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Case Report
2025
:4;
100566
doi:
10.1016/j.jorep.2025.100566

Real time 3D O-arm based extremity bone tumour surgery

, , , ,
a Department of Musculoskeletal Surgical Oncology, Rajiv Gandhi Cancer Institute & Research Centre, New Delhi, India
b Department of Pathology, Rajiv Gandhi Cancer Institute & Research Centre, New Delhi, India

⁎Corresponding author: Vaibhav Sahu. sahu.vaibhav@rgcirc.org

Received: , Accepted: ,
The Authors
Disclaimer:
This article was originally published by Reed Elsevier India Pvt. Ltd. and was migrated to Scientific Scholar after the change of Publisher.

Abstract

Abstract

Aneurysmal bone cyst (ABC) within the diaphyseal cortex is a very rare finding because most of the ABCs are of metaphyseal origin and within medullary cavity. Imaging of the cortical ABC may be inconclusive; therefore role of biopsy is extremely crucial in pre-operative period for the proper management and surgical plan.

A 59-year-old female came to us with complaints of pain and swelling right arm since last 6 months. Radiograph of the involved arm was suggestive of cystic intracortical lesion without fracture. Core needle biopsy report came out to be aneurysmal bone cyst. O-arm and Computer navigation assisted en-bloc resection of the lesion was done as it was involving around half of the cortex and there was risk of iatrogenic fracture in intraoperative and postoperative period if done without navigation.

O-arm provides real time 3D images which were quite helpful in navigation assisted resection of the lesion so that negative margins can be obtained and maximum cortex of the bone can be saved.

Keywords

Aneurysmal bone cyst
Computer navigation
O-arm
Bone tumour
1

1 Introduction

The term 'aneurysmal bone cyst' is a misnomer, as these lesions are neither aneurysmal nor genuinely cystic, lacking a true endothelial lining.1 Rather, these lesions are benign and expansile which create spaces within the bone that become filled with blood, and are characterized by a lining composed of proliferative fibroblasts, giant cells, and trabecular bone. Therefore, they are defined as ‘active’ benign lesion. About 90 % of these lesions occur in patients younger than 30 years with the median age of 11.1 years.2 The male to female ratio of ABC is 1.8:1.2 The annual incidence of ABC is 1.4 per 100,000 individuals.3,4 ABC has predilection for metaphysis of long bones and usually involve medullary cavity.5 These lesions are generally solitary and eccentric in location. Recurrence rates of the aneurysmal bone cyst in the literature has been documented between 10 and 60 % following curettage with or without adjuvant therapy like phenol or burr.6–10

Bone marrow oedema has been reported in association with various benign primary bone lesions; however, to our knowledge, it has only been described on two occasions in the bone marrow adjacent to an aneurysmal bone cyst.11,12

The navigation system aids surgeons in performing more precise procedures and has become a standard tool in spine and joint surgeries, with increasing use in tumor surgeries. In cases of ABC in the humeral diaphyseal cortex, where exact tumor removal and preservation of bony and neurovascular structures are critical, the navigation system offers a potential solution. However, there is limited Indian literature reporting the bone and soft tissue tumor surgeries using the O-arm and computer navigation system. We present a case of intracortical humeral ABC with intramedullary oedema managed with O-arm and navigation guided resection.

2

2 Case presentation

A 59-year old female presented to our opd with the complaints of pain and swelling of the right arm since last 6 months. The patient had complaint of pain in the movement of the right shoulder joint especially in abduction. Thorough clinical examination was done and the patient was advised for radiographic studies. X-ray images were suggestive of intracortical lytic lesion over lateral aspect of shaft of humerus measuring 4x2 cm. Magnetic resonance imaging (MRI) of the right arm was done which was suggestive of well defined exophytic lesion in outer cortex of mid shaft of humerus measuring 4× 2 × 1.5 cm (CC x AP x TR) (Fig. 1). The lesion appears hypointense on T1 and hyperintense on T2W images with fluid-fluid level (Fig. 2). Post-contrast study reveals peripheral enhancement of lesion. No evidence of cortical break is seen. Underlying humerus shows long segment marrow signal changes over 10 cm segment with no significant enhancement-likely marrow oedema (Fig. 1). MRI report suggested diagnosis of surface aneurysmal bone cyst. Computed tomography (CT) guided core needle biopsy was taken which revealed giant cell rich lesion with bony trabeculae and new bone formation.

MRI T1W and T2W images showing lesion in the outer cortex of the humerus with bone marrow oedema.
Fig. 1 MRI T1W and T2W images showing lesion in the outer cortex of the humerus with bone marrow oedema.
MRI scan (axial cut) showing fluid-fluid level characteristic of aneurysmal bone cyst.
Fig. 2 MRI scan (axial cut) showing fluid-fluid level characteristic of aneurysmal bone cyst.

On the basis of the above findings, patient was advised for the surgical removal of the lesion. Several treatment methods (such as curettage only, curettage with bone grafting or cementing to fill the defect, or the use of fibrosing agents such as phenol) were discussed with the patient and she opted for wide resection of the lesion as it is associated with recurrence of almost 0 %. Since the lesion was involving half of the cortex circumferentially, so, there was risk of fracture post-operatively because of thinning of cortex afterwards with wide resection. Therefore, it was finally decided to take help of O-arm and navigation for complete resection of the tumour without damaging adjacent normal bone and other structures.

She underwent surgery after getting clearance from anesthesia department. O-arm and computer navigation assisted surgery was done (Figs. 3 and 4). The benefit of using O-arm during the surgery is that it does not require point-to-point matching with preoperative CT data; therefore, errors due to surgical positions can be avoided and the exact resection of the tumour along the margin can be done without spillage of the tumour to the surrounding structures. Following resection of the lesion, reconstruction of the cortical defect was done by cementing and plating of the humerus shaft (Fig. 5). Her post-operative period was uneventful. Physiotherapy was started from 2nd post-operative day after the first dressing. Her histopathology report was compatible with the diagnosis of aneurysmal bone cyst with reactive new bone formation was seen.

With the help of computer navigation, bone drilling was done for en-bloc resection.
Fig. 3 With the help of computer navigation, bone drilling was done for en-bloc resection.
This image is showing the projection line of the drill bit outside the ABC lesion.
Fig. 4 This image is showing the projection line of the drill bit outside the ABC lesion.
Post-op X-ray images showing fixation of the humerus shaft with the cement and plate.
Fig. 5 Post-op X-ray images showing fixation of the humerus shaft with the cement and plate.
3

3 Discussion

Aneurysmal bone cyst (ABC) lesions are benign, blood-filled tumor that grows inside the bones.13 Although they are benign, but can be aggressive and leads to destruction of bones causing pain, and sometimes pathological fracture of the involved extremity.14 ABCs are of two types: 1. Primary ABCs and 2. Secondary ABCs.15 The exact cause of Primary ABCs is currently not known, but recently some studies have shown that it is due to the translocation of the ubiquitin specific peptidase 6 (USP6) gene on chromosome 17 (t16; 17)(q22; p23) which leads to up regulation of USP6 gene and induction of matrix metalloproteinases (MMPs).16 The action of these MMPs is responsible for osteolysis, inflammation and increased vascularization in primary ABCs. The risk factors of secondary ABCs are trauma, neoplasms and vascular malformation which lead to cascade of reaction causing increase in venous pressure, vasodilation and engorged vascular bed which activate osteoclasts leading to bone resorption and erosion of cortex.

Enneking divided ABC lesions into 3 stages where stage 1 was described as lesions with the surrounding rim of cortical bone. Lesions with clearly defined border but no cortical bone were defined as stage 2 and lesions with no clearly defined border were described as stage 3 lesions.17

The treatment of ABC should be individualized depending on the location, aggressiveness and extent of the lesion. Although curettage and other treatment modalities (including percutaneous intralesional injection, cryotherapy, radiation and embolization) were described in the literature but they are associated with the recurrence of the disease in 10–60 % cases as documented. En-bloc resection of the ABCs are not associated with recurrence of the lesion in the literature.18–20

According to a study by Döring et al., they found that the overall recurrence rate of the ABCs, treated with different modalities, was 31 % (28 cases had recurrence out of 90 operated).21 They found recurrence rate of 25 % and 24 % in the patients who underwent curettage with adjuvant burring and phenolization respectively. Patients treated with curettage and filling of cavity with either autograft, allograft or cement had recurrence rate of 14 %, 40 % and 50 % respectively. They found no recurrence in the group underwent en-bloc resection of the lesion.

A study conducted by Gibbs et al. on 40 patients of ABCs, found 12 % of recurrence rate in patients treated with curettage and high speed burring while there was no recurrence in patients treated with excision through the margins.22 In another study, ABCs treated with curettage alone with bone-grafting is associated with a recurrence rate of approximately 31 %.23 There are studies suggestive of 50–71 % recurrence rate in patients treated with different intralesional procedures but 0 % of recurrence in patients underwent resection.24

Similarly, various studies suggestive of recurrence rates with curettage and an adjuvant treatment such as polymethylmethacrylate (PMMA), phenol, liquid nitrogen, or the use of a high-speed burr are lower and have ranged from 3.7 % to 18 %.25–30 In a study by Steffner et al., recurrence rate after curettage and high speed burr was 20.6 % but it came down to 7.5 % when the above treatment was augmented with argon beam coagulation.31 In a study on sclerotherapy with polidocanol on ABC lesions, no response to treatment was seen in approximately 12 % of patients.32

Adjuvant treatments can have several adverse effects. Radiation is linked to growth arrest, organ damage, nonunion, and secondary sarcomatous changes.33,34 Alcoholic zein/Ethibloc may cause infections (such as abscesses, osteitis, cellulitis), cutaneous fistula and lung infarcts.23,35–40 Sclerotherapy often requires multiple sittings and can lead to local induration (82 %) and skin hypopigmentation (24 %).41–43 Phenol can result in denaturation of protein, coagulative necrosis and hemolysis.44–47 Cryotherapy may lead to growth arrest, gangrene, nerve palsy, artrhrofibrosis, infections, fractures, and embolism.33,44,48,49 PMMA can cause heat necrosis of local site and damage to the physis in juxtaphyseal tumour.44,50

In a study by Sharma et al. on diaphyseal ABC lesions of long bones, they suggested that the chances of pathological fractures are more in comparison to metaphyseal lesions because of long lever arm at diaphysis and stress riser at the site of lesion and suggested internal fixation for the diaphyseal lesion. Similarly, we also fixed the humerus cortex post excision of the lesion with the plate and cement.51

O-arm-guided resection is an accurate and safe method for bone tumor removal, offering several advantages, such as superior image quality, reduced radiation exposure, and intra-operative confirmation of tumor excision. It can also shorten surgical time, decrease blood loss, and enhance post-operative recovery.52

A study by Fujiwara et al. on 6 patients of extremity musculoskeletal tumours, found clear margins on histopathological examination of resected specimens in all patients. There were no issues or complications associated with the use of navigation, and none of the patients experienced a recurrence of the tumor at the surgical site.52

The CT navigation system relies on preoperative data acquired in the supine position. However, tumours located in different regions may necessitate various surgical positions, leading to potential discrepancies between preoperative and intraoperative imaging.53,54 Such mismatches can cause errors in tumour resection and extend surgical duration due to the need for time-consuming point-to-point registration of the affected bone. The use of intra-operative O-arm/Stealth CT navigation system (Medtronic, Minneapolis, MN, USA) represents the latest advancement in surgical technology, offering high-resolution, 3-D fluoroscopic imaging that registered to the StealthStation (Medtronic) automatically without the need for point-to-point matching with preoperative CT data.55–57 This feature eliminates errors related to surgical positioning. However, to date, there are no published Indian studies documenting bone and soft tissue tumor surgeries of extremities utilizing the O-arm/Stealth navigation system.

Navigation-assisted surgery for bone tumor resection has become more common since its introduction in 200457,58; though there are some challenges. A study by Farfalli et al. on 78 cases found a 5 % registration failure, requiring surgeries to proceed without navigation.59 Takao et al. highlighted the importance of understanding the error range in CT-based navigation for proximal femoral osteotomy60 Positional errors in navigation-guided bone tumor resections typically range up to 1 mm, with additional cutting errors around 1 mm corresponding to the width of bone saw.59 For most bone tumours, errors under 2 mm, including positional and angle discrepancies, are generally negligible, but could be more significant for small, deep tumor.

4

4 Conclusion

Aneurysmal bone cyst in the diaphyseal/cortical region are rarity in itself and are prone for pathological fracture because of long lever arm and stress riser in this region. En-bloc excision of the tumour is very important for the prevention of recurrence but makes bone weak because of hemicortical excision. O-arm assisted navigated surgery could be a possible solution which provides real time 3D images helpful in navigation assisted resection of the lesion so that negative margins can be obtained and maximum cortex of the bone can be saved.

Written consent was taken from the patient for inclusion in the study.

CRediT authorship contribution statement

Vaibhav Sahu: Conceptualization, Formal analysis, Writing – review & editing. Himanshu Rohela: Conceptualization, Validation, Formal analysis, Writing – review & editing. Sunil Pasricha: Validation, Formal analysis, Writing – review & editing. Anila Sharma: Conceptualization, Resources. Anurag Mehta: Conceptualization, Supervision.

Ethical statement and Patients’ consent

Institutional Ethical Committee Approval was taken for this study and Patient Consent has been obtained.

Funding/Sponsorship

This research did not receive any specific grant from funding agencies in the public, commercial or not-for-profit sectors.

References

  1. , , , et al . Aneurysmal bone cysts of the Craniofacial origin: a Systematic review. OTO Open. 2021 Oct 24;5(4)
    [Google Scholar]
  2. , , , et al . Prevalence of aneurysmal and solitary bone cysts in young patients. Clin Orthop Relat Res. 2005 Oct;439:136-143.
    [Google Scholar]
  3. , , , et al . Epidemiology of aneurysmal bone cyst in children: a multicenter study and literature review. J Pediatr Orthop B. Nov 2004;13(6):389-394.
    [Google Scholar]
  4. , , , , , , . Aneurysmal bone cyst: a population based epidemiologic study and literature review. Clin Orthop Relat Res. 1999;363:176-179.
    [Google Scholar]
  5. , , , et al . Current management of aneurysmal bone cysts. Curr Rev Musculoskelet Med. 2016;9(4):435-444.
    [Google Scholar]
  6. , , , et al . Aneurysmal bone cyst of the mobile spine: report on 41 cases. Spine. 2001;26:27-35.
    [Google Scholar]
  7. , , , . Aneurysmal bone cyst of the spine: 31 cases and the importance of the surgical approach. J Pediatr Orthop B. 1998;7:286-292.
    [Google Scholar]
  8. , , , et al . Aneurysmal bone cyst of the spine. Management and outcome. Spine. 1998;23:621-628.
    [Google Scholar]
  9. , , , . Modern surgical treatment of primary aneurysmal bone cyst of the spine in children and adolescents. J Pediatr Orthop. 2005;25:387-392.
    [Google Scholar]
  10. , , , , . Aneurysmal bone cysts. A clinicopathology study of 66 cases. Cancer. 1970;26:615-625.
    [Google Scholar]
  11. , , , . Subperiosteal aneurysmal bone cyst with associated bone marrow oedema: an unusual appearance. Australas Radiol. 2003;47(4):475-478.
    [Google Scholar]
  12. , . Magnetic resonance imaging of musculoskeletal tumors: skeletal and soft tissue masses. Curr Probl Diagn Radiol. 1999;28:34-62.
    [Google Scholar]
  13. , , , . Aneurysmal bone cyst: its pathogenesis based on angiographic, immunohistochemical and electron microscopic studies. Pathol Oncol Res. 1998;4:277-281.
    [Google Scholar]
  14. , , , . Bone cysts: Unicameral and aneurysmal bone cyst. J Orthop Traumatol: Surgery & Research. 2015;101(Issue 1):S119-S127.
    [Google Scholar]
  15. , , , , , . Aneurysmal bone cyst Plus lesions: a case report and a literature review. Cureus. 2022 Aug 12;14(8)
    [Google Scholar]
  16. , , , , , . Soft-tissue aneurysmal bone cyst with translocation t(17;17)(p13;q21) corresponding to COL1A1 and USP6 loci. Skeletal Radiol. 2015;44(11):1695-1699.
    [Google Scholar]
  17. , . A system of staging musculoskeletal neoplasms. Clin Orthop Relat Res. 1986;204:9-24.
    [Google Scholar]
  18. , , , et al . Aneurysmal bone cyst recurrence in children: a review of 56 patients. J Pediatr Orthop. 2007;27(8):938-943.
    [Google Scholar]
  19. , , , , . Aneurysmal bone cyst. A review of 52 primary and 16 secondary cases. Arch Orthop Trauma Surg. 1992;111(6):318-322.
    [Google Scholar]
  20. , . Treatment of aneurysmal bone cysts in childhood. J Pediatr Orthop. 1986;6(3):326-329.
    [Google Scholar]
  21. , , , et al . Results in the surgical treatment of aneurysmal bone cysts – a retrospective data analysis. J Orthop Traumatol: Surgery & Research. 2022;108(4)
    [Google Scholar]
  22. , , , , , , . Aneurysmal bone cyst of the extremities. Factors related to local recurrence after curettage with a high-speed burr. J Bone Joint Surg Am. 1999;81:1671-1678.
    [Google Scholar]
  23. , , . Modern concepts of primary aneurysmal bone cyst. Arch Orthop Trauma Surg. Feb. 2007;127:105-114.
    [Google Scholar]
  24. , , . Aneurysmal bone cyst in 29 children. J Pediatr Orthop. 2002;22(4):533-539.
    [Google Scholar]
  25. , , , , , . Aneurysmal bone cyst: a review of 150 patients. J Clin Oncol. 2005;23:6756-6762.
    [Google Scholar]
  26. , , , , , . Aneurysmal bone cysts recur at juxtaphyseal locations in skeletally immature patients. Clin Orthop Relat Res. 2008;466:722-728.
    [Google Scholar]
  27. , , , , , . Aneurysmal bone cyst: the role of cryosurgery as local adjuvant treatment. J Surg Oncol. 2009;100:719-724.
    [Google Scholar]
  28. , , , , . The treatment of aneurysmal bone cyst. Clin Orthop Relat Res. 1995;311:157-163.
    [Google Scholar]
  29. , , , , . Aneurysmal bone cysts. A clinicopathologic study of 66 cases. Cancer. 1970;26:615-625.
    [Google Scholar]
  30. , , , , , , . Aneurysmal bone cysts treated by curettage, cryotherapy and bone grafting. J Bone Joint Surg Br. 1997;79:20-25.
    [Google Scholar]
  31. , , , et al . Factors associated with recurrence of primary aneurysmal bone cysts: is argon beam coagulation an Effective adjuvant treatment? The Journal of Bone and Joint Surgery-American. 2011;93(21):e122.
    [Google Scholar]
  32. , , , , . Is sclerotherapy better than intralesional excision for treating aneurysmal bone cysts? Clin Orthop Relat Res. 2010;468:1649-1659.
    [Google Scholar]
  33. , , , , . The treatment of aneurysmal bone cyst. Clin Orthop Relat Res. 1995;311:157-163.
    [Google Scholar]
  34. , , , , , . Aneurysmal bone cyst. Long-term follow-up of 20 cases. Arch Orthop Trauma Surg. 1990;109:221-223.
    [Google Scholar]
  35. , , , et al . Recurrence of aneurysmal bone cysts in young children: a multicentre study. J Pediatr Orthop B. 2005;14:212-218.
    [Google Scholar]
  36. , , , , , . Intralesional Ethibloc injections in primary aneurysmal bone cysts: an efficient and safe treatment. Skeletal Radiol. 2003;32:559-566.
    [Google Scholar]
  37. , , , et al . Aneurysmal bone cysts: treatment with direct percutaneous Ethibloc injection: long-term results. Cardiovasc Intervent Radiol. 2002;25:282-290.
    [Google Scholar]
  38. , , , et al . Aneurysmal bone cysts: percutaneous embolization with an alcoholic solution of zein—series of 18 cases. Radiology. 1998;208:369-373.
    [Google Scholar]
  39. , , , , , . Aneurysmal bone cysts in children: complications of fibrosing agent injection. Radiology. 2004;232:522-526.
    [Google Scholar]
  40. , , , , , , . Percutaneous Ethibloc injection in the treatment of primary aneurysmal bone cysts. J Pediatr Orthop B. 2005;14:367-370.
    [Google Scholar]
  41. , , , , , , . Treatment of aneurysmal bone cysts with percutaneous sclerotherapy using polidocanol. A review of 72 cases with long-term follow-up. J Bone Joint Surg Br. 2006;88:1212-1216.
    [Google Scholar]
  42. , , . Sclerosing foam in the treatment of varicose veins and telangiectases: history and analysis of safety and complications. Dermatol Surg. 2002;28:11-15.
    [Google Scholar]
  43. , , , , . Is sclerotherapy better than intralesional excision for treating aneurysmal bone cysts? Clin Orthop Relat Res. 2010;468:1649-1659.
    [Google Scholar]
  44. , , , , , , . Aneurysmal bone cysts treated by curettage, cryotherapy and bone grafting. J Bone Joint Surg Br. 1997;79:20-25.
    [Google Scholar]
  45. , , , , , , . Argon beam coagulation as an adjuvant for local control of giant cell tumor. Clin Orthop Relat Res. 2006;454:192-197.
    [Google Scholar]
  46. , , , , . Cytotoxicity of phenol to musculoskeletal tumours. J Bone Joint Surg Br. 1996;78:984-985.
    [Google Scholar]
  47. , , , . Characteristics of phenol. Instillation in intralesional tumor excision of chondroblastoma, osteoclastoma and enchondroma. Arch Orthop Trauma Surg. 1998;117:43-46.
    [Google Scholar]
  48. , , , , , , . Cryosurgery in the treatment of giant cell tumor. A long-term followup study. Clin Orthop Relat Res. 1999;359:176-188.
    [Google Scholar]
  49. , . A 17-year review of cryosurgery in the treatment of bone tumors. Clin Orthop Relat Res. 1982;163:231-234.
    [Google Scholar]
  50. , , , , . Aneurysmal bone cysts in children. J Cancer Res Clin Oncol. 1996;122:767-769.
    [Google Scholar]
  51. , , , , . Diaphyseal aneurysmal bone cysts (ABCs) of long bones in extremities: analysis of surgical management and comparison with metaphyseal lesions. J Clin Orthop Trauma. 2021 Apr 17;18:74-79.
    [Google Scholar]
  52. , , , et al . Intraoperative O-arm-navigated resection in musculoskeletal tumors. J Orthop Sci. Nov 2018;23(6):1045-1050.
    [Google Scholar]
  53. , , . Computer-assisted tumor surgery in malignant bone tumors. Clin Orthop Relat Res. 2013;471:750-761.
    [Google Scholar]
  54. , , , , , , . Does intraoperative navigation assistance improve bone tumor resection and allograft reconstruction results? Clin Orthop Relat Res. 2015;473:796-804.
    [Google Scholar]
  55. , . Extraarticular endoscopic excision of femoral head chondroblastoma. J Pediatr Orthop. 1995;15:578-581.
    [Google Scholar]
  56. , , , , , , . Image guided radiofrequency thermo-ablation therapy of chondroblastomas: should it replace surgery? Skeletal Radiol. 2014;43:513-522.
    [Google Scholar]
  57. , , , , , , . New indications for computer-assisted surgery: tumor resection in the pelvis. Clin Orthop Relat Res 2004:219-225.
    [Google Scholar]
  58. , , , , , . Is computer navigation when used in the surgery of iliosacral pelvic bone tumours safer for the patient? Bone Jt. J.. 2017;99–B:261-266.
    [Google Scholar]
  59. , , , , , , . What is the expected learning curve in computer-assisted navigation for bone tumor resection? Clin Orthop Relat Res. 2017;475:668-675.
    [Google Scholar]
  60. , , , , . Error range in proximal femoral osteotomy using computer tomography-based navigation. Int J Comput Assist Radiol Surg. 2017;12:2087-2096.
    [Google Scholar]

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