Pigmented macules were also seen on the palms, soles, tongue and cheek mucosa. Blood and urinary electrolyte levels were within normal ranges. Plasma cortisol was low, plasma ACTH high, and the synacthen test negative. These findings led us to diagnose Addison's disease. Antithyroid antibodies were noted without antinuclear factors or antiadrenal antibodies. Outcome was favorable. Because of the presence of isolated melanoderma of the sun-exposed areas and macular pigmentation of oral mucosa in this patient, we tested for Addison's disease: hormonal testing confirmed the diagnosis.
Because our patient had no electrolyte disorders, Addison's disease might have remained unknown until the onset of acute adrenal insufficiency. Etiology in this case was probably autoimmune. In the case of persistent pigmentation of sun-exposed areas, even isolated, Addison's disease must be considered and hormonal testing performed to avoid acute adrenal insufficiency.
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In addition, in patients with dark skin, this lesion can be found on the tongue or the palate [ 11 — 13 ]. When these macules are multiple, we talk about lentiginosis. The histological examination finds epithelial hyperplasia, acanthosis, melanic pigmentation of the basal layers of the epithelium hypermelanocytosisand many melanophages in the underlying layers.
There is no regrouping in thecas. Nevocellular nevus is infrequent in the oral mucosa and more often affect subjects aged 30—40 years. It is frequently located on exercices pour perdre 1 kilo or palate, to the inside of the cheeks and sometimes gum and the labial mucosa in the form of a macule with a size 0. It can also manifest in the form of a nodule. Clinical distinction is difficult between a nevocellular nevus and melanoma, thus the need for histological analysis [ 15 ].
Histological analysis found a proliferation of nevus cells, regular, rounded, grouped in thecas or layers within the basal membrane and the lamina propria. Blue nevus is rare in the oral mucosa, and is most often located on the palate. It is most often found in children and young adults, and in women. It has bluish gray color, and can be macular or nodular.
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Fusiform cells located exclusively in the lamina propria can be found on the histological sections. The deep location of these cells explains the bluish coloration of the nevus. Blue nevus generally does not degenerate, but its resemblance to melanoma is an indication for surgical excision. Melanoacanthoma is exceptionally rare in the oral mucosa and is more often found on the palate and inner cheeks.
It affects young adults and most frequently women with black skin. Local trauma or a history of chronic irritation are triggering factors.
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It can measure up to several centimeters in diameter. The pigmentation of exogenous origin can be either accidental or voluntary. Pigmentation of accidental causes may be due to tar, pigmentation due to the metals present in the fillings or crowns. Sometimes a mucosal break with intramucosal deposit of dental material may be observed.
Pigmentation of voluntary origin may be because of ethnic pigmentations in African tribes or because of tattoos with the presence of pigmented scar on cheeks or the buccal gingiva [ 16 ]. Pigmentation of vascular origin is dark red or purplish, close to black. These lesions are represented by bruises, angiomas plans, and Kaposi's disease. The vascular nature of the lesion manifests as diurnal changes in angioma volumes, the position of the head, the heat and by their blanching to the pressure.
In Kaposi's disease, the lesion is macular or nodular, with dark purplish coloring, and diffuse location, with a possible extension to the whole body. Essential melanosis or Laugier—Hunziker syndrome belongs to the group of single pigmented macules occurring in adults.
Hyperpigmented Patches on the Tongue of a Young Girl—Quiz Case
Although rare, it is necessary to know how to clinically diagnose when encountering a single, flat pigmented lesion of the oral cavity, particularly in women of Caucasian origin. Even if clinical suspicion is strong, the only way for a final diagnosis of a pigmented oral lesion remains the histopathological examination. Therefore, it is necessary to reinforce the importance of a comprehensive intraoral examination during any consultation with the dental surgeon, complemented by full dermatological examination if there is a mucosal lesion as required.
Early detection of melanoma and prompt treatment allows for a decrease the morbidity and mortality of this pathology. Data correspond to usage on the plateform after The current usage metrics is available hours after online publication and is updated daily on week days.
Open Access. Issue Med Buccale Chir Buccale. Lésions pigmentées de la cavité buccale. Ann Pathol ;— The normal and pathological pigmentation of oral mucous membrane: a review. J Contemp Dent Pract ;— Oral and perioral endogenous pigmented lesions. Laugier-Hunziker syndrome: a case report and review of the literature.
Dermatol Online J ; Mélanose essentielle ou syndrome de Laugier-Hunziker. Med Buccale Chir Buccale ;— Primary mucosal malignant melanoma of the head and neck.
Head Neck ;— Oral melanoma and other pigmented lesions of the oral cavity. It represents 0. Melanoma is a tumor of the adult. It is often said that there is a slight male predilection of [ 1246 — 8 ]. Whereas other studies showed no sex predilection, even more there was a female superiority [ 910 ].
High risk sites are the palate and the maxillary gingiva. However, all oral mucosa sites can be affected by OMM [ 1246 — 9 ]. Oral malignant melanoma is usually painless. It is often described as a uniformly pigmented black or brown lesion. But sometimes several shades co-exist: black, brown, gray, pink and red. The lesions are asymmetric and irregular in outline. Sometimes, they are multiple. The elemental lesion may be a flat macula, a low elevated plaque or a soft nodule. These clinical aspects can be present at the same time [ 124 ].
Unfortunately, this classification has no prognostic value. Ulceration and bleeding are late signs. Unlike squamous cell carcinoma, there is no induration [ 124 ]. So far, the etiology of oral malignant melanoma is poorly understood, unlike cutaneous melanoma which has well-defined risk factors.
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Therefore, it is still difficult to recognize subjects at highest-risk for developing OMM [ 5 ]. Some authors suggest tobacco smoking, inhaled or aspirated environmental carcinogens and chronic irritation as possible risk factors [ 2 ]. It is true that most of these tumors appear to be de novo.
Haïtami et al. However, it needs to be mentioned that about one-third of melanomas have had a history of benign pigmented lesions for months and even years before malignant transformation [ 1 — 3 ].
Unfortunately, the exact mechanism of malignant transformation is unknown. We will present here a non-exhaustive summary of literature cases about melanoma emerging from flat precursor lesions Tab. Several descriptive terms have been used to describe premalignant lesions: oral benign melanosis, pre-malignant melanosis, oral melanotic macule, melanocytic dysplasia, melanocytic hyperplasia, neoplastic melanocytic proliferation Tab.
No sex predilection was noted. Precursor lesions were irregularly shaped flat, preferentially localized in keratinized tissues, at the same sites of melanomas.
Pigmentation over tongue
Lesions' size ranged from 0. Lesions have evolved for months and years and they were characterized by their tendency to recur after excision, persistence and enlargement [ 3 ]. It is accepted that clinical evaluation of cutaneous melanocytic lesions is guided by ABCDE criteria.
Many authors claimed that these criteria do not sit well with oral pigmented lesions. However, most of premalignant lesions including our patient transgressed these criteria when progressing to oral malignant melanoma. According to the western society of teachers of oral pathology WESTOPboth clinical and histological criteria should be used for the monitoring of pigmented lesions and recognition of potentially malignant ones [ 18 ].
Biopsy of oral pigmented lesion is still controversial. Umeda involved the biopsy as well as any procedures before the definitive intervention such as teeth extraction, incision Without biopsy, the 5-year survival rate was of However, it dropped to On the other hand, without histological analysis, we can never recognize the exact nature of an oral pigmented lesion.
So no early treatment could be started. Until tangible evidence, it is not asserted that biopsy increases the risk of local or distant spread of melanoma [ 2 ].
Moreover, when we find melanocytic hyperplasia or dysplasia in histological reports, the entire lesion should be completely excised with clear margins [ 318 ]. For our patient, the lesion covered the entire palate, and histo-pathological report reported no sign of dysplasia. In principle, incisional biopsy was sufficient. But close clinical follow up had to be maintained. Histological features of potentially malignant pigmented lesions were multifarious.
We can find hyperpigmentation in the basal layers, proliferation of dendritic melanocytes with or without atypia, proliferation of clear cell [ 31315 — 1719 ].
These data are found in Umeda's gradual enlarging pattern of oral malignant melanoma. Meticulous examination revealed that OMM went through three phases: a nodular phase usually affecting the centre, a slightly elevated, deep brownish-black pigmented plaque phase, and a flat light-brown macular phase [ 11 ].
In the macular phase, a simple hyperpigmentation in the basal layer is found. Lentiginous proliferation of dendritic melanocytes without apparent cellular atypia may be also noted. The pigmented plaque lesion contains atypical melanocytes nests or individually proliferating tumor cells in the lower epithelial layers. It's considered pre-invasive phase. Macular and plaque phases form radial growth pattern of OMM [ 311 ].
Overall, most of premalignant lesions histological findings appear to be consistent with features found in the macular and plaque phases of OMM [ 3 ]. Only the nodular phase corresponds to true invasive melanoma when vertical growth pattern begins with spindle- shaped or epithelioid tumor cells in the submucosa [ 311 ]. So, in OMM cellular morphology shows a wide range of features [ 1248920 ].
Immunohistochemistry is strongly indicated. Melanocytes markers commonly used are protein S, melan-A, tyrosinase, HMB45 [ 247820 ]. OMM treatment is based on surgery with wide clear margins.
To meet this requirement, it is necessary to extend the excision to the soft palate, the tonsillar pillar, and into the pterygomaxillary space. But, the proximity of vital structures makes this objective difficult. No consensus exists so far.