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Case Report

A Case of Synchronous Dual Malignancy with the Index

Malignancy in the Lower Alveolus and a Second Primary
Malignancy in the Left Breast
Satya Prakash Agarwal1, Jayendra K. Arya2, Priyanka Gupta2*, Bina Ravi2, Anjum Syed2
1
Department of Surgical Oncology, AIIMS, Rishikesh, Uttarakhand, India, 2Department of Integrated Breast Care Centre, AIIMS, Rishikesh, Uttarakhand, India

Abstract
Multiple primary malignancies are nowadays commonly detected due to better radiological techniques, greater awareness and an increase
in number of elderly cancer survivors. We are reporting a rare case of patient having dual synchronous malignancy of carcinoma left lower
alveolus and ductal carcinoma in left breast who presented to the department of surgical oncology and integrated breast care center. The patient
is on regular follow‑up, and currently, there was no evidence of recurrent or residual disease. No such case has ever been reported, and despite
a thorough check on all, we are unable to find any previously reported case. Hence, to the best of our knowledge, this case is the first of its
kind to have simultaneous presentation of synchronous dual malignancies involving left lower alveolar and left breast cancers.

Keywords: Alveolar and ductal carcinoma, squamous cell carcinoma, synchronous malignancy

Introduction
Multiple primary malignancies are uncommon and suggest
genetic predisposition due to mutations in cell line and
persistent exposure to one or more etiological risk factor(s);
however, due to better diagnostic techniques they are now-a-
days more frequently detected.
Case Report
A 62‑year‑old female was referred to the department of
surgical oncology with difficulty in chewing and mouth
opening for 6 months. On examination, there was only one
finger mouth opening. Ultrasound showed a 5 cm × 3 cm
lesion over the lower alveolus extending anteriorly up to
canine, posteriorly till second molar and laterally up to buccal
mucosa. No lymph nodes were palpable in the cervical region.
These findings were further confirmed on contrast‑enhanced
computed tomography neck and face which further showed
underlying bony erosion of the mandible which was not visible
on clinical examination, following which biopsy was done
which showed squamous cell carcinoma of the lower alveolus.
The patient also complained of lump in the left breast for 6
months and was referred to Integrated Breast Care Center.
No history of radiation to the head‑and‑neck area before the
examination was there. The breasts were examined, and triple
assessment of bilateral breast was done. On examination, the
left breast had a 3 cm × 3 cm lump. On mammography and
ultrasound correlation, left breast BIRADS 5 (Breast Imaging
Reporting and Data System) mass lesion was detected, and
further, fine‑needle aspiration cytology and biopsy from left
breast lesion were done. Histopathology reported invasive
carcinoma (modified Bloom–Richardson Grade III). Positron
emission tomography–computed tomography was also done
which showed metabolically active lesion in the left breast at
9 o’clock position and in the left retromolar trigone eroding
the mandible. The clinical tumour, node and metastasis (TNM)
stage of oral cavity was cT4aN0M0.
Address for correspondence: Dr. Priyanka Gupta,
Department of Integrated Breast Care and Centre, AIIMS, Rishikesh,
Uttarakhand, India.
E‑mail: dr.priyanka2jan@gmail.com

Received: 25‑05‑2020 Revised: 30‑05‑2020 Accepted: 09‑06‑2020 Available Online: 20-07-2020

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How to cite this article: Agarwal SP, Arya JK, Gupta P, Ravi B, Syed A.
DOI: A case of synchronous dual malignancy with the index malignancy in the
10.4103/JME.JME_22_20 lower alveolus and a second primary malignancy in the left breast. J Med
Evid 2020;1:33-4.

© 2020 Journal of Medical Evidence | Published by Wolters Kluwer - Medknow 33

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Agarwal, et al.: Primary synchronous dual malignancy of lower alveolus and left breast
A team of doctors from different specialties including surgical
oncology, anaesthesiology, breast onco‑surgery, plastic and
reconstructive surgery, radiation oncology and psychiatry
was formed. The patient was admitted and planned for
upfront surgery of oral cavity lesion and breast cancer. The
patient consented for modified radical mastectomy (MRM)
instead of breast conservation surgery. Left MRM was done
by the breast onco‑surgeon. Composite resection involving
segmental mandibulectomy of the left lower canine to the
ramus and modified radical neck dissection Type III was done
by the team of surgical oncology. Reconstruction with tongue
advancement flap and sternocleidomastoid rotation flap was
done by the team of plastic and reconstructive surgery at the
same time.
Intraoperative finding was 5 cm × 4 cm ulceroproliferative
growth at the left lower alveolus, extending anteriorly from the
left first premolar, posteriorly involving the retromolar trigone,
medially extending on the floor of the mouth and laterally
going into the lower gingivobuccal sulcus with extension on
buccal mucosa.
The total operative time was approximately 4 h. The breast and
oral cavity specimens were sent to the pathology department.
The TNM stage for alveolar cancer was T4aN0Mx and for
breast cancer was T2N0Mx. The post‑operative patient was
stable and discharged on the 7th day. The patient was further
sent to the radiation oncology department after 6 weeks for
adjuvant radiotherapy (RT).
Discussion
Multiple primaries were first reported by Billroth and Reimer.
The patient with a known cancer may have second primary
malignancy (SPM) which is defined as malignant neoplasm in a
patient with a known cancer.[1] There are few criteria laid down
by Warren and Gates that help in the diagnosis of SPM which
are; tumor should present a definite picture of malignancy;
each tumour should be histologically distinct; the possibility
that one is a metastasis of the other must be excluded, and if
present in the same organ, they must be at least be 2 cm apart.
If the tumours are in the same location, then they should be
separated in time by at least 5 years.[2]
The North American Association of Central Cancer Registries
has classified multiple primary tumours into two categories: (a)
synchronous, in which the cancers occur at the same time (the
Surveillance, Epidemiology and End Results Program
definition is within 6 months), and (b) metachronous, in which
the cancers follow in sequence, that is, more than 6 months
apart.[3]
‘Field cancerisation’ is thought to be common carcinogen‑
induced multiple cancers in an exposed epithelial surface and
is theorised to be the pathophysiology behind the occurrence of
multiple primary malignancies in the head‑and‑neck region.[4]
Other possible causal factors include tumor and patient
characteristics including persistent carcinogen exposure
34
from environment, progressive ozone depletion and effects
of ionizing radiation, increased use of organ transplant,
and the increasing use of newer treatment modalities like
hormonal manipulation, target therapies, genetic manipulation,
immunomodulators, HER2 receptor positivity, low estrogen
receptor expression level, and BRCA1 or BRCA2 mutation,
along with mutation in tumor suppressor genes such as p16,
p53, PTEN, and Rb gene has an effect on the risk of secondary
primary malignancy.[5-7]
Conclusion
The present case of synchronous dual primary malignancy
of the lower alveolus and breast was confirmed by
immunohistochemistry and pathological analysis. Simultaneous
removal of resectable primary malignancies should be
attempted, and RT/chemotherapy should be considered.
A regular follow‑up after treatment of such patients should
be performed.
Acknowledgment
This study has been seen and approved by all authors; it is an
original contribution that is not previously published; it is not
under consideration for publication elsewhere.
Declaration of patient consent
The authors certify that they have obtained all appropriate
patient consent forms. In the form the patient(s) has/have
given his/her/their consent for his/her/their images and other
clinical information to be reported in the journal. The patients
understand that their names and initials will not be published
and due efforts will be made to conceal their identity, but
anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
References
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51 Lectures-A handbook for students and doctors, 14th Edition,
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4. Hsieh WC, Chen YM, Perng RP. Temporal relationship between cancers
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Risk of second primary malignancy in breast cancer survivors: A nested
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Journal of Medical Evidence ¦ Volume 1 ¦ Issue 1 ¦ May-August 2020