ARTICLE
Focal epithelial hyperplasia (FEH) of the oral mucosa (Heck disease)
has been observed among American Indians and less frequently in Eskimos
from Greenland and Alaska. On the contrary, isolated cases have been reported
among Caucasians in the rest of the world. This prevalence of the disease
seems to be of genetic predisposition [1-4].
The cause of the disease is a human papilloma virus (HPV) [5-7] and
in the majority of the cases DNA of HPV 13 and less frequently of HPV
32 has been identified [4, 8].
In this study we present the first case of FEH in Greece in which HPV
13 was found with PCR analysis and which has been successfully treated
with CO2 laser [9-11].
Case report
A 17 year-old healthy Greek Caucasian girl was referred for persisting
oral lesions which the patient had had for one year. On examination she
presented multiple, soft and slightly elevated sessile papules, of 2 to
12 mm in diameter. They were located mainly on the inner aspect of the
lower lip and to a lesser extent on the parietal mucous membranes and
the underside of the tongue. Their colour was reddish to whitish or red,
similar to the normal surrounding mucosa (Fig.
1).
The patient complained only of mild discomfort during speaking and eating.
No other viral diseases were clinically seen on the patient's skin or
the mucosa. A small cavernous haemangioma had been present on the right
half of the lower lip since birth.
Personal and family history were negative.
A diagnosis of HPV infection was suggested and biopsy specimens were
surgically removed from the inner aspect of the lower lip under local
anesthesia. A part was fixed with 10% formalin and embedded in paraffin
for histological examination which revealed: acanthosis, dyskeratosis,
parakeratosis, balloon cells, hyperchromasia of nucleus, binucleation
and perinuclear clear halo, findings compatible with HPV infection (Fig.
2). The other half of the specimen was snap-frozen and stored
at 70° C for PCR examination. This method was used because
of its high sensitivity and specificity in the detection and typing of
HPVs in humans. Total DNA extraction was performed by the salt extraction
method [12]. For the detection and typing of the specific HPV two step
analysis was used. A multiprimer set for the L1 gene detecting HPV types
6, 8, 11, 13, 16, 18, 30, 31, 32, 33 and 45 was used for the initial PCR
amplification. A band was produced indicating the presence of the virus.
A second step including primers for each of the above HPV types was done.
The second PCR analysis was positive for HPV 13 only. All reactions were
run in duplicate with positive, negative controls and blanks (Fig.
3).
The lesions were removed under local anaesthesia with CO2
laser set at 5-7 W. The laser operated in a continuous mode. The procedure
was carried out with no bleeding and the traumas left with no sutures
or dressing. After two weeks all the lesions were healed with no scarring.
There have been no recurrences for a period of 20 months (Fig.
4).
Discussion
FEH is clinically characterized by multiple circumscribed, sessile,
soft elevated nodules of the oral mucosa which sometimes form clusters.
They are reddish or whitish or like the adjoining oral mucosa. In our
case the clinical manifestations were similar to those usually reported
[1, 4].
The disease affects both sexes and all ages but it has been reported
especially in Indians and Eskimos. However, there have been a few cases
reported in Caucasians [1, 2]. As far as we know our case is the first
one in Greece in a young Caucasian girl.
The diagnosis of FEH can be made clinically but histological examination
may show characteristics of viral infection, as in our case. However,
the precise diagnosis is made by in situ hybridization or PCR analysis,
where two types of viruses have been reported, namely HPV 13 and HPV 32
[5-12]. In our case HPV 13, which is the most common, was found using
PCR.
The course of the disease is with mild subjective symptoms and a spontaneous
remission is possible. Different therapeutic procedures have been reported
and CO2 laser surgery seems to be one of the best [9-11]. Advantages
of this method are the haemostatic effect, the healing without scarring
and the compliance of the patient. Our patient has been followed up for
20 months without recurrences.
Accepted on 10/4/00
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