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The Annals of Otology, Rhinology, and... Jul 2024to investigate the ability of mandibular distraction osteogenesis (MDO) to avoid gastrostomy tube (G-tube). (Review)
Review
Pre-Operative Characteristics Helping to Avoid Gastrostomy Tube After Mandibular Distraction in Neonates With Pierre-Robin Sequence: A Institutional Case-Series and Review of the Literature.
OBJECTIVE
to investigate the ability of mandibular distraction osteogenesis (MDO) to avoid gastrostomy tube (G-tube).
DATA SOURCES
PubMed, EBSCOhost, Cochrane, and Embase.
REVIEW METHODS
We retrospectively reviewed the number of MDO cases performed at our institution for patients with Robin Sequence (RS) over the past 10 years. In our institutional review, patients were excluded if they had a G-tube already placed at the time of surgery. We also performed a systematic review of the literature. Articles were excluded if they did not detail feeding outcomes after MDO, or if MDO was performed on patients that did not have RS.
RESULTS
In our systematic review, 12 articles were included that comprised a total of 209 neonates with RS that underwent MDO. A total of 174 (83.3%) patients avoided a G-tube once MDO was performed. A total of 14 patients met the inclusion criteria at our institution. Of the 14 RS patients, 9 (64%) avoided having a G-tube placed and all (14/14) avoided tracheostomy. The average birth weight of patients avoiding a G-tube was 3.11 kg compared to 2.25 kg ( = .045) in the group requiring a G-tube. In the group avoiding a G-tube, the average weight at time of operation was 3.46 kg compared to 2.83 kg ( = .037) in the group requiring a G-tube.
CONCLUSION
MDO may be considered as a surgical option to prevent G-tube placement for neonates with non-syndromic RS who have difficulty with PO feeding but whose airway obstruction is not severe enough to require respiratory support. Based on our institutional experience, a minimum weight of 3.00 kg correlated with higher success rates of PO intake and avoiding a G-tube.
Topics: Humans; Pierre Robin Syndrome; Infant, Newborn; Gastrostomy; Osteogenesis, Distraction; Retrospective Studies; Mandible; Male; Female; Enteral Nutrition; Intubation, Gastrointestinal
PubMed: 38712740
DOI: 10.1177/00034894241249547 -
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 -
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 -
Advances in Neonatal Care : Official... Dec 2008Stickler syndrome is an autosomal dominant, genetic connective tissue disorder characterized by ocular, orofacial, auditory, and skeletal anomalies. The diagnosis of... (Review)
Review
Stickler syndrome is an autosomal dominant, genetic connective tissue disorder characterized by ocular, orofacial, auditory, and skeletal anomalies. The diagnosis of Stickler syndrome is often missed in the newborn period and diagnosed as simply Pierre Robin sequence. For these patients, complications and disabilities could be lessened by further analysis of the family history.Neonatal caregivers are responsible for the immediate care of the newborn with Pierre Robin sequence. The prudent healthcare profession will explore the origin of the condition to provide optimal comprehensive care to the infant and family. This article reviews the physical findings of Stickler syndrome including differentiation between similar syndromes. A systematic review of physical findings is included along with treatment options.
Topics: Connective Tissue Diseases; Craniofacial Abnormalities; Diagnosis, Differential; Humans; Infant; Infant, Newborn; Nursing Assessment; Pierre Robin Syndrome; Syndrome
PubMed: 19060576
DOI: 10.1097/01.ANC.0000342763.64240.69 -
The Journal of Craniofacial Surgery Mar 2018This study presents a systematic review of randomized controlled trials (RCTs) in cleft and craniofacial surgery. All studies reporting on RCTs in cleft and craniofacial... (Review)
Review
This study presents a systematic review of randomized controlled trials (RCTs) in cleft and craniofacial surgery. All studies reporting on RCTs in cleft and craniofacial surgery were identified on PubMed using the search terms "cleft," "velopharyngeal insufficiency," "velopharyngeal dysfunction," "nasoalveolar molding," "gingivoperiosteoplasty," "Pierre Robin sequence," "craniofacial," "craniosynostosis," "craniofacial microsomia," "hemifacial microsomia," "hypertelorism," "Le Fort," "monobloc," "distraction osteogenesis," "Treacher Collins," and "Goldenhar." Studies were excluded if they were not randomized, did not focus primarily on topics related to cleft or craniofacial surgery, included repeat publications of data, or were unavailable in English. Studies were evaluated on demographic and bibliometric data, study size, specific area of focus, and findings reported. Four hundred forty-seven unique studies were identified. One hundred eighty-three papers met inclusion criteria (115 cleft lip and palate, 65 craniofacial, and 3 spanning both disciplines). Sixty-six (36%) were dedicated to topics related to surgical techniques. There were no studies comparing current cleft lip or soft palate repair techniques and no studies on cleft rhinoplasty. The most frequently reported surgical topic was cleft palate. There were several studies on orthognathic techniques which compared distraction osteogenesis to traditional advancement. Most craniofacial operations, such as cranial vault remodeling and frontofacial advancement/distraction, were not represented. Several standard operations in cleft and craniofacial surgery are not supported by Level I evidence from randomized controlled trials. Our community should consider methods by which more RCTs can be performed, or redefine the acceptable standards of evidence to guide our clinical decisions.
Topics: Alveolar Bone Grafting; Bibliometrics; Cleft Lip; Cleft Palate; Humans; Orthognathic Surgical Procedures; Randomized Controlled Trials as Topic; Velopharyngeal Insufficiency
PubMed: 29084117
DOI: 10.1097/SCS.0000000000004100 -
Otolaryngology--head and Neck Surgery :... Jan 2012In this systematic review, the authors summarize the current evidence in the literature regarding diagnosis, treatment, and long-term outcomes in neonates with... (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
In this systematic review, the authors summarize the current evidence in the literature regarding diagnosis, treatment, and long-term outcomes in neonates with tongue-based airway obstruction (TBAO) and assess the level of evidence of included studies.
DATA SOURCES
The terms Pierre Robin syndrome/sequence, micrognathia, retrognathia, and cleft palate were combined with airway obstruction, treatment, tongue-lip plication, and osteogenesis distraction to perform an Ovid literature search, yielding 341 references. The authors excluded references containing patients with isolated choanal/nasal obstruction, patients older than 12 months, and expert opinion papers, yielding 126 articles.
REVIEW METHODS
The authors searched 3 electronic databases and reference lists of existing reviews from 1980 to October 2010 for articles pertaining to the diagnosis, treatment, and outcomes of TBAO. Reviewers assigned a level of evidence score based on Oxford's Centre for Evidence Based Medicine scoring system and recorded relevant information.
RESULTS
Most studies were case studies and single-center findings. The lack of standardization of diagnostic and treatment protocols and the heterogeneity of cohorts both within and between studies precluded a meta-analysis. There was little evidence beyond expert opinion and single-center evaluation regarding diagnosis, treatment, and long-term outcomes of neonates with TBAO.
CONCLUSIONS
The variability in the phenotype of the cohorts studied and the absence of standardized indications for intervention preclude deriving any definitive conclusions regarding diagnostic tools to evaluate this patient population, treatment choices, or long-term outcomes. A coordinated multicenter study with a standardized diagnostic and treatment algorithm is recommended to develop evidence for the diagnosis and treatment of neonates with TBAO.
Topics: Airway Obstruction; Humans; Infant, Newborn; Otorhinolaryngologic Surgical Procedures; Tongue
PubMed: 21926259
DOI: 10.1177/0194599811421598 -
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 -
Frontiers in Pediatrics 2018The use of long-term non-invasive ventilation (NIV) to treat sleep and breathing disorders in children has increased substantially in the last decade; however, less data...
BACKGROUND
The use of long-term non-invasive ventilation (NIV) to treat sleep and breathing disorders in children has increased substantially in the last decade; however, less data exist about its use in infants. Given that infants have distinct sleep and breathing patterns when compared to older children, the outcomes of infants on long-term NIV may differ as well. The aim of this study is to systematically review the use and outcomes of long-term NIV in infants.
METHODS
Ovid Medline, Ovid Embase, CINAHL (via EbscoHOST), PubMed, and Wiley Cochrane Library were systematically searched from January 1990 to July 2017. Studies on infants using long-term NIV outside of an acute care setting were included. Data were extracted on study design, population characteristics, and NIV outcomes.
RESULTS
A total of 327 studies were full-text reviewed, with final inclusion of 60. Studies were distributed across airway (40%), neuromuscular (28%), central nervous system (10%), cardio-respiratory (2%), and multiple (20%) disease categories. Of the 18 airway studies reporting on NIV outcomes, 13 (72%) reported improvements in respiratory parameters. Of the 12 neuromuscular studies exclusively on spinal muscular atrophy type 1 (SMA1), six (50%) reported decreased hospitalizations and nine (75%) reported on mortality outcomes. Risk of bias was moderate to serious, and quality of the evidence was low to very low for all studies. Most studies had an observational design with no control group, limiting the potential for a meta-analysis.
CONCLUSION
The outcomes reported in studies differed by the disease category being studied. Studies on airway conditions showed improvements in respiratory parameters for infants using NIV. Studies on neuromuscular disorder, which were almost exclusively on SMA1, reported decreased hospitalizations and prolonged survival. Overall, it appears that NIV is an effective long-term therapy for infants. However, the high risk of bias and low quality of the available evidence limited strong conclusions.
PubMed: 29484287
DOI: 10.3389/fped.2018.00013 -
Frontiers in Pediatrics 2021Cyproheptadine is a first-generation H1-antihistamine drug first that was distributed in the 1960s. While its orexigenic effect was observed early, cyproheptadine is...
Cyproheptadine is a first-generation H1-antihistamine drug first that was distributed in the 1960s. While its orexigenic effect was observed early, cyproheptadine is not yet authorized for this indication in all countries today. There is an increasing medical interest and demand for the orexigenic effect of cyproheptadine, especially in children with poor appetite. As cyproheptadine might be evaluated in future clinical trials, we wanted to assess its safety profile. Using the French national pharmacovigilance database, we retrospectively analyzed all pediatric and adult reports of adverse effects of cyproheptadine recorded since its first distribution in France. Next, we performed a systematic review of the literature of cyproheptadine adverse effects. Since 1985, 93 adverse effects were reported in the French pharmacovigilance database (adults 81.7%, children 18.3%); these were mainly neurological symptoms ( = 38, adults 71%, children 28.9%), and hepatic complications ( = 15, adults 86.7%, children 13.3%). In the literature, the most frequent adverse effect reported was drowsiness in adults or children, and five case reports noted liver complications in adults. We estimated the frequency of hepatic adverse effects at 0.27 to 1.4/1000, regardless of age. Cyproheptadine can be considered a safe drug. Mild neurological effects appear to be frequent, and hepatotoxicity is uncommon to rare. Randomized controlled trials are needed to evaluate the safety and efficacy of cyproheptadine before authorization for appetite stimulation, especially in young children as studies at this age are lacking. Possible hepatic complications should be monitored, as very rare cases of liver failure have been reported.
PubMed: 34676184
DOI: 10.3389/fped.2021.712413