Int. J. Oral Surg. 1975: 4:168-171

(Key words: epithelinm; mouth, neoplasms) Case Reports

Focal epithelial hyperplasia in a child from Surinam

I. V A N D E R WAAL, CHR. M. TEN B R U G G E N K A T E A N D W. A. M. V A N DER K W A S T

Departments O[ Oral Surgery and Oral Pathology, Free University, Amsterdam, the Netherlands

AnST~tCT -- A 12-year-old Negro boy, born in Surinam and living in Amsterdam, is described. The diagnosis was focal epithelial hyper- plasia of the oral mucosa. Some of the clinical and histopathologic aspects of the condition are discussed.

(Received Jot publication 14 April, accepted 15 May 1975)

In 1965 the t e r m foca l epithelial hyper- p las ia was in t roduced b y ARCHARD, HECK & STANLEY t. A synonym that somet imes is used is " H e c k ' s disease." This disorder of the oral m u c o s a is charac te r ized by mul- t iple, m o r e or less papi l lomatous- l ike lesions and is poss ib ly caused by a virus 4.

Ini t ia l ly , foca l epithelial hyperplas ia was though t to be an ex t remely rare entity, oc- curr ing o n l y in ch i ldren of A m e r i c a n Indian

origin, w i th an equal sex distribution. Since 1965, h o w e v e r , many articles have been

wri t ten, s h o w i n g that foca l epi thel ial hyper- p las ia is n o t so rare and m a y occur in

several r aces and cer ta inly is not l imited to youngsters . A m o n g E s k i m o s in Green land 4,

CLAUSEN, MOGELTOFT, ROED-PETERSEN r la lNDBORG 3 even repor ted a preva lence of

about 19 %, I t is gene ra l l y accepted that focal epi-

thel ia l h y p e r p l a s i a is a harmless and self- hea l ing cond i t ion , n o t result ing in scar- fo rma t ion . So far , the d i sorder has not been repor ted f r o m the Nether lands .

Case report

In October 1974 a 12-year-old weU-nourished and healthy Negro boy, born in Surinam and living in Amsterdam for 3 years, was referred by his dentist to the Oral Surgery Department because of multiple, symptomless, papillo- matous-like swellings of the oral mueosa. The lesions were located on the lower and upper lip, buccal mucosa, gingiva and on the borders of the tongue (Figs. 1 & 2). The hard palate was involved as well. The lesions had been noticed by his mother some months before. On stretching the mucosa, the lesions flattened somewhat, but did not disappear. Skin lesions were absent,

The clinical diagnosis of focal epithelial hyperplasia was confirmed by microscopic examination of a biopsy taken of one of the lesions of the lower lip (Figs. 3-5).

During a 6 month's follow-up hardly any change has been noticed. The patient's only sister, a 10-year-old girl, was examined too. In the oral cavity several pin-point sized ero- sions were noticed on the cheeks, the upper frenulum and the palate. These lesions were very unlike the lesions seen in focal epithelial hyperplasia. A biopsy was refused.

In the patient's mother no abnormalities of

FOCAL EPITHELIAL HYPERPLASIA I69

Fig. 1. Papillomatous-like lesions on the upper lip. There is also a small lesion on the buecal aspect of the gingiva between central and lateral incisors. Initially, this gingival lesion was over- looked.

Fig. 2. Identical lesions, as shown in Fig. 1, on lower lip.

170 VAN I)ER WAAL, TEN BRUGGENKATE AND VAN DER KWAST

Fig. 3. Low-power view of a lesion in focal epithelial hyperplasia. No changes in under- lying connective tissue, nor in salivary glands.

Fig. 4. Rather typical aspect of rete pegs in focal epithelial hyperplasia, showing a tendency to form buds. • 130.

the oral mucosa could be detected. The father had died some years before. The disorder was not known to have occurred in other relatives. According to the mother, the boy's school- mates were not affected by the disease either.

Discussion In this case report nothing new has been added on the etiology of focal epithelial hyperplasia. A familial occurrence as re- ported by GOMEZ, CALLE, ARCILA & PIND- BORG ~, NCHOCK s a n d BUCHNER ~I; MASS~ was

absent, al though the multiple pin-point sized erosions of the oral mucosa of the patient 's sister were not understood. The tongue and gingiva are thought to be in- frequent sites, except in Greenland Eski- mos~, We doubt, however, whether this is true. In our patient the lesions of the tongue were very small, as were the lesions on the

gingiva, and were only detected at the sec- ond and third visit. It is conceivable that some of the lesions in patients affected by focal epithelial hyperplasia are overlooked or just appear after each other.

The lesions in our patient did not dis- appear on stretching the adjoining mucosa, which does not exclude the clinical diagnosis of focal epithetial hyperplasia, as is more or less suggested by some authorsv.

The histopathologic aspects of focaI epithelial hyperplasia are not pathognomon- ic by themselves and are somewhat subjec- tive. The changes that have been described in the literature as occurring in the under- lying connective tissue and the salivary glands, do not seem to be of great diagnostic importance either. Nevertheless, the clin- ical diagnosis of focal epithelial hyperplasia has to be supported by a histologic exam-

FOCAL EPITHELIAL HYPERPLASIA t71

Fig. 5. Mitotic-like figures and nuclear inclusions in upper part of stratum spinosum. These features can be seen in, but are not pathognomonic of, focal epithelial hyperplasia. • 530.

inat ion. Jus t t ak ing a cytologic scraping

does no t seem to be sufficient~.

References 1. ARCHARD, H. O., HECK, I. W. & STANLEY,

H. R.: Focal epithelial hyperplasia: an un- usual oral mucosal lesion found in Indian children. Oral Sttrg. 1965: 20: 201-222.

2. BUCI-INER, A. & MASS, E.: Focal epithelial hyperplasia in an Israeli family. Oral Sttrg. 1973: 36: 507-511.

3. CLAUSEN, F. P , MOGELTOFT, M., ROED- PETERSEN, B. & PINDBORO, J. J.: Focal epithelial hyperplasia of the oral mucosa in a south-west Greenlandic population. Scand. J. Dent. Res. 3.970: 78: 287-294.

4. CLAUSEN, F. P.: Rare oral viral disorders (molluscum contagiosum, localized kerato- acanthoma, verrucae, condyloma acumin- atum, and focal epithelial hyperplasia. Oral Surg. 1972: 34: 604-618.

5. FISCHMAN, S. L.: Focal epithelial hyper- plasia; case reports from Paraguay and Peru. Oral Surg. 1969: 28: 389-393.

6. GOMEZ, A., CALLE, C., ARClLA, G. & PrND- BORG, J. J.: Focal epithelial hyperplasia in a half-breed family of Colombians. J. Am. Dent. Assoc. 1969: 79: 663-669.

7. GUSTERSAN, B. A. & GREENSPAN, I. S.: Mul- tiple polypoid conditions of the oral mucosa. Br. J. Oral Surg, 1974: 12: 91-95.

8. SCHOCK, R. K.: Familial focal epithelial hyperplasia; repor t of case. Oral Surg. 1969: 28: 598-602.

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Aid. Mondziekten en KaakcMrurgie Aeademisch Ziekenhuis der Vri]e Universiteit Amsterdam 1011 De Boelelaan 1117 Postbus 7057 The Netherlands