4 cm × 4 cm) with margin positive and all five resected axillary nodes positive for malignancy [Figure 1]. Ovarian histopathology was papillary serous cystadenocarcinoma of the bilateral ovaries with involvement of external surfaces [Figure 2]. Both the hormone receptors (estrogen and progesterone receptors) and human epidermal growth factor receptor 2/neu were negative. She was planned for adjuvant chemotherapy, followed by adjuvant radiation to chest wall and regional lymph nodes. She received three cycles of paclitaxel (260 mg) and carboplatin (450 mg) every 21 days. Following this, her serum CA 125 level was within normal limit (15 IU/ml), but her contrast-enhanced computed tomography of the abdomen showed well-defined hypodense pelvic lesion invading adjacent bowel loops and rectum with metastatic deposits over bowel loops and uterine fibroid. In May 2010, she underwent interval debulking surgery. Histopathology revealed metastatic deposits on omentum, chronic cervicitis with all the resected nodes showing reactive changes. After this, she received three more cycles of same chemotherapy. Then, she was treated with irradiation to chest wall and regional lymph nodes. Her treatment was completed in September 2010.
Figure 1: Histopathological features of infiltrated duct carcinoma of the breast

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Figure 2: Histopathological features of serous papillary adenocarcinoma of the ovary

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She was on regular follow-up. During her follow-up period, she underwent thorough physical examination with regular monitoring of serum CA 125 level and radiological investigations as per requirement. In April 2012, she presented with bleeding per vaginum and ultrasonography (USG) of the abdomen revealed a 65 mm × 63 mm mass in the uterine fossa. All other organs were normal with no evidence of metastasis, but her CA 125 level was raised to 1276 U/ml. She got symptomatic improvement, following conservative treatment. She was again planned for chemotherapy and received six cycles of cyclophosphamide (750 mg D1), adriamycin (70 mg D1), and cisplatin (100 mg D1) repeated every 21 days. After the completion of chemotherapy, radiologically, the pelvic mass completely resolved and serum CA 125 level was within normal limit. In June 2013, she again presented with complaints of bleeding per vaginum. Clinical examination revealed an ulceroproliferative growth involving uterine vault with induration of right-sided parametrium short of the pelvic wall. Vault biopsy was suggestive of squamous cell carcinoma [Figure 3] (no immunohistochemistry marker was not done for vault growth as it was clearly evident as squamous cell carcinoma from histopathology examination only and there was no confusion in the diagnosis). She received pelvic external beam radiotherapy dose of 50 Gray in 25 fractions along with concurrent weekly cisplatin-based chemotherapy. It was followed by intravaginal brachytherapy dose of three fractions of 6.5 Gray each prescribed at vaginal mucosa. After radiotherapy, there was complete response of vault growth. The patient was kept on regular follow-up. In November 2014, she had recurrence of vault growth reaching up to introitus. Biopsy confirmed it as moderately differentiated squamous cell carcinoma. USG of the abdomen was suggestive of irregular mass in the pelvis posterior to the base of urinary bladder [Figure 4], and serum CA 125 level was 226.13 U/ml. She received six cycles of three weekly gemcitabine (1.4 gm) and carboplatin (450 mg) based chemotherapy. The pelvic disease responded to chemotherapy and the patient is kept on regular follow-up. Our patient tolerated all her treatment well with minimal side effects and without complications. In the last follow, all the three primary malignancies are well controlled with evidence of treatment-related late toxicities. At present, the patient is alive and disease-free with no evidence of any recurrent or residual disease.
Figure 3: Histopathological features of squamous cell carcinoma of the uterine vault

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Figure 4: Ultrasonography of the abdomen showing irregular mass in the pelvis posterior to the base of urinary bladder

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 > Discussion Top


MPM are rare, but now with increasing survival of cancer patients, the incidence of MPM is increasing. These newly developed primary cancers can be due to previous therapy, some syndrome-related or some common etiologic factors.[7]

Multiple metachronous malignancies are often seen with hematological, lung, thyroid, breast, skin, and genitourinary malignancies.[3] Familial carcinoma syndrome is a combination of carcinoma of breast and ovary. In the present case, there were two primary synchronous malignancies – carcinoma breast and ovary. However, our patient does not have family history of any type of cancer and also no known major risk factor for these malignancies. Hence, the present case is a sporadic occurrence which might be related to polygenic model and environmental modifications.[8] Noh et al. reported a case of quadruple malignancy involving ovary and endometrium, but after breast and rectal cancer.[9]

The choice of treatment and their sequencing was difficult task. There are no standard guidelines for management of MPM.

Among the cases of MPM, there are only about 10% cases of true triple malignancies, with reporting of approximately 135 cases only. With this background, it is felt that this case warrants recording in the literature.


 > Conclusion Top


Awareness, suspicion of MPM, and aggressive diagnostic workup play crucial role in the detection of MPMs at early stage for better outcome. In addition, choice of appropriate treatment and their sequencing remains the cornerstone.

Acknowledgment

We are thankful and would like to acknowledge the contributions of the doctors and staffs of Departments of Radiotherapy and Obstetrics and Gynaecology of J. N. Medical College and Hospital, Aligarh, Uttar Pradesh, India.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
 > References Top

1.
Warren S, Gates O. Multiple primary malignant tumors: A survey of the literature and a statistical study. Am J Cancer 1932;16:1358-414.  Back to cited text no. 1
    
2.
Moertel CG. Multiple primary malignant neoplasms: Historical perspectives. Cancer 1977;40 4 Suppl: 1786-92.  Back to cited text no. 2
    
3.
Jayaraman S, Balakrishnan S, Rao D. Multiple metachronous malignancies, one patient with three primary malignancies. Indian J Surg 2011;73:377-9.  Back to cited text no. 3
[PUBMED]    
4.
Hu NC, Hsieh SC, Chen TJ, Chang JY. Multiple primary malignancies including colon, stomach, lung, breast, and liver cancer: A case report and literature review. Chin Med J (Engl) 2009;122:3091-3.  Back to cited text no. 4
[PUBMED]    
5.
Cercato MC, Colella E, Ferraresi V, Diodoro MG, Tonachella R. Report of two cases of quintuple primary malignancies and review of the literature. Anticancer Res 2008;28:2953-8.  Back to cited text no. 5
[PUBMED]    
6.
Wood ME, Vogel V, Ng A, Foxhall L, Goodwin P, Travis LB. Second malignant neoplasms: Assessment and strategies for risk reduction. J Clin Oncol 2012;30:3734-45.  Back to cited text no. 6
    
7.
Travis LB, Hill D, Dores GM, Gospodarowicz M, van Leeuwen FE, Holowaty E, et al. Cumulative absolute breast cancer risk for young women treated for Hodgkin lymphoma. J Natl Cancer Inst 2005;97:1428-37.  Back to cited text no. 7
    
8.
Hemminki K, Aaltonen L, Li X. Subsequent primary malignancies after endometrial carcinoma and ovarian carcinoma. Cancer 2003;97:2432-9.  Back to cited text no. 8
    
9.
Noh SK, Yoon JY, Ryoo UN, Choi CH, Sung CO, Kim TJ, et al. A case report of quadruple cancer in a single patient including the breast, rectum, ovary, and endometrium. J Gynecol Oncol 2008;19:265-9.  Back to cited text no. 9
    


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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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