Multifocal epithelial hyperplasia confined to the interdental papilla of an adult Chinese man: a rare case report and literature review.

BACKGROUND: Multifocal epithelial hyperplasia (MEH), or focal epithelial hypertension (FEH), or Heck's disease, is an uncommon, benign oral mucosal disease associated with human papillomavirus infection. It is mostly observed in indigenous populations of the world, and has been rarely reported in China. However, previous research suggested there might be a greater prevalence of MEH in the Chinese population. While predominantly involves the lips, buccal mucosa and tongue, MEH was occasionally reported to affect the hard palate and gingiva as well. CASE PRESENTATION: This paper reports a case of extensive MEH lesions that confined to the interdental papilla of a Chinese male without detection of HPV, and summarizes the published gingiva-involved MEH reports from 1966 until present. The lesions were excised with an Er: YAG laser after scaling and root planning, no recurrence was observed after 6-month follow-up. CONCLUSIONS: The present report illustrates the need for clinicians to be aware of rare presentations of MEH to facilitate a prompt diagnosis and proper management. More reports are encouraged to determine a correct prevalence rate of MEH in China.

Lesiones papilomatosas orales: hiperplasia epitelial focal / Oral papillomatous lesions: Focal epithelial hyperplasia

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Useful cytological confirmation of HPV 13 in lesional mucosa enhances diagnosis of focal epithelial hyperplasia.

An 11-year-old female presented with multiple oral lesions for several months. Histopathological findings suggested focal epithelial hyperplasia (FEH), also known as Heck disease. FEH is strongly associated with Human papillomavirus (HPV), especially genotypes 13 and 32. An oral swab of a mucosal lesion was subsequently obtained for cytology, immunohistochemistry and in situ hybridization. In addition, in situ hybridization and immunohistochemistry were also performed retrospectively on the biopsy specimen for correlation. The cytology specimen showed squamous cells with enlarged, slightly atypical nuclei and rare perinuclear halos. The histology findings included papillomatosis with acanthosis, mild nuclear atypia and focal perinuclear halos. The immunohistochemistry for the consensus HPV L1 capsid protein was found in both the cytology and biopsy specimens indicating that the lesion was HPV-related. High viral copy numbers of HPV 13 were detected by in situ hybridization in both the cytology and histology specimens. Although histologic features of FEH have been well characterized in the literature, to our knowledge, this is the first case to describe in FEH with adjunct immunohistochemistry and in situ hybridization results. Furthermore, these findings assisted in our diagnosis since the patient's clinical presentation was a diagnostic challenge with smooth dome-shaped papules instead of the typically described flat-topped verrucous lesions seen in FEH. In summary, our case reveals that there is a high concordance between the HPV 13 detection in the cytology and histology of FEH, and that performing cytology in addition to histology can be used to optimize diagnostic evaluation towards appropriate patient care.

Hiperplasia epitelial focal: relato de caso clínico / Focal epithelial hyperplasia: case report

Introdução: a hiperplasia epitelial focal (HEF) ou doença de Heck, caracteriza-se por ser uma lesão benigna rara da mucosa bucal. Geralmente, está associada a proliferação do epitélio pavimentoso atribuída principalmente ao papiloma vírus humano (HPV) dos subtipos 13 e 32. Objetivo: relatar o caso de um paciente, com suspeita diagnóstica de HEF, descrever suas manifestações clínicas bucais e histopatológicas. Metodologia: homem, 38 anos, melanoderma, compareceu no Serviço Universitário com queixa de ardência em mucosa bucal. Ao exame clínico, apresentava múltiplas lesões papulares, firmes à palpação, de coloração rósea, com pápulas aglomeradas ou isoladas, localizadas em rebordo alveolares, gengiva inserida e livre da arcada superior e inferior, mucosas jugais e labiais, compatíveis com lesões causadas por HPV. Realizou-se biópsia incisional das lesões bucais, o laudo histopatológico descreveu o fragmento de mucosa bucal que estava revestida por epitélio pavimentoso estratificado paraceratinizado com acantose proeminente, projeções focais confluentes para o tecido conjuntivo, confirmando a suspeita diagnóstica clínica. Conclusão: a HEF é uma patologia rara, benigna da mucosa, indolor e que pode se resolver espontaneamente. Portanto, é necessário o diagnóstico adequado através da biopsia e laudo histopatológico.

Introduction: focal epithelial hyperplasia (FEH) or Heck's disease is characterized as a rare benign lesion of the oral mucosa. It is usually associated with proliferation of the squamous epithelium, mainly attributed to human papillomavirus (HPV) subtypes 13 and 32. Objective: to report the case of a patient with a diagnostic suspicion of HEF, to describe its oral clinical and histopathological manifestations. Methodology: hale, 38 years old, black, attended the University Service with a complaint of burning in the oral mucosa. On clinical examination, she presented multiple papular lesions, firm to palpation, pink in color, with agglomerated or isolated papules, located in the alveolar ridge, inserted gingiva free from the upper and lower arch, jugal and labial mucosa, compatible with lesions caused by HPV. An incisional biopsy of the oral lesions was performed, the histopathological report described the fragment of oral mucosa that was lined with parakeratinized stratified squamous epithelium with prominent acanthosis, focal projections confluent to the connective tissue, confirming the clinical diagnostic suspicion. Conclusion: FEH is a rare, benign mucosal pathology, painless and can resolve spontaneously. Therefore, proper diagnosis through biopsy and histopathological report is necessary.

Oral human papilloma virus infection: an overview of clinical-laboratory diagnosis and treatment.

OBJECTIVE: The aim of this review is to describe the "hot points" of current clinical governance for oral HPV comprising the use of new diagnostic molecular procedures, namely, Pyrosequencing and Next Generation Sequencing. MATERIALS AND METHODS: The data on oral HPV was collected through two levels of research. First for all, we used the canonical medical search engines, PubMed, and Medline, followed by the study of current commercial tools for HPV diagnosis, particularly within commercial companies involved in the molecular procedures for HPV detecting and genotyping. RESULTS: Different medical procedures are now described and used throughout the world in HPV diagnosis and treatment. However, the laboratory methods are often validated and used for genital infections, and, in these cases, data are missing in the literature as regards the clinical approach for oral lesions. CONCLUSIONS: Dental care units are often the front line for a clinical evaluation of a possible HPV lesion in the oral cavity, which means that correct clinical governance could avoid a viral neoplastic progression of this disease with great advantages for the patient. In this case, the problem is due to the difficulty in lesion recognition but also and more especially the absence of correct laboratory diagnosis and subsequent treatment in the clinical course.

Unusually high incidence of multifocal epithelial hyperplasia in children of the Nahuatl population of Mexico.

BACKGROUND: Multifocal epithelial hyperplasia is an uncommon disease of the oral mucosa caused by the human papilloma virus. AIM: To study the clinical and pathological findings of multifocal epithelial hyperplasia detected during an oral examination of 343 Mexican Nahuatl children from a single primary school in El Paso de Cupilco, Mexico. METHODS: A thorough oral examination was performed in all children and clinical data (age, gender, location and number of lesions) were documented and analyzed. RESULTS: Multifocal epithelial hyperplasia was diagnosed in 110 of the 343 children (32.3%). The ages of the children varied from 5 to 15 years, and of these, 56.3% were girls. The lesions were asymptomatic, 0.2 to 3.0 cm in diameter, soft, round to oval, smooth surfaced, sessile papulonodules, similar in colour to that of the surrounding mucosa. The lesions were commonly seen on the buccal mucosa and tongue, and most affected children (85%) had less than 5 lesions. Children in the 7 to 10 years age group were most often affected. LIMITATIONS: Human papillomavirus typing was not done owing to a lack of facilities. CONCLUSIONS: There is a high incidence of multifocal epithelial hyperplasia in Nahuatl children with a predilection for females.

Multifocal Epithelial Hyperplasia.

Multifocal epithelial hyperplasia is a rare disease associated with human papilloma virus types 13 and 32. Diagnosis is based on clinical and histopathological findings, and most lesions are asymptomatic and regress spontaneously with time. The purpose of this paper is to describe a five-year-old girl who presented with multiple intraoral lesions on the buccal mucosa and tongue, which regressed spontaneously in 15 months.

Multifocal epithelial hyperplasia: a forgotten condition in the elderly.

Multifocal epithelial hyperplasia (MEH), also known as focal epithelial hyperplasia and Heck disease, is a relatively rare condition caused by the human papillomavirus. This case report describes a 92-year-old man who presented with multiple, asymptomatic, circumscribed, soft, flattened papules in different sizes on the retrocommissure of his lower lip. The lesions were of 2 months' duration and had the same coloration as the adjacent normal mucosa. Histopathologic examination showed orthokeratinized stratified squamous epithelium with acanthosis, some areas of club-shaped rete ridges, and a few superficial epithelial cells with koilocytic changes and a mitosoid-like appearance. A diagnosis of MEH was established. Although MEH tends to occur in the first 2 decades of life, it can be encountered in elderly patients as well.

Focal epithelial hyperplasia by human papillomavirus (HPV)-32 misdiagnosed as HPV-16 and treated with combination of retinoids, imiquimod and quadrivalent HPV vaccine.

Focal epithelial hyperplasia (FEH) or Heck's disease is a rare, benign and asymptomatic mucosal proliferation associated with human papillomavirus (HPV) infection, mainly with genotypes 13 and 32. We report a florid case of FEH in an 11-year-old Haitian girl with systemic lupus erythematosus receiving immunosuppressive therapy. Cryotherapy was previously performed on numerous occasions with no results. We decided to prescribe a non-invasive and more comfortable treatment. A combination of topical retinoid and imiquimod cream was well tolerated and led to an important improvement. The evidence of infection by HPV-16 detected by polymerase chain reaction (PCR) technique, prompted us to prescribe the quadrivalent HPV vaccine (types 6, 11,16 and 18). Subsequent PCR sequencing with generic primers GP5-GP6 and further BLAST comparative analysis confirmed that genomic viral sequence in our case truly corresponded with HPV-32. This molecular misdiagnosis can be explained by the similarity between genomic sequences of both HPV-16 and -32 genotypes. At the 1-year follow up, we observed total clinical improvement and no recurrences of the disease. Complete healing in this case may correspond to a potential action of topical retinoid, imiquimod and the cross-protection mechanism of the quadrivalent HPV vaccine.

Hiperplasia epitelial focal / Focal epithelial hyperplasia

La hiperplasia epitelial focal (HEF) o enfermedad de Heck es una patolog¡a poco frecuente de la mucosa oral, caracterizada por una proliferaci¢n benigna del epitelio debida a la infecci¢n por el papilomavirus humano (PVH) tipos 13 y 32. El nombre enfermedad de Heck deriva del nombre del m‚dico que evalu¢ al primer paciente, en M‚xico, en 1961. Sin embargo, una revisi¢n de la literatura latinoamericana revela publicaciones previas realizadas por Estrada, en 1956, quien observ¢ esta entidad en indios colombianos. Algunos a¤os despu‚s, Soneira y Fonseca realizaron la primera descripci¢n completa de la enfermedad, y se¤alaron sus caracter¡sticas cl¡nicas e histol¢gicas y, adem s, sugirieron el posible origen viral. Finalmente, en 1965, Archard y col. la denominaron HEF, aunque en la actualidad se sugiere utilizar el nombre de hiperplasia multifocal del epitelio, que describe mejor las caracter¡sticas cl¡nicas y microsc¢picas de la enfermedad. La HEF, aunque.

The focal epithelial hyperplasia (HEF) or Heck's disease is a rare disease of the oral mucosa, characterized by benign epithelial proliferation due to infection with human papillomavirus (HPV) ttypes 13 and 32. The disease name derives Heck the name of the doctor who evaluated the first patient in Mexico in 1961. However, a review of Latin American literature reveals previous publications by Estrada, in 1956, who observed this entity in Colombian Indians. Some years later, Soneira and Fonseca made the first complete description of the disease, noting their clinical and histological features and also suggested the possible viral origin. Finally in 1965, Archard et al. HEF called it, although at present it is suggested to use the name multifocal epithelial hyperplasia, which best describes the clinical and microscopic features of the disease. The HEF is presented with a clear predominance of Americans, Eskimos and Indians some communities in South Africa, although isolated cases in other ethnic groups. In South America it is more common in the Amazon basin of Peru, Venezuela, Colombia, Bolivia, Brazil and Paraguay.

Expresión de calretinina en el tumor de células granulares: análisis del gradiente de intensidad en la inmunotinción con correlación clínico-patológica / Calretinin expression in granular cell tumour: an analysis of the immunostaining intensity gradient and its clinicopathological correlation

Introducción. El tumor de células granulares (TCG) es una lesión poco frecuente que aparece predominantemente en mujeres y en el área de cabeza y cuello. Múltiples estudios apuntan a que deriva de las células de Schwann y su perfil inmunohistoquímico ha sido ampliamente estudiado. La expresión de calretinina (CRT) es, sin embargo, la peor caracterizada en el TCG, por ello en este estudio se pretende analizar en detalle su expresión en esta forma tumoral. Material y método. Se analizó una amplia muestra de TCG (n = 43), de los que se recogieron datos clínico-epidemiológicos y se realizó inmunotinción en todos ellos con anticuerpo anti CRT estudiando su patrón de expresión. Resultados. Se encontró que este tumor muestra positividad global frente a CRT en una amplia mayoría de los casos (74%), encontrando un gradiente en la inmunotinción en el 53% de los TCG estudiados, situación observada en los tumores con superficies epiteliales en proximidad y que no ocurre cuando estos epitelios se encuentran ausentes o alejados del TCG. Conclusiones. La tinción frente a CRT es útil para el diagnóstico del TCG y se demuestra la existencia de un gradiente en la inmunotinción cuando el TCG se encuentra en la proximidad de epitelios escamosos, sobre todo cuando estos muestran hiperplasia epitelial pseudoepiteliomatosa, lesión a la que frecuentemente se encuentran asociados (AU)

Introduction. Granular cell tumour (GCT) is a rare lesion which predominately affects women and occurs most frequently in the head and neck. Many reports suggest that the tumour cells are probably derived from Schwann cells. The immunohistochemical profile of GCT has been extensively studied, although calretinin expression (CRT) is the least understood. Our aim is to undertake a detailed analysis of its expression in this tumour. Materials and methods. A large sample of GCT (n = 43) was analysed. Clinical-epidemiological data were collected and immunostaining with CRT was performed in all of them with anti-CRT antibody to study its expression pattern. Results. We found that most of the GCT cases were positive to CRT (74%). Furthermore, in 53% of cases, we detected an intensity gradient in the immunostaining in tumour cells with epithelial surfaces in proximity, which does not occur when these epithelia are absent or distant from the GCT. Conclusions. CRT is a useful marker for the diagnosis of GCT. We found the existence of an immunostaining intensity gradient when the tumour is in close proximity to squamous epithelia, especially when these show pseudoepitheliomatous epithelial hyperplasia, which is often associated with GCT (AU)

Oral focal fibrous hyperplasia and squamous cell papilloma treated with an erbium laser. Case presentation.

Mouth and oropharynx cancer constitute 5% of all malignancies; 95% of them are head and neck squamous cell carcinomas. Carcinogenesis is a multifactor process. Mutagenesis is also determined by the human papilloma virus which has recently been found to be etiologically associated with 20 to 25% of head and neck squamous cell carcinomas, mostly in the oropharinx. Focal fibrous hyperplasia of the connective tissue comes up as an answer to a chronic irritation in which a big amount of collagen can be found. As there exist certain clinical resemblance between squamous cell papilloma, fibrous focal hyperplasia and other mesenchimal tumors it is recommended to proceed, always, with removal and study. Two cases, one of an oral papilloma and another of a focal fibrous hyperplasia in pediatric patients, treated with an Er,Cr:YSGG laser wave length (mu) of 2780 nm are presented.

Hiperplasia epitelial multifocal: reporte de tres casos clínicos y revisión de la literatura / Multifocal epithelial hyperplasia: a report of three clinical cases and review of the literature

La hiperplasia epitelial multifocal es una enfermedad benigna que se caracteriza por la presencia de múltiples pápulas en la mucosa de la cavidad bucal, asociada con el virus del papiloma humano 13 y 32. En el presente trabajo se describen tres casos con características clínicas similares, donde se evidencian múltiples pápulas no dolorosas localizadas en mucosa bucal y lengua, mostrando un curso lento y benigno las cuales no habían sido diagnosticadas previamente por dentistas de práctica general. Se llevó a cabo el diagnóstico basado en las características clínicas patognomónicas de las lesiones, historia familiar y evolución; se les dio información a los pacientes acerca de su padecimiento y se mantienen periódicamente en observación. El objetivo de este trabajo es mostrar las características clínicas de la hiperplasia epitelial multifocal y hacer una revisión de la literatura en cuanto a la etiología de las lesiones su evolución, diagnóstico diferencial y tratamiento.

Multifocal epithelial hyperplasia is a benign disease characterized by the presence of multiple papules in oral cavity mucosa. It is as-sociated with HPV 13 and 32. In this paper, we describe three cases with similar clinical features, involving the presence of multiple painless papules localized in the oral mucosa and on the tongue, and displaying a slow and benign course. The condition had not been previously diagnosed by general dental practitioners. We made our diagnosis based on the pathognomonic clinical features of the lesions, family history, and evolution of the condition. The patients concerned were provided with information about their disease and kept under regular observation. The aim of this article is to describe the clinical features of multifocal epithelial hyperplasia and present a review of the literature on the etiology of the lesions, their evolution, differential diagnosis, and treatment.

Focal epithelial hyperplasia - an update.

Focal epithelial hyperplasia (FEH) is an asymptomatic benign mucosal disease, which is mostly observed in specific groups in certain geographical regions. FEH is usually a disease of childhood and adolescence and is generally associated with people who live in poverty and of low socioeconomic status. Clinically, FEH is typically characterized by multiple, painless, soft, sessile papules, plaques or nodules, which may coalesce to give rise to larger lesions. Human papillomavirus (HPV), especially genotypes 13 and 32, have been associated and detected in the majority of FEH lesions. The clinical examination and social history often allow diagnosis, but histopathological examination of lesional tissue is usually required to confirm the exact diagnosis. FEH sometimes resolves spontaneously however, treatment is often indicated as a consequence of aesthetic effects or any interference with occlusion. There remains no specific therapy for FEH, although surgical removal, laser excision or possibly topical antiviral agents may be of benefit. There remains no evidence that FEH is potentially malignant.