Total prosthetic replacement of the humerus following extensive sarcoma: a case report

¹Department of Orthopedic Surgery and Traumatology, CHU Mohammed VI of Tangier, Tangier, Morocco, ²Department of Vascular Surgery, CHU Mohammed VI of Tangier, Tangier, Morocco

^&Corresponding author
Jaouad Yasser, Department of Orthopedic Surgery and Traumatology CHU Mohammed VI of Tangier, Tangier, Morocco

Introduction    

Osteosarcoma is a rare pathology. The humerus is considered to be the third site most affected by osteosarcomas [1]. Neoadjuvant chemotherapy and wide surgical resection are the main pillars of the oncological management of osteosarcomas of the humerus. Situations, the indication of which is a total humeral resection, present a real challenge in terms of reconstruction. In this case, prosthetic replacement is the best choice for limb salvage [2]. Indeed, salvage surgery gives good functional results without impact on patient survival compared to amputations [3]. We present a case of a young patient aged 17 who underwent total prosthetic replacement of the humerus after resection of the humerus secondary to extensive osteosarcoma. The presentation of this case highlights the importance of being wary of suspicious images to avoid certain serious consequences. It also shows an interest in prosthetic replacement for saving the limb and preserving the function of the hand and elbow.

Patient and observation     

Patient information: the young patient who´s 17 years old has no notable pathological history.

Clinical findings: swelling of the upper two-thirds of the left arm was observed, good skin condition without inflammatory signs on the side, and amyotrophy with an unremarkable vascular-nervous examination.

Timeline: his history of illness dates back two years; the patient was the victim of a public road accident with a direct impact on the left arm. The radio-clinical examination revealed a mid-diaphyseal fracture of the left humerus on a lacunar image, with an unremarkable vascular-nervous examination. The patient was operated on by a colleague using the Haquetal-type pinning technique with a biopsy of the suspicious lesion. The pathological study came back without signs of malignancy. The evolution after three months was marked by a painful swelling of the proximal two-thirds of the left humerus, with deterioration of the general condition.

Diagnostic assessment: the computed tomography (CT) examination revealed an osteolytic tissue process involving the entire humeral diaphysis and the proximal metaphysis, extending over 24 cm in height and measuring 6 cm in transverse diameter at the height of the middle third of the diaphysis with contact humeral pedicle. The proximal epiphysis, the distal metaphyseal epiphyseal region, and the glenohumeral, and elbow joints are intact. He also noted the presence of intramedullary pinning (Figure 1). An ultrasound-guided biopsy was performed, which revealed a high-grade malignant osteoblastic osteosarcoma.

Therapeutic intervention: the patient initially received neoadjuvant chemotherapy, and resection-replacement surgery was planned. The replacement parts consist of an elbow hinge prosthesis, a reverse shoulder prosthesis, and a rod that is used to join the components and which has adjustable sizes depending on the total prosthetic length. The patient was placed in a supine semi-sitting position with general anesthesia. The approach was a combination of the deltopectoral shoulder, internal brachial, and anterior elbow approaches. The first surgical stage is aimed at the release of the pedicle in the axillary, internal brachial region, and at the elbow with the release of the radial nerve via the same approach. The second phase was devoted to the one-piece resection of the humerus (Figure 2). The third stage was for prosthetic replacement. The fourth stage is aimed at the reinsertion of the muscles: deltoid, biceps brachii, the distal part of the brachialis muscle, and the triceps, the closure is done under a suction drain. Postoperatively, the patient was placed under arm-to-body immobilization with an abduction cushion.

Follow-up and outcomes: the vascular-nervous examination was unremarkable, apart from paresthesia of the radial territory. The control X-ray showed subluxation of the prosthetic shoulder joint (Figure 3). After 15 months, no local recurrence was detected. The patient has preserved elbow and hand function but with limited shoulder range of motion.

Patient perspective: the patient and the patient's family were pleased with the quality of the surgery of the salvage. The patient's family was surprised by the preservation of the functions of the hand and elbow.

Informed consent: an informed consent was obtained from the patient and the patient's family to publish this work.

Discussion     

The humerus ranks third among the preferred locations for osteosarcomas [1]. The metaphyseal region is the most affected by this pathology [4]. Whereas in our case, the location is initially diaphyseal before its dissemination. The epidemiological study of osteosarcomas finds a peak in the rate of occurrence in the second decade of life and a slight male predominance of 58% [4]. Thanks to advances in chemotherapy, amputations are being replaced by limb salvage in the management of malignant bone tumors of the long bones [3-5]. Apart from high rates of local recurrence in patients receiving limb salvage compared to amputations, overall survival is the same [5]. Total prosthetic replacement of the humerus is considered a practical solution for limb salvage by reconstructing the elbow and shoulder joints. The preservation of the residual function of the hand and elbow and the emotional acceptance of patients are the main achievements of this salvage surgical option [2]. Following an insufficiency of the intrinsic stabilizing elements of the shoulder and in particular the rotator cuff; the difficulty of recovering active anti-gravity movements is among the main deficiencies of this surgery [2]. Indeed, the incompetence of the rotator cuff and the presence of an unopposed contraction of the deltoid which causes superior migration of the head of the prosthesis are the main elements responsible for the alteration of active movements postoperatively [6]. Consequently, patients must be informed of a possible high probability of a reduction in active joint range of motion. To compensate for this impairment of active shoulder movements; a rehabilitation program is required, which must target the activation of the scapulo-thoracic joint. In addition, the preservation of hand and elbow function allows good adaptation to the lifestyle [6].

Natarjan et al. reviewed 11 cases of total prosthetic replacements of the humerus after radical excision for malignant tumors. The one-year and five-year survival rates are found to be 90% and 77.9% successively. The study demonstrated good elbow and hand function with an MSTS score of 80%, but with a significant reduction in active shoulder range of motion [2]. Bernthal et al. reviewed a series of 20 cases of total prosthetic replacement of the humerus for tumors with a median survival of 148 months; the study showed survival rates at 5 years, 10 years, and 15 years successively at 100%, 86%, and 86%. Also, the study found 3 cases of local recurrence and 2 cases that were candidates for revision following symptomatic dislocations of the shoulder joint. The study did not find cases of infection or radial palsy [7]. According to the literature, the rate of local recurrence in total prosthetic replacements is estimated at 15 to 26% [8], but this rate is linked to poor survival. On the other hand, amputation is not associated with an increase in survival [9]. These studies analyzing the results of humeral prostheses are limited by a small sample size, the largest contains 34 prostheses [10], hence the interest in prospective and multicenter studies with a large sample size. Neoadjuvant chemotherapy based on cisplatin, doxorubicin, and methotrexate helps combat the emergence of chemoresistance and reduces the risk of lung metastases. As a result, it restores a high survival rate to salvage surgery [4]. In addition, chemotherapy has the role of reducing the tumor mass and isolating the tumor in the calcified periosteum. The effectiveness of chemotherapy in osteosarcomas is evaluated using the HUVOS classification. From this case report, we can conclude that limb salvage by total humeral replacement may be preferred for extensive osteosarcoma of the humerus compared with amputation. The result is the same in tumoral recurrence, while preserving elbow and hand function.

Conclusion     

Total prosthetic replacement after resection secondary to osteosarcoma is an excellent alternative to amputation. It allows functional preservation of the elbow and hand, a low rate of complications with good psychological tolerance and acceptance of patients. At 15 months of follow-up, the patient in this case report had a good clinical outcome with preservation of elbow and hand function, and without local recurrence.

Competing interests     

The authors declare no competing of interests.

Authors' contributions     

Patient management: Mohammed Shimi, Amine Azghari and Hicham Aitbenali. Data collection: Jaouad Yasser, Mohamed Aoulad Omar and Marouane Kaibech. Manuscript drafting: Jaouad Yasser. Manuscript revision: Jaouad Yasser, Mohamed Aoulad Omar and Marouane Kaibech. All the authors have read and agreed to the final manuscript.

Figures     

Figure 1: computed tomography appearance of the tumor

Figure 2: postoperative appearance of the surgical specimen

Figure 3: the control X-ray showed subluxation of the prosthetic shoulder joint

References