There is currently limited evidence on the optimal management of CNO, with only few small case series and case reports in the literature. Evidence concerning segmental bone excision and reconstruction is even scarcer and, to our knowledge, limited to a single case report where the authors opted to use a bone transport technique with external fixation [6].
The complex anatomy of the clavicle is characterized by the strong sternoclavicular joint capsule and costoclavicular ligaments on the medial aspect and the coracoclavicular and acromioclavicular ligaments laterally [8]. Therefore, the clavicle not only represents a link between the trunk and the shoulder but also contributes to shoulder motion and strength by transmitting the forces of the trapezius muscle to the scapula [9]. In the case of a claviclectomy, protraction and retraction of the scapula is increased, leading to secondary weakness in overhead activities [10]. Furthermore, the clavicle also acts as a shield to protect the underlying neurovascular structures, which are located just beneath its medial third [11]. Finally, esthetics has also been pointed out as a potential concern after total or partial claviclectomy, particularly in female patients [12].
Several case reports of total claviclectomy reported satisfactory results in terms of pain control and shoulder function. However, associated complications such as infections and vascular lesions should not be overlooked [13,14,15]. These observations were further questioned by other reports showing less satisfactory outcomes in terms of pain control, ROM, strength and patients’ satisfaction [16, 17]. A recent systematic review did not suggest any evidence of better clinical outcome after surgical reconstruction of the clavicle [18]. Various clavicle reconstruction techniques have been proposed in the literature (mainly segmentary and mostly in cases of nonunion or reconstruction after tumor excision), including autograft, allograft and synthetic materials. Autografts have been described in the form of free fibula or “Eve procedure” with a costal graft [19,20,21]. The main advantage of this procedure is to bring healthy vascularized tissue and improve the long-term outcome by enhancing the healing of the surrounding soft tissues. On the other hand, fibula harvesting may be associated with infection, fracture and knee or ankle instability at the donor site.
In our case, the indication to perform a total claviclectomy was dictated by the occurrence of an infectious complication with development of a chronic cutaneous fistula, as well as by pain and neurologic complaints due to local mass effect. We thus opted to reconstruct the clavicle based on the patient’s young age, the good long-term evolution of the disease and the aim for both an esthetic and functional surgical reconstruction. We opted for a two-stage approach to total clavicle reconstruction, with a custom-made antibiotics-loaded cement spacer. This option had two main advantages. First, the infectious background was in our opinion not suitable for a single-stage procedure and we further recommend this approach in case of doubtful diagnosis. The second argument was that the cement spacer would favor stability and biological integration of the vascularized peroneal graft. We used contralateral computed tomography images and preferred to maintain clavicle length rather than its proper anatomical shape for our surgical reconstruction. While clavicular length has been linked with improved functional outcome in clavicle fractures, there is limited evidence concerning the importance of its shape. Furthermore, this choice spared osteotomy of the peroneal graft and thus eliminated the risk of nonunion and symptomatic metal hardware [22, 23]. The improvement in the Constant-Murley score noted in our case might indicate that clavicular length is sufficient to improve or maintain shoulder function after total clavicle excision, even though the ROM returned to its preoperative level but did not surpass it.
We opted to fix our peroneal graft using an anatomic ligamentous reconstruction. To the best of our knowledge, the association of a free vascularized bone graft with ligamentous reconstruction has not been described so far. When dealing with total clavicle reconstruction, this combination makes even more sense. It not only avoids any need for subsequent surgery to remove symptomatic or infected foreign materials, but the biologic fixation also ensures a long-lasting stability of the reconstruction. Another key point in this case was the combination of a free flap with the concept of induced biological membrane [24]. While not mandatory, this might facilitate integration in a compromised environment.
Our technique showed an effective and persistent pain remission associated with a functional improvement at two years follow-up. The closest report to ours is the one by Kalbermatten et al., with a case of late clavicular reconstruction using a free peroneal graft 23 years after the index procedure [25]. They used an osteotomy guide to reconstruct the S-shaped clavicle, which was fixed with a plate and screws to maintain its alignment as well as Kirchner wires and suture anchors to attach it to the acromion and sternum. Besides a modest improvement of 10° in shoulder ROM in the sagittal plane, the procedure was reported as effective on pain control, which was unfortunately not assessed using VAS. On the other hand, Lin et al. reported results in five oncologic patients with subtotal (instead of total) claviclectomy using a cement prosthesis to replace the clavicle [26]. Any direct comparison with this study should thus be made with caution. The authors also used VAS pain score and found significant improvements from 8.0 (range, 6–9) to 1.4 (range, 1–2). They further reported functional outcome using the American Shoulder and Elbow Surgeons shoulder outcome score, which improved from 42.4 ± 9.0 to 92.4 ± 3.3. While this score does not directly assess strength, it is comparable to the Constant-Murley score. The long-term outcome of this technique is not yet known, this method having mainly been used in patients with limited life-time expectancy. Finally, Li et al. reviewed 6 clavicle reconstructions using massive allografts, all of them performed after subtotal claviclectomy for malignancies [27]. In their retrospective study, the mean postoperative Constant-Murley score was 85. However, it was associated with a high complication rate with two allografts secondarily removed due to infection and nonunion.
In conclusion, advanced complicated CNO is a rare indication for claviclectomy. This implies that advantages and complications of clavicle reconstruction should be carefully discussed with patients due to limited evidence of superior clinical outcome and potential local and donor-site complications. While in our case the outcome met the patient’s satisfaction, it remains an isolated case and further reports are awaited to help surgeons and patients in their decision process.
