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Advancements in biological reconstruction of proximal humerus tumor: A case study and review of literature
, Rahul Karn, Vikas Olkha, Hawaibam Nongdamba, Bishwa Niruala, Vikas Maheshwari, Mohit Dhingra
⁎Corresponding author: Mohit Dhingra. mohit.orth@aiimsrishikesh.edu.in
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Received: ,
Accepted: ,
This article was originally published by Reed Elsevier India Pvt. Ltd. and was migrated to Scientific Scholar after the change of Publisher.
Abstract
Abstract
Ewing sarcoma is the second most common primary bone malignancy in adolescents. Advancement in field of imaging, revolution in reconstruction techniques, have made management of Ewings sarcoma a near success with least recurrence and better function outcomes in post operative period.
This case report presents Ewings Sarcoma of left proximal humerus, managed with wide excision and reconstruction by vascularized fibular autograft with tibial Allograft with emphasizes on the technique of reconstruction.
Wide excision and reconstruction by vascularized fibular autograft with tibialautografta feasible option in the management of Ewings Sarcoma of proximal humerus with good functional outcomes and no local recurrence.
The choice of the most appropriate surgical treatment should be considered based on resection extent as well patient’ systemic and local clinical picture, life expectancy, and functional requests. Biologic reconstructions are indicated mainly for young patients. In fact, the osteo integration and mechanic performances promised by grafts could lead to great functional results through the long life of the implant.
Keywords
EwingsSarcoma
Proximal humerus
Autologous bone grafting
Allograft
1 Introduction
Ewing sarcoma, the second most prevalent primary bone malignancy among adolescents and young adults, typically manifests around the age of 15, with nearly 30 % of cases occurring in children under 10.1 This disease exhibits a notable male predominance, with a ratio of 3 to 1. Historically, its aggressive nature has led to a mere 20 % five-year survival rate, largely due to recurrence and metastasis.2(see Table 1)
| Author Year | Type of reconstruction | Number of cases (N) | MSTS Score (% ± mean) | Complications |
| I. Antal et al., 2023 | Endoprosthesis | 90 | 84 ± NS | Implant failure |
| Cannon et al., 2009 | Endoprosthesis | 83 | 63 ± NS | Implant failure |
| Rödl et al. 20029 | Autograft | 15 | 82 ± NS | Non union and Delayed union |
| Padiolleau et al., 2014 | Autograft | 12 | 71 ± NS | Pseudo orthosis |
| DeGroot et al., 2004 | Osteoarticular allograft | 32 | 74.67 ± NS | FRACTURE |
| Jamshidi et al., 2017 | Osteoarticular allograft | 32 | 85 ± NS | FRACTURE |
The prognosis for Ewing sarcoma remains challenging, particularly given its high-grade nature and propensity for vascular spread. Patients with localized disease face a 55–65 % five-year survival rate, while those with metastases at presentation fare significantly worse, with less than 40 % survival at five years.
While both amputation and limb salvage techniques have shown comparable outcomes in terms of functionality and local recurrence rates, the emphasis on reconstruction techniques for proximal humerus lesions is crucial. Effective reconstruction not only offers comparable outcomes to amputation but also enhances functionality and reduces the patient's burden in terms of energy expenditure.3
In pursuit of surgical goals centered around curative excision and functional reconstruction, orthopedic surgeons have explored various methods. One such innovative approach is highlighted in the following case report, showcasing a novel biological reconstruction technique.4
2 Case report
An 8-year-old male presented to our institute reporting persistent pain and swelling in his left shoulder for the past ten months. The pain had developed gradually, intensifying over time, while the swelling had progressed from an initial size of 2 × 2 cm to its current dimensions of 14 × 8 cm. There were no reports of trauma, fever, weight loss, or constitutional symptoms.
Upon examination, a diffuse swelling was observed over the left proximal arm's anterolateral aspect, with no apparent skin changes or venous prominence. The swelling was characterized by an irregular surface and border. The swelling exhibited a bony-hard consistency, was firmly attached to the underlying bone, tender to palpation, and the overlying skin remained unaltered. Distal neurovascular status was found to be intact, and the patient exhibited normal range of motion in the shoulder, elbow, and wrist joints.
A biopsy was performed, confirming the diagnosis of Ewing's Sarcoma upon histopathological examination. The patient underwent neo-adjuvant chemotherapy, receiving a regimen consisting of Vincristine, Actinomycin, and Cyclophosphamide. As part of the pre-operative protocol, non-invasive investigations including radiographs of the left arm, encompassing both the shoulder and elbow joints, as well as an MRI scan of the entire length of the left humerus, were conducted.
2.1 Surgical technique
Following routine investigations and under general anesthesia, the patient was positioned supine. A skin incision was made encircling the biopsy site, with the approach lateral to the deltopectoral groove, including resection of the anterior deltoid muscles. The pectoralis major tendon was divided, and the tendon of the long head of the biceps was cut distally to facilitate resection with the operative specimen. The upper limb was externally rotated to expose the tendons of the latissimus dorsi and teres major muscles, both of which were divided close to the bone. The proximal humerus was externally rotated, and the subscapularis was vertically divided at the myotendinous junction. The medial axillary nerve and anterior circumflex artery were identified and secured using loops. Before tumor resection, the radial nerve was dissected and retracted. The tumor was excised as a single block measuring the length from the preoperative MRI. A frozen section of marrow from the distal fragment was examined for malignant cells, yielding a negative result, allowing the reconstruction procedure to commence with a new set of instruments. Meanwhile, the tumor specimen was measured for dimensions and sent for histopathological examination.
A non-antigenic, non-reactive, and non-infectious tibial allograft, which tested negative for nucleic acid amplification testing (NAAT) and culture, was thawed, and approximately 10 cm of allograft was prepared. Under aseptic precautions, a vascularized fibular autograft was harvested from the right leg. The lateral collateral ligament was sutured together with the hamstring, and the donor site was closed in layers. The vascularized fibular autograft was inserted into the medullary canal of the tibial allograft, and the composite was fixed to the distal end of the humerus using a dynamic compression plate. The shoulder joint was reconstructed using prolene mesh, and adequate hemostasis was achieved. Closure was performed in layers, and an arm pouch was applied in the immediate postoperative period.
In the postoperative period, a plain radiograph was obtained, depicting a well-fixed composite with the distal humerus. During follow-up evaluations, suture removal was performed on postoperative day 14, and patient satisfaction scores were assessed at 3 and 8 weeks using the SF-36 scoring system for patient-based satisfaction. Functionality of the left shoulder joint and donor site (right knee joint) and SF-36 scores at 3 weeks and 8 weeks postoperatively were assessed. There were no systemic or local complications noted at the 1-year postoperative mark.
The histopathological sample confirmed evidence of necrosis in the tumor, with margins deemed adequately sufficient. The patient underwent adjuvant chemotherapy cycles following suture removal. At 1 year of follow-up, the patient was disease and pain-free, with a functional arm and no evidence of recurrence.
Proximal humerus is a frequent site for primary bone tumors, ranking as the foremost location among upper limb bones. In the past, amputations and arthrodesis were the preferred treatments for musculoskeletal tumors in this area. However, modern surgical progress has revolutionized options for orthopedic oncologists. Improved imaging and surgical technologies have enabled safer and more precise resections, increasing the likelihood of achieving tumor-free surgical margins and reducing the occurrence of local recurrence. The reconstructive phase has also witnessed the development of various approaches to restore the shoulder's original shape and function.
Current modalities for proximal humerus reconstruction include osteoarticular allografts, bone autografts, prostheses, and graft-prosthesis composites. Despite the advantages of prostheses such as shorter operative time and early functional recovery, they are not without drawbacks. Prostheses carry risks of dislocation, aseptic loosening, and infection due to the presence of large metal segments ideal for bacterial biofilm formation.
I. Antal et al. concluded that prosthetic reconstruction in adults offers good functional outcomes and a lower rate of recurrence, with fewer complications post-adjuvant chemotherapy. However, instability, dislocation, infection, and the need for frequent component replacement remain concerns.7
Cannon et al. observed that post reconstruction, prosthesis depicted better functional outocomes with earlier return to activities. But they were frequent incidence of implant failure (Henderson Type 3 failure) in longer follow up.8
Autografts serve not only as scaffolds for nearby osteoblasts but also stimulate osteogenesis and are osteoinductive. They promote local bone growth and aid in postoperative healing, with potential benefits for pediatric patients, including mimicking native bone growth and restoring limb length. However, limited bone stock and donor site morbidity are significant drawbacks.
Padiolleau et al. concluded that autografts serve as execellent tools for post resection reconstruction owing to their to osteogenic potential. Autografts are often burdened by the risk of non unions and delayed unions as well as their usage is always limited owing to limited bone stock.9
Jamshidi et al. also concluded on similar lines while usage of osteoarticular autografts but complications of graft fractures were reported in post op period.10
Allografts, suitable for young patients with good healing potential, provide scaffolds for osteocyte incorporation and osteointegration but carry risks such as host versus graft reaction and implant fracture.
In our pediatric case, we opted for reconstruction using a vascularized fibular autograft and tibial allograft. The autograft's open physis facilitates native bone growth, aiding in limb length restoration and postoperative healing. The limited bone stock disadvantage of the autograft is offset by the allograft, resulting in improved reconstruction of the proximal arm. Shoulder joint reconstruction utilized prolene mesh, with follow-up radiographs indicating shoulder arthrodesis and good osseointegration. The patient achieved good functional outcomes with a fair range of shoulder joint movements. The postoperative period was uneventful, with no complications at the graft donor site. The patient underwent six cycles of postoperative chemotherapy and achieved a 1-year Musculoskeletal Tumor Society (MSTS) score of 94 %.
3 Conclusion
The management of any operative case of bone cancer adheres to the fundamental principles of orthopedic oncology: the primary goal is the complete resection of the tumor with wide margins to minimize the risk of local recurrence. Equally important is the restoration of anatomy and function post-surgery.
A combination of vascularized autograft and allograft has emerged as a reliable and durable option for reconstructing musculoskeletal tumors of the proximal humerus. This approach aims to not only remove the tumor but also to reconstruct the affected area, preserving as much function and structural integrity as possible.
Patients who undergo this combined grafting technique often exhibit good functional scores postoperatively, with a reduced risk of local recurrence. This underscores the efficacy and success of this approach in orthopedic oncology.
Funding
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
Authors contribution
P. B. – Planning of study, writing, and revising the manuscript.
A. R. – Planning of study, revising the manuscript.
B. B. N. – Data Management.
R. K. – Data Management.
V. O. – Data Management.
V. M. - Data Management.
H. N.– Data Management.
M. D. – Planning of study, revising the manuscript.
Informed consent
Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.
Ethical approval
This study was performed in line with the principles of the Declaration of Helsinki.
The intuitional ethical clearance was taken for the study and for paper publication. A copy of the ethical clearance is available for review by the Editor-in-Chief of this journal on request.
Consent to publish
The authors affirm that human research participants provided informed consent for publication of the images inFigs. 1–5.
Availability of data and materials
All data and materials as well as software application support published claims and comply with field standards.
Code availability
Not Applicable.
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