Aggressive osteoblastoma with a secondary aneurysmal bone cyst treated with denosumab

Osteoblastomas and aneurysmal bone cysts (ABC) are rare benign bone tumors that make up about 1%–2% of primary bone malignancies, typically occurring in young patients with a median age of 20 years, most commonly effecting the axial skeleton. ABCs may develop independently as primary lesions, or secondary to other bony lesions including osteoblastomas, chondroblastomas, and giant cell tumors. Treatment of unresectable or extensive osteoblastomas can be challenging. In 2013, the Food and Drug Administration (FDA) approved denosumab for the treatment of giant cell tumors of the bone due to its efficacy in these morbid bony lesions. Various case reports have shown that osteoblastomas can respond to denosumab. Furthermore, numerous ABC case reports have described the efficacy of denosumab in these situations. We herein describe a unique case of a young patient with an aggressive osteoblastoma and secondary ABCs who was successfully treated with denosumab.

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Aneurysmal Bone Cyst and Giant Cell Tumor of the Foot

Foot & Ankle International ◽

10.1177/107110079601700810 ◽

1996 ◽

Vol 17 (8) ◽

pp. 487-495 ◽

Cited By ~ 50

Author(s):

Roberto Casadei ◽

Pietro Ruggieri ◽

Manuela Moscato ◽

Andrea Ferraro ◽

Piero Picci

Keyword(s):

Giant Cell ◽

Bone Cyst ◽

Bone Destruction ◽

Lytic Lesion ◽

Joint Involvement ◽

X Rays ◽

Giant Cell Tumors ◽

Pathologic Fractures ◽

Tarsal Bones ◽

Aneurysmal Bone Cysts

From 1950 to 1994, 257 cases of benign bone tumors of the foot were treated at the Bone Tumor Center of the Rizzoli Institute. Aneurysmal bone cysts (ABC) and giant cell tumors (GCT) are rare and often they are misdiagnosed. To define the clinical and radiographic features useful for diagnosis, x-rays of 24 ABCs and 21 GCTs of the foot were reviewed. Adding our series to the cases reported in the literature, the only important clinical data that emerged was the average age of patients affected by ABC and GCT (15 and 27 years, respectively). ABCs localized in small tarsal bones were rare (6%), whereas 19% of GCTs were found in this site. The eccentric, round shape of a lytic lesion was more characteristic of GCT. An aggressive pattern of radiographic bone destruction was observed more frequently in GCT than in ABC ( P = 0.01). Septation was seen in both tumors. Typical of ABC was a sharp and trabeculated margin, whereas GCT had an ill-defined edge with normal cancellous bone ( P = 0.007). The growth of ABCs often expanded the cortex, while GCTs eroded, thinned, and broke the cortical bone ( P = 0.001). A saucerized cortex was observed only in ABCs. A bony shell was more characteristic of ABC than of GCT ( P = 0.002). Pathologic fractures (67%) and joint involvement, evaluated by computed tomography and magnetic resonance imaging (83%), were more frequent in GCTs, whereas fluid levels were more frequently observed in ABCs (47%). The majority of the tumors were stage 2, but GCTs represented 70% of stage 3 aggressive lesions.

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Chondroblastoma: An Update

Archives of Pathology & Laboratory Medicine ◽

10.5858/arpa.2016-0281-rs ◽

2017 ◽

Vol 141 (6) ◽

pp. 867-871 ◽

Cited By ~ 21

Author(s):

Wenqian Chen ◽

Lisa M. DiFrancesco

Keyword(s):

Aneurysmal Bone Cyst ◽

Bone Cyst ◽

Cyst Formation ◽

Giant Cells ◽

Lytic Lesion ◽

Primary Bone Tumor ◽

Hemosiderin Deposition ◽

Secondary Aneurysmal Bone Cyst ◽

Primary Bone ◽

Chondroblastic Osteosarcoma

Chondroblastoma is a rare primary bone tumor of young people that typically arises in the ends of the long bones. Radiologic investigations show a small, circumscribed, lytic lesion. The tumor is characterized histologically by the proliferation of chondroblasts along with areas of mature cartilage, giant cells, and occasionally, secondary aneurysmal bone cyst formation. Chondroblastoma, however, may also present with atypical features, such as prominent hemosiderin deposition, numerous giant cells, or the presence of a large aneurysmal bone cyst component. Malignant entities such as clear cell chondrosarcoma and chondroblastic osteosarcoma must also be considered. Recently, immunohistochemical stains such as DOG1 and SOX9 have been described in chondroblastoma, and K36M mutations in either the H3F3A or H3F3B genes have also been identified. While generally regarded as a benign entity, chondroblastoma manifests an intermediate type of behavior, given its ability to recur locally, and rarely, metastasize.

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Therapeutic Management of a Substantial Pelvic Aneurysmatic Bone Cyst Including the Off-Label Use of Denosumab in a 35-Year-Old Female Patient

Case Reports in Orthopedics ◽

10.1155/2017/9125493 ◽

2017 ◽

Vol 2017 ◽

pp. 1-5

Author(s):

D. Ntalos ◽

M. Priemel ◽

C. Schlickewei ◽

D. M. Thiesen ◽

J. M. Rueger ◽

...

Keyword(s):

Monoclonal Antibody ◽

Female Patient ◽

Therapeutic Agent ◽

Surgical Intervention ◽

Case Reports ◽

Bone Cyst ◽

Off Label Use ◽

Aneurysmal Bone Cysts ◽

Promising Therapeutic Agent

Aneurysmal bone cysts (ABC) are benign bone tumors, which are highly vascularized. The main course of treatment is curettage followed by bone grafting or cement insertion. Still recurrence remains a main problem for patients. Denosumab is a monoclonal antibody, which acts as an inhibitor of the RANK/RANKL pathway, diminishing bone turnover. Recent case reports have shown that Denosumab can be a promising therapeutic agent for people suffering from therapy-resistant ABC. We report the case of a 35-year-old female patient presenting with a pronounced ABC of the pelvis. Since the tumor was inoperable, Denosumab was administered, leading to a significant shrinkage of the lesion, which allowed surgical intervention. Upon recurrence, Denosumab was restarted putting the patient once more into remission. Follow-up was four years overall with a clinical and radiological stable disease for fifteen months after final discontinuation of the monoclonal antibody. Therefore, our case further underlines the potential of Denosumab in the treatment of ABC.

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Giant Cell Tumor of the Talar Neck

Journal of the American Podiatric Medical Association ◽

10.7547/0970225 ◽

2007 ◽

Vol 97 (3) ◽

pp. 225-228 ◽

Cited By ~ 7

Author(s):

Hakan Selek ◽

Hamza Özer ◽

Sacit Turanli ◽

Özlem Erdem

Keyword(s):

Giant Cell Tumor ◽

Giant Cell ◽

Clinical Signs ◽

Cell Tumor ◽

Giant Cell Tumors ◽

Radiographic Appearance ◽

Aneurysmal Bone Cysts ◽

Tumors Of Bone ◽

Brown Tumors ◽

Small Bones

We describe a patient with a giant cell tumor in the talar head and neck of the left foot who was diagnosed as having osteochondritis dissecans and treated with arthroscopic drilling in this same location 3 years earlier. Giant cell tumors can be confused with several conditions, including giant cell reparative granulomas, brown tumors, and aneurysmal bone cysts. Giant cell tumors of bone typically occur in the epiphysis of long bones, including the distal femur and proximal tibia. They are uncommonly found in the small bones of the foot or ankle, and talar involvement is rare. Despite this rarity, the radiographic appearance and clinical signs of talar lesions should be considered in the differential diagnosis of nontraumatic conditions in the foot. (J Am Podiatr Med Assoc 97(3): 225–228, 2007)

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Temporal bone giant-cell tumor with secondary aneurysmal bone cyst: A rare case report and review of literature

International Journal of Neurooncology ◽

10.4103/ijno.ijno_1_20 ◽

2020 ◽

Vol 3 (1) ◽

pp. 60

Author(s):

VenkateswaraRao Kommu ◽

SudhaS Murthy ◽

Rashmi Sudhir ◽

SwarnaCh Kumari

Keyword(s):

Case Report ◽

Giant Cell Tumor ◽

Giant Cell ◽

Aneurysmal Bone Cyst ◽

Rare Case ◽

Bone Cyst ◽

Cell Tumor ◽

Review Of Literature ◽

Rare Case Report ◽

Secondary Aneurysmal Bone Cyst

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Aneurysmal bone cyst in thoracolumbar spine

BJR|case reports ◽

10.1259/bjrcr.20190133 ◽

2020 ◽

Vol 6 (3) ◽

pp. 20190133

Author(s):

Alex Kiu ◽

Tiffany Fung ◽

Pranav Chowdhary ◽

Sungmi Jung ◽

Tom Powell ◽

...

Keyword(s):

Treatment Options ◽

Bone Cyst ◽

Thoracolumbar Spine ◽

Cord Compression ◽

Central Canal ◽

Neurological Deficits ◽

Primary Bone Tumors ◽

Canal Stenosis ◽

Aneurysmal Bone Cysts ◽

Primary Bone

Aneurysmal bone cysts (ABC) are rare, benign primary bone tumors. Although benign, they can be locally aggressive resulting in erosion of bone and surrounding tissues over time. In later stages, depending on the clinical urgency, immunotherapy or surgical resection remain treatment options. This report illustrates a case of a 32-year-old female who presented with chronic worsening low back pain without neurological deficits. Radiological imaging revealed a large destructive mass arising from the thoracic spine invading into the central canal, causing critical central stenosis and cord compression. Histopathology revealed ABC. This case highlights the importance of including ABCs and other ‘benign’/locally aggressive lesions in the differential of patients with insidious musculoskeletal complaints. This case also demonstrates that one can be neurologically asymptomatic despite having critical central canal stenosis and cord compression if the causative lesion is slow growing. Understanding this allows us to arrange for most appropriate management.

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Aneurysmal Bone Cysts and Giant Cell Tumors of the Spine

Seminars in Spine Surgery ◽

10.1053/j.semss.2009.03.008 ◽

2009 ◽

Vol 21 (2) ◽

pp. 106-111 ◽

Cited By ~ 1

Author(s):

Sigurd Berven ◽

Shane Burch

Keyword(s):

Giant Cell ◽

Bone Cysts ◽

Giant Cell Tumors ◽

Aneurysmal Bone Cysts

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Primary Bone Lymphoma in Axial Skeleton in a Middle-Aged Female Presented as Recurrent Anemia

Case Reports in Oncology ◽

10.1159/000506362 ◽

2020 ◽

Vol 13 (1) ◽

pp. 276-280

Author(s):

Tala Batia ◽

Mohamed A. Yassin ◽

Deena S. Mudawi ◽

Omnia A. Hamid ◽

Ahmed M.A. Abdalhadi

Keyword(s):

Cell Lymphoma ◽

Case Reports ◽

B Cell Lymphoma ◽

Axial Skeleton ◽

Primary Bone Lymphoma ◽

Large B Cell Lymphoma ◽

Pathological Type ◽

Primary Bone ◽

Insight Into ◽

Bone Lymphoma

Primary bone lymphoma (PBL) is a peculiar extranodal presentation of non-Hodgkin’s lymphoma. Primary bone diffuse large B-cell lymphoma (DLBCL) is the most common pathological type, comprising about 80% of PBL. The diagnosis of PBL depends on the combined clinical examination and imaging studies and is confirmed with immunohistochemical examination. Due to the rarity of this disease, more relative studies and case reports are needed to provide insight into this obscure lymphoproliferative malignancy. Here, we report one rare case of primary bone DLBCL involving the axial skeleton in a 37-year-old female.

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Giant cell tumors of the calvaria

Journal of Neurosurgery ◽

10.3171/jns.1975.42.5.0535 ◽

1975 ◽

Vol 42 (5) ◽

pp. 535-540 ◽

Cited By ~ 24

Author(s):

Constantin Arseni ◽

Lenke Horvath ◽

Mlhail Maretsis ◽

Nlcolae Carp

Keyword(s):

Giant Cell ◽

Benign Tumor ◽

Young Patients ◽

Cell Tumor ◽

Surgical Removal ◽

Minor Trauma ◽

Giant Cell Tumors ◽

Content Type ◽

Low Rate ◽

Fine Print

✓ The authors report and discuss four rare cases of primary, giant cell tumor of the cranial vault, a usually benign tumor that develops in young patients and has an extremely low rate of latent malignancy. The authors believe the preferred treatment to be total surgical removal of the tumor; only in rare cases of malignancy when the tumor is impossible to remove totally should this be combined with radiotherapy. Associated minor trauma played only a tumor-revealing role and was not involved in the etiology.

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