Bone Plating in Patients with Type III Osteogenesis Imperfecta: Results and Complications

William J Enright, MD and Kenneth J Noonan, MD

Abstract
The results of bone plating in four children (6 femurs, 2 tibias) with osteogenesis imperfecta type III were analyzed. Average age at time of operation was 44 months. In three of the femurs, multiple platings were performed for a total of 13 bone platings in the eight bones studied. Average time to revision following plating was 27 months. Indications for revision included fracture, deformity, hardware failure, and nonunion. Other complications included one case of compartment syndrome. All eight bones were ultimately revised to elongating intramedullary Bailey-Dubow rods. Bone plating in skeletally immature patients with osteogenesis imperfecta does not provide better outcome than elongating rods. Complications from bone plating leading to revision, such as refracture or hardware failure, are higher than in those children managed with elongating rods, as previously reported in the literature.

Introduction
Osteogenesis imperfecta (OI) is a group of inherited disorders caused by defective type I collagen synthesis. Using the Sillence classification, one can determine the type of OI based on clinical, radiographic, and genetic findings. Patients with OI can suffer from frequent fractures and deformity of the long bones during development, resulting in impaired ambulation. The goal of orthopedic surgery for OI is twofold: Reduce the incidence of fractures and correct long bone deformity. Contemporary surgical options for deformed bones in OI include osteotomy and stabilization with non-elongating nails (Rush rods, flexible nails), elongating nails (Bailey- Dubow, Frasier-Duval), and bone plating. Elongating rods allow for growth of the bone, thereby decreasing the number of repeat operations. The advantages of elongating rods over fixed intramedullary rods include benefit to growing bones, lower incidence of re-fracture, and longer time to re-operation. There is also evidence that suggests that elongating rods used in the femur do not require revision as often as those placed in the tibia. At our institution, we have utilized plate fixation for stabilization of osteotomies in young patients with severe OI. Plate fixation was initially appealing in this group of patients given the age of the individuals and the difficulty of placing expandable rods in small bones. In addition, the treating surgeon felt that these rods were too large for the smallest children, thus resulting in stress shielding and bone atrophy. The purpose of this study is to review our experience in this small but select group of patients.

MATERIALS AND METHODS This study is a retrospective review of all patients with osteogenesis type III treated with bone plating for correction of deformity or treatment of fracture. All operations were performed by the same pediatric orthopedic surgeon between 1994 and 2001. Inclusion criteria for this study were a diagnosis of type III osteogenesis imperfecta, history of bone plating, and recent clinical follow-up. After review of the medical files of all patients treated for osteogenesis imperfecta at the University of Wisconsin Hospital and Clinics, we were able to find four patients who had undergone at least one bone plating as treatment for fracture or deformity. Clinical records and imaging studies were reviewed. We recorded the indications for initial plating, types of plates and screws used, time to evidence of healing

on radiograph, time to revision, indications for revisions, hardware used in revision (plate or rod), number and location of fractures following each plating, complications including hardware failure, and number of revisions for each bone. Regarding the measurement of time to fracture and time to revision, the authors considered each plating separately. There were cases of sequential platings of the same bone in patients where initial plating was revised with further plating. Initial plating was considered as the start point for this study, and revision with expandable rods was considered the end point.

RESULTS All four patients were diagnosed with osteogenesis type III using the Sillence classification.13 There were three males and one female, ranging in age from 14 to 82 months (44.7 months mean age) at the time of surgery. Ages ranged from five to eight years (7.7 years mean age) at last follow-up. The average time from initial plating to final follow-up period was four years. Thirteen bone platings were performed on eight bones. The eight bones included six femurs and two tibias. Three of the femurs underwent multiple platings before being ultimately revised to Bailey-Dubow rods. Of these three femurs, two were plated twice, and one femur was plated four times for a total of 13 platings. All platings were separate operations. No two bones were plated at the same time. The indications for initial plating of the eight bones included fracture and deformity. Of the six femurs, four were plated because of fracture, and two were plated for correction of deformity. Of the two tibias, one was plated for correction of deformity and the other because of fracture. All eight bones ultimately required revision. Three of the femurs underwent further plating for revision, while the two tibias and three other femurs were revised to Bailey-Dubow rods. Indications for revision included fracture (6), deformity (3), hardware failure (3), and nonunion (1). Rate of fracture following plating was 46% (six fractures). Location of fracture was distal to the plate in two cases, under the plate in two cases, and through the plate in two cases. In the two cases of fracture through the plate, fracture of bone with broken plate was considered the reason for revision and also considered a complication (Figures 1 and and2).2). The average time to revision was 27 months (range 4 to 71 months). The average time from initial plating to final revision with Bailey-Dubow rods was 42 months (range 9 to 89 months) for all bones.

Figure 2 Radiograph showing screw pull-out. Note the proximal and distal cancellous screws have pulled out, allowing the plate to displace from the bone and leading to hardware prominence. The complication rate in these patients was 69.2% (9 plates). The most common complication following plating was screw pull-out. Screw pull-out was seen following plating in five cases. One case involved multiple screws and required revision for stabilization. Two fractures through the plate were seen, and these underwent revision. Bending of two of the plates was observed. Of these nine complications, three instances of hardware failure led to revision: Screw pull-out required revision in one case, and two fractures went through the plate as mentioned above. Complications are listed in Table 1.

TABLE 1 There was one case of compartment syndrome following plating of a tibia, which required fasciotomy. There was one case of nonunion in a femur. This nonunion was noted five months after the initial plating and was revised with bone plating seven months after the initial operation. There were three instances of prominent hardware, one of which was symptomatic. Go to: DISCUSSION Bone plating is an option in the treatment of fracture and deformity in children with osteogenesis imperfecta. Previous studies in the orthopedic literature report treatment of these patients with intramedullary rods, both fixed and elongating. The benefits of elongating rods over fixed rods have been demonstrated in regard to reduction in the number of operations performed and facilitation of growth.4,5,12 There are no studies, however, examining the results of bone plating in comparison to the results obtained with elongating intramedullary rods.

The purpose of this study was to examine the results of bone plating in patients with osteogenesis imperfecta. Average time to revision following plating was 27 months. This compares quite unfavorably with the five years to revision following placement of Bailey-Dubow rods reported by Luhmann et al.9 It compares more favorably with the average time to revision of 2.5 years following placement of nonelongating rods reported by Marafioti and Westin.10 The most common indication for revision following plating was fracture (6), followed by deformity (3) and hardware failure (3). The complication rate of plating was 69.2%. This rate was slightly higher than the 63.5% complication rate previously reported by Jerosch et al. and significantly higher than the 27% complication rate reported by Marafioti and Westin in their treatment of patients with OI.6,10 Jerosch et al. implanted Bailey-Dubow rods in 107 bones and Kirschner wires in eight bones.6 In their study, the Kirschner wires were implemented because of small bone diameter. The most common complication of Bailey-Dubow rods has been reported to be rod migration.6,7 The most common complication seen after plating was screw pull-out. This seems intuitive given the quality of bone in patients with osteogenesis imperfecta. Not only does the bone quality not allow for purchase of the screws, the bowing of bones may act to further any screw pull-out. Screw pull-out was not a clinical problem in this series unless it was associated with increasing deformity or fracture. The treatment plan for skeletally immature patients with osteogenesis imperfecta must include consideration of growth. The advantage of the elongating rod is that it allows for longitudinal bone growth. The rod does cross the physis, but the diameter of the rod is small enough not to affect growth.2Bone plating does not disturb the physis in most cases, but it does not migrate with growth, thus leaving unsupported bone. Higher revision and failure rates in the bone adjacent to the plate are also most likely due to the sharp disparity in construct rigidity and osteopenic metaphyseal bone. Considering the higher complication rates, shorter length of time to revision, and unknown effect on longitudinal growth, bone plating does not compare favorably to elongating rods in patients with osteogenesis imperfecta. We recommend elongating rods when considering treatment of deformity or fracture in patients with osteogenesis imperfecta. Go to: Footnotes Study conducted at University of Wisconsin Hospital and Clinics, Madison, WI Go to: References 1. Bailey RW. Studies of longitudinal bone growth resulting in an extensible nail. Surg Forum.1963;14:455458. [PubMed] 2. Bailey RW. Further clinical experience with the extensible nail. Clin Orthop. 1981;159:171 176.[PubMed] 3. Bailey RW, Dubow HI. Evolution of. the concept of an extensible nail accommodating to normal longitudinal bone growth: clinical considerations and implications. Clin Orthop. 1981;159:157 170.[PubMed] 4. Gamble JG, Strudwick WJ, Rinsky LA, Bleck EE. Complications of intramedullary rods in osteogenesis imperfecta: Bailey-Dubow rods versus nonelongating rods. J Pediatric Ortho.1988;8(6):645649. 5. Harrison WJ, Rankin KC. Osteogenesis imperfecta in Zimbabwe: a comparison between treatment with intramedullary rods of fixed-length and self-expanding rods. J Royal College Surg Edinburgh.1998;43(5):328332.

6. Jerosch J, Mazzotti I, Tomasevic M. Complications after treatment of patients with osteogenesis imperfecta with a Bailey-Dubow rod. Archives Ortho & Trauma Surg. 1998;117(4-5):240245. 7. Lang-Stevenson AI, Sharrard WJ. Intramedullary rodding with Bailey-Dubow extensible rods in osteogenesis imperfecta: an interim report of results and complications. J Bone Joint Surg Br. 1984;66B(2):227232. [PubMed] 8. Li YH, Chow W, Leong JC. The Sofield-Millar operation in osteogenesis imperfecta: a modified technique. J Bone Joint Surg Br. 2000;82-B(1):1116. [PubMed] 9. Luhmann SJ, Sheridan JJ, Capelli AM, Schoenecker PL. Management of lower extremity deformities in osteogenesis imperfecta with extensible intramedullary rod technique: a 20-year experience. J Pediatric Ortho. 1998;18(1):8894. 10. Marafioti RL, Westin GW. Elongating intramedullary rods in the treatment of osteogenesis imperfecta. J Bone Joint Surg Am. 1977;59A(4):467472. [PubMed] 11. Mulpuri K, Joseph B. Intramedullary rodding in osteogenesis imperfecta. J Pediatric Ortho.2000;20(2):267273. 12. Porat S, Heller E, Seidman DS, Meyer S. Functional results of operation in osteogenesis imperfecta: elongating and nonelongating rods. J Pediatric Ortho. 1991;11(2):200203. 13. Sillence D. Osteogenesis imperfecta: an expanding panorama of variants. Clin Orthop. 1982;159:11 25. [PubMed] 14. Sofield HA, Millar EA. Fragmentation, realignment, and intramedullary rod fixation of deformities of the long bones in children. J Bone Joint Surg. 1959;41A:1371. 15. Stockley I, Bell MJ, Sharrard WJ. The role of expanding intramedullary rods in osteogenesis imperfecta. J Bone Joint Surg Br. 1989;71-B(3):422427. [PubMed] 16. Zionts LE, Ebramzadeh E, Stott NS. Complications in the use of the Bailey-Dubow extensible nail.Clin Orthop. 1998;348:186195. [PubMed]