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Reviews in Endocrine & Metabolic... Dec 2023Fibrous dysplasia (FD) is a rare skeletal disorder in which normal bone is replaced by a fibro-osseous tissue, resulting in possible deformities and fractures. The aim... (Meta-Analysis)
Meta-Analysis Review
Fibrous dysplasia (FD) is a rare skeletal disorder in which normal bone is replaced by a fibro-osseous tissue, resulting in possible deformities and fractures. The aim of this systematic review and meta-analysis was to synthesize the available evidence on the use of antiresorptive drugs in FD in terms of changes in bone turnover markers (BTMs), bone mineral density (BMD), and reducing pain. Three databases were searched in October 2022, with an update in July 2023. Of the 1037 studies identified, 21 were retained after eligibility assessment. A random-effects model was used to calculate global effect size and the corresponding standard error. Pamidronate and Denosumab were the most reported drugs in a total of 374 patients assessed. The initiation of treatments was accompanied by an average reduction of 40.5% [CI -51.6, -29.3] in the bone resorption parameters, and 22.0% [CI -31.9, -12.1] in the parameters of bone formation after 6-12 months. BMD was increased in both FD lesions and in the unaffected skeleton. Pain was reduced by 32.7% [CI -52.7, -12.6] after 6-12 months of treatment, and by 44.5% [CI -65.3, -23.6] after a mean 41.2 months of follow-up. The variation in pain was highly correlated to variation in bone resorption (R = 0.08, p < 0.0001) and formation parameters (R = 0.17, p < 0.0001). This study supports the overall efficacy of antiresorptive therapies in terms of reducing bone remodeling, improving bone density, and pain in FD.
Topics: Humans; Bone Density Conservation Agents; Fibrous Dysplasia, Polyostotic; Diphosphonates; Fibrous Dysplasia of Bone; Bone Resorption; Pain
PubMed: 37632645
DOI: 10.1007/s11154-023-09832-2 -
Early Human Development May 2023There is lack of evidence synthesis on the global consequences of bronchopulmonary dysplasia (BPD) in adolescence. (Review)
Review
BACKGROUND
There is lack of evidence synthesis on the global consequences of bronchopulmonary dysplasia (BPD) in adolescence.
AIM
Assess the impact of bronchopulmonary dysplasia on respiratory and non-respiratory outcomes in adolescents.
METHODS
A systematic review of studies assessing the outcomes of adolescents aged 10 to 19 years-old with BPD was conducted. We independently screened studies published until 6th March 2023 in PubMed® and Scopus® databases. Data on methodologic design, sample descriptive and findings were extracted from each study. Risk of bias was assessed using quality assessment tools.
RESULTS
Thirty-one studies were included. Adolescents with a history of BPD present with more respiratory symptoms (wheezing, respiratory exacerbations, need for respiratory medication) and twenty-five studies showed a reduction in pulmonary function, with varying impact according to BPD severity and no differences before and after the surfactant era. Spirometry evaluation throughout the years is not consensual, but methacholine and salbutamol response in BPD groups is increased compared to non-BPD groups. Markers of eosinophilic airway inflammation are not increased as in asthma patients. Exercise potential is identical, but data regarding physical capacity and activity are inconsistent. More frequent radiologic abnormalities translate into higher high-resolution computed tomography scores, with linear (72.2 %) and triangular subpleural opacities (58.3 %) as the most common findings. There is a higher risk for special needs in education, but quality of life seems to be equal to non-BPD adolescents.
CONCLUSIONS
BPD negatively impacts both pulmonary and non-pulmonary outcomes in adolescents.
Topics: Infant, Newborn; Humans; Adolescent; Child; Young Adult; Adult; Bronchopulmonary Dysplasia; Quality of Life; Lung; Asthma; Spirometry
PubMed: 36965348
DOI: 10.1016/j.earlhumdev.2023.105756 -
Journal of Stomatology, Oral and... Dec 2023Craniofacial fibrous dysplasia (CFD) may be associated with major cosmetic or functional consequences. However, management recommendations for CFD are currently...
Craniofacial fibrous dysplasia (CFD) may be associated with major cosmetic or functional consequences. However, management recommendations for CFD are currently unavailable. Therefore, this systematic literature review aimed to review the existing approaches for CFD management and propose a management algorithm. The focus question was "What are the different options for CFD treatment and their complication rates?" The MEDLINE database was searched, and 33 articles evaluating a total of 1154 patients were reviewed. The bias assessment showed that 20 of the 33 studies had a high or intermediate risk of bias, mainly because of retrospective data collection and small patient numbers. Radical surgery showed a lower recurrence rate than debulking, but its use should be weighed against the morbidity caused by the reconstruction performed in this technique. Orbital decompression using a radical technique or debulking is effective in cases showing exophthalmos or dystopia. Surveillance is a viable option for asymptomatic and/or non-progressive lesions. In cases showing optic nerve compression, prophylactic decompression should be avoided, and decompression should be performed only when patients show diminished visual acuity or visual field defect. Although bisphosphonates have shown efficacy in pain management, their posology requires further discussion. A management algorithm is presented.
Topics: Humans; Craniofacial Fibrous Dysplasia; Retrospective Studies; Decompression, Surgical; Face; Optic Nerve Diseases
PubMed: 37866506
DOI: 10.1016/j.jormas.2023.101660 -
Gene Feb 2023Developmental dysplasia of the hip (DDH) is a complex developmental deformity whose pathogenesis and susceptibility-related genes have yet to be elucidated. This... (Review)
Review
BACKGROUND
Developmental dysplasia of the hip (DDH) is a complex developmental deformity whose pathogenesis and susceptibility-related genes have yet to be elucidated. This systematic review summarizes the current literature on DDH-related gene mutations, animal model experiments, and epigenetic changes in DDH.
METHODS
We performed a comprehensive search of relevant documents in the Medline, Scopus, Cochrane, and ScienceDirect databases covering the period from October 1991 to October 2021. We analyzed basic information on the included studies and summarized the DDH-related mutation sites, animal model experiments, and epigenetic changes associated with DDH.
RESULTS
A total of 63 studies were included in the analysis, of which 54 dealt with the detection of gene mutations, 7 presented details of animal experiments, and 6 were epigenetic studies. No genetic mutations were clearly related to the pathogenesis of DDH, including the most frequently studied genes on chromosomes 1, 17, and 20. Most gene-related studies were performed in Han Chinese or North American populations, and the quality of these studies was medium or low. GDF5 was examined in the greatest number of studies, and mutation sites with odds ratios > 10 were located on chromosomes 3, 9, and 13. Six mutations were found in animal experiments (i.e., CX3CR1, GDF5, PAPPA2, TENM3, UFSP2, and WISP3). Epigenetics research on DDH has focused on GDF5 promoter methylation, three microRNAs (miRNAs), and long noncoding RNAs. In addition, there was also a genetic test for miRNA and mRNA sequencing.
CONCLUSIONS
DDH is a complex joint deformity with a considerable genetic component whose early diagnosis is significant for preventing disease. At present, no genes clearly involved in the pathogenesis of DDH have been identified. Research on mutations associated with this condition is progressing in the direction of in vivo experiments in animal models to identify DDH susceptibility genes and epigenetics analyses to provide novel insights into its pathogenesis. In the future, genetic profiling may improve matters.
Topics: Humans; Animals; Developmental Dysplasia of the Hip; Hip Dislocation, Congenital; Epigenesis, Genetic; Mutation; Asian People; Membrane Proteins; Nerve Tissue Proteins
PubMed: 36435507
DOI: 10.1016/j.gene.2022.147067 -
Knee Surgery, Sports Traumatology,... Oct 2022The literature on hip injuries in ballet dancers was systematically evaluated to answer (1) whether the prevalence of morphological abnormalities and pathology of hip... (Review)
Review
PURPOSE
The literature on hip injuries in ballet dancers was systematically evaluated to answer (1) whether the prevalence of morphological abnormalities and pathology of hip injuries in dancers differs from the general population (2) if there are any specific risk factors which contribute to a higher rate of hip injury and (3) what are the outcomes of primary and secondary intervention strategies.
METHODS
A systematic literature search of Medline, EMBASE and the Cochrane Library was undertaken for all literature relating to hip injuries in ballet dancers using the PRISMA guidelines. Reference lists were also searched for relevant literature. Clinical outcome studies, prospective/retrospective case series published between 1989 and October 2021 were included. Review articles (non-original data), case reports, studies on animals as well as book chapters were excluded.
RESULTS
The search yielded 445 studies, of which 35 were included for final analyses after screening. This included 1655 participants, of which 1131 were females. The analyses revealed that damage at the chondrolabral junction and degenerative disease of the hip may develop at a higher rate in ballet dancers than in the general population (odds ratio > 1 in 15/18 cohorts). The intra-articular lesions were more frequently found in postero-superior region of the hip suggesting an alternative impingement mechanism. Furthermore, numerous risk factors specific for hip injury in ballet were highlighted amidst a wide body of literature which consistently reports risk factors for a more generic 'dancer vulnerability'.
CONCLUSION
Ballet dancers may suffer from both higher rates of chondrolabral damage and degenerative disease in their hips. In contrast to other sports, the intra-articular lesions are more frequently found in postero-superior region of the hip. Future research clarifying the prevalence of osseous abnormalities and prevention strategies in dancers may be pivotal in delaying the development of hip disease in this cohort.
LEVEL OF EVIDENCE
Level IV.
Topics: Dancing; Female; Hip; Hip Injuries; Humans; Male; Prospective Studies; Retrospective Studies
PubMed: 35305112
DOI: 10.1007/s00167-022-06928-1 -
Journal of Laparoendoscopic & Advanced... Dec 2023Gastroesophageal reflux disease is a common gastrointestinal disorder with one of its most feared complications being Barrett's esophagus (BE). Currently, most of the...
Gastroesophageal reflux disease is a common gastrointestinal disorder with one of its most feared complications being Barrett's esophagus (BE). Currently, most of the recommendations of BE management are driven by the level of dysplasia. However, the length of BE might also be related to the risk of dysplasia/malignant transformation. We aimed to determine the appropriate management of BE based on its length. A systematic literature review was conducted with searches made on PubMed, Embase, and Cochrane databases. Long-segment BE (LSBE) was defined as 3 cm or longer and short-segment BE (SSBE) as under 3 cm. Studies evaluating the behavior and management of SSBE and/or LSBE were included for analysis. LSBE have greater risk of dysplasia or progression to esophageal adenocarcinoma compared to SSBE. Despite this greater risk, LSBE and SSBE are currently managed similarly based on the presence and degree of dysplasia. Endoscopic and ablative techniques may have higher level of success and less complications in SSBE, compared to LSBE. Decreasing time interval between surveillance may be a viable option for managing LSBE. Although many algorithms of monitoring and treatment of BE remain the same regardless of segment length, current evidence suggests that more aggressive management for LSBE might be needed due to its higher risk of malignant progression.
Topics: Humans; Barrett Esophagus; Esophageal Neoplasms; Adenocarcinoma; Gastroesophageal Reflux; Endoscopy
PubMed: 37796531
DOI: 10.1089/lap.2023.0321 -
BMJ Paediatrics Open Oct 2023To estimate the prevalence of developmental dysplasia of the hip (DDH) in infants with a systematic review and meta-analysis. (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
To estimate the prevalence of developmental dysplasia of the hip (DDH) in infants with a systematic review and meta-analysis.
METHOD
A literature search was conducted in April 2023, using databases such as Cochrane Library, PubMed, MEDLINE, CNKI, and SinoMed, without language restrictions. Eligible studies included cross-sectional studies reporting the prevalence of DDH among infants aged 0-12 months. Two independent reviewers manually selected and coded the studies, with any disagreements resolved by a third reviewer. Meta-analysis was performed using a random-effects model to calculate the prevalence of DDH. Regression analysis examined the trend of DDH prevalence, and stratification analysis explored heterogeneity between studies.
RESULTS
A total of 65 studies involving 3 451 682 infants were included in the meta-analysis. None of the studies were classified as high quality, four were medium-to-high quality, 50 were low-to-medium quality, and eight were low quality. The pooled prevalence of DDH was 1.40% (95% CI: 0.86 to 2.28, I=100%), and prevalence of dysplasia, subluxation, and dislocation was 1.45% (95% CI: 0.93 to 2.24, I=97%), 0.37% (95% CI: 0.22 to 0.60, I=94%), and 0.21% (95% CI: 0.13 to 0.34, I=92%), respectively. Notably, the overall prevalence has a slight upward trend in the last three decades (β=0.24, p=0.35), but the dysplasia was downward trend (β=-0.48, p<0.01). Girls have higher risk of DDH than boys (1.46% vs 0.66%; Q=5.83, df=1, p=0.02). There were no significant differences based on gender, country, setting, or screening technique.
CONCLUSION
The prevalence of DDH among infants is approximately one in a 100, with girls being at higher risk. Though the prevalence of dysplasia has decreased, there is a slight upward trend in overall DDH. Therefore, routine screening for DDH in infants is recommended to prevent more serious developmental problems.
Topics: Male; Female; Humans; Infant; Prevalence; Cross-Sectional Studies; Developmental Dysplasia of the Hip; Hip Dislocation, Congenital; Mass Screening
PubMed: 37879719
DOI: 10.1136/bmjpo-2023-002080 -
Bronchopulmonary dysplasia prediction models: a systematic review and meta-analysis with validation.Pediatric Research Jul 2023Prediction models could identify infants at the greatest risk of bronchopulmonary dysplasia (BPD) and allow targeted preventative strategies. We performed a systematic... (Meta-Analysis)
Meta-Analysis
Prediction models could identify infants at the greatest risk of bronchopulmonary dysplasia (BPD) and allow targeted preventative strategies. We performed a systematic review and meta-analysis with external validation of identified models. Studies using predictors available before day 14 of life to predict BPD in very preterm infants were included. Two reviewers assessed 7628 studies for eligibility. Meta-analysis of externally validated models was followed by validation using 62,864 very preterm infants in England and Wales. A total of 64 studies using 53 prediction models were included totalling 274,407 infants (range 32-156,587/study). In all, 35 (55%) studies predated 2010; 39 (61%) were single-centre studies. A total of 97% of studies had a high risk of bias, especially in the analysis domain. Following meta-analysis of 22 BPD and 11 BPD/death composite externally validated models, Laughon's day one model was the most promising in predicting BPD and death (C-statistic 0.76 (95% CI 0.70-0.81) and good calibration). Six models were externally validated in our cohort with C-statistics between 0.70 and 0.90 but with poor calibration. Few BPD prediction models were developed with contemporary populations, underwent external validation, or had calibration and impact analyses. Contemporary, validated, and dynamic prediction models are needed for targeted preventative strategies. IMPACT: This review aims to provide a comprehensive assessment of all BPD prediction models developed to address the uncertainty of which model is sufficiently valid and generalisable for use in clinical practice and research. Published BPD prediction models are mostly outdated, single centre and lack external validation. Laughon's 2011 model is the most promising but more robust models, using contemporary data with external validation are needed to support better treatments.
Topics: Infant; Infant, Newborn; Humans; Infant, Premature; Bronchopulmonary Dysplasia; Infant, Very Low Birth Weight; Infant, Premature, Diseases; England
PubMed: 36624282
DOI: 10.1038/s41390-022-02451-8 -
Journal of Perinatal Medicine Nov 2023Dexamethasone administration can reduce bronchopulmonary dysplasia, our objective was to identify long term adverse effects. (Review)
Review
BACKGROUND
Dexamethasone administration can reduce bronchopulmonary dysplasia, our objective was to identify long term adverse effects.
CONTENT
A systematic review was performed to determine the childhood and adolescent cardiopulmonary and cognitive effects of dexamethasone systemically administered to preterm infants during neonatal intensive care. Relevant studies were identified by searching two electronic health databases and the grey literature. Spirometry assessments were used as respiratory outcomes, blood pressure and echocardiography assessments as cardiovascular outcomes and cognitive and motor function as cognitive outcomes. From 1,479 articles initially identified, 18 studies (overall 1,609 patients) were included (respiratory n=8, cardiovascular n=2, cognitive n=10); all were observational cohort studies. Dexamethasone exposure was associated with worse pulmonary outcomes in children and adolescents (more abnormal FVC and FEV1:FVC z scores). Dexamethasone exposure was associated in one study with lower IQ scores compared to preterm controls (mean 78.2 [SD 15.0] vs. 84.4 [12.6], [p=0.008]) and in two others was associated with lower total and performance IQ when compared to term controls (p<0.001).
SUMMARY AND OUTLOOK
Postnatal dexamethasone exposure has a negative influence on pulmonary and cognitive outcomes in childhood and adolescence. Medications with a better benefit to risk profile need to be identified.
Topics: Adolescent; Child; Humans; Infant; Infant, Newborn; Adrenal Cortex Hormones; Anti-Inflammatory Agents; Bronchopulmonary Dysplasia; Chronic Disease; Dexamethasone; Glucocorticoids; Infant, Premature
PubMed: 37606507
DOI: 10.1515/jpm-2023-0297 -
Endoscopy International Open Sep 2023Upper gastrointestinal (UGI) endoscopy lacks established quality indicators. We conducted an umbrella systematic review of potential quality indicators for the... (Review)
Review
Upper gastrointestinal (UGI) endoscopy lacks established quality indicators. We conducted an umbrella systematic review of potential quality indicators for the detection of UGI cancer and dysplasia. Bibliographic databases were searched up to December 2021 for systematic reviews and primary studies. Studies reporting diagnostic accuracy, detection rates or the association of endoscopy or endoscopist-related factors with UGI cancer or dysplasia detection were included. AMSTAR2 and JBI checklists were used to assess systematic review and primary study quality. Clinical heterogeneity precluded meta-analysis and findings are summarized narratively. Eight systematic reviews and nine primary studies were included. Image enhancement, especially narrow band imaging, had high diagnostic accuracy for dysplasia and early gastric cancer (pooled sensitivity 0.87 (95% CI 0.84-0.89) and specificity 0.97 (0.97-0.98)). Higher detection rates with longer endoscopy examination times were reported in three studies, but no difference was observed in one study. Endoscopist biopsy rate was associated with increased gastric cancer detection (odds ratio 2.5; 95% confidence interval [CI] 2.1-2.9). Early esophageal cancer (0.17% vs 0.14%, =0.04) and gastric cancer (0.16% vs 0.12%, =0.02) detection rates were higher with propofol sedation compared to no sedation. Endoscopies performed by trained endoscopists on dedicated Barrett's surveillance lists had higher detection rates (8% vs 3%, <0.001). The neoplasia detection rate during diagnostic endoscopies for Barrett's esophagus was 7% (95% CI 4%-10%). Image enhancement use, longer examination times, biopsy rate and propofol sedation are potential quality indicators for UGI endoscopy. Neoplasia detection rate and dedicated endoscopy lists are additional potential quality indicators for Barrett's esophagus.
PubMed: 37719799
DOI: 10.1055/a-2117-8621