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

2017 ◽  
Vol 2017 ◽  
pp. 1-5
Author(s):  
D. Ntalos ◽  
M. Priemel ◽  
C. Schlickewei ◽  
D. M. Thiesen ◽  
J. M. Rueger ◽  
...  

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.

2020 ◽  
Vol 102-B (2) ◽  
pp. 186-190 ◽  
Author(s):  
Ajay Puri ◽  
Prateek Hegde ◽  
Ashish Gulia ◽  
Mishil Parikh

Aims The aim of this study was to analyze the complications and outcomes of treatment in a series of previously untreated patients with a primary aneurysmal bone cyst (ABC) who had been treated by percutaneous sclerosant therapy using polidocanol. Methods Between January 2010 and December 2016, 56 patients were treated primarily with serial intralesional sclerosant injections. Their mean age was 20 years (1 to 54). The sites involved were clavicle (n = 3), humeri (n = 11), radius (n = 1), ulna (n = 3), hand (n = 2), pelvis (n = 12), femur (n = 7), tibia (n = 13), fibula (n = 3), and foot (n = 1). After histopathological confirmation of the diagnosis, 3% polidocanol (hydroxypolyaethoxydodecan) was injected into the lesion under image intensifier guidance. Patients were evaluated clinically and radiologically every six to eight weeks. In the absence of clinical and/or radiological response, a repeat sclerosant injection was given after eight to 12 weeks and repeated at similar intervals if necessary. Results There were no complications of treatment. One patient was lost to follow-up. Overall, 46/55 (84%) of lesions healed after one or more injections of polidocanol: 24/55 (44%) patients healed with a single injection, and 43/55 (78%) within two injections. Of these 46, four (9%) patients developed local recurrence, two of whom healed with a repeat sclerosant injection. Thus, 44/55 (80%) patients of primary ABC healed with sclerotherapy. The mean follow-up was 62 months (20 to 111). The local recurrence free survival (LRFS) with percutaneous sclerosant therapy with polidocanol was 100%, 98% (95% confidence interval (CI) 85 to 100) and 93% (95% CI 78 to 98) at two, three, and five years, respectively. Conclusion Percutaneous sclerotherapy using polidocanol is a safe, effective, minimally invasive and inexpensive method of treating a primary ABC of the limbs or pelvis. Cite this article: Bone Joint J 2020;102-B(2):186–190.


2010 ◽  
Vol 11 (4) ◽  
pp. 56-62 ◽  
Author(s):  
Yusuf Ziya Bayindir ◽  
Yahya Orcun Zorba ◽  
Cagatay Barutcugil

Abstract Aim The aim of this report is to present five-year follow-ups of two different applications for the use of direct laminate resin-based composite veneers to improve esthetics. Background Defects in the maxillary anterior teeth, such as enamel hypoplasia and peg lateral, can present esthetic challenges. Furthermore, a treatment plan that can be completed in a single appointment is highly desirable. This case report presents two different clinical cases involving the use of direct laminate resin-based composite veneers with five-year follow-ups. Case Report Case 1: A 17-year-old female patient was referred for treatment of her anterior teeth, which were unesthetically altered due to enamel hypoplasia and dental caries. A treatment plan was developed that included restoring the affected teeth with direct resin-based composite laminate veneers to improve the patient's appearance. The six maxillary anterior teeth were prepared for and restored with direct resinbased composite laminate veneers. At the fiveyear follow-up, the patient was satisfied with the restorations both esthetically and functionally. Case 2: A 15-year-old female patient also was referred for treatment to improve the appearance of her maxillary anterior teeth. A treatment plan was developed with two objectives: (1) to restore the undersized supernumerary crown in the area of the maxillary right lateral incisor and (2) to close the anterior diastemas. The facial surfaces were conservatively prepared and resin-based composite was applied with the aid of transparent crown forms. After completion of the treatment, the patient was recalled at six-month intervals. At the five-year follow-up appointment, the restorations were intact, no adverse effects were noted, and the resultant appearance was highly satisfactory for the patient. Summary The use of direct resin-based composite laminate veneers and adhesive bonding systems has been shown to provide an esthetic alternative to metal-ceramic or all-ceramic crowns for the rehabilitation of anterior teeth. This treatment option offers another advantage, namely a lower cost compared to an indirect technique. Other more complex and costly treatment options in the future are not ruled out. Clinical Significance In the present two cases, the initial and five-year follow-up results support the use of direct resin-based composite laminate veneers with minimal altering of healthy tooth structure. Such results should encourage clinicians to seek a cost-effective technique such as direct resin restorations to improve a patient's esthetic appearance in a single appointment. Citation Zorba YO, Bayindir YZ, Barutcugil C. Direct Laminate Veneers with Resin Composites: Two Case Reports with Five-Year Follow-ups. J Contemp Dent Pract [Internet]. 2010 July; 11(4):056-062. Available from: http://www. thejcdp.com/journal/view/volume11-issue4-zorba


Rare Tumors ◽  
2021 ◽  
Vol 13 ◽  
pp. 203636132110347
Author(s):  
Karlton Wong ◽  
Jomjit Chantharasamee ◽  
Scott Nelson ◽  
Mark A Eckardt ◽  
Kambiz Motamedi ◽  
...  

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.


2018 ◽  
Vol 104 (5) ◽  
pp. 344-351 ◽  
Author(s):  
Emanuela Palmerini ◽  
Pietro Ruggieri ◽  
Andrea Angelini ◽  
Stefano Boriani ◽  
Domenico Campanacci ◽  
...  

Purpose: Aneurysmal bone cyst (ABC) is a rare skeletal tumor usually treated with surgery/embolization. We hypothesized that owing to similarities with giant cell tumor of bone (GCTB), denosumab was active also in ABC. Methods: In this observational study, a retrospective analysis of ABC patients treated with denosumab was performed. Patients underwent radiologic disease assessment every 3 months. Symptoms and adverse events were noted. Results: Nine patients were identified (6 male, 3 female), with a median age of 17 years (range 14–42 years). Primary sites were 6 spine–pelvis, 1 ulna, 1 tibia, and 1 humerus. Patients were followed for a median time of 23 months (range 3–55 months). Patients received a median of 8 denosumab administrations (range 3–61). All symptomatic patients had pain relief and 1 had paresthesia improvement. Signs of denosumab activity were observed after 3 to 6 months of administration: bone formation by computed tomography scan was demonstrated in all patients and magnetic resonance imaging gadolinium contrast media decrease was observed in 7/9 patients. Adverse events were negligible. At last follow-up, all patients were progression-free: 5 still on denosumab treatment, 2 off denosumab were disease-free 11 and 17 months after surgery, and the last 2 patients reported no progression 12 and 24 months after denosumab interruption and no surgery. Conclusions: Denosumab has substantial activity in ABCs, with favorable toxicity profile. We strongly support the use of surgery and/or embolization for the treatment of ABC, but denosumab could have a role as a therapeutic option in patients with uncontrollable, locally destructive, or recurrent disease.


2021 ◽  
Vol 111 (1) ◽  
Author(s):  
Olgun Bingol ◽  
Guzelali Ozdemir ◽  
Niyazi Erdem Yasar ◽  
Alper Deveci

The aneurysmal bone cysts, usually found in the tibia, femur, pelvis, or humerus, are expansile pseudotumor lesions of unknown etiology. An aneurysmal bone cyst is rarely seen in the medial cuneiform. In this case report, a 43-year-old man with an aneurysmal bone cyst in the left medial cuneiform is presented. The cyst was curetted, and the defect was filled with an en bloc iliac crest graft. A screw was placed to fix the graft in the proper position. In the 2-year follow-up of the patient, recurrence was not detected radiologically.


2013 ◽  
Vol 20 (2) ◽  
pp. 93-101
Author(s):  
Rasa Aurelija Vankevičiūtė ◽  
Arijanda Neverauskienė ◽  
Evaldas Lukoševičius

Background. The aim of this article is to present rare clinical cases of craniofacial fibro-osseous lesions in pediatric patients. Materials and methods. In this report we present 3 cases of fibroosseous craniofacial lesions in pediatric patients. Lesions of bones resembled fibrous dysplasia, dysplastic fibroma and chronic osteomyelitis which were confirmed or suspected by clinical signs, radiographic visualization methods and histopathological investigations for these patients. The diagnostic workup, treatment, follow-up procedures and a literature review are presented. Results and conclusions. Fibro-osseous lesions of the jaws and skull manifest in a similar pattern. Conditions are easily revealed upon examination and with the help of conventional diagnostics, however, differential diagnosis using only radiological imaging is impossible. Histological findings remain a tool of choice determining the treatment strategy and outcome of the patients but it is not always available due to anatomical and functional considerations. Treatment of fibro-osseous lesions of the face and skull is highly individualized and depends on various qualities of the lesion and the patient himself. All patients received palliative symptomatic treatment to alleviate their conditions and in all cases obtaining a histology specimen and surgical intervention for treatment of the lesion was considered as a treatment option only if it was seen being safe and as least traumatic as possible because of the functional and psychological importance of the craniofacial region.


Neurosurgery ◽  
1982 ◽  
Vol 10 (1) ◽  
pp. 111-115 ◽  
Author(s):  
Philippe Bret ◽  
Christian Confavreux ◽  
Hervé Thouard ◽  
Jean Pialat

Abstract The authors report the case of a 62-year-old woman with an aneurysmal bone cyst of the cervical spine. The patient presented with a progressive tetraparesis. Cervical computed tomographic scanning revealed a mass involving three contiguous vertebral bodies. The patient underwent two consecutive surgical procedures: (a) laminectomy and metallic plate osteosynthesis and (b) an anterior approach for curettage of the cyst and autologous grafting. Twelve months after operation, the patient was given a course of radiation therapy in the cervical area. She was found to be asymptomatic at the 20-month follow-up examination. The authors discuss the etiological, anatomical, and radiological features of vertebral aneurysmal bone cysts and evaluate treatment methods.


2020 ◽  
pp. 107815522095000
Author(s):  
Alicia Rodríguez-Alarcón ◽  
David Conde-Estévez

Introduction Extravasation is a rare complication from intravenous chemotherapy administration. Literature about monoclonal antibody (MoAb) extravasations is scarce and also conflicting in how they are classified. Case report We reported two different cases of MoAb extravasations with cetuximab and nivolumab outcome respectively. The administration site appeared inflamed and patients did not report disturbances. Management and outcome: Both extravasations did not require specific treatment. General unspecific measures suffice to properly manage these extravasations and no sequels were observed after long follow-up. Both patients received all further courses of MoAb without any adverse events. Discussion To our knowledge, we reported the first case-report of nivolumab extravasation in the literature. In addition, the cetuximab extravasation management and outcome was in accordance with previously published reports. Both MoAb may be considered as non-aggressive or neutral. We reviewed published information about MoAb extravasations. In conclusion, not all MoAb should be classified in the same category when extravasated and special precautions are warranted with conjugated MoAb and bevacizumab.


2021 ◽  
Vol 48 (4) ◽  
pp. 476-483
Author(s):  
Minah Sung ◽  
Nanyoung Lee ◽  
Sangho Lee ◽  
Myeongkwan Jih

Traumatic bone cyst (TBC) is an asymptomatic lesion seen most in adolescents. TBC is found incidentally on routine panoramic examinations and appears as a relatively well-demarcated unilocular radiolucency with scalloped margins. Histological examination reveals a vacant cavity of cancellous bone usually unlined or very occasionally lined with a thin connective tissue layer. The lack of lining epithelial membrane is common histological feature. The most affected site is between the mandibular canine and third molar. The involvement of the mandibular symphysis is rare. The etiopathogenesis of the TBC is unclear. TBC is treated with surgical exploration and curettage; new bone is formed in place of the lesion within 6 - 12 months of surgery. Diagnosis of TBC prior to surgical intervention has limitations in most of the cases. Both of our patients were diagnosed through radiological examination and biopsy. Neither patient had a history of trauma. After surgery, the panoramic radiograph and CBCT were used to confirm bone healing.


2019 ◽  
Vol 25 (5) ◽  
pp. 564-569 ◽  
Author(s):  
Sudhen B Desai ◽  
Cormac O’Brien ◽  
Raja Shaikh ◽  
Daniel Hedequist ◽  
Mark Proctor ◽  
...  

Objective The management of spinal aneurysmal bone cysts (ABCs) is complex and often requires multimodality therapy, including surgical intervention to stabilize the axial skeleton, and avoid neurologic injury or death. With en bloc resection, ABCs have a recurrence rate of 12%, which increases to >50% with subtotal resection. The use of doxycycline sclerotherapy has been reported to reduce the recurrence rate of non-spinal ABCs to 5% at >24 month follow-up. We retrospectively reviewed our institutional results for sodium tetradecyl sulfate (STS)/doxycycline sclerotherapy and surgical intervention for spinal ABCs, to assess our treatment paradigm for these tumors and inform our future approach to these lesions. Methods Three cervical, two thoracic and two lumbar spine ABCs were treated in seven patients with spine-exclusive disease at our institution from 2011 to the present. The most common presenting complaint was pain. Each patient was retrospectively reviewed for clinical symptomology, number of treatments, technique and clinical follow-up. Qualitative assessment of improvement was based on the most recent clinical evaluation. Results The cohort underwent a mean of three treatment sessions (range 2–15). All were treated with STS and/or doxycycline. Five patients underwent surgical intervention at some point, either before or following sclerotherapy. After the last sclerotherapy session, four patients reported stable or improved pain symptoms, while two reported progressive pain that required surgical intervention for that indication. One patient, who underwent both multiple rounds of sclerotherapy and surgical resection, died due to acute on chronic cervical spine collapse with cord compression and inability to control disease. Conclusion We report our experience in the treatment of spinal column ABCs. Stabilization or improvement in pain was seen in four patients, while the remainder had progressive disease. Our multidisciplinary approach allows patients to receive the most appropriate treatment at presentation and thereafter, for symptom amelioration or spinal stability. Important future goals are to quantitatively assess changes in symptoms over time and to incorporate a reproducible radiographic endpoint for the assessment of treatment efficacy.


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