Good functional outcome evaluation of free vascularized fibular head graft (FVFHG) as treatment after resection of giant cell tumor (GCT) campanacci 3 at proximal humerus: A case report

Highlights • Vascularized fibula could be used to reconstruct wide resected proximal humerus.• Peroneal artery pedicle ensure vascularization of harvested long graft.• Rehabilitation for either recipient or donor site is crucial.


Introduction
Giant cell tumor (GCT) is a rare primary bone tumor that typically occurs in the meta-epiphyseal region of a long bone [1]. It is benign locally aggressive tumor with a high rate of recurrence and capacity to metastasize [1][2][3]. Wide excision is the management of choice to solve this problem, but this creates a defect [4]. The preferred modalities for the defect reconstruction include vascularized/non-vascularized bone graft, osteoarticular allografts, and custom-made prosthesis. Satisfying target to achieve by orthopedic surgeon team towards limb salvage in upper extremity musculoskeletal tumor must include limb function preservation while maintaining tumor resection and halt recurrence [5]. Limb-sparing procedures has been slowly replacing amputation regarding the technology of radiology diagnostic, microsurgery advancement and more effective adjuvant therapy following surgery [5].
In reconstruction as the treatment after resection of bone tumor at proximal humerus, we prefer to use free vascularized fibular head graft (FVFHG) transfer method. This became the first choice for reconstruction of such defects at many institutions as well as ours due to its safety as well as predictable outcome and able to confidently applied especially in defect of proximal humerus or distal radius [6].
We present our experience on wide resection and modified free vascularized autogenous fibula head grafting for GCT at the proximal humerus of a 32 years-old-male with fair long term outcome evaluation. This case is arranged and reported in line with the Surgical Case Report Guidelines (SCARE) criteria [7].

Presentation of case
A 32 years-old-male came to the hospital with the chief complaint of a lump at the proximal part of the right arm since 3 months before admitted to the hospital. The lump was as the size of a marble at the beginning and has enlarged as it is now. Pain is felt continuously, irritating and does not radiate. There was no history of trauma, fever and no remarkable past history.  is right-hand dominant and work as a mechanic. From physical examination, we found a lump at proximal humerus, firm, welldemarcated, no discoloration, no wound or ulcer. Patient has a good distal neurovascular (Fig. 1).
His plain radiograph showed an eccentric lytic lesion at epimetaphyseal proximal humerus which extends to soft tissue, destructs the subchondral and cortical bone of proximal humerus (Fig. 2). Magnetic Resonance Imaging (MRI) showed edema around the lesion with diffuse non homogenous enhancement extended to glenohumeral joint, but no histology examination before surgery was performed. From the radiograph and MRI, we concluded the diagnose was GCT Campanacci 3.
We performed FVFHG as a reconstruction modality (Fig. 3), in which long head of biceps brachii was attached to the fibular head, sutured the bicep femoris tendon to the Superior Glenohumeral Ligament (SGHL) to maintain the continuity and stability of fibular head for the purpose of preserving shoulder joint stability as well [3]. Peroneal vascular (artery and vein) were attached to the circumflex humeral artery and vein to ensure graft vascular supply. Resection of the tumor was followed by FVFHG to the proximal humerus in order to preserve good shoulder stability (Fig. 4). We are not the pioneer for this method but provide a solid successful evi-dence for this approach. Three years target follow up for anatomical, functional and radiological outcomes evaluation was undergone.
After 3 years follow up, the patient showed good functional outcome with radiographic union of the graft and no recurrence of tumor (Fig. 5). The patient is currently able to perform daily activities, such as maintain personal hygiene, write, self-feed, hold a glass, grooming, and drive his motorcycle (Fig. 6). The functional outcome rating after reconstruction of the proximal humerus was 80 percent, while score 1 is the worst and 5 is the best for each item using the Musculoskeletal Tumor Society scores [8]. Assessment of pain, emotional acceptance, and manual dexterity from this patient was in scores 5, means satisfactory. We concluded that the functional outcome is satisfactory with no further symptom nor complaint of dexterity.

Discussion
Upper limb function after resection of GCT at the proximal humerus which depends solely on reconstruction technique are to ensure revascularization, conserve shoulder joint stability and provide good elbow joint function. One of the technique choices was vascularized autograft. The advantages of the vascularized autograft are more rapid healing (due to immediate vascularity), graft hypertrophy and strength to mechanical failure compared to avascularized graft [5]. The fibula is currently the most favored donor for free vascularized transfer since it could be used for segmental defect reconstruction up to 26 cm thanks to its long and straight structure. It can be harvested without many difficulties, and the nutrient artery arises from the peroneal artery [5]. Harvesting procedure itself require a long operation time performed by a skillful microsurgery orthopedic surgeon, not to mention autologous tissue sacrifice.
In our case presentation, the FVFHG for reconstruction modality as the treatment after resection of giant cell tumor on proximal humerus showed satisfactory result following long term evaluation on anatomical, functional, and radiological parameters without any sign of tumor recurrence on three years after surgery. It shows the same with a study performed by Rose et al. [9] that reported the Fig. 2. a).Plain radiograph, b).MRI. success in limb salvage of the humerus by using vascularized fibula graft. The outcome was excellent, with no loss of limb or local recurrence, and donor site morbidity was low and no complications were found.
Theoretically, the anterior tibial artery is a major blood supplier for proximal epiphysis and the proximal two-thirds of the diaphysis of the fibula. However, in this patient, we chose the peroneal artery as a vascular pedicle because of a long defect up to two-thirds of the humerus or more than 10 cm. It is the same procedure as done by Onoda et al. [6] to their three of eight patients study due to bone defect after tumor resection more than 10 cm. This is because the vascularity of the head and peripheral parts of the fibula is safer by using the peroneal artery as a vascular pedicle in adult patients with defect more than 10 cm [6].
The other advantages by using peroneal artery as vascular pedicle are bone union due to well vascularity to the peripheral part of the fibula was obtained [6]. In this patient, it can only be followed up in the eighth months, when the bone union was occurred. Hypertrophy was seen in three years follow up and no fibular head reabsorption. This is different from the other with the FVFHG study that reported no hypertrophy occurred in all of the study patients [10]. In addition, there are also reabsorption of the fibular head in all patients. 10 Donor site morbidity from free vascularized fibula graft including mild pain at the donor site, and some complaints of numbness on the side of the leg and dorsum of the foot. The impaired flexion or extension of the great toe is not uncommon [5]. Feuvrier et al. [11] revealed that they took more cautious approaches during the walk to reduce the risk of falling. An early rehabilitation program is important to improve the physical abilities following the vascularized free fibula harvest. In these patients, no donor site morbidity was found as mentioned above. The patient is recently able to take care of personal hygiene, write, self-feed, hold glass, groom, even drive on his motorcycle. The outcome is satisfactory with no further symptom nor complaint of dexterity. Some studies observed that there were some predictive values for tumor recurrence from giant cell tumors, including the classification of Campanacci from tumors, surgical methods, the involvement of the cortical bone and the involvement of soft tissue. The recurrence rate in the Campanacci -as an independent recurrence factor-grade 1 group was 0, whereas that in grades 2 and 3 was 13.51% and 41.67%, respectively [12]. Fortunately, there was no recurrence in this patient after a three-year follow-up even though this patient had grade Campanacci 3.

Conclusion
FVFHG for reconstruction modality as the treatment after resection of GCT grade Campanacci 3 at proximal humerus shows satisfactory result following long term evaluation on anatomical, functional, and radiological parameters without any sign tumor recurrence.

Declaration of Competing Interest
The authors declare that they have no competing interests.

Sources of funding
This study was funded independently.

Ethical approval
This study was approved by the ethical board of Hasanuddin University of Makassar. Our patient has signed terms of consent to participate in the research of this case report. The institutional ethical committee has approved the publication of this case report

Consent
Written informed consent was obtained from the patient for publication of this case report and accompanying images.

Registration of research studies
We are not pioneer of this method as described in page 3 of manuscript

Provenance and peer review
Not commissioned, externally peer-reviewed