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j.joms.2018.08.003

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Accepted Manuscript
Palatal Basal Cell Adenocarcinoma from misdiagnosis to cure: a unique case report
and review
Ramzey Tursun, DDS, FACS, Dhafer Alsuwied, DDS, Lieutenant Commander J
Marshall Green, III, DDS, FACS, James Melville, DDS, FACS
PII:
S0278-2391(18)30878-4
DOI:
10.1016/j.joms.2018.08.003
Reference:
YJOMS 58413
To appear in:
Journal of Oral and Maxillofacial Surgery
Received Date: 4 May 2018
Revised Date:
7 August 2018
Accepted Date: 7 August 2018
Please cite this article as: Tursun R, Alsuwied D, Green III JM, Melville J, Palatal Basal Cell
Adenocarcinoma from misdiagnosis to cure: a unique case report and review, Journal of Oral and
Maxillofacial Surgery (2018), doi: 10.1016/j.joms.2018.08.003.
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ABSTRACT TITLE:
Palatal Basal Cell Adenocarcinoma from misdiagnosis to cure: a unique case report and review.
AUTHORS:
Ramzey Tursun, DDS, FACS, Dhafer Alsuwied DDS, LCDR J Marshall Green III DDS, James Melville
DDS, FACS
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INSTITUTIONAL AFFILIATION:
1) Ramzey Tursun, DDS, FACS University of Miami- Miller School of Medicine, Miami Fl
Associate Professor of Clinical Surgery
Director, Head & Neck Oncologic and Microvascular Reconstructive Surgery
2) Dhafer Alsuwied DDS University of Texas Health Science Center at Houston
Fellow- Oral, Head & Neck Oncology and Microvascular Reconstructive Surgery
3) Lieutenant Commander J Marshall Green III DDS, FACS
Associate Program Director- Naval Medical Center Portsmouth
Assistant Professor of Surgery-Uniformed Services University of Health Sciences
Adjunct Assistant Professor of Surgery- University of Miami-Miller School of Medicine
4) James Melville DDS, FACS University of Texas Health Science Center at Houston
Assistant Professor of Surgery
Oral, Head & Neck Oncology and Microvascular Reconstructive Surgery
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CORRESPONDING AUTHOR AND CONTACT INFORMATION:
Ramzey Tursun DDS
Associate Professor of Surgery
Director of Head and Neck Oncology and Microvascular Reconstructive Surgery
Division of Oral and Maxillofacial Surgery
University of Miami- Miller School of Medicine
Deering Medical Plaza
9380 S.W. 152nd St.,
Miami, FL 33157
Office#: 305.256.5277
Email address: r.tursun@med.miami.edu
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Disclaimer
The views expressed in this article are those of the author(s) and do not necessarily reflect the official policy or position of the
Department of the Navy, Department of Defense, or the United States Government.
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I am a military service member. This work was prepared as part of my official duties. Title 17 U.S.C. 105 provides that “Copyright
protection under this title is not available for any work of the United States Government.” Title 17 U.S.C. 101 defines a United States
Government work as a work prepared by a military service member or employee of the United States Government as part of that
person’s official duties.
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Palatal Basal Cell Adenocarcinoma from misdiagnosis to cure: a unique case report and
review.
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INTRODUCTION:
Basal cell adenocarcinoma (BCAC) is a rare malignant lesion of the salivary gland,
accounting for less than 2% of all salivary neoplasms.1 In 2005, World Health
Organization defined basal cell adenocarcinoma (BCAC) as "an epithelial neoplasm that
has cytological characteristics of basal cell adenoma (BCA), but a morphologic growth
pattern indicative of malignancy."2 Here we describe a 66-year-old woman treated by
University of Miami Oral/Head and Neck Oncology Service that was misdiagnosed twice
as benign neoplasms prior appropriate diagnosis of the BCAC. At the time of her
recurrence, her care was assumed by the senior author and was appropriately
diagnosed and treated to cure including transoral microvascular reconstruction, as well
as adjuvant radiation. A review of the current literature of this disease entity is also
included.
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CASE REPORT:
A 66-year-old woman originally presented for evaluation of a swelling in her right
posterior palate to the Oral/Head and Neck Oncology service at the University of Miami.
In August 2011, she the patient had undergone a biopsy and definitive resection of the
area. Upon histopathologic evaluation, the lesion was definitively described as a
canalicular adenoma. The patient then failed to follow up as scheduled. Following this
period of time, the patient returned for evaluation after a biopsy by an oral and
maxillofacial surgeon at an outside facility with concerns of swelling in the area of the
right hard and soft palate. The senior author then took over her care and upon
evaluation noted a mass in the right posterior palate with mild facial swelling on the
patient's right side (Image 1). In the right posterior palate, a boggy lesion, approximately
2.5 X 2.5 cm was palpated. No ulceration noted over the lesion. Mucosa over lesion was
noted to be pink but thinned, with mild erythema and no pulsation (Image 2). The
biopsy that had been performed before arrival had the histopathologic diagnosis of
ameloblastoma and was then reviewed by two general pathologist as well as an oralmaxillofacial pathologist (OMP) on consultation who agreed with the diagnosis.
A computed tomography (CT) scan showed a radiolucent lesion in the right
posterior maxilla extending on to the palate with elevation of the right nasal floor and a
small perforation through bone. Osseous erosion of the alveolus is also noted seen in
this region (Images 3,4). A magnetic resonance image (MRI) was obtained to mitigate
the CT scan artifact and to better visualize the lesion boundaries. The axial view
revealed a hyperdense lesion of the right maxilla and palate and further elucidated the
location and extension (Image 5).
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Based upon clinical impression, her past surgical history, and a detailed radiographic
examination, the patient was offered the treatment of surgical resection and immediate
reconstruction with vascularized free tissue transfer. Subsequently, the patient was
taken to the operating room, the lesion outlined and the resection was performed. Due
to the history of recurrence and differing previous diagnoses, frozen sections were
utilized in surgery to ensure disease control. While nonspecific, intraoperative frozen
sections returned the finding of a malignant neoplasm of minor salivary gland origin.
Wide local excision was completed bearing the new malignant pathologic diagnosis in
mind and immediate reconstruction was performed with an anteromedial thigh
fasciocutaneous flap utilizing an intraoral anastomosis technique (Image 6-8).
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The patient's post-operative course was uneventful and she was discharged
home with the appropriate care. Final pathology was reviewed and found to be basal
cell adenocarcinoma. Furthermore, despite the frozen margins being clear upon
completion, final pathology was read as a close margin in one focal area. The case was
reviewed by our multidisciplinary tumor board where adjuvant radiation was
recommended. Due to the finding of carcinoma, a workup for metastasis was performed
and no distant disease was identified. The patient then successfully underwent a course
of postoperative radiation without complication and to the date of submission is 38
months out with no evidence of disease.
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DISCUSSION:
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Basal cell adenocarcinoma (BCAC) is a rare malignant lesion of the salivary glands,
accounting for less than 2% of all salivary neoplasms.1 In 2005, the World Health
Organization defined basal cell adenocarcinoma (BCAC) as "an epithelial neoplasm that
has cytological characteristics of basal cell adenoma (BCA), but a morphologic growth
pattern indicative of malignancy."3 It is considered to be the malignant counterpart of
the BCA. Histological differentiation between the two is difficult, and frequently they
can only be separated by identification of the invasion of local structures or by perineural or vascular invasion.2 This case is a perfect example of the difficulties in the
pathologic differentiation where the patient was twice diagnosed with a benign lesion
prior to the malignant BCAC diagnosis being rendered. Most BCACs arise de novo, but
approximately 20% arise from a preexisting basal cell adenoma. Approximately 90% of
BCACs are found in the parotid gland, and the remainder occur in the submandibular
gland. 4 Lo et al were the first to report a basal cell adenocarcinoma of the palate, but
the clinicopathologic behavior of this tumor, which originates from the minor salivary
gland, is still unclear. 5
It is necessary to differentiate BCAC from other basaloid cell tumors of the minor
salivary glands such as canalicular adenoma, BCA, adenoid cystic carcinoma,
polymorphous low-grade adenocarcinoma, myoepithelial tumors,
epithelial-myoepithelial carcinoma, and basaloid squamous cell carcinoma because of
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the prognosis and potential differences in treatment.6 BCAs with capsular invasion
shared several pathological features with BCACs, including large size and frequent
cribriform patterns but the malignant potential of these tumors seems highly limited
and should be reexamined (Image 9a,b). Additionally, β-catenin immunostaining may
aid when differentiating between a basal cell neoplasm and adenoid cystic carcinoma.7
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Overall, BCAC has a more favorable outcome than other common malignant salivary
gland tumors and is considered to be a low-grade malignancy.8 The most common
location for minor salivary gland tumor is the palate (42-54%) followed by the lips, but
any other oral site harboring minor salivary glands may be affected.9 The diagnosis of
BCAC depends on the presence of features similar to adenoma but with an infiltrative
growth pattern. Furthermore, adenoid cystic carcinoma, sialoblastoma, and basaloid
carcinoma must be excluded.10 The widely-accepted treatment for BCAC is complete
surgical excision.11 Ellis and Wiscovitch have reported that among 29 patients with basal
cell adenocarcinoma of the major salivary gland, 7 patients (28%) experienced at least 1
incidence of local tumor recurrence, and 3 patients (12%) had metastasis to the lung or
cervical lymph nodes.12 An extensive literature review performed by Cuthbertson et al.
included international and non-English literature in addition to their reported case
identified a total of 72 total cases of minor salivary gland BCAC.13 Regional lymph node
dissection is recommended only if there is evidence of metastatic disease to the cervical
chain, and adjuvant radiation therapy has been used for invasive tumors and local
recurrences.14 It is unclear whether radiotherapy plays a major role in the management
of this tumor, but it can be effective if the margin of the surgical specimen is close or the
lesion demonstrates local recurrence.15
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A discussion could also be considered with regards to the plan for the immediate
reconstruction of the maxillectomy defect. In review of the literature, this topic remains
controversial. It has been the experience of the authors as well as the findings of others,
as discussed by Moreno et al, that in maxillectomy defects consisting of >50% resection
of the hard palate, immediate free flap reconstruction results in superior functional
outcomes when compared to obturation. Furthermore, it is often posited, that
immediate reconstruction leads to a delay in the diagnosis of recurrence when
compared to obturation, but this assertion has not been the experience of the authors
nor the finding of Moreno et al. 16 In this case, an aggressive resection and clear frozen
margins led the surgical team toward an immediate reconstruction. In retrospect, the
permanent specimen finding of a close surgical margin could be used in making a case
for delayed reconstruction.
CONCLUSION:
Diagnosis of BCAC can be difficult to make with a simple biopsy and the lesion may be
easily mistaken for a benign basaloid tumor. Retrospectively, in review of this case, the
new finding of ameloblastoma by an outside pathologist with a previous diagnosis of
canalicular adenoma necessitated an oral-maxillofacial pathology consultation which
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Disclosures:
Dr Tursun serves as a consultant to AxoGen, Inc.
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confirmed the diagnosis. In this case, it would have also been beneficial for an original
slide request to be performed and a comparison completed of both sets of specimens
despite the recurrence diagnosis having been confirmed by two general pathologists as
well as an OMP. Due to the many similar features, despite this review, inadequate
sampling of the lesion may have left the pathologist without the ability to determine the
infiltrative growth pattern that differentiates BCAC from other similar lesions. In many
such cases, the authors prefer to rebiopsy or personally review all outside pathologic
diagnoses, and in this case that may have proven beneficial. Basal cell adenocarcinoma
should be considered rare but none the less included in the differential diagnosis of
lesions in this anatomical location when the lesions demonstrate similar clinical
behavior. Furthermore, in this case, the patient not only underwent surgical ablation
but also adjuvant radiation due to the history of recurrence as well as a close margin
and is currently disease-free 3 years following surgery. (Image 10a,b, 11)
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References
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2. Barnes L, Eveson JW, Reichart P, Sidransky D. Pathology and genetics of tumors
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January 2, 2015
6. Barnes, L: World health organization, international agency for research on
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14. Gross M, Maly B, Goldfarb A, Eliashar R. Basal cell adenocarcinoma in a buccal
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Images
Image 1. Patient at representation with noted mild fullness on the patient's right side.
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Image 2 Preoperative clinical photograph of the lesion and right posterior palate
Image 3 Axial view of soft tissue window of facial CT scan showing Right posterior
maxilla and palate soft tissue mass
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Image 4 A Coronal views of soft tissue and bone windows of facial CT
B. Multiple windows of the CT scan are limited secondary to dental prosthesis
artifact
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Image 5 A. T1 axial view shows hyperdense lesion of the right maxilla and palate;
B. T2 axial view shows hyperdense lesion of the right maxilla and palate;
C. Sagittal views elucidating lesion location and extension
D. Coronal views elucidating lesion location and extension
Image 6 Ablative defect of wide excision following intraoperative finding of BCAC on
frozen section.
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Image 7 Intra-oral anastomosis of the antero-medial thigh flap prior to inset. No
extraoral incision was used in preparing the facial vessels for microvascular
anastomosis
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Image 8 Inset of the antero-medial thigh flap into the ablative defect.
Image 9 A. Low power view of histological examination with signs of invasion, the
absence of a capsule, and evidence of mitotic figures
B. Higher power view showing evidence of mitotic figures
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Image 10 A. Frontal view of the patient at 3 years without significant deformity well
healed.
B. Lateral view with no scarring to neck or face due to intraoral approach for
vascular anastomosis to facial vessels.
Image 11 Intraoral view of the well-healed flap and no signs of clinical recurrence.
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