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Focal epithelial hyperplasia in a child from Surinam
Int. J. Oral Surg. 1975: 4:168-171
Case Reports
(Key words: epithelinm; mouth, neoplasms)
Focal epithelial hyperplasia in a child from Surinam I. V A N D E R WAAL, CHR. M. T E N B R U G G E N K A T E A N D W. A. M. V A N D E R 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 hyperplasia 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)
I n 1965 t h e t e r m f o c a l epithelial hyperplasia was i n t r o d u c e d b y ARCHARD, HECK & STANLEYt. A s y n o n y m that sometimes is u s e d is " H e c k ' s disease." T h i s disorder of the oral m u c o s a is c h a r a c t e r i z e d by multiple, m o r e or less p a p i l l o m a t o u s - l i k e lesions a n d is p o s s i b l y caused b y a virus 4. Initially, f o c a l epithelial hyperplasia was t h o u g h t to be a n e x t r e m e l y rare entity, occurring o n l y in c h i l d r e n of A m e r i c a n Indian origin, w i t h an equal sex distribution. Since 1965, h o w e v e r , m a n y articles h a v e been written, s h o w i n g that f o c a l epithelial hyperp l a s i a is n o t so rare and m a y occur in several r a c e s a n d certainly is not limited to youngsters. A m o n g E s k i m o s in G r e e n l a n d 4, CLAUSEN, MOGELTOFT, ROED-PETERSEN r l a l N D B O R G 3 e v e n r e p o r t e d a p r e v a l e n c e of a b o u t 19 % , It is g e n e r a l l y a c c e p t e d that focal epithelial h y p e r p l a s i a is a harmless and selfh e a l i n g c o n d i t i o n , n o t resulting in scarf o r m a t i o n . So f a r , the d i s o r d e r has not been reported from the Netherlands.
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, papillomatous-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 erosions 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 o n the buecal aspect of the gingiva between central and lateral incisors. Initially, this gingival lesion was overlooked.
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 underlying 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 schoolmates were not affected by the disease either.
gingiva, and were only detected at the second 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 disappear 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 pathognomonic by themselves and are somewhat subjective. The changes that have been described in the literature as occurring in the underlying connective tissue and the salivary glands, do not seem to be of great diagnostic importance either. Nevertheless, the clinical diagnosis of focal epithelial hyperplasia has to be supported by a histologic exam-
Discussion I n this case report nothing new has been added o n the etiology of focal epithelial hyperplasia. A familial occurrence as reported b y GOMEZ, CALLE, ARCILA & PINDBORG ~, NCHOCK s a n d BUCHNER ~I; MASS~ w a s
absent, although the multiple pin-point sized erosions of the oral mucosa of the patient's sister were n o t understood. The tongue a n d gingiva are thought to be infrequent sites, except in Greenland Eskimos~, W e doubt, however, whether this is true. I n our patient the lesions of the tongue were very small, as were the lesions on the
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.
ination. Just t a k i n g a cytologic does n o t seem to b e sufficient~.
scraping
References 1. ARCHARD, H. O., HECK, I. W. & STANLEY, H. R.: Focal epithelial hyperplasia: an unusual 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., ROEDPETERSEN, 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 keratoacanthoma, verrucae, condyloma acuminatum, and focal epithelial hyperplasia. Oral Surg. 1972: 34: 604-618.
5. FISCHMAN, S. L.: Focal epithelial hyperplasia; case reports from Paraguay and Peru. Oral Surg. 1969: 28: 389-393. 6. GOMEZ, A., CALLE, C., ARClLA, G. & PrNDBORG, 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.: Multiple polypoid conditions of the oral mucosa. Br. J. Oral Surg, 1974: 12: 91-95. 8. SCHOCK, R. K.: Familial focal epithelial hyperplasia; report of case. Oral Surg. 1969: 28: 598-602.
Address:
Aid. Mondziekten en KaakcMrurgie Aeademisch Ziekenhuis der Vri]e Universiteit Amsterdam 1011 De Boelelaan 1117 Postbus 7057 The Netherlands
Case Reports
(Key words: epithelinm; mouth, neoplasms)
Focal epithelial hyperplasia in a child from Surinam I. V A N D E R WAAL, CHR. M. T E N B R U G G E N K A T E A N D W. A. M. V A N D E R 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 hyperplasia 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)
I n 1965 t h e t e r m f o c a l epithelial hyperplasia was i n t r o d u c e d b y ARCHARD, HECK & STANLEYt. A s y n o n y m that sometimes is u s e d is " H e c k ' s disease." T h i s disorder of the oral m u c o s a is c h a r a c t e r i z e d by multiple, m o r e or less p a p i l l o m a t o u s - l i k e lesions a n d is p o s s i b l y caused b y a virus 4. Initially, f o c a l epithelial hyperplasia was t h o u g h t to be a n e x t r e m e l y rare entity, occurring o n l y in c h i l d r e n of A m e r i c a n Indian origin, w i t h an equal sex distribution. Since 1965, h o w e v e r , m a n y articles h a v e been written, s h o w i n g that f o c a l epithelial hyperp l a s i a is n o t so rare and m a y occur in several r a c e s a n d certainly is not limited to youngsters. A m o n g E s k i m o s in G r e e n l a n d 4, CLAUSEN, MOGELTOFT, ROED-PETERSEN r l a l N D B O R G 3 e v e n r e p o r t e d a p r e v a l e n c e of a b o u t 19 % , It is g e n e r a l l y a c c e p t e d that focal epithelial h y p e r p l a s i a is a harmless and selfh e a l i n g c o n d i t i o n , n o t resulting in scarf o r m a t i o n . So f a r , the d i s o r d e r has not been reported from the Netherlands.
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, papillomatous-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 erosions 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 o n the buecal aspect of the gingiva between central and lateral incisors. Initially, this gingival lesion was overlooked.
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 underlying 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 schoolmates were not affected by the disease either.
gingiva, and were only detected at the second 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 disappear 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 pathognomonic by themselves and are somewhat subjective. The changes that have been described in the literature as occurring in the underlying connective tissue and the salivary glands, do not seem to be of great diagnostic importance either. Nevertheless, the clinical diagnosis of focal epithelial hyperplasia has to be supported by a histologic exam-
Discussion I n this case report nothing new has been added o n the etiology of focal epithelial hyperplasia. A familial occurrence as reported b y GOMEZ, CALLE, ARCILA & PINDBORG ~, NCHOCK s a n d BUCHNER ~I; MASS~ w a s
absent, although the multiple pin-point sized erosions of the oral mucosa of the patient's sister were n o t understood. The tongue a n d gingiva are thought to be infrequent sites, except in Greenland Eskimos~, W e doubt, however, whether this is true. I n our patient the lesions of the tongue were very small, as were the lesions on the
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.
ination. Just t a k i n g a cytologic does n o t seem to b e sufficient~.
scraping
References 1. ARCHARD, H. O., HECK, I. W. & STANLEY, H. R.: Focal epithelial hyperplasia: an unusual 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., ROEDPETERSEN, 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 keratoacanthoma, verrucae, condyloma acuminatum, and focal epithelial hyperplasia. Oral Surg. 1972: 34: 604-618.
5. FISCHMAN, S. L.: Focal epithelial hyperplasia; case reports from Paraguay and Peru. Oral Surg. 1969: 28: 389-393. 6. GOMEZ, A., CALLE, C., ARClLA, G. & PrNDBORG, 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.: Multiple polypoid conditions of the oral mucosa. Br. J. Oral Surg, 1974: 12: 91-95. 8. SCHOCK, R. K.: Familial focal epithelial hyperplasia; report of case. Oral Surg. 1969: 28: 598-602.
Address:
Aid. Mondziekten en KaakcMrurgie Aeademisch Ziekenhuis der Vri]e Universiteit Amsterdam 1011 De Boelelaan 1117 Postbus 7057 The Netherlands