Quiste óseo aneurismático del atlas: Tratamiento con denosumab y reporte de caso / Aneurysmal bone cyst of the atlas: treatment with denosumab and case report

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

Estudio radiológico simple de quiste óseo simple y aneurismático: diagnóstico diferencial / Plain radiologic assessment of unicameral and aneurysmal bone cyst: differential diagnosis

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.

Percutaneous treatments of primary aneurysmal bone cysts: systematic review and meta-analysis.

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.

Aneurysmal bone cyst and osteoblastoma: an extremely rare combination in the pediatric spine.

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.

Aneurysmal Bone Cyst of the Skull Base ­ Case Report

Introduction Aneurysmal bone cysts (ABCs) are pseudotumoral bone lesions of unknown etiology that are also hypervascularized, benign, and locally destructive. They are rare in the base of the skull. The present case report describes a case of aneurysmal bone cyst in the sella turcica. Case Report The present study was developed at the department of neurosurgery of the Hospital Universitário Professor Alberto Antunes of the Universidade Federal de Alagoas (HUPAA-AL, in the Portuguese acronym), Maceió, state of Alagoas, Brazil, and is accompanied by a review of the literature from the PubMed database. A 17-year-old female patient with bitemporal hemianopia and intense left hemicranial headache associated with symptoms from the cranial nerves contained in the cavernous sinus. Neuroimaging evidenced a large lesion in the suprasellar region with calcification foci, sellar erosion, and extension to the cavernous sinus. The patient was submitted to a partial lesion resection and the histopathological analysis showed an aneurysmal bone cyst. Conclusion A rare case of intracranial aneurysmal bone cyst, with the important differential diagnosis from pituitary adenoma.

Quiste óseo aneurismático mandibular / Aneurysmal mandibular bone cyst

El quiste óseo aneurismático es definido como una lesión osteolítica expansiva que consiste en espacios llenos de sangre y canales divididos por tabiques de tejido conectivo, los cuales contienen tejido osteoide y células gigantes multinucleadas. El objetivo es presentar un caso clínico poco común de un quiste óseo aneurismático de la región del cuerpo mandibular. Se trata de una paciente femenina de 39 años de edad que acudió a consulta externa del Servicio de Cirugía Maxilofacial del Hospital Universitario General Calixto García por aumento de volumen en región mandibular derecha y dolor intenso de 1 mes de evolución. Radiográficamente se detectó un área radiolúcida unilocular de bordes bien definidos; se realizó curetaje de la cavidad, y estudio histopatológico de la lesión que informó la presencia de un quiste óseo aneurismático. Se concluye que el quiste óseo aneurismático es más común en los huesos largos y en la región del ángulo mandibular en el esqueleto facial, por lo que la presentación de este en el cuerpo mandibular resulta de interés(AU)

The aneurysmal bone cyst is defined as an expansive osteolytic lesion consisting of blood-filled spaces and divided by partitions of connective tissue, which contain bone tissue and giant cells channels. The objective is to present a rare case of an aneurysmal bone cyst in the region of the mandibular body. This is a case of a 39-year-old female who attended the outpatient Maxillofacial Surgery Service of General Calixto Garcia University Hospital due to an increased volume in right mandibular region and intense pain of a month, both in evolution. Radiographically, a unilocular radiolucent area with well-defined edges was detected; curettage of the cavity and histopathological examination of the lesion were performed, which reported the presence of an aneurysmal bone cyst. It is concluded that the aneurysmal bone cyst is most commonly presented in the long bones and in the region of the mandibular angle in the facial skeleton, so the presentation of this in the mandibular body is of interest(AU)

Destructive aneurysmal bone cyst of the mandible.

Aneurysmal bone cyst (ABC) is an uncommon osteolytic lesion of the bones, usually affecting the long bones and spine. The lesion is rare in the jaws and is found most commonly in the body and ramus of the mandible. In some cases, ABCs may be present as rdestructive lesions simulating malignancies. In these cases, a careful diagnosis should be done, including in the differential diagnosis malignant tumors such as telangiectasic osteosarcoma and intraosseous fibrosarcoma. Removing the lesion is usually easy if it is confined within the bone, but it may prove difficult if the lesions are multilocular, expansive, divided by multiple bony septa, or destructive or when the cortical is perforated. Therefore, the surgical treatment of the destructive ABCs should be more radical.

Quiste óseo aneurismático / Aneurysmal bone cyst

El quiste óseo aneurismático (QOA) es una lesión benigna rara en huesos maxilares, su mayor frecuencia es en huesos largos del esqueleto y columna vertebral. Algunos autores especulan que el origen sea por trauma, mala formación o un neoplasma. El aspecto radiográfico puede mostrar desde una imagen difusa hasta una imagen bien definida, muy semejante a las otras lesiones de los maxilares volviendo el diagnóstico diferencial amplio. Con la destrucción del córtex óseo puede todavía formar una imagen reaccional del periostio en forma de rayos de sol, característico de una lesión maligna. En este estudio, se presenta caso de paciente femenino, 17 años de edad, que buscó el Servicio de Cirugía y Traumatología Buco maxilofacial del Hospital São Vicente de Paulo-RS, que presentó dolor y aumento de volumen en la mandíbula. Informó que hace cerca de un año empezó a sentir malestar en la región junto al ángulo mandibular derecho, lo cual fue progresivamente aumentando de volumen. El aspecto radiográfico acordaba una lesión maligna. Después de la confirmación histopatológica de QOA, el tratamiento seleccionado para el caso fue quirúrgico conservador, con legrado de la cavidad ósea, y fue mantenido en control posoperatorio de rutina con 6 y 18 meses. En las radiografías panorámicas realizadas se verifica la neoformación ósea y cura del caso(AU)

The aneurysmal bone cyst (ABC) is a benign and rare lesion in maxillary bones, is more frequent in largo bones of skeletal and spinal column. Some authors speculate about that its origin be traumatic, malformation of neoplasm. The radiographic appearance may to show from a diffuse image up to a well defined image very similar to the other maxillary lesions turning wide the differential diagnosis. With destruction of the bon cortex it is possible to create a reaction image of periosteum in sunbeams characteristic of malignant lesion. This is the case of a female patient aged 17 seeking help in the Bucco-maxillofacial Surgery and Traumatology Service of the São Vicente de Paulo Hospital _ RS, presenting with pain and an increased mandibular volume. She manifested that around one year she began to feel a discomfort next to he right mandibular angle, which was progressively increasing the volume. The radiographic appearance resembled a malignant lesion. After the histopathology verification of ABC, treatment choosed for this case was of type conservative surgical with bone cavity curettage and was maintained under systemic postoperative control for 6 and 18 months. In the taken panoramic radiographies is verified the bone neoformation and the cure of case(AU)

Clinical pathological analysis of nine cases of aneurysmal bone cyst of the jaws in a Brazilian population.

Aneurysmal bone cysts (ABCs) are benign osteolytic lesions that occur rarely in the jaws. The aim of this study was to investigate the clinical, radiographical and pathological features of ABCs of the jaws. A retrospective analysis of the content of a 39-year database, including nine cases of ABCs of the jaws diagnosed from the archives of the Oral Pathology Service. Nine patients (3 males and 6 females), ranging in age from 5 to 33 years were included. Seven (7/9) lesions were located in the mandible and two (2/9) in the maxilla. A painful swelling was the most common clinical finding (n = 4, 4/9). Radiologically, the lesions frequently presented as multilocular (5/9), well defined (4/9), bone expansion and perforation (2/9). Pathological analysis revealed that two cases were associated with central ossifying fibroma and one case with central giant cell lesion. Histomorphology showed a predominance of the solid type (5/9) and of sinusoidal pseudocystic spaces (4/9). Giant cells, osteoid material, calcified material, blood vessels and hemosiderin deposits were observed in 6/9, 7/9, 8/9, 9/9 and 7/9, respectively. The patients with ABCs presented clinical and radiographical features, which often posed a diagnostic dilemma. Knowledge about the most common characteristics of ABCs may contribute to the establishment of a more accurate diagnosis.

Aneurysmal bone cyst at C2: imaging evaluation after intralesional injection of calcitonin and methylprednisolone.

OBJECTIVE: To demonstrate imaging findings during the follow-up of patients with aneurysmal bone cyst (ABC) of C2 treated with intralesional injection of calcitonin and methylprednisolone. METHOD: Three patients with ABC of C2 were treated percutaneously with intralesional injection of calcitonin and methylprednisolone. All the patients were females, with ages varying from 6 to 13 years. All of them presented with cervical masses, but without neurological symptoms. RESULTS: Imaging follow-up with CT and plain films showed progressive ossification and reduction of the blood-filled cavities with volume reduction of all lesions. No recurrence was noted during the follow-up. CONCLUSION: Intralesional injection of calcitonin and methylprednisolone is a safe treatment option for cervical ABC. The CT scan is especially valuable as a guide of injection site and for the follow-up of ossification of the treated ABCs.

[Aneurysmal bone cyst of the maxilla. Report of one case]. / Quiste aneurismático del maxilar superior. Reporte de un caso.

Aneurysmal bone cysts are benign fibro osseous lesions, rarely present in maxillary region. We report the CT and morphological findings of aneurysmal bone cyst of the maxilla in a 15 years old female. The patient was subjected to a maxillectomy, preserving the orbit floor. The postoperative evolution has been uneventful. The tumor mainly involved the maxillary sinus with extension to the orbit wall and osseous palate. Very few cases of aneurysmal bone cysts of the maxilla have been reported in the literature.

Aneurysmal bone cyst of the maxilla.

An aneurysmal bone cyst of the maxilla is a nonneoplastic, uncommon, solitary bone lesion recognized by distinct radiographic and histopathological characteristics. It is described as a localized and quickly expandable benign tumor, which can reach a considerable size. It is characterized by the replacement of bone by spongy fibro-osseous tissue and a locally destructive and multicystic lesion filled with blood. It is attributed to a circulatory disturbance leading to locally increased venous pressure but with an unclear etiology. It takes place mainly in the long bones and in patients less than 20 years old, with no sex predilection. Its treatment is by surgical excision. An aneurysmal bone cyst in the facial bone is rare. The present case describes the lesion arising in the maxilla of a 14-year-old girl.

Quiste óseo aneurismático

Hemos estudiado 85 casos de quistes óseos aneurismáticos, en los que hallamos como principal manifestación clínica, el tumor y el dolor. Radiológicamente, imágenes osteolíticas multicavitarias expansivas, preferentemente en los sectores metafisarios de los huesos largos, donde predomina (fémur, 21,2 por ciento). La mayor incidencia por edad correspondió a la 2da. década de la vida (47,1 por ciento), con un ligero predominio, en nuestro material, del sexo masculino (53,0 por ciento). El estudio anatomopatológico hizo evidente, como se conoce a través de las descripciones clásicas, una lesión ósea multicavitaria con contenido hemático y paredes o septos parduscos, con trama ósea, las que histológicamente no tienen cubierta endotelial, sino condensación de elementos celulares fusiformes. El tratamiento de elección es el quirúrgico, decidiéndose el curetaje o la resección, según el tamaño y la localización. Las recidivas en nuestro material fueron muy bajas (5,8 por ciento), pero carecemos de información completa como para considerar ese dato representativo