22 CLINICAL CASES IN MINERAL AND BONE METABOLISM The Official Journal of the Italian Society of Orthopaedics and Medicine (OrtoMed) CIC Edizioni Internazionali Roma, Italyinfo@gruppocic.ithttp://www.gruppocic.com/http://www.ccmbm.com 2017 September - December; 14(3): 317–323. ISSN: 1724-8914 ISSN: 1971-3266
Published online 2017 December 27. doi: 10.11138/ccmbm/2017.14.3.317.

Surgical treatment of neglected hip fracture in children with cerebral palsy: case report and review of the literature

Giuseppe Toro,1 Antimo Moretti,1 Gabriella Toro,2 Assunta Tirelli,3 Giampiero Calabrò,4 Antonio Toro,4 and Giovanni Iolascon1

1Department of Medical and Surgical Specialties and Dentistry, University of Campania “Luigi Vanvitelli”, Naples, Italy
2Unit of Radiology and Nuclear Medicine, “Santa Maria della Speranza” Hospital, Battipaglia (SA), Italy
3“AOU San Giovanni di Dio e Ruggi d’Aragona”, Salerno, Italy
4Unit of Orthopaedics and Traumatology, “Martiri del Villa Malta” Hospital, Sarno (SA), Italy

Address for correspondence: Giovanni Iolascon, MD, Department of Medical and Surgical Specialties and Dentistry, University of Campania “Luigi Vanvitelli”, Naples, Italy, E-mail: giovanni.iolascon@gmail.com

Summary

Case
A clinical case of a 15-year-old cerebral palsy child with a Sandhu type 2 neglected femoral neck fracture is presented. The patient was treated using cannulated screws and cancellous bone graft augmented with mesenchymal stem cells. At 6 months after the surgery complete fracture healing was observed.

Conclusion
To early diagnose this fractures, it is mandatory to perform a comprehensive clinical and radiological evaluation including also a second level imaging. The use of cannulated screws with cancellous bone graft and MSCs is a viable treatment option in these patients.

Keywords: cerebral palsy, children’s hip fracture, neglected femoral neck fractures, mesenchymal stem cells, diamond concept, non-union

Introduction

Femoral neck fractures in children are uncommon and usually associated with high energy trauma (1, 2), but physically disabled children are at higher fracture risk with an increased incidence of low energy fractures (3) linked to lower bone density (4, 5) and impaired postural control (6). The treatment of neglected femoral neck fractures is still a challenge for the orthopaedic surgeon (7).

Case report

A 15-year-old boy with hemiplegic cerebral palsy (CP), causing a disability classified as Gross Motor Function Classification System (GMFCS) level II (8), sustained a femoral neck fracture (Delbet 2) after a fall from a standing position (9). It was initially misdiagnosed as a hip bruise (Figure 1). Over the next three months, the patient was forced to a wheelchair by an increasing hip pain. Because of the persistence of hip pain and flexion contracture and inability to support full weight bearing a standard radiographic exam was carried out showing a neglected femoral neck fracture classified as Sandhu type 2 (Figure 2) (10). Bone scan and MRI evaluations of the femoral head showed no signs of AVN (Figures 3, 4); an attempt of hip preservation surgery was performed using an antero-lateral hip approach. A T-shaped capsulotomy with postero-lateral flap preservation was performed to obtain a freshening fracture surfaces with a direct reduction (Figure 5 a). Three cannulated screws were inserted with a transverse one in the calcar. Autologous bone marrow stem cell concentrate, obtained by an iliac crest aspiration needle, was prepared using RegenExtracell® BMC protocol (RegenLab, Le Montsur-Lausanne, Switzerland) (Figure 5 b). The stem cells concentrate was then applied in hip fracture gap as a gelled membrane (Figure 5 c). Finally, a cancellous bone auto-graft from the greater trochanter was used to fill the fracture gap. Starting from the first day after surgery a gently mobilization of the fractured hip was implemented. In the third post-operative day, a specific rehabilitation protocol was prescribed in order to progressively regain the normal hip range of motion. Partial weight bearing was allowed as radiological fracture healing was demonstrated. Figure 6 showed X-ray evaluation at 6 months after surgery, reporting a complete fracture healing and no signs of AVN.

Figure 1Figure 1
Antero-posterior (a) and axial (b) X-rays at the time of the injury. The subsequent evaluation performed at the time of our observation allowed to identify a Delbet type 2 fracture, initially misdiagnosed as a hip bruise.
Figure 2Figure 2
In “a” antero-posterior radiograph at 9 months after the injury (in “b” detail), showing a Sandhu type 2 neglected femoral neck fracture.
Figure 3Figure 3
Bone scan performed at the time of our observation. No signs of AVN can be observed in the right hip.
Figure 4Figure 4
MRI performed at the time of our observation showing no signs of femoral head AVN.
Figure 5Figure 5
Intraoperative pictures. In “a” the antero-lateral approach performed in order to refresh and reduce the fracture; in “b” the stem cells concentrate in liquid and gelled physical state; in “c” application of the stem cells.
Figure 6Figure 6
Antero-posterior (a) and axial (b) hip radiographs at 6 months after surgery showing fracture healing and no signs of AVN.

At 12 months after surgery, the patient was able to walk without pain and aids. Hip flexion contracture was completely resolved, whereas a slight equinus (<10°) and a lower limb discrepancy of 0.5 centimetres were observed. The X-ray and MRI evaluations confirmed the bone healing and the absence of AVN (Figures 7, 8).

Figure 7Figure 7
Antero-posterior (a) and axial (b) hip radiographs at 12 months after surgery confirmed the absence of AVN.
Figure 8Figure 8
MRI performed at the time of our observation showing no signs of femoral head AVN.

Discussion

The main challenges for the orthopedic surgeon treating children affected by CP are spasticity, delay in developmental milestones, contractures of the soft tissue, bone deformities and their consequences on postural and gait control (11). Hip fractures occur more frequently in this population than in non-disabled children (3) and they are associated to a higher rate of complications compared to others injuries, such as AVN, non-union, coxa vara, and premature physeal closure (12). The overall complication rate was higher in Delbet type I and II (13), and in neglected fractures (7). According to Meyers et al. neglected femoral neck fractures are subacute fractures diagnosed at least 30 days after the injury (14). Sandhu et al. classified these fractures in 3 stages (Table 1) (10).

Table 1Table 1
Sandhu classification for neglected femoral neck fractures (10, 15).

Henderson et al. hypothesized that a misdiagnosis of fracture in CP could be related to the following conditions: 1) non-significant or recognized trauma; 2) communication impairments; 3) minimally displacement or angulation of the fracture (4).

The treatment of neglected femoral neck fractures is challenging and different techniques are available. Valgus intertrochanteric osteotomy, muscle pedicle bone grafting, reduction and fixation with fibular grafting, and reduction and fixation without fibular grafting are among the options reported (15). In our patient presenting a Sandhu type 2, according to Jain et al. (15), the treatment should be an open reduction (allowing fracture refreshing) and internal-fixation with bone grafting or valgisation osteotomy or open reduction and internal fixation with compression screws and free fibular graft. Valgisation osteotomy is one of the most common procedures performed in these patients. The Putti osteotomy showed its reliability in clinical outcome since 1956 (16) that was lately confirmed also by Zinghi et al. (17). However, these Authors suggested the use of Pauwel’s osteotomy with a degree of lateralization in order to compensate the shortening and prevent the Trendelenburg gait (17). The most common valgisation osteotomy performed is generally the Pauwel’s osteotomy with its several modifications. Recently Min et al. reported good mid-term outcomes in patients treated with a Pauwel-Muller’s osteotomy in 11 cases of non-union femoral neck fractures. However, the Authors hypothesized AVN of the femoral head as a possible complication (18).

The effectiveness of Pauwel’s osteotomy in neglected femoral neck fractures had been largely reported (15, 1922).

Magu et al. (20) investigating the effectiveness and safety of a modified Pauwel’s osteotomy in 10 children observed a bone union in 9 of them recommending their procedure as the treatment of choice for neglected femoral neck fractures in this population.

On the other hand, valgisation osteotomy is technically demanding and presents some limitations.

Kainth et al. supported the use of valgisation osteotomy in neglected femoral fractures only in selected patients, such as those with a femoral neck resorption less than 2.5 cm, absence of posterior comminution and good bone quality. The Authors argued that valgus osteotomy is technically more demanding and might result in intraoperative anterior angulation and difficulties in reduction in case of posterior comminution. Moreover, a low bone quality was associated with a blade plate cutting out. In these cases, the Authors suggested to use cancellous screw fixation and fibular graft (21). Varghese et al., studying factors affecting valgus osteotomy outcomes in neglected fractures, observed that reduced preoperative femoral neck absorption ratio (<0.5) was associated to non-union, whereas excessive valgus alignment was associated with worse functional outcome (22).

In particular, excessive valgus alignment could be related to persistent limp and early osteoarthritis and might affect the abductor lever length (23). Wu et al., comparing the use of a compression sliding screw with and without valgus osteotomy, observed that the addition of valgus osteotomy was able to correct a limb shortening of at least 1.5 cm, but it was not associated with a higher rate of bone union, and a higher complication rate (24).

Our patient presented a limb shortening of 1 cm, therefore we preferred to not perform the valgisation osteotomy to preserve the abductor lever arm.

Muscle pedicle bone graft (MPBG) is another available option with good outcomes (14, 15, 2527). Several MPBGs had been described (i.e. quadratus femoris, tensor fasciae latae and gluteus medius) and the choice among them depends on the surgeon preference. Hou et al. showed complete fracture healing in all of 5 cases treated using vascularized iliac bone graft (26). However, MPBG use is technically demanding and requires microsurgical facilities and experience, which are not widely available (27). Although vascularized fibular bone graft might provide a plus for femoral head vitality (28), nonvascularized one seems to be a dependable and technically less demanding choice (23) in cases of neglected fracture Sandhu 1 and 2 (15). In fact, the fibular graft provides mechanical support and a channel for revascularisation (26, 27). Nagi et al. reported a 70% of good results and only 4 AVN at 6-year-follow-up in 40 patients treated with a free fibular graft and one cancellous screw (29). However, their use was associated with some complications such as donor site pain, graft fracture, and gait pattern modification (15, 23, 2931). We preferred to avoid the use of fibular strut graft considering the risk of gait impairment as reported in literature (31).

Several Authors showed good outcomes using cannulated screws associated with both fibular strut graft (29,30,32) and cancellous bone graft (33). Recently, a biomechanical study demonstrated that placing a third cannulated cancellous screw in the calcar provide a stability advantage for femoral neck fractures fixation (34).

Therefore, we planned to perform an open reduction to refresh the fracture surfaces and to fix it using three cannulated screws with a transverse one in the calcar. The recent introduction of stem cells implantation for fracture non-union treatment is part of the “diamond concept” conceived by Giannoudis et al. (35). Marcucio et al. hypothesized that mesenchymal stem cells (MSCs) might influence bone repair through different mechanisms: 1) differentiation into osteoblasts; 2) triggering the division and differentiation of native connective tissue progenitors; 3) modulating cells of the immune system; 4) secretion of trophic molecules that inhibit apoptosis and fibrosis and/or promote angiogenesis; 5) homing to the fracture site through chemokine receptors, such as CCR1, CCR7, CCR9, and CXCR4–6 and other pathways (36). MSCs use is widely accepted for the treatment of AVN of the femoral head (37, 38) and recently their use has been demonstrated to improve intertrochanteric fracture healing (39). The effectiveness of MSCs in neglected femoral neck fractures was tested by Ayoub et al. in a series of 36 patients between 15 and 50 years (33) that reported an union rate of 94.4% using cancellous screws and MSCs. The RegenExtracell® BMC protocol (RegenLab, Le Mont-sur-Lausanne, Switzerland) is a promising technique of MSCs concentration that allows to achieve recovery rates from bone marrow aspirate of 87% of MSCs (40). In our case, we used the MSCs in two different physical states: liquid and gelled by injecting them into the femoral head and filling the fracture gap. Finally, we used cancellous bone graft from the greater trochanter to completely fill the fracture gap. To identify the appropriate surgical treatment of neglected femoral neck fractures in CP children, we suggest that the surgeon should perform a comprehensive clinical and radiological evaluation, including the GMSCF. In our experience, the use of cannulated screws with cancellous bone graft and MSCs is a viable treatment option in these patients.

Informed consent

This case report respects the Declaration of Helsinki, approved by the Ethical Committee of Unit of Orthopaedics and Traumatology, Martiri del Villa Malta Hospital, Sarno, Italy. Parents were asked to carefully read and sign an informed consent.

Acknowledgements

No funding or sponsorship was received for this study or publication of this article. All named Authors meet the International Committee of Medical Journal Editors (ICMJE) criteria for authorship for this manuscript, take responsibility for the integrity of the work as a whole, and have given final approval for the version to be published.

References
1.
Bagatur AE, Zorer G. Complications associated with surgically treated hip fractures in children. J Pediatr Orthop Part B. 2002;11:219–28.
2.
Eberl R, Singer G, Ferlic P, Weinberg AM, Hoellwarth ME. Post-traumatic coxa vara in children following screw fixation of the femoral neck. Acta Orthop. 2010;81:442–5. doi:10.3109/17453674.2010.501744.
3.
Maruyama K, Nakamura K, Nashimoto M, Kitamoto F, Oyama M, Tsuchiya Y, et al. Bone fracture in physically disabled children attending schools for handicapped children in Japan. Environ Health Prev Med. 2010;15:135–40. doi:10.1007/s12199-009-0121-x.
4.
Henderson RC, Berglund LM, May R, Zemel BS, Grossberg RI, Johnson J, et al. The relationship between fractures and DXA measures of BMD in the distal femur of children and adolescents with cerebral palsy or muscular dystrophy. J Bone Miner Res Off J Am Soc Bone Miner Res. 2010;25:520–6. doi:10.1359/jbmr.091007.
5.
Finbråten A-K, Syversen U, Skranes J, Andersen GL, Stevenson RD, Vik T. Bone mineral density and vitamin D status in ambulatory and non-ambulatory children with cerebral palsy. Osteoporos Int. 2015;26:141–50.
6.
Szopa A, Domagalska-Szopa M. Postural stability in children with hemiplegia estimated for three postural conditions: Standing, sitting and kneeling. Res Dev Disabil. 2015;39:67–75.
7.
Roshan A, Ram S. The Neglected Femoral Neck Fracture in Young Adults: Review of a Challenging Problem. Clin Med Res. 2008;6:33–9.
8.
Palisano R, Rosenbaum P, Walter S, Russell D, Wood E, Galuppi B. Development and reliability of a system to classify gross motor function in children with cerebral palsy. Dev Med Child Neurol. 1997;39:214–23.
9.
Delbet MP. Fractures du col de femur. Bull Mem Soc Chir. 1907;35:387–9.
10.
Sandhu HS, Sandhu PS, Kapoor A. Neglected fractured neck of the femur: a predictive classification and treatment by osteosynthesis. Clin Orthop. 2005:14–20.
11.
Ajami S, Maghsoudlorad AA. The Role of Information Systems to Manage Cerebral Palsy. Iran J Child Neurol. 2016;10:1–9.
12.
Forlin E, Guille JT, Kumar SJ, Rhee KJ. Complications associated with fracture of the neck of the femur in children. J Pediatr Orthop. 1992;12:503–9.
13.
Yeranosian M, Horneff JG, Baldwin K, Hosalkar HS. Factors affecting the outcome of fractures of the femoral neck in children and adolescents: a systematic review. Bone Jt J. 2013;95-B:135–42.
14.
Meyers MH, Harvey JP, Moore TM. Treatment of displaced subcapital and transcervical fractures of the femoral neck by muscle-pedicle-bone graft and internal fixation. A preliminary report on one hundred and fifty cases. J Bone Joint Surg Am. 1973;55:257–74.
15.
Jain A, Mukunth R, Srivastava A. Treatment of neglected femoral neck fracture. Indian J Orthop. 2015;49:17.
16.
Ponzoni A. Results of intertrochanteric osteotomy in inveterate femur neck fractures. Minerva Ortop. 1956;7:672–3.
17.
Zinghi GF, Specchia L, Ruggieri N, Galli G. The role of osteotomy in the treatment of pseudarthrosis of the neck of the femur in younger patients. Ital J Orthop Traumatol. 1985;11:341–8.
18.
Min B-W, Bae K-C, Kang C-H, Song K-S, Kim S-Y, Won Y-Y. Valgus intertrochanteric osteotomy for non-union of femoral neck fracture. Injury. 2006;37:786–90.
19.
Kalra M, Anand S. Valgus intertrochanteric osteotomy for neglected femoral neck fractures in young adults. Int Orthop. 2001;25:363–6.
20.
Magu NK, Singh R, Sharma AK, Ummat V. Modified Pauwels? Intertrochanteric Osteotomy in Neglected Femoral Neck Fractures in Children: A Report of 10 Cases Followed for a Minimum of 5 Years. J Orthop Trauma. 2007;21:237–43.
21.
Kainth GS, Yuvarajan P, Maini L, Kumar V. Neglected femoral neck fractures in adults. J Orthop Surg Hong Kong. 2011;19:13–7.
22.
Varghese VD, Boopalan PR, Titus VTK, Oommen AT, Jepegnanam TS. Indices affecting outcome of neglected femoral neck fractures after valgus intertrochanteric osteotomy. J Orthop Trauma. 2014;28:410–6.
23.
Azam MQ, Iraqi A, Sherwani M, Sabir A, Abbas M, Asif N. Free fibular strut graft in neglected femoral neck fractures in adult. Indian J Orthop. 2009;43:62.
24.
Wu CC, Shih CH, Chen WJ, Tai CL. Treatment of femoral neck non-unions with a sliding compression screw: comparison with and without subtrochanteric valgus osteotomy. J Trauma. 1999;46:312–7.
25.
Huang CH. Treatment of neglected femoral neck fractures in young adults. Clin Orthop. 1986:117–26.
26.
Hou SM, Hang YS, Liu TK. Ununited femoral neck fractures by open reduction and vascularized iliac bone graft. Clin Orthop. 1993:176–80.
27.
Nair N, Patro DK, Babu TA. Role of muscle pedicle bone graft as an adjunct to open reduction and internal fixation in the management of neglected and ununited femoral neck fracture in young adults: a prospective study of 17 cases. Eur J Orthop Surg Traumatol. 2014;24:1185–91.
28.
Soucacos PN, Dailiana Z, Beris AE, Johnson EO. Vascularised bone grafts for the management of non-union. Injury. 2006;37(Suppl 1):S41–50.
29.
Nagi ON, Dhillon MS, Goni VG. Open reduction, internal fixation and fibular autografting for neglected fracture of the femoral neck. J Bone Joint Surg Br. 1998;80:798–804.
30.
Pal CP, Kumar B, Dinkar KS, Singh P, Kumar H, Goyal RK. Fixation with cancellous screws and fibular strut grafts for neglected femoral neck fractures. J Orthop Surg Hong Kong. 2014;22:181–5.
31.
Ling XF, Peng X. What Is the Price to Pay for a Free Fibula Flap? A Systematic Review of Donor-Site Morbidity following Free Fibula Flap Surgery. Plast Reconstr Surg. 2012;129:657–74.
32.
Roshan A, Ram S. Early return to function in young adults with neglected femoral neck fractures. Clin Orthop. 2006;447:152–7.
33.
Ayoub MA, Gad HM. Neglected neck femur fractures in adolescents and young adults: factors predicting the surgical outcome. J Orthop Sci. 2013;18:93–100.
34.
Gumustas SA. Influence of number and orientation of screws on stability in the internal fixation of unstable femoral neck fractures. ACTA Orthop Traumatol Turc. 2014;48:673–8.
35.
Giannoudis PV, Ahmad MA, Mineo GV, Tosounidis TI, Calori GM, Kanakaris NK. Subtrochanteric fracture non-unions with implant failure managed with the “Diamond” concept. Injury. 2013;44:S76–81.
36.
Marcucio RS, Nauth A, Giannoudis PV, Bahney C, Piuzzi NS, Muschler G, et al. Stem Cell Therapies in Orthopaedic Trauma. J Orthop Trauma. 2015;29:S24–7.
37.
Papakostidis C, Tosounidis TH, Jones E, Giannoudis PV. The role of “cell therapy” in osteonecrosis of the femoral head. A systematic review of the literature and meta-analysis of 7 studies. Acta Orthop. 2016;87:72–8.
38.
Calori GM, Mazza E, Colombo M, Mazzola S, Mineo GV, Giannoudis PV. Treatment of AVN using the induction chamber technique and a biological-based approach: indications and clinical results. Injury. 2014;45:369–73.
39.
Torres J, Gutierres M, Lopes MA, Santos JD, Cabral AT, Pinto R, et al. Bone marrow stem cells added to a hydroxyapatite scaffold result in better outcomes after surgical treatment of intertrochanteric hip fractures. BioMed Res Int. 2014;2014:451781.
40.
RegenLab SA. RegenExtracell BMC & GLUE product characteristics n.d. [accessed December 30, 2016]. https://www.regenlab.com/regenextracell-bmc/product/