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Orthodontics & Craniofacial Research Feb 2020To provide an overview on the prevalence and types of dental anomalies in patients with craniofacial microsomia (CFM). Eligibility criteria: Inclusion criteria were...
OBJECTIVE
To provide an overview on the prevalence and types of dental anomalies in patients with craniofacial microsomia (CFM). Eligibility criteria: Inclusion criteria were CFM and dental anomalies. The following data were extracted: number of patients, methodology, mean age, sex, affected side, severity of mandibular hypoplasia, dentition stage and dental anomalies.
INFORMATION SOURCES
Cochrane, EMBASE, PubMed, MEDLINE Ovid, Web of Science, CINAHL EBSCOhost and Google Scholar, searched until the 30 August 2019. Risk of bias: The quality was examined with the OCEBM Levels of Evidence.
INCLUDED STUDIES
In total, 13 papers were included: four retrospective cohort studies, four prospective cohort studies, four case-control studies and one case series. Synthesis of results: The studies reported information on dental agenesis, delayed dental development, tooth size anomalies, tooth morphology and other dental anomalies. Description of the effect: Dental anomalies are more often diagnosed in patients with CFM than in healthy controls and occur more often on the affected than on the non-affected side. Strengths and limitations of evidence: This is the first systematic review study on dental anomalies in CFM. However, most articles were of low quality.
INTERPRETATION
Dental anomalies are common in CFM, which might be linked to the development of CFM. The pathophysiology of CFM is not entirely clear, and further research is needed.
Topics: Anodontia; Goldenhar Syndrome; Humans; Prevalence; Prospective Studies; Retrospective Studies
PubMed: 31608577
DOI: 10.1111/ocr.12351 -
The Journal of Craniofacial Surgery 2019Hemifacial microsomia is the most common congenital disorder of the face after cleft lip and palate. While treatment of the bony abnormalities has been well documented,...
INTRODUCTION
Hemifacial microsomia is the most common congenital disorder of the face after cleft lip and palate. While treatment of the bony abnormalities has been well documented, less attention has been paid to correction of the soft tissue deformity. This study aimed to systematically review the literature addressing the techniques utilized to correct the soft tissue deformity in hemifacial microsomia.
MATERIALS AND METHODS
A comprehensive review of peer-reviewed literature regarding the management of soft tissue reconstruction in patients with hemifacial microsomia was performed according to the Preferred Reporting Items for Systematic Reviews and Meta-analyses statement. Odds ratio was used to assess differences in complication rates between reconstructions performed with free tissue transfer and fat grafting.
RESULTS
The literature search yielded 38 articles that were fully reviewed for data extraction. Articles were grouped into one of 5 reconstructive modalities-pedicled flap, microvascular free tissue transfer, structural fat grafting, alloplastic implant, and functional reconstruction. Microvascular free tissue transfer had a complication rate of 27.1%, while structural fat grafting had a complication rate of 4.2% (odds ratio 6.7, P = 0.0003).
CONCLUSIONS
Microvascular free tissue transfer likely provides greater volume than fat grafting at the cost of a higher complication rate. Mild to moderate soft tissue deformity can be corrected with serial fat grafting. However, severe soft tissue deformity is better corrected with microvascular free tissue transfer. When performing free tissue transfer, one should be prepared for recipient vessel anomalies. To provide adequate augmentation with isolated fat grafting, multiple sessions are likely required.
Topics: Adipose Tissue; Goldenhar Syndrome; Humans; Prostheses and Implants; Plastic Surgery Procedures; Surgical Flaps
PubMed: 30817535
DOI: 10.1097/SCS.0000000000005320 -
Journal of Cranio-maxillo-facial... Sep 2018Mandibular reconstruction in craniofacial microsomia (CFM) has been described and reviewed at length although final results are not always (aesthetically) satisfactory...
INTRODUCTION
Mandibular reconstruction in craniofacial microsomia (CFM) has been described and reviewed at length although final results are not always (aesthetically) satisfactory due to maxillo-mandibular asymmetry, for which optimal correction techniques remain unclear. The aim of this systematic review is to provide an overview of the surgical options for maxillary correction in patients with unilateral CFM.
MATERIAL AND METHODS
MEDLINE/Pubmed, Embase, Cochrane and Web of Science databases were searched up to April 15, 2017. Inclusion criteria were: studies reporting patients with unilateral CFM (n > 4) who had maxillary correction (with/without simultaneous mandibular correction) with a minimal follow-up of 6 months. The outcome measures included type of treatment (including preceding facial procedures), type and severity of mandibular deformity (by Pruzansky-Kaban system: Types I/IIa/IIb/III), asymmetry analysis method, outcome (i.e. occlusion, canting, stability, esthetic result, facial symmetry), complications and additional treatment needed.
RESULTS
Nine studies met the inclusion criteria. Analysis showed that Le Fort I + mandibular distraction osteogenesis (LeFort + MDO) and BiMaxillary osteotomy (BiMax) were used for treatment, as single or multiple-stage procedures. All studies reported aesthetic and functional improvement.
CONCLUSION
Types I/IIa benefited from LeFort + MDO; Type IIb from LeFort + MDO or BiMax; and Type III from BiMax (with 50% of cases having preceding mandibular procedures, including patient-fitted prosthesis) at a mean age of 20.2 years. Four studies recommended additional (esthetic) procedures.
Topics: Esthetics, Dental; Facial Asymmetry; Goldenhar Syndrome; Humans; Mandibular Reconstruction; Maxillofacial Development; Osteogenesis, Distraction; Osteotomy, Le Fort
PubMed: 29907434
DOI: 10.1016/j.jcms.2018.05.043 -
The Journal of Craniofacial Surgery Mar 2018Accessory auricles are rare congenital abnormalities embryologically derived from the first branchial arch. Presentation may be variable with locations grouped into... (Review)
Review
Accessory auricles are rare congenital abnormalities embryologically derived from the first branchial arch. Presentation may be variable with locations grouped into anatomical zones based on the frequency of location found in the literature. This study reviewed the papers between 1981 and 2017. Findings included an association with syndromes including Goldenhar, VACTERL, Treacher-Collins, Townes-Brocks, and Wolf-Hirschhorn. Based on histological and embryological evidence, the term "accessory auricle" is best used as an umbrella term to define this difference. Finally, indications are provided for imaging and laboratory investigations to guide the physician treating patients with accessory auricles.
Topics: Abnormalities, Multiple; Anal Canal; Anus, Imperforate; Branchial Region; Ear, External; Esophagus; Goldenhar Syndrome; Hearing Loss, Sensorineural; Heart Defects, Congenital; Humans; Kidney; Limb Deformities, Congenital; Mandibulofacial Dysostosis; Spine; Thumb; Trachea
PubMed: 29239919
DOI: 10.1097/SCS.0000000000004201 -
International Journal of Oral and... Jan 2018Extracraniofacial anomalies, including central nervous system (CNS) anomalies, may occur in craniofacial microsomia (CFM). This systematic review was performed to... (Review)
Review
Extracraniofacial anomalies, including central nervous system (CNS) anomalies, may occur in craniofacial microsomia (CFM). This systematic review was performed to provide an overview of the literature on the prevalence and types of CNS anomalies and developmental disorders in CFM, in order to improve the recognition and possible treatment of these anomalies. A systematic search was conducted and data on the number of patients, patient characteristics, type and prevalence of CNS anomalies or developmental delay, and correlations between CFM and CNS anomalies were extracted. Sixteen papers were included; 11 of these described developmental disorders. The most common reported anomalies were neural tube defects, corpus callosum agenesis or hypoplasia, intracranial lipoma, Arnold-Chiari malformations, hydrocephaly, ventriculomegaly, and cerebral hypoplasia. The prevalence of CNS anomalies in CFM varied from 2% to 69%. The prevalence of developmental disorders, such as intellectual disability, language or speech developmental delay, and neuropsychomotor delay, varied from 8% to 73%. This study suggests that CNS anomalies and developmental disorders are seen in a substantial proportion of patients with CFM. Further research should focus on determining which features of CFM are correlated with CNS anomalies to allow adequate screening and timely care.
Topics: Abnormalities, Multiple; Child; Child, Preschool; Developmental Disabilities; Goldenhar Syndrome; Humans; Infant; Infant, Newborn; Nervous System Malformations; Prevalence
PubMed: 28736116
DOI: 10.1016/j.ijom.2017.06.009 -
International Journal of Oral and... Oct 2017Craniofacial microsomia (CFM) is characterized by a heterogeneous underdevelopment of the facial structures arising from the first and second branchial arches, but... (Review)
Review
Craniofacial microsomia (CFM) is characterized by a heterogeneous underdevelopment of the facial structures arising from the first and second branchial arches, but extracraniofacial malformations such as vertebral anomalies also occur. This systematic review provides an overview of the literature on the types and prevalence of vertebral anomalies found in patients with CFM. A systematic search was conducted. Data on the number of patients, patient characteristics, types and prevalence of vertebral anomalies, and other associations between CFM and vertebral anomalies were extracted from the articles identified. Thirty-one articles were included. Seventeen articles described both the prevalence and types of vertebral anomalies in CFM, five articles described solely the types of vertebral anomalies in CFM, and nine articles reported solely the prevalence of vertebral anomalies in CFM. The vertebral anomalies most often reported in CFM are hemivertebrae, block vertebrae, scoliosis/kyphoscoliosis, and spina bifida. These anomalies are mostly present in the cervical and thoracic spine and ribs. The reported prevalence of vertebral anomalies in CFM varies from 8% to 79%. To diagnose vertebral anomalies early in patients with CFM, further research should focus on determining which patients with CFM are at risk of vertebral anomalies.
Topics: Abnormalities, Multiple; Goldenhar Syndrome; Humans; Spine
PubMed: 28669484
DOI: 10.1016/j.ijom.2017.04.025 -
Plastic and Reconstructive Surgery Jan 2016No reviews or guidelines are available on evidence-based treatment for the multidisciplinary approach in Treacher Collins syndrome. The authors' aim is to provide an... (Review)
Review
BACKGROUND
No reviews or guidelines are available on evidence-based treatment for the multidisciplinary approach in Treacher Collins syndrome. The authors' aim is to provide an evidence-based review of multidisciplinary treatment of Treacher Collins syndrome based on levels of evidence and supported with graded recommendations.
METHODS
A systematic search was performed by means of the PubMed, Web-of-Science, Embase, and Cochrane Central databases (1985 to January of 2014). Included were clinical studies (with five or more Treacher Collins syndrome patients) related to therapy, diagnosis, or risk of concomitant diseases. Level of evidence of the selected articles was rated according to the American Society of Plastic Surgeons evidence-based clinical practice guidelines. After two panelists had reviewed each abstract separately, a consensus method was used to solve any disagreements concerning article inclusion.
RESULTS
Of the 2433 identified articles, 63 studies (Level of Evidence II through V) were included. Conclusions and recommendations were extracted consecutively for the following items: upper airway; ear, hearing, and speech; the eye, eyelashes, and lacrimal system; growth, feeding, and swallowing; the nose; psychosocial factors; and craniofacial reconstruction.
CONCLUSIONS
In this systematic review, current evidence for the multidisciplinary treatment of Treacher Collins syndrome is provided, recommendations for treatment are made, and a proposed algorithm for treatment is presented. Although some topics are well supported, others, especially ocular, nasal, speech, feeding, and swallowing problems, lack sufficient evidence. In addition, craniofacial surgical reconstruction lacks a sufficient level of evidence to provide a sound basis for a full treatment protocol. Despite the rarity of the syndrome, more research is needed to compare outcomes of several surgical treatments, especially in orbitozygomatic/maxillary regions.
Topics: Combined Modality Therapy; Disability Evaluation; Evidence-Based Medicine; Female; Humans; Male; Mandibulofacial Dysostosis; Netherlands; Patient Care Planning; Practice Guidelines as Topic; Prognosis; Rare Diseases; Severity of Illness Index; Treatment Outcome
PubMed: 26710023
DOI: 10.1097/PRS.0000000000001896 -
International Journal of Oral and... Jun 2015Patients with craniofacial microsomia are at higher risk of developing obstructive sleep apnoea (OSA), as described in the previous article entitled "Obstructive sleep... (Review)
Review
Patients with craniofacial microsomia are at higher risk of developing obstructive sleep apnoea (OSA), as described in the previous article entitled "Obstructive sleep apnoea in craniofacial microsomia: a systematic review". These patients are also more likely to develop feeding difficulties. The present systematic review provides an overview of the literature on the prevalence, treatment, and follow-up of feeding difficulties in children with craniofacial microsomia (CFM). A search was performed in PubMed, Embase, Cochrane Library, and Web of Science for articles on CFM and feeding difficulties. The following data were extracted from the articles: number of patients, patient characteristics, presence of feeding difficulties, and the treatment and outcomes of feeding difficulties. Eight articles on CFM and feeding difficulties were included, two of which reported the prevalence of feeding difficulties (range 42-83%). Treatment mostly consisted of tube feeding. No information regarding follow-up was found in these articles. According to the literature, feeding difficulties are related to CFM. However, as there have been no prospective studies and few studies have presented objective measurements, no definitive conclusions can be drawn. Prospective studies are needed to determine the prevalence of feeding difficulties in patients with CFM.
Topics: Child; Child, Preschool; Feeding and Eating Disorders of Childhood; Goldenhar Syndrome; Humans; Infant; Infant, Newborn; Sleep Apnea, Obstructive
PubMed: 25771070
DOI: 10.1016/j.ijom.2015.02.014 -
International Journal of Oral and... May 2015Children with craniofacial microsomia (CFM) are at risk of obstructive sleep apnoea (OSA). This systematic review provides an overview of the literature on the... (Review)
Review
Children with craniofacial microsomia (CFM) are at risk of obstructive sleep apnoea (OSA). This systematic review provides an overview of the literature on the prevalence of OSA in children with CFM. A search was performed in PubMed, Embase, Cochrane Library, and Web of Science for articles on CFM and OSA. The following data were extracted from the articles: number of patients, patient characteristics, presence of OSA, polysomnography outcomes, and the treatments and outcomes of OSA. We included 16 articles on CFM and OSA, four of which reported the prevalence of OSA (range 7-67%). Surgical treatment was more often described in these patients than conservative treatment. According to the literature, OSA is related to CFM. However, as there have been no prospective studies and few studies have presented objective measurements, no definitive conclusions can be drawn. Prospective studies are needed to determine the prevalence of OSA in patients with CFM.
Topics: Child; Goldenhar Syndrome; Humans; Sleep Apnea, Obstructive
PubMed: 25769220
DOI: 10.1016/j.ijom.2015.01.023 -
International Journal of Oral and... Mar 2014The 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... (Review)
Review
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.
Topics: Child; Facial Asymmetry; Goldenhar Syndrome; Humans; Mandible; Mandibular Reconstruction
PubMed: 24332589
DOI: 10.1016/j.ijom.2013.11.001