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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 53  |  Issue : 4  |  Page : 152-155

Functional outcome in critical-size defect aneurysmal bone cyst of the distal humerus patient treated with curettage and nonvascularized autologous fibular graft


Department of Orthopaedics and Traumatology, Faculty of Medicine, Sardjito General Hospital, Universitas Gadjah Mada, Yogyakarta, Indonesia

Date of Submission18-Feb-2020
Date of Decision09-Mar-2020
Date of Acceptance22-Apr-2020
Date of Web Publication20-Aug-2020

Correspondence Address:
Rizky Admagusta
Jalan Kesehatan No. 1, Sekip, Yogyakarta 55284
Indonesia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/fjs.fjs_16_20

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  Abstract 


Aneurysmal bone cyst (ABC) is a rare benign expansive cystic lesion characterized by blood-filled cavernous spaces separated by septae containing osteoid tissue and osteoclast giant cells. Treatment goals are to stop progression of the lesion, relieve pain, stabilize pathologic fractures, and reduce recurrence rates. The standard of care for ABCs is curettage with or without bone graft depending on the resultant void. Despite best efforts at curettage, clinical series have shown highly variable recurrence rates, with some series showing rate ranges from 0% to 100%. Tumor resection may be associated with critical bone loss. Critical-size defects require reconstruction. Other than that, functional outcome after surgical treatment in patients with critical size defect is still a challenge even in benign tumor case. We reported a 32-year-old female presenting an entirely contained ABC to the distal metaphysis of the humerus treated with curettage and nonvascularized autologous fibular bone graft.

Keywords: Aneurysmal bone cyst, curettage, fibular graft, functional outcome


How to cite this article:
Magetsari R, Putro YA, Admagusta R. Functional outcome in critical-size defect aneurysmal bone cyst of the distal humerus patient treated with curettage and nonvascularized autologous fibular graft. Formos J Surg 2020;53:152-5

How to cite this URL:
Magetsari R, Putro YA, Admagusta R. Functional outcome in critical-size defect aneurysmal bone cyst of the distal humerus patient treated with curettage and nonvascularized autologous fibular graft. Formos J Surg [serial online] 2020 [cited 2020 Sep 25];53:152-5. Available from: http://www.e-fjs.org/text.asp?2020/53/4/152/292725




  Introduction Top


Aneurysmal bone cyst (ABC) is a rare benign expansive cystic lesion characterized by blood-filled cavernous spaces separated by septae containing osteoid tissue and osteoclast giant cells.[1] ABCs occur with an estimated incidence of 0.14–0.32 per 100.000 individuals.[1] ABCs are commonly seen during childhood and young adulthood with a median age of 13 years, and 90% of lesions are found prior to 30 years.[2] Females are slightly more affected with an estimated male-to-female sex ratio of 1:1.16.[2] ABCs have a predilection for the metaphysis of long bones including the femur, tibia/fibula, and upper extremity.[2] ABCs are generally solitary and are currently thought to arise either as a primary neoplasm (translocation driven) or secondary lesions arising adjacent to osteoblastomas, chondroblastomas, or giant cell tumors (GCTs), among others.[3] Treatment goals are to stop progression of the lesion, relieve pain, stabilize pathologic fractures, and reduce recurrence rates. To this end, various treatment modalities have been described, ranging from surgical procedures to radiation to medical therapies. The standard of care for ABCs is curettage with or without bone graft depending on the resultant void. Curettage is associated with an acceptable rate of local control, which is approximately 90% in large series.[4] Despite best efforts at curettage, clinical series have shown highly variable recurrence rates, with some series showing rate ranges from 0% to 100%.[4]

The etiology of critical bone defects is varied, and tumor resection may be associated with critical bone loss.[5] General guidelines that have been suggested in the literature include defect length >1–2 cm and >50% loss of the circumference of the bone.[5] Critical-size defects require reconstruction. Autogenous bone graft is still the gold standard for management, and it remains unclear when alternative biologic therapies should be considered.[5] Other than that, functional outcome after surgical treatment in patients with critical size defect is still a challenge even in benign tumor case. We reported a 32-year-old female presenting an entirely contained ABC to the distal metaphysis of the humerus treated with curettage and nonvascularized autologous fibular bone graft.


  Case Report Top


A 32-year-old female presented to the outpatient clinic with complaints of right arm pain of 1-week duration. The patient fell on outstretched hand then unable to move her arm. There were no other symptoms or complaints. On physical examination, there were pain and tenderness located over the right aspect of the distal third of the right arm. There was limited range of motion of the shoulder and elbow caused by pain. The overlying skin was intact with no changes. The neurovascular examination was normal.

The initial radiographs [Figure 1] showed a fracture and a well-defined, eccentric, radiolucent lesion in the middle third of the humerus. Magnetic resonance imaging (MRI) [Figure 2] showed fluid–fluid levels caused by the different densities of the cyst fluid owing to the setting of red blood cells. The patient underwent fine-needle aspiration biopsy-guided ultrasonography.
Figure 1: Initial radiographs

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Figure 2: Magnetic resonance imaging of the patient

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After the diagnosis of ABC was confirmed with no signs of any other associated tumors, she underwent a surgery and bone grafting during the same anesthetic event [Figure 3]. The surgical plan was curettage of the lesion with incorporation of an autogenous interpositional nonvascularized fibular graft. In the surgery, a 15 cm posterior approach of humerus incision was placed over the cystic lesion. The incision passed through layers to the level of the cyst, exposing the full length of the affected bone. The radial nerve could be preserved well. As we could see from the initial radiograph and MRI, the cortical breakage already took place. The thin-walled tumor was then performed curettage. The bone defect was 4 cm and then replaced by a segment from the ipsilateral fibula and fixed by two reconstruction plates.
Figure 3: Surgery and Bone Grafting Performed to the Patient

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All lesion tissue was sent for pathologic evaluation intraoperatively. The final histopathologic analysis showed histologic features that were most consistent with those of an ABC. Postoperatively, neurovascular deficit was found. The patient used an arm sling for 3 weeks, then gradually moved the elbow. At the 3-month follow-up, the patient complained no pain when using her arm and good neurovascular status. There was slight limitation in shoulder forward flexion and abduction. There was also a limitation in elbow flexion, but the functional status of the upper extremity is fine. It was represented by a good result of Dash score, 10.4. The radiological evaluation also showed a good result [Figure 4].
Figure 4: (a) Postoperative plain film. (b) Plain-film at 3-month follow-up. (c) Plain film at 6-month follow-up

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  Discussion Top


ABC is classified as an aggressive benign bone tumor, which means if not treated properly, it may recur, or if left untreated, it may get larger and be called giant ABC.[1] The pathogenesis of ABC is still unknown, and the two broad types are either primary (70%) or secondary (30%).[6] Primary ABCs arise de novo. A secondary ABC develops in association with other neoplasms, most commonly GCT of the bone, osteoblastoma, chondroblastoma, and fibrous dysplasia.[7] The diagnosis of an ABC shows five classic findings in radiographic examination: (1) an expansive lytic lesion with a soap bubble appearance, (2) an eccentric lesion outlined by a thin layer of subperiosteal new bone, (3) a centric lesion, (4) metaphyseal lesion that occupies a large percentage of the bone with trabeculations at the edges, and (5) soft-tissue expansion and destruction of the cortex. This patient fit with the criteria definitely.[7]

Most of the available surgical methods are suitable for treating metaphyseal localized lesions. Bone defect after the use of surgical method must be treated with bone grafting as a primary treatment along with or without adjuvant methods. [8,9] In our patients, curettage would have been sufficient to cause permanent cure. To deal with the critical size bone defect, we chose to use non-ascularized autologous fibular graft. Nonvascularized autologous bone grafts are biologically active grafts with relatively low donor-site complications that will be replaced completely by living bone and that are capable of remodeling to fulfill the function need.[10] Critical-size defects require reconstruction, and the gold standard has been iliac crest bone graft, but we chose to use nonvascularized autologous fibular graft instead because the contour of the fibular mimics the humerus.[4] It provides osteoconduction and osteogenesis properties to the bone defect.[11] The long-term possible complications of this procedure include fracture of the graft, nonunion, failure of incorporation of the graft, and stiffness or degenerative arthrosis in the joints.[12] To achieve graft consolidation, the graft should be in a stable condition. In our case, we use two reconstruction plates to fix the graft with the donor site. This will promote union of the cortical graft to the host bone.[13] Our patient showed no sign of infection, and consolidation of the fibula at the defect site occurred within 3 months after the surgery; Basarir et al. also showed the same result.

We also evaluated the patient's functional outcome pre- and postoperatively using DASH score. The DASH can detect and differentiate small and large changes in disability over time after surgery in patients with upper extremity musculoskeletal disorders. A 10-point difference in the mean DASH score might be considered as a minimal important change. The effect size and standardized response mean (commonly used indices of the magnitude of health change measured by questionnaires) may yield substantially differing results.[14] Our patient shows 6-point difference in the mean DASH score. The patient showed good functional outcome, even though there is a limitation of range of movement after the surgery.


  Conclusion Top


ABC patients of the distal humerus with critical-size defect treated with curettage and non-vascularized autologous fibular graft showed good radiological evaluation, clinical result, and range of movement and functional outcome, with the result of 10.4 measured using DASH Score.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Grzegorzewski Pogonowicz E, Sibinski M, Marciniak M, Synder M. Treatment of benign lesions of humerus with resection and non-vascularised, autologous fibular graft. Int Orthopaed (SICOT) 2010;34:1267-72.  Back to cited text no. 1
    
2.
Leithner A, Windhager R, Lang S, Haas OA, Kainberger F, Kotz R. Aneurysmal bone cyst. A population based epidemiologic study and literature review. Clin Orthop Relat Res 1999;(363):176-9.  Back to cited text no. 2
    
3.
Martinez V, Sissons HA. Aneurysmal bone cyst. A review of 123 cases including primary lesions and those secondary to other bone pathology. Cancer 1988;61:2291-304.  Back to cited text no. 3
    
4.
Mankin HJ, Hornicek FJ, Ortiz-Cruz E, Villafuerte J, Gebhardt MC. Aneurysmal bone cyst: A review of 150 patients. J Clin Oncol 2005;23:6756-62.  Back to cited text no. 4
    
5.
Nauth A, Schemitsch E, Norris B, Nollin Z, Watson JT. Critical-size bone defects: Is there a consensus for diagnosis and treatment? J Orthop Trauma 2018;32 Suppl 1:S7-11.  Back to cited text no. 5
    
6.
Bonakdarpour A, Levy WM, Aegerter E. Primary and secondary aneurysmal bone cysts: A radiological study of 75 cases. Radiology 1978;126:75-83.  Back to cited text no. 6
    
7.
Tsagozis P, Brosjö O. Current strategies for the treatment of aneurysmal bone cysts. Orthop Rev (Pavia) 2015;7:6182.  Back to cited text no. 7
    
8.
Abuhassan FO, Shannak A. Non-vascularized fibular graft reconstruction after resection of giant aneurysmal bone cyst (ABC). Strat Traum Limb Recon 2010;5:149-54.  Back to cited text no. 8
    
9.
Campanacci M, Capanna R, Picci P. Unicameral and aneurysmal bone cysts. Clin Orthop 1986;204:25-36.  Back to cited text no. 9
    
10.
George B, Abudu A, Grimer RJ, Carter SR, Tillman RM. The treatment of benign lesions of the proximal femur with nonvascularised autologous fibular strut grafts. J Bone Joint Surg Br 2008;90:648-51.  Back to cited text no. 10
    
11.
Zwierzchowski H, Zwierzchowska D, Synder M. The value of fibular autografts in block resection of bone tumours and tumour like conditions. Int Orthop 1989;13:113-7.  Back to cited text no. 11
    
12.
Finkemeier CG. Bone grafting and bone graft substitutes. J Bone Joint Surg Am 2002;84:454-64.  Back to cited text no. 12
    
13.
Basarir K, Selek H, Yildiz Y, Saglik Y. Nonvascularized fibular grafts in the reconstruction of bone defects in orthopedic oncology. Acta Orthop Traumatol Turc 2005;39:300-6.  Back to cited text no. 13
    
14.
Gummesson C, Atroshi I, Ekdahl C. The disabilities of the arm, shoulder and hand (DASH) outcome questionnaire: Longitudinal construct validity and measuring self-rated health change after surgery. BMC Musculoskelet Disord 2003;4:11.  Back to cited text no. 14
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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