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Conde RA, Silva Oliveira Filho AR, Machinski E, Furtado da Cruz V, Melinte MA, Varone BB, Pires de Camargo O, Helito CP, Leal DP. Can bioactive glass replace bone grafts after curettage of benign bone Tumors? A systematic review and meta-analysis of randomized controlled trials. J Orthop 2025; 69:208-215. [PMID: 40538410 PMCID: PMC12173806 DOI: 10.1016/j.jor.2025.05.061] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/12/2025] [Accepted: 05/26/2025] [Indexed: 06/22/2025] Open
Abstract
Background and purpose Curettage followed by autografts or allografts has been the preferred method for treating benign bone tumors. However, the development of synthetic substances, such as bioactive glass (BAG), has prompted interest in the medical community. This meta-analysis of randomized controlled trial (RCTs) aimed to compare the adverse event rates between BAG and autograft or allograft following curettage for benign bone tumors. Methods We searched PubMed, EMBASE, and Cochrane Central for RCTs comparing BAG versus autograft or allograft after curettage for benign bone tumors. The primary endpoint was tumor recurrence. Secondary endpoints included rates of overall complications, mild complications, and reoperations. Risk ratios (RRs) with 95 % CIs were pooled across trials. Results Four RCTs encompassing 156 patients were included, with 80 (51.3 %) receiving BAG. The most common lesions were enchondromas (24.4 %) and aneurysmal bone cysts (21.8 %). Recurrence rates were comparable between the BAG group (18.7 %) and the bone grafts (21.0 %) group (RR 0.91; 95 % CI 0.52 to 1.61; p = 0.75; I2: 0 %). Reoperation (RR 1.53; 95 % CI 0.64 to 3.66; p = 0.34; I2 = 0 %), overall complications (RR 0.72; 95 % CI 0.46 to 1.13; p = 0.15; I2 = 0 %), and mild complications (RR 0.54; 95 % CI 0.16 to 1.86; p = 0.33; I2 = 21 %) were also comparable. Longer follow-up subanalysis showed analogous results. Conclusions BAG appears as effective as autografts or allografts regarding recurrence, reoperation, and complication rates after curettage of benign bone tumors. Larger RCTs with more standardized approaches are needed for validation.
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Affiliation(s)
- Rodrigo Arruda Conde
- Fundación H.A. Barcelo, Instituto Universitario de Ciencias de La Salud, Buenos Aires, CABA, Argentina
| | | | - Elcio Machinski
- Universidade Estadual de Ponta Grossa, Ponta Grossa - PR, Brazil
| | | | - Marian Andrei Melinte
- George Emil Palade University of Medicine, Pharmacy, Science, and Technology of Targu Mures, 540142, Targu Mures, Romania
| | - Bruno Butturi Varone
- Instituto de Ortopedia, Hospital Das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil
| | - Olavo Pires de Camargo
- Instituto de Ortopedia, Hospital Das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil
| | - Camilo Partezani Helito
- Instituto de Ortopedia, Hospital Das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil
| | - Daniel Peixoto Leal
- Instituto de Ortopedia, Hospital Das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil
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Anwar U, Padua HM, Chaudry G, Alomari AI, Amarneh M, Kim KR, Shaikh R. Qualitative evaluation of magnetic resonance imaging features in aneurysmal bone cysts after percutaneous cryoablation. Pediatr Radiol 2025; 55:1112-1117. [PMID: 40088277 DOI: 10.1007/s00247-025-06199-1] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/09/2024] [Revised: 01/31/2025] [Accepted: 02/10/2025] [Indexed: 03/17/2025]
Abstract
BACKGROUND Aneurysmal bone cysts (ABCs) are benign vascular lesions commonly affecting the pediatric population. OBJECTIVE To assess magnetic resonance imaging (MRI) features of ABCs after percutaneous cryoablation. MATERIALS AND METHODS A retrospective review of the hospital's electronic medical record database was conducted to identify patients who underwent percutaneous image-guided cryoablation for aneurysmal bone cysts between March 2014 and April 2024. Patients were excluded if they lacked pre- or post-treatment MRI, underwent surgical curettage and bone grafting between cryoablation treatments, or received adjunctive therapies such as sclerotherapy or bone grafting. Demographic data and pre- and post-cryoablation MRI characteristics of the lesions were collected and analyzed. RESULTS We retrospectively analyzed 20 patients (mean age = 14.6 years). The average ABC volume decreased from 83 cm3 pre-treatment to 32.7 cm3 post-treatment. In the final post-treatment MRI, there was a 50% decrease in the cystic component in 90% (n = 18) of patients, improved cortical integrity in 75% (n = 9), resolution of peri-lesional edema and mass effect in all (n = 20), and development of organized fibrosis in 50% (n = 10) of the patients. The average time was 8.3 months for a 50% reduction in cystic component, 7.3 months for improvement in cortical integrity, 6.4 months for improvement in mass effect, 15 months for complete resolution of peri-lesional enhancement, and 16.1 months for the development of organized fibrosis. CONCLUSION These findings suggest MRI is a valuable tool for monitoring ABC response to cryotherapy, guiding future treatment protocols.
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Affiliation(s)
- Usama Anwar
- Division of Interventional Radiology, Department of Radiology, Boston Children's Hospital, Longwood Ave., Boston, MA, 02492, USA
| | - Horacio M Padua
- Division of Interventional Radiology, Department of Radiology, Boston Children's Hospital, Longwood Ave., Boston, MA, 02492, USA
| | - Gulraiz Chaudry
- Division of Interventional Radiology, Department of Radiology, Boston Children's Hospital, Longwood Ave., Boston, MA, 02492, USA
| | - Ahmad I Alomari
- Division of Interventional Radiology, Department of Radiology, Boston Children's Hospital, Longwood Ave., Boston, MA, 02492, USA
| | - Mohammad Amarneh
- Division of Interventional Radiology, Department of Radiology, Boston Children's Hospital, Longwood Ave., Boston, MA, 02492, USA
| | - Kyung Rae Kim
- Division of Interventional Radiology, Department of Radiology, Boston Children's Hospital, Longwood Ave., Boston, MA, 02492, USA
| | - Raja Shaikh
- Division of Interventional Radiology, Department of Radiology, Boston Children's Hospital, Longwood Ave., Boston, MA, 02492, USA.
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Al Kadhim N, Akintayo AA, Ree A, Rezai P. Synchronous primary aneurysmal bone cysts: A rare case report and literature review. Radiol Case Rep 2025; 20:1461-1469. [PMID: 39811051 PMCID: PMC11730204 DOI: 10.1016/j.radcr.2024.11.092] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/06/2024] [Revised: 11/24/2024] [Accepted: 11/28/2024] [Indexed: 01/16/2025] Open
Abstract
Aneurysmal bone cysts (ABCs) are rare benign osteolytic tumors which occur most frequently within the first two decades of life and can lead to pathological fractures. They have characteristic imaging and histologic features and have historically been classified into two types: primary ABCs, which are true neoplasms linked to specific oncogenes, and secondary ABCs, which are preceded by other bone lesions. The occurrence of multiple concurrent primary ABCs is exceedingly rare; with only a few cases reported in the published literature. Here, we present an unusual case of this condition, along with a review of the typical presentation, imaging and pathological findings, and available treatment options.
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Affiliation(s)
- Noori Al Kadhim
- Department of Radiology, John H. Stroger, Jr. Hospital of Cook County, 1969 West Ogden Avenue, Chicago, Illinois 60612, USA
| | - Akinyemi A Akintayo
- Department of Radiology, John H. Stroger, Jr. Hospital of Cook County, 1969 West Ogden Avenue, Chicago, Illinois 60612, USA
| | - Alexander Ree
- Department of Radiology, John H. Stroger, Jr. Hospital of Cook County, 1969 West Ogden Avenue, Chicago, Illinois 60612, USA
| | - Pedram Rezai
- Department of Radiology, John H. Stroger, Jr. Hospital of Cook County, 1969 West Ogden Avenue, Chicago, Illinois 60612, USA
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Singh H, Aw SJ, Mahadev A, Zainuddin MAB, Wong KPL. Complete remodelling post-intralesional resection of an aggressive proximal humerus aneurysmal bone cyst mimicking telangiectatic osteosarcoma. BMJ Case Rep 2025; 18:e263437. [PMID: 40010756 DOI: 10.1136/bcr-2024-263437] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/28/2025] Open
Abstract
We describe a case of an aggressive proximal humerus aneurysmal bone cyst (ABC) mimicking telangiectatic osteosarcoma with complete remodelling and reconstitution of the bone. A boy in middle childhood presented with a pathological fracture preceded by progressive right shoulder pain and swelling. Imaging and open biopsy supported the diagnosis of primary ABC with positive ubiquitin-specific protease 6 gene fusion. Treatment was initially refused, leading to significant bone erosion. Subsequently, intralesional resection and grafting were performed, confirming the ABC diagnosis. Three years post-surgery, radiographs show excellent bone remodelling, with the child maintaining premorbid shoulder function. By performing an intralesional resection, there was no doubt of tissue representation, and it allows contamination to be minimised should a wide resection be necessary later. Interestingly, there was complete reconstitution and remodelling of the proximal humerus; likely from intact periosteum like that seen in the donor sites of fibula strut grafts.
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Affiliation(s)
| | - Sze Jet Aw
- Anatomical Pathology, KK Women's and Children's Hospital, Singapore
| | - Arjandas Mahadev
- Orthopaedic Surgery, KK Women's and Children's Hospital, Singapore
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Chaudhary S, Dhawale R, Sawarbandhe S, Agrawal P, Kate A, Sakhare K, Sakharkar N. Aneurysmal Bone Cyst of Femoral Head : A Rare Case Report. J Orthop Case Rep 2025; 15:133-138. [PMID: 39801849 PMCID: PMC11723746 DOI: 10.13107/jocr.2025.v15.i01.5152] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/09/2024] [Revised: 11/23/2024] [Indexed: 01/16/2025] Open
Abstract
Introduction Aneurysmal Bone Cyst (ABC) is a benign, non neoplastic, expansile lesion of bone characterized by channels of blood and spaces separated by fibrous septae. ABC of femoral head is an extremely rare condition and is difficult to treat as the surgical approach is a dilemma . Case Report We hereby report a case of aneurysmal bone cyst in femoral head of a 19 year old female patient. To preserve the vascularity of the femoral head, we approached the lesion by ganz safe surgical dislocation of the affected femoral head, trapdoor procedure of femoral head followed by curettage and bone grafting . The patient was followed up for 6 months & at latest follow up, lesion has healed completely & patient is walking pain free . Conclusion In rare cases like ABC of femoral head, a trapdoor osteotomy of femoral head with bone grafting followed by stabilization of the bone graft and osteotomy gives excellent functional and radiological outcomes.
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Affiliation(s)
- Sumedh Chaudhary
- Department of Orthopaedics, Government Medical College and Hospital , Nagpur 43GW+CR2, Hanuman Nagar, Ajni Rd, Nagpur, Maharashtra 440003, India
| | - Raunak Dhawale
- Department of Orthopaedics, Government Medical College and Hospital , Nagpur 43GW+CR2, Hanuman Nagar, Ajni Rd, Nagpur, Maharashtra 440003, India
| | - Sarang Sawarbandhe
- Department of Orthopaedics, Government Medical College and Hospital , Nagpur 43GW+CR2, Hanuman Nagar, Ajni Rd, Nagpur, Maharashtra 440003, India
| | - Pravin Agrawal
- Department of Orthopaedics, Government Medical College and Hospital , Nagpur 43GW+CR2, Hanuman Nagar, Ajni Rd, Nagpur, Maharashtra 440003, India
| | - Amit Kate
- Department of Orthopaedics, Government Medical College and Hospital , Nagpur 43GW+CR2, Hanuman Nagar, Ajni Rd, Nagpur, Maharashtra 440003, India
| | - Kuber Sakhare
- Department of Orthopaedics, Government Medical College and Hospital , Nagpur 43GW+CR2, Hanuman Nagar, Ajni Rd, Nagpur, Maharashtra 440003, India
| | - Nilesh Sakharkar
- Department of Orthopaedics, Government Medical College and Hospital , Nagpur 43GW+CR2, Hanuman Nagar, Ajni Rd, Nagpur, Maharashtra 440003, India
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Beyer RS, Steiner Q, Hennessy DW, Rosas HG, Goodspeed DC, Spiker AM. Assessment and management of periacetabular aneurysmal bone cysts-a series of four cases. J Hip Preserv Surg 2025; 12:11-19. [PMID: 40331076 PMCID: PMC12051852 DOI: 10.1093/jhps/hnae040] [Citation(s) in RCA: 1] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/07/2024] [Revised: 09/26/2024] [Accepted: 11/04/2024] [Indexed: 05/08/2025] Open
Abstract
Aneurysmal bone cysts (ABCs) in the periacetabular region can be challenging to treat because they create unique problems, given their proximity to articular cartilage and a significant weight bearing surface. This case series details the assessment and treatment of four periacetabular ABCs with a review of pertinent current literature. Treatment approaches used include curettage with 6% phenol chemical adjuvant, type III hemipelvectomy, serial image-guided sclerotherapy injections, and in one case, an exostotic lesion was treated with hip arthroscopy.
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Affiliation(s)
- Reagan S.H Beyer
- Department of Orthopedic Surgery, University of Wisconsin—Madison, 1685 Highland Ave, Madison, WI 53705, USA
| | - Quinn Steiner
- Department of Orthopedic Surgery, University of Wisconsin—Madison, 1685 Highland Ave, Madison, WI 53705, USA
| | - David W Hennessy
- Department of Orthopedic Surgery, University of Wisconsin—Madison, 1685 Highland Ave, Madison, WI 53705, USA
| | - Humberto G Rosas
- Department of Radiology, University of Wisconsin–Madison, 600 Highland Ave, Madison, WI 53792, USA
| | - David C Goodspeed
- Department of Orthopedic Surgery, University of Wisconsin—Madison, 1685 Highland Ave, Madison, WI 53705, USA
| | - Andrea M Spiker
- Department of Orthopedic Surgery, University of Wisconsin—Madison, 1685 Highland Ave, Madison, WI 53705, USA
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7
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Erdoğan F, Cengiz T, Aydın Şimşek Ş, Şay CŞ, Coşkun HS, Dabak N. Infantile Aggressive Aneurysmal Bone Cyst of the Proximal Femur: A Rare Clinical Presentation. Cureus 2024; 16:e75708. [PMID: 39807466 PMCID: PMC11728812 DOI: 10.7759/cureus.75708] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 12/12/2024] [Indexed: 01/16/2025] Open
Abstract
Aneurysmal bone cysts (ABCs) are aggressive, osteolytic lesions usually seen in childhood and young adulthood. The patient's age, location, and behavior of the lesion in the bone may cause patients to present with different clinical findings. Appropriate treatment of these rare, aggressive bone lesions is essential for recurrence. This case report aims to present the diagnosis and treatment approach in the case of an infantile aggressive ABC and to present the long-term follow-up results. A 14-month-old baby boy was admitted to the clinic with the complaint of difficulty in walking after a fall. After evaluation with advanced imaging methods, a fluid-filled cystic lesion and pathological fracture were detected in the proximal femur. After surgical treatment of the lesion, which showed an aggressive course in the follow-up, the patient was able to give a whole load six months after surgery. Although the ABC was aggressive in this case, it was controlled with surgical treatment and long-term follow-up. It should be kept in mind that ABCs in children, especially in the proximal femur, may be aggressive and may require surgical treatment.
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Affiliation(s)
- Furkan Erdoğan
- Orthopaedics and Traumatology, Faculty of Medicine, Ondokuz Mayıs University, Samsun, TUR
| | - Tolgahan Cengiz
- Orthopaedics and Traumatology, Faculty of Medicine, Ondokuz Mayıs University, Samsun, TUR
| | - Şafak Aydın Şimşek
- Orthopaedics and Traumatology, Faculty of Medicine, Ondokuz Mayıs University, Samsun, TUR
| | - Cahit Şemsi Şay
- Orthopaedics and Traumatology, Faculty of Medicine, Ondokuz Mayıs University, Samsun, TUR
| | - Hüseyin Sina Coşkun
- Orthopaedics and Traumatology, Faculty of Medicine, Ondokuz Mayıs University, Samsun, TUR
| | - Nevzat Dabak
- Orthopaedics and Traumatology, Faculty of Medicine, Ondokuz Mayıs University, Samsun, TUR
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8
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Price A, Fredricks N, Bartman M, D’Souza P, Hayworth M, Campbell G, Kan P, Lall R. Thoracic aneurysmal bone cyst secondary to osteoblastoma: unique surgical management. Illustrative case. JOURNAL OF NEUROSURGERY. CASE LESSONS 2024; 8:CASE24471. [PMID: 39556816 PMCID: PMC11579909 DOI: 10.3171/case24471] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 07/20/2024] [Accepted: 08/27/2024] [Indexed: 11/20/2024]
Abstract
BACKGROUND Aneurysmal bone cysts (ABCs) are rare, benign, yet locally aggressive lesions that contain blood-filled channels that rarely occur in the thoracic spine of adults. The literature on the treatment of spinal ABCs is sparse, but the consensus is to achieve gross-total resection (GTR) due to these lesions being locally aggressive and to prevent recurrence. OBSERVATIONS This report describes a 35-year-old female admitted with back pain and right T5 dermatome radiculopathy without any inciting events. Magnetic resonance imaging revealed a 3.0 × 4.3 × 4.0-cm solid, enhancing, multicystic lesion with multiple fluid levels centered in the right posteromedial chest wall, involving the right fifth rib and costovertebral junction. Because of the high suspicion for an ABC, later found to be secondary to an osteoblastoma, surgical intervention was planned via preoperative embolization and T4-6 fusion with right T5 laminectomy and costotransversectomy to obtain GTR. LESSONS This case of an ABC secondary to osteoblastoma of the spine showcases a strategy for unique surgical management, given the limited information on treatment considerations for secondary ABCs. https://thejns.org/doi/10.3171/CASE24471.
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Affiliation(s)
- Anthony Price
- John Sealy School of Medicine, The University of Texas Medical Branch at Galveston, Texas
- Department of Neurosurgery, The University of Texas Medical Branch at Galveston, Texas
| | - Nathan Fredricks
- John Sealy School of Medicine, The University of Texas Medical Branch at Galveston, Texas
- Department of Neurosurgery, The University of Texas Medical Branch at Galveston, Texas
| | - Mason Bartman
- John Sealy School of Medicine, The University of Texas Medical Branch at Galveston, Texas
- Department of Neurosurgery, The University of Texas Medical Branch at Galveston, Texas
| | - Preston D’Souza
- Department of Neurosurgery, The University of Texas Medical Branch at Galveston, Texas
| | - Miranda Hayworth
- Department of Neurosurgery, The University of Texas Medical Branch at Galveston, Texas
| | - Gerald Campbell
- Department of Neurosurgery, The University of Texas MD Anderson Cancer Center, Houston, Texas
| | - Peter Kan
- Department of Neurosurgery, The University of Texas Medical Branch at Galveston, Texas
| | - Rishi Lall
- Department of Neurosurgery, The University of Texas Medical Branch at Galveston, Texas
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Jain M, Pradhan SS, Khan S, Tripathy S, Lubaib KP, Raj KS. Aneurysmal Bone Cyst of the Distal Ulna in an Elderly Lady. J Orthop Case Rep 2024; 14:79-83. [PMID: 39524295 PMCID: PMC11546030 DOI: 10.13107/jocr.2024.v14.i11.4922] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/17/2024] [Revised: 09/03/2024] [Indexed: 11/16/2024] Open
Abstract
Introduction Aneurysmal bone cyst (ABC) is a rare, benign, cystic, hemorrhagic expansile osteolytic lesion of unknown etiology resembling multiple intraosseous blood-filled spaces mostly seen in the first two decades of life. It accounts for <1% of all bone tumors and the usual sites are the metaphyses of the proximal and distal femur, proximal tibia, and posterior elements of the spine. Affection of other sites is unusual. Case Report A 55-year-old female with a 5-month history of pain and swelling of distal ulna. She was operated on with curettage, bone grafting, and plating. At 1-year follow-up, she regained complete movements without any recurrence. Conclusion In this article, we present a case of a primary ABC of distal ulna in an unusual age group of 55 years in a female. The clinical presentation, diagnosis, surgical management, and clinical outcome at 1 year are described.
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Affiliation(s)
- Mantu Jain
- Department of Orthopedics, AIIMS, Bhubaneswar, Odisha, India
| | | | - Shahnawaz Khan
- Department of Orthopedics, AIIMS, Bhubaneswar, Odisha, India
| | - Sujit Tripathy
- Department of Orthopedics, AIIMS, Bhubaneswar, Odisha, India
| | - KP Lubaib
- Department of Orthopedics, AIIMS, Bhubaneswar, Odisha, India
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10
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Cubitt C, Ariyaratne S, Evans S, Vaiyapuri S, Hughes S, Botchu R. A rare case of a solitary osseous metastasis from breast carcinoma presenting with fluid-fluid levels on MRI. Int Cancer Conf J 2024; 13:401-406. [PMID: 39398900 PMCID: PMC11464755 DOI: 10.1007/s13691-024-00698-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/24/2023] [Accepted: 06/22/2024] [Indexed: 10/15/2024] Open
Abstract
Osseous metastatic disease is commonly encountered in breast carcinoma, which typically presents as either osteolytic, osteoblastic, or mixed lesions on imaging. Osseous metastasis presenting as a multiloculated cystic lesion with fluid-fluid levels resembling that of an aneurysmal bone cyst (ABC) is sparsely described in the literature, and even less so in the case of breast carcinoma. We report an unusual case of fluid-fluid levels in a bone metastasis to the spine in a 66-year-old female with a prior history of breast carcinoma. Magnetic resonance imaging (MRI) demonstrated a cystic lesion with fluid levels resembling that of an ABC. Computed tomography (CT)-guided biopsy revealed the lesion to be a metastasis from breast carcinoma. The management of ABCs and osseous metastases differ drastically. Accurate diagnosis and distinction between these lesions is paramount as the management of metastatic disease can have a significant impact on the quality and length of life. The presentation, differential diagnoses and imaging features of this atypical case are discussed.
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Affiliation(s)
- Catherine Cubitt
- College of Medical and Dental Sciences, University of Birmingham, Birmingham, UK
| | - Sisith Ariyaratne
- Department of Musculoskeletal Radiology, The Royal Orthopaedic Hospital, Birmingham, UK
| | - Scott Evans
- Department of Orthopaedic Oncology, The Royal Orthopaedic Hospital, Birmingham, UK
| | - Sumathi Vaiyapuri
- Department of Pathology, The Royal Orthopaedic Hospital, Birmingham, UK
| | - Simon Hughes
- Department of Spinal Surgery, The Royal Orthopaedic Hospital, Birmingham, UK
| | - Rajesh Botchu
- Department of Musculoskeletal Radiology, The Royal Orthopaedic Hospital, Birmingham, UK
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11
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Gall C, Allison DC. Recurrent Aneurysmal Bone Cyst Treated with Percutaneous Doxycycline Sclerotherapy: A Case Report. JBJS Case Connect 2024; 14:01709767-202412000-00057. [PMID: 39637081 DOI: 10.2106/jbjs.cc.24.00371] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/07/2024]
Abstract
CASE A 7-year-old girl with an aggressive proximal femur aneurysmal bone cyst (ABC) who sustained a pathological fracture of the femoral neck required an open reduction and internal fixation with curettage and bone grafting. This was followed by early aggressive recurrence of the ABC, which was treated successfully with percutaneous doxycycline sclerotherapy. CONCLUSION Doxycycline sclerotherapy seems to be an effective and relatively low-risk treatment for recurrent ABCs.
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Affiliation(s)
- Cory Gall
- Community Memorial Health System, Department of Orthopedic Surgery, Ventura, California
- Cedars-Sinai Medical Center, Department of Orthopedic Surgery, Los Angeles, California
| | - Daniel C Allison
- Cedars-Sinai Medical Center, Department of Orthopedic Surgery, Los Angeles, California
- Children's Hospital of Los Angeles, Department of Pediatric Orthopedic Surgery, Los Angeles, California
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12
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Saraf A, Kumar SK. Sclerotherapy as a Primary or Salvage Procedure For Aneurysmal Bone Cyst: A Case Report. J Orthop Case Rep 2024; 14:102-106. [PMID: 39381282 PMCID: PMC11458240 DOI: 10.13107/jocr.2024.v14.i10.4826] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/25/2024] [Revised: 08/10/2024] [Indexed: 10/10/2024] Open
Abstract
Introduction A benign and locally aggressive tumour, aneurysmal bone cysts (ABCs) can develop in any bone but are more common in the metaphysis of long bones. Case Report A 10 year old Female patient arrived at our outpatient department two years ago with a history of recurring discomfort, edema, and limited movement in her right shoulder and proximal 1/3 of her right arm. X-ray and Magnetic Resonance Imaging (MRI) of the humerus was performed and was diagnosed as aneurysmal bone cyst of proximal humerus. Patient was managed with sclerotherapy with polidocanol injections. The patient experienced significant symptoms improvement was seen two months after starting treatment, and there were no post operative side effects. Monthly progress reports were started, and after three months, physiotherapy was added to improve shoulder range of motion because there were no indications of a recurrence. A two-year follow-up showed improvement and no indications of a relapse. Conclusion Percutaneous Sclerotherapy can be used as a primary procedure for aneurysmal bone cyst.
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Affiliation(s)
- Amit Saraf
- Department of Orthopaedics, Teerthanker Mahaveer Medical College and Research Centre, Moradabad, Uttar Pradesh, India
| | - S Krishna Kumar
- Department of Orthopaedics, Teerthanker Mahaveer Medical College and Research Centre, Moradabad, Uttar Pradesh, India
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Jokela A, Aho J, Kosola J, Stenroos A, Sinikumpu JJ, Maffulli N, Lempainen L. Heel pain in young athletes - not always Sever's Disease: A Narrative Review. Foot (Edinb) 2024; 60:102114. [PMID: 39029380 DOI: 10.1016/j.foot.2024.102114] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/28/2021] [Revised: 06/06/2024] [Accepted: 06/15/2024] [Indexed: 07/21/2024]
Abstract
Heel pain is a prevalent issue in young athletes, often arising from overuse and increased sporting demands. While Sever's Disease is the predominant cause, various other entities, including stress-related injuries and pathologies like tumors and bone lesions, contribute to this condition. The complex hind foot anatomy, encompassing ossicles, physis, and soft tissues, may lead to heel pain. This study aims to provide physicians with a clinically oriented narrative review of adolescent heel pain, supported by illustrative cases. CONCLUSION: This study aims to offer physicians a comprehensive understanding of the concepts surrounding heel pain in adolescents. By presenting clinically relevant information and illustrated cases, it seeks to enhance medical practitioners' ability to diagnose and manage heel pain effectively in this specific demographic.
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Affiliation(s)
- Aleksi Jokela
- Department of Orthopaedics and Traumatology, Turku University Hospital, Turku, Finland
| | - Joni Aho
- University of Turku, Turku, Finland
| | - Jussi Kosola
- Department of Orthopaedics and Traumatology, Kanta-Häme Central Hospital, Hämeenlinna, Finland; University of Helsinki, Helsinki, Finland; Department of Physical Activity and Health, Paavo Nurmi Centre, University of Turku, Turku, Finland; Department of Orthopaedics and Traumatology, Hyvinkää Hospital, Hyvinkää, Finland
| | - Antti Stenroos
- Department of Orthopaedics and Traumatology, Helsinki University Hospital, University of Helsinki, Helsinki, Finland
| | - Juha-Jaakko Sinikumpu
- Department of Children and Adolescents, PEDEGO unit and MRC Oulu, Oulu University Hospital and University of Oulu, Oulu, Finland
| | - Nicola Maffulli
- Department of Musculoskeletal Disorders, University of Salerno School of Medicine, Surgery and Dentistry, Salerno, Italy; Centre for Sports and Exercise Medicine, Queen Mary University of London, London, UK; Institute of Science and Technology in Medicine, Keele University School of Medicine, Stoke on Trent, UK
| | - Lasse Lempainen
- Department of Physical Activity and Health, Paavo Nurmi Centre, University of Turku, Turku, Finland; FinnOrthopaedics / Hospital Pihlajalinna, Turku, Finland; Ripoll y De Prado, FIFA Medical Centre of Excellence, Madrid, Spain.
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14
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Cekov A, Tarev I, Kanev A. Spinal aneurysmal bone cysts - case series. Folia Med (Plovdiv) 2024; 66:402-408. [PMID: 39365632 DOI: 10.3897/folmed.66.e121395] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/21/2024] [Accepted: 05/07/2024] [Indexed: 10/05/2024] Open
Abstract
Aneurysmal bone cysts are benign neoplastic lesions that are extensively vascularized and can occasionally exhibit destructive and expanding activity. They typically affect young people, with a small predilection toward females, and most frequently arise during the second decade of life. Aneurysmal bone cysts account for about 1% of all bone tumors and usually develop in long tubular bones, with the spine being much less commonly affected.
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Affiliation(s)
- Asen Cekov
- Acibadem City Clinic University Hospital Tokuda, Sofia, Bulgaria
| | - Ivan Tarev
- Acibadem City Clinic University Hospital Tokuda, Sofia, Bulgaria
| | - Anastas Kanev
- Acibadem City Clinic University Hospital Tokuda, Sofia, Bulgaria
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15
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Mahajan NP, Pande KP, Mhatre JA, Chaudhari KM. Management of Proximal Femur Aneurysmal Bone Cyst with 10-year Follow-up - A Case Report. J Orthop Case Rep 2024; 14:140-146. [PMID: 38910999 PMCID: PMC11189069 DOI: 10.13107/jocr.2024.v14.i06.4532] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/23/2024] [Revised: 04/22/2024] [Indexed: 06/25/2024] Open
Abstract
Introduction Aneurysmal bone cyst is a benign, most often non-neoplastic lesion affecting the bone. Malignant transformation is reported in certain cases, although rare. It commonly occurs in young adults with around 75% of the cases occurring in the first two decades and 95% occurring in the first 3 decades. The management depends on the individual case parameters with ABC occurring in a weight-bearing area of a bone warranting an aggressive treatment plan with the consideration of preventing a pathological fracture. Case Report A 20-year-old male patient with no other comorbidities presented to us with complaints of atraumatic right hip pain of 9-month duration. On preliminary clinical examination and imaging studies with X-rays, CT scan, and MRI, the patient was diagnosed to have an aneurysmal bone cyst (ABC) of the right proximal femur. The patient was operated with intra-lesional excision with extended curettage and prophylactic fixation with DHS plating and kept on yearly follow-up. 10-year follow-up showed complete resolution of the lesion and implant removal was done after that. Conclusion As is clear from this case, the treatment of an aneurysmal bone cyst needs to individualized based on the patient's age, complaints, and tumor behavior in terms of its aggressiveness, size, and its extent. Often, en bloc excision with extensive curettage is required along with bone grafting and prophylactic fixation to support the bone graft and to prevent a pathological fracture and further recurrence. Implant removal if indicated should be done after complete resolution of the lesion, patient willingness, and any other medical indication.
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Affiliation(s)
- Neetin Pralhad Mahajan
- Department of Orthopaedics, Grant Government Medical college, Mumbai, Maharashtra, India
| | - Kartik Prashant Pande
- Department of Orthopaedics, Grant Government Medical college, Mumbai, Maharashtra, India
| | - Jayesh Anant Mhatre
- Department of Orthopaedics, Grant Government Medical college, Mumbai, Maharashtra, India
| | - Kunal M Chaudhari
- Department of Orthopaedics, Grant Government Medical college, Mumbai, Maharashtra, India
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16
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Madani H, El Hassnaoui Y, Ait Benali H, Shimi M. Aneurysmal bone cyst of the calcaneus: A rare case report and review of the literature. Int J Surg Case Rep 2024; 119:109802. [PMID: 38788635 PMCID: PMC11140776 DOI: 10.1016/j.ijscr.2024.109802] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/25/2024] [Revised: 05/17/2024] [Accepted: 05/21/2024] [Indexed: 05/26/2024] Open
Abstract
INTRODUCTION AND IMPORTANCE Aneurysmal bone cyst (ABC) is a benign lytic bone tumor of the skeletal system but locally destructive lesion of unknown origin. It often occurs in childhood and usually involves the metaphyseal region of long bones; thus, its localisation in the calcaneum remains rare; its atypical presentation in our case makes it remarkable. CASE PRESENTATION We describe a case of a young patient who suffered from chronic heel pain, in whom the clinical examination finds swelling and pain on palpation. A complete radiological assessment was carried out, which revealed a solitary and expansive osteolytic lesion within the calcaneus. Treatment included tumor curettage and reconstruction with allograft and cement. The biopsy report was consistent with an ABC. The postoperative follow-up at 18 months was marked by a good radio-clinical evolution and no recurrence. CLINICAL DISCUSSION ABCs are benign cystic expansive tumors that are reactive, locally destructive and blood-filled, their occurrence in the calcaneus has been reported in only 1.6 % of total reported cases. Talalgia is the most frequent sign associated with swelling. Liquid-liquid level images on MRI is a pathognomonic sign of these lesions but the gold standard diagnosis remains histology. Their treatment is based on curettage - filling by grafting. CONCLUSION Curettage combined graft makes results butter and improves the quality of life.
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Affiliation(s)
- Hamza Madani
- Traumatology and Orthopedics Department, University Hospital Center of Tangier, Abdelmalek Essaâdi University, Faculty of Medicine and Pharmacy, Tangier, Morocco.
| | - Youssef El Hassnaoui
- Traumatology and Orthopedics Department, University Hospital Center of Tangier, Abdelmalek Essaâdi University, Faculty of Medicine and Pharmacy, Tangier, Morocco
| | - Hicham Ait Benali
- Traumatology and Orthopedics Department, University Hospital Center of Tangier, Abdelmalek Essaâdi University, Faculty of Medicine and Pharmacy, Tangier, Morocco
| | - Mohammed Shimi
- Traumatology and Orthopedics Department, University Hospital Center of Tangier, Abdelmalek Essaâdi University, Faculty of Medicine and Pharmacy, Tangier, Morocco
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Ahmed HA, Almasoudi EA, Nail LRL, Samargandi R. Response of Recurrent Aggressive Aneurysmal Bone Cyst of Distal Tibia to Denosumab Treatment. J Orthop Case Rep 2024; 14:157-162. [PMID: 38910984 PMCID: PMC11189066 DOI: 10.13107/jocr.2024.v14.i06.4538] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/25/2024] [Revised: 04/22/2024] [Indexed: 06/25/2024] Open
Abstract
Introduction Aneurysmal bone cysts (ABCs) are aggressive and benign tumors that primarily affect children and adolescents. The standard course of treatment for ABCs involves surgical excision or curettage with a bone transplant or cement to repair the deficiency. Denosumab, a monoclonal antibody that inhibits receptor activator of nuclear kappa B ligand, is used to treat osteoporosis, skeletal metastasis, and giant cell tumors of the bones. Case Report This case study details the therapeutic treatment of a female patient, age 22, who had a recurring aggressive ABC of the distal tibia. The patient was initially treated using curettage and lesion filling. However, recurrence of the osteolysis was observed 9 months later that led to subsequent interventions involving absolute alcohol sclerotherapy in multiple sessions. However, these interventions failed to achieve ossification. Following unsuccessful surgical and sclerotherapy treatments, the patient was administered denosumab, which led to a positive response. Regular radiographic and clinical follow-up demonstrated significant improvements in ossification and pain reduction. During the course of the 12-month treatment, the frequency of visits was gradually reduced. Further, follow-up and monitoring revealed the effectiveness of the local control and long-term treatment. Conclusion This case report highlights the ability of denosumab to manage recurrent aggressive ABCs after surgical or sclerotherapy failure.
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Affiliation(s)
- Hassan A Ahmed
- College of Medicine, University of Jeddah, Jeddah, Saudi Arabia
| | - Eid A Almasoudi
- College of Medicine, University of Jeddah, Jeddah, Saudi Arabia
| | - Louis-Romée Le Nail
- Department of Orthopaedics Surgery and Traumatology, Centre Hospitalier Régional Universitaire de Tours, Tours, France
| | - Ramy Samargandi
- Department of Orthopaedics Surgery and Traumatology, Centre Hospitalier Régional Universitaire de Tours, Tours, France
- Department of Orthopedic Surgery, College of Medicine, University of Jeddah, Jeddah, Saudi Arabia
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18
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Gyemi L, Selznick A, Petrisor B, Ghert M. Time to full weight-bearing with the use of a calcium sulfate-calcium phosphate bone substitute as a bone void filler following extended curettage in the treatment of primary benign bone tumours. J Orthop Surg (Hong Kong) 2024; 32:10225536241254200. [PMID: 38733211 DOI: 10.1177/10225536241254200] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 05/13/2024] Open
Abstract
PURPOSE The primary objective of this study was to determine time to full weight-bearing after the use of a calcium-sulfate-calcium phosphate bone substitute (CaSO4/CaPO4) as a bone void filler in the treatment of primary benign bone tumours following intralesional curettage. The secondary objectives were to determine surgical complications and recurrence rates. METHODS Retrospective review of patients identified from a surgeon-specific orthopaedic oncology database, who underwent curettage of benign bone tumours and subsequent bone void filling with CaSO4/CaPO4. RESULTS A total of 39 patients (20 males, 19 females) met inclusion criteria with an average age of 31 years (range: 13 to 62 years), a median follow-up of 3.7 years, and a maximum follow-up of 11 years. The most common tumour diagnosis was giant cell tumour of bone (GCT) (n = 19), and the most common location was the proximal tibia (n = 9). The mean volume of tumour excised was 74.1 cm3 including extraosseous bone expansion due to tumour growth, with a mean of volume of 21.4 mL of CaSO4/CaPO4 used to fill the intraosseous cavitary defects to restore normal bone anatomy. None of the lesions required additional internal fixation. The primary outcome measure, average time to full weight-bearing/full range of motion, was 11 weeks and 6 weeks for upper and lower extremity lesions, respectively. Secondary outcomes included tumour recurrence requiring reoperation in five patients and infection requiring reoperation in two patients. CONCLUSION This study demonstrates that CaSO4/CaPO4 is a viable option as a bone void filler in the reconstruction of cavitary defects following removal of primary benign bone tumours. CaSO4/CaPO4 provides sufficient bone regeneration early in the post-operative period to allow progression to full weight-bearing within weeks without the need for internal fixation. There were no graft-specific complications noted.
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Affiliation(s)
- Lauren Gyemi
- Division of Orthopaedic Surgery, Department of Surgery, McMaster University, Hamilton, ON, Canada
| | - Asher Selznick
- Division of Orthopaedic Surgery, Department of Surgery, McMaster University, Hamilton, ON, Canada
| | - Brad Petrisor
- Division of Orthopaedic Surgery, Department of Surgery, McMaster University, Hamilton, ON, Canada
| | - Michelle Ghert
- Division of Orthopaedic Surgery, Department of Surgery, McMaster University, Hamilton, ON, Canada
- Department of Orthopaedics, University of Maryland, Baltimore, MD, USA
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19
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Costa DD, Gabrielli E, Cerrone M, Di Gialleonardo E, Maccauro G, Vitiello R. Pathological Fractures in Aneurysmal Bone Cysts: A Systematic Review. J Clin Med 2024; 13:2485. [PMID: 38731012 PMCID: PMC11084630 DOI: 10.3390/jcm13092485] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/14/2024] [Revised: 04/18/2024] [Accepted: 04/21/2024] [Indexed: 05/13/2024] Open
Abstract
Background: Aneurysmal bone cysts (ABCs) are osteolytic, non-malignant, vascular lesions of the bone. Pathological fractures can be a manifestation of the ABCs, which occur in about 8% of ABCs. Different treatments have been described in the literature, but, nowadays, an optimal management of the pathological fractures in patients with ABCs is still a matter of debate and there are no standard guidelines for treatment nor any shared indication about the best surgical intervention. The aim of our study is to review the current literature available on this matter exploring and confronting different surgical treatments for pathological fractures in ABC in order to clarify the surgical approach to these patients. Methods: A systematic review of the literature indexed in PubMed, MEDLINE, and Cochrane Library databases was carried out. The Preferred Reporting Items for Systematically Reviews and Meta-Analyses (PRISMA) were followed. Results: A total of 37 articles were relevant and were finally included in the study. In total, we reached a population of 140 patients. Of the 140 patients included in the review, 124 patients (88.6%) underwent curettage surgery, 15 patients (10.7%) underwent en bloc resection surgery. A total of 47% of patients (70) underwent synthesis surgery with a plate, screw, nail, or external fixator. Adjuvant treatments were used in 8.6% of patients (12). Complications involved 20.7% of the patients (29). Conclusions: In conclusion, the treatment of pathological fractures in aneurysmal bone cysts requires careful patient assessment, considering factors such as age, the presence of open growth plates, the location of the lesion, and the surgeon's expertise.
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Affiliation(s)
- Doriana Di Costa
- Department of Orthopaedics, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168 Rome, Italy; (D.D.C.); (M.C.); (E.D.G.); (G.M.); (R.V.)
- Department of Orthopaedic Surgery, Università Cattolica del Sacro Cuore, 00168 Rome, Italy
| | - Elena Gabrielli
- Department of Orthopaedics, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168 Rome, Italy; (D.D.C.); (M.C.); (E.D.G.); (G.M.); (R.V.)
- Department of Orthopaedic Surgery, Università Cattolica del Sacro Cuore, 00168 Rome, Italy
| | - Mariagrazia Cerrone
- Department of Orthopaedics, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168 Rome, Italy; (D.D.C.); (M.C.); (E.D.G.); (G.M.); (R.V.)
- Department of Orthopaedic Surgery, Università Cattolica del Sacro Cuore, 00168 Rome, Italy
| | - Emidio Di Gialleonardo
- Department of Orthopaedics, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168 Rome, Italy; (D.D.C.); (M.C.); (E.D.G.); (G.M.); (R.V.)
- Department of Orthopaedic Surgery, Università Cattolica del Sacro Cuore, 00168 Rome, Italy
| | - Giulio Maccauro
- Department of Orthopaedics, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168 Rome, Italy; (D.D.C.); (M.C.); (E.D.G.); (G.M.); (R.V.)
- Department of Orthopaedic Surgery, Università Cattolica del Sacro Cuore, 00168 Rome, Italy
| | - Raffaele Vitiello
- Department of Orthopaedics, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168 Rome, Italy; (D.D.C.); (M.C.); (E.D.G.); (G.M.); (R.V.)
- Department of Orthopaedic Surgery, Università Cattolica del Sacro Cuore, 00168 Rome, Italy
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20
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Singh R, Madasswery S, Colman M, Kent PM. Denosumab and sclerotherapy for recurrent spinal aneurysmal bone cyst in a child. BMJ Case Rep 2024; 17:e257450. [PMID: 38589235 PMCID: PMC11015305 DOI: 10.1136/bcr-2023-257450] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 04/10/2024] Open
Abstract
Aneurysmal bone cyst (ABC) is a non-malignant, locally destructive, blood-filled lesion in the bone that tends to grow aggressively. A young girl presented with a rapid recurrence after aggressive surgery of a large symptomatic sacral-spinal ABC. After a multidisciplinary tumour board, she was successfully treated with sclerotherapy and monthly intravenous denosumab. The patient has maintained asymptomatic for over 36 months now and has returned to full activity and strength. She never required surgery and has had radiologic resolution of the lesions. Treatment of recurrent ABC requires a multidisciplinary team approach. We believe this to be the first report to use this combined therapy to provide an alternative to morbid surgery for children with ABCs.
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Affiliation(s)
- Raj Singh
- College of Medicine, Rush University Medical Center, Chicago, Illinois, USA
| | | | - Matt Colman
- Rush University Rush Medical College, Chicago, Illinois, USA
| | - Paul McKeegan Kent
- Medical Director, FibroFighters Foundation LLC, Temecula, California, USA
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21
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Radalov I, Páez-Carpio A, Macías Rodríguez N, Inarejos Clemente EJ, Gómez FM. Minimizing neurovascular complications during image-guided percutaneous cryoablation of a cervical spinal aneurysmal bone cyst using protective doxycycline sclerotherapy: a case report. JOURNAL OF SPINE SURGERY (HONG KONG) 2024; 10:159-164. [PMID: 38567007 PMCID: PMC10982925 DOI: 10.21037/jss-23-147] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 12/26/2023] [Accepted: 02/23/2024] [Indexed: 04/04/2024]
Abstract
Background Aneurysmal bone cysts (ABC) are rare, locally aggressive bone tumors primarily observed in pediatric patients. Surgical curettage is the treatment of choice. Image-guided percutaneous cryoablation (CYOA) is a recently implemented alternative technique in cases not amenable to surgery. CYOA may be limited if the lesion is close to critical neurovascular structures. In this case report, a cervical spinal ABC was successfully treated using CYOA in combination with complementary and protective image-guided percutaneous doxycycline sclerotherapy (DS) to dissect and treat the portion of the lesion in contact with critical structures. Case Description A 4-year-old male presented with a symptomatic ABC within the C5 vertebral body, which encompassed the right vertebral artery and contacted the right C5-C6 spinal cord and nerve roots. After ruling out surgery due to the proximity of critical neurovascular structures, treatment with CYOA was performed. However, subsequent follow-up showed recurrence in the part of the lesion contacting critical structures. A second approach was then decided, using DS to dissect and treat the most vulnerable portion and CYOA to treat the remaining tumor. Follow-up showed almost complete sclerosis of the lesion and total resolution of symptoms 3 years after treatment. Conclusions Using DS to dissect and treat the portion of the lesion in contact with critical neurovascular structures during CYOA treatment of a cervical spinal ABC allowed for a safe and effective approach in our case.
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Affiliation(s)
- Igor Radalov
- Musculoskeletal Radiology Unit, Department of Radiology, Hospital del Mar, Barcelona, Spain
| | - Alfredo Páez-Carpio
- Department of Medical Imaging, University of Toronto, Toronto, Canada
- Interventional Radiology Unit, Department of Radiology, Hospital Clínic Barcelona-Sant Joan de Déu Children’s Hospital, Barcelona, Spain
| | - Napoleón Macías Rodríguez
- Interventional Radiology Unit, Department of Radiology, Hospital Clínic Barcelona-Sant Joan de Déu Children’s Hospital, Barcelona, Spain
| | - Emilio J. Inarejos Clemente
- Pediatric Diagnostic Radiology Unit, Department of Radiology, Sant Joan de Déu Children’s Hospital, Barcelona, Spain
| | - Fernando M. Gómez
- Interventional Radiology Unit, Department of Radiology, Hospital Universitari I Politècnic La Fe, València, Spain
- Interventional Radiology Unit, Department of Radiology, The Netherlands Cancer Institute, Amsterdam, The Netherlands
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22
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Wahyudi M, Aminata IW, Satria O, Aprilya D. Gigantic aneurysmal bone cyst of the scapula in children treated with left total scapulectomy and humeral suspension: A case report. Int J Surg Case Rep 2024; 116:109339. [PMID: 38320415 PMCID: PMC10850966 DOI: 10.1016/j.ijscr.2024.109339] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/08/2023] [Revised: 01/30/2024] [Accepted: 01/31/2024] [Indexed: 02/08/2024] Open
Abstract
INTRODUCTION AND IMPORTANCE Aneurysmal bone cyst (ABC) is a benign, rapidly growing, osteolytic and could have hemorrhagic lesion. This cyst mostly occurs in females under 20 years old and is located predominantly in the metaphysis of long bones, followed by the pelvis and spine. ABC of the scapula is a rare case, and their treatment still remains controversial. CASE PRESENTATION A 7-year-old girl has had a growing lump on her left shoulder since two years ago. Physical examination revealed local tenderness and a firm and immobile mass, which limit shoulder motion. The radiological and histopathological results confirmed the diagnosis of ABC of the scapula. The patient underwent a left total scapulectomy and humeral suspension reconstruction and was augmented with surgical mesh. There were no signs of postoperative infection or recurrence during the 1-year follow-up. The MSTS score was 24. CLINICAL DISCUSSION There are many options for ABC treatment. Resection of the cyst is the treatment of choice for aggressive tumors (Enneking stage 3) or lesions in less essential bones. In scapula, the ABC treatment with total scapulectomy followed by humeral suspension is an effective method to decrease complications and reduce the risk of recurrence with a good clinical outcome. CONCLUSION ABC of the scapula is a rare case. Tumor resection for aggressive ABC should be considered to decrease the recurrence rate. Total scapulectomy with humeral suspension is an effective method for treating ABC scapula (Enneking stage 3).
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Affiliation(s)
- Muhammad Wahyudi
- Orthopaedic and Traumatology Departement, Fatmawati Central General Hospital Jakarta, Indonesia
| | - Iman Widya Aminata
- Orthopaedic and Traumatology Departement, Fatmawati Central General Hospital Jakarta, Indonesia
| | - Oryza Satria
- Orthopaedic and Traumatology Departement, Fatmawati Central General Hospital Jakarta, Indonesia
| | - Dina Aprilya
- Orthopaedic and Traumatology Departement, Fatmawati Central General Hospital Jakarta, Indonesia.
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23
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Jahangiri P, Roohollahi F, Habibi Z, Mirbolouk MH, Rostami M. Management of aggressive recurrent thoracic spine aneurysmal bone cyst in a 7-year-old male: A case report and review of the literature. Surg Neurol Int 2024; 15:30. [PMID: 38468663 PMCID: PMC10927180 DOI: 10.25259/sni_886_2023] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/31/2023] [Accepted: 12/29/2023] [Indexed: 03/13/2024] Open
Abstract
Background Spinal aneurysmal bone cysts (ABCs) are rare, histologically benign tumors with aggressive behavior, which may cause bone and soft-tissue destruction, particularly affecting neural elements. Management of these tumors, including treatment modalities and follow-up protocols, remains challenging. Case Description A 7-year-old boy presented with chest wall pain persisting for two months before admission, accompanied by progressive mono paresis lasting ten days before admission. Myelopathy signs were evident during the examination. Imaging confirmed a multicystic lesion at the T6 level involving the posterior elements of the vertebra, with significant cord compression. Due to deteriorating neurological function, he underwent urgent laminectomy and neural decompression, followed by subtotal tumor resection. Postoperative histopathological examination confirmed the diagnosis of an ABC, and the patient experienced significant neurological recovery. However, after 21 days, the patient was readmitted to the emergency department with severe paraparesis. Magnetic resonance imaging revealed rapid growth of the residual tumor, leading to cord compression. He underwent aggressive total tumor resection, T6 vertebral body corpectomy, and fixation with pedicle screws and cage insertion. Following the second surgery, prompt neurological recovery occurred. Conclusion This rare case report emphasizes the importance of a close follow-up protocol for spinal ABCs in the pediatric population. It highlights the challenges in managing these tumors and the need for vigilant monitoring to detect and address rapid recurrences.
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Affiliation(s)
- Pedram Jahangiri
- Department of Neurosurgery, Shariati Hospital, Tehran University of Medical Sciences, Tehran, Iran
| | - Faramarz Roohollahi
- Sport Medicine Research Center, Tehran University of Medical Sciences, Yas Hospital, Tehran, Iran
| | - Zohreh Habibi
- Department of Pediatric Neurosurgery, Tehran University of Medical Sciences, Children’s Medical Center, Tehran, Iran
| | | | - Mohsen Rostami
- Spine Center of Excellence, Yas Hospital, Tehran University of Medical Sciences, Tehran, Iran
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El Masry AM, Azmy SI, Mustafa MAR, Abuelhadid MA. Using Denosumab as a Nonsurgical Management of Aneurysmal Bone Cysts in the Pelvis. Clin Orthop Surg 2024; 16:149-156. [PMID: 38304208 PMCID: PMC10825244 DOI: 10.4055/cios22228] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/09/2022] [Revised: 06/09/2023] [Accepted: 06/09/2023] [Indexed: 02/03/2024] Open
Abstract
Background Aneurysmal bone cysts (ABCs) are infrequent, benign, and locally destructive lesions that most commonly occur during the first two decades of life. They usually affect the metaphysis of the long bones, but the pelvis is involved in 8%-12% of the cases. The management of pelvic ABCs is a challenging issue due to difficulties in choosing the appropriate approach, adjacent neurovascular bundles, the risk of intraoperative bleeding with difficulty achieving good hemostasis, and the risk of injury to the hip or sacroiliac joints. Limited data exist concerning the use of denosumab as a non-surgical treatment for pelvic ABCs. Our hypothesis was that denosumab might be an effective and safe solo treatment of cases with ABCs in the pelvis. Methods We retrospectively assessed 20 patients with ABCs in the pelvis, who were treated by denosumab as a solo agent without surgery. Patients were assessed regarding disease control, the incidence of recurrence and non-oncological complications, and functional outcome. Results The mean follow-up period was 38.5 months. Disease control was achieved in 16 patients (80%), with no local recurrence. Tolerable drug-related complications occurred in 15% of cases. The mean Musculoskeletal Tumor Society score was 92.3%. Conclusions Denosumab may provide a reliable option in the nonsurgical treatment of ABCs of pelvic origin with expected lower morbidity than the surgical solution and tolerable complications. Further studies on the safety profile and long-term effects of denosumab especially in skeletally immature patients are required.
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Affiliation(s)
| | - Sherif Ishak Azmy
- Department of Orthopedic Surgery, Faculty of Medicine, Ain Shams University, Cairo, Egypt
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25
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Bakarman KA. Diagnosis and Current Treatment of Aneurysmal Bone Cysts. Cureus 2024; 16:e53587. [PMID: 38449944 PMCID: PMC10915701 DOI: 10.7759/cureus.53587] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 02/04/2024] [Indexed: 03/08/2024] Open
Abstract
The purpose of this review is to increase awareness about the evolution and development of current trends in the diagnosis and treatment of aneurysmal bone cysts (ABCs). ABCs are benign, but locally aggressive bone tumors that mainly affect children. ABCs comprise 1% of all primary bone tumors and occur most frequently during the first two decades of life. The diagnosis is made using a variety of imaging modalities and has the characteristic features of an expansile, radiolucent lesion that is often seen in the metaphyseal region of the bone and has fluid-fluid levels that are apparent on MRI. In the pediatric population, telangiectatic osteosarcoma and unicameral bone cyst (UBC) are the main differential diagnoses of an ABC. Giant cell tumors (GCTs) also include in differential diagnosis, which often manifest in patients older than 15 and do not penetrate the open physis although they develop after the physeal closure. Imaging alone cannot rule out telangiectatic osteosarcoma; therefore, a biopsy is recommended. A variety of treatment options have been described; traditionally, most patients are treated with curettage and bone grafting. Curettage alone, however, usually results in tumor recurrence following excision. A variety of adjuvants have been utilized with varying degrees of effectiveness to reduce the risk of local recurrence. When a cyst is in the pelvis, its location and size are such that surgery is a very risky option. Selective arterial embolization has significantly contributed to the development of effective treatments for these situations. Embolization or radiation, as well as denosumab therapy, are widely used as therapies for ABCs in anatomic locations where surgery would significantly increase morbidity.
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Gettleman BS, Padilla AN, Kumar S, Wren TAL, Miller J, Pawel BR, Tolo VT, Christ AB. Use of Surgical Adjuvants Does Not Decrease Recurrence of Aneurysmal Bone Cysts in Surgical Intervention With Pediatric Patients. J Pediatr Orthop 2024; 44:e79-e83. [PMID: 37815299 PMCID: PMC11195430 DOI: 10.1097/bpo.0000000000002536] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 10/11/2023]
Abstract
BACKGROUND Aneurysmal bone cysts (ABCs) are benign active tumors often requiring intralesional curettage with or without adjuvants. The primary aim of this study was to analyze whether recurrence is influenced by the use of surgical adjuvants in pediatric patients with ABCs. Secondary aims examined recurrence rates based on age, sex, and physeal contact. METHODS A retrospective review was performed at a tertiary pediatric hospital from 2004 to 2020. Inclusion criteria consisted of patients treated surgically for histologically confirmed ABCs with a minimum of 6 months follow-up. Patients with treatment for a recurrent tumor or incomplete records were excluded. Patient demographics, location of the lesion, treatment technique, and incidence of recurrence were collected. Statistical analyses were performed using STATA. RESULTS There were 129 patients (74 males and 55 females) with a mean age of 11.5 ± 4.1 years and an average follow-up of 29.0 ± 25.4 months. The most common locations for ABCs were the femur and tibia. Of the patients, 53.5% had tumors abutting the physis, 28.7% had no physeal contact, and 17.8% had insufficient imaging to evaluate physeal contact. Surgical adjuvants (high-speed burr, coagulation, liquid nitrogen, and/or hydrogen peroxide) were used in 91 of the 129 cases (70.5%). There was no significant difference in recurrence when comparing those who received an adjuvant and those who did not (25.3% vs 23.7%, P = 1.000). Physeal contact was also not significantly associated with recurrence ( P = 0.146). Finally, patients younger than 6 years old were significantly more likely to have recurrence compared with those 6 years old or older (66.7% vs 21.7%, P = 0.007). CONCLUSION Our study found no association between the use of surgical adjuvants and the risk of recurrence after intralesional curettage for ABCs. Although our study did demonstrate that patients 6 years old or younger had an increased rate of recurrence, no significant association was found regarding physeal contact or sex. These data indicate that surgical adjuvant may not affect the recurrence rates of pediatric patients with ABCs. LEVEL OF EVIDENCE Level III. This retrospective review compares rates of recurrence based on the choice of surgical adjuvant.
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Affiliation(s)
| | | | - Shourya Kumar
- Texas A&M School of Engineering Medicine, Houston, TX
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Cevolani L, Staals E, Campanacci L, Dozza B, Martella C, Spinnato P, Di Carlo M, Peta G, Donati DM, Miceli M, Facchini G. Aneurysmal bone cyst: Is selective arterial embolization effective as curettage and bone grafting? J Surg Oncol 2023; 128:1428-1436. [PMID: 37638388 DOI: 10.1002/jso.27422] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/18/2023] [Revised: 07/31/2023] [Accepted: 08/09/2023] [Indexed: 08/29/2023]
Abstract
INTRODUCTION Aneurysmal bone cyst (ABC) is a lytic benign bone lesion representing about 1% of all primary bone tumors. Method to treat ABC's have developed over time. The standard of care cure for ABC has been curettage with or without bone grafting of the defect but is burdened by recurrence rates of approximately 25%-31%. Based on the assumption that ABCs usually supplied by one or more pathological feeding arteries, selective arterial embolization has been described as an adjuvant preoperative procedure to reduce intra-operative hemorrhage, and as primary treatment for lesions in difficult surgical access. In the current study, we therefore asked whether (1) a single or a repeat selective arterial embolization (SAE) for treating ABCs would produce comparable healing rates compared with curettage and bone grafting; (2) evaluated the relationship of recurrence in relation to the site of the cyst, the age, and gender of the patients; and (3) the two techniques differ in term of long-term complication. MATERIAL AND METHODS We retrospectively reviewed 265 patients who underwent curettage and bone grafting or SAE performed at our institute from 1994 to 2018. The diagnosis of ABC was always established with percutaneous CT-guided biopsy or open biopsy. Patients were followed clinically with plain radiographs or CT scan at 3, 6, 9, and 12 months then annually in the absence of symptoms. Treatment success was determined evaluating pre- and postprocedural imaging according to Chang classification. RESULTS Two hundred and nineteen were treated with curettage and bone grafting (curettage group), and 46 with SAE Group. Of the 219 patients treated with Curettage and bone grafting (curettage group), 165 out of 219 (75.3%) experienced bone healing, while local recurrence was observed in 54 cases (24.7%) after 12 months on average (range: 3-120 months) from surgery. After the first SAE, bone ossification was seen in 27 (58.7%), without needing any further treatment. Eleven recurred patients were treated with SAE (four patients need two while seven need three SAE to heal), and eight patients with curettage and bone grafting. Thirty-eight out of 46 (82%) patients experienced bone ossification regardless the number of SAE. The overall rate of local recurrence for all patients was 26.7%. SAE group presented a lower complication rate (6%) where two patients experienced skin necrosis, and one limb-length discrepancies (2% of all cohort). DISCUSSION The use of SAE is an attractive option to treat ABC as it combines on one hand a lower complication rate than curettage and bone grafting, on the other it can be carried out in case of nonresectable ABCs, significantly reducing the size of viable ABC lesions, fostering bone remodeling and mineralization, and most importantly, significantly improving the patient's quality of life.
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Affiliation(s)
- Luca Cevolani
- Unit of 3rd Orthopaedic and Traumatologic Clinic Prevalently Oncologic, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy
| | - Eric Staals
- Unit of 3rd Orthopaedic and Traumatologic Clinic Prevalently Oncologic, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy
| | - Laura Campanacci
- Unit of 3rd Orthopaedic and Traumatologic Clinic Prevalently Oncologic, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy
| | - Barbara Dozza
- Department of Biomedical and Neuromotor Sciences (DIBINEM), Alma Mater Studiorum University of Bologna, Bologna, Italy
| | - Claudia Martella
- Diagnostic and Interventional Radiology, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy
| | - Paolo Spinnato
- Diagnostic and Interventional Radiology, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy
| | - Maddalena Di Carlo
- Diagnostic and Interventional Radiology, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy
| | - Giuliano Peta
- Diagnostic and Interventional Radiology, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy
| | - Davide M Donati
- Unit of 3rd Orthopaedic and Traumatologic Clinic Prevalently Oncologic, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy
| | - Marco Miceli
- Diagnostic and Interventional Radiology, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy
| | - Giancarlo Facchini
- Diagnostic and Interventional Radiology, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy
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Annamalai S, Kandasamy V, Chandrasekaran S, Muthu S. Cross-beam Non-vascular Fibular Graft Reconstruction in the Management of Long Bone Giant Aneurysmal Bone Cyst - A Case Series. J Orthop Case Rep 2023; 13:141-147. [PMID: 38162373 PMCID: PMC10753678 DOI: 10.13107/jocr.2023.v13.i12.4112] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/17/2023] [Revised: 10/03/2023] [Indexed: 01/03/2024] Open
Abstract
Introduction Aneurysmal bone cyst (ABC) is a benign expansile osteolytic lesion, characterized by a blood-filled cavity in the bone. Giant ABC (GABC) is an uncommon condition due to the delayed presentation of an ABC that is difficult to handle when it occurs in long weight-bearing bones due to its aggressive nature. The common treatment relies on total resection of tumor with reconstruction of the resultant defect. Case Report We present the results of 5 cases of GABCs of long bones managed with non-vascularized fibular graft in a cross-beam fashion along with internal fixation. All patients achieved complete consolidation of the lesion by 12 months along with full incorporation of the graft with good-excellent musculoskeletal tumor society scores. None of the patients had recurrence/pathological fracture till 2 years of follow-up. Conclusion We suggest the method of using a non-vascularized fibula graft in a cross-beam fashion to reconstruct the void from the resection of long-bone GABC as a safe, reliable technique with excellent functional and radiological results.
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Affiliation(s)
- Saravanan Annamalai
- Department of Orthopaedics, Thiruvallur Medical College, Thiruvallur, Tamil Nadu, India
| | - Velmurugan Kandasamy
- Department of Orthopaedics, Government Kilpauk Medical College, Chennai, Tamil Nadu, India
| | | | - Sathish Muthu
- Orthopaedic Research Group, Coimbatore, Tamil Nadu, India
- Department of Biotechnology, Faculty of Engineering, Karpagam Academy of Higher Education, Coimbatore, Tamil Nadu, India
- Department of Orthopaedics, Government Medical College, Karur, Tamil Nadu, India
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Ahmad S, Alam I, Khan AQ, Abbas MB, Chowdhry M. Polidocanol sclerotherapy for the treatment of aneurysmal bone cyst, with or without pathological fractures: A prospective, comparative study. J Orthop 2023; 46:143-149. [PMID: 38009081 PMCID: PMC10665595 DOI: 10.1016/j.jor.2023.10.022] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/08/2023] [Revised: 10/18/2023] [Accepted: 10/25/2023] [Indexed: 11/28/2023] Open
Abstract
Introduction Polidocanol sclerotherapy in the treatment of aneurysmal bone cysts (ABCs) with or without pathological fractures has been a topic of debate. While some studies recommend intralesional curettage as the preferred treatment, others suggest intralesional sclerosants as an effective alternative. Material and methods This study evaluates the use of polidocanol-based percutaneous sclerosant therapy in 28 patients with ABCs, of which 8 patients had pathological fracture. The patients received injections every four weeks based on clinical and radiological evaluation of the cyst, and the study assessed the results and complications of the treatment. Results In group A (without pathological fractures), 14 out of 17 (82.4 %) lesions healed without any significant residual lesion. Three out of 17 (17.6 %) patients required further surgical intervention and were treated by curettage and bone grafting, definitively. Minor local inflammatory reaction was observed in 3 patients, which resolved without intervention within 3-4 days. In group B, all the 8 patients (100 %) with pathological fracture in their lesions healed as Grade 1 healing, without any significant residual lesion. One patient developed an episode of anesthesia related complication (delayed recovery) which resolved within an hour, without any residual effect. At final follow up, no recurrence was seen in any patient. Conclusion The study concludes that polidocanol sclerotherapy is an effective treatment option for patients with ABCs, with or without pathological fractures, and has the potential to become the new treatment of choice for pathological fractures due to its low invasiveness, low morbidity, and affordability. However, further research is needed to confirm the efficacy of polidocanol sclerotherapy in larger patient populations and to compare its effectiveness with other treatment options.
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Affiliation(s)
- Sohail Ahmad
- Department of Orthopaedic Surgery, Jawaharlal Nehru Medical College, Aligarh Muslim University, Aligarh, 202001, Uttar Pradesh, India
| | - Intekhab Alam
- Department of Orthopaedic Surgery, Jawaharlal Nehru Medical College, Aligarh Muslim University, Aligarh, 202001, Uttar Pradesh, India
| | - Abdul Qayyum Khan
- Department of Orthopaedic Surgery, Jawaharlal Nehru Medical College, Aligarh Muslim University, Aligarh, 202001, Uttar Pradesh, India
| | - Mohammad Baqar Abbas
- Department of Orthopaedic Surgery, Jawaharlal Nehru Medical College, Aligarh Muslim University, Aligarh, 202001, Uttar Pradesh, India
| | - Madhav Chowdhry
- Department of Orthopaedic Surgery, Jawaharlal Nehru Medical College, Aligarh Muslim University, Aligarh, 202001, Uttar Pradesh, India
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Gaballah M, Worede F, Alexander C, Arkader A, Cahill AM, Acord MR. Novel method to biopsy aneurysmal bone cysts in children. Skeletal Radiol 2023; 52:2479-2486. [PMID: 37191930 DOI: 10.1007/s00256-023-04360-1] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/09/2023] [Revised: 05/02/2023] [Accepted: 05/02/2023] [Indexed: 05/17/2023]
Abstract
PURPOSE The thin and friable septations composing aneurysmal bone cysts (ABC) may be challenging to target for percutaneous biopsy. The purpose of this study was to describe and evaluate a novel method of ABC biopsy using endomyocardial biopsy forceps as an attempt to capture larger fragments of tissue for diagnosis. METHODS AND MATERIALS This was a retrospective study performed over a 17-year period. Patients <18 years old who underwent percutaneous biopsy for a presumed ABC, based on pre-procedure imaging, were included. Medical records were reviewed to identify age, sex, lesion location, biopsy procedure details, complications, and pathology results. A diagnostic biopsy was defined as conclusive histologic confirmation. Inconclusive findings or findings suggestive of but not diagnostic of an ABC were treated as non-diagnostic, even if imaging and clinical findings were characteristic. Biopsy device selection and quantity of tissue obtained was at the discretion of the pediatric interventional radiologist. Fisher's exact test was used to compare the diagnostic yield of standard biopsies to those employing biopsy forceps. RESULTS Twenty-three biopsies were performed in 18 patients (11 female) with a median age of 14.7 years (IQR 10.6-15.6). Lesions were located in the extremities (7, 30.4%), chest (6, 26.1%), pelvis (5, 21.7%), spine (4, 17.4%), and mandible (1, 4.3%). Specimens were obtained using a 13- or 15-gauge bone coring needle (11, 47.8%); 14-, 16-, or 18-gauge soft tissue needle (6, 26.1%); or a combination of bone and soft tissue (4, 17.4%) needles. Endomyocardial biopsy forceps were utilized in 7 cases (30.4%), 2 in which it was the only device used. Overall, a conclusive pathologic diagnosis was made in 13/23 (56.5%) biopsies. Of the diagnostic biopsies, 1 was a unicameral bone cyst and all others were ABCs. No malignancy was identified. Compared to the standard approach, the use of forceps was more likely to result in a diagnostic biopsy (40.0% vs 100.0%, p = 0.008). There were no complications. CONCLUSIONS Endomyocardial biopsy forceps offer an additional, novel technique to biopsy presumed ABCs and may improve diagnostic yield.
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Affiliation(s)
- Marian Gaballah
- Department of Radiology, Children's Hospital of Philadelphia, 3401 Civic Center Blvd, Philadelphia, PA, 19104, USA.
- Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.
| | - Fikadu Worede
- Department of Medicine, Mercy Catholic Medical Center, Darby, PA, 19023, USA
| | - Caitlin Alexander
- Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA
- Division of Anatomic Pathology, Children's Hospital of Philadelphia, 3401 Civic Center Blvd, Philadelphia, PA, 19104, USA
| | - Alexandre Arkader
- Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA
- Division of Orthopedic Surgery, Children's Hospital of Philadelphia, 3401 Civic Center Blvd, Philadelphia, PA, 19104, USA
| | - Anne Marie Cahill
- Department of Radiology, Children's Hospital of Philadelphia, 3401 Civic Center Blvd, Philadelphia, PA, 19104, USA
- Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA
| | - Michael R Acord
- Department of Radiology, Children's Hospital of Philadelphia, 3401 Civic Center Blvd, Philadelphia, PA, 19104, USA
- Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA
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Pena-Burgos EM, Serra-Del Carpio G, Tapia-Viñe M, Iglesias Urraca C, Cordero García JM, Ortiz-Cruz EJ, Pozo-Kreilinger JJ. Primary aneurysmal bone cyst of hands and feet: A series of 14 cases. Ann Diagn Pathol 2023; 66:152169. [PMID: 37295038 DOI: 10.1016/j.anndiagpath.2023.152169] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/17/2023] [Revised: 05/24/2023] [Accepted: 05/25/2023] [Indexed: 06/12/2023]
Abstract
Aneurysmal bone cyst (ABC) is a relatively rare, benign bone tumor that occurs exceptionally in the hands and feet. The objective of this article is to present clinical, radiological, histopathological features and management of a series of 14 primary ABC cases in these unusual locations. Eight cases occurred in hands and six in feet. We present the first reported subungual case to occur in the hand. The average age of the patients was 26 years (range 7 to 49), with half being over the age of twenty at diagnosis. The male to female ration was 9:5. In radiological terms, ABC appeared as an expansive lesion with internal septa and without cortical disruption. Twelve cases displayed the classic multicystic morphology and two cases were of the solid variant. "Blue bone" was detected in 50 % of the specimens. Treatment consisted of curettage, excision, or amputation in all cases. Recurrence rate was observed in 35 % of the cases, with the similar ABC morphology as the original samples. New therapeutic options have been proposed on their own or in combination with surgery to reduce local recurrence rates.
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Affiliation(s)
| | | | - M Tapia-Viñe
- La Paz University Hospital, Radiology Department, Spain
| | | | | | - E J Ortiz-Cruz
- La Paz University Hospital, Orthopaedic Surgery and Traumatology Department, Spain
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Yahaya JJ, Morgan ED, Abraham ZS, Othieno E. Aneurysmal bone cyst of the mandible: a rare case report and literature review. Ann Med Surg (Lond) 2023; 85:5133-5137. [PMID: 37811038 PMCID: PMC10553088 DOI: 10.1097/ms9.0000000000001168] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/18/2023] [Accepted: 08/02/2023] [Indexed: 10/10/2023] Open
Abstract
Introduction and importance Aneurysmal bone cysts (ABCs) are benign, non-neoplastic cystic lesions composed of multiple blood-filled cavities, which are separated by connective tissue septa and they constitute ~6 and 1.5% of all cases of the skull and jaws lesions, respectively. Case presentation Herein, the case of an 80-year-old male with a histologically confirmed diagnosis of ABC involving the body of the left mandible was presented. The patient underwent partial mandiblectomy after confirmation of the diagnosis using orthopantomography, a computed tomography scan, and a tissue biopsy. The patient was free from pain after 1-year of follow-up, and the control orthopantomography showed no evidence of recurrence. This was followed by reconstruction of the healed part of the bone with titanium plates and a piece of bone from the lateral two-thirds of the left femur and thereafter he was cosmetically well. Clinical discussion Patients with ABCs present with expansile and radiolucent bone lesions, which may be associated with displacement and loss of teeth due to alveolar bone erosion. Complete excision of the lesions is mandatory for the prevention of recurrence and increased morbidity. Conclusion ABCs that involve the jaws are extremely rare and are more likely to pose a diagnostic challenge as they are more likely to be confused clinically with other expansile radiolucent bone lesions such as ameloblastoma, osteoblastoma, and giant cell tumor among many others. Also, those with extensive bone matrix formation may sometimes be confused histologically with other bone forming tumors including osteosarcoma. Recurrence is common but it can be avoided or minimized by complete resection of the lesion.
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Affiliation(s)
- James J. Yahaya
- Department of Pathology, School of Health Sciences, Soroti University, Soroti, Uganda
| | - Emmanuel D. Morgan
- Department of Pathology, School of Health Sciences, Soroti University, Soroti, Uganda
| | - Zephania S. Abraham
- Department of Surgery, School of Medicine and Dentistry, University of Dodoma, Dodoma, Tanzania
| | - Emmanuel Othieno
- Department of Pathology, School of Health Sciences, Soroti University, Soroti, Uganda
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Weber KS, Jensen CL, Petersen MM. Sclerotherapy as a primary or salvage procedure for aneurysmal bone cysts: A single-center experience. World J Orthop 2023; 14:698-706. [PMID: 37744716 PMCID: PMC10514711 DOI: 10.5312/wjo.v14.i9.698] [Citation(s) in RCA: 4] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/03/2023] [Revised: 08/17/2023] [Accepted: 08/23/2023] [Indexed: 09/14/2023] Open
Abstract
BACKGROUND Aneurysmal bone cysts (ABC) are benign cystic bone tumors of an osteolytic and locally aggressive nature. As an alternative to the primary treatment of choice, which consists of curettage with bone grafting, alternative treatment methods with promising results have been described. At our department, we have, in recent years, used percutaneous sclerotherapy with polidocanol. The objective of this study was to identify the healing rate and safety of sclerotherapy with polidocanol. AIM To identify the efficacy and safety of sclerotherapy with polidocanol in primary and recurrent ABC. METHODS Twenty-two consecutive patients (median age 12.5 years; range 1-27) with 23 ABCs treated with sclerotherapy with polidocanol from 2016-2021 were included retrospectively. Eleven patients (48%) had undergone different forms of previous treatment with recurrence. Under general anesthesia and fluoroscopic guidance, repeated percutaneous injections of 4mg polidocanol/kg body weight were performed. Through review of the electronic medical records, the following were identified: healing and recurrence rate, number of treatments, gender, age, comorbidity, location of the tumor and side effects / complications, as well as any previous surgery for ABC. The median length of radiographic follow-up was 19.5 mo. RESULTS All ABCs except one (96%) showed healing or stable disease after a median of 4 (range 1-8) injections. Complete clinical and radiographic healing was observed in 16 cysts (70%), while partial radiographic healing with resolution of pain was seen in 6 cases (26%) and considered as stable disease. The cyst that failed to heal had previously undergone curettage twice with recurrence. One patient with a large pelvic ABC experienced, right after two injections, a sudden drop in blood pressure, which could quickly be reversed. One patient with a juxtaphyseal ABC in the femoral neck showed a minor limb length discrepancy because of deformity. Beyond that, no complications were observed. CONCLUSION Percutaneous sclerotherapy with polidocanol appears to be a safe alternative for treatment of aneurysmal bone cysts. In our series of both primary and recurrent cysts, it showed the ability to achieve healing or stable disease in 22 of 23 cases (96%). Further studies are needed to decide if this provides a long-lasting effect.
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Castro Castro J, Villa Fernández JM, Agulleiro Díaz JP. Craniofacial fibrous dysplasia and aneurismal bone cyst in a patient with McCune-Albright syndrome. A case report and review of the literature. NEUROCIRUGIA (ENGLISH EDITION) 2023; 34:268-272. [PMID: 36192338 DOI: 10.1016/j.neucie.2022.09.002] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 01/30/2022] [Revised: 05/17/2022] [Accepted: 06/23/2022] [Indexed: 06/16/2023]
Abstract
McCune-Albright syndrome (MAS) is a rare heterogeneous genetic disorder that is characterized by a triad of polyostotic fibrous dysplasia (FD), café au lait spots (CAL), and multiple hyperfunctional endocrinopathies. In general, it is diagnosed clinically. From the triads, 2 of the findings are enough to make the diagnosis. Craniofacial fibrous dysplasia is a term that is used to describe the fibrous dysplasia, which was localized at the craniofacial skeleton and is common in MAS patients. Aneurysmal bone cyst (ABC) is a rare non-neoplastic bone lesion that involves mostly the long bones and vertebrae and may occur very rarely in the craniofacial bones. ABCs may occur as secondary bony pathologies in association with various benign and malignant bone tumors and with fibrous dysplasia. Secondary ABC occurring in craniofacial FD is extremely rare. We present the case of a 21-year-old patient treated at our center for a right orbital aneurysmal bone cyst associated with MAS and provide a review of the relevant literature.
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Affiliation(s)
- Julián Castro Castro
- Servicio de Neurocirugía, Complexo Hospitalario Universitario de Ourense, Instituto de Investigación Sanitaria Galicia Sur, Ourense, Spain.
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Korkoman AJ, AlQahtani AA. Aneurysmal bone cyst of the clavicle: A case report. Int J Surg Case Rep 2023; 110:108755. [PMID: 37678034 PMCID: PMC10509914 DOI: 10.1016/j.ijscr.2023.108755] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/05/2023] [Revised: 08/23/2023] [Accepted: 08/25/2023] [Indexed: 09/09/2023] Open
Abstract
INTRODUCTION An aneurysmal bone cyst (ABC) is a locally aggressive primary bone neoplasm. ABC of the clavicle is rare with only a few reported cases in the literature. PRESENTATION OF CASE We report the case of a 10-year-old boy who presented with an ABC at the right acromial end of the clavicle. The patient underwent intralesional curettage and allogenic bone grafting. Moreover, the patient's arm was placed in a sling for 2 weeks postoperatively. The patient had a satisfactory outcome after 11 years, with excellent Toronto Extremity Salvage, Quick-Dash, and Musculoskeletal Tumor Society scores. DISCUSSION Clavicular ABCs are uncommon. Early diagnosis helps to prevent pathological fractures. Adjuvant therapies might help decrease recurrence. CONCLUSION ABC should be considered an important differential diagnosis for clavicular swelling and masses. The best results can be achieved using curettage and void-filled bone grafts. LEVEL OF EVIDENCE: 4
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Affiliation(s)
| | - Abdulaziz Ali AlQahtani
- Orthopedic Surgery Department at the Prince Sultan Military Medical City, Riyadh, Saudi Arabia
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Gajewski CR, Alhushki W, Benhaim P, Bernthal NM, Wessel L. Approach to the Treatment of Expansile Metacarpal Aneurysmal Bone Cysts: A Report of Two Cases. JOURNAL OF HAND SURGERY GLOBAL ONLINE 2023; 5:689-694. [PMID: 37790824 PMCID: PMC10543794 DOI: 10.1016/j.jhsg.2023.05.004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/28/2023] [Accepted: 05/06/2023] [Indexed: 10/05/2023] Open
Abstract
The two cases presented demonstrate the management of aneurysmal bone cysts of the metacarpal, which destroyed the normal bone architecture. Treatment of both cases included wide resection and metacarpal reconstruction with an intercalary fibular allograft. Denosumab use contrasts these two cases and is helpful in reestablishment of a cortical rim for fixation in the absence of a 1-cm margin proximally or distally to preserve the native carpometacarpal and metacarpophalangeal joints. Surgical resection and allograft reconstruction is a viable treatment for expansile metacarpal aneurysmal bone cysts, and neoadjuvant denosumab has utility in creating an ossified margin for fixation.
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Affiliation(s)
| | | | | | | | - Lauren Wessel
- UCLA Department of Orthopaedic Surgery, Los Angeles, CA
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Smolle MA, Roessl V, Leithner A. Effect of Local Adjuvants Following Curettage of Benign and Intermediate Tumours of Bone: A Systematic Review of the Literature. Cancers (Basel) 2023; 15:4258. [PMID: 37686534 PMCID: PMC10487159 DOI: 10.3390/cancers15174258] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/14/2023] [Revised: 08/24/2023] [Accepted: 08/24/2023] [Indexed: 09/10/2023] Open
Abstract
Local adjuvants are used upon intralesional resection of benign/intermediate bone tumours, aiming at reducing the local recurrence (LR) rate. However, it is under debate whether, when and which local adjuvants should be used. This PRISMA-guideline based systematic review aimed to analyse studies reporting on the role of adjuvants in benign/intermediate bone tumours. All original articles published between January 1995 and April 2020 were potentially eligible. Of 344 studies identified, 58 met the final inclusion criteria and were further analysed. Articles were screened for adjuvant and tumour type, follow-up period, surgical treatment, and development of LR. Differences in LR rates were analysed using chi-squared tests. Altogether, 3316 cases (10 different tumour entities) were analysed. Overall, 32 different therapeutic approaches were identified. The most common were curettage combined with high-speed burr (n = 774; 23.3%) and high-speed burr only (n = 620; 18.7%). The LR rate for studies with a minimum follow-up of 24 months (n = 30; 51.7%) was 12.5% (185/1483), with the highest rate found in GCT (16.7%; 144/861). In comparison to a combination of curettage, any adjuvant and PMMA, the sole application of curettage and high-speed burr (p = 0.015) reduced the LR rate in GCT. The overall complication rate was 9.6% (263/2732), which was most commonly attributable to postoperative fracture (n = 68) and osteoarthritis of an adjacent joint during follow-up (n = 62). A variety of adjuvants treatment options are reported in the literature. However, the most important step remains to be thorough curettage, ideally combined with high-speed burring.
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Affiliation(s)
- Maria Anna Smolle
- Department of Orthopaedics and Trauma, Medical University of Graz, Auenbruggerplatz 5, 8036 Graz, Austria; (V.R.); (A.L.)
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Kotaka S, Fujiwara Y, Ota R, Manabe H, Adachi N. The Efficacy of Denosumab in Treating Spinal Aneurysmal Bone Cyst: A Case Report. Cureus 2023; 15:e39954. [PMID: 37415990 PMCID: PMC10319940 DOI: 10.7759/cureus.39954] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 06/04/2023] [Indexed: 07/08/2023] Open
Abstract
The optimal treatment for aneurysmal bone cysts (ABCs) of the spine remains controversial. No treatment guidelines exist for the use of denosumab in aneurysmal bone cysts. In this report, we describe the results from a representative case and compare our experience with those of previously published reports. A 38-year-old male was referred for pain in the lower back and left leg. Radiographs and a needle biopsy specimen revealed a lumbar aneurysmal bone cyst, which was treated with denosumab chemotherapy. The pain in the lower back and left leg gradually improved, and at 16 weeks, the symptoms had resolved. Once a satisfactory local effect was achieved, denosumab therapy was discontinued. However, the erosive lesion subsequently expanded. After re-initiation of treatment, there was no subsequent evidence of recurrence. Single-therapy denosumab is an option for aneurysmal bone cysts. However, recurrences have been documented after denosumab termination, and the timing for cessation of denosumab is controversial.
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Affiliation(s)
- Shinji Kotaka
- Orthopaedic and Microscopic Spine and Spinal Cord Surgery Center, Hiroshima City North Medical Center Asa Citizens Hospital, Hiroshima, JPN
| | - Yasushi Fujiwara
- Orthopaedic and Microscopic Spine and Spinal Cord Surgery Center, Hiroshima City North Medical Center Asa Citizens Hospital, Hiroshima, JPN
| | - Ryo Ota
- Orthopaedic and Microscopic Spine and Spinal Cord Surgery Center, Hiroshima City North Medical Center Asa Citizens Hospital, Hiroshima, JPN
| | - Hideki Manabe
- Department of Orthopaedic Surgery, Hiroshima Hiramatsu Hospital, Hiroshima, JPN
| | - Nobuo Adachi
- Department of Orthopaedic Surgery, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima, JPN
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Razii N, Docherty LM, Halai M, Mahendra A, Gupta S. Injectable Synthetic Beta-Tricalcium Phosphate/Calcium Sulfate (GeneX) for the Management of Contained Defects Following Curettage of Benign Bone Tumours. Curr Oncol 2023; 30:3697-3707. [PMID: 37185394 PMCID: PMC10137257 DOI: 10.3390/curroncol30040281] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/25/2023] [Revised: 03/17/2023] [Accepted: 03/20/2023] [Indexed: 03/29/2023] Open
Abstract
Benign and low-grade malignant bone tumours are often treated with curettage and filling of the resultant defect using any of a number of materials, including autologous bone grafts, allografts, or synthetic materials. The objective of this study was to report our experience using a synthetic bone graft substitute in these patients. Ten consecutive cases (four males, six females; mean age, 36 years) of benign bone tumours were treated surgically at a tertiary musculoskeletal oncology centre, between 2019 and 2021. Following curettage, the contained defects were managed with injectable beta-tricalcium phosphate/calcium sulfate (GeneX; Biocomposites Ltd., Keele, UK). The desired outcomes were early restoration of function and radiographic evidence of healing. No other graft materials were used in any of the patients. The mean follow-up was 24 months (range, 20–30 months). All patients in this series (100%) demonstrated radiographic evidence of healing and resumed their daily living activities. There were no tumour recurrences and no complications were encountered with the use of GeneX. In patients with contained defects following curettage of benign bone tumours, we found GeneX to be a safe and effective filling agent. These findings contrast with some existing studies that have reported local complications with the use of injectable beta-tricalcium phosphate/calcium sulfate.
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40
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Minimally Invasive Treatment of a Cervical Aneurysmal Bone Cyst Through Percutaneous Doxycycline-Albumin Foam Injection. Cardiovasc Intervent Radiol 2023; 46:408-410. [PMID: 36319714 PMCID: PMC10014782 DOI: 10.1007/s00270-022-03292-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/04/2022] [Accepted: 09/24/2022] [Indexed: 11/05/2022]
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Vanderniet JA, Tsinas D, Wall CL, Girgis CM, London K, Keane C, Briody J, Hibbert S, Poon M, Padhye B, Biggin A, Dalla-Pozza L, Gray RJ, Munns CF. Surgical Management and Denosumab for Aneurysmal Bone Cysts of the Spine in an Australian Tertiary Paediatric Centre. Calcif Tissue Int 2023; 112:592-602. [PMID: 36810677 DOI: 10.1007/s00223-023-01068-1] [Citation(s) in RCA: 5] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/10/2022] [Accepted: 01/30/2023] [Indexed: 02/23/2023]
Abstract
Aneurysmal bone cysts (ABC) are rare osteolytic, benign but often locally aggressive tumours of the long bones or vertebrae. For spinal ABC, surgical management, embolisation or sclerotherapy alone often carry high morbidity and/or high recurrence rates. Interruption of receptor activator of nuclear factor-kappa B ligand (RANKL) signalling holds promise as an effective therapeutic strategy for these tumours. We aimed to review the approach to surgical management and evaluate the efficacy and safety of denosumab for ABC of the spine in children. Retrospective review of 7 patients treated with denosumab using a standardised protocol for ABC of the spine in a tertiary paediatric centre. Surgical intervention was only conducted if there was spinal instability or significant neurological impairment. Denosumab 70 mg/m2 was given 4-weekly for at least 6 months, followed by 2 doses of zoledronate 0.025 mg/kg, aiming to prevent rebound hypercalcaemia. All patients achieved stability of the spine and resolution of neurological impairment, if present. Six patients achieved metabolic remission and have ceased denosumab without recurrence to date; the other showed clinical and radiological improvement without complete metabolic remission. Three patients developed symptomatic hypercalcaemia 5-7 months after cessation of denosumab, requiring additional bisphosphonate treatment. We present our algorithm for the surgical and medical management of paediatric spinal ABC. Denosumab produced a radiological and metabolic response in all patients, with complete remission in most. Follow-up time was not long enough to evaluate the endurance of response after cessation in some patients. Incidence of rebound hypercalcaemia in this paediatric cohort was high, prompting a change to our protocol.
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Affiliation(s)
- Joel A Vanderniet
- Institute of Endocrinology and Diabetes, The Children's Hospital at Westmead, Locked Bag 4001, Westmead, Sydney, NSW, 2145, Australia.
| | - Dionysios Tsinas
- Department of Orthopaedics, The Children's Hospital at Westmead, Sydney, NSW, Australia
| | - Christie-Lee Wall
- Institute of Endocrinology and Diabetes, The Children's Hospital at Westmead, Locked Bag 4001, Westmead, Sydney, NSW, 2145, Australia
| | - Christian M Girgis
- Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia
- Department of Endocrinology, Royal North Shore Hospital, Sydney, NSW, Australia
- Department of Diabetes and Endocrinology, Westmead Hospital, Sydney, NSW, Australia
| | - Kevin London
- Department of Nuclear Medicine, The Children's Hospital at Westmead, Sydney, NSW, Australia
| | - Corinne Keane
- Department of Orthopaedics, The Children's Hospital at Westmead, Sydney, NSW, Australia
| | - Julie Briody
- Department of Nuclear Medicine, The Children's Hospital at Westmead, Sydney, NSW, Australia
| | - Sally Hibbert
- Department of Dentistry, The Children's Hospital at Westmead, Sydney, NSW, Australia
- Department of Paediatric Dentistry, Westmead Centre for Oral Health, Sydney, NSW, Australia
| | - Myra Poon
- Institute of Endocrinology and Diabetes, The Children's Hospital at Westmead, Locked Bag 4001, Westmead, Sydney, NSW, 2145, Australia
| | - Bhavna Padhye
- Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia
- Cancer Centre for Children, The Children's Hospital at Westmead, Sydney, NSW, Australia
| | - Andrew Biggin
- Institute of Endocrinology and Diabetes, The Children's Hospital at Westmead, Locked Bag 4001, Westmead, Sydney, NSW, 2145, Australia
- Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia
| | - Luciano Dalla-Pozza
- Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia
- Cancer Centre for Children, The Children's Hospital at Westmead, Sydney, NSW, Australia
| | - Randolph J Gray
- Department of Orthopaedics, The Children's Hospital at Westmead, Sydney, NSW, Australia
| | - Craig F Munns
- Child Health Research Centre, The University of Queensland, Brisbane, QLD, Australia
- Mayne Academy of Paediatrics, The University of Queensland, Brisbane, QLD, Australia
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Abstract
PURPOSE OF REVIEW Aneurysmal bone cysts are rare, locally aggressive bone tumors. Optimal treatment of ABCs is still matter of debate as therapies including sclerotherapy, selective arterial embolization and systemic treatment with denosumab are increasingly utilized, in addition to or instead of traditional curettage. The purpose of this review is to discuss current concepts and difficulties in diagnosing and treating primary ABCs, based on latest available literature. RECENT FINDINGS In diagnostics, multiple new fusion partners of USP-6 have been described on next-generation sequencing specifically for primary ABCs. In a recent systematic review, failure rates of percutaneous injections and surgery were comparable. In a literature review, the use of denosumab seemed effective but resulted in multiple cases of severe hypercalcemia in children. SUMMARY Accurately diagnosing primary ABC is crucial for treatment decisions. Curettage remains a valid treatment option, especially with adjuvant burring, autogenous bone grafting and phenolization. Percutaneous sclerotherapy represents a solid alternative to surgery, with polidocanol showing good results in larger studies. Systematic therapy with denosumab exhibits favorable results but should be reserved in the pediatric population for unresectable lesions, as it may result in severe hypercalcemia in children. When selecting a treatment option, localization, stability and safety should be considered.
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Do's and Don'ts in Primary Aneurysmal Bone Cysts of the Proximal Femur in Children and Adolescents: Retrospective Multicenter EPOS Study of 79 Patients. J Pediatr Orthop 2023; 43:37-45. [PMID: 36102541 PMCID: PMC9746336 DOI: 10.1097/bpo.0000000000002267] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Abstract
BACKGROUND Aneurysmal bone cysts (ABC) are rare benign cystic bone tumors, generally diagnosed in children and adolescents. Proximal femoral ABCs may require specific treatment strategies because of an increased pathologic fracture risk. As few reports are published on ABCs, specifically for this localization, consensus regarding optimal treatment is lacking. We present a large retrospective study on the treatment of pediatric proximal femoral ABCs. METHODS All eligible pediatric patients with proximal femoral ABC were included, from 11 tertiary referral centers for musculo-skeletal oncology (2000-2021). Patient demographics, diagnostics, treatments, and complications were evaluated. Index procedures were categorized as percutaneous/open procedures and osteosynthesis alone. Primary outcomes were: time until full weight-bearing and failure-free survival. Failure was defined as open procedure after primary surgery, >3 percutaneous procedures, recurrence, and/or fracture. Risk factors for failure were evaluated. RESULTS Seventy-nine patients with ABC were included [mean age, 10.2 (±SD4.0) y, n=56 male]. The median follow-up was 5.1 years (interquartile ranges=2.5 to 8.8).Index procedure was percutaneous procedure (n=22), open procedure (n=35), or osteosynthesis alone (n=22). The median time until full weight-bearing was 13 weeks [95% confidence interval (CI)=7.9-18.1] for open procedures, 9 weeks (95% CI=1.4-16.6) for percutaneous, and 6 weeks (95% CI=4.3-7.7) for osteosynthesis alone ( P =0.1). Failure rates were 41%, 43%, and 36%, respectively. Overall, 2 and 5-year failure-free survival was 69.6% (95% CI=59.2-80.0) and 54.5% (95% CI=41.6-67.4), respectively. Risk factors associated with failure were age younger than 10 years [hazard ratios (HR)=2.9, 95% CI=1.4-5.8], cyst volume >55 cm 3 (HR=1.7, 95% CI=0.8-2.5), and fracture at diagnosis (HR=1.4, 95% CI=0.7-3.3). CONCLUSIONS As both open and percutaneous procedures along with osteosynthesis alone seem viable treatment options in this weight-bearing location, optimal treatment for proximal femoral ABCs remains unclear. The aim of the treatment was to achieve local cyst control while minimizing complications and ensuring that children can continue their normal activities as soon as possible. A personalized balance should be maintained between undertreatment, with potentially higher risks of pathologic fractures, prolonged periods of partial weight-bearing, or recurrences, versus overtreatment with large surgical procedures, and associated risks. LEVEL OF EVIDENCE Level IV, therapeutic study.
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Muacevic A, Adler JR, Young SM, Ray J, Shah A, Conklin MJ. A Review of Pediatric Heel Pain. Cureus 2023; 15:e34228. [PMID: 36852370 PMCID: PMC9960861 DOI: 10.7759/cureus.34228] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 01/25/2023] [Indexed: 01/28/2023] Open
Abstract
The objective of this review article is to provide orthopaedic surgeons and general practitioners a reference and guidance for the evaluation and workup of heel pain in pediatric patients. The authors performed a comprehensive literature search to review the etiologies and management of heel pain in patients <18 years of age. Relevant studies in Medline/PubMed and EMBASE were searched from inception to March 3, 2022 using medical subject headings and text words without limitations on language or study type. The initial search utilized the following Boolean operators: (children) AND (heel pain); (pediatric) AND (heel pain). Heel pain in the pediatric population is usually a benign condition. Sever's apophysitis is the most common etiology of heel pain in pediatric patients. Most causes of heel pain in the pediatric population do not require imaging or extensive workup. However, providers must maintain a high index of suspicion for symptoms that could indicate a more severe pathology.
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Affiliation(s)
- Alexander Muacevic
- Orthopaedic Surgery, University of Alabama at Birmingham School of Medicine, Birmingham, USA
| | - John R Adler
- Orthopaedic Surgery, University of Alabama at Birmingham School of Medicine, Birmingham, USA
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45
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Giantini-Larsen AM, Chakravarthy VB, Barzilai O, Newman WC, Wexler L, Bilsky MH. The role of neoadjuvant denosumab in the treatment of aneurysmal bone cysts: a case series and review of the literature. J Neurosurg Pediatr 2022; 30:547-554. [PMID: 36282899 DOI: 10.3171/2022.8.peds22314] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/23/2022] [Accepted: 08/22/2022] [Indexed: 11/06/2022]
Abstract
OBJECTIVE Aneurysmal bone cysts (ABCs) are benign cystic lesions most commonly occurring in the long bones of pediatric patients. Spinal ABCs may be difficult to resect given their invasive, locally destructive nature, proximity to critical structures such as the spinal cord, and their intrinsic hypervascularity, for which complete embolization is often constrained by radiculomedullary segmental feeders. Denosumab, a monoclonal antibody that binds the receptor activator of nuclear factor kappa B (NF-κB) ligand, has been utilized in the treatment of ABCs most often as a rescue therapy for recurrent disease. Here, the authors present 3 cases of neoadjuvant denosumab use in surgically unresectable tumors to calcify and devascularize the lesions, allowing for safer, more complete resection. METHODS This is a single-center, retrospective case series treated at a tertiary care cancer center. The authors present 3 cases of spinal ABC treated with neoadjuvant denosumab. RESULTS All 3 patients experienced calcification, size reduction, and a significant decrease in the vascularity of their ABCs on denosumab therapy. None of the patients developed new neurological deficits while on denosumab. Subsequently, all underwent resection. One patient continued denosumab during the immediate postoperative period because a subtotal resection had been performed, with stabilization of the residual disease. No complications were associated with denosumab administration. CONCLUSIONS The use of denosumab in unresectable ABCs can cause calcification and devascularization, making safe resection more likely.
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Affiliation(s)
- Alexandra M Giantini-Larsen
- Departments of1Neurosurgery and
- 2Department of Neurosurgery, Weill Cornell Medical Center, New York, New York
| | | | | | | | - Leonard Wexler
- 3Pediatrics, Memorial Sloan Kettering Cancer Center, New York; and
| | - Mark H Bilsky
- Departments of1Neurosurgery and
- 2Department of Neurosurgery, Weill Cornell Medical Center, New York, New York
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Aljamaan YM, Alhathloul HS, Aleissa SI, Abaalkhail MS, Alhelal FH, Konbaz FM. Unusual presentation of aneurysmal bone cyst with scoliosis: a case report. J Med Case Rep 2022; 16:446. [PMID: 36443749 PMCID: PMC9706952 DOI: 10.1186/s13256-022-03685-0] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/17/2022] [Accepted: 11/20/2022] [Indexed: 11/30/2022] Open
Abstract
BACKGROUND Aneurysmal bone cysts are benign bone tumors that not uncommonly involves the spine. However, this involvement can cause scoliosis, albeit rarely. This report focuses on the importance of proper management for complete tumor resection to prevent recurrence and spinal deformity. CASE PRESENTATION A 12-year-old Middle Eastern boy, with a history of T11 aneurysmal bone cyst resection and bone grafting carried out at another hospital, presented with spine deformity of 4 months' duration. The deformity was not associated with pain or neurological deficit. A whole-spine magnetic resonance imaging with contrast confirmed the recurrence of the aneurysmal bone cyst. Posterior spinal instrumentation with corpectomy of T11 was then performed, and confirmed with histopathology the recurrence of aneurysmal bone cyst. Two years post-corpectomy, deformity correction was done from T5-L4. CONCLUSION Management of aneurysmal bone cysts requires meticulous planning and full excision to prevent recurrence, especially in the growing spine. If neglected, it can cause major spinal deformities and cord compression, which places a medical burden on the patient and family. To avoid such complications, treating aneurysmal bone cysts along with scoliosis correction can prevent deformity progression.
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Affiliation(s)
- Yousef M Aljamaan
- Department of Orthopedic Surgery, College of Medicine, King Fahad Hospital of the University, Imam Abdulrahman Bin Faisal University, Dammam, Saudi Arabia.
| | - Hisham S Alhathloul
- Department of Orthopedics, Armed Forces Hospital, King Abdulaziz Air Base, Dhahran, Saudi Arabia
| | - Sami I Aleissa
- Department of Orthopedics, Ministry of The National Guard-Health Affairs, Riyadh, Saudi Arabia.,King Abdullah International Medical Research Center, Riyadh, Saudi Arabia.,King Saud Bin Abdulaziz University for Health Science, Riyadh, Saudi Arabia
| | - Majid S Abaalkhail
- Department of Orthopedics, Ministry of The National Guard-Health Affairs, Riyadh, Saudi Arabia.,King Abdullah International Medical Research Center, Riyadh, Saudi Arabia.,King Saud Bin Abdulaziz University for Health Science, Riyadh, Saudi Arabia
| | - Fahad H Alhelal
- Department of Orthopedics, Ministry of The National Guard-Health Affairs, Riyadh, Saudi Arabia.,King Abdullah International Medical Research Center, Riyadh, Saudi Arabia.,King Saud Bin Abdulaziz University for Health Science, Riyadh, Saudi Arabia
| | - Faisal M Konbaz
- Spine Surgery Section, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia
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Yasin M, Alisi M, Hammad Y, Samarah O, Hassan FA. Treatment of Aneurysmal Bone Cysts by Minimally Invasive Curettage and Allogenic Bone Impaction Grafting: Mid-to Long-term Results. Orthop Surg 2022; 14:3171-3177. [PMID: 36250563 PMCID: PMC9732591 DOI: 10.1111/os.13511] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/24/2021] [Revised: 08/02/2022] [Accepted: 08/23/2022] [Indexed: 12/14/2022] Open
Abstract
OBJECTIVE Recurrence is the main hinderance in treatment of aneurysmal bone cysts (ABCs). Therefore, several treatment modalities and adjuvant therapies have been proposed. In this study, we aim to evaluate the long-term effectiveness of percutaneous curettage and allogenic bone grafting as a new, minimally invasive modality in treating ABCs. METHODS We performed a retrospective review of the medical records of patients diagnosed with primary ABCs at a university hospital over a 10-year period (2000-2010). We selected all patients who were diagnosed with primary ABC in the extremities and pelvis, treated with the same surgical procedure, and were followed for at least 2 years postoperatively. All patients underwent the same procedure of percutaneous curettage and impaction of allogenic pulverized fine bone fragments (harvested from locally stored femoral heads) mixed with autologous bone marrow aspirate from the iliac bone. We reported patient's characteristics (age and gender), site and size of the lesion, presenting symptoms, Capanna classification, follow-up duration, and post-operative complications. Assessment of cyst healing was based on the appearance on radiographs according to the modified Neer classification. RESULTS Nineteen patients were included in this study; 10 patients were males and nine were females. The mean age was 9.6 years (range 3-15). The location of the lesions was as follows: femur (eight), tibia (four), pelvis (four), proximal humerus (one), distal radius (one), and calcaneus (one). The most common presenting symptom was pain in the involved area. Pathological fracture was the presenting feature in two patients. The mean follow-up duration was 6.4 years (range 2-18). The earliest radiological sign of incorporation of the allograft was seen at 3 months after surgery. All patients showed bone remodeling and radiographic resolution (classified as either A or B on the modified Neer classification) of their cystic lesions within 6 months. No local recurrence, infection, or pathological fractures occurred during the follow-up period. CONCLUSION Percutaneous curettage and impaction of allogenic bone graft mixed with autogenic bone marrow aspirate is an efficient, minimally invasive, reproducible, and affordable procedure for the treatment of primary ABCs.
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Affiliation(s)
- Mohamad Yasin
- Department of Special Surgery, Division of Orthopaedics, School of MedicineThe University of JordanAmmanJordan
| | - Mohammed Alisi
- Department of Special Surgery, Division of Orthopaedics, School of MedicineThe University of JordanAmmanJordan,Faculty of MedicineIslamic University of GazaGazaPalestine,Faculty of MedicineAl‐Azhar UniversityGazaPalestine
| | - Yazan Hammad
- Department of Special Surgery, Division of Orthopaedics, School of MedicineThe University of JordanAmmanJordan
| | - Omar Samarah
- Department of Special Surgery, Division of Orthopaedics, School of MedicineThe University of JordanAmmanJordan
| | - Freih Abu Hassan
- Department of Special Surgery, Division of Orthopaedics, School of MedicineThe University of JordanAmmanJordan
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48
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Femoral Reconstruction Using Long Tibial Autograft After Resection of Giant Aneurysmal Bone Cyst. Tech Orthop 2022. [DOI: 10.1097/bto.0000000000000608] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/05/2023]
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Stewart HL. Aneurysmal bone cysts as a diagnostic consideration in juvenile patients: Considerations from humans and animals. EQUINE VET EDUC 2022. [DOI: 10.1111/eve.13717] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Affiliation(s)
- Holly L. Stewart
- Department of Clinical Sciences, College of Veterinary Medicine and Biomedical Sciences Colorado State University Fort Collins Colorado USA
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50
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Castro Castro J, Villa Fernández JM, Agulleiro Díaz JP. Displasia fibrosa craneofacial y quiste óseo aneurismático en una paciente con síndrome de McCune-Albright. Presentación de un caso y revisión de la literatura. Neurocirugia (Astur) 2022. [DOI: 10.1016/j.neucir.2022.06.004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/15/2022]
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