Rhomboid glossitis in atypical location: case report and differential diagnosis
Glositis romboidal: localización atípica. Caso clínico y diagnóstico diferencial

Lucía Lago Méndez ⁽¹⁾, Andrés Blanco Carrión ⁽²⁾, Márcio Diniz Freitas ⁽³⁾, Pilar Gándara Vila ⁽³⁾,
Abel García García ⁽⁴⁾, José Manuel Gándara Rey ⁽⁵⁾

(1) Alumna del Máster de Medicina Oral, Cirugía Oral e Implantología
(2) Profesor Titular de Medicina Oral de la Universidad de Santiago de Compostela
(3) Profesor Colaborador del Máster de Medicina Oral, Cirugía Oral e Implantología
(4) Profesor Titular de Cirugía Oral y Maxilofacial de la Universidad de Santiago de Compostela
(5) Catedrático de Medicina Oral de la Universidad de Santiago de Compostela

Address:
José Manuel Gándara Rey
Facultad de Odontología
C/ Entrerríos s/n 15705
Santiago de Compostela (Coruña)
Tlfno: 981563100 (12357)
Fax: 981562226
E-mail: luci_lago@hotmail.com

Received: 5-09-2003. Accepted: 22-02-2004

INTRODUCTION

Median rhomboid glossitis (MRG), first described by Brocq in 1914, occurs in less than 1% of the general population (1). About 70 - 80% of cases are in men (2). Its aetiology is unknown, although it has been proposed that it may be derived from chronic candidiasis, or that it may be of embryological, inflammatory, or even immunological origin. Reported associated factors include smoking, dental prostheses and small traumas (1). It is considered to be a dysembryoplastic process, with the lesion provoked by the mechanical actions of mastication and swallowing, and in which other complications and secondary infections may occur (notably chronic candidiasis or pigmented nevus) (3). It typically presents in the posterior region of the dorsum of the tongue, at the midline, anterior to the lingual "V"; however, it sometimes appears in paramedial location (2). It appears as a rounded or rhomboid painless plaque with well-defined margin, intense reddish or pinkish in colour due to atrophy or depapillation, and firm to palpation. It may present in two distinct clinical forms: atrophic non-raised macular, or mamillated exophytic, raised by about 2 - 5 mm (1). Occasionally, the lesion may be fissured or lobulate.

Clinical diagnosis is based on culture for Candida. Treatment is based on the elimination of known aetiological factors, or antifungal treatment in cases of confirmed candidiasis.

CASE REPORT

A 61-year-old male presented with no relevant personal or family antecedents, no known allergies, and no medication. He showed a haemangioma on the left cheek. He reported a daily intake of 0.5 -1 l of wine, but did not smoke. He consulted for an exophytic formation on the dorsum of the tongue. At anamnesis the patient reported that the lesion had arisen 46 years ago, with little increase in size over the intervening period. Clinical examination indicated a lump in the lingual mucosa, located about 3 cm anterior to the lingual "V" in left paramedial position, elongated and slightly fissured, about 2 x 0.5 x 0.75 cm in size, deep reddish in colour, alternating with paler or pinkish areas (Fig. 1). The lump was firm to palpation, non-fluctuant, not attached to deep tissue layers, painless, and non-bleeding.

Candida culture tested positive and indicated the presence of Candida albicans. Colour changes were not observed under vitropressure. In view of clinical characteristics we obtained an incisional biopsy, which was stained with haematoxylin-eosin and PAS. Histopathological examination revealed a non-keratinized stratified flat epithelium, with irregular acanthosis and papillomatosis without evident cellular atypia. Moderate lymphocyte/plasma cell infiltration was observed in the adjacent lamina propria (Fig. 2a). PAS staining was positive and revealed the presence of hyphae in the upper layers of the epithelium (Fig. 2b).

The lesion was thus diagnosed as rhomboid glossitis with presence of C. albicans.

Treatment was with fluconazole (Diflucan 50, capsules) for 2 weeks at 50 mg per day. The patient consulted again a week after termination of the treatment; samples obtained at this visit tested negative for Candida, and the lesion showed reduced redness and was less raised than previously (Fig. 3).

DISCUSSION

Diagnosis of MRG is basically clinical, although sometimes histopathology is required for differential diagnosis. In the present case, all lesion characteristics except location were consistent with MRG. In view of the unusual location, the present case can be considered as atypical rhomboid glossitis (ARG), the term "atypical" covering both posterior and medial positions; some author have used the alternative term "paramedial" (2). In view of the unusual location and shape of lesions of this type, various differential diagnoses need to be taken into account, as detailed in what follows.

Congenital haemangiomas of the oral cavity tend to locate on the floor of the mouth or on the tongue, affecting all or part of the tongue and leading to macroglossia. Clinically, haemangiomas present as bluish or intense reddish purple masses, soft and fluctuant to palpation, with detectable pulse (3). The main characteristic for diagnosis is paling or disappearance of colour under vitropressure. In the present case there was no change in colour, and the lesion is firm to palpation.

Pyogenic granuloma presents as a smooth or lobulate lesion, generally pediculated, though sometimes sessile. The surface is generally ulcerated, and colour may be pink, red or purple (depending on the age of the lesion and the degree of fibrosis), sometimes whitish due to the presence of an overlying pseudomembrane. The size of the lesion ranges from a few millimetres to several centimetres. The lesion is not painful, but bleeds readily due to its abundant vascularization. It is solid to palpation. This disorder is most frequent in children and young adults, and in females (4). In the present case the lesion was neither pediculated, ulcerate nor bleeding, and was firm to palpation.

Amyloidosis lesions are due to intercellular accumulation of a fibrous glycoprotein. They may be primary, or secondary to disorders like tuberculosis or rheumatoid arthritis. They may also be associated with multiple myeloma, but occasionally present as localized depositions without evidence of generalized disease (5). Clinically, amyloidosis lesions differ little from MRG, as noted by Yamaoka et al. (6), and diagnosis requires histopathological studies. Cases have been described in which an initial MRG lesion subsequently developed amyloid deposits (6). In the present case no amyloid deposits were detected.

Granular cell tumour (Abrikossoff's tumour) is an uncommon benign neoplasm of soft tissues, occurring preferentially on the tongue (3), with about 35% of the lesion surface occupied by nodules (7). This tumour presents as a subcutaneous nodule on the dorsum of the tongue, or on the margin. Size ranges from a few millimetres to several centimetres, and the overlaying mucosa is normal, with the lingual papillae unaltered. It is painless (3). Definitive diagnosis is based on histopathological findings.

Cancers of the tongue (mostly squamous cell carcinomas) account for about two-thirds of intraoral cancers. Such tumours are most frequently located on the margins of the tongue, at the union of the anterior two-thirds with the posterior third, or on the ventral surface. The dorsum of the tongue appears to be relatively resistant to the onset of new malignant lesions, although lesions in this location frequently arise from adjacent lesions (3,8). Cancer of the tongue is classically described as an ulcerate or indurated tumour, or as an ulcer, painful or painless, accompanied by adenopathies. The initial lesion is usually an erythematous plaque, generally painless, which may or not be associated with leucoplastic images. At advanced stages tumours may be endophytic, exophytic, mixed, or warty (3,8).

Clinical examination typically reveals a non-fluctuant immobile indurated lesion, not attached to the deep musculature. When the patient shows his/her tongue, it displaces to the lesion side, and loss of mobility is observed. All of these signs becoming increasingly marked as the tumour progresses (3,8).

There have been three previous reports of malignant transformation of MRG (9,10). However, Delamarre and van der Wall (11) have suggested that there is no clear relationship between MRG and cancer.

In the present case the lesion did not extend deep into the tongue musculature, and histopathology did not reveal any signs of malignancy.

In conclusion, the diagnosis of rhomboid glossitis (whether medial or atypical) is largely based on clinical examination, though histopathological studies of biopsies may be required if clinical appearance does not allow other possible diagnoses to be ruled out.

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