RESUMO
Aneurysmal bone cyst is a rare osteolytic lesion of uncertain etiology, commonly observed in the lower limbs, with only 1-2% of reports in gnathic bones. We present the case of a 27-year-old male patient referred to the oral and maxillofacial surgery and traumatology service due to complaints of paresthesia in the mental region and increased mandibular volume. Physical examination revealed midline shift and hard consistency. Imaging examinations demonstrated a radiolucent/hypodense lesion with disruption of the mandibular cortices. The histopathological examination of incisional biopsy material led to the diagnosis of a central giant cell lesion. The patient underwent surgical resection, and the histopathological analysis of the specimen revealed a predominantly solid lesion, characterized by blood-filled spaces of varying size, not covered by epithelium or endothelium, with the presence of spindle cells, multinucleated giant cells, and basophilic osteoid material, concluding the diagnosis of mixed-type aneurysmal bone cyst. Despite being uncommon, aneurysmal bone cysts should be considered in the differential diagnosis of volumetric increase in the gnathic bones of young patients.
Assuntos
Cistos Ósseos Aneurismáticos , Doenças Mandibulares , Humanos , Cistos Ósseos Aneurismáticos/patologia , Masculino , Adulto , Doenças Mandibulares/patologiaRESUMO
Presentamos un caso de quiste óseo aneurismático (QOA) de ubicación infrecuente y comportamiento agresivo en un paciente masculino de 28 años, en que la resección quirúrgica es controversial por el riesgo de iatrogenia y eventual recurrencia. El tratamiento con denosumab ha sido recientemente propuesto como una alternativa para el manejo de QOAs irresecables o recurrentes; sin embargo, la literatura disponible es escasa. Reportamos nuestra experiencia en un caso y analizamos la bibliografía disponible
We present a case of aneurysmal bone cyst (ABC) of infrequent location and aggressive behavior in a 28-year-old male patient, in which surgical resection is controversial due to the risk of iatrogenicity and eventual recurrence. Treatment with denosumab has been recently proposed as an alternative for the management of unresectable or recurrent ABCs; however, the available literature is sparse. We report our experience with one case and analyze the available literature
Assuntos
Humanos , Cistos Ósseos Aneurismáticos/tratamento farmacológico , Cistos Ósseos Aneurismáticos/diagnóstico por imagem , Conservadores da Densidade Óssea/uso terapêutico , Denosumab/uso terapêutico , Tomografia Computadorizada por Raios X/métodosRESUMO
Se presenta un caso clínico poco frecuente de un quiste óseo aneurismático localizado en el cuboides de un niño de 13 años. Se llegó al diagnóstico mediante la tríada de síntomas, estudios por imágenes (radiografía, resonancia magnética) y anatomía patológica. El tratamiento consistió en el abordaje y curetaje minuciosos dentro de la lesión, el fresado de alta velocidad, la electrocauterización y el relleno con aloinjerto óseo liofilizado con chips cortico-esponjosos. Pese al diagnóstico tardío, la evolución clínica fue favorable con remisión completa de los síntomas y retorno a las actividades deportivas, sin recidiva local. Nivel de Evidencia: IV
A rare clinical case of an aneurysmal bone cyst located at the level of the cuboid in a 13-year-old boy is presented. The diagnosis was reached through clinical evaluation, imaging studies (radiograph-MRI), and a pathological anatomy analysis. Its treatment consisted of thorough intralesional curettage, high speed burring, electrocautery, and filling with lyophilized bone allograft with cortical/cancellous chips. Despite the late diagnosis, we would like to highlight the favorable clinical evolution of the patient, with ad integrum remission of the symptoms and return to his sport activities, without elements of local recurrence. Level of Evidence: IV
Assuntos
Adolescente , Ossos do Tarso , Cistos Ósseos Aneurismáticos , Doenças do PéRESUMO
RESUMEN Los quistes óseos aneurismáticos son frecuentes en la edad pediátrica. Para su determinación se cuenta con diversos estudios imagenológicos como la radiografía y la tomografía axial computarizada que pueden colaborar al diagnóstico diferencial con otras lesiones. Existen disímiles opciones terapéuticas, el uso de factores de crecimiento autólogos se ha considerado como alternativa eficaz. Se presentan dos pacientes consultados en el servicio de Ortopedia del Hospital Provincial Pediátrico Universitario «José Luis Miranda¼, de Villa Clara, con diagnóstico clínico e imagenológico de quiste óseo aneurismático que recibieron tratamiento mediante terapia celular con células mononucleares con buena evolución clínica y radiográfica. Esta técnica es aplicable en nuestro medio ya que no requiere de estimables recursos materiales lo que constituye una fortaleza para su implementación. Las radiografías permiten reconocer la evolución posterior al tratamiento.
ABSTRACT Aneurysmal bone cysts are common in children. For its determination, various imaging studies are available, such as radiography and computerized axial tomography, which can collaborate in the differential diagnosis with other lesions. There are dissimilar therapeutic options, the use of autologous growth factors has been considered as an effective alternative. We present two patients seen in the Orthopedics service at "José Luis Miranda" University Pediatric Hospital in Villa Clara, with a clinical and imaging diagnosis of aneurysmal bone cyst who received treatment with mononuclear cell therapy having a good clinical and radiographic evolution. This technique is applicable in our environment since it does not require considerable material resources, which constitutes a strength for its implementation. X-rays allow us to recognize the evolution after treatment.
Assuntos
Cistos Ósseos Aneurismáticos/terapia , Leucócitos Mononucleares , Plasma Rico em PlaquetasRESUMO
OBJETIVO Dar a conocer nuestra experiencia en el diagnóstico diferencial de los quistes óseos simple y aneurismático mediante estudio radiológico simple. MATERIALES Y METODOS Se incluyeron pacientes menores de 20 años con radiografía de quiste óseo simple o aneurismático histológicamente confirmado pertenecientes al Registro Nacional de Tumores Óseos. Las radiografías fueron analizadas por dos radiólogos experimentados. Se compararon las variables demográficas de los pacientes, y las variables clínicas y radiológicas de ambos quistes. RESULTADOS Un total de 97 pacientes cumplieron los criterios de inclusión (65% presentaba quistes óseos simples y 35%, aneurismáticos). No se observaron diferencias en cuanto a la edad, al hueso comprometido, al tamaño de la lesión, a la expansión ósea, ni al adelgazamiento cortical. El género, la forma de presentación, la presencia de interrupción de la cortical, y la localización de la lesión en los ejes longitudinal y transversal del hueso son parámetros que podrían ser útiles en el diagnóstico diferencial de ambos quistes. DISCUSIÓN Los quistes óseos simple y aneurismático son lesiones benignas frecuentes que, de acuerdo con la literatura, serían difíciles de diferenciar únicamente con radiografía simple. La resonancia magnética permite una mejor caracterización anatómica, y aporta sensibilidad y especificidad al diagnóstico. Sin embargo, debe ser precedida por la radiografía simple, y su disponibilidad es limitada. CONCLUSIÓN Aun en centros con disponibilidad de resonancia magnética, la radiografía simple sigue siendo el estudio inicial de elección en el diagnóstico de tumores óseos. Ciertas características demográficas y radiográficas permiten orientar el diagnóstico diferencial inicial entre el quiste óseo simple y el aneurismático
OBJETIVE To describe our experience in the differential diagnosis of unicameral and aneurysmal bone cysts using plain radiography. PATIENCES AND METHODS We included patients under 20 years of age with radiographs of histologically-confirmed unicameral or aneurysmal bone cysts found on the Chilean National Bone-Tumor Registry. The radiographs were evaluated by two experienced radiologists. We compared the demographic variables of the patients, and the clinical and radiological variables of both tumors.. RESULTADOS A total of 97 patients met the inclusion criteria, 65% of whom had simple bone cysts, and 35%, aneurysmal bone cysts. No differences were found regarding age, the bone affected, the size of the lesion, bone expansion, nor cortical thinning. Gender, clinical presentation, cortical interruption, and location of the lesion on the longitudinal and transverse bone axes are parameters that could be used in the differential diagnosis of both cysts. DISCUSSION According to the literature, unicameral and aneurysmal bone cysts are frequent benign lesions that are difficult to differentiate merely through plain radiographs. Magnetic resonance imaging enables a better anatomical characterization and provides sensitivity and specificity to the diagnosis. However, its availability is limited, and it should be preceded by plain radiography. CONCLUSION Plain radiography is still the initial imaging study of choice in patients with clinical suspicion of bone tumor, even in those centers where magnetic resonance imaging is available. Certain demographic and radiological characteristics guide physicians in the differential diagnosis of unicameral and aneurysmal bone cysts.
Assuntos
Humanos , Masculino , Feminino , Criança , Adolescente , Adulto , Cistos Ósseos/diagnóstico por imagem , Cistos Ósseos Aneurismáticos/diagnóstico por imagem , Radiografia/métodosRESUMO
INTRODUCCIÓN Los quistes óseos aneurismáticos (QOAs) son tumores benignos, localmente agresivos, y con importante potencial de recidiva, que representan aproximadamente el 1% de todos los tumores óseos. Se describen múltiples tratamientos, como: escisión intralesional, embolización arterial selectiva, inyección de agentes esclerosantes, y radiación. Estos tratamientos tienen una tasa variable de eficacia, ya que la recurrencia puede llegar al 20% y puede estar asociada a comorbidades graves como la pérda funcional de la extremidad. OBJETIVO Realizar una revisión integradora de la literatura sobre el uso de denosumab para el tratamiento de QOAs, describiendo el perfil epidemiológico, la dosis utilizada, y las complicaciones. MÉTODO Se recopilaron artículos publicados en los últimos cinco años en la base de datos PubMed. La información recogida de los casos reportados fue la edad, el sexo, la ubicación del tumor, la realización de cirugía antes y/o después del tratamiento con denosumab, la dosis utilizada, las complicaciones, y la recurrencia. RESULTADOS Se analizaron 7 artículos, 4 reportes de casos y 3 series de casos, escritos en inglés, y publicados de 2014 a 2019. La mayoría de los pacientes eran del sexo femenino, con una edad promedio de 14 años, y el tumor localizado en la columna. CONCLUSIÓN El uso de denosumab en el tratamiento de QOAs ha tenido una buena respuesta, ya que tiene bajas tasas de recurrencia y complicaciones; sin embargo, hacen falta más estudios para definir el protocolo de tratamiento.
INTRODUCTION Aneurysmal Bone Cysts (ABCs) are locally-aggressive benign tumors with relevant potential for recurrence, representing approximately 1% of all bone tumors. Multiple treatments are described for them, such as: intralesional excision, selective arterial embolization, injection of sclerosing agents, and radiation. These treatments have a variable efficacy rate, can reach 20% and may be associated with serious comorbidities such as functional loss of the limb. OBJETIVE To perform an integrative review of the literature on the use of denosumab in the treatment of ABCs, describing the epidemiological profile, the dosage used, and the complications. METHODOLOGY Articles published in the past five years were retrieved from the PubMed database. The information collected from the cases reported was age, gender, tumor location, the performance of surgery before and/or after the denosumab treatment, the dose used, the complications, and recurrence. RESULTS We analyzed 7 articles, 4 case reports and 3 case series, written in English, and published from 2014 to 2019. Most patients were female, with an average age of 14 years, with the tumor located in the spine. CONCLUSION The use of denosumab in the treatment of ABCs yielded good results, with low rates of recurrence and complications. However, further studies are needed to define a treatment protocol.
Assuntos
Humanos , Masculino , Feminino , Pré-Escolar , Criança , Adolescente , Adulto , Cistos Ósseos Aneurismáticos/tratamento farmacológico , Conservadores da Densidade Óssea/uso terapêutico , Denosumab/uso terapêuticoRESUMO
Introducción: El quiste óseo aneurismático es una lesión seudotumoral lítica, expansiva, compuesta por espacios llenos de sangre separados por tabiques. Representa el 15% de los tumores primarios en la columna, y su presentación de comportamiento agresivo es aún más infrecuente. Se han descrito diferentes opciones terapéuticas en función de su estadio y tasa de recurrencia. Presentamos a un paciente con compromiso neurológico secundario a un quiste óseo aneurismático en la columna torácica, de comportamiento agresivo, que requirió resolución quirúrgica. Conclusión: Las opciones de tratamiento del quiste óseo aneurismático se deben adecuar a cada caso en particular, según sus características. Nivel de Evidencia: IV
Introduction: An aneurysmal bone cyst is an expansive, lytic, pseudotumoral lesion that consists of blood-filled spaces separated by septa. It represents 15% of primary spinal tumors. An aggressive presentation is even rarer. Different therapeutic options are described depending on its stage and recurrence rate. We present the case of a patient with neurological involvement due to the aggressive behavior of an aneurysmal bone cyst in the thoracic spine, which required surgical treatment. Conclusion: Treatment options for aneurysmal bone cysts must adapt to each case, depending on its characteristics. Level of Evidence:IV
Assuntos
Adulto , Neoplasias da Coluna Vertebral , Vértebras Torácicas , Cistos Ósseos AneurismáticosRESUMO
PURPOSE: To systematically review the literature to determine recurrence rates of percutaneous treatments for primary aneurysmal bone cysts (ABC). METHODS: Search strategies were performed in the following databases: PubMed, SCIELO, LILACS and BVS, using terms in English, Spanish and Portuguese (PROSPERO Registration Number: CRD42020170340). Longitudinal studies, either observational or clinical trials, with at least five patients and with a mean of 18 months of follow-up were included. Studies had to use any type of percutaneous treatments and report the recurrence rates of primary ABC treatment. Studies selection, data extraction and risk of bias assessment were performed independently by two researchers. A global meta-analysis was carried out to assess the proportion of recurrence. Studies were categorized into two subgroups: selective arterial embolization and sclerotherapy. RESULTS: Thirteen studies were included in the present study. The average success rate of percutaneous treatments for ABC was 91.11%, with a total of 37 lesions recurrences in the 416 patients. The sex ratio was 1:1. The subgroup of sclerotherapies presented a lower proportion of recurrence. The proportion of recurrence in the subgroup of selective arterial embolization was 19% (95%IC 12.11-27.54) and that of sclerotherapies was 6% (95%IC 3.65-9.19). CONCLUSIONS: Both percutaneous treatments for ABC are effective, showing a lower rate of recurrence. Sclerotherapy treatments seem to be promising, but further clinical trials must be conducted with a longer follow-up.
Assuntos
Cistos Ósseos Aneurismáticos , Embolização Terapêutica , Cistos Ósseos Aneurismáticos/diagnóstico por imagem , Cistos Ósseos Aneurismáticos/terapia , Humanos , Recidiva Local de Neoplasia , Escleroterapia , Resultado do TratamentoRESUMO
INTRODUCTION: The aneurysmal bone cyst is a rare benign but aggressive osteolytic tumor for which there is still no ideal treatment, the reports on treatment by sclerotherapy in the pediatric population are scarce and in our region even less. The objective is to communicate the experience of the treatment of aneurysmal bone cyst with polydocanol 3%. MATERIAL Y METHODS: Retrospective, descriptive and cross-sectional study. Period: June/2017 to June/2021. Inclusion: patients with histological diagnosis of aneurysmal bone cyst; Under general anesthesia and fluoroscopic guidance, intralesional puncture with 16G needle was performed through which 3% polydocanol was slowly administered. Data: medical history. Quantitative variables shall be expressed in measures of central tendency and dispersion; qualitative variables shall be expressed as frequencies or percentages. RESULTS: Nine consecutive patients were included, all of whom had pain and tumor in one of the extremities. Gender: 3 female and 6 male. Age: median 10.5 years (range: 2-15.1). Weight: median 32.8 kg (range: 11-44.5). Total procedures: 44; procedures per patient: mean 4.9 (SD: ± 2.0). Procedure time: mean 33.9 minutes (SD: ± 18.3); radiation: mean 1.34 mGy (SD: ± 1.55). Hospitalization: one day, except one patient. Complications: skin damage in one case, no recurrences follow-up: 12 to 50 months. CONCLUSION: In this pediatric series, polydocanol 3% was useful and effective for the treatment of aneurysmal bone cyst, with few complications. One disadvantage is that it requires several sessions and in addition, no significant difference has been demonstrated between other forms of treatment in terms of the recurrence rate.
INTRODUCCIÓN: El quiste óseo aneurismático es un raro tumor osteolítico benigno, pero agresivo para el que aún no existe tratamiento ideal. Los comunicados sobre tratamiento mediante escleroterapia en población pediátrica son escasos y en nuestra región más aún. El objetivo es comunicar la experiencia del tratamiento del quiste óseo aneurismático con polidocanol 3%. MATERIAL Y MÉTODOS: Estudio retrospectivo, descriptivo y transversal. Período: Junio de 2017 a Junio de 2021. Inclusión: pacientes con diagnóstico histológico de quiste óseo aneurismático. Bajo anestesia general y guía fluoroscópica se realizó punción intralesional con aguja 16G a través de la cual se administró lentamente el polidocanol 3%. Datos: historia clínica. Las variables cuantitativas se expresarán en medidas de tendencia central y de dispersión; las variables cualitativas se expresarán como frecuencias o porcentajes. RESULTADOS: Se incluyeron nueve pacientes consecutivos, todos presentaban dolor y tumoración en alguna de las extremidades. Género: tres femeninos y seis masculinos. Edad: mediana 10.5 años (rango: 2-15.1). Peso: mediana 32.8 kg (rango: 11-44.5). Total de procedimientos: 44, procedimientos por paciente: promedio 4.9 (DE: ± 2.0). Tiempo de procedimiento: promedio 33.9 minutos (DE: ± 18.3); radiación: promedio 1.34 mGy (DE: ± 1.55). Hospitalización: un día, excepto un paciente. Complicaciones: Daño de piel en un caso, no recidivas. Seguimiento: de 12 a 50 meses. CONCLUSIÓN: En esta serie pediátrica el polidocanol 3% fue útil y efectivo para el tratamiento del quiste óseo aneurismático con escasas complicaciones. Una desventaja es que requiere varias sesiones y además, no se ha demostrado una diferencia significativa entre otras formas de tratamiento en términos de la tasa de recurrencia.
Assuntos
Cistos Ósseos Aneurismáticos , Adolescente , Cistos Ósseos Aneurismáticos/diagnóstico , Cistos Ósseos Aneurismáticos/terapia , Criança , Pré-Escolar , Estudos Transversais , Feminino , Humanos , Masculino , Uso Off-Label , Polidocanol/uso terapêutico , Estudos RetrospectivosRESUMO
STUDY DESIGN: Case report. OBJECTIVE: To report the clinical and imaging findings of a patient with the extremely rare association of aneurysmal bone cyst and osteoblastoma in the cervical spine. To our knowledge, only three cases have been reported in the published literature in children under 16 years of age with this condition in the cervical spine. METHODS: The patient's history, physical examination, imaging findings, and management with a complete 4-year medical history, surgical intervention and radiological follow-up are reported. RESULTS: A 4-year 11-month-old boy was diagnosed with aneurysmal bone cyst in association of osteoblastoma and was treated with CT-guided intralesional injection calcitonin and methylprednisolone. During the course of intralesional therapy, a pathological fracture of C2 was produced. Subsequently, a widened intralesional excision and instrumented fusion from occiput to cervical spine (C0-C4) was performed. CONCLUSION: The association of aneurysmal bone cyst and osteoblastoma in spine is extremely rare. Although both are benign lesions, in the cervical location, complete removal of the tumors is challenging. Wide resection with reconstruction of the segments for stability associated with adjuvant treatment with calcitonin and corticosteroids provides a good option.