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Oral melanoacanthoma of a rare intraoral site: case report and review of literature.
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PMID:  25206187     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Oral melanoacanthoma is rare pigmented mucosal lesion that presents most commonly on the buccal mucosa, characterized by sudden appearance and rapid radial growth, thus clinically mimicking malignant melanoma. It was originally described as a mixed tumor of melanocytes and keratinocytes, but appears to be a reactive process; formed in areas prone to trauma, and regressing after the removal of trauma or incomplete excision. The clinical appearance of oral melanoacanthoma is nondiagnostic, and biopsy is mandatory to rule out malignancy. We report a case of melanoacanthoma of a rarer oral mucosal site in a 12-year-old Asian male. A brief review of the current literature is also presented. How to cite this article: Rohilla K, Ramesh V, Balamurali PD, Singh N. Oral Melanoacanthoma of a Rare Intraoral Site: Case Report and Review of Literature. Int J Clin Pediatr Dent 2013;6(1):40-43.
Authors:
Kshitiz Rohilla; V Ramesh; Pd Balamurali; Namrata Singh
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Publication Detail:
Type:  Journal Article     Date:  2013-04-26
Journal Detail:
Title:  International journal of clinical pediatric dentistry     Volume:  6     ISSN:  0974-7052     ISO Abbreviation:  Int J Clin Pediatr Dent     Publication Date:  2013 Jan 
Date Detail:
Created Date:  2014-09-10     Completed Date:  2014-09-10     Revised Date:  2014-09-15    
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Nlm Unique ID:  101585405     Medline TA:  Int J Clin Pediatr Dent     Country:  India    
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Languages:  eng     Pagination:  40-3     Citation Subset:  -    
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Journal Information
Journal ID (nlm-ta): Int J Clin Pediatr Dent
Journal ID (iso-abbrev): Int J Clin Pediatr Dent
Journal ID (publisher-id): IJCPD
ISSN: 0974-7052
ISSN: 0975-1904
Publisher: Jaypee Brothers Medical Publishers
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Copyright © 2013; Jaypee Brothers Medical Publishers (P) Ltd.
open-access:
Received Day: 11 Month: 1 Year: 2013
Accepted Day: 14 Month: 3 Year: 2013
Print publication date: Season: Jan-Apr Year: 2013
Electronic publication date: Day: 26 Month: 4 Year: 2013
Volume: 6 Issue: 1
First Page: 40 Last Page: 43
PubMed Id: 25206187
ID: 4034627
DOI: 10.5005/jp-journals-10005-1185

Oral Melanoacanthoma of a Rare Intraoral Site: Case Report and Review of Literature
Kshitiz Rohilla Affiliation: Demonstrator, Department of Oral Pathology, Postgraduate Institute of Dental Sciences, Rohtak, Haryana, India
V Ramesh Affiliation: Dean, Professor and Head, Department of Oral Pathology and Microbiology, Mahatma Gandhi Postgraduate Institute of Dental Sciences, Puducherry, India
PD Balamurali Affiliation: Professor, Department of Oral Pathology and Microbiology, Mahatma Gandhi Postgraduate Institute of Dental Sciences, Puducherry, India
Namrata Singh Affiliation: Ex-Senior Lecturer, Department of Orthodontics and Dentofacial Orthopedics, Indira Gandhi Institute of Dental Sciences, Puducherry India
Correspondence: Demonstrator, Department of Oral Pathology, Postgraduate Institute of Dental Sciences, Rohtak, Haryana, India, dr.k.rohilla@gmail.com

INTRODUCTION

Melanoacanthoma is an uncommon, benign, mucocutaneous pigmented lesion characterized by dendritic melanocytes dispersed throughout the acanthotic epithelium.1, 2 Though it was originally described as a benign skin tumor of keratinocytes and dendritic melanocytes, there is now evidence that the intraoral lesions are unlike those occurring on skin.3

Cutaneous melanoacanthoma was first described in 1927 by Bloch, but the term melanoacanthoma was introduced by Mishima and Pinkus in 1960.1 The first case of oral melanoacanthoma was reported in 1978 by Tomich.4

Melanoacanthoma of the skin is a benign mixed proliferation of keratinocytes and melanocytes and is considered to be a variant of seborrheic keratosis. Most patients are adults, beyond 40 years of age. Sex predominance is not known.5 Most melanoacanthomas are located on the trunk, though lesions have been reported on the scalp, neck and extremities too.5, 6 These lesions are almost exclusive to whites in middle to late age, developing slowly over a long period, and usually having a roughened or papillary surface.7

On the contrary, intraoral melanoacanthomas tend to affect a much younger population, occurring almost exclusively in blacks, with a female predilection. These lesions show rapid increase in size and may attain dimensions of several centimeters in a few weeks. Buccal mucosa is the most frequently reported intraoral site, although masticatory mucosa subject to chronic trauma (palate, gingiva) may also be affected.8-11 Involvement of labial mucosa12-14 and alveolar ridge15 has also been reported. Mostly unilateral and solitary,16 these deeply pigmented lesions may have a flat or slightly raised surface. The other end of the spectrum of clinical presentation includes lesions that may be bilateral,17 and even multifocal,1, 8, 18, 19 as well as those which even have a proliferative or warty surface. These intraoral hypermelanotic macules or papules are typically brown, black or blue-black in color, with possible variation in the intensity of pigmentation.1, 3, 18, 20

Intraoral melanoacanthoma still continues to be a rare entity.21-24 Some of the previously reported cases have been summarized in Table 1.


CASE REPORT

A 12-year-old male patient presented for evaluation of a lesion in the left maxillary gingiva, which was present for the past 6 months. The patient was under medication with valproic acid for the treatment of petit mal seizures, till the age of 8 years, after which it was discontinued. Otherwise, the medical history was noncontributory.

Extraoral examination revealed no clinically significant findings. Intraorally, there was a soft tissue growth in maxillary left quadrant (Fig. 1), involving the attached and the marginal gingiva on the buccal aspect. The lesion was brownish black in color and had a smooth, slightly raised surface (Fig. 2). The patient denied any association of pain with the lesion. The other three quadrants showed macular pigmentation of the attached and marginal gingivae, which was clinically labeled as racial pigmentation.

The lesion was excised and sent for histopathological examination. Hematoxylin and eosin (H&E) stained sections revealed surface stratified squamous epithelium and underlying fibrous connective tissue. The epithelium exhibited parakeratosis and acanthosis and the rete ridges were irregular in shape. There was a prominence of melanocytes in the basal layer, in a linear fashion (Fig. 3). There was a suspicion of pigmented melanocytes even in the suprabasal layers. The underlying connective tissue appeared normal, showing some evidence of melanophagic activity in the subepithelial zone. Masson-Fontana silver stain supplemented the presence of dendritic melanocytes filling up almost the entire epithelium (Fig. 4). The presence of benign appearing melanocytes was salient, and there was no evidence of any cytological atypia, pleomorphism or nuclear hyperchromasia. In light of the history, clinical features and the histopathological picture with H&E and Masson-Fontana stain, the final diagnosis of oral melanoacanthoma was rendered.

The patient has been on a regular follow-up (Fig. 5), and the lesion was observed to be healing well 10 months postoperatively.


DISCUSSION

The credit for the first fully documented case of oral melanoacanthoma goes to Matsouka (1979).14 Since then, there has been an addition of more than 65 cases to the available literature. The pathogenesis still remains obscure, though some authors have ascribed the potential role of chronic trauma in these cases.

The intraoral melanoacanthoma is essentially reactive in nature'a fact supported by the clinical course of the lesion. It is characterized by a tendency to affect the mucosal sites that are exposed to trauma, and typically shows rapid growth and observable regression of the lesion'spontaneously or following incomplete removal15 or elimination of local irritants.18 The histologic picture of subepithelial inflammatory cell infiltrate and slightly increased vascularity further add evidence to this concept. To differentiate these lesions from cutaneous melanoacanthoma and to emphasize their reactive nature, several terms have been suggested, including melanoacanthosis, reactive melanocytic hyperplasia and mucosal melanotic macule.6, 8, 21

The clinical picture of this lesion is indistinguishable from many other oral pigmented lesions. All pigmented lesions should be observed for evolution with respect to size, shape, color, surface or symptom overtime.2 The observation of any of these features mandates a biopsy, because of potential resemblance to a malignant melanoma. The alarming growth rate of oral melanoacanthoma makes it clinically indistinguishable from oral malignant melanoma, especially the radial growth phase of an in situ melanoma. The biopsy, hence, should be performed invariably to rule out the possibility of a melanoma. Once the diagnosis has been established, no further treatment is indicated, and many cases document spontaneous regression.

The diagnosis of oral melanoacanthoma can be made solely on the basis of histological features and special staining. In order to emphasize the presence of melanin and to demonstrate the dendritic melanocytes, Masson-Fontana silver impregnation technique can be used.10 The immunohistochemical profile of these lesions is limited to the melanocytic markers, but is not necessary for diagnosis, as strong reactivity to HMB-45 and S100 is seen in both oral melanoacanthoma and malignant melanoma.8-10

Some authors6, 8 opine that in contrast to the other pigmented lesions, the melanin in oral melanoacanthoma is restricted mainly to melanocytes, the adjacent keratinocytes being devoid of melanin. Interestingly, in our case, the histopathological picture showed the presence of ‘dusty’ melanin in the basal as well as parabasal keratinocytes.

Melanoacanthoma is a reparative lesion with no malignant potential. The treatment should be directed toward removing all local causes of trauma and excluding any other causes of oral pigmentation, particularly malignant melanoma.

The authors advocate the replacement of the misnomer ‘melanoacanthoma’ with a more appropriate description ‘melanoacanthosis', a term which gives due credit to the clinical behavior and histopathological picture of this rare and interesting lesion.


SUMMARY

We present a case of a rare entity, oral melanoacanthoma, occurring at a rarer oral mucosal site, that is, gingiva, in a 12-year-old Asian male. The diagnosis was based mainly on the histologic findings with H&E and Masson-Fontana stains. The patient is on a regular follow-up and the lesion has regressed completely after the initial surgery.


Notes

n1Source of support: Nil

Conflict of interest: None

REFERENCES
1. Chandler K,Chaudhry Z,Kumar N,Barrett A,Porter SR. Melanoacanthoma: A rare cause of oral pigmentation.Oral Surg Oral Med Oral Pathol Oral Radiol EndodYear: 19978454924949394380
2. Neville BW,Damm DD,Allen CM,Bouquot J. Oral and maxillofacial pathology.3rd ed.PhiladelphiaWB Saunders CompanyYear: 2009984 p
3. Wright JM. Intraoral melanoacanthoma: A reactive melanocytic hyperplasia: Case report.J PeriodontolYear: 198859153553422293
4. Tomich CE. Oral presentation. Paper presented at: 32nd Annual Meeting of the American Academy of Oral PathologyYear: 19782328
5. LeBoit PE,Burg G,Weedon D,Sarasin APathology and genetics of skin tumours (IARC WHO Classification of tumours).OxfordOxford University PressYear: 2009355 p
6. Tomich C,Zunt S. Melanoacanthosis (melanoacanthoma) of the oral mucosa.J Dermatol Surg OncolYear: 19901632312362179347
7. Buchner A,Merrell PW,Hansen LS,Leider AS. Melanocytic hyperplasia of the oral mucosa.Oral Surg Med Oral PatholYear: 19917115862
8. Marocchio LS,Junior DS,de Sousa SC,Fabre RF,Raitz R. Multifocal diffuse oral melanoacanthoma: A case report.J Oral SciYear: 200951346346619776516
9. Fornatora ML,Reich RF,Haber S,Solomon F,Freedman PD. Oral melanoacanthoma: A report of 10 cases, review of the literature, and immunohistochemical analysis for HMB-45 reactivity.Am J DermatopatholYear: 2003251121512544093
10. Carlos-Bregni R,Contreras E,Netto AC,Mosqueda-Taylor A,Vargas PA,Jorge J,Leon JE,de Almeida OP. Oral melanoacanthoma and oral melanotic macule: A report of 8 cases, review of the literature, and immunohistochemical analysis.Med Oral Patol Oral Cir BucalYear: 2007125374379
11. Flaitz CM. Oral melanoacanthoma of the attached gingiva.Am J DentYear: 200013316211763954
12. Maize JC. Mucosal melanosis.Dermatol ClinYear: 1988622832933378373
13. Sexton FM,Maize JC. Melanotic macules and melano- acanthomas of the lip: A comparative study with census of the basal melanocyte population.Am J DermatopatholYear: 1987954384443688369
14. Matsouka LY,Glasser S,Barsky S. Melanoacanthoma of the lip.Arch DermatolYear: 1979115911161117485190
15. Goode R,Crawford B,Callihan M,Neville B. Oral melano- acanthoma: Review of the literature and report of ten cases.Oral Surg Oral Med Oral PatholYear: 19835666226286581460
16. Landwehr DJ,Halkias LE,Allen CM. A rapidly growing pigmented plaque.Oral Surg Oral Med Oral Pathol Oral Radiol EndodYear: 19978443323349347492
17. Heine B,Drummond JF,Damm DD,Heine RD 2nd. Bilateral oral melanoacanthoma.Gen DentYear: 19964454514529171047
18. Fatahzadeh , Sirois DA. Multiple intraoral melanoacanthomas: A case report with unusual findings.Oral Surg Oral Med Oral Pathol Oral Radiol EndodYear: 2002941545612193894
19. Kauzman A,Pavone M,Blanas N,Bradley G. Pigmented lesions of the oral cavity: Review, Differential diagnosis, and case presentations.J Can Dent AssocYear: 2004701068268315530266
20. Wright JM,Binnie WH,Byrd DL,Dunsworth AR. Intraoral melanoacanthoma.J PeriodontolYear: 19835421071116572716
21. Horlick HP,Walther RR,Zegarelli DJ,Silvers DN,Eliezri YD. Mucosal melanotic macule, reactive type: A simulation of melanoma.J Am Acad DermatolYear: 19881957867913056992
22. Schneider LC,Mesa ML,Haber SM. Melanoacanthoma of the oral mucosa.Oral Surg Oral Med Oral PatholYear: 19815232842876945539
23. Frey VM,Lambert WC,Seldin RD,Schneider LC,Mesa ML. Intraoral melanoacanthoma.J Surg OncolYear: 198427293966482463
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Figures

[Figure ID: F1]
Fig. 1 

Clinical photograph showing the melanotic gingival enlargement in the maxillary left quadrant



[Figure ID: F2]
Fig. 2 

The lesion covering the entire buccal surfaces of the teeth and also partially covering the occlusal surface of the first molar



[Figure ID: F3]
Fig. 3 

Photomicrograph showing acanthosis and parakeratosis of the surface epithelium as well as linear melanocytic hyperplasia in the basal layer. Benign melanocytes are seen in parabasal layers, and there is evidence of melanophagic activity



[Figure ID: F4]
Fig. 4 

Photomicrograph highlighting the dendritic morphology of the melanocytes, which are seen extending above the basal layers (Masson-Fontana, 400×)



[Figure ID: F5]
Fig. 5 

Postoperative clinical appearance of the site after 4 weeks



Tables
[TableWrap ID: Table1]

Table 1: Summary of previously reported cases


Authors Years Number of cases Affected oral site
Tomich4     1978     1     Buccal mucosa    
Matsouka et al14     1979     1     Labial mucosa    
Schneider et al22     1981     1     Buccal mucosa    
Wright et al20     1983     2     Buccal mucosa    
Goode et al15     1983     10     Buccal mucosa, palate, labial mucosa, alveolar ridge, attached gingiva    
Frey et al23     1984     1     Buccal mucosa    
Sexton and Maize13     1987     3     Labial mucosa    
Wright3     1988     1     Buccal mucosa    
Whitt et al24     1988     1     Buccal mucosa    
Horlick et al21     1988     2     Buccal mucosa    
Heine et al17     1996     1     Buccal mucosa    
Chandler et al1     1997     1     Palate, tonsillar fossae, upper nasopharynx    
Landwehr et al16     1997     1     Buccal mucosa    
Flaitz11     2000     1     Attached gingiva    
Fatahzadeh et al18     2002     1     Buccal mucosa, palate    
Fornatora et al9     2003     10     Buccal mucosa, gingiva, hard palate, lower lip, floor of mouth, retromolar pad    
Kauzman et al19     2004     1     Buccal mucosa, labial mucosa, tonsillar pillars    
Carlos-Bregni et al10     2007     4     Gingiva, buccal mucosa, hard palate    
Marocchio et al8     2009     1     Buccal mucosa, lips, gingiva, tongue    


Article Categories:
  • Case Report

Keywords: Keywords Melanoacanthoma, Oral pigmented lesion, Melanocytes.

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