Journal of Cranio-Maxillo-Facial Surgery 39 (2011) 412e419

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Journal of Cranio-Maxillo-Facial Surgery

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Distraction osteogenesis as followed by CT scan in Pierre Robin sequenceq,qq

Ahmed Mahrous Mohamed a, *, Awad Al Bishri b,1, Ahmed Haroun Mohamed c, 2
a

Maxillofacial and Plastic surgery, Plastic Surgery Department, Faculty of medicine, El Minia University, Egypt Maxillofacial Surgery Department, Al Noor Specialist Hospital, Makkah, Saudia Arabia c Diagnostic Radiology, Faculty of Medicine, Mansoura University, Al Noor Specialist Hospital, Makkah, Saudia Arabia
b

a r t i c l e i n f o
Article history: Paper received 3 April 2010 Accepted 11 October 2010 Keywords: Distraction osteogenesis Three-dimensional CT scan Pierre Robin sequence

a b s t r a c t
The aim: The aim of this work was to assess the multislice CT scan for analysis of the craniofacial anatomic changes in Pierre Robin sequence both predistraction and postdistraction, and to assess the use of unidirectional internal distractors in this patient group. Patients & methods: The study involved 11 patients. Their age at the time of distraction ranged from 2 to 7 months. Six were females and 5 were males. All had retromicrognathia, glossoptosis and obstructive sleep apnoea. All were diagnosed clinically and by CT scan. All were managed by distraction osteogenesis and were followed postoperatively by multislice CT. Results: The distance between the base of the tongue and the posterior pharyngeal wall increased by a mean of 141%, and the total mandibular length increased by a mean of 26%. The increase in the distance between the hyoid bone and the posterior pharyngeal wall increased by a mean of 42% .The distance between the hyoid bone and the genoid process increased by a mean of 9%. Conclusion: Multislice CT scan was found to be a practical imaging technique to evaluate the morphologic changes in the airway and the mandible after distraction osteogenesis. It rules out the need for other traditional methods. Owing to the plasticity and malleability of the infant mandible that allow for sufcient bone remodelling, unidirectional internal distractors achieved a satisfactory maxillomandibular relationship which was tolerated by the infants and accepted by the parents. 2010 European Association for Cranio-Maxillo-Facial Surgery.

1. Introduction Pierre Robin (PR) is a sequence of events arising from the poor development of the mandible. Micrognathia causes the tongue to be displaced to the back of mouth (glossoptosis). Concomitant reduction of the oropharyngeal airway leads to upper airway obstruction. The patients have symptoms of obstructive sleep apnoea (OSA) that in severe cases needs tracheostomy. The tongue may interfere with the closure of the palate resulting in a horse

q The department to which the work is attributed to: The work is attributed to the department of maxillofacial surgery, Al Noor Specialist Hospital, Makkah, Saudia Arabia; Head of this institution and head of the maxillofacial department is Dr Awad El Bishri who is one of the authors; the work has been approved by the ethical committee of the hospital. Parents gave informed consent to the work. qq Sources of support in the form of grants: There were no sources in the form of grants. * Corresponding author. Tel.: 20 145051930; fax: 20 862342502/3. E-mail addresses: mahrous_ahmed2004@yahoo.com (A. Mahrous Mohamed), albishri@hotmail.com (A. Al Bishri), ahmharon@gmail.com (A. Haroun Mohamed). 1 Tel.: 966 50064067; fax: 966 25664314. 2 Tel.: 966 0502561208; fax: 966 25664314.

shoe-shaped cleft palate (Cohen et al., 1998; Dauria and Marsh, 2008; Marques et al., 1998). The management of Pierre Robin sequence (PRS) varies according to the degree of severity of the individual case. Conservative management includes prone positioning and the placement of nasopharyngeal airway stents. Signicant airway obstruction mandates more aggressive therapy, including tongueelip adhesion and hyomandibulopexy. Tracheostomy has been considered the conventional alternative for management of upper airway obstruction in PRS; however, it is associated with a high cost in terms of morbidity and mortality rates. The average age of decanulation for children with PRS is 3.1 years (Denny and Kalantarian, 2002; Mandell et al., 2004). Distraction osteogenesis (DO) has become an accepted method of treatment for patients with a hypoplastic mandible. It achieves mandibular lengthening without need for a bone graft. During the recent past, mandibular reconstruction by DO has been demonstrated to be effective in resolving upper airway obstruction and for tracheostomy decanulation. It has also been used in respiratory distressed neonates and infants to avoid tracheostomy (Denny and Kalantarian, 2002; Looby et al., 2009; Morovic and Monasterio, 2000).

1010-5182/$ e see front matter 2010 European Association for Cranio-Maxillo-Facial Surgery. doi:10.1016/j.jcms.2010.10.016

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Fig. 3. Preoperative axial CT showing narrowing of the transverse and the anteroposterior dimensions of the airway.

Fig. 1. Preoperative 3DCT showing the craniofacial skeleton of one patient with retromicrognathic mandible and maxillomandibular ridge disharmony.

Fig. 2. Preoperative sagittal CT showing glossoptosis and severe airway obliteration.

Fig. 4. Preoperative 3DCT for the transverse dimensions of the airway. The arrow points to the site of narrowing.

Traditional methods of mandibular distraction use multidirectional external distractors. These distractors allow multiplanar manipulation of the mandibular segments and allow ne adjustment of the maxillomandibular relationship. New, unidirectional internal microdistractors allow placement of inconspicuous internal devices with minimal morbidity. Infants tolerate these internal distractors more readily than the external distractors. The internal distractors also offer minimal risk of dislodgement and they avoid the scars associated with the insertion of external distractor pins through soft tissues. They do not allow ne adjustment of mandibular segments to correct any occlusal disharmony that occurs during the distraction process which is a reported drawback (Roy et al., 2009).

Computed tomography (CT) and its 3-dimensional (3D) reconstructive imaging technique have been widely used in the diagnosis, treatment planning, surgical guidance, evaluation of results, and follow-up studies of maxillofacial deformities. Helical CT scanning technique in combination with 3D rendering techniques enables the use of high-quality 3DCT images. The helical CT scanner provides adequate image data to create 3D images with reduced scanning time and radiation dose compared with the conventional CT scans because of the continuous scanner rotation and Tabletop movement (Cademartiri et al., 2004; Girod et al., 1995; Hoponik et al., 1983; Metes et al., 1993; Ono et al., 1992). The scan time has been reduced signicantly and the smallest details can be scanned within a predictable scan time (Springer and Heidelberg, 2007).

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Fig. 7. Postoperative 3DCT showing the craniofacial skeleton of the same patient in Fig. 1 with increase in the total mandibular length and normal maxillomandibular ridge relationship.

Fig. 5. Preoperative 3DCT for the anteroposterior dimensions of the airway. The arrow points to the site of narrowing.

Fig. 8. Postoperative sagittal CT showing improvement in the anteroposterior airway dimensions and forward displacement of the hyoid bone.

Fig. 6. Intraoperative view showing distractor application.

Because the information within the CT image data involves both hard and soft tissue structures, it is possible to design a threedimensional computerized imaging technique to allow threedimensional visualization of the pharyngeal airway and the mandible (Calhoun et al., 1999; Kawamata et al., 2000). A new innovative 3D cephalometric analysis has been recommended for making a precise diagnosis. It uses the set-up and validation of a voxel-based semi-automatic 3D cephalometric reference system. The 3DCT method is widely used and acceptable for many reasons. Actual measurement can be obtained, and the 3D

image can be rotated easily by changing the rotational axis. In addition, 3DCT image can also show asymmetry of the midface and the cranial base that is difcult to be detected in ordinary 2D X-ray lm (Maeda et al., 2006; Netherway et al., 2006; Swennen et al., 2006). Olszewski et al. (2010), proposed a classication scheme and exclusion criteria for reference landmarks used in 3D cephalometrics, based on inter-observer reproducibility and anatomical reality. 2. Patients & methods Over the past 3 years, 27 patients having retromicrognathia, glossoptosis, obstructive sleep apnoea and cleft palate were

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Fig. 9. Postoperative axial CT of the same patient in Fig. 3 showing improvement in the transverse and the anteroposterior dimensions of the airway.

Fig. 11. Postoperative 3DCT showing widening of the anteroposterior dimensions of the airway.

Fig. 10. Postoperative 3DCT showing widening of the transverse dimensions of the airway.

diagnosed as Pierre Robin sequence. These infants were managed by conservative measures including prone positioning, application of nasopharyngeal airway or tongue stitch. Most of these patients (16 patients) responded to these conservative measures, but 11 patients did not respond well. They were subjected to DO. Six of them were females and 5 were males. Their age at initial presentation ranged from 3 days to 7 months age. All had retromicrognathia, glossoptosis, cleft palate and symptoms of OSAS as noisy breathing during sleep, fragmented sleep, pauses in respiration and daytime somnolence. All patients were unable to control their airway during feeding, as evidenced by repeated episodes of choking and obstruction. Birth weights ranged from 2.9 to 3.3 kg. Their age at the time of DO ranged from 2 to 7 months. Polysomnographic sleep studies revealed that the respiratory disturbance index ranged between 9 and 20 apnoeas per hour (mean 14), and oxygen saturation ranged between 75 and 85% (mean was 81%). Patients were assessed by multidetector CT scan in a low dose technique with the eye closed using a sedating dose of midazolam (0.01e0.1 mgm/kgm) under care of an anaesthiologist. The device used was 64 multidetector CT, GE(General Electric), CTV(CT volume), ADW (Advantage workstation) 4.3 (Figs. 1e5). The surgical procedure was performed under GA with nasotracheal intubation in 7 patients and by tracheostomy in the

Fig. 12. 3DCT of the distracted mandible with the distractors in place at the end of the consolidation period showing a good quality of deposited bone as shown by smooth intact regular cortices.

remaining 4 patients because of severe upper airway obstruction. A submandibular incision was done bilaterally and the ap elevated to the lower border of the mandible. Buccal and lingual corticotomies were done in the distal part of the mandibular body. Unidirectional internal microdistractors were used bilaterally (modied track plus alveolar distractor e KLS Martin, L.P., modied by removing its distractor arms on one side). The distractor was xed by screws anterior and posterior to the corticotomy. Completion of the corticotomy into osteotomy was then done. Activation of the distractor in place was tried to assess the completeness of the osteotomy. The tissues were closed over the distractor. The distractor arm was placed subcutaneously and its free end was left uncovered to facilitate the distraction process (Fig. 6). Distraction started 24 h after application of the distractor and activated 1 mm daily (0.5 mm morning and 0.5 mm evening). Patients with severe OSAS were kept in the intensive care unit for several days for monitoring the postoperative airway and oxygen

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Fig. 13. Intraoperative view at the time of distractor removal showing good quality of bone as appears between the distractor arms.

Fig. 15. Preoperative prole view of one of the patients with retromicrognathia and tracheostomy tube in place.

Fig. 14. One year postdistraction 3DCT of the head and neck shows normal growth of the mandible with normal alveolar ridge relationship.

saturation until the swelling reduced and the distraction was nearly completed. After 10 weeks of consolidation period, the distractors were removed under GA. With respect to the airway, either uneventful decanulation of the tracheostomy, uneventful removal of the tongue stitch, uneventful removal of the nasopharyngeal airway or oxygen saturation above 95% was considered as success. Follow up of the distraction process was done by CT scan in axial, sagittal planes and three-dimensional CT of the facial bones and upper airway (Figs. 7e12). The measurements were made on 2D images (of 3D models) and were cephalometrically calibrated. The CT data from the in vivo scan were extracted and analysed for the following: 1. Distance from the posterior pharyngeal wall to the tongue base at the level of the C1 arch measured in the axial and midsagittal planes. This represented the anteroposterior dimensions of the airway (Figs. 8 and 9). 2. Distance from the lateral pharyngeal wall on one side to that on the other side at the level of the C1 arch measured in the axial plane. This represented the transverse dimensions of the airway (Figs. 3 and 9). 3. Mandibular ramus length as measured from a point on the posterior condyle (articulare: Ar) to the midpoint of the mandibular angle (gonion: G) (Figs. 17 and 18). 4. Mandibular body length as measured from the gonion to the most protrusive point of the symphysis (pogonion: Pg) (Figs. 17 and 18).

Fig. 16. Prole view of the same previous patient after distractor removal with improvement of the micrognathia and inconspicuous scar.

5. Total mandibular length which is the sum of both the length of the ramus and the body (Figs. 17 and 18). 6. The distance from the posterior pharyngeal wall to the hyoid bone and the distance from the hyoid to the genoid process. This represented the degree of forward displacement of the hyoid bone with distraction (Fig. 8). 7. The maxillomandibular alveolar ridge distances were measured by extending their long axis up and down. The distance between the maxillary and the mandibular axis was measured at the occlusal plane. It ranged from 14 to 18 mm (mean 15 mm).

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Fig. 17. Extracted 3DCT of the mandible showing its predistraction dimensions. Ar points to the point articulare, G points to the gonion point and Pg points to the pogonion point.

The follow-up period was from 12 to 24 months after removal of the distractor.
Fig. 18. Extracted 3DCT of the mandible showing its postdistraction dimensions.

3. Results Mandibular distraction was successful in all patients (Figs. 15e18). The deposited bone was of good quality (Figs. 12 and 13). Bone quality was dened subjectively at the time of distractor removal (intact smooth regular cortices), from the 2D CT and from the 3D CT. From 2D CT the bone density of the newly formed bone was the same density of the adjacent bone. From 3D CT, the newly formed bone was homogenous with the adjacent bone. The average linear mandibular lengthening was 18 mm (range 15e23 mm). One patient had an incomplete osteotomy on one side. The distraction stopped on this side. He was diagnosed both clinically and radiologically. The osteotomy was completed again and the distraction continued. One patient developed unilateral mild weakness of the marginal branch of the facial nerve which improved spontaneously before removal of the distractor. The surgical scar was hidden in the submandibular region. Polysomnographic sleep studies revealed that the respiratory disturbance index ranged between 0 and 2 apnoeas per hour (mean 1), and oxygen saturation ranged between 95 and 99% (mean 97%). All patients with a tracheostomy were decanulated, patients with tongue stitches had them removed and patients with nasopharyngeal airways had them removed, all uneventfully. Symptoms of OSAS improved and weight gain was excellent. Clinical examination showed good arch harmony without open bite or cross bite deformity (Fig. 7). There were signs of mild infection in 3 patients at the distractor site. These were successfully managed with systemic antibiotics and dressings. Analysis of the CT demonstrated the followings (Table 1): 1. The increase in the anteroposterior dimensions of the airway ranged from 81 to 248% with a mean of 141% (Figs. 8, 9, 11). 2. The increase in the transverse dimensions of the airway ranged from 1 to 8% with a mean of 4% (Figs. 9 and 10). 3. The ramus height increased between 15 and 20% with a mean of 18% (Figs. 7, 17 and 18). 4. The body length increased between 5 and 30% with a mean of 22% (Figs. 7, 17 and 18). 5. The total mandibular length increased between 16 and 33% with a mean of 26%. The forward lengthening of the mandible resulted in forward traction of the tongue and increased hypopharyngeal airway space. A reciprocal relationship was found between the % of increase in the total mandibular length and the % of increase in the anteroposterior dimensions of the airway (Figs. 7, 17 and 18). 6. The increase in the distance between the hyoid bone and the posterior pharyngeal wall ranged from 33 to 56% with a mean of 42%. The distance between the hyoid bone and the genoid process increased between 8% and 12% with a mean of 9% (Fig. 8). 7. Maxillomandibular alveolar ridge distances were corrected to a mean of 2 mm after distraction. After 6e12 months (Fig. 14), all patients had grown normally as evidenced clinically, were able to feed themselves and had no breathing problems. 4. Discussion The use of mandibular DO in alleviating upper airway obstruction in infants with micrognathia is rapidly expanding. Series presented by Denny et al. (2001); Denny and Kalantarian (2002); Sidman et al. (2001); Wittenborn et al. (2004), and Mandell et al. (2004), conrm the utility of this option. New internal devices have been shown to have signicant advantages over the bulky external devices. This was also shown by Lin et al. (2007), and by Roy et al. (2009). One purpose of this study was to determine if unidirectional vector forces are sufcient to achieve a good maxillomandibular relationship. The study has shown that this have been achieved clinically with no open bite or cross bite deformities. Functional loading and bony remodelling in this young age group are important in achieving this relationship. Imaging evaluation of the morphological airway changes has been performed in patients with OSAS. Imaging techniques include cephalometric radiography (Ppin et al., 1999), CT (Hoponik et al.,

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Table 1 Patients demographic data, average increase in airway dimensions, distance of the hyoid bone from the posterior pharyngeal wall and total mandibular length. No Age at the time of distraction in months 2 5 2 2 5.5 5.5 3 7 2 6 5 4.5 Sex The % of increase in the anteroposterior dimensions of the airway 81 248 128 116 138 140 135 122 126 124 198 141 The % of increase in the transverse dimensions of the airway 1 8 5 3 4 6 4 4 2 1 6 4 The % of increase in the anterior displacement of the hyoid 33 56 42 41 41 44 38 36 43 43 45 42 The % of increase in the total mandibular length 16 33 26 20 30 31 28 22 25 24 32 26

1 2 3 4 5 6 7 8 9 10 11 Mean

Male Female Male Female Male Female Female Female Male Female Male

1983; Ppin et al., 1999), and magnetic resonance imaging (Ryan et al., 1991). With conventional cephalometric radiography, it is difcult to observe the pharyngeal airway 3-dimensionally. In frontal cephalometric radiography, hard tissue structures such as the anterior teeth and the pharyngeal airway overlap. With 3DCT imaging it is possible to visualize the pharyngeal airway 3-dimensionally without obstruction by hard tissue structures (Iatrou et al., 2009). CT scans create 3D images using ionizing radiation. According to the alliance for radiation safety in paediatric imaging, a CT scan of the head exposes a child to the amount of radiation he should naturally experience during approximately 8 months. Apart from scientic reasons, it is better to avoid duplicating diagnostic CT. It is mandatory to discuss the benets and risks with the parents (Zoetelief and Geleijns, 1998). A study performed by Williams et al. (1999), demonstrated expansion of the mandibular framework with advancement of the base of the tongue, leading to increased pharyngeal airway for early decanulation of tracheostomy dependent patients. This is determined on the basis of cephalometric study measuring the advancement of hyoid bone along the axis of the mandibular body after distraction. Despite the great increase in the distance between the posterior pharyngeal wall and the hyoid bone (42%), the genioehyoid relationship remained relatively constant after mandibular distraction, with only a 9% increase. This relationship supports the notion that the hyoid moves forward as the distal mandibular segment is anteriorly distracted. This is in accordance with that reported by Roy et al. (2009), who reported only a 14% increase in the genioehyoid distance. Roth et al. (1997) measured the advancement of the hyoid bone along the axis of the mandibular body after distraction. An average advancement of 14.5 mm was found. Any displacement of the hyoid bone away from the posterior pharyngeal wall was believed to aid in decreasing airway obstruction. In their CT analysis after distraction osteogenesis in Pierre Robin sequence, Roy et al. (2009), showed that the total mandibular length had been increased by 26%, and the posterior distance from the pharyngeal wall had been increased by 198.9%. Three recent studies have examined both mandibular volume and upper airway volume after DO of the mandible on the basis of 3DCT calculations. The rst study was done by Perlyn et al. (2002), who examined four patients (two with Nager syndrome and two with Treacher Collins syndrome) who underwent mandibular distraction. The mandibular volume and upper airway volume increased on postdistraction CT scan by an average of 27% and 37%, respectively. The second study was by Rachmiel et al. (2005), who studied 12 patients who similarly underwent mandibular

distraction to correct airway obstruction. The mandibular volume and upper airway volume increased on postdistraction CT scan by an average of 28% and 72%. Neither study, however, was performed on neonates (the youngest child in these studies was 12 months old). The third study was by Roy et al. (2009), who studied three infants with PRS and severe upper airway obstruction who underwent DO. A 32.0% increase in mandibular bone volume was present after distraction osteogenesis compared with the remainder of the craniofacial skeleton. When mandibular bone volume was compared with maxillary growth, they found a 39.1% increase in bone growth of the mandible after distraction osteogenesis. The midsagittal airway area improved by 150.0%, and the 3D airway volume increased by 192.0%. Expansion of the width of the airway over part or the entire airway will be reected in an increase of the internal volume of the airway, as this volume is the longitudinal integral of the crosssectional area and consequently reects a decrease in the ow resistance over the length of the airway (Perlyn et al., 2002). We reviewed papers to relevant DO in the mandible published in the last 3 years. Kruse et al. (2008) utilized micro-CT to dene bone quality and density. Wolvius et al. (2008) described a new custom made distractor xed by wires for distraction of four PR patients. Schendel et al. (2008) described the use of an internal curvilinear distractor and evaluated their work clinically and by CT scan after DO of 16 micrognathic infants. Rohit et al. (2008) stated that mandibular DO is a viable option in the paediatric age group with intervention as early as 8 months of age. They utilized an intraoral ramus distractor. Iatrou et al. (2009) reported one case of severe PRS that was distracted extraorally bilaterally by an internal distractor due to lack of intraoral space. They followed the patient by anteroposterior cephalogram and by 3DCT. Sadaka et al. (2009), utilized intraoral distractors to relieve airway obstruction in seven patients. Their age ranged from 7 months to 8 years. They followed their patients by conventional radiography. Olszewski et al. (2010) measured the reproducibility of osseous landmarks identied from two recently described cephalometric analysis: 3D-ACRO and 3D-Swennen analysis. Rania et al. (2010) showed that there is a wide variety in treatment approaches for craniofacial anomalies in Europe. There is disagreement on the essential steps in the distraction process. 5. Conclusions Multislice CT scan was found to be a practical imaging technique to evaluate the morphologic changes in the airway and the mandible after distraction osteogenesis. It rules out the need for other traditional methods. Owing to the plasticity and malleability of the infant mandible, that allow good bone remodelling, unidirectional internal distractors achieved satisfactory maxillomandibular

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relationship which was tolerated by the infants and accepted by the parents. Conict of interest There was no conict of interest. Acknowledgments The authors express their great appreciation to the staff members of the anaesthesia department, ENT department and Paediatric ICU in Al-Noor specialist hospital, Makkah who provided pure technical help and general support. References
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