|Year : 2014 | Volume
| Issue : 4 | Page : 1109-1111
Triple primary malignant neoplasms including breast, esophagus and base tongue in an elderly male: A case report
Madhup Rastogi1, Sharad Singh2, Sudhir Singh3, Seema Gupta3, Raghav C Dwivedi4
1 Department of Radiation Oncology, Dr. Ram Manohar Lohia Institute of Medical Sciences, Lucknow, India
2 Department of Radiotherapy, J. K. Cancer Institute, Kanpur, India
3 Department of Radiotherapy, King George's Medical University, Lucknow, Uttar Pradesh, India
4 Head-Neck Unit, Royal Marsden Hospital, Fulham Road, London, United Kingdom
|Date of Web Publication||9-Jan-2015|
Associate Professor, Department of Radiation Oncology, Dr. Ram Manohar Lohia Institute of Medical Sciences, Vibhuti Khand, Gomti Nagar, Lucknow - 226 010, Uttar Pradesh
Source of Support: None, Conflict of Interest: None
Cases involving more than two primary malignant neoplasms are very rare. The present article reports a case of multiple primary malignant neoplasms including esophagus initially followed by right breast and later base of tongue in an elderly male patient, which is extremely a rare combination.
Keywords: Breast carcinoma, carcinoma base tongue, carcinoma esophagus, triple primary malignant tumor
|How to cite this article:|
Rastogi M, Singh S, Singh S, Gupta S, Dwivedi RC. Triple primary malignant neoplasms including breast, esophagus and base tongue in an elderly male: A case report. J Can Res Ther 2014;10:1109-11
|How to cite this URL:|
Rastogi M, Singh S, Singh S, Gupta S, Dwivedi RC. Triple primary malignant neoplasms including breast, esophagus and base tongue in an elderly male: A case report. J Can Res Ther [serial online] 2014 [cited 2020 Jan 28];10:1109-11. Available from: http://www.cancerjournal.net/text.asp?2014/10/4/1109/144639
| > Introduction|| |
Multiple primary malignant neoplasm (PMN) has been identified in the cases harboring the multiple tumors that meet the criteria by Warren and Gates  as follows; [A] Each neoplasm must be malignant on histological evaluation. [B] Each neoplasm must be geographically separate and distinct. If the intervening mucosa demonstrates dysplasia, it must be considered as a multicentric primary lesion and not as two separate neoplasms. [C] The possibility that the second neoplasm represents a metastasis should be excluded. Documentation that invasive carcinoma is arising from overlying epithelium with transition from carcinoma in situ at the border is helpful. The case reported here met these criteria, and presented each tumor with interval over 1 year, indicating that they were metachronous triple PMNs. Cases involving more than two primary malignant neoplasms are very rare. The present article reports a case of multiple primary malignant neoplasms including esophagus initially followed by right breast and later base of tongue in an elderly male patient, which is an extremely rare combination.
| > Case report|| |
A 70-year-old male was referred to our head and neck cancer clinic in June 2007 with chief complaints of gradually increasing difficulty in swallowing with pain in the neck for the past 2 months. His past history and examination revealed some interesting facts. In 1997, he had dysphagia for solids. Barium Swallow examination revealed irregularity of mucosa in upper esophagus [Figure 1]. An Upper gastrointestinal (GI Endoscopy examination showed growth of 21 cm extending up to 25 cm [Figure 2] suggestive of carcinoma esophagus of upper one third. Endoscopic biopsy confirmed the diagnosis of squamous cell carcinoma. Patient was subjected to radiation therapy after metastatic work up. He was given 60 Gy in 30 fractions using 2 Gy fractions, 5 days a week. The radiotherapy was well tolerated and patient became apparently asymptomatic following radiotherapy. In January 2000, he developed a small lump in the right breast of approximately 3 × 3 cm in size, with no fixity to underlying structures. The fine needle aspiration cytology revealed few duct carcinoma cells in it. Thereafter, the patient underwent simple mastectomy with axillary clearance. Histopathological examination of postoperative specimen revealed infiltrating ductal carcinoma of right breast without lymphatic metastases [Figure 3]. The receptor study was also performed, which showed 32% estrogen receptor (ER) and 36% progesterone receptor (PR) nuclear positivity. Postoperative phase was uneventful and scar healed properly [Figure 4]. He was given Tab. tamoxifen 20 mg daily for 3 years, and remained under regular follow-up.
|Figure 1: Barium Swallow AP view showing narrowing and irregular mucosa of upper esophagus|
Click here to view
|Figure 2: Upper G.I. Endoscopy film showing irregular growth in the upper esophagus suggestive of Carcinoma esophagus|
Click here to view
|Figure 3: Photomicrograph (H and E×1000) showing round and cuboidal cells disposed in groups and in thin strands. The cells present anisnucleosis and anisocytosis|
Click here to view
|Figure 4: Healed scar of the right breast with the radiation changes of the skin in the neck due to previous radiotherapy for carcinoma esophagus|
Click here to view
In June 2007, the patient presented again with complaints of dysphagia. Recurrence of esophageal carcinoma was thought but barium swallow examination did not show any irregularity or obstruction. Indirect laryngoscopy examination revealed a proliferative growth from posterior third of the tongue [Figure 5]. Histopathological examination turned out to be squamous cell carcinoma of base tongue. His family history was non-contributory. Physical examination revealed cachexia and icterus. Routine examinations like hemogram and renal function test were well within normal limits, but liver function test was deranged. Sonological study of abdomen revealed multiple calculi in the gall bladder causing obstructive jaundice. Search for metastases by chest skiagram and bone scan did not show any positive results. Patient did not receive any treatment further because of high levels of bilirubin and finally succumbed to the disease.
|Figure 5: Vide Endoscopy film showing proliferative growth in the posterior third of tongue|
Click here to view
| > Discussion|| |
Multiple primary cancers are defined by the International Association of Cancer Registries as the occurrence of two or more primary cancers, where each cancer originates in a separate primary site and is either an extension, recurrence or metastases.  Recent advances in the treatment and the diagnostic modality of malignant neoplasm have led to increased survival rate and life expectancy. As a result, many of the patients, who survived the first cancer live long enough to develop additional primary cancers. Multiple primary squamous cell carcinomas of head and neck are increasing in frequency, especially in cases of hypopharynx and esophagus, because the capability to control squamous cell carcinomas of the head and neck has improved recently. ,, The concept of "field cancerization" has been widely accepted for such multiple cancers in the upper aero digestive tract region.  Up to 10% of the cancer patients have been reported to acquire multiple primary cancers of separate organ sites in the ten year following the diagnosis of their first cancer.  Sometimes, host susceptibility is sited as the cause of these multiple primaries, with BRAC1 and BRAC2 genetic mutation related to breast and ovarian cancers, as being one example.  Infection and immunodeficiency also have been implicated, as in the association between Kaposi sarcoma and non-Hodgkin's lymphoma.  Environmental or lifestyle factors can also be instrumental for PMN. Smoking and drinking behaviors, especially, in elderly males compared to females of same age are at more risk of developing PMN in sites like esophagus, stomach, colon, rectum and lung.  It is well known that cancer treatment can cause second primary cancers. Example includes acute leukemias that arise due to chemotherapy and these along with other cancers that arise from radiotherapy.  There is also the potential for an increased detection of second primary cancers from medical investigations that follow an initial cancer diagnosis.  This would contribute, for example, to the common occurrence of multiple cancers of the bladder and prostate. There are number of instances where identifying cancers that occur as multiple primaries can be useful. For example, females with history of uterine or ovarian cancer have been found to be at increased risk of breast cancer, which can lead to genetic testing or routine screening of the person at risk.  Also, by comparing the treatment profiles based on how second primaries were experienced, carcinogenic effects of treatment can be identified and safer treatment options can be adopted. 
Only few articles pertaining to the multiple neoplasms are present in the literature dealing with mostly second primary of upper aero digestive tract. , However, occurrence of such a combination in male patient has not been reported so far.
| > Conclusion|| |
Nevertheless, the occurrence of second and third malignant tumors being very rare, this study needs to be considered. The differential diagnosis with cancer metastasis is very important, because of the involvement of different kinds of treatment and prognosis. These results suggest that genetic background might play an important role in tumorigenesis of PMN in the younger group, whereas epigenetic factors would be more important in the older group. Characteristic organ association and factors influencing carcinogenesis, such as aging, environmental carcinogens, and underlying genetic alterations in these tumors are to be further discussed.
| > References|| |
Warren S, Gates D. Multiple primary malignant tumors: A survey of the literature and a statistical study. Am J Cancer 1932:1358-414.
International Association of Cancer Registeries. Multiple Primeries Internal Report No. 00/003;2000Lyon: IARC .
Cohn AM, Peppart SB. Multiple primary malignant tumors of the head and neck. Am J Otolaryngol 1980;1:411-7.
Bruke M. Multiple primary cancers. Am J Cancer 1936;27:316-25.
Gluckman JL, Crissman JD. Survival rates in 548 patients with multiple neoplasm of the upper aerodigestive tract. Laryngoscope 1983;93:71-4.
Slaughter DP, Southwick HW, Smejkl W. ′Field cancerization′ in oral stratified squamous epithelium. Cancer 1953;6:963-8.
Horri A, Han HJ, Shimada M, Yanagisawa A, Kato Y, Ohta H, et al
. Frequent replication errors at micro satellite loci in tumors of patients with multiple primary cancers. Cancer Res 1994;54:3373-5.
Gertig D, Hunter D. Ovarian cancer. In: Adami H-O Hunter D, Trichopoulos D, editors. Textbook of Cancer Epidemilogy. Oxford: Oxford University Press; 2002. p. 378-99.
Oksenhendler E, Boulanger E, Galicier L, Du MQ, Dupin N, Diss TC, et al
. High incidence of Kaposi sarcoma-assiciated herpesvirus-related non-Hodgkins lymphoma in patients with HIV infection and multicentric Castleman disease. Blood 2002;99:2331-6.
Storm HH, Jensen OM, Ewertz M, Lynge E, Olsen JH, Schou G, et al
. Summary: Multiple primary cancers in Denmark, 1943−1980. Natl Cancer Inst Monogr 1985;68:41-430.
Van Leeuwen FE, Travis LB. Second cancers. In DeVita VT, Hellman S, Rosenberg SA, editors. Cancer: Principles and Practice of Oncology. 6 th
ed. Philadelphia: Lippincott, Williams and Wilkins; 2001. p. 2939-64.
Ikeda Y, Tsukuda M, Ishitoya J, Arai Y, Matsuda H, Katori H, et al
. Four cases of simultaneous triple primary cancers of the hypopharynx, esophagus, and stomach. Otolaryngol Head Neck Surg 2005;132:788-93.
Kohmura T, Hasegawa Y, Matsuura H, Terada A, Takahashi M, Nakashima T. Clinical analysis of multiple primary malignancies of the hypopharynx and esophagus. Am J Otolaryngol 2001;22:107-10.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]