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      Histiocitosis de células de Langerhans localizada en hueso malar: Presentación de un caso Translated title: Localized langerhans cell histiocytosis of the zygomatic bone: A case report

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          Abstract

          La histiocitosis de células de Langerhans localizada (HCLL), conocida como granuloma eosinófilo, representa entre el 50 y el 60% de todos los casos de histiocitosis de células de Langerhans. El tratamiento clásico para la HCLL ha sido el curetaje o la resección de las lesiones óseas. Hay publicaciones de casos tratados con inyección intralesional de corticosteroides, combinado con curetaje. Se presenta un caso clínico de un paciente de tres años de edad con diagnóstico de HCLL que compromete en su extensión el hueso malar, tratado con infiltraciones de corticosteroides y posterior curetaje de la lesión. A un año de realizado el tratamiento, el paciente se encuentra asintomático y con una regeneración ósea del hueso malar, evidenciable en la tomografía axial computarizada.

          Translated abstract

          Localized Langerhans cell histiocytosis (LLCH), also known as eosinophilic granuloma, represents 50 to 60% of all cases of Langerhans cell histiocytosis. The standard treatment for LLCH has been lesion curettage or resection. Cases treated with intralesional corticosteroid injections combined with curettage have been described. We report the case of a three-year-old patient diagnosed of LLCH with extensive zygomatic bone involvement, who was treated with corticosteroid infiltrations and subsequent curettage of the lesion. One year after treatment, the patient is asymptomatic with zygomatic reossification evidenced on computed tomography.

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          Most cited references 20

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          Percutaneous techniques for the diagnosis and treatment of localized Langerhans-cell histiocytosis (eosinophilic granuloma of bone).

          We retrospectively studied the outcome of percutaneous needle biopsy and intralesional injection of a corticosteroid (methylprednisolone) in thirty-nine patients who had localized Langerhans-cell histiocytosis (eosinophilic granuloma of bone). All thirty-nine patients had a solitary symptomatic lesion at presentation; a second lesion developed in two patients, two and four months after the first lesion was diagnosed. Therefore, there were forty-one lesions in thirty-nine patients. Fine-needle aspiration with or without core-needle biopsy was performed for all forty-one lesions, and the diagnosis of Langerhans-cell histiocytosis was established for thirty-seven (90 per cent). A corticosteroid was injected into thirty-five lesions. Twenty-nine received the injection at the time of the fine-needle aspiration on the basis of the cytological findings in the aspirate. Six patients who had a solitary lesion had a two-stage procedure because the injection was delayed until the diagnosis was confirmed with histological evaluation of specimens obtained by core-needle biopsy. Thirty-four (97 per cent) of the thirty-five lesions healed. The clinical symptoms associated with thirty-one lesions resolved within two weeks after a single injection of the corticosteroid. There were no complications associated with either the biopsy or the injection. At a median of ninety months (range, twenty-four to 199 months), no patient had recurrence of symptoms or of radiographic evidence of the lesion. All patients who had been managed with an intralesional injection of the corticosteroid had full range of motion of the affected extremity and had resumed unlimited activities. Although the mechanism of action of intralesional injection of a corticosteroid has not been defined, use of percutaneous needle biopsy to diagnose localized Langerhans-cell histiocytosis and treatment with intralesional administration of methylprednisolone relieved pain expeditiously, enabled the patient to avoid an operative procedure, and resulted in osseous healing. The specific role of corticosteroid therapy remains to be determined by prospective, randomized studies.
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            Histiocytosis X: a review of 114 cases with oral involvement.

             K Hartman (1979)
            A review of 1,120 cases of histiocytosis X yielded 114 cases with oral involvement. Seventy-eight percent of the oral lesions in this series were categorized as monostotic or polyostotic eosinophilic granuloma. The great majority of cases occurred in males, with 40 percent of the lesions having developed before the patient had reached the age of 10 years. The mandible was involved in 73 percent of the cases, and the posterior jaw region was the predominant site. Extraoral involvement occurred in 70 percent, with the most common sites being the skull and lower extremity. The preferred treatment for oral lesions was surgical curettage, and the prognosis was generally very favorable, as evidenced by the over-all recurrence rate of 16 percent.
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              Intralesional infiltration of corticosteroids in the treatment of localised langerhans' cell histiocytosis of the mandible Report of known cases and three new cases.

              Langerhans' cell histiocytosis (LCH) can involve virtually any site and organ of the body, either as an isolated lesion or as a widespread systemic disease. Bone can be involved as a part of generalised disease or as a separate entity, formerly known as eosinophilic granuloma. The clinical cause of localised LCH (LLCH) to bone is generally benign. Solitary bone lesions may resolve spontaneously after diagnostic biopsy in a period of months to years. If treatment is indicated, as in larger lesions with pain, swelling and risk for spontaneous fracture, the disease can be controlled by chemotherapy or local measures such as surgical curettage, low-dose radiotherapy and intralesional injection of corticosteroids. There are no controlled studies in the literature comparing outcomes of these treatment modalities because of low incidence of the disease and general tendency to regress. Three patients (female, 28 months; male, 9 years; male, 15 years) with LLCH of the mandible were treated in an one stage procedure with intralesional injection of 80, 40 and 80 mg methylpredisolone succinate, respectively, as the primary form of treatment. Patients were seen for clinical and radiological evolution 1, 3, 6, 9 and 12 months after treatment, and yearly thereafter. The overall outcome was excellent. The lesions showed clinically and radiologically complete remission approximately 6 months after treatment. There were no complications nor morbidity of the treatment. After a follow-up period of 12 months, the patients are well with no evidence of residual disease on radiographs. Local injection of corticosteroids should be the initial choice of treatment of LLCH of the mandible, because of the favourable treatment outcome in this disease and possible complications and lasting effects of surgery, radiotherapy and chemotherapy.
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                Author and article information

                Contributors
                Role: ND
                Role: ND
                Role: ND
                Role: ND
                Journal
                maxi
                Revista Española de Cirugía Oral y Maxilofacial
                Rev Esp Cirug Oral y Maxilofac
                Sociedad Española de Cirugía Oral y Maxilofacial (Barcelona, Barcelona, Spain )
                1130-0558
                2173-9161
                September 2011
                : 33
                : 3
                : 114-119
                Affiliations
                Santiago de Chile orgnameHospital Luis Calvo Mackenna Chile
                Santiago de Chile orgnameUniversidad Mayor orgdiv1Facultad de Odontología Chile
                Article
                S1130-05582011000300004
                10.4321/S1130-05582011000300004

                This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.

                Page count
                Figures: 0, Tables: 0, Equations: 0, References: 13, Pages: 6
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                Product Information: SciELO Spain

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