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At least 199 records · Page 11Linked to original sources

PubMed2004

Vertical distraction osteogenesis of the mandible applied to an iliac crest graft: report of a case.

OBJECTIVES: This clinical case represents a particular application of vertical distraction osteogenesis of the mandible. The surgical technique was applied to an iliac crest bone graft previously positioned on an anterior mandibular defect with the aim to restore the vertical height after a partial resorption in a trauma patient. CASE REPORT: Distraction osteogenesis was applied because it was necessary to restore the mandibular height in a young male patient who presented with a post-trauma anterior mandibular bony deficiency. Because of the irregularity of the mandibular surface and the depth of the defect, the patient first underwent an iliac crest bone grafting that, after 3 months, showed partial resorption. Vertical distraction osteogenesis was performed on the grafted mandible in order to obtain a satisfactory bony height of the mandibular ridge. Then the patient was successfully rehabilitated by means of an implant-supported prosthesis. CONCLUSION: Vertical distraction osteogenesis appears to be a versatile surgical procedure, which can be applied as a secondary treatment in patients presenting with mandibular trauma sequelae.

Adult↗

PubMed2007

Enamel matrix derivative stimulates osteogenesis- and chondrogenesis-related transcription factors in C3H10T1/2 cells.

Our purpose was to determine how enamel matrix derivative (EMD) affects the expression of osteogenesis- and chondrogenesis-related transcription factors in undifferentiated mesenchymal cells. C3H10T1/2 cell line, a typical pluripotential mesenchymal cell line, was cultured with or without EMD for up to 7 d. Expression of mRNAs encoding osteogenesis- and chondrogenesis-related transcription factors (Runx2, Osterix, AJ18, Dlx5, Msx2, Sox5, Sox9 and Zfp60) was measured using real-time polymerase chain reaction. Runx2 and Sox9 protein expression and the presence of bone morphogenetic protein (BMP)-6-like molecules in EMD were determined by Western blotting. EMD substantially increased mRNA levels of osteogenesis- and chondrogenesis-related transcription factors. EMD also induced Runx2 and Sox9 protein expression. Western blotting analysis of EMD using anti-BMP-6 antibody revealed immunoreactive bands corresponding to about 14 kDa and 60 kDa. These results suggest that EMD stimulates osteogenesis- and chondrogenesis-related transcription factors, and these activities may be mediated, at least in part, by BMP-6 in EMD.

Animals↗

PubMed2006

Intravenous pamidronate treatment of infants with severe osteogenesis imperfecta.

OBJECTIVE: Children with the severe forms of osteogenesis imperfecta have in several studies been treated with intravenous pamidronate, but there are only few reports of the effect of early treatment. AIM: To evaluate the effect of treatment started in infancy. METHODS: In a prospective observational study, with a historic control group, intravenous disodium pamidronate (APD) was given as monthly infusions to 11 children with osteogenesis imperfecta aged 3-13 (median 3.6) months, who had severe osteogenesis imperfecta with congenital bowing of the femora and vertebral compression fractures. RESULTS: During treatment of children aged between 3 and 6 (median 4.5) years, dual-energy x ray absorptiometry measurements of the lumbar spine showed a gradual increase in bone density. Bone metabolism parameters in serum (alkaline phosphatase, osteocalcin, procollagen 1 carboxy-terminal peptide, collagen 1 teleopeptide) and in urine (deoxypyridinoline) indicated a decrease in bone turnover. An improvement of mobility was seen and at the latest recording, at the age of 3.3-6.5 (median 4.8) years, the children could all walk. Vertebral remodelling was seen, with increased vertebral height, and no child developed scoliosis, kyphosis or basilar impression. All children required femoral intramedullar rods for fractures, and five needed tibial rodding for extreme curvatures that prevented functional standing and walking. No adverse effects were seen on growth, fracture healing or blood chemistry. CONCLUSIONS: APD is an efficient symptomatic treatment for infants with severe osteogenesis imperfecta, but additional orthopaedic surgery is often needed. Early treatment may prevent scoliosis and basilar impression. Long-term follow-up is important.

Absorptiometry, Photon↗

PubMed2006

Treatment of craniosynostosis by distraction osteogenesis.

PURPOSE: Craniosynostosis patients treated with one-stage cranioplasty often have bleeding from the dura mater, fluid collection in the extradural space, and poor wound healing due to skin overstretching. To avoid these complications, we began using distraction osteogenesis. To determine the advantages and disadvantages of the procedure, we retrospectively compared distraction osteogenesis with conventional cranioplasty. PATIENTS AND RESULTS: We treated 24 patients with fronto-orbital advancement. Fifteen had one-stage cranioplasty; 9 received distraction osteogenesis. The one-stage operation patients averaged 25.5 months of age at surgery, 289 min of operating time, and 154 ml of intraoperative blood loss. For the distraction group, the average age was 16.6 months, average operating time was 196 min, and average blood loss was 76 ml. CONCLUSIONS: Compared with one-stage cranioplasty patients, distraction osteogenesis patients had significantly less intraoperative bleeding and shorter operating times. The disadvantages of distraction treatment were the need for multiple surgeries, prolonged hospitalization, wound infection, and dislocation of the distraction device.

Blood Loss, Surgical↗

PubMed2002

Similar COL1A1 expression in fibroblasts from some patients with clinical otosclerosis and those with type I osteogenesis imperfecta.

Because of the clinical and histopathologic similarities between otosclerosis and type I osteogenesis imperfecta, we examined COL1A1 messenger RNA (mRNA) expression in cultured fibroblasts from patients with clinical otosclerosis to determine whether abnormalities of expression of COL1A1 were present, as has been reported in type I osteogenesis imperfecta. Type I osteogenesis imperfecta has been found to result from mutations in the COL1A1 gene that result in null expression of the mutant allele. Patients with clinical otosclerosis were genotyped for the presence of an expressed 4 base-pair insertion polymorphism in the 3' region of the COL1A1 gene. Skin biopsies were performed, and cultured fibroblast cell lines were established from patients who were heterozygous for the polymorphism. Allelic expression was examined by reverse transcription-polymerase chain reaction and silver-stained polyacrylamide gel electrophoresis. Two of 9 patients with clinical otosclerosis demonstrated null or reduced expression of one COL1A1 allele. The differential expression of the two COL1A1 alleles in all subjects was also examined by a semiquantitative method using an ABI Prism 7700 Sequence Detection System (Taqman). We did this examination to determine whether milder abnormalities in COL1A1 expression might account for the development of otosclerosis in the 7 clinical cases that did not reveal evidence of null expression by the gel technique. Of the same 2 cases of otosclerosis that demonstrated evidence of null expression by gel electrophoresis, both were found to have significant differences in COL1A1 mRNA expression by the Taqman analysis. The remaining 7 cases revealed equal expression of the two COL1A1 alleles similar to that seen in controls. These results suggest that mutations in COL1A1 that are similar to those that occur in type I osteogenesis imperfecta may account for a small percentage of cases of otosclerosis, and that the majority of cases of clinical otosclerosis are related to other genetic abnormalities that have yet to be identified.

Alleles↗

PubMed2002

Osteoblastic response to the defective matrix in the osteogenesis imperfecta murine (oim) mouse.

This work examines the cellular pathophysiology associated with the weakened bone matrix found in a murine model of osteogenesis imperfecta murine (oim). Histomorphometric analysis of oim/oim bone showed significantly diminished bone mass, and the osteoblast and osteoclast histomorphometric parameters were increased in the oim/oim mice, compared with wild-type (+/+) mice. To assess osteoblast activity, a rat Col1a1 promoter linked to the chloramphenicol acetyltransferase reporter transgene was bred into the oim model. At 8 d and 1 month of age, no difference in transgene activity between oim and control mice was observed. However, at 3 months of age, chloramphenicol acetyl transferase activity was elevated in oim/oim;Tg/Tg, compared with +/+;Tg/Tg and oim/+;Tg/Tg. High levels of urinary pyridinoline crosslinks in the oim/oim;Tg/Tg mice were present at all ages, reflecting continuing high bone resorption. Our data portray a state of ineffective osteogenesis in which the mutant mouse never accumulates a normal quantity of bone matrix. However, it is only after the completion of the rapid growth phase that the high activity of the oim/oim osteoblast can compensate for the high rate of bone resorption. This relationship between bone formation and resorption may explain why the severity of osteogenesis imperfecta decreases after puberty is completed. The ability to quantify high bone turnover and advantages of using a transgene that reflects osteoblast lineage activity make this a useful model for studying interventions designed to improve the bone strength in osteogenesis imperfecta.

Amino Acids↗

PubMed1997

Limb salvage using distraction osteogenesis. A classification of the technique.

We report the results of distraction osteogenesis (callotasis) for the reconstruction of extensive defects after the excision of skeletal tumours in the limbs. Bone transport was performed in ten patients (five osteosarcomas and five giant-cell tumours), shortening-distraction in three (two osteosarcomas and one Ewing's sarcoma), and distraction osteogenesis combined with an intramedullary nail to reduce the time of external fixation in six (three osteosarcomas, two chondrosarcomas, and one malignant fibrous histiocytoma). The mean length of the defects after excision of the lesion was 8.4 cm. The mean external fixation index was 39.5 days/cm for the group treated by bone transport, 34.1 days/cm for the shortening-distraction group, and 24.0 days/cm for the group treated by distraction and an intramedullary nail. Functional evaluation gave excellent results in 12 patients, good in five and fair in two. There were ten complications in 19 patients, all of which were successfully treated. We also classified reconstruction using distraction osteogenesis into five types based on the location of the defects after resection of the tumour: type 1, diaphyseal; type 2, metaphyseal; type 3, epiphyseal; type 4, subarticular reconstruction; and type 5, arthrodesis. Our results suggest that reconstruction using distraction osteogenesis provides bone which will develop sufficient biomechanical strength and durability. It is beneficial in patients with an expectation of long-term survival and in growing children.

Adolescent↗

PubMed2003

Zoledronic acid prevents osteopenia and increases bone strength in a rabbit model of distraction osteogenesis.

UNLABELLED: Prolonged healing times and stress-shielding osteopenia remain problematic in distraction osteogenesis. In this study of 30 rabbits, zoledronic acid increased regenerate volume, mineralization, and tibial strength and prevented osteopenia over a 6-week period. Translation to the clinical setting, if safe, could improve outcomes in distraction osteogenesis in children. INTRODUCTION: Because the external fixators for limb lengthening and reconstruction are designed to control the positions of bone fragments accurately, they also produce stress-shielding effects on the forming regenerate and surrounding bone. Osteopenia, leading to refracture and limitations on rehabilitation, are common consequences, potentially increasing morbidity and detracting from final clinical outcome. MATERIALS AND METHODS: We examined the effect of zoledronic acid on distraction osteogenesis in 42 immature male NZW rabbits. The model chosen results in reliable regenerate formation and stress-shielding osteopenia. Fourteen animals received either Saline, zoledronic acid 0.1 mg/kg at surgery (ZOL), or another dose 2 weeks postoperatively (Redosed ZOL). Rabbits underwent DXA for bone mineral content and bone mineral density in regenerate and surrounding segments of operated and contralateral tibias. After death at 6 weeks, 30 pairs of tibias underwent quantitative computerized tomography (QCT) and four-point bend testing, and 12 were examined by histomorphometry. The study was powered at 0.8 to show differences of 1.3 SDs for mineral and mechanical parameters. RESULTS: Osteopenia observed in tibias of the Saline group was absent in ZOL and Redosed ZOL tibias, the latter exhibiting higher bone mineral density and bone mineral content over contralateral regions (p < 0.01). Regenerate bone mineral content was higher in ZOL and Redosed ZOL versus Saline groups at 4 and 6 weeks (p < 0.01). Cross-sectional area was 49% and 59% greater at 6 weeks in ZOL and Redosed ZOL regenerates compared with the Saline group (p < 0.01). ZOL and Redosed ZOL tibias were 29% and 89% stronger by four-point bending than the Saline group (p < 0.01). Histomorphometry in the regenerate of ZOL and Redosed ZOL groups revealed higher trabecular bone volume and trabecular number compared with the Saline group (p < 0.001). CONCLUSIONS: Zoledronic acid administration led to significantly greater bone area, mineral content, strength, and trabecular number with reduced stress-shielding osteopenia in this model of distraction osteogenesis. These data suggest that intraoperative and postoperative zoledronic acid administration could improve outcomes in children undergoing limb lengthening.

Animals↗

PubMed2005

A comparison of oral and intravenous bisphosphonate therapy for children with osteogenesis imperfecta.

Bone mineral density and fracture rates in children with osteogenesis imperfecta improve with intravenous bisphosphonates. The efficacy of oral bisphosphonates has not been established. This report is an analysis of an open-label, prospective, randomized clinical trial of oral compared to intravenous bisphosphonate medications in children with osteogenesis imperfecta. Children were stratified according to bone age, pubertal stage, and type of osteogenesis imperfecta and then randomized to receive intravenous pamidronate, 3 mg/kg over 3 days every 4 months, or oral alendronate 1 mg/kg, from a minimum of 10 mg to a maximum of 20 mg daily. The primary efficacy outcome was change in bone mineral density. Secondary outcomes included change in biomarkers of bone turnover, fracture incidence, and growth rate. Ten children were randomized (6 oral and 4 intravenous). Two other children were assigned to intravenous treatment due to chronic abdominal pain. In each group, three patients had type III/IV osteogenesis imperfecta, while three had type I. All 12 children completed 8 months of therapy; nine completed 12 months. Bone mineral density increased in both oral and intravenous groups equally and beyond that expected with normal growth. All children had a decrease in biochemical markers of bone turnover. Linear growth showed a moderate increase above that for age. There was a non-significant decrease in fracture incidence in both groups.

Administration, Oral↗

PubMed2000

Chest compressions in an infant with osteogenesis imperfecta type II: No new rib fractures.

The case report of a newborn female with osteogenesis imperfecta type II who underwent cardiopulmonary resuscitation (CPR) with manual chest compressions for several minutes is presented. Chest radiographs taken before and after the chest compressions were administered were reviewed by several radiologists from 3 different hospitals and demonstrated no new radiographically visible rib fractures. Collagen analysis, the patient's clinical appearance, and clinical course, as well as a consultant's opinion aided in confirmation of the diagnosis of osteogenesis imperfecta type II. A review of 4 previous studies concerning rib fractures and CPR is included. This unique case supports previous articles that have concluded that rib fractures rarely, if ever, result from CPR in pediatrics, even in children with a lethal underlying bone disease, such as osteogenesis imperfecta type II. cardiopulmonary resuscitation, chest compressions, osteogenesis imperfecta, rib fractures, bone disease.

Biomechanical Phenomena↗

PubMed2004

Mandibular widening by distraction osteogenesis in the treatment of a constricted mandible and telescopic bite.

OBJECTIVE: Documentation of the application of mandibular widening by distraction osteogenesis and orthodontics. PATIENTS: Three patients with telescopic bite resulting from an extremely constricted mandible related to hypoglossia-hypodactyly syndrome. INTERVENTION: Mandibular widening by distraction osteogenesis using an extraoral device and subsequent orthodontic treatment. RESULTS: The extremely constricted mandible and telescopic bite were dramatically improved by mandibular widening using distraction osteogenesis and subsequent orthodontics. Two of the three patients had transient complications; one reported temporomandibular joint pain and the other showed evidence of periodontal damage. CONCLUSION: Mandibular widening by distraction osteogenesis is an effective technique for the treatment of telescopic bite resulting from an extremely constricted mandible.

Child↗

PubMed1994

Experimental and clinical experience with distraction osteogenesis.

G.A. Ilizarov's clinical insights and experimental biology have developed into what is known today as distraction osteogenesis. Initially used for the treatment of fractures and nonunions, his methods have proven successful for limb lengthening and bone transportation. A brief history of the development of distraction osteogenesis, definition of terms, methods, and monitoring techniques are described. The primary mechanism, intramembranous ossification, is direct bone formation in uniform gradients of mineralization from a central fibrous interzone. New bone is produced from the local host surfaces and quickly remodels to the equivalent macro and microstructure. Blood supply at the focus of distraction, dependency on a latency period, different rates, and rhythms of distraction are all tested and discussed. Data from experimental tibial lengthening in more than 125 animals (dogs, rabbits, and rats) show that distraction osteogenesis provides unlimited new bone formation that remodels at daily rates ranging from 200 to 400 microns. In over 100 clinical cases, patients ranging in age from 18 months to 49 years have regenerated bone at an average rate of 213 microns in adults and 385 microns in children. Approximately 10% of these cases required supplemental bone grafts. The article proposes that distraction osteogenesis might be successfully applied in craniofacial surgery.

Adolescent↗

PubMed2002

Ethical and scientific decision making in distraction osteogenesis.

OBJECTIVE: To propose a prospective registry for distraction osteogenesis research. RESULTS: The search strategy identified 82 reports, of which 11 were review articles, 17 were animal studies, 5 were theoretical models, and 49 were case reports/series. CONCLUSIONS: So far, the literature concerning distraction osteogenesis does not allow reliable choices to be made on the most appropriate form of distraction osteogenesis in different clinical decisions or whether it is superior to osteotomy or nontreatment. A prospective registry is proposed to hasten critical appraisal of distraction osteogenesis.

Bias↗

PubMed2003

Strain induced osteogenesis of the craniofacial suture upon controlled delivery of low-frequency cyclic forces.

Static forces have been used for more than a century to modulate osteogenesis of craniofacial sutures in not only laboratory research, but also clinical practice. Whether cyclic forces more effectively stimulate sutural osteogenesis than static forces is unknown. Here, the premaxillomaxillary sutures of growing rabbits received in vivo exogenous static forces with peak magnitude of 2 Newtons, or cyclic forces also at 2 Newtons but with frequencies of 0.2 Hz and 1 Hz. The static force and two cyclic forces did not evoke significant differences in the peak magnitude of static bone strain (506 microstrain 182; mean S.D.), 0.2-Hz cyclic strain (436 microstrain 191) or 1-Hz cyclic strain (461 microstrain 229). However, cyclic forces at 0.2 Hz delivered to the premaxillomaxillary suture for 10 min/d over 12 days (120 cyclic per day) induced significantly more craniofacial growth (p< 0.01), marked sutural separation, and islands of newly formed bone, in comparison with both sham controls and static force of matching peak magnitude. The bone strain threshold of approximately 500 microstrain for inducing sutural osteogenesis is lower than the minimum effective strain capable of inducing bone apposition in long bones. These data demonstrate, for the first time, that application of brief doses of cyclic forces induces sutural osteogenesis more effectively than static forces with matching peak magnitude.

Animals↗

PubMed1994

Variable prenatal appearance of osteogenesis imperfecta.

Osteogenesis imperfecta is a heterogeneous group of disorders of type I collagen with both lethal and nonlethal forms. Prenatal sonographic findings in affected fetuses are variable and depend on the severity of the disease. Six cases of osteogenesis imperfecta in which prenatal sonography had been performed were reviewed. Two cases of lethal type II osteogenesis imperfecta revealed short femurs at 16 to 17 weeks' gestation with development of bowing and fractures by 19 weeks' gestation. Four fetuses with the nonlethal type III or IV had femoral bowing with or without shortening in the late second or third trimester with grossly normal mineralization. Fractures in this latter group did not develop until 1 to 12 months after delivery. Understanding the progressive nature and variability of osteogenesis imperfecta is crucial in the prenatal diagnosis and management of this disease.

Adolescent↗

PubMed1994

Osteogenesis imperfecta and campomelic dysplasia: difficulties in prenatal diagnosis.

The classic features of osteogenesis imperfecta and campomelic dysplasia typically are so specific that these two forms of skeletal dysplasia are among the few that can be diagnosed in the second trimester of pregnancy. We report a series of seven examples of osteogenesis imperfecta and one example of campomelic dwarfism to illustrate the difficulty of differentiating these two disorders in utero. The prenatal sonographic findings in three of the osteogenesis imperfecta cases mimicked campomelic dwarfism, whereas the case of campomelic dwarfism was antenatally diagnosed as osteogenesis imperfecta.

Adolescent↗

PubMed1999

Evaluation of the treatment for micromandibular deformity by distraction osteogenesis with submerged intraoral device.

OBJECTIVE: To evaluate the results of distraction osteogenesis for correcting the micromandible with an intraoral device. MATERIALS AND METHODS: Eight cases of micromandible resulting from temporomandibular joint (TMJ) ankylosis and first and second branchial arch syndrome received treatment by distraction osteogenesis. Subjects included 5 females and 3 males, ranging in age from 10 to 43 years (avg 20.25). Five micromandibles were associated with obvious mandibular asymmetry. An osteotomy was performed on bilateral mandibular bodies by intraoral approach. Elongation was started on the 5th or 6th day postoperation at a rate of 0.8 to 1 mm per day. The distractors were removed after a consolidation period from 71 to 139 days. RESULTS: The distractions were fulfilled with a range of 8.5 to 24.4 mm. Pain appeared in osteotomy region when the 1 mm distraction was carried out once a day and disappeared when it was divided into twice a day. Mandibular lengthening was successful. Micromandible and facial asymmetries were corrected satisfactorily. The follow-up period ranged from a minimum of 1 month to a maximum of 10 months. There was no recurrence. One case of soft tissue infection and one case of lower lip numbness were reported. There were no cases of infection or other disturbance of wound healing or pseudarthrosis. CONCLUSIONS: Because of its advantage in little injury, avoiding bone grafting, and few complications, intraoral distraction osteogenesis is a valuable approach to correct mandibular agenesis. However, in the management of mandibular deformities, distraction osteogenesis achieves better results when combined with other orthognathic operations. How to control the direction of distraction is a problem that needs further study.

Adolescent↗

PubMed2000

Anaemia, osteogenesis imperfecta and valve diseases. The preoperative treatment with epoetin-alpha to increase haematocrit and haemoglobin levels in patients with high risk of perioperative bleeding.

The case of a patient with Osteogenesis imperfecta is reported who underwent surgery for mitral valve replacement. Osteogenesis imperfecta is a hereditary disease of the connective tissue, associated with bone fragility, bluish colouring of the sclerae, loss of hearing and dental anomalies. Osteogenesis imperfecta is included in a group of hereditary pathologies with Ehlers-Danlos syndrome, Hurler syndrome, pseudoxanthoma elasticum and Marfan syndrome. In the literature there are few cases of patients with osteogenesis imperfecta operated for mitral valve disease secondary to such disease. The patient also has a positive family history and a severe anaemia: this was treated with epoetin-alpha and ferrous sulphate during the three weeks before surgery. The response to epoietin treatment was good while clinical outcome was poor due to rupture of the posterior ventricular wall in the 12th postoperative hour. The use of epoietin-alpha is discussed.

Anemia↗