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International Journal of Pediatric... Nov 2020To evaluate outcomes of injection laryngoplasty (IL) and endoscopic surgical repair for the treatment of type 1 laryngeal clefts (LC1) and to determine the most common... (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
To evaluate outcomes of injection laryngoplasty (IL) and endoscopic surgical repair for the treatment of type 1 laryngeal clefts (LC1) and to determine the most common presentations of LC1.
METHODS
Primary studies were searched for in PubMed, Scopus, OVID, and Cochrane Library using search terms laryngeal cleft and laryngotracheoesophageal cleft for articles published from database inception through August 2019. The search identified all articles pertaining to the symptomatology and management of LC1. Meta-analysis was performed on presenting symptoms and improvement of clinical symptoms and swallow function.
RESULTS
Twenty-four studies, comprising 713 patients with a mean age of 33.7 months (range 0-168 months), were included. The most common presenting symptoms were aspiration (80%, CI [55%-96%]), dysphagia (61%, CI [47%-74%]), and choking (57%, CI [37%-76%]). Gastroesophageal reflux disease (GERD) was the most common comorbidity (68%, CI [51%-83%]) and premature birth comprised 27% [21%-32%] of the patients. Overall, 38% of patients received IL as a primary therapy. Prior to injection, 91% [87%-94%] of patients aspirated on swallow evaluations, and 62% [55%-68%] aspirated post-injection. At an average follow-up time of 6.8 months, 90% [70%-100%] of parents reported symptom improvement. An additional 54% of patients underwent endoscopic surgical repair as primary treatment. The rate of aspiration decreased from 73% [43%-94%] to 28% [13%-46%] after repair. At a mean follow-up of 14.2 months, 80% [67%-91%] reported symptom improvement.
CONCLUSION
A high level of clinical suspicion is necessary to detect LC1 since its most common presenting symptoms overlap with other diseases commonly found in this patient population. Both IL and formal surgical repair were effective in managing LC1.
Topics: Adolescent; Child; Child, Preschool; Congenital Abnormalities; Deglutition Disorders; Humans; Infant; Infant, Newborn; Laryngoplasty; Larynx; Retrospective Studies
PubMed: 33152963
DOI: 10.1016/j.ijporl.2020.110370 -
The British Journal of Dermatology Sep 2016Psoriasis is a chronic inflammatory disorder with significant physical and psychological sequelae. The majority of individuals experience disease onset in early adult... (Meta-Analysis)
Meta-Analysis Review
Psoriasis is a chronic inflammatory disorder with significant physical and psychological sequelae. The majority of individuals experience disease onset in early adult life - for women this often occurs during their reproductive years. While some autoimmune diseases have been shown to affect pregnancy outcomes adversely, such a relationship has not been well studied in psoriasis. We searched PubMed, Embase and the Cochrane database for published articles examining psoriasis and adverse pregnancy outcomes, and included observational studies and clinical trials evaluating direct measures of maternal and fetal morbidity and mortality. Four of the nine included articles reported a statistically significant increase in the risk of at least one outcome, including spontaneous abortion, caesarean delivery, low birth weight, macrosomia, large-for-gestational age, and a composite outcome consisting of both prematurity and low birth weight. However, these associations were not always consistent across studies. Overall, there was no clear evidence of increased adverse outcomes in pregnant women with psoriasis.
Topics: Abortion, Spontaneous; Cesarean Section; Congenital Abnormalities; Epidemiologic Studies; Female; Fetal Macrosomia; Humans; Infant, Low Birth Weight; Observational Studies as Topic; Pregnancy; Pregnancy Complications; Pregnancy Outcome; Psoriasis
PubMed: 26991866
DOI: 10.1111/bjd.14547 -
Human Reproduction Update 2013It has been 10 years since we carried out a systematic search of the literature on birth defect risk in infants born following assisted reproductive technology (ART)... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
It has been 10 years since we carried out a systematic search of the literature on birth defect risk in infants born following assisted reproductive technology (ART) compared with non-ART infants. Because of changes to ART practice since that review and the publication of more studies the objective of this review was to include these more recent studies to estimate birth defect risk after ART and to examine birth defect risk separately in ART singletons and multiples.
METHODS
We searched Medline, Embase and Current Contents databases (1978-2012). We used the same data extraction sheet and questionnaire we had used previously with the addition of a quality score to the questionnaire. Pooled relative risk (RR) estimates were calculated using a random effects model. All data were analysed using Comprehensive Meta-Analysis V2.
RESULTS
There were 45 cohort studies included in this review. ART infants (n = 92 671) had a higher risk of birth defects [RR 1.32, 95% confidence interval (CI) 1.24-1.42] compared with naturally conceived infants (n = 3 870 760). The risk further increased when data were restricted to major birth defects (RR 1.42, 95% CI 1.29-1.56) or singletons only (RR 1.36, 95% CI 1.30-1.43). The results for ART multiples were less clear. When all data for multiples were pooled the RR estimate was 1.11 (95% CI 0.98-1.26) but this increased to 1.26 (0.99-1.60) when the analysis was restricted to studies of ART twins where some adjustment was made for differences in zygosity distribution between ART and non-ART multiples.
CONCLUSIONS
Birth defects remain more common in ART infants. Further research is required to examine risks for important subgroups of ART exposure.
Topics: Congenital Abnormalities; Humans; Infant; Reproductive Techniques, Assisted; Risk Factors; Twins
PubMed: 23449641
DOI: 10.1093/humupd/dmt006 -
Phlebology Jul 2017Objectives Parkes Weber syndrome is a congenital vascular malformation which consists of capillary malformation, venous malformation, lymphatic malformation, and... (Review)
Review
Objectives Parkes Weber syndrome is a congenital vascular malformation which consists of capillary malformation, venous malformation, lymphatic malformation, and arteriovenous malformation. Although Parkes Weber syndrome is a clinically distinctive entity with serious complications, it is still frequently misdiagnosed as Klippel-Trenaunay syndrome that consists of the triad capillary malformation, venous malformation, and lymphatic malformation. Methods We performed a systematic review investigating clinical, diagnostic, and treatment modalities of Parkes Weber syndrome (PubMed/MEDLINE, Embase, and Cochrane databases). Thirty-six publications (48 patients) fulfilled the eligibility criteria. Results The median age of patients was 23 years (IQR, 8-32), and 24 (50.0%) were males. Lower extremity was affected in 42 (87.5%) and upper extremity in 6 (12.5%) patients; 15 (31.3%) patients developed high-output heart failure; 12 (25.0%) patients had chronic venous ulcerations, whereas 4 (8.3%) manifested distal arterial ischemia. The spinal arteriovenous malformations were reported in six (12.5%) patients and coexistence of aneurysmatic disease in five (10.4%) patients. The most frequently utilized invasive treatments were embotherapy followed by amputation and surgical arteriovenous malformation resection, and occasionally stent-graft implantation. All modalities showed clinical improvement. However, long follow-up and outcome remained unclear. Conclusion A diagnosis of Parkes Weber syndrome should be made on the presence of capillary malformation, venous malformation, lymphatic malformation, and arteriovenous malformation (as main defect) in overgrowth extremity. Arteriovenous malformation presents the criterion for distinguishing Parkes Weber syndrome from Klippel-Trenaunay syndrome, which is substantial for treatment strategy. The primary management goal should be patient's quality of life improvement and complication reduction. Embolization alone/combined with surgical resection targeting occlusion or removal of arteriovenous malformation "nidus" reliably leads to clinical improvement.
Topics: Adolescent; Adult; Arteriovenous Malformations; Child; Diagnosis, Differential; Female; Humans; Klippel-Trenaunay-Weber Syndrome; Lymphatic Abnormalities; Male; Pregnancy; Pregnancy Complications, Cardiovascular; Quality of Life; Risk; Sturge-Weber Syndrome; Young Adult
PubMed: 27511883
DOI: 10.1177/0268355516664212 -
Ultrasound in Obstetrics & Gynecology :... Apr 2017The majority of studies on fetal ventriculomegaly have focused on the perinatal and long-term outcomes in fetuses with an antenatal diagnosis of bilateral... (Meta-Analysis)
Meta-Analysis Review
OBJECTIVES
The majority of studies on fetal ventriculomegaly have focused on the perinatal and long-term outcomes in fetuses with an antenatal diagnosis of bilateral ventriculomegaly. The aim of this study was to undertake a systematic review and meta-analysis to quantify the perinatal and long-term outcomes in fetuses diagnosed in the second or third trimester of pregnancy with isolated unilateral ventriculomegaly.
METHODS
MEDLINE, EMBASE and The Cochrane Library were searched electronically. Outcomes investigated included incidence of aneuploidy, congenital infection, progression of ventriculomegaly, associated brain and extracerebral abnormalities in the apparently isolated cases and neurodevelopmental delay in both apparently and truly isolated cases. Sensitivity analysis was performed according to whether the ventriculomegaly was mild/moderate (atrial width < 15 mm) or severe (atrial width ≥ 15 mm). Reference lists within relevant articles and reviews were hand-searched for additional reports. Cohort and case-control studies were included. Meta-analysis of proportions was used, and between-study heterogeneity was assessed using the I test.
RESULTS
The search yielded 2053 citations. The full text was retrieved for 202, and 11 studies were included in the systematic review. In fetuses with apparently isolated unilateral ventriculomegaly, no chromosomal abnormalities were identified and the pooled prevalence of congenital infection was 8.2% (95% CI, 3.6-14.5%). The pooled prevalence of additional brain abnormalities detected prenatally and postnatally by magnetic resonance imaging was 5.1% (95% CI, 0.2-16.1%) and 6.4% (95% CI, 0.3-19.4%), respectively. The pooled prevalence of abnormal neurodevelopment was 5.9% (95% CI, 2.2-11.2%) in apparently isolated cases with an atrial width of < 15 mm, and it was 7.0% (95% CI, 3.2-12.2%) in fetuses with truly isolated unilateral ventriculomegaly. Most cases with apparently isolated ventriculomegaly were classified as mild/moderate (93.5%) and therefore the outcomes in this group were similar to those in the whole cohort of apparently isolated ventriculomegaly.
CONCLUSIONS
The prevalence of aneuploidy, congenital infection and neurodevelopmental delay in fetuses with a prenatal diagnosis of isolated unilateral ventriculomegaly is likely to be low. Copyright © 2016 ISUOG. Published by John Wiley & Sons Ltd.
Topics: Aneuploidy; Congenital Abnormalities; Female; Fetal Diseases; Humans; Hydrocephalus; Infections; Neurodevelopmental Disorders; Perinatal Care; Pregnancy; Pregnancy Outcome; Pregnancy Trimester, Second; Pregnancy Trimester, Third; Ultrasonography, Prenatal
PubMed: 27091707
DOI: 10.1002/uog.15943 -
Obstetrical & Gynecological Survey Dec 2016The human vertebral column has a stable number of vertebrae and ribs, which is presumably the result of evolutionary selection. An association between an abnormal... (Review)
Review
IMPORTANCE
The human vertebral column has a stable number of vertebrae and ribs, which is presumably the result of evolutionary selection. An association between an abnormal vertebral pattern, especially in the cervical region, and congenital anomalies or adverse fetal outcome has been reported.
OBJECTIVE
The aim of this study was to review the current literature concerning an abnormal vertebral pattern and prevalence of cervical ribs in healthy subjects and in subjects with adverse outcome.
EVIDENCE ACQUISITION
Scientific databases were searched systematically. Studies assessing the number of vertebrae and/or ribs were included, and data concerning anomalies and outcome were extracted.
RESULTS
Thirty-nine studies including 75,018 healthy subjects and 6130 subjects with structural or chromosomal anomalies or adverse outcome were selected. The majority of these studies focused on the prevalence of cervical ribs. The prevalence of cervical ribs was considerably higher in fetuses with adverse outcome, including aneuploidies, compared with healthy individuals in the vast majority of studies. Studies suggest an association between cervical ribs and other structural anomalies.
CONCLUSIONS
These results demonstrate that detailed assessment of the fetal vertebral column, especially of the cervicothoracic region, could provide valuable information regarding fetal and neonatal prognosis. Based on the available evidence, the application of 3-dimensional (3D) ultrasound to assess the vertebral column and ribs, in particular the cervical region, warrants further research.
RELEVANCE
Prenatal assessment of the ribs and vertebral pattern by 3D ultrasound, which is currently not routinely performed, might be useful in the assessment of the fetus, because this can predict fetal and neonatal outcome in some cases.
Topics: Cervical Rib; Female; Humans; Infant, Newborn; Musculoskeletal Abnormalities; Patient Care Management; Pregnancy; Pregnancy Outcome; Prognosis; Ultrasonography, Prenatal
PubMed: 28005137
DOI: 10.1097/OGX.0000000000000388 -
Journal of Obstetrics and Gynaecology... Apr 2013Fluoxetine is the selective serotonin reuptake inhibitor (SSRI) with the longest clinical use. Published reports regarding its fetal safety are contradictory. We aimed... (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
Fluoxetine is the selective serotonin reuptake inhibitor (SSRI) with the longest clinical use. Published reports regarding its fetal safety are contradictory. We aimed to establish the fetal safety of the drug.
METHODS
We performed a systematic review of the literature, searching PubMed, Medline, and Embase from inception to August 31, 2012, for cohort and case-control studies in which women were exposed to fluoxetine during the first trimester and compared outcomes with those of unexposed control subjects.
RESULTS
Twenty-one studies met the inclusion criteria. The odds ratio for major malformations associated with maternal fluoxetine use in cohort studies was 1.12 (95% CI 0.98 to 1.28). The studies included were homogeneous. Fifteen cohort studies evaluated cardiac malformations and yielded an overall odds ratio of 1.6 (95% CI 1.31 to 1.95). These studies also were homogeneous. In contrast, two case-control studies assessing cardiac malformations yielded a combined odds ratio of 0.63 (95% CI 0.39 to 1.03).
CONCLUSION
The apparent increased risk of fetal cardiac malformations associated with maternal use of fluoxetine has recently been shown also in depressed women who deferred SSRI therapy in pregnancy, and therefore most probably reflects an ascertainment bias. Overall, women who are treated with fluoxetine during the first trimester of pregnancy do not appear to have an increased risk of major fetal malformations.
Topics: Abnormalities, Drug-Induced; Case-Control Studies; Cohort Studies; Depression; Female; Fluoxetine; Heart Defects, Congenital; Humans; MEDLINE; Odds Ratio; Pregnancy; Pregnancy Complications; Pregnancy Trimester, First; Selective Serotonin Reuptake Inhibitors
PubMed: 23660045
DOI: 10.1016/S1701-2163(15)30965-8 -
Journal of Pediatric and Adolescent... Dec 2014The nonobstructive group of anatomic variants involving the reproductive tract includes vaginal agenesis as well as the congenital anomalies of the vagina and uterus,... (Review)
Review
BACKGROUND
The nonobstructive group of anatomic variants involving the reproductive tract includes vaginal agenesis as well as the congenital anomalies of the vagina and uterus, occurring without pain during the pubertal years.
OBJECTIVE
The objective is to discuss the non-obstructive morphologic variations in anatomy of the uterus and vagina.
DESIGN
Systematic review using the GRADE system.
RESULTS
These congenital anomalies are not associated with abnormalities of the external genitalia and therefore may be missed on routine physical examination. When these anomalies do cause symptoms they may be as minor as difficulty with menstrual hygiene or more significant such as primary amenorrhea, dyspareunia, recurrent pregnancy loss, and reproductive complications.
CONCLUSIONS
Women with non-obstructive reproductive tract anomalies present at various ages due to the asymptomatic nature or late symptom onset of certain conditions. An MRI is the gold standard in evaluation of such conditions to aid in confirming the müllerian variant. Each condition requires careful counseling because obstetric and gynecologic risks and consequences may differ. Treatment is individualized in cases of uterovaginal agenesis with both nonsurgical and surgical options available for neovagina creation. In cases of uterine or vaginal septae, the treatment timing may vary depending on patient history. Finally, in cases of non-obstructive communicating uterine horns, the risk of ectopic pregnancy is high in the remnant horn. Should a pregnancy occur in this small underdeveloped horn, therefore, excision is recommended.
Topics: Adult; Amenorrhea; Congenital Abnormalities; Dilatation; Dyspareunia; Female; Humans; Infertility, Female; Mullerian Ducts; Pregnancy; Pregnancy Complications; Surgically-Created Structures; Urogenital Abnormalities; Uterus; Vagina
PubMed: 25438707
DOI: 10.1016/j.jpag.2014.07.001 -
Pediatrics Jun 2015Ankyloglossia is a congenital condition characterized by an abnormally short, thickened, or tight lingual frenulum that restricts tongue mobility. The objective of this... (Review)
Review
OBJECTIVE
Ankyloglossia is a congenital condition characterized by an abnormally short, thickened, or tight lingual frenulum that restricts tongue mobility. The objective of this study was to systematically review literature on surgical and nonsurgical treatments for infants with ankyloglossia.
METHODS
Medline, PsycINFO, Cumulative Index of Nursing and Allied Health Literature, and Embase were searched up to August 2014. Two reviewers independently assessed studies against predetermined inclusion/exclusion criteria. Two reviewers independently extracted data regarding participant and intervention characteristics and outcomes and assigned quality and strength-of-evidence ratings.
RESULTS
Twenty-nine studies reported breastfeeding effectiveness outcomes (5 randomized controlled trials [RCTs], 1 retrospective cohort, and 23 case series). Four RCTs reported improvements in breastfeeding efficacy by using either maternally reported or observer ratings, whereas 2 RCTs found no improvement with observer ratings. Although mothers consistently reported improved effectiveness after frenotomy, outcome measures were heterogeneous and short-term. Based on current literature, the strength of the evidence (confidence in the estimate of effect) for this issue is low. We included comparative studies published in English. The evidence base is limited, consisting of small studies, short-term outcomes, and little information to characterize participants adequately. No studies addressed nonsurgical interventions, longer-term breastfeeding or growth outcomes, or surgical intervention compared with other approaches to improve breastfeeding, such as lactation consultation.
CONCLUSIONS
A small body of evidence suggests that frenotomy may be associated with mother-reported improvements in breastfeeding, and potentially in nipple pain, but with small, short-term studies with inconsistent methodology, strength of the evidence is low to insufficient.
Topics: Ankyloglossia; Breast Feeding; Child; Humans; Mouth Abnormalities; Treatment Outcome
PubMed: 25941303
DOI: 10.1542/peds.2015-0658 -
Clinical Oral Investigations Aug 2022To evaluate the association between orofacial clefts (OFC) and tooth abnormalities (TA). (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
To evaluate the association between orofacial clefts (OFC) and tooth abnormalities (TA).
METHODS
We searched PubMed, Scopus, Web of Science, Cochrane Library, LILACS, and BBO, and in the gray literature and selected observational studies that evaluated the association between TA and OFC. The risk of bias was analyzed using the Newcastle-Ottawa Scale. A random-effects meta-analysis was performed comparing the presence and absence of OFC, cleft type-cleft palate (CP) and cleft lip with or without palate (CL/P)-and cleft laterality-unilateral and bilateral. The certainty of evidence was evaluated using the GRADE approach.
RESULTS
A total of 99 studies were included in the qualitative analysis, and 37 were included in the meta-analysis. Only four studies were classified as low risk of bias. Significant associations were observed between the presence of OFC and tooth agenesis (OR = 19.46; 95%CI = 4.99-75.96), supernumerary teeth (OR = 4.04; 95%CI = 1.26-12.99), developmental defects of enamel (OR = 3.15; 95%CI = 1.28-7.80), microdontia (OR = 15.57; 95%CI = 1.06-228.51), and taurodontism (OR = 1.74; 95%CI = 1.74-2.86). Individuals with CP had a lower frequency of supernumerary teeth (OR = 0.22; 95%CI = 0.08-0.64), peg-shaped tooth (OR = 0.31; 95%CI = 0.12-0.80), and morphological TA (OR = 0.13; 95%CI = 0.04-0.45) than individuals with CL/P. No TA was significantly associated with cleft laterality (p > 0.05). The quality of the evidence was very low in all analyses.
CONCLUSION
Individuals with OFC had a higher frequency of TA than those without OFC. Individuals with CP had a lower frequency of TA than individuals with CL/P. No TA was associated to cleft laterality.
CLINICAL RELEVANCE
Help to identify the treatment needs of individuals affected by OFC, improving the services provided to this population.
Topics: Cleft Lip; Cleft Palate; Humans; Tooth Abnormalities; Tooth, Supernumerary
PubMed: 35729285
DOI: 10.1007/s00784-022-04540-8