[Show abstract][Hide abstract] ABSTRACT: The aim of this study was to describe directional and fluctuating mandibular asymmetry over time in children with Crouzon or Apert syndrome. Mandibular asymmetry of children between 7.5 and 14 years of age with Crouzon syndrome (n = 35) and Apert syndrome (n = 24) were compared with controls (n = 327). From panoramic radiographs, mandibular directional and fluctuating asymmetry was determined for the three groups. Multilevel statistical techniques were used to describe mandibular asymmetry changes over time. Patients with Crouzon and Apert syndromes showed statistically significant more fluctuating asymmetry for mandibular measures than did controls. Between the Crouzon and Apert syndromes groups, no statistical differences were found in directional and fluctuating asymmetry. The control group showed statistically significantly more directional asymmetry than did patients with Crouzon or Apert syndrome. The controls showed no change over time for the directional asymmetry of condylar-ramal height; however, the directional asymmetry of the gonial angle increased. Patients with Crouzon syndrome showed side dominance for only condylar-ramal height; whereas, patients with Apert syndrome did not show dominance for any of the measurements. Apert and Crouzon syndromes showed developmental instability, in contrast to the controls. No statistically significant longitudinal differences were found for either the directional or the fluctuating asymmetry between Crouzon and Apert syndromes. Findings for fluctuating and directional asymmetry for both syndromes may indicate an inability to cope with genetic and environmental stress during development and treatment, compared with untreated nonsyndromic individuals.
The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association. 05/2014;
[Show abstract][Hide abstract] ABSTRACT: The purpose of this systematic review is to provide an overview of the surgical correction of the mandible in unilateral craniofacial microsomia (UCM) performed in the growing patient, and its long-term outcome and stability. The following databases were searched: PubMed, Embase, Cochrane, and Web of Science. Articles reporting prospective and retrospective studies of patients not older than 16 years (N ≥ 4) who had undergone surgical correction of a craniofacial microsomia spectrum condition using grafts, osteotomies, distraction, or combinations of these, were reviewed. The period of follow-up was selected to be ≥1 year. After inclusion, the articles were evaluated on short- and long-term outcomes, relapse, and any increase in asymmetry following treatment. Thirty of 1611 articles were included in the qualitative synthesis. Analysis of the surgical mandibular correction of UCM showed that the outcome is not so much treatment-dependent, but patient-dependent, i.e. deformity gradation-dependent. The type I–IIa Pruzansky–Kaban patient had the best results with regard to minimal relapse and/or minimal increase in asymmetry. Single-stage correction of the asymmetry should be postponed until the permanent dentition stage. It can be concluded that in the treatment of the severely hypoplastic mandible, the patient will benefit from a multi-stage treatment protocol if indicated for functional or psychological problems.
International Journal of Oral and Maxillofacial Surgery 01/2013; · 1.52 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: In a 48-year-old woman and a 35-year-old man, a sinusitis maxillaris resulting from overextension of root canal filling material after an endodontic treatment ofa maxillary molar could be diagnosed through computed tomography. The woman complained of nasal congestion and in the man 2 radiopaque abnormalities in the maxillary sinus were discovered coincidentally. The corpora aliena were removed surgically. Root canal filling materials containing zinc oxide or hydrocortisone may cause aspergillosis of the maxillary sinus because they encountergrowth of Aspergillus species. In case ofmaxillary sinusitis, surgical removal of overextended root canal filling material from the maxillary sinus is indicated. No treatment consensus consists for cases without maxillary sinusitis symptoms.
Nederlands tijdschrift voor tandheelkunde 10/2012; 119(10):480-3.
[Show abstract][Hide abstract] ABSTRACT: The aim of this study was to design mandibular ramal height growth curves for patients with HFM and compare those with the curves for a Dutch reference population. Two hundred fifty-one pre-operative orthopantomograms (OPTs) from 84 patients with unilateral HFM were used in conjunction with a control set of 2260 OPTs from 329 healthy individuals from the Nijmegen Growth Study (NGS) to determine mandibular ramal distances. For grades I/IIa and IIb/III, and for both sides, growth curves were constructed for mandibular ramal height with a linear curve-fitting procedure. This procedure revealed a significant difference between HFM patients and the NGS control group (p < 0.001); both in the mild and severe group mandibular ramal height differed significantly between the affected and non-affected side (p < 0.001). Growth was similar between HFM patients and the NGS control group. HFM patients therefore start with a smaller mandible and end with a smaller mandible, but experience growth similar to the Dutch normal population. These growth curves may aid the timing and determination of the combined surgical orthodontic treatment plan for HFM patients.
Journal of cranio-maxillo-facial surgery: official publication of the European Association for Cranio-Maxillo-Facial Surgery 07/2012; · 1.25 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: This cohort study in 35 patients (13 children) evaluates the prevalence, severity and anatomical cause of obstructive sleep apnoea syndrome (OSAS) in patients with Treacher Collins syndrome. Ambulatory polysomnography was performed cross-sectionally to determine OSAS prevalence and severity. All upper airway related surgical interventions were evaluated retrospectively. In 11 patients, sleep endoscopy, and flexible and rigid endoscopy were applied to determine the level of anatomical obstruction of the upper airway. The overall prevalence of OSAS in Treacher Collins patients was 46% (54% in children; 41% in adults). Thirty-eight upper airway related surgical interventions were performed in 17 patients. Examination of the upper airway revealed various anatomical levels of obstruction, from the nasal septum to the trachea. Most significant obstruction was found at the level of the oro/hypopharynx. OSAS in Treacher Collins patients is an important problem so all patients should be screened for OSAS by polysomnography. Endoscopy of the upper airways was helpful in determining the level of obstruction. Surgical treatment at one level will not resolve OSAS in most patients because OSAS in Treacher Collins has a multilevel origin. Non-invasive ventilation (continuous positive airway pressure or bilevel positive airway pressure) or tracheotomy should be considered as a treatment modality.
International Journal of Oral and Maxillofacial Surgery 06/2012; 41(6):696–701. · 1.52 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: Facilities for hyperbaric oxygen therapy that are suitable for animal experimental research are scarce. In this paper, the authors introduce a hyperbaric oxygen chamber that was developed specifically for animal experimental purposes. The hyperbaric oxygen chamber was designed to meet a number of criteria regarding safety and ease of use. The hyperbaric oxygen chamber conforms to 97/23/EC (Pressure Equipment Directive), Conformity Assessment Module G Product Group 1. It provides easy access, and can be run in manual mode, semi-automatic mode and full-automatic mode. Sensors for pressure level, oxygen level, temperature, humidity and carbon dioxide level allow full control. This state-of-the-art hyperbaric oxygen chamber for animal experimental purposes permits the investigation of the biological mechanisms through which hyperbaric oxygen therapy acts at a fundamental level.
International Journal of Oral and Maxillofacial Surgery 12/2011; 41(2):271-4. · 1.52 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: Hypoplastic mandible related airway obstruction therapy may vary from non-surgical to surgical, depending on the severity of the obstruction. Since its introduction in 1992, distraction osteogenesis (DO) has been used to lengthen the mandible. A new intraoral device is presented here. It has no need of screw fixation, whilst placement and activation are intraoral, leading to simple placement and removal. Seven cases with hypoplastic mandible related airway obstruction were treated between 2001 and 2008 with intraoral DO at the authors' institution. Six patients had a tracheostomy and one received continuous positive airway pressure (CPAP). After the distraction phase, five patients could be decanulated and in one case CPAP was no longer required. In one case the desired effect was not achieved. The findings suggest that early mandibular DO leads to damage to the permanent tooth buds resulting in missing teeth. The effectiveness of mandibular DO regarding early discharge of the patient is shown, leading to an overall cost reduction. Knowledge of alternative modalities such as mandibular DO in case of hypoplastic mandible related airway obstruction in neonatal healthcare should lead to early referral to a dedicated maxillofacial surgeon, possibly avoiding long-term airway support or tracheotomy.
International Journal of Oral and Maxillofacial Surgery 12/2011; 41(2):186-91. · 1.52 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: A systematic review of mandibular midline distraction (MMD) was carried out to assess effectiveness, treatment related difficulties, complications and biomechanical effects of this treatment modality objectively.
Randomized controlled trials (RCT), controlled clinical trials (CCT) and case series concerning MMD with a sample size of >5 were searched electronically in Pubmed/Medline, Embase, Cochrane and CENTRAL up to September 6th 2010.
85 unique articles were found of which 22 met our inclusion criteria. The study designs of the articles found were prospective (9), retrospective (9) or uncertain (2). One clinical trial and no randomized clinical trials were found. The methodological quality was considered low in most articles.
Generally MMD is a safe and effective treatment modality to treat transverse mandibular discrepancies, however controversies still exist: choice of distractor, surgical setting, distraction rate, start of orthodontic treatment and relapse. In addition, little is known about patient experience and quality of life after treatment. Further prospective trials are necessary to address these controversies and questions.
Journal of cranio-maxillo-facial surgery: official publication of the European Association for Cranio-Maxillo-Facial Surgery 06/2011; 40(3):248-60. · 1.25 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: Severe midface hypoplasia in patients with various craniofacial anomalies can be corrected with Le Fort III or monobloc advancement. Often additional corrective orthognathic surgery is indicated to achieve Class I occlusion and a normal inter-jaw relationship. This study evaluated the incidence of, and the surgical indications for, secondary orthognathic surgery following Le Fort III/monobloc advancement. The total study group consisted of 41 patients: 36 patients with Le Fort III advancement and 5 patients with monobloc advancement. Seven patients underwent additional orthognathic surgery. Of the resulting 18 non-operated patients older than 18 years at the end of follow-up, Class I occlusion was observed in 11 patients. In the remaining patients malocclusions were dentally compensated with orthodontic treatment. None of the patients was scheduled for additional orthognathic surgery due to the absence of functional complaints and/or resistance to additional surgery. Le Fort III and monobloc advancement aim to correct skeletal deformities on the level of zygoma, orbits, nasal area and forehead, but Class I occlusion is frequently not achieved. Additional orthognathic surgery is often indicated in patients undergoing Le Fort III or monobloc advancement. Naso-endoscopic analysis of the upper airway and the outcomes of sleep studies may influence the orthognathic treatment plan.
International Journal of Oral and Maxillofacial Surgery 03/2011; 40(7):679-84. · 1.52 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: Almost 50% of patients with Apert, Crouzon or Pfeiffer syndrome develop obstructive sleep apnea (OSA), mainly due to midface hypoplasia. Midface advancement is often the treatment of choice, but the few papers on long-term outcome report mixed results. This paper aimed to assess the long-term respiratory outcome of midface advancement in syndromic craniosynostosis with OSA and to determine factors contributing to its efficacy. A retrospective study was performed on 11 patients with moderate or severe OSA, requiring oxygen, continuous positive airway pressure (CPAP), or tracheostomy. Clinical symptoms, results of polysomnography, endoscopy and digital volume measurement of the upper airways on CT scan before and after midface advancement were reviewed. Midface advancement had a good respiratory outcome in the short term in 6 patients and was ineffective in 5. In all patients without respiratory effect or with relapse, endoscopy showed obstruction of the rhino- or hypopharynx. The volume measurements supported the clinical and endoscopic outcome. Despite midface advancement, long-term dependence on, or indication for, CPAP or tracheostomy was maintained in 5 of 11 patients. Pharyngeal collapse appeared to play a role in OSA. Endoscopy before midface advancement is recommended to identify airway obstruction that may interfere with respiratory improvement after midface advancement.
International Journal of Oral and Maxillofacial Surgery 02/2010; 39(2):115-21. · 1.52 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: Little is known about the long-term prevalence of elevated intracranial pressure (ICP), obstructive sleep apnoea (OSA), level of education, language and motor skills, impaired sight and hearing in craniosynostosis syndromes. The objective of this study was to define the prevalence per syndrome of elevated ICP, OSA, impaired sight and impaired hearing.
A retrospective study was undertaken on 167 consecutive patients diagnosed with Apert, Crouzon, Pfeiffer, Muenke or Saethre-Chotzen syndrome, aged 1-25 years and treated between 1983 and 2008. The mean age at time of referral and review was 1 years and 2 months and 10 years and 3 months, respectively.
Patients with Apert and Crouzon/Pfeiffer syndromes had the highest prevalence of elevated ICP (33% and 53%, respectively) and OSA (31% and 27%, respectively), while Saethre-Chotzen syndrome was also associated with a fair risk for elevated ICP (21%). The prevalence of impaired sight (61%) and hearing (56%) was high in all syndromes.
Based on these data, a syndrome-specific risk profile with suggestions for screening and treatment is presented.
Journal of Plastic Reconstructive & Aesthetic Surgery 11/2009; 63(10):1635-41. · 1.44 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: Experimental research on optimising the distraction protocol has been performed extensively in the past. However, relatively little research has been done on the rhythm of distraction. Findings in the orthopaedic literature showed that the outcome of distraction osteogenesis (DO) is positively influenced by increasing the rhythm of distraction. The aim of this study is to quantitatively compare continuous with discontinuous rhythms of distraction in rabbits.
Tissue blocks of regenerated bone were harvested from thirty-eight young adult female New-Zealand White rabbits. After a latency period of three days, rabbits were subjected for eleven days to either single daily activation of the distractor at a rate of 0.9 mm/d, or triple daily activation at a rate of 0.9 mm/d, or continuous activation at a rate of 0.9 mm/d. After three weeks of consolidation, bone regenerates were analysed using histomorphometry.
The continuous DO group showed significantly (p<.01) more regenerate bone volume in the central part of the regenerate than the discontinuous DO groups. Higher osteoblastic activity was seen, as well as more blood vessels (p<.05). Bone volume and the number of blood vessels correlated significantly in the central part of the regenerate (p<.05). Also, the early mineral apposition rate (MAR) was higher than the late MAR (p<.05).
Continuous DO significantly accelerates bone formation when compared with discontinuous DO.
Journal of cranio-maxillo-facial surgery: official publication of the European Association for Cranio-Maxillo-Facial Surgery 06/2009; 37(7):398-404. · 1.25 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: This study evaluated stability, tipping and relapse after surgically assisted rapid maxillary expansion (SARME), comparing bone-borne and tooth-borne devices, in skeletally matured non-syndromal patients with transverse maxillary hypoplasia. The study is a randomized, open-label, clinical trial. Patients were randomized to bone-borne (n=25) and tooth-borne (n=21) groups. The surgical technique for corticotomy was the same in both groups. Expansion was performed using a bone-borne or tooth-borne device. Dental study casts, lateral and postero-anterior cephalograms were taken before treatment, after the distraction phase and at 12-month follow up. Stability, segmental maxillary tipping and relapse were studied. 23 bone-borne and 19 tooth-borne patients were analyzed. There were no significant differences between the two groups. Widening was comparable at canine, premolar and molar level. Relapse was not significant and at follow up the significant increase in distance was sustained. A significant increase in palatal width, at premolar and molar level, occurred in both groups. The maxilla moves slightly downward in SARME. Segmental maxillary tipping occurred in both groups and did not affect relapse. There is no significant difference between the two groups. In SARME, the widening achieved at dental level is stable after 12 months. Over-correction is not necessary. Tipping of the maxillary segments and increases in the retention period are equal in both groups.
International Journal of Oral and Maxillofacial Surgery 05/2009; 38(4):308-15. · 1.52 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: Unilateral maxillary hypoplasia is a characteristic feature of an oblique facial cleft. This three-dimensional shortage of osseous structures and soft tissue becomes more prominent over the years and is difficult to correct. The authors describe a 17-year-old boy born with a unilateral nasomaxillary dysplasia and nasal dysplasia (Tessier type 1, 2, 3) who underwent a hemi-Le Fort III distraction with a rigid external distraction (RED) system. This distraction, in combination with initial peroperative advancement and retained with elastic traction with a facial mask, achieved a substantial horizontal improvement of the hemi-midface; this resulted in a better projection of the left cheek, infra-orbital rim, nasal ala, and improved occlusion. Owing to the underdevelopment of the maxilla and zygoma in all three dimensions, contour deformities remain. Creating several bone segments for multidirectional distraction would jeopardize vascularization of the bone. Good long-term planning is essential in these complicated cases, and more long-term results need to be assessed. The major reconstructive challenge for this complex pathology continues.
International Journal of Oral and Maxillofacial Surgery 05/2009; 38(7):790-4. · 1.52 Impact Factor