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\begin{document}

\title{Adenoid Cystic Carcinoma of Buccal Mucosa: A Report of Two Rare Cases
and Review of Literature}



\author[1]{abbas karimi}%
\author[1]{Alireza Parhiz}%
\author[1]{Negin Eslamiamirabadi}%
\author[1]{monir Moradzadeh}%
\author[1]{samira derakhshan}%
\affil[1]{Affiliation not available}%


\vspace{-1em}



  
  \date{May 30, 2024}


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\selectlanguage{english}
\begin{abstract}
Adenoid Cystic Carcinoma (AdCC) is a malignant tumor mostly occurring in
the head and neck salivary glands. In this article, two rare cases of
adenoid cystic carcinoma occurring in the buccal mucosa with their
treatment and long-term follow-up are presented.%
\end{abstract}%



\sloppy


\textbf{Introduction}

Adenoid Cystic Carcinoma (AdCC) was initially described as
''cylindroma'' in 1856 by Billroth, as it contained long amorphous
compartments named ''cylinders'' in its histological view. Later on, the
term ``Adenoid Cystic Carcinoma''(AdCC) replaced the term cylindroma for
defining this tumor(1). AdCC accounts for 10\% of salivary gland tumors,
and for approximately 1\% of all malignancies of head and neck(2). AdCC
arises more frequently in minor salivary glands, in comparison to major
salivary glands(3). AdCC of minor salivary glands are believed to have a
worse prognosis, compared to those of the major salivary glands. Pain
can be a paramount symptom of the disease, due to the tumor's proneness
towards perineural invasion(4). Although this tumor is likely to occur
at almost any age, it is most commonly observed in women, in the
5\textsuperscript{th} and 6\textsuperscript{th} decades of life(5). In a
recent study, the AdCC occurrence rate in women and men was 60:40,
respectively(6).

AdCC often manifests itself as a small and slowly-growing tumor.
However, it is diagnosed at an advanced stage in most cases(7). It grows
with a slower rate, in comparison to other carcinomas, and has a low
prevalence of spreading into local and regional lymph nodes.
Nevertheless, local and distant recurrences and also hematogenous spread
are relatively common(3). Distant metastasis is quite common, with the
highest prevalence in the lungs, followed by bones, liver and the
brain(8).

In this article, two cases of adenoid cystic carcinoma which occurred in
the right buccal mucosa are presented.

\textbf{Case Presentation:}

Case No. 1:

A 39-year-old female was referred to the department of oral and
maxillofacial surgery with the chief complaint of a mass in the right
buccal mucosa, which had been formed more than 2 months ago. In the
intraoral examination, an ulcerative sessile painless exophytic mass in
the right buccal mucosa was observed. Lymphadenopathy was not detected.
No past medical or allergic history was found. Computered tomography
scan (CT-scan) of the patient showed large massin right buccal mucosa
which is attached to superficial skinand also due to bone depression in
right zygomatic bone (figure 1). Incisional biopsy was carried out. The
histopathologic examination revealed a neoplasm composed of small
hyperchromatic basaloid cells arranged mostly in cribriform, and
occasionally in solid and tubular patterns within a fibromyxoid stroma.
The cyst-like spaces among the tumoral cells contained eosinophilic or
basophilic material. The tubular pattern consisted of small ducts lined
by several cuboidal cells which contained hyalinized material (figure
2). Immunohistochemistry (IHC) was performed to confirm the diagnosis
and to rule out other adenocarcinomas containing basaloid cells. C-kit
antigen was diffusely positive in the tumoral cells. P63 was positive
and scattered. Ki67 was positive in about 10\% of the tumoral cells
(figure 2).

These results were compatible with adenoid cystic carcinoma with a
salivary gland origin. The patient underwent examinations to rule out
distant metastasis by computed tomography of the chest and abdominal
areas. Fortunately, no evidence of distant metastasis was observed.

The lesion was excised completely and reconstruction of the surgical
area was done by pedicle temporalis muscle flap (figure 3). Adjuvant
radiotherapy was also carried out. After 18 months of follow up, the
patient is alive without any noticeable problems (figure 4).

Case No. 2:

A 37-year-old male was presented to the department of oral and
maxillofacial surgery with a chief complaint of a mass in the buccal
mucosa of the left side. Intraoral examination revealed a pink
submucosal lesion with a duration of 8 months. Incisional biopsy was
conducted. The histopathologic examination indicated a neoplastic lesion
composed of islands of myoepithelial and ductal cells that were arranged
in the tubular pattern in some areas. The tumor cells were small and
cuboidal exhibiting basophilic nuclei and scant cytoplasm. The IHC
findings were compatible with adenoid cystic carcinoma (figure 5).

The lesion was surgically excised and post-operative radiotherapy was
performed for the patient. The patient suffered from trismus for 18
months after treatment due to the complications of surgery and
radiotherapy. No distant metastasis was eventually detected in the
patient's workups every three months.

\textbf{Discussion:}

The world health organization (WHO) has defined AdCC as ''a basaloid
tumor containing epithelial and myoepithelial cells in diverse
morphological configurations, such as tubular, cribriform, and solid
patterns. Its clinical course is relentless and usually has a fatal
outcome''(9).

A review of literature was carried out in this study, which is
demonstrated in table 1.

Eleven cases of AdCC in the buccal mucosa have been reported via
articles thus far. In this article, two other cases of AdCC in the
buccal mucosa have been reported. The first case was a 39-year-old
female with a painless exophytic mass in the buccal mucosa for more than
two months. The second case was a 37-year-old male with a mass in the
left buccal mucosa which had been existent for 8 months.

In Vidyalakshmi et al's case, the swelling had arisen after a left upper
posterior tooth was extracted. Furthermore, a single submandibular lymph
node on the left side of the patient's face was palpable, a phenomenon
rarely occurring in adenoid cystic carcinoma. The lymph node was firm,
mobile, non-tender, and had a size of less than 1 cm(14).

The patient in Dalirsani et al's study had a leukoedema accompanying the
AdCC lesion on the same side. Superficial ulceration was evident in the
aforementioned tumoral lesion, unlike the reported cases in this
article, which were free of ulcers. At the time, a stage I diagnosis
(T\textsubscript{2}N\textsubscript{0}M\textsubscript{0}) was made, and
the patient underwent 35 cycles of radiotherapy and chemotherapy. Two
years later, the patient was once again referred to the department, with
a chief complaint of a 1.5 cm firm mass on his right frontal region.
Following an incisional biopsy, a diagnosis of Adenoid Cystic Carcinoma
was established, and the patient was referred for chemotherapy and
further investigations to an oncologist. Following a CT-scan, a stage IV
lung metastasis was diagnosed. The patient underwent 3 cycles of
cisplatin and 5-fluorouracil chemotherapy, which were ineffective in
reducing the tumor's size. Therefore, 3 more cycles were carried out
with Taxol and carboplatin. The patient remained under oncologists'
supervision until date(16).

In the case reported by Bansal et al, the patient who had received
treatment for adenoid cystic carcinoma of the buccal mucosa in 2005, was
referred again, with a chief complaint of coughs for 4 months,
breathlessness for the last 2 months, and fever for 20 days. A CECT scan
view of thorax and upper abdomen identified parenchymal metastatic
deposits. Multiple osseous metastatic deposits and a few small
mediastinal lymph nodes were observed. Fine needle aspiration cytology
(FNAC) confirmed that the metastatic deposits belonged to the Adenoid
Cystic Carcinoma, of which the patient was diagnosed with in 2005. The
patient was referred to the department of oncology for further
management(8).

In Kumar et al's study (2018), Brain MRI and Chest X-ray revealed no
evidence of distant metastases(17).

There are three histopathological views for adenoid cystic carcinoma:
the cribriform, tubular, and solid patterns. The cribriform pattern,
being the most common, has a view of islands of basaloid cells,
surrounded by cyst-like spaces in different sizes, forming a ''Swiss
cheese'' pattern. The tubular histologic subtype has a closely similar
display, but with cells arranged and organized in nests surrounded by
different amounts of often hyalinized eosinophilic stroma. The solid
subtype manifests aggregates of basaloid cells without tubular or
pseudocystic formations(3).Since polymorphism is a common phenomenon in
AdCC, it's possible to see all three aforementioned patterns in one
specimen. Therefore, MD Anderson introduced a pathological grading
system, to which is now contributed worldwide(19):

Grade I: tubular and cribriform together, without a solid pattern.

Grade II: mostly cribriform, with less than 30\% of solid pattern.

Grade III: solid being the predominant subtype.

The differential diagnosis includes polymorphous low grade
adenocarcinoma (PLGA), salivary duct carcinoma, and basaloid squamous
cell carcinoma(18). PLGA demonstrates large tumoral cells than AdCC with
vesicular nuclei versus small basaloid cells of AdCC. Single file
appearance can also help to distinguish PLGA from AdCC. Salivary duct
carcinoma (SDC) does not show cribriform pattern of AdCC and PLGA. On
the other hand, SDC is a high grade carcinoma with prominent nuclear
pleomorphism and atypical mitotic figures. Comedo-type necrosis can help
to diagnosis between SDC and other tumors of salivary glands(20).
Basaloid squamous cell carcinoma (BSCC) is a high grade malignancy of
keratinocytes which can mimic AdCC, histopatologically. Superficially
squamous cell carcinoma, large eosinophilic cytoplasm of tumoral cells
and keratin pearls can help to distinguish between BSCC and AdCC.
Ancillary studies such as immunohistochemistry examinations can help to
definitive diagnosis.

Perineural invasion, sometimes associating Adenoid Cystic Carcinoma, is
defined as ''a form of direct primary spread of neoplasm which may not
necessarily be macroscopically continuous with the main focus of the
tumor, but is usually microscopically continuous''. The second and third
branches of the trigeminal nerve are most frequently affected. The
descending branches of the facial nerve and smaller cranial branches can
also get involved. Perineural involvement is claimed to be an indicator
of poor prognosis(19). Furthermore, it can increase the chances of
recurrence. Recent data argues that intraneural invasion, rather than
perineural, can have a higher impact on the survival rate in the AdCC of
the head and neck(3).

In Naik et al's case, slight perineural thickening of the facial nerve
was observed. In a more precise examination, perineural and intraneural
invasion were evident, due to which the patient had mild degrees of
facial palsy(13).

Contrary to other types of carcinomas, distant hematogenous metastases
are much more common than regional lymph node metastases in AdCC.
Hematogenous metastases in AdCC can remain asymptomatic for a
considerable period of time, especially lung metastases, which
apparently has a slow progress rate(19).

Singh et al (2010) used paraclinical assessments, such as
ultrasonography of upper abdomen, non-contrast proton MRI of the oral
cavity, PA view of the chest, axial CT scan of the head and face,
lateral neck radiograph, submentovertex view of the skull (35\selectlanguage{ngerman}°), PA view
of the pelvic girdle, and CT scan of chest, to rule out metastasis(10).

The primary goal of treatment for patients affected with AdCC is local
control of the tumor, normal function, and preventing distant
metastases(21). Radical surgery with wide resection margins may not be
sufficient per se, as achieving disease-free margins can be difficult
due to AdCC's propensity to perineural invasion and some lesions'
challenging anatomical access(2). Radiation is noncompulsory for small
tumors (T\textsubscript{1}N\textsubscript{0}) but must be considered for
cases having low-grade tumors in association with perineural invasion,
or evidence of tumor seeding during surgery. A lower radiation dose has
also been recommended for patients with tumors located in lymphatically
rich areas (22). Using radiotherapy as primary treatment is proposed
when surgery is not feasible(3). In addition, radiation can be a
standard treatment for alleviating bone and brain metastases(23).

As AdCC's sensitivity to chemotherapy is relatively low, systemic
chemotherapy for AdCC remains controversial. On the other hand,
chemotherapeutic treatment has proven to be effective in a rather low
percentage of patients with recurrences or metastases. The first-line
chemotherapy choice is highly dependent on patients' comorbidities and
characteristics(2).

Tumors of the minor salivary glands can more easily infiltrate the
surrounding extra-glandular tissues, increasing dissemination of the
tumor cells and thus rendering resection with disease-free margins more
difficult. The cribriform variant is believed to have the best prognosis
while the solid pattern has the worst, with the tubular subtype having
an intermediate-level prognosis(19).

\textbf{Conclusion:}

Adenoid cystic carcinoma is a malignant tumor characterized by features
such as slow growth, high infiltration potential, and hematogenous
distant metastasis. Minor salivary grand AdCCs should receive aggressive
treatment to achieve negative surgical margins to inhibit recurrence. In
this article, two cases of adenoid cystic carcinomas having occurred in
the buccal mucosa were reported. For both cases, long-term follow-ups
were carried out, which are essential for monitoring signs of recurrence
or distant metastasis.

\textbf{Patient Consent:}

The authors of this article have obtained all required consent forms
from both patients. Their consent has been given for their images and
other clinical information to be reported in this article without
including their names.

\textbf{Author contribution:}

Abbas Karimi and Alireza Parhiz: patients's treatment and follow up
modalities; Negin Eslamiamirabadi: manuscript writing and data
collection; Monir Moradzadeh Khiavi: pathologic report; Samira
Derakhshan: manuscript writing, pathologic report and supervision.

\textbf{References:}

1. Dutta NN, Baruah R, Das L. Adenoid cystic carcinoma - Clinical
presentation and cytological diagnosis. Indian journal of otolaryngology
and head and neck surgery : official publication of the Association of
Otolaryngologists of India. 2002;54(1):62-4.

2. De Berardinis R VA, Micarelli A, Alessandrini M, Bruno E. Adenoid
Cystic Carcinoma of Head and Neck. Am J Otolaryngol Head Neck Surg.
2018;1(2):1010.

3. Dillon PM, Chakraborty S, Moskaluk CA, Joshi PJ, Thomas CY. Adenoid
cystic carcinoma: A review of recent advances, molecular targets, and
clinical trials. Head \& Neck. 2016;38(4):620-7.

4. Giannini PJ, Shetty KV, Horan SL, Reid WD, Litchmore LL. Adenoid
cystic carcinoma of the buccal vestibule: A case report and review of
the literature. Oral Oncology. 2006;42(10):1029-32.

5. Yaga US, Gollamudi N, Mengji AK, Besta R, Panta P, Prakash B, et al.
Adenoid cystic carcinoma of the palate: case report and review of
literature. The Pan African medical journal. 2016;24:106.

6. Jesse Jaso, Reenu Malhotra. Adenoid Cystic Carcinoma. Archives of
Pathology \& Laboratory Medicine. 2011;135(4):511-5.

7. Michel G, Joubert M, Delemazure AS, Espitalier F, Durand N, Malard O.
Adenoid cystic carcinoma of the paranasal sinuses: Retrospective series
and review of the literature. European Annals of Otorhinolaryngology,
Head and Neck Diseases. 2013;130(5):257-62.

8. S Bansal , K Goyal , Ahir G. Metastasis of adenoid cystic carcinoma
of buccal mucosa to lungs - a case report with review of literature.
IJCMR. 2016;3(10):3066-8.

9. Barrett AW, Speight PM. Perineural invasion in adenoid cystic
carcinoma of the salivary glands: A valid prognostic indicator? Oral
Oncology. 2009;45(11):936-40.

10. Singh S, Gokkulakrishnan, Jain J, Pathak S, Singh KT. Adenoid cystic
carcinoma of buccal mucosa. J Maxillofac Oral Surg. 2010;9(3):273-6.

11. Ajila V, Hegde S, Nair GR, Babu SG. Adenoid cystic carcinoma of the
buccal mucosa: A case report and review of the literature. Dent Res J
(Isfahan). 2012;9(5):642-6.

12. Kumar AN, Harish M, Alavi YA, Mallikarjuna R. Adenoid cystic
carcinoma of buccal mucosa. BMJ Case Rep. 2013;2013:bcr2013009770.

13. Naik K, Shetty P, Hegde P. Adenoid cystic carcinoma of buccal mucosa
with extensive hyalinization: A unique case report. Annals of Tropical
Medicine and Public Health. 2013;6(5):571-4.

14. S V, R A. Adenoid cystic carcinoma of the buccal mucosa: a case
report with review of literature. J Clin Diagn Res. 2014;8(3):266-8.

15. Garg V, Roy S, Khanna KS, Bakshi PS, Chauhan I. Adenoid Cystic
Carcinoma of Buccal Mucosa: A Rare Case Report. Indian J Otolaryngol
Head Neck Surg. 2016;68(3):370-3.

16. Dalirsani Z, Mohtasham N, Pakfetrat A, Delavarian Z, Ghazi A, Rahimi
SA, et al. Adenoid Cystic Carcinoma of the Buccal Mucosa with Rare
Delayed Frontal Bone Metastasis: A Case Report. Journal of Dental
Materials and Techniques. 2016;5(4):208-12.

17. A K, Rawat G, S G, S D. Adenoid Cystic Carcinoma of Buccal Mucosa: A
Rare Presentation. Journal of Dental and Maxillofacial Surgery.
2018;1(1):37-41.

18. Venkatesh V, Murugan P. Solid variant of adenoid cystic carcinoma of
oral cavity: A diagnostic difficulty in histopathological examination. .
J Oral Med, Oral Surg, Oral Pathol, Oral Radiol. 2018;4(4):186-9.

19. Pinakapani R, Chaitanya NC, Lavanya R, Yarram S, Boringi M, Waghray
S. Adenoid Cystic Carcinoma of the Head and Neck--literature review.
Qual Prim Care. 2015;23(5):309-14.

20. Sadeghi HM, Karimi A, Rahpeima A, Derakhshan S. Salivary Duct
Carcinoma with Unusual Cutaneous Metastasis; A Case Report. Iran J
Pathol. 2020 Summer;15(3).

21. Triantafillidou K, Dimitrakopoulos J, Iordanidis F, Koufogiannis D.
Management of Adenoid Cystic Carcinoma of Minor Salivary Glands. Journal
of Oral and Maxillofacial Surgery. 2006;64(7):1114-20.

22. Balamucki CJ, Amdur RJ, Werning JW, Vaysberg M, Morris CG, Kirwan
JM, et al. Adenoid cystic carcinoma of the head and neck. American
Journal of Otolaryngology. 2012;33(5):510-8.

23. Brill Ii LB, Kanner WA, Fehr A, Andrén Y, Moskaluk CA, Löning T, et
al. Analysis of MYB expression and MYB-NFIB gene fusions in adenoid
cystic carcinoma and other salivary neoplasms. Modern Pathology.
2011;24:1169.\selectlanguage{english}
\begin{longtable}[]{@{}lllllllllllll@{}}
\toprule
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Age \& Sex}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Pain}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Duration}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Consistency}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Paraclinical} \textbf{auxiliary} \textbf{examinations}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Size}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{AdCC Subtype}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Perineural Invasion}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Metastasis}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Lymph Node Involvement}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Treatment}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Follow up}\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Singh(10)} \textbf{(2010)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
50/M\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
yes\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
1.5 year\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
firm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
3 cm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Cribriform\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Negative\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Excision under general anesthesia\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Till date(for 3 years)\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Ajila \&} \textbf{Hegde(11)} \textbf{(2012)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
45/F\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Yes (mild and continuous)\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
3 months\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
soft to firm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Panoramic radiography Ultrasonography\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
1\selectlanguage{ngerman}×1 cm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Cribriform\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
yes\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Excision without adjuvant therapy\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
3 years\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Kumar(12)} \textbf{(2013)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
26/F\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Yes (in palpation)\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
1 year\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
hard\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
3cm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Cribriform\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Negative\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Negative\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Complete surgical removal\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
till date\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Naik(13)} \textbf{(2013)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
48/M\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Yes\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
1 month\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
firm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Conventional radiography Ultrasonography MRI\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
3\selectlanguage{ngerman}×3×2 cm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Cribriform\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
yes (\& intraneural invasion)\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Excision without adjuvant therapy\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
10 years\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Vidyalakhshmi(14)} \textbf{(2014)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
48/F\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
no\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
1 year\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
firm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Panoramic radiography\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
2\selectlanguage{ngerman}×3 cm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Cribriform\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Excision \& referral to oncologist\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
6 months\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Garg(15)} \textbf{(2016)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
40/F\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
yes\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
1 year\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
firm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
1.5\selectlanguage{ngerman}×1 cm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Solid (with few areas of cribriform and tubular)\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Excision under local anesthesia\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
till date\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Dalirsani(16)} \textbf{(2016)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
47/M\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Yes (in eating and palpation)\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
3 years\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
firm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Sonography CT scan\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
3 cm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Cribriform\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Positive\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Negative\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Excision, Radiotherapy and Chemotherapy\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
till date\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Bansal(8)} \textbf{(2016)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
5/M\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
9 years\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
yes\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Excision,Neck dissection, Radiotherapy and Chemotherapy, referral to the
department of oncology\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Kumar(17)} \textbf{(2018)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
16/M\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Yes\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
4 months\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
firm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Sialadenoscopy Sialography face and neck MRI\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
1.08\selectlanguage{ngerman}×0.7× 1.13 cm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Cribriform\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
yes\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Negative\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Negative\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Excision, Radiotherapy\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
1 year\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Venkatesh(18)} \textbf{(2018)} \textbf{(case 1)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
70/M\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
2\selectlanguage{ngerman}×1×1 cm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Solid\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
no\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Excision\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage}\tabularnewline
\begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
\textbf{Venkatesh(18)} \textbf{(2018)} \textbf{(case 2)}\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
60/M\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
0.5\selectlanguage{ngerman}×0.2 cm\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
Solid\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage} & \begin{minipage}[t]{0.07\columnwidth}\raggedright\strut
-\strut
\end{minipage}\tabularnewline
\bottomrule
\end{longtable}

Table 1 -- Literature review of the reported cases of Adenoid Cystic
Carcinoma of the buccal mucosa (2010 -- present)

Figure legends:

Figure 1- CT-scan view of the lesion demonstrates ill-defined mass of
right buccal area with invasion to the surrounding structures of the
skin and zygomatic bone.

Figure 2- Histopathologic views of the lesion. a) small basaloid tumoral
cells in various sized nests (H\&E staining, \selectlanguage{ngerman}×100 magnification). b)
classic cribriform pattern in tumoral cells (H\&E staining, ×100
magnification). c) immunohistochemistry results show severe diffuse
positive immunoreaction for C-kit (×100 magnification). d) ki67 was
positive in about 10\% of tumoral cells.

Figure 3- Surgical intervention. a) whole excited lesion with safe
margin. b) excited zygomatic bone and maxillary tuberosity. c) pedicled
right temporalis muscle flap was used for reconstruction of the surgical
area. d) transferred flap to the surgical area.

Figure 4- Follow up of the patient after one and half year shows ideal
healing without significant problem.

Figure 5- a) microscopic examinations shows tubular pattern of small
basaloid tumoral cells (H\&E staining, ×100 magnification). b) tumoral
cells demonstrate admixture of cribriform and tubular pattern (H\&E
staining, ×100 magnification). c) diffuse positive C-kit immunoreaction
in tumoral cells (IHC, ×400 magnification). d) positive immunoreaction
for ki67 in about 10\% of tumoral cells (IHC, ×400 magnification).\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/1/1}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/2a/2a}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/2b/2b}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/2c/2c}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/2d/2d}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/3a/3a}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/3b/3b}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/3c/3c}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/3d/3d}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/4/4}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/5a/5a}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/5b/5b}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/5c/5c}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/5d/5d}

\end{center}
\end{figure}

\selectlanguage{english}
\FloatBarrier
\end{document}

