Advances in Oral and Maxillofacial Surgery 6 (2022) 100274

Contents lists available at ScienceDirect

Advances in Oral and Maxillofacial Surgery

journal homepage: www.sciencedirect.com/journal/advances-in-oral-and-maxillofacial-surgery

Case report

Extensive primary oral malignant melanoma: A case report with review

of literature
Walid Bijou a, Mohammed Laachoubi a, *, Youssef Oukessou a, b, Sami Rouadi a, b, Reda Abada a, b,
Mohamed Roubal a, b, Mohamed Mahtar a, b
a
ENT Department, Face and Neck Surgery, Hospital August, 20’1953, University Hospital Center IBN ROCHD, Casablanca, Morocco
b
Faculty of Medicine and Pharmacy, Hassan II University of Casablanca, B.P 5696, Casablanca, MoroccoENT Department, Face and Neck Surgery, Hospital August,
20’1953, University Hospital Center IBN ROCHD, Casablanca, Morocco

A R T I C L E I N F O A B S T R A C T

Keywords: Oral mucosal melanoma is an exceptionally rare tumor with the tendency to metastasize and locally invade
Melanocytes tissues more readily than other malignant tumor of the oral cavity. Here we present a case of 53-year-old women
Oral malignant melanoma patient with chief complaint of a discoloration over the hard palate since 6 months. On the basis of a through
Hard palate
clinical and paraclinical assessment, a diagnosis of oral malignant melanoma was evoked. On histopathological
and immunohistochemical analysis with S-100 and homatropine methylbromide 45 the diagnosis of malignant
melanoma of the had palate was confirmed.

1. Introduction
Malignant melanoma of the oral cavity is an exceptionally rare tumor
arising from the uncontrolled growth of melanocytes, initiate in the
basal layer of oral mucous membrane which is derived from cells of
neural crest origin and migrate to the skin, mucous membranes and
several other sites [1,2].
Primary oral malignant melanoma accounts for approximately
0.2–8% of all melanomas. Intra orally, hard palate and maxillary gingiva
are the most common involved sites [3].
Clinically, lesion may presents single or multiple as a pigmented
macular or proliferative lesion to a non pigmented lesion which may be,
primary or metastatic [4]. Non pigments forms often cannot be distin­
guished clinically from other benign or malignant oral tumors which can
be diagnosed through biopsy [5].
The tumor surface may be smooth or ulcerated. Other signs and
symptoms include bleeding, pain, increased mobility of teeth, and ill-
fitting dentures. Regional lymphadenopathy may be present and indi­
cate a poor prognosis [4].
In the present case study, we hereby report a rare case of primary oral
malignant melanoma of hard palate and maxillary gingiva, in a 53-year-
old women patient. with cervical lymph node metastasis, which has
been discussed with detailed investigations such as biochemical, histo­
pathology, and contrast enhanced computed tomography to emphasize
the necessity for early diagnosis and treatment of this deadly condition.
This study has been reported in accordance with the SCARE criteria
[13].
2. Case report
A 53-year-old woman in excellent health presented to our depart­
ment of head and neck Surgery, with chief complaint of a discoloration
over the hard palate since 6 months.
Interrogation revealed that the discoloration developed suddenly,
was painless, and had grown rapidly during the last month. No other
abnormal body pigmentation was reported by the patient nor were any
observed on the exposed body surfaces.
The intraoral examination found a large brown, poorly circum­
scribed patch, painless, involving the posterior palate. The discoloration
extended from the molar region to the anterior alveolar ridge and
covered most of the palatal vault (Fig. 1). No other abnormal intraoral
pigmentation was present.
Extraorally, There was no palpable cervical lymphadenopathy. Rest
of systemic examination was within normal limits.
Computed Tomography scan showed abnormal enhancement
measuring 22.6*13.3*25mm at the mucosal surface of hard palate
(Fig. 2). There was no destruction in surrounding area.
Positron emission tomography showed an intense hypermetabolic

* Corresponding author.
E-mail address: laachoubim@gmail.com (M. Laachoubi).

https://doi.org/10.1016/j.adoms.2022.100274
Received 22 February 2022; Accepted 15 March 2022
Available online 31 March 2022
2667-1476/© 2022 Published by Elsevier Ltd on behalf of British Association of Oral and Maxillofacial Surgeons. This is an open access article under the CC
BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
W. Bijou et al. Advances in Oral and Maxillofacial Surgery 6 (2022) 100274

Fig. 1. Brown color discoloration of hard palate. (For interpretation of the references to color in this figure legend, the reader is referred to the Web version of
this article.)

Fig. 2. Computed tomography scan abnormal enhancement of mucosa of hard palate.

focus of the right paramedian palatal mucosa adjacent to the maxillary
alveolar ridge below the palatal process with bilateral jugular adenop­
athy in territory II (Fig. 3).
On the right: maximum SUV at 1.8 measuring 9.6*8 mm.
On the left: intense signal with maximum SUV at 14.9 measuring
16.2*11 mm.
On the thoracic level: left hilar mediastinal adenopathy (maximum
SUV at 7.8) measuring 16*11 mm.
Under local anesthesia we performed a biopsy of the lesion. The gross
tissue examination revealed a mass 4 mm in size, black in colour,
smooth surface and firm in consistency. On histopathological and
immunohistochemical analysis with S-100 and homatropine methyl­
bromide 45 the diagnosis of malignant melanoma, of hard palate was
made.
The patient was referred to a cancer institute for further management
where she underwent 2 cycles of chemotherapy for which she failed to
respond and eventually passed away.
3. Discussion
Primary melanoma of the oral cavity is an uncommon malignant
neoplasm that tends to metastasize or invade local tissue more rapidly
than other malignancies. Melanomas of the oral cavity account for 0.5%
of all oral neoplasms and 0.2%–8% of all malignant melanomas [6]. The
exact etiology of melanoma of the oral cavity is unknown, unlike its
cutaneous counterpart, which is concomitant with chronic sun exposure.

2
W. Bijou et al.
Fig. 3. Positron emission tomography showed an intense hypermetabolic focus of the right paramedian palatal mucosa, with bilateral jugular adenopathy in ter­
ritory II.
Possible risks include smoking, alcohol, denture irritation, and envi­
ronmental carcinogens [3,7].
Nearly 80% of oral melanomas arise in the mucosa of the upper jaw,
with the majority occurring on keratinizing mucosa of the palate and
alveolar gingivae [8]. It occurs slightly more often in males 2.8:1 male to
female ratio) and the age range is from. 20–83 years worth an average
age of 56 years [9].
clinically, besides "de novo" appearance a possible transformation of
pre-existing oral benign melanotic lesions to OM has been reported [3].
In the present case, the patient did not report any pre-existing pigmented
lesion at the site where the OM arose.
The clinical presentation of this condition may vary widely which is
divided into following five types: Pigmented nodular type, pigmented
macular type, pigmented mixed type, non-pigmented nodular type and
non-pigmented mixed type [8–10]. Nevertheless, the diagnosis of non­
pigmented melanomas is more diffucult than that of pigmented lesions,
because they may not exhibit the classic criteria « ABCDE signs » [11]:
3
Advances in Oral and Maxillofacial Surgery 6 (2022) 100274
• A: asymmetry
• B: border irregularity
• C: color variation
• D: diameter
• E: evolving
The confirmation of diagnosis is done by histopathological exami­
nation of biopsy of clinically suspicious lesion. Oral MM usually shows
junctional activity and upward migration of the malignant cells. Special
stains like Fontana silver stain is helpful in approximately 75% the cases.
Immunohistochemical demonstration of the neuronal specific S-100
protein, HMB- 45 and Mart- 1 (Melan A) [3]. is a useful diagnostic in­
dicator, especially if the tumor is of amelanotic type. In our case, tumor
cells were positive for S-100 and HMB- 45.
The differential diagnosis includes melanotic macule, smoking
associated with melanosis, post-inflammatory pigmentation, melano­
plakia, melanoacanthoma, nevi, Addison’s disease, Peutz-Jeghur
W. Bijou et al.
syndrome, Kaposi’s sarcoma [12]. And many other conditions sharing
macroscopic characteristics and drug induced pigmentation more often
the culprit is azidothymidine [6].
Surgical excision with adequate margins was the treatment of choice,
followed by chemotherapy and radiotherapy as a complementary mo­
dality. The authors advocate aiming for margins of 1.5–2 cm, with 5 mm
of clearance considered a negative margin [14,15]. Some authors
recommend elective prophalactic neck dissection because of this low
risk of lymph node failure. Primary and adjuvant radiotherapy should be
considered if surgical management is not possible due to unresectable
disease or in patients unfit for surgery. A number of studies have shown
that postoperative radiation therapy improves locoregional control in
patients with oral mucosal melanoma [16]. The role of chemotherapy in
oral mucosal melanoma has been in the management of advanced or
disseminated disease. To date, no systemic regimen has been recognized
as effective for metastatic melanoma, as such regimens have been unable
to show any overall survival benefit [17].
The worse prognosis of oral mucosal melanomas is due to the late
presentation of patients. Rich vascularity and lymphatic drainage of
mouth favors earlier metastatic spread to regional lymph nodes and to
distant sites such as the lungs and vertebral column [18].
4. Conclusion
Malignant melanoma of hard palate is rare tumor, but aggressive
with very low survival rates which can metastasize rapidly. all pig­
mented lesions in the oral cavity should be examined with suspicion.
The treatment of choice for oral melanomas is wide surgical resection
with or without neck dissection depending upon chemotherapy as an
adjuvant or palliative therapy.
Provenance and peer review
Not commissioned, externally peer reviewed.
Ethical approval
Written informed consent was obtained from the patient for publi­
cation of this case report and accompanying images. A copy of the
written consent is available for review by the Editor-in-Chief of this
journal on request.
Sources of funding
This research did not receive any specific grant(s) from funding
agencies in the public, commercial, on non-for-profit sectors..
Authors contribution
Bijou walid: writing the paper.
Laachoubi Mohammed: Corresponding author, writing the paper.
Youssef Oukessou: study concept.
Advances in Oral and Maxillofacial Surgery 6 (2022) 100274
Sami Rouadi: revising the paper.
Reda Abada: revising the paper.
Mohamed Roubal: revising the paper.
Mohamed Mahtar: final approval.
Research registration unique identifying number (UIN)
Not needed.
Guarantor
Laachoubi Mohammed.
Declaration of competing interest
The authors declare no conflicts of interest for this article.
References
[1] Ullah H, Vahiker S, Singh M, Baig M. Primary malignant mucosal melanoma of the
oral cavity: a case report. Egypt J Ear Nose Throat Allied Sci 2010;11:48–50.
[2] Nambiar S, Vishwanath MN, Bhat S, et al. Oral malignant melanoma: a brief
review. J Clin Exp Pathol 2016;6:2161–681.
[3] Meleti M, Leemans CR, Mooi WJ, et al. Oral malignant melanoma: a review of the
literature. Oral Oncol 2007;43:116–21.
[4] Aguas SC, et al. Primary melanoma of the oral cavity: ten cases and review of 177
cases from literature. Med Oral, Patol Oral Cirugía Bucal 2009;14(6):E265–71.
[5] Magliocca KR, Rand MK, Su LD, Helman JI. Melanoma-in-situ of the oral cavity.
Oral Oncol Extra 2006;42:46–8.
[6] Kumar SV, Swain N. Oral malignant melanoma-A case report. Int J Oral Maxillofac
Surg 2011;2:50–4.
[7] Chang AE, Karnell LH, Menck HR. The National Cancer Data Base report on
cutaneous and noncutaneous melanoma: a summary of 84,836 cases from the past
decade. Cancer 1998;83:1664–78.
[8] Dimitrakopoulos I, Lazaridis N, Skordalaki A. Primary malignant melanoma of the
oral cavity. Report of an unusual case. Aust Dent J 1998;43:379–81.
[9] Tanaka N, Mimura M, Ichinose S, Odajima T. Malignant melanoma in the oral
region: ultrastructural and immunohistochemical studies. Med Electron Microsc
2001;34:198–205.
[10] Cockerell CJ. The pathology of melanoma. Dermatol Clin 2012;30:445–68.
[11] Auluck A, Lewei Z, Rajeev D, Miriam P. Primary malignant melanoma of maxillary
gingiva–a case report and review of the literature. J Can Dent Assoc 2008 May;74
(4). 367-71.
[12] Magliocca KR, Rand MK, Su LD, Helman JI. Melanoma-in-situ of the oral cavity.
Oral Oncol Extra 2006;42:46–8.
[13] Agha RA, Franchi T, Sohrabi C, Mathew G. For the SCARE Group, the SCARE 2020
guideline: updating consensus surgical CAse REport (SCARE) guidelines. Int J Surg
2020;84:226–30.
[14] Breik O, Sim F, Wong T, et al. Survival outcomes of mucosal melanoma in the head
and neck: case series and review of current treatment guidelines. J Oral Maxillofac
Surg 2016;74:1859–71. https://doi.org/10.1016/j.joms.2016.03.008.
[15] Sun CZ, Chen YF, Jiang YE, et al. Treatment and prognosis of oral mucosal
melanoma. Oral Oncol 2012;48:647–52.
[16] Benlyazid A, Thariat J, Temam S, et al. Postoperative radiotherapy in head and
neck mucosal melanoma: a GETTEC study. Arch Otolaryngol Head Neck Surg 2010;
136:1219–25.
[17] American Joint Committee on Cancer. NCCN guidelines version 1. Staging: head
and neck cancers. Fort Washington, PA: National Comprehensive Cancer Network;
2015.
[18] Calabrese V, Cifola M, Pareschi R, et al. Primary malignant melanoma of the oral
cavity. J Laryngol Otol 1989;103:887–9.