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Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology 28 (2016) 197201

Contents lists available at ScienceDirect

Journal of Oral and Maxillofacial Surgery,


Medicine, and Pathology
journal homepage: www.elsevier.com/locate/jomsmp

Case Report

A rare case of mucous adenocarcinoma with gingival metastasis:


A case report and review of literature
Sumit Majumdar 1 , Divya Uppala 2 , Rajyalakshmi Boddepalli 3 , Ayyagari Kameswara Rao
Department of Oral Pathology and Microbiology, GITAM Dental College and Hospital, Rushikonda, Visakhapatnam 530045, India

a r t i c l e

i n f o

Article history:
Received 25 March 2015
Received in revised form 27 August 2015
Accepted 2 September 2015
Available online 21 October 2015
Keywords:
Metastasis
Mucous adenocarcinoma
Stomach
Immunohistochemistry

a b s t r a c t
Oral cavity metastatic tumors are considered rare and represent approximately 1% of all oral malignancies. Because they are rare and due to atypical clinical and radiographic presentation, metastatic lesions
are considered a diagnostic challenge. Primary tumors which metastasize to the oral cavity are most
commonly from lung, breast, and kidney and colon. Oral cavity metastases represent distant spread
and are associated with poor prognosis. The purpose of this report is to present a rare case of mucous
adenocarcinoma of stomach which metastasized to mandibular gingiva.
2015 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.

1. Introduction
Oral region is an uncommon site for metastatic tumor. It
accounts for only 1% of all oral malignant neoplasms and most commonly involve the posterior region of mandible [1]. Primary tumors
from breast, lung, kidney, bone and colon accounts for 70% of all oral
metastatic cases [1], while stomach accounts for only for 2.2% of all
cases [8]. Several types of carcinomas arise from the stomach, with
adenocarcinoma being the most common and is the second most
common cancer worldwide accounting for 3% to 10% of all cancer related deaths [3]. Gastric carcinoma can spread by direct local
invasion of adjacent organs, peritoneal spread, lymphatic spread or
by haematogenous dissemination [4,5]. Gastric carcinomas metastasize most commonly to liver, lung, and pancreas [4]. The oral
cavity is an uncommon site for metastatic spread of gastric adenocarcinoma very few cases are reported in literature and according

AsianAOMS: Asian Association of Oral and Maxillofacial Surgeons; ASOMP: Asian


Society of Oral and Maxillofacial Pathology; JSOP: Japanese Society of Oral Pathology; JSOMS: Japanese Society of Oral and Maxillofacial Surgeons; JSOM: Japanese
Society of Oral Medicine; JAMI: Japanese Academy of Maxillofacial Implants.
Corresponding author. Tel.: +91 9440074890.
E-mail addresses: drsumitmajumdar1971@gmail.com (S. Majumdar),
uppala.divya@gmail.com (D. Uppala), drrajii@gmail.com (R. Boddepalli),
calmua@gmail.com (A.K. Rao).
1
Tel.: +91 9831132632.
2
Tel.: +91 9966413710.
3
Tel.: +91 9611129360.

to Sauerborn D et al. [4] only 18 cases were reported including their


case and our search revealed only 2 more such cases [5,6] of which
only 10 cases are reported in the mandible (Table 1).

2. Case report
A 60-year-old female patient reported to the hospital with
the chief complaint of non-healing socket and painless growth of
gums since 1 month (Fig. 1A). Her Medical history revealed that
she had gastritis and using proton pump inhibitors for the past
2 years and was anemic. Past dental history reveals an extraction of mobile mandibular right rst molar. She was a chronic
reverse smoker, quit the habit recently. On intra oral examination a pinkish soft tissue growth surrounding the extracted socket
was noted measuring about 2 cm 1 cm which bleeded on probing
and the extracted socket was dry (Fig. 1B). Extraoral examination revealed a painful, xed right submandibular lymph node.
The radiological ndings were insignicant to the clinical ndings (Fig. 2). Clinical diagnosis was made as pyogenic granuloma
and an incisional biopsy of the soft tissue growth was done under
local anesthesia. The Hematoxylin and Eosin stained soft tissue
section revealed cells with a peripherally located crescent shaped
nucleus and intracytoplasmic mucin, the cells are discohesive in
the connective tissue with few tubulo ductal areas (Fig. 3A and
B). Histochemical stains were performed in an attempt to localize and characterize the tumor cells. Histochemical studies using
Periodic acidSchiff staining revealed the strong positivity in the

http://dx.doi.org/10.1016/j.ajoms.2015.09.002
2212-5558/ 2015 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.

198

S. Majumdar et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology 28 (2016) 197201

Table 1

S. no

Sex/age

Site of soft tissue in oral cavity

Author/year

1.
2.
3.
4.
5.
6.
7.
8.
9.
10.
11.
12.
13.
14.
15.
16.
17.
18.
19.
20.
21.

F/63
M/58
M/51
F/65
M/59
M/65
M/69
M/56
M/66
M/60
M/65
M/65
M/56
F/61
M/65
F/82
M/58
M/70
M/50
M/50
F/60

Mandible
Mandible
Mandible
Maxilla
Maxilla
Soft palate
Mandible
Mandible
Maxilla
Mandible
Maxilla
Tongue
Mandible
Maxilla
Mandible
Mandible
Maxilla
Mandible
Maxilla
Maxilla
Mandible

Lund et al. (1968) [4]


Astacio and Alfaro (1969) [4]
Ohba et al. (1974) [4]
Lopez and Loboz (1976) [4]
Osaki et al. (1978) [4]
Arjona et al. (1989) [4]
Tojo et al. (1989) [4]
Hamakawa et al. (1993) [4]
Florio and Hurd (1995) [4]
Makino et al. (1997) [4]
Yajima and Miyazaki (1999) [4]
Yasumoto et al. (1999) [4]
Shimoyama et al. (2004) [4]
Colombo et al. (2005) [4]
Kwon et al. (2006) [4]
Nishide and Kanamura (2006) [4]
Hwang et al. (2007) [4]
Sauerborn et al. (2011) [4]
Umashankar et al. (2013) [5]
Manjunath et al. (2013) [6]
Present case

Fig. 1. (A) Extra oral and (B) Intra oral picture of 60 year old female patient.

cytoplasm of tumor cells, which was interpreted as mucin (Fig. 4A).


Immunohistochemically tumor cells showed intra cytoplasmic and
luminal positivity for MUC1 (Fig. 4B). Based on this a diagnosis of
mucous adenocarcinoma was made. The possibility of a metastatic

Fig. 2. Orthopantomogram.

lesion was considered because mucous adenocarcinoma may not


develop in the gingiva as the primary site. An immunohistochemical study was done to identify the most probable site of the primary
tumor. As the most common metastatic tumors are from breast
and lungs immuno proling was started with Cytokeratins 7 and
20 (CK7/20). The tumor cells are widely immunopostive for CK7.
Though the tumor cells were widely positive for CK7, they were
negative for Estrogen Receptor (ER), Napsin, TTF1 and focally positive for CK20. The tumor cells are strongly positive for Carcino
Embryonic Antigen (CEA) (Fig. 5). Based on the above immunohistochemical studies nal diagnosis of mucous adenocarcinoma
from stomach was conrmed and the patient was referred to an
oncologist for further treatment. Endoscopic examination showed
an ulceroproliferative growth of the gastric mucosa and the histological features of the endoscopic biopsy specimen demonstrated
the same histological pattern of the metastatic tumor (Fig. 6A and
B). Patient was not able to retain in any form solid or liquid diet due
to frequent regurgitation even under constant care and supervision.
Patient expired due to poor nutrition, before any further investigations could be performed to diagnose metastatic deposits in other
organs.

S. Majumdar et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology 28 (2016) 197201

199

Fig. 3. demonstrating mucous cells having a peripherally placed crescent shaped nucleus with intracytoplasmic mucin (A); with few tubulo ductal areas (B) (Hematoxylin
and eosin Stain); Original magnication 25 and 10, respectively.

3. Discussion
The primary metastases to soft tissues are extremely rare,
accounts only 1% of oral malignancies with a predilection for the
gingiva, tongue and post extraction socket [1]. The breast is the
most common site for tumors that metastasize to the jawbones,
whereas lung is common site for cancers that metastasize to the
oral soft tissues [2]. Most cancers are usually diagnosed at their initial site, because of the symptoms that are produced by the primary
tumor. Some 1015% of them, however, present as metastases in
solid organs, body cavities or lymph nodes. Most of these secondary
tumors are adenocarcinomas for which the commonest primary
sites are pancreas, hepatobiliary system, and lungs [7]. The most
common metastatic malignancies in women are from primary cancers in the breasts, genital organs, lungs and kidneys and in men
they are from the lungs, kidneys and prostrate. In 25% of such cases,
oral metastases were found to be the rst sign of the metastatic
spread. There was almost equal gender distribution in jawbone
metastases and a male to female ratio of 2:1 in oral soft tissues [10].

Possible routes of metastasis to oral cavity are by hematogenous


and lymphatic routes. Mandible is considered susceptible to metastasis because of the presence of red bone marrow, local blood
vessels branching and slowing of blood ow [1,2]. Probable reason for metastases to the head and neck area bypassing the lungs is
by Batsons plexus which is a valveless vertebral venous plexus that
might allow for the retrograde spread of tumor cells [1,4,5,9]. Hirshberg also suggested that inammation plays an important role
in attracting metastatic cells to the gingiva [2]. According to Hirshberg in 55 cases, tooth extraction preceded the discovery of the
metastases [1] both of which are correlative to the present case.
A wide range of non specic clinical signs and symptoms may be
seen in association with oral metastatic lesions including rapidity
in growth, pain, difculty in chewing, intermittent bleeding, altered
sensation, gum irritation, tooth loosening, exophytic masses of the
soft tissues, trismus and rarely pathologic fractures ultimately leading to poor quality of life [10]. Radiographic examination is rarely
considered diagnostically important as the lesion may not produce
a radiographic appearance in its initial stages. Hirshberg et al. found

Fig. 4. (A) The strong positivity in the cytoplasm of tumor cells, which was interpreted as mucin (Periodic acidSchiff staining); (B) Tumor cells with intracytoplasmic and
luminal positivity for MUC1; Original magnication 10.

200

S. Majumdar et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology 28 (2016) 197201

Fig. 5. (A) Tumor cells positivity for CK7; (B) focal positivity for CK 20; (C) negativity for ER; (D) negativity for TTF-1; (E) negativity for Napsin; (F) positivity for CEA. Original
magnication 10.

Fig. 6. (A) Ulceroproliferative growth of the gastric mucosa; (B) the same histological features of the metastatic tumor (Hematoxylin and eosin stain, Original magnication
10)

that out of 390 cases, 5.4% cases did not show any important radiographic change [2]. So, early detection of jaw metastasis can be
challenging and the delay in diagnosis may turn fatal to the patient.

Conict of interest
No conict of interest.

4. Conclusion
References
The discovery of an oral metastasis is extremely important for
better patient care by appropriate application of advanced diagnostic tools, starting with immunohistochemistry which plays a pivotal
role in developing countries as it is costeffective.

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Eur J Cancer B Oral Oncol 1995;3:35560.
[2] Hirshberg A, Leibovich P, Buchner A. Metastatic tumor to the jawbones: analysis
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[7] John D, Hainsworth F, Anthony Greco. Adenocarcinoma of Unknown Primary Site. Holland-Frei Cancer Medicine. 6th ed. Hamilton, ON: BC Decker;
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