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Focal epithelial hyperplasia (FEH) of the oral mucosa occurring in a HIV-infected man is described. Molecular biology disclosed an HPV-32 type in oral lesions. The association of FEH and AIDS is uncommon although many HPV subtypes may manifest during HIV infection. R. Viraben, Service de Dermato-vénéréologie, CHU La Grave, Place Lange, F-31052 Toulouse Cedex (France) D ow nl oa de d by : 54 .7 0. 40 .1 1 10 /3 1/ 20 17 1 :2 6: 29 A M Focal epithelial hyperplasia (FEH) of the oral mucosa was defined as circumscribed slightly elevated pink or white papules located mainly on the lower lip and buccal mucosa [1]. Most commonly two types of human papillomavirus, HPV-13 and HPV-32, were recognized in FEH and considered as the causative agents [2]. We report a new case of an HPV-32 infection correlated with oral FEH developing during HIVinfection. Case Report A 37-year-old Caucasian homosexual man with known AIDS since 1987 presented with flat smooth oral papules on the mucosal part of the upper and lower lip. The tongue remained unaffected and the general examination disclosed only discrete papilloma on the chin. At the time of examination biological evaluation of the immune status revealed: CD4 lymphocytes 72/mm3; CD4/CD8 ratio 1/7; positive P24 antigen 225 pg/ml. A general treatment with zidovudine and sulfarnethoxazole-trimethoprim was followed for 6 months. We first considered the mucosal lesions as atypical oral hairy leukoplakia and therapy with aciclovir (1 g/day) was begun. A month Fig. 1. Lesions of FEH on the lower labial mucosa. KARGER E-Mail karger¢1⁄8karger.ch Fax + 4I 61 306 12 34 http://www.karger.ch D ow nl oa de d by : 54 .7 0. 40 .1 1 10 /3 1/ 20 17 1 :2 6: 29 A M © 1996 S.KargerAG. Basel 1018-8665/96/1933-0261 $ 10.00/0 later, the mucosal papules were unaffected and the treatment was stopped. Biopsy of oral papules was made because of new lesions on the mucosal surface of the cheek. Routine histological examination revealed epidermal hyperplasia with acanthosis and papillomatosis. A few cells were dysker-atotic, some with a clear cytoplasm and an irregular voluminous nucleus and marginated chromatin. All these changes were compatible with a viral origin. In situ hybridization was also performed as previously described [3]. Epsfein-Barr virus (EBV) genome was detected with bio Bamiil W cDNA probe. A strong signal with FITC-labeled anti-sense BHLF1 oligonucleotide probes demonstrated a lytic phase of EBV infection; labeling with EBER 1/2 oligoprobe remained negative and was supportive of absence of an EBV latent cycle. Molecular cloning of extracted DNA found an HPV-32 band pattern (G. Orth, Insti-tut Pasteur, Paris, France). During the 3 following years, no further specific therapy was initiated and the lesions spontaneously resolved, but recurrences were noted. Histological and virological examinations were repeated during this period on the newly developed lesions, showing similar results concerning morphological and virological analysis, but EBV remained unde-tectable. Discussion FEH of the oral mucosa was first described in 1965 by Archard et al. [1]. The diagnosis is based on clinical features, histology and detection of HPV types 13 and 32. However, additional HPV types have been detected in FEH (types 1,6-related, 11, 16, 18) [4, 5] and the clinical picture may be almost indistinguishable from condylomata of the oral cavity [6]. FEH occurs mostly among American Indians and Eskimos of Greenland but is sporadically reported in Caucasians [4]. An inherited predisposition is further supported by cases of familial recurrence with similar HPV types [7]. Initial reports of FEH postulated an associated undefined immunodeficiency [1]. In some cases, FEH has developed in immune-deficient patient: in 1 case [8], FEH occurred after immunosuppressive treatment, the other [9] developed in 2 siblings with leukocyte adhesion deficiency. Unusual HPV-associated oral lesions have been investigated in HIV-positive patients [10, 11] but to our best knowledge, only 1 case of FEH associated with HIV infection has previously been reported in the studies by Vilmeretal. [12]. In HIV infection, oral warts harbor different HPV types: if HPV-7 appears more prevalent in the studies by Greenspan et al. [10] and de Villiers [13], other HPV types, including 13,18 and 32, were also detected without clinical, histomorphological and virological correlations. In our case the clinical appearance, the histological features and the natural course are highly suggestive of FEH. The virological analysis of two different biopsy specimens disclosed HPV-32. This HPV type is closely associated with FEH but also found in limited cases of condylomata acuminata and squamous cell papilloma [2, 14]. The simultaneous occurrence of EBV infection initially noted may be coincidental because of the high prevalence of both HPV and EBV infections during AIDS. A conspicuous clinical and virological assessment of mucosal lesions of HIV patients might provide further argument about the relationship between FEH and immunodeficiency. D ow nl oa de d by : 54.70.40.11-10/31/20171:26:29AM ReferencesArchard HO, Heck JW, Stanley HR: Focal epithelial hyperplasia: An unusual oral mucosallesion found in Indian children. Oral Surg 1965;20:201-212.Beaudenon S, Praetorius F, Kremsdorf D, Lutzner M, Worsaal N, Pehau-Arnaudet G, Orth G: Anew type of human papillomavirus associated with oral focal hyperplasia. J Invest Dermatol1987;88:130-135.Courtade M, Brousset P, Delsol M, Gorguet B, Viraben R, Voigt JJ, Delsol G: Detection si-multanée de papillomavirus humains et du virus d’Epstein-Barr en cycle lytíque dans des lesionsde leucoplasie orale chevelue par hybridation in situ non isotopique. Ann Pathol 1992;12:353-357.Bodokh I, Lacour JPh, Rainero C, Orth G, Per-rin C, Hoffman P, Santini J, Ortonne JP: Hyper-plasie épithéliale focale: aspect clinique in-habituel. Ann Dermatol Vénéréol 1993; 120: 555-557. Cohen PR, Hebert AA, Adler-Storthz KA: Focal epithelial hyperplasia: Heck disease. Pe-diatrDermatol 1993;10:245-251.Pardisi M, Mostaccioli S, Celano C, Angelo C, Ruatti P, Onetti Muda A, Faraggiana T: Infantilecondylomata of the oral cavity. Pediatr Dermatol 1992;9:107-Ill.Premoli-De-Percoco G, Cisternas JP, Ramirez JL, Galindo I: Focal epithelial hyperplasia:Human-papillomavirus-induced disease with a genetic predisposition in a Venezuelan family.Hum Genet 1993;91:386-388.Tan KN, Medak H, Cohen L, Burlahou P: Focal epithelial hyperplasia in a Mexican Indian. ArchDermatol 1969;100:474-477.Mealey BL, Hallmon WW, Waldrop TC: Occurrence and resolution of focal epithelialhyperplasia in two siblings with leukocyte adhesion deficiency. J Periodontol 1993;64: 149-152. Greenspan D, de Villiers EM, Greenspan JS, de Souza YG, zur Hausen H: Unusual HPV types inthe oral warts in association with HIV infection. J Oral Pathol Med 1988;17:482-487.Itin PH, Lautenschlager S, Flückiger R, Rufli T: Oral manifestations in HIV infected patients:Diagnosis and management. J Am Acad Dermatol 1993;29:749-760.Vilmer C, Cavelier-Balloy B, Pinquier L, Blanc F, Dubertret L: Focal epithelial hyperplasia andmultifocal human papillomavirus infections in an HIV-seropositive man. J Am Acad Dermatol1994;30:497-498.De Villiers EM: Prevalence of HPV-7 papillo-mas in the oral mucosa and facial skin of a patientwith human immunodeficiency virus (letter). Arch Dermatol 1989; 125:1590.Praetorius F, Praetorius-Clausen P: Cytopathic changes in human oral focal epithelialhyperplasia; in Howley P, Broker T (eds): The Eighth International Papillomavirus WorkshopTaos 1989, p 223. 262 Dermatology 1996;193:261-262Viraben/Aquilina/Broussel/Bazex Downloadedby: 54.70.40.11-10/31/20171:26:29AM