The most common sites of involvement include the labial, buccal, and lingual mucosa, but gingival and palatal lesions also have been reported. This disease normally manifests as multiple soft, flattened or rounded papules, which are usually clustered and the color of normal mucosa, although they may be scattered, pale, or rarely white. Notably, the lesions have a tendency to disappear on their own[1,2].
There is an etiological link between papillomavirus and this lesion. Moreover, living conditions such as malnutrition, poor hygiene and also genetic factors have been related to this disease[3].
This paper reports 3 cases of focal epithelial hyperplasia that have familial predominance in an Afghan family group. Besides, the diagnosis is based on histopathological features.
Case 1 was a 30 years old man suffering from painless small elevations on oral mucosa in the last 6 months. He had no history of previous systemic disease, and he had not taken any medications. His oral examination revealed normalcolored to white papules, mainly located on the buccal mucosa, mucosa of the lower lip and tongue. The lesions were not ulcerated nor inflamed (Figure 1A).
Figure 1A, B: Patient with multiple lesions in labial and buccal mucosa.
Case 2 was the nephew of case 1. She was a 12-year-old girl presented with about 5 months history of asymptomatic growths on her mouth. The patient did not report any systemic disease. Her oral examination revealed several papules measuring 0.2 to 1.0 cm extended over the buccal and labial mucosa. Moreover, the lesions on the right cheek occasionally had interference with mastication that resembled a string of beads (Figure 1B).
Case 3 was the brother of case 2. He was a 5-year-old boy without any systemic condition who complained of multiple asymptomatic lesions in his mouth for about 5 months. Intraoral examination showed multiple elevated pinkish papules on the buccal, upper and lower lip mucosa. All three patients described here were presented with poor dental hygiene.
The clinical diagnosis of focal epithelial hyperplasia was straightforward; however, in order to confirm the diagnosis a biopsy was performed on the largest lesions of labial and buccal mucosa after applying local anesthesia and the specimens were submitted for histopathologic evaluation. The histopathological assessment showed a squamous epithelium displaying regional parakeratosis, acanthosis (Figure 2A), basal cell hyperplasia, vacuolization of numerous epithelial cells (koilocytosis) (Figure 2B), Occasional binucleation and nuclear irregularity. There was no epithelial dysplasia. Also, collapsed nucleus that resembles a mitotic figure (mitosoid cell) was obvious (Figure 2C).
We advised the patients to have better oral and general hygiene. The lesions disappeared remarkably after 4 months illustrated in figure 3A and 3B.
Figure 3A,B: Remission of the lesions after 4 months of promoting good oral hygiene.
The human papillomavirus is associated with proliferations of squamous mucosa in this process. Type 13 and 32 has been detected with polymerase chain reaction[9,10]. It should be mentioned that HPV 6, 11 and 18 have been found as well[11]. The oral cavity can be the site of a variety of HPV-related lesions, some of which are microscopically and behaviorally benign. These include focal epithelial hyperplasia (Heck's disease), verruca vulgaris, condyloma acuminatum and squamous papilloma[12]. Human-tohuman transmission has been assumed to be the most important mode of contact. However, the virus could be transmitted from mother to child[12]. This helps to explain why Heck disease occurs in child group and its familial tendency. Notably, poor nutrition, hygiene and overall health may be related to disease[5]. These conditions comply with our cases.
Histological examination illustrated acanthosis of the oral epithelium. The rete ridges were widened, often confluent, and sometimes club shaped. Some superficial keratinocytes showed a koilocytic change similar to that seen in other HPV infections. Others occasionally demonstrate an altered nucleus that resembles a mitotic figure (mitosoid cell)[13].
Spontaneous regression of multifocal epithelial hyperplasia has been reported after months or years by enhancing oral and general hygiene (as we observed in all 3 patients), nevertheless, application of local cryotherapy has been used as well[14]. Also, application of vitamin A and sulfamides has been recommended by Archard and co-workers[1].
1) A rchard HO, Heck JW, Stanley HR. Focal epithelial hyperplasia:
An unusual oral mucosal lesion found in Indian children. Oral
Surg Oral Med Oral Pathol. 1965;20:201-12.
2) Terezhalmy GT, Riley CK, Moore WS. Focal epithelial hyperplasia
(Heck's disease). Quintessence Int. 2001;32:664-5.
3) Carlos R, Sedano HO. Multifocal papilloma virus epithelial
hyperplasia. Oral Surg Oral Med Oral Pathol. 1994;77:631-5.
4) Moussavi S. Focal epithelial hyperplasia: Report of two cases and
review of literature. J Am Dent Assoc. 1986;113:900-2.
5) H ashemipour MA, Shoryabi A, Adhami S, Mehrabizadeh
Honarmand H. Extensive focal epithelial hyperplasia. Arch Iran
Med. 2010;13:48-52.
6) Bennett LK, Hinshaw M. Heck's disease: Diagnosis and
susceptibility. Pediatr Dermatol. 2009;26:87-9.
7) Yasar S, Mansur AT, Serdar ZA, Goktay F, Aslan C. Treatment
of focal epithelial hyperplasia with topical imiquimod: Report of
three cases. Pediatr Dermatol. 2009;26:465-8.
8) Viraben R, Aquilina C, Brousset P, Bazex J. Focal epithelial
hyperplasia (Heck disease) associated with AIDS. Dermatology.
1996;193:261-2.
9) Syrjanen SM, Syrjanen KJ, Happonen RP, Lamberg MA. In situ
DNA hybridization analysis of human papillomavirus (HPV)
sequences in benign oral mucosal lesions. Arch Dermatol Res.
1987;279:543-9.
10) Premoli-de-Percoco G, Christensen R. Human papillomavirus in
oral verrucal-papillary lesions. A comparative histological, clinical
and immunohistochemical study. Pathologica. 1992;84:383-92.
11) Pfister H, Hettich I, Runne U, Gissmann L, Chilf GN.
Characterization of human papillomavirus type 13 from focal
epithelial hyperplasia Heck lesions. J Virol.1983;47:363-6.
12) Kumaraswamy KL, Vidhya M. Human papilloma virus and oral
infections: An update. J Cancer Res Ther. 2011;7:120-7.