Cranial Distraction Osteogenesis Research Articles

Impact of Anterior and Posterior Vault Distraction Osteogenesis (A-PVDO) and 3D-Printed Positioning/Shaping Templates in Apert Syndrome: A Case Series Study.

This study seeks to examine the impact of anterior and posterior vault distraction osteogenesis (A-PVDO) in conjunction with 3D-printed positioning and shaping templates for the management of Apert syndrome. From January 2018 to February 2022, a retrospective analysis was conducted on 6 cases of Apert syndrome employing fronto-orbital 3D-printed positioning and molding templates. The cranium underwent surgical modification in accordance with the template's configuration and was affixed with absorbable plates. Subsequently, distraction devices were applied, encompassing both anterior and posterior craniotomies. The evaluation encompassed clinical outcomes, complications (including cerebrospinal fluid leakage and infection), safety, and the feasibility of the distraction osteogenesis procedure. Six patients diagnosed with Apert syndrome underwent treatment involving the integration of fronto-orbital 3D-printed positioning and shaping templates in conjunction with anterior and posterior cranial distraction osteoplasty. Follow-up durations ranged from 18 to 32 months (average: 22mo). No instances of fronto-orbital retraction, cerebrospinal fluid leakage, or intracranial infection were noted during the follow-up period. The sole reported complication entailed an infection at the extension rod site in 1 case. All patients conveyed satisfaction with the treatment outcomes. The application of 3D-printed positioning and shaping templates in tandem with anterior and posterior cranial distraction osteogenesis demonstrates efficacy in addressing Apert syndrome. Notably, significant enhancements in head shape and orbit were observed, and the incidence of postoperative complications such as cerebrospinal fluid leakage and infection remained minimal. Moreover, long-term follow-up affirmed stability.

Cranial distraction osteogenesis for craniosynostosis associated with osteopetrosis: A case report

Background: Osteopetrosis is a rare disease characterized by systemic osteosclerosis and hematopoietic disturbances. Childhood-onset cases are often accompanied by hydrocephalus and craniosynostosis; however, there have been no established treatments. We performed cranial distraction in a child with osteopetrosis who presented with craniosynostosis and intracranial hypertension. Case Description: The patient was a 4-year-1-month-old boy. His pregnancy and birth were normal, but at 4 months of age, he was diagnosed with osteopetrosis based on generalized osteosclerosis and family history. A computed tomography scan of the head revealed early sagittal suture fusion and ventricular enlargement. A ventriculoperitoneal shunt was placed for intracranial hypertension; however, slit ventricle syndrome ensued and pansynostosis developed. To improve uncontrolled high intracranial pressure, cranial distraction was performed for intracranial volume expansion. No perioperative hemorrhagic or infectious complications were observed. After the start of distraction, the intracranial pressure gradually decreased, and clinical findings such as disturbance of consciousness and bradycardia disappeared. Bone regeneration in the defect site was good, and the extension device was removed 6 months after the operation. Conclusion: For osteopetrosis with poorly controlled intracranial hypertension, cranial distraction was considered to be an effective treatment.

Effectiveness of immediate fixation after cranial distraction osteogenesis

Distraction osteogenesis (DO) is an established safe and effective treatment of craniosynostosis (CS) deformities. However, conventional methods demonstrate some complications, such as long-term maintenance of the distractor and relapse after distractor removal. Only a few studies have overcome these limitations. Therefore, we hypothesized that placing a resorbable plate after removing the distractor will provide additional stability to the newly formed bone, shortening the consolidation period and minimizing relapse. Twenty-six children diagnosed with CS who underwent DO between 2000 and 2019 were retrospectively analyzed. A resorbable plate was fixed across the regenerated bone when distractors were removed. The consolidation period and complication rate were obtained from medical records, and both two- and three-dimensional analyses were performed to obtain relapse rate and brain volume changes using three-dimensional computed tomography. Among 26 patients, the average consolidation period was 90.75±23.75 days in the conventional group (n=11) and 22.77±8.69 days in the intervention group (n=15). In the two-dimensional analysis, the relapse rate was lower in the intervention group. Moreover, in the three-dimensional analysis, the relapse rate of unilateral CS between the affected and unaffected sides was lower in the intervention group. The conventional group had more complications (skin defect and distractor exposure). Resorbable plate placement after distractor removal helps shorten the consolidation period and prevent relapse in pediatric patients with cranial DO. It reduces complications and shows stable results in terms of cranial morphology and symmetric brain growth in patients with CS.

Multidirectional Cranial Distraction Osteogenesis for Craniosynostosis

In this article we explain in detail the Multidirectional Cranial Distraction Osteogenesis(MCDO)procedure for treating craniosynostosis, in which the multiple cranial bone flaps are pulled up in different directions by the external distractor with the helmet-type frame. MCDO has several advantages over conventional procedures such as unilateral cranial distraction osteogenesis(UCDO). First, the design of the osteotomy in MCDO is flexible depending on the deformities. Each bone flap can be moved to the desired direction in MCDO, while being moved only in one direction along the axis of the distractor in UCDO. This enables remodeling of the deformed cranium to the desired shape. Second, compared with UCDO, the distance between each bone flap after distraction is narrower in MCDO, allowing earlier bone formation and fusion, and thus a shorter consolidation period. Third, the expansion efficacy of MCDO is greater than that of UCDO. The maximum defects of MCDO were the large amount of osteotomy and the resulting heavy bleeding. Therefore, we have recently attempted to improve the design and procedure of osteotomy. The MCDO method allows all phenotypes of skull deformity to be reshaped by distraction osteogenesis.

Evaluation and Management of Nonsyndromic Craniosynostosis.

Nonsyndromic craniosynostosis (NSC) is more common than syndromic craniosynostosis and predominantly involves single suture. It affects sagittal, coronal, metopic, and lambdoid sutures in the decreasing order of frequency. A surgery for NSC is generally recommended to avoid potential neurodevelopmental delays and sequelae of raised intracranial pressure. Open calvarial vault reconstruction, strip craniectomy with/without the use of a postoperative molding helmet, strip craniectomy with spring implantations, endoscopic suture release, and cranial distraction osteogenesis are various surgical options used for NSC cases. The ideal age for intervention is 6-12 months for open procedures and 3-4 months for endoscopic approaches. The management is directed toward minimizing operative trauma and improving the neurocognitive outcome. The role of nonsurgical intervention by the use of genetic manipulation is still not a reality because of the nature of disease and time of presentation.

A Single-Center Retrospective Review of Perioperative Complications and Reoperation Rates Between Open Cranial Vault Remodeling and Distraction Osteogenesis for Unilateral Coronal Craniosynostosis.

Unilateral coronal craniosynostosis (UCS) is the third most prevalent form of craniosynostosis. Traditional treatment of UCS has been achieved with fronto-orbital advancement and cranial vault remodeling (FOAR), but utilization of cranial distraction osteogenesis (DO) techniques has increased. This study aims to compare perioperative complications and reoperation trends in FOAR versus DO techniques at a single institution. An Institutional Review Board-approved retrospective review was performed from January 1999 to November 2018 at a single institution. Patients were those that have undergone FOAR or DO with an anterior rotational flap technique as previously described. Indications for secondary procedures included: contour deformities, relapse, surgical site infection, and persistent cranial defects. Eighty-one patients with UCS were identified, 64 patients underwent FOAR and 17 patients underwent DO. When perioperative characteristics were compared, patients who underwent DO were younger in age, however, there was no significant difference in transfusion requirement or length of stay between patient cohorts. Surgery time was increased in DO patients. When perioperative complications were compared, more intraoperative dural tears were observed in the FOAR cohort. When unplanned reoperation rates were compared, patients who had undergone FOAR had a statistically significant higher reoperation rates at 5 years of follow up. When including routine distractor removal as a reoperation, reoperative rate was increased in the DO cohort. No difference in reoperation rates was noted at 5 years following index operation. The safety profile of DO is similar to that of traditional FOAR techniques for treatment of UCS. Longer-term follow-up studies are needed to elucidate whether outcomes are durable, but the unplanned reoperation rate in DO is less than that of FOAR at 5 years and presents several advantages that warrants its use in patients with UCS.

Morphological improvement after multi-directional cranial distraction osteogenesis procedure for syndromic craniosynostosis.

The multidirectional cranial distraction osteogenesis (MCDO) procedure, which uses an external distraction device, enables tailor-made distraction in an arbitrary direction, eliminating the disadvantage of unidirectional distraction with an internal distraction device. Multiple-suture synostosis cases for syndromic craniosynostosis patients are better indicated for this procedure. Here the authors describe seven cases in which the MCDO procedure was used to treat syndromic craniosynostosis. In each case, the MCDO procedure and postoperative distraction, with reference to midsagittal vector analysis of normal morphology in Japanese children, resulted in morphological improvement.The video can be found here: https://vimeo.com/519006555

学童期まで診断が遅れた頭蓋縫合早期癒合症に対してMCDO法を適用した 1 例

学童期まで診断が遅れた頭蓋縫合早期癒合症に対してMCDO法を適用した 1 例

Thirty Years Later: What Has Craniofacial Distraction Osteogenesis Surgery Replaced?

After studying this article and viewing the video, the participant should be able to: 1. Compare the relative stability and neurosensory changes following mandible distraction osteogenesis with those after traditional advancement and fixation. 2. Describe the condylar changes that can occur after mandible distraction osteogenesis and list three ways to mitigate these changes. 3. Propose clinical situations where segmental or rotational movements of the midface may allow improved outcomes compared to en bloc linear distraction advancement. 4. Summarize the advantages and risks associated with anterior and posterior cranial distraction osteogenesis compared to traditional one-stage expansion. Over the past 30 years, distraction forces have been applied to the spectrum of craniofacial osteotomies. It is now time to assess critically the current understanding of distraction in craniofacial surgery, identifying both traditional procedures it has replaced and those it has not. This article provides a review of comparative studies and expert opinion on the current state of craniofacial distraction compared with traditional operations. Through this critical evaluation, the reader will be able to identify when distraction techniques are appropriate, when traditional techniques are more favorable, and what the future of distraction osteogenesis is.

Utilization of a Simple Surgical Guide for Multidirectional Cranial Distraction Osteogenesis in Craniosynostosis.

Background:Multidirectional cranial distraction osteogenesis (MCDO) can achieve a desired shape for deformities of the cranium. In the past, visual estimation was used to reflect on the actual skull, but it was time-consuming and inaccurate. Here we demonstrate an effective osteotomy navigation method using surgical guides made from a dental impression silicone.Methods:Seven patients who underwent MCDO between August 2013 and September 2016 were included in the study. Five cases involved utilization of the surgical guide for osteotomy. Three-dimensional (3D) printed cranium models were made using 3D computed tomography (3DCT) imaging data and dental impression silicone sheets were molded using the printed cranium models. These surgical guides were sterilized and used for intraoperative osteotomy design. Vertical distance between nasion/porion and osteotomy lines were calculated using 3D printed cranial models and postoperative 3DCT images to assess reproducibility.Results:The average surgical time/design time was 535/37.0 minutes for the nonsurgical guide group and 486.8/11.8 minutes for the surgical guide group (SG).Treatment using the surgical guide was significantly shorter in terms of operative time and time required for design. For the vertical distance comparison, the average distance was 5.7mm (SD = 0.3) in the non-SG and 2.5mm (SD = 0.44) in the SG, and SG was more accurate.Conclusions:Shorter operative times and higher reproducibility rates could be achieved by using the proposed surgical guide, which is accurate, low-cost, and easily accessible.

S4A-10 SESSION 4A: GENERALITIES IN CRANIOSYNOSTOSIS – PART I EFFECTS OF DIFFERENT DIRECTIONS OF ANTERIOR CRANIAL DISTRACTION OSTEOGENESIS ON ANTERIOR CRANIAL VOLUME

Introduction: Anterior cranial distraction osteogenesis has been proposed as an effective technique for expending the anterior cranial volume to avoid intracranial hypertension. Mostly studies have focused on volumetric changes in different surgical approaches such as fronto-orbital advancement and posterior cranial vault distraction osteogenes. However the direction of distraction can also affect the outcome. So the aim of this study was to analyze the effects of three different directions of anterior cranial distraction osteogenesis, these are 15 degrees forward and downward, horizontal distraction, 15 degrees forward and upward. Methods: This was a retrospective chart review performed between 2015 to 2018 at a the department of plastic surgery in the children`s hospital of Nanjing medical university. 24 patients were included in this study. We divided the patients into three groups according to the three directions of anterior cranial distraction osteogenesis. Each group has ten (10) patients. All the patients were performed the CT scan preoperatively and postoperatively. The CT databases were processed by DICOM files into MIMICS software, which were automatically calculated into anterior cranial volume. Then we calculated the percentage increase in anterior cranial volume of each group. Results: The mean increase volume was 64±17ml for 15 degrees forward and downward group 44±15 mlfor horizontal distraction group and 38±12ml for 15 degrees forward and upward group. The mean difference in anterior cranial volume between the 15 degrees forward and downward group and horizontal distraction group and 15 degrees forward and upward group was significantly increased (P <0.05) Conclusion: In this retrospective study, 15 degrees forward and downward group in anterior cranial vault distraction osteogenesis provided statistically greater anterior cranial volume expansion than the other two angles.

Multidirectional Cranial Distraction Osteogenesis with Simplified Modifications for Treating Sagittal Synostosis.

Multidirectional cranial distraction osteogenesis (MCDO) is a procedure of ours developed earlier for treating craniosynostosis. However, the numerous bone flaps led to prolonged operative time and occasional bone detachment from dura. We have since simplified the osteotomy design. In treating sagittal synostosis, required bone flaps have been reduced to 11 (from ~20). In a 2-year period (2014-2015), 5 boys with sagittal synostosis underwent MCDO using our simplified and fixed-form osteotomy. Mean age at surgery was 9.4 months (range, 8-11 months). Pre- and postoperative cranial morphology was assessed by cephalic index and by mid-sagittal vector analysis. Improved cranial shape was confirmed by 3-dimensional CT scans and by mid-sagittal vector index. Mean preoperative cephalic index (68.7) progressively increased to means of 78.5 immediately after distraction device removal, 75.2 at postoperative month 6, and 75.1 at 1 year postoperatively. There were no major complications, although transient cerebrospinal fluid leakage and loosening of anchor pins occurred in 1 patient. Simplified MCDO has a number of advantages over conventional distraction procedures such as discretionary reshaping/expansion of cranium and predictable osteogenesis and is a valid treatment option for patients with sagittal synostosis.

Distraction Osteogenesis Update: Introduction of Multidirectional Cranial Distraction Osteogenesis.

In this review, we discuss in detail our current procedure for treating craniosynostosis using multidirectional cranial distraction osteogenesis (MCDO). The MCDO method allows all phenotypes of skull deformity to be reshaped by distraction osteogenesis, except in patients who are 5 months of age or younger and patients with posterior cranial vault problems. We report the results of clinical data of 36 children with craniosynostosis who underwent MCDO between 2005 and 2014 in our institute. This method has the following benefits, such as a high flexibility of reshaping, shorter treatment period and less invasive secondary intervention. We also discuss the other distraction osteogenesis techniques that are used to treat craniosynostosis and compare them with MCDO. The preferred procedure for correction of craniosynostosis may depend on the patient's age, the extent of deformity, and the extent of correction achievable by surgery. We can arrange the combinations of various methods according to the advantage and disadvantage of each technique.

Cranial distraction osteogenesis: a proposal of minimal consolidation period.

Cranial distraction osteogenesis (DO) has many advantages for correcting skull deformities: Thus, DO is extensively used for the treatment of skull deformities. However, diverse, unexpected complications are associated with this procedure. In this study, we present the surgical outcomes and complications of DO. Moreover, we propose a modified protocol for DO to reduce complications. This is a retrospective study on managed patients that underwent DO between March 2008 and May 2013. Their clinical courses were reviewed. Distraction protocols were individually inspected, and the final surgical outcomes, including complications, were evaluated. During the study period, a total of ten patients (seven boys and three girls) were treated at our institute. The median distraction period was 20.5days (ranging from 17 to 50days). The range of total distraction length was 19-22mm. The median consolidation period was 96days (ranging from 0 to 343days). All patients achieved the goals of distraction. At follow-up evaluations, all patients, except one, showed good surgical outcomes in both head shape and neurologic symptoms. There were six patients with wound complications during the treatment period. Among them, the distractors were removed early in three patients. Interestingly, even these three patients, without a sufficient consolidation period, showed good outcomes. Although DO has many merits for correcting skull deformities, it frequently causes severe wound complications. To reduce these complications, we propose a modified protocol with a minimal or even no consolidation period.

Intracerebral foreign body granuloma caused by a resorbable plate with passive intraosseous translocation after cranioplasty

Numerous reports have demonstrated the usefulness of bioresorbable materials, but few have described severe complications caused by delayed degradation. The authors present the case of an intracranial foreign body granuloma caused by plates made of unsintered hydroxyapatite (uHA) particles and poly-l-lactide (PLLA; Super Fixsorb MX, Takiron) after cranioplasty. This 1-month-old boy presented to the authors' department with Pfeiffer syndrome. He had multiple-suture synostosis causing turribrachycephaly, Chiari malformation Type 1, and obstructive sleep apnea syndrome. At 6 months old, the child was treated with multidirectional cranial distraction osteogenesis. The uHA-PLLA plates were applied as base stones to reinforce the pins. After 16 days of distraction and 3 weeks of consolidation, the pins were removed. Seventeen months postoperatively, the plate on the right temporal bone showed passive intraosseous translocation (PIT), and by 2 years postoperatively, the plate was completely left behind in the cerebrum. At 3.5 years postoperatively, MRI disclosed a contrast-enhanced mass with surrounding brain edema at the site of the plate. The lesion was resected. The clinical history and histological specimens led to a diagnosis of foreign body granuloma surrounding the nonabsorbed resorbable plate in the dura mater. Resorbable plates are clearly useful resources in cases in which delayed absorption will not prove problematic, but careful application and follow-up is required when dealing with the growing skull given the possibility of intracranial displacement after PIT.

Cranial distraction osteogenesis for syndromic craniosynostosis: Long-term follow-up and effect on postoperative cranial growth

Although cranial distraction osteogenesis (CDO) is beneficial, few studies have reported on detailed operative procedures and postoperative cranial growth. Herein, we demonstrated the objective effectiveness of CDO in younger infants. The study included infants who underwent primary cranial distraction for craniosynostosis. Infants who had hydrocephalus were excluded and those who underwent additional osteotomy surgeries were analysed before the subsequent procedures. The infants' computed tomography data were analysed using Mimics(®) software (Materialise, Leuven, Belgium) to calculate the cranial volumes and compare them with the Abbott curve for a normal population. We defined cranial growth gap as the difference between the subject data and normal infant data to demonstrate the perioperative effects on cranial growth. CDO was performed in 10 infants. The mean infant age at the time of surgery was 6.4 months (range, 24--61 months) and the mean duration of postoperative follow-up was 38.9 months (range, 24--61 months). Five infants with Crouzon syndrome and five with Apert's syndrome were included. All infants showed postoperative cranial growth, but cranial growth gap showed postoperative declines for a certain period, indicating cranial growth suppression immediately following expansion. At the last follow-up, all cases were within ±2 standard deviation (SD) compared with the normal population. We derived a formula to predict the CDO target volume using the declining cranial growth gap curve. CDO was applicable and suitable for younger infants requiring aggressive cranial expansion.

Suitable Indication for the Application of Distraction Osteogenesis

In recent years, cranial distraction osteogenesis (DOG) for craniosynostosis has often been performed in Japan. Since 1998, DOG has been conducted in more than 80 patients with craniosynostosis in our institution. It is now important to define the appropriate indications for the application of DOG for craniosynostosis. From the point of view of our more than 10 years of experience with the application of cranial DOG, posterior enlargement is one of the most appropriate applications. Bilateral posterior expansion is conducted for hypertension because of bilateral lambdoidal synostosis, whereas unilateral posterior expansion is conducted for asymmetry because of unilateral posterior plagiocephaly. Posterior expansion by gradual distraction was conducted for 12 cases. Eight cases of syndromic craniosynostosis and 4 of simple unilateral lambdoid synostosis were included. Clinical and radiological improvement was observed in all cases. Postdistraction computed tomography demonstrated a decrease in digital printing within a few months in all cases. Distraction osteogenesis is a useful procedure for posterior enlargement by gradual expansion and guarantees postdistraction rigidity without collapse in a supine position in bilateral and unilateral lambdoid synostosis.

Evidence for long-term effectiveness of cranial distraction osteogenesis for syndromic craniosynostosis

Evidence for long-term effectiveness of cranial distraction osteogenesis for syndromic craniosynostosis

Multidirectional Cranial Distraction Osteogenesis for the Treatment of Craniosynostosis

Although many operative methods for the treatment of craniosynostosis exist, whether any difference in outcome could be associated with either the child's age at surgery or the extent of the operation remains to be elucidated. The authors have developed a method of distraction osteogenesis for craniosynostosis that uses a new multidirectional cranial distraction osteogenesis system. From 2003 to 2008, 26 selected patients with syndromic and nonsyndromic craniosynostosis were treated with this method. The ages of the patients ranged from 9 to 139 months (median, 27 months). The follow-up period ranged from 13 to 81 months (median, 49 months). The postoperative course was uneventful in all cases. The mean blood transfusion was 20.9 ml/kg. The mean postoperative hospital stay was 12 days. Sixteen of 49 (32.7 percent) anchor pins used in the first eight patients loosened because of bone absorption during the consolidation period. After the authors altered the screw shape, loosening occurred in seven of 81 pins (8.6 percent) in the last 18 patients. Loosening occurred in 28 of 204 traction pins (13.7 percent) among all patients. The phase of activation ranged from 8 to 14 days (mean, 10.5 days) and the consolidation period ranged from 21 to 42 days (mean, 29 days). Overall, distraction osteogenesis with the multidirectional cranial distraction osteogenesis method is safe and effective. The authors conclude that the multidirectional cranial distraction osteogenesis method constitutes an excellent alternative for all phenotypes of syndromic or nonsyndromic craniosynostosis.

The Bone Remodeling and Cellular Responses to Cranial Suture Distraction Osteogenesis in Growing Goats

The purpose of the present study was to observe the response and changes of cranial suture to the distraction forces in growing goats and to examine the expression patterns of TGF-β and BMP during suture distraction.Twenty growing goats were divided into three groups: control (n=4), experimental (n=12), and sham (n=4). A pure titanium distractor was placed in the coronal suture in both the sham and experimental groups. After healing, the distractor was activated for distraction of the coronal suture at a rate of 0.5 mm/day for 8 days in the experimental group. Three animals were killed respectively, at 0，2，4 and 8 weeks after completion of suture distraction. No force was applied in the sham group. X- Ray examination was taken and the coronal suture samples were harvested and processed for histological analysis and scanning electron microscopic analysis and immunohistochemistry of TGF-β and BMP. The coronal sutures of experimental group were separated successfully. Signs of intramembranous bone formation and remodeling were found in the distracted suture，and the sutural structure almost return to its normal state at 8 weeks after end of distraction. At 0 and 2 weeks after completion of suture distraction, the collagen fiber bundles were strengthened and aligned in the direction of the distracted forces. Strong expression of BMP and TGF-β were detected in the fibroblast-like cells and the active osteoblasts. At 4 weeks after suture distraction, signs of intramembranous ossification were found in the edge areas of the distracted suture, and the positive staining of BMP and TGF-β was still noted in the osteoblasts around the newly formed bone trabeculae. This study suggests that cranial suture expansion can be achieved in growing animal by distraction osteogenesis. Mechanical strain resulted from distractor can induce the adaptive remodeling in the cranial suture of growing goats. It also suggests BMP and TGF-β may play very important roles in the process of bone formation and remodeling during suture distraction osteogenesis.