Focal epithelial hyperplasia. A rare disease in our area
Hiperplasia epitelial focal. Una rara enfermedad en nuestro medio

 

Rafael Segura Saint-Gerons ⁽¹⁾, Mariano Toro Rojas ⁽²⁾, Alejandro Ceballos Salobreña ⁽³⁾,
Jose Luis Aparicio Soria ⁽⁴⁾, Helena Fuentes Vaamonde ⁽⁵⁾

(1) Estomatólogo de Atención Primaria. C.S. Peñarroya. Córdoba
(2) Catedrático de Anatomía Patológica. Facultad de Medicina de Córdoba
(3) Catedrático de Medicina Oral. Facultad de Odontología de Granada
(4) Pediatra de Atención Primaria. C.S. Peñarroya. Córdoba
(5) Medico Adjunto. Servicio de Anatomía Patológica. Hospital Comarcal de Pozoblanco. Córdoba. España

Address:
Rafael Segura Saint-Gerons
Pza. Dr. Emilio Luque 3,3º.14003 Córdoba
Tfno: 957-485094
E-mail: 957485094@terra.es

Received: 31-01-2004. Accepted: 10-10-2004

 

INTRODUCTION

Focal epithelial hyperplasia is a rare disease of the oral cavity in our society.

It was first described by March in 1881, who published a case of papular tumors of the oral mucosa in a 9-year-old girl, whose clinical signs coincided with those later observed by Helms in Eskimos in Greenland in 1894, and by Sterm in a Caucasian in Berlin in 1922 (1,2).

The term focal epithelial hyperplasia was again used by Archard in 1965 to describe oral lesions found in certain groups of Indians in New Mexico (3).

The frequency of this disease varies widely from one geographic region to another, and can vary, according to Clausen, from 0.002 to 35% depending on the population studied (4).

In our region the incidence is even lower still, existing, until now, one case described by Ficarra in Italy (5), another in Madrid by Simón (6), a case described by Bassioukas in Greece (7) and two cases described by Bagán in HIV+ patients (8).

The papillomavirus is involved in the etiology of this process, genotypes 13 and 32 being those detected with highest frequency by DNA in situ hybridization techniques (9, 10), although cross reactions with HPV 6, 11 and 18 have also been described (3). In addition, genetic factors, malnutrition, poor hygiene and social conditions have also been associated with this disease (11).

Clinically, the process is characterized by the presence of multiple papular lesions on the lower lip, jugal mucosa or tongue, small in size and of the same colour as the surrounding mucosa.

Histologically, the disease presents parakeratosis, acanthosis with epithelial projections that anastomose horizontally, and usually koilocytes and other cellular changes indicative of viral infection. Cells with nuclear degeneration, called mitosoid cells, can also appear, although not consistently, perhaps due to the fact that a serial study of the preparations has not been undertaken (12).

The differential diagnosis should be made against condyloma acuminatum, florid oral papillomatosis, Cowden's syndrome or Crohn's disease (11, 13).

This disease tends to spontaneously regress, although it can persist for many years, with cases having been described in the elderly (14).

Surgical treatment is only required when its presence in the red area of the lip causes aesthetic problems.

We present the case of an HIV- Saharan girl, diagnosed with focal epithelial hyperplasia.

CLINICAL CASE

A 9-year-old patient, referred by her pediatrician for multiple papular lesions in the mouth. On exploration, multiple lesions of varying size (1-4mm) were found on the retro-commisural area and mucosa of the lower lip, of the same colour as the mucosa, with no inflammatory appearance (Fig. 1), soft on palpation and not pediculated. Various verrucae were found on the ring finger, and since a papilloma of the gingiva had been extirpated the year before, we decided to carry out a biopsy in order to discount either a papillomatosis or a verrucal infection of the oral mucosa.

Under local anesthetic, we extirpated a fragment of mucosa that contained a quantity of these lesions.

The histopathological study revealed a squamous epithelium with parakeratosis, acanthosis and marked papillomatosis, with horizontal fusion of papillae (Fig. 2), hyperplasia of basal cells, and isolated perinuclear cellular vacuolization (koilocytosis), cellular binucleation and nuclear irregularities. The presence of epithelial dysplasia was not detected. There were very isolated mitosoid cells (Fig. 3).

Given the diagnosis of focal epithelial hyperplasia, no treatment was established, indicating a revision the following year since the girl is on a regular holiday exchange program.

DISCUSSION

Focal epithelial hyperplasia, although rare in our society, is a diagnosis that must be taken into consideration due to the high number of immigrants living in our country.

While the greatest incidence of these cases occurs in certain populations in America and Greenland according to Clausen (4), Cohen indicates that although the first cases came to light in certain ethnic groups (13), owing to certain conditions, such as the ease at which a disease can be transported, it is a disease which is being seen in other geographic areas.

Although some authors have established this as a childhood disease, others include older age groups. It will perhaps be necessary to study its incidence in larger populations of different ages in order to definitively establish this point.

The higher incidence of this disease in women than in men could be explained by the conditions under which women of some ethnic groups live.

In the case presented here, the patient matches the characteristics of age and sex in which this disease presents with greatest frequency, and although the in situ hybridization detection of papillomavirus was not possible, the histology showed signs of viral infection, such as the presence of koilocytes or characteristic nuclear alterations (10).

This hypothesis is supported by the presence of verrucae vulgaris on the fingers, also indicated by González López (14), as well as by the fact that a papilloma of the gingiva had been extirpated the previous year.

The differential diagnosis of this pathology should be made against florid oral papillomatosis, condyloma acuminatum, Cowden's syndrome or Crohn's disease. The differential diagnosis is arrived at through clinical means and likewise the differential diagnosis with Cowden's syndrome is carried out in the absence of hamartomas in other parts of the body such as the skin, breasts, thyroids, stomach or colon. With Crohn's disease, the absence of regional enteritis, typical of this illness, will discount this diagnosis. In any case, it is the histopathological analysis that will provide the definitive diagnosis.

With respect to condyloma acuminatum, the differential diagnosis is important given that when not very keratinized lesions are found, the lesions are of the same colour as the surrounding mucosa. The absence of previous sexual contact discounts the possibility of condyloma acuminatum; even so it would be the anatomopathological analysis that provides the precise diagnosis of this disease (15,16).

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