Immunohistopathology of the Newly Discovered Giant Papillae Tongue Disorder in Organ-Transplanted Children.

BACKGROUND: Giant papillae tongue disorder (GPTD) is a newly discovered, long-lasting clinical disorder that may develop in organ-transplanted pediatric recipients. The key feature of this disorder is the unique tongue lesion, which comprises swollen fungiform papillae. The aim of this study was to characterize the immunohistopathology of this novel inflammatory condition. METHODS: Six organ transplanted children with GPTD were included in the study. Routine histopathology and immunohistochemical stainings for CD3, CD4, CD8, CD25, FOXP3, CD20, CD138, CD68, CD1a, CD15, CD23, and mast cell tryptase were performed. RESULTS: Immunohistochemical analyses of the oral lesions revealed a subepithelial infiltrate that was primarily composed of CD3- and CD4-positive T cells, CD20-expressing B cells, macrophages, and CD138-positive plasma cells. The CD20-positive cells did not display the typical B cell morphology, having in general a more dendritic cell-like appearance. The CD138-expressing plasma cells were distinctly localized as a dense infiltrate beneath the accumulation of T cells and B cells. Increased numbers of CD1a-expressing Langerhans cells were detected both in the epithelium and connective tissue. Because no granulomas were observed and only single lesional eosinophils were detected, GPTD does not resemble a granulomatous or eosinophilic condition. CONCLUSIONS: We describe for the first time the immunopathological characteristics of a novel inflammatory disorder of the oral cavity, which may develop after solid organ transplantation in children.

Amlodipine-induced gingival overgrowth in a child after liver transplant.

Drug-induced gingival overgrowth (GO) has been associated with phenytoin, cyclosporine, and calcium channel blocker therapies. This study reports the case of an 11-year-old girl who was referred for evaluation of GO, which had occurred over the last 6 months. Her medical history included a liver transplant due to biliary atresia 3 years ago, immunosuppressive therapy, and hypertension, which is why she was started on a daily intake of amlodipine. The intraoral examination showed generalized GO, and the treatment consisted of a gingivectomy. Subsequently, amlodipine was replaced with captopril and oral hygiene instructions. There was no recurrence of GO after 28 months of follow-up. Although GO may be related to the chronic use of amlodipine, such an association is uncommon in pediatrics, and the treatment consists of the replacement of medication combined with a surgical approach and plaque control.

Oral metastasis of metaplastic breast carcinoma in a patient with neurofibromatosis 1.

Neurofibromatosis type 1 (NF1) has been associated with an increased risk for development of malignancy, especially malignant peripheral nerve sheath tumors. In addition, recently, literature has demonstrated an increased risk of breast cancer in women with NF1. The present paper shows a 53-year-old woman with NF1 who presented with metaplastic breast carcinoma and developed multiple metastases, including mandible. Furthermore, we reviewed the English literature, found 63 cases showing the association between NF1 and breast cancer, and added one more case. The present study demonstrated an important association between NF1 and breast cancer. Until the present time, there has been only one case of metaplastic breast carcinoma associated with NF1. Curiously, in our case the oral metastasis corresponded to sarcomatous component of metaplastic breast carcinoma.

Clinical and histological features of multifocal canalicular adenomas of the upper lip.

Canalicular adenoma (CA) is an uncommon benign salivary gland tumor that mostly occurs in the upper lip. Despite its benign nature, some patients may present multiple nodules and also microscopic growth that can be multifocal with foci of tumor within the adjacent gland. Microscopically, CA can be mistaken for a polymorphous low-grade adenocarcinoma (PLGA) or basal cell adenoma (BCA), and immunohistochemistry can be helpful in differentiating them. Two cases of canalicular adenoma showing tumor foci within the adjacent gland are described. Both cases occurred in the upper lip of white females aged 72 and 79 years. In addition to the histological multifocal aspect, one of the patients presented bi-lateral nodules. Histological examination of both lesions revealed interconnecting and branching canal-like structures composed by uniform columnar and sometimes cuboidal cells, partially encapsulated. Immunohistochemical analysis showed intense expression of cytokeratins (CK) 7, a weak reaction to CK14 and a negative reaction to vimentin. Upon the diagnosis of CA the lesions were excised. In conclusion, it is important to properly diagnose CA and differentiate its multifocal aspect from a malignant behavior.

Clinical and Histological Features of Multifocal Canalicular Adenomas of the Upper Lip

Canalicular adenoma (CA) is an uncommon benign salivary gland tumor that mostly occurs in the upper lip. Despite its benign nature, some patients may present multiple nodules and also microscopic growth that can be multifocal with foci of tumor within the adjacent gland. Microscopically, CA can be mistaken for a polymorphous low-grade adenocarcinoma (PLGA) or basal cell adenoma (BCA), and immunohistochemistry can be helpful in differentiating them. Two cases of canalicular adenoma showing tumor foci within the adjacent gland are described. Both cases occurred in the upper lip of white females aged 72 and 79 years. In addition to the histological multifocal aspect, one of the patients presented bi-lateral nodules. Histological examination of both lesions revealed interconnecting and branching canal-like structures composed by uniform columnar and sometimes cuboidal cells, partially encapsulated. Immunohistochemical analysis showed intense expression of cytokeratins (CK) 7, a weak reaction to CK14 and a negative reaction to vimentin. Upon the diagnosis of CA the lesions were excised. In conclusion, it is important to properly diagnose CA and differentiate its multifocal aspect from a malignant behavior.

O adenoma canalicular (AC) é uma rara neoplasia benigna de glândula salivar que ocorre com maior frequência em lábio superior. Apesar de sua natureza benigna, alguns pacientes podem apresentar múltiplos nódulos, além de apresentar microscopicamente um crescimento multifocal com focos de neoplasia na glândula adjacente. Microscopicamente, AC pode ser confundido com adenocarcinoma polimorfo de baixo grau ou adenoma de células basais, sendo a imunohistoquímica útil na diferenciação entre eles. Dois casos de AC com focos de neoplasia dentro da glândula são descritos. Ambos os casos ocorreram em lábio superior de dois indivíduos do sexo feminino com 72 e 79 anos de idade. Além do aspecto histológico multifocal, um dos pacientes apresentou nódulos bilaterais. A análise histológica dos dois casos revelou estruturas canaliculares interligadas, compostas por células colunares uniformes e cuboidais, parcialmente encapsuladas. A análise imunohistoquímica mostrou intensa marcação de citoqueratina (CK) 7, uma fraca expressão para CK14, e reação negativa para vmentina. Após o diagnóstico as lesões foram excisionadas. Conclusão: É importante o correto diagnóstico do AC e a diferenciação do aspecto multifocal de um comportamento maligno.

Avaliação de manifestações bucais em pacientes pediátricos submetidos ao transplante hepático / Evaluation of oral manifestations in liver transplanted pediatric patients

Introdução. O transplante hepático se tornou a principal opção terapêutica para o tratamento de várias doenças hepáticas. Subsequentemente ao transplante, é necessária a administração de terapia imunossupressora para evitar rejeição ao órgão transplantado. A avaliação odontológica é fundamental para eliminação ou prevenção do surgimento de focos infecciosos. Além disso, faz-se necessário o acompanhamento dos efeitos colaterais em cavidade bucal relacionados ao uso de drogas imunossupressoras. Objetivo. Avaliar as condições odontológicas previamente ao transplante hepático e identificar as alterações bucais apresentadas após o transplante hepático. Pacientes e métodos. Foi realizado estudo retrospectivo de 265 pacientes pediátricos submetidos ao transplante hepático no Hospital A.C. Camargo, São Paulo-SP, entre janeiro de 2002 e dezembro de 2009. As informações clínicas como idade, gênero, diagnóstico da doença hepática, data do transplante, terapia imunossupressora (tipo, dose e duração), tratamento odontológico e a presença de alterações bucais pós-transplante foram coletadas dos prontuários médicos. Análise estatística foi realizada buscando estabelecer informações relevantes quanto aos riscos e possíveis fatores preditivos para o desenvolvimento de manifestações bucais. Resultados. A idade ao transplante hepático variou de 3,5 a 210,7 meses, tendo uma mediana de 15,5 meses. Dos 265 pacientes, 150 pacientes (56,6%) eram do gênero feminino e 165 (62,3%) eram leucodermas. Dentre as doenças de base, a atresia de vias biliares foi a mais frequente, acometendo 170 (64,1%) pacientes.

Um total de 73 pacientes foi avaliado pelo Departamento de Estomatologia previamente ao transplante, e destes, 34 (46,6%) apresentaram cárie. Quanto à presença de pigmentação dentária por bilirrubina, 172 pacientes foram avaliados e destes, 100 (58,1%) apresentaram pigmentação. Em relação à presença de hipoplasia do esmalte dentário, a alteração foi observada em 56 (34,4%) de 163 pacientes. Interessantemente, dos 100 pacientes com pigmentação dentária por bilirrubina, 97 apresentavam doenças colestáticas (p<0,001). Quanto aos casos de hipoplasia do esmalte, 52 (92,9%) pertenciam ao grupo de doenças colestáticas (p<0,001). Diversas alterações em mucosa bucal foram encontradas após o transplante, sendo que 135 pacientes apresentaram alguma complicação. Dos 265 pacientes estudados, o principal problema encontrado foi infecção pelo vírus herpes simples, em 48 pacientes, sendo que 66,7% dos casos ocorreram a partir de 12 meses após o transplante. A segunda doença infecciosa mais comum em cavidade oral foi a candidose, observada em 39 pacientes e 61,5% dos casos ocorreram durante o primeiro semestre pós-transplante. Importantes alterações bucais foram encontradas em pacientes em uso de tacrolimus, como a hipertrofia de papilas linguais, ressecamento labial, edema labial, fissuras labiais, fissuras linguais, queilite angular, mucosa com aspecto de pedra de calçamento e língua despapilada, observadas em 45, 39, 39, 38, 27, 11 e 6 pacientes, respectivamente. A hiperplasia gengival medicamentosa foi observada em 13 pacientes, sendo que 8 estavam em uso de ciclosporina e todos os outros usavam além do tacrolimus, drogas bloqueadoras de canais de cálcio. A complicação que ocorreu mais precocemente nos pacientes após o transplante foi a doença linfoproliferativa, em média 5,2 meses após o transplante. Conclusões. O índice de cáries foi elevado refletindo as precárias condições de saúde bucal desses pacientes. A pigmentação por bilirrubina.

Introduction. Liver transplantation has become the main therapeutic option for the treatment of various liver diseases. Subsequent to transplantation, it is necessary to administer immunosuppressive treatment to avoid rejection of the graft. A dental evaluation is critical to eliminate or prevent the emergence of infectious foci. Moreover, it is necessary to monitor the side effects in the oral cavity related to the use of immunosuppressive drugs. Objectives. To evaluate the dental conditions prior to liver transplantation and to identify oral abnormalities presented after liver transplantation. Pacients and methods. A retrospective study of 265 pediatric patients who underwent liver transplantation at Hospital A.C. Camargo, São Paulo-SP, between January 2002 and December 2009 was performed. Clinical information such as age, gender, diagnosis of liver disease, date of transplantation, immunosuppressive therapy (type, dose and duration), dental treatment and oral changes after transplantation were collected from medical records. Statistical analysis was performed in order to establish relevant information about the risks and possible predictive factors for the development of oral manifestations.