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Journal of Oral and Maxillofacial... Jul 2016A variety of surgical protocols are available in the literature for performing mandibular distraction. This study aims to determine the ideal rate of distraction and... (Review)
Review
PURPOSE
A variety of surgical protocols are available in the literature for performing mandibular distraction. This study aims to determine the ideal rate of distraction and compare outcomes between internal and external distractors in children and infants with upper airway obstruction due to micrognathia.
PATIENTS AND METHODS
A systematic review was performed. The databases searched included PubMed, Embase, Scopus, Web of Knowledge, and gray-literature sources. The intervention was bilateral mandibular distraction for upper airway obstruction in children with clinical evidence of micrognathia or Pierre Robin sequence. The variables for comparison included distraction rates of 1 mm/d and 2 mm/d and external versus internal distractors. The outcome was successful completion of distraction. Complications also were recorded and compared. The data were analyzed by cross tabulation to calculate odds ratios.
RESULTS
Overall, 43 studies were included in the surgical outcomes analysis. The overall success rate of distraction was 95.4%. There was no statistically significant difference in success rate with distraction at 1 mm/d or 2 mm/d (P = .18). Distraction at 1 mm/d was associated with a higher rate of technical failures. External distractors had a higher failure rate than internal distractors (P = .012). Internal distractors had a lower rate of significant scarring (P = .006) and had a lower incidence of technical failures (P = .039).
CONCLUSIONS
In children younger than 12 months, distraction at a rate of 2 mm/d is safe and appears to have a similar success rate to distraction at 1 mm/d. Internal distractors have a higher success rate than external distractors and should be used when possible.
Topics: Child; Child, Preschool; Craniofacial Abnormalities; Humans; Infant; Infant, Newborn; Micrognathism; Osteogenesis, Distraction
PubMed: 26920642
DOI: 10.1016/j.joms.2016.01.049 -
The Cleft Palate-craniofacial Journal :... Mar 2016Objective To gather data from relevant experimental and observational studies to determine the relationship between micrognathia and cleft palate. The goal is to raise... (Review)
Review
Objective To gather data from relevant experimental and observational studies to determine the relationship between micrognathia and cleft palate. The goal is to raise awareness and motivate clinicians to consider the cause and effect relationship when confronted with patients with cleft palate, even if there is no clearly noticeable mandibular abnormality. Design Several electronic databases were systematically examined to find articles for this review, using search terms including "cleft palate," "micrognathia," "tongue," and "airway obstruction." PubMed was the source of all the articles chosen to be included. Exclusion criteria included case reports, articles focused on treatment options, and articles only tangentially related to cleft palate and/or micrognathia. Results A total of 930 articles were screened for relevance, and 82 articles were chosen for further analysis. Evidence gathered in this review includes a variety of etiological factors that are causative or associated with both micrognathia and cleft palate. Observational studies relating the two abnormalities are also included. Much of the included literature recognizes a cause-and-effect relationship between micrognathia and cleft palate. Conclusion On the basis of the published data, we suggest that micrognathia does induce cleft palate in humans and animals. With knowledge of this causative relationship, clinicians should consider the importance of gathering cephalometric data on the mandibles and tongues of patients presenting with isolated cleft palate to determine whether they have micrognathia as well. With more data, patterns may emerge that could give insight into the complex etiology of nonsyndromic cleft palate.
Topics: Animals; Cephalometry; Cleft Palate; Humans; Micrognathism
PubMed: 25658963
DOI: 10.1597/14-238 -
Bone Feb 2024Arthrogryposis multiplex congenita (AMC) is a heterogeneous group of disorders associated with decreased fetal movement, with a prevalence between 1/3000 and 1/5200 live... (Review)
Review
INTRODUCTION
Arthrogryposis multiplex congenita (AMC) is a heterogeneous group of disorders associated with decreased fetal movement, with a prevalence between 1/3000 and 1/5200 live births. Typical features of AMC include multiple joint contractures present at birth, and can affect all joints of the body, from the jaw, and involving the upper limbs, lower limbs and spine. The jaws may be affected in 25 % of individuals with AMC, with limited jaw movement and mouth opening. Other oral and maxillofacial deformities may be present in AMC, including cleft palate, micrognathia, periodontitis and delayed teething. To our knowledge, oral and maxillofacial abnormalities have not been systematically assessed in individuals with AMC. Therefore, this scoping review was conducted to identify, collect, and describe a comprehensive map of the existing knowledge on dental and maxillofacial involvement in individuals with AMC.
METHODOLOGY
A scoping review was conducted in accordance with the Preferred Reporting Items for Systematic reviews and Meta-Analyses extension for Scoping Reviews guidelines. The PRISMA guidelines for scoping reviews were followed and databases were searched for empirical articles in English and French published until October 2022. We searched MEDLINE, Embase, Web of Science and ERIC databases. Two authors independently reviewed the articles and extracted the data.
RESULTS
Of a total of 997 studies that were identified, 96 met the inclusion criteria and were subsequently included in this scoping review. These 96 studies collectively provided insights into 167 patients who exhibited some form of oral and/or maxillofacial involvement. Notably, 25 % of these patients were within the age range of 0-6 months. It is worth highlighting that only 22 out of the 96 studies (22.9 %), had the primary objective of evaluating dental and/or maxillofacial deformities. Among the patients studied, a prevalent pattern emerged, revealing that severe anomalies such as micrognathia (56 %), high-arched palate (29 %), cleft palate (40 %), limited mouth opening (31 %), and dental anomalies (28 %) were frequently observed. Importantly, many of these patients were found to have more than one of these anomalies. Even though these maxillofacial impairments are known to be associated with dental problems (e.g., cleft palate is associated with oligodontia, hypodontia, and malocclusion), their secondary effects on the dental phenotype were not reported in the studies.
CONCLUSION
Our findings have uncovered a notable deficiency in existing literature concerning dental and maxillofacial manifestations in AMC. This underscores the need for interdisciplinary collaboration and the undertaking of extensive prospective cohort studies focused on AMC. These studies should assess the oral and maxillofacial abnormalities that can impact daily functioning and overall quality of life.
Topics: Infant, Newborn; Humans; Infant; Arthrogryposis; Cleft Palate; Micrognathism; Prospective Studies; Quality of Life
PubMed: 37951521
DOI: 10.1016/j.bone.2023.116955 -
International Journal of Pediatric... Jun 2016Mandibular distraction osteogenesis (MDO) is becoming increasingly more commonly used as in neonates and infants with upper airway obstruction secondary to micrognathia.... (Review)
Review
UNLABELLED
Mandibular distraction osteogenesis (MDO) is becoming increasingly more commonly used as in neonates and infants with upper airway obstruction secondary to micrognathia. A significant number of these children are dependent on nasoenteric feeding or gastrostomies after birth for adequate nutrition and often suffer from gastro-esophageal reflux (GERD).
OBJECTIVE
This analysis is a subset of a larger systematic review. The objective of this study is to determine the effects of MDO on feeding and GERD.
DATA SOURCES
The databases searched included PubMed, Embase, Scopus, Web of Knowledge and grey literature sources.
STUDY SELECTION
The inclusion criterion included studies in children with clinical evidence of micrognathia/Pierre Robin Sequence (PRS) who have failed conservative treatments, including both syndromic (sMicro) and non-syndromic (iPRS) patients. 21 studies relevant to feeding and 4 studies relevant to GERD outcomes were included. All studies included were case series and case reports.
RESULTS
MDO leads to a significant improvement in feeding, with 82% of children feeding exclusively orally after surgery. The overall percentage of children with iPRS who were feeding orally was 93.7% compared with only 72.9% in the sMicro group (p<0.004). A growth decline within the first six weeks after surgery was observed in multiple studies. Overall, out of 70 patients with pre-operative GERD, only four had evidence of GERD after surgery.
CONCLUSIONS
Considering the limitations of this systematic review, this study found that successful relief of airway obstruction by MDO leads to improvement of feeding and improvement in symptoms of GERD in children with upper airway obstruction secondary to micrognathia. Clinicians need to be aware of the risk of growth decline in the initial post-operative period.
Topics: Airway Obstruction; Child; Eating; Feeding Behavior; Gastroesophageal Reflux; Humans; Infant; Infant, Newborn; Mandible; Micrognathism; Osteogenesis, Distraction; Pierre Robin Syndrome; Postoperative Period; Treatment Outcome
PubMed: 27240511
DOI: 10.1016/j.ijporl.2016.03.033 -
International Journal of Oral and... Jun 2016Mandibular distraction osteogenesis (MDO) is increasingly used for neonates and infants with upper airway obstruction secondary to micrognathia. This systematic review... (Review)
Review
Mandibular distraction osteogenesis (MDO) is increasingly used for neonates and infants with upper airway obstruction secondary to micrognathia. This systematic review was conducted to determine the effectiveness of MDO in the treatment of airway obstruction. The databases searched included PubMed, Embase, Scopus, and grey literature sources. The inclusion criteria were applied to identify studies in children with clinical evidence of micrognathia/Pierre Robin sequence (PRS) who had failed conservative treatments, including both syndromic and non-syndromic patients. Overall 66 studies were included in this review. Primary MDO for the relief of upper airway obstruction was found to be successful at preventing tracheostomy in 95% of cases. Syndromic patients were found to have a four times greater odds of failure compared to those with isolated PRS. The most common causes of failure were previously undiagnosed lower airway obstruction, central apnoea, undiagnosed neurological abnormalities, and the presence of additional cardiovascular co-morbidities. MDO was less effective (81% success rate) at facilitating decannulation of tracheostomy-dependent children (P<0.0001). Failure in these patients was most commonly due to severe preoperative gastro-oesophageal reflux disease, swallowing dysfunction, and tracheostomy-related complications. The failure rate was higher when MDO was performed at an age of ≥24 months. More studies are needed to evaluate the long-term implications of MDO on facial development and long-term complications.
Topics: Airway Obstruction; Child; Humans; Infant; Infant, Newborn; Mandible; Micrognathism; Osteogenesis, Distraction; Pierre Robin Syndrome; Retrospective Studies; Treatment Outcome
PubMed: 26867668
DOI: 10.1016/j.ijom.2016.01.009 -
The Cleft Palate-craniofacial Journal :... Jan 2017Tongue-lip adhesion (TLA), mandibular distraction osteogenesis (MDO), and subperiosteal release of the floor of the mouth (SPRFM) are the most commonly performed... (Review)
Review
BACKGROUND
Tongue-lip adhesion (TLA), mandibular distraction osteogenesis (MDO), and subperiosteal release of the floor of the mouth (SPRFM) are the most commonly performed surgical procedures to treat severe airway obstruction in infants born with Pierre Robin sequence (PRS).
OBJECTIVES
To determine the rate of failure of each type of procedure, in terms of mortality and the need for tracheostomy, and to determine what proportion of infants have significant airway obstruction postoperatively as determined by polysomnography (PSG) and compare the data by procedure type.
METHOD
A comprehensive literature search (1981 through June 2015) was done of the National Library of Medicine database using PubMed. Extracted data included diagnosis, type of surgery, and outcome including mortality, need for postoperative tracheostomy and details of PSG. Persistence of significant airway obstruction was defined as an apnea-hypopnea index >15 events/h on PSG.
RESULTS
Both mortality rate and need for tracheostomy were low for all procedures. Many studies lacked sufficient detail to identify significant airway obstruction postoperatively. In studies with sufficient data, MDO was associated with the lowest percentage of significant airway obstruction postprocedure (3.6%) compared to 50% for infants who underwent TLA. Insufficient PSG data was available for patients who were treated with SPRFM.
CONCLUSIONS
There is a paucity of objective PSG data to definitively assess postoperative airway outcomes for PRS. MDO appears to be the most effective technique based on the available PSG data. Standardized use of PSG may lead to better identification and treatment of patients at risk for suboptimal airway outcomes postoperatively.
Topics: Airway Obstruction; Humans; Infant; Micrognathism; Osteogenesis, Distraction; Pierre Robin Syndrome; Polysomnography; Tracheostomy; Treatment Outcome
PubMed: 27414091
DOI: 10.1597/15-282 -
International Journal of Oral and... Apr 2020Maxillary hypoplasia in cleft lip and palate is a complex deformity. Despite surgical improvements, postoperative relapse persists. This systematic review was performed... (Meta-Analysis)
Meta-Analysis
Maxillary hypoplasia in cleft lip and palate is a complex deformity. Despite surgical improvements, postoperative relapse persists. This systematic review was performed to determine the mean horizontal relapse rates for the surgical techniques used to treat maxillary hypoplasia: Le Fort I osteotomy with rigid fixation, Le Fort I distraction osteogenesis, and anterior maxillary distraction osteogenesis. This study followed the PRISMA statement. The PubMed, Embase, Science Direct, and Web of Science databases were searched through to June 2018. Studies on non-growing cleft lip and palate patients who had undergone one of the three surgical procedures and who had postoperative horizontal maxillary changes assessed at >6 months post-surgery were included. Stata SE was used to estimate pooled means, heterogeneity, and publication bias. The search strategy identified 326 citations, from which 24 studies were selected. Relapse rates following Le Fort I osteotomy with rigid fixation, Le Fort I distraction osteogenesis, and anterior maxillary distraction osteogenesis were 20%, 12%, and 12%, respectively. Relapse rates with and without bone grafting were 19% and 66%, respectively. The relapse rate following distraction osteogenesis with internal distraction was lower than that with external distraction. Study limitations were heterogeneity, which was above moderate, the low number of high-quality studies, and unidirectional assessment of postoperative maxillary movement.
Topics: Cephalometry; Cleft Lip; Cleft Palate; Humans; Maxilla; Micrognathism; Osteogenesis, Distraction; Osteotomy, Le Fort; Recurrence; Treatment Outcome
PubMed: 31526673
DOI: 10.1016/j.ijom.2019.08.012