OPEN ACCESS SCIENTIFIC JOURNAL VOLUME 20 N0 02 - 2017 Ciência Odontológica Brasileira Benign fibrous histiocytoma of the tongue: a case report Ciência Odontológica Brasileira UNIVERSIDADE ESTADUAL PAULISTA “JÚLIO DE MESQUITA FILHO” Instituto de Ciência e Tecnologia Campus de São José dos Campos UNIVERSIDADE ESTADUAL PAULISTA “JÚLIO DE MESQUITA FILHO” Instituto de Ciência e Tecnologia Campus de São José dos Campos CASE REPORT doi: 10.14295/bds.2017.v20i2.1302 Benign fibrous histiocytoma of the tongue: a case report Histiocitoma fibroso benigno em língua: relato de caso Michele Fonseca BRANTES1, Rebeca de Souza AZEVEDO1, Silvia Paula de OLIVEIRA2, Adriele Ferreira GOUVÊA1, Ademar TAKAHAMA JR3 1 - Departamento de Formação Específica - Universidade Federal Fluminense (UFF) - Nova Friburgo - Rio de Janeiro – RJ - Brazil. 2 - Odontoclínica Central do Exército (OCEx) - Rio de Janeiro – RJ - Brazil. 3 - Departamento de Medicina Oral e Odontologia Infantil - Universidade Estadual de Londrina (UEL) – Londrina - PR - Brazil. ABSTRACT RESUMO The fibrous histiocytoma is a soft tissue neoplasm O Histiocitoma Fibroso é uma neoplasia de partes moles that affects the dermis and the subcutaneous que acomete a derme e o tecido subcutâneo, raramente tissue, rarely is found in the oral cavity and perioral é encontrado na cavidade oral e regiões periorais, e regions, and is originated from the proliferation tem origem a partir da proliferação de fibroblastos ou of fibroblasts and histiocytes. The objective of histiócitos. O objetivo deste artigo é relatar um caso de this paper is to report a case of Benign Fibrous Histiocitoma Fibroso Benigno em um paciente masculino, Histiocytoma in a 30-year-old male patient, 30 anos de idade, com um nódulo indolor, bem delimitado, complaining of a painless nodule in the tongue com duração de cerca de seis meses, localizado no dorso for about six months. With diagnostic clinical anterior da língua. Com as hipóteses clínicas diagnósticas hypotheses of Fibrous Hyperplasia, Neurofibroma, de Hiperplasia Fibrosa, Neurofibroma, Neuroma Traumatic Neuroma, Fibrous Histiocytoma, Traumático, Histiocitoma Fibroso, Tumor de Células Granular Cell Tumor or Ectomesenchymal Granulares e Tumor Condromixoide Ectomesenquimal Chondromyxoid Tumor a excisional biopsy was uma biópsia foi realizada sob anestesia local e a lesão performed. The histopathological examination foi fixada em formol a 10% e enviada para análise revealed a non-encapsulated proliferation of histopatológica. O exame histopatológico revelou uma spindle cells with some giant multinucleated cells in proliferação não-encapsulada de células fusiformes com the periphery of the lesion. Immunohistochemical algumas células gigantes multinucleadas na periferia reactions were performed, staining only for da lesão. A marcação imunohistoquímica foi positiva vimentin in the spindle cells and for CD68 in the para CD68 nas células gigantes multinucleadas e para multinucleated giant cells. According to these vimentina nas células fusiformes. O diagnóstico final foi characteristics, the final diagnosis was Benign de Histiocitoma Fibroso Benigno. Para um diagnóstico Fibrous Histiocytoma. The correct diagnosis of correto, este deve ser feito correlacionando características spindle shaped cell neoplasia must be performed clínicas, análise histopatológica e imunohistoquímica with the aid of histopathological analysis and devido à similaridade microscópica do Histiocitoma immunohistochemistry, mainly because the Fibroso com outras lesões com aspecto fusocelular, assim morphological similarities with other benign and como similaridade clínica com outras lesões benignas e malignant lesions. malignas. KEYWORDS PALAVRAS-CHAVE Fibroblasts; Benign fibrous histiocytoma; Fibroblastos; Histiocitoma fibroso benigno; Histiocitoma Malignant fibrous histiocytoma; Histiocytes, fibroso maligno; Histiócitos; Lesões em língua. Tongue diseases. 152 Braz Dent Sci 2017 Apr/Jun;20(2) Brantes MF et al. Benign fibrous histiocytoma of the tongue: a case report LITERATURE REVIEW CASE REPORT Male patient, 30 years of age, was referred enign fibrous histiocytoma (BFH) is a rare for evaluation of a nodule in the tongue for Bneoplasm of soft tissue, which affects the the last six months. During the anamnesis dermis and subcutaneous tissue, has origin he did not report any symptoms such pain or as from the fibroblasts and histiocytes. [1] difficulty to speak. The physical examination It was first described by Stout and Lattes revealed a small, asymptomatic and sessile in 1967. [2] Before 1960, the BFH was not white nodule in the anterior dorsum of the considered a distinct clinic-pathological tongue (Figure 1). The diagnostic hyphoteses entity. But, with electronic microscopy and was fibrous Hyperplasia, Neurofibroma, immunohistochemistry advancement, it was Traumatic Neuroma, Fibrous Histiocytoma, possible to differentiate this diagnosis. [3] Per Granular Cell Tumor and Ectomesenchymal present a variable nature this type of neoplasm Chondromyxoid Tumor. An excisional biopsy has many synonyms as: Nodular Fibrosis was performed under local anesthesia and the lesion was fixed on formaldehyde 10% and sent Subepidermic, Sclerosing Hemangioma and for histopathological analysis. Simple Fibroid when it involves the skin. [4] The histopathological examination The BFH occurs most frequently on the skin revealed a non-encapsulated proliferation surface of the extremities, however can develop of spindle-shaped cells somewhat disposed in deeper tissues, in tissue of the oral mucosa in a storiform pattern. In addition, some or even in bone. [5] This neoplasm must be giant multinucleated cells occupied the differentiated of Malignant Fibrous Histiocytoma periphery of the lesion (Figure 2 A,B,C and that often presents a more aggressive course, can D). Immunohistochemistry reactions were present recurrences and metastases. [6] performed for cytokeratin AE1/AE3, vimentin, Little cases of HFB occurs in the oral S100, smooth muscle actin, desmin and cavity and perioral regions. The intraoral sites CD68, being positive only for CD68 in the most commonly of involved soft tissue are the giant multinucleated cells and for vimentin gums, lips lower and upper, soft palate, floor in the spindle-shaped cells (Figure 3 A and of the mouth and tongue being one of the least B). According to clinical, histopathological reported locations. [4] and immunohistochemical analysis, it was established a final diagnosis of Benign Fibrous This type of neoplasm is reported at any Histiocytoma, once there was not any sign of age with predominance during the third and malignant changes. In a follow up of 2 years, no fourth decades of life. Its distribution by gender evidence of recurrence was observed. varies between different populations. The clinical features of oral HFB are a painless solitary tumor, slow growth, 2-3 cm to over 10 cm, along a period of several months. [7] The symptoms are specifically caused by interference with the normal anatomy and physiology of the area in which it appears, when the mass is located on the tongue difficulty speaking may be present, for Figure 1 - Clinical Aspects: intraoral exam revealing a white example. [3,8] nodule in the anterior dorsum of the tongue. 153 Braz Dent Sci 2017 Apr/Jun;20(2) Brantes MF et al. Benign fibrous histiocytoma of the tongue: a case report Figure 2 - Histopathological characteristics: A- General aspect in larger magnification (HE – 40 x); B- Subepithelial proliferation of spindle cells (HE – 100x); C- Presence of giant multinucleated cells on the periphery of the lesion (HE – 400 x); D- Spindle cells in a storiform arrangement (HE – 400 x). Figure 3 - Immunohistochemical Aspects: A- Positive staining for vimentin in spindle cells (Immunoperoxidase, 200 x); B- Positive staining for CD68 in multinucleated giant cells (Immunoperoxidase, 400 x). 154 Braz Dent Sci 2017 Apr/Jun;20(2) Brantes MF et al. Benign fibrous histiocytoma of the tongue: a case report DISCUSSION spindle cells similar to fibroblasts and also histiocytes. The findings of Pandey et al. [13] are The etiology of Fibrous Histiocytoma is equivalent to this study, wherein was described not well established, is questionable whether a case of HFB in to tongue what microscopically it is a reactive proliferation to an inflammatory showed spindle cells and histiocytes. Jo et al. stimulus or neoplastic process. [4] Some cases [10] described the same pattern storiform are reported after local injuries, such as trauma, insect bites or folliculitis, strengthening the of spindle cells in a buccal mucosa lesion. conception of inflammatory origin. MacLeod Even intra osseous lesions in the mandible, and Jones [9] reported a case of oral HFB in lip described by De-ming et al. [14] and Shoor et than occurred after continuous trauma to bite. al. [15] revealed spindle cells and giant cells In opposition, some examples support the idea with positive staining for vimentin and CD68 clonal expansion, strengthening the conception respectively. Immunohistochemistry for CD68 of neoplastic origin. [4] can be used in these cases because it acts by marking the various cells of the macrophage Due to uncertainty about the natural lineage including monocytes, histiocytes, history of fibrohistiocytic lesions, the HFB was giant cells and osteoclasts. [15] Lesions such not identified as a distinct clinical entity until as Nodular Fasciitis, Fibrous Tumor Solitaire, years 1960. [3] Still today, this type of lesion Neurofibroma and Dermatofibroma are the often