-
The British Journal of Oral &... Nov 2022The main purpose of this study was to identify an algorithm for the surgical management of fibrous dysplasia in syndromic (McCune-Albright syndrome) and non-syndromic... (Meta-Analysis)
Meta-Analysis Review
The main purpose of this study was to identify an algorithm for the surgical management of fibrous dysplasia in syndromic (McCune-Albright syndrome) and non-syndromic patients (monostotic and polyostotic subtypes). The secondary objectives were to assess the prevalence of affected craniofacial bones and the main clinical presentation. The authors performed a systematic review and meta-analysis by conducting a comprehensive electronic search from 1 January 2000 to 31 December 2019. A total of 1260 patients were included. The maxilla was the most affected facial bone (41%) (p<0.001, CI 38.3 to 43.8) and facial asymmetry was the chief complaint (p<0.001, CI 31.7 to 37.1). Conservative surgery registered higher recurrence rates than radical resection in both syndromic (84%) (p<0.001, CI 70.9 to 92.8) and non-syndromic patients (26%) (p<0.001, CI 21.8 to 30.6). Compared with prophylactic decompression, therapeutic optic nerve decompression (OND) showed better postoperative outcomes in both syndromic (p=0.9, CI 18.6 to 55.9) and non-syndromic patients (p=0.09, CI 9.3 to 28.4). Watchful waiting showed excellent results in both subgroups when asymptomatic (p<0.001). Syndromic and non-syndromic patients share the same treatment strategies. Radical resection is the preferred surgical technique to eradicate the disease, but it is often difficult to perform due to the extent and location of the disease. Furthermore, the authors advise early therapeutic over prophylactic OND to prevent optic nerve atrophy. Asymptomatic patients should be managed expectantly. Finally, medical management helps reduce the symptoms of bone pain (p=0.02 in non-syndromic and p<0.001 in syndromic patients).
Topics: Humans; Craniofacial Fibrous Dysplasia; Skull; Fibrous Dysplasia, Polyostotic; Orbit; Maxilla; Fibrous Dysplasia of Bone
PubMed: 35817637
DOI: 10.1016/j.bjoms.2022.06.002 -
Frontiers in Pediatrics 2023We aimed to determine the association between red blood cell transfusions (RBCT) and bronchopulmonary dysplasia (BPD) in neonates. (Review)
Review
BACKGROUND
We aimed to determine the association between red blood cell transfusions (RBCT) and bronchopulmonary dysplasia (BPD) in neonates.
METHODS
A systematic review and meta-analysis were conducted using data obtained from literature search of PubMed, Embase, and Web of Science from their inception till May 1, 2022. Two reviewers independently selected potentially relevant studies, and after data extraction, they assessed the methodological quality of the included studies using the Newcastle-Ottawa scale. Data were pooled using random-effects models in Review Manager 5.3. Subgroup-analysis was performed based on the number of transfusions and adjusted results.
RESULTS
Of the 1,011 identified records, 21 total case-control, cross-sectional, and cohort studies were selected, which included a total of 6,567 healthy controls and 1,476 patients with BPD. The pooled unadjusted odds ratio ([OR], 4.01; 95% confidence interval [CI] 2.31-6.97) and adjusted OR (5.11; 95% CI 3.11-8.4) showed significant association between RBCT and BPD. A substantial heterogeneity was noted, which could be due to different variables controlled for in each study. The subgroup analysis showed that heterogeneity may be partially explained by the extent of transfusion.
CONCLUSION
The association between BPD and RBCT remains unclear based on the current data due to the substantial heterogeneity among the results. Well-designed studies are still needed in the future.
PubMed: 37325359
DOI: 10.3389/fped.2023.1095889 -
Indian Journal of Orthopaedics Dec 2021Developmental dysplasia of hip (DDH) is a common disorder of childhood and has a good prognosis when treated at an early age. In spite of being a significant concern,... (Review)
Review
BACKGROUND
Developmental dysplasia of hip (DDH) is a common disorder of childhood and has a good prognosis when treated at an early age. In spite of being a significant concern, many children with DDH are not picked early and present late at walking age. In our country, it is presumed to be due to absence of a national policy for screening of DDH. Screening programmes including the combination of clinical and radiological methods in different ways have been suggested. However, the exact method of screening is controversial.
PURPOSE
To analyze effectiveness and cost-effectiveness of various screening methods for DDH.
STUDY DESIGN
Systematic review.
METHODS
This review was conducted in accordance with PRISMA guidelines. Medline database was explored for original case series and randomized clinical trials. Inclusion criteria were English language, screening for DDH in neonates, sample size more than 500, and studies with minimum duration of one year.
RESULTS
Thirty-four studies were selected to write the manuscript. This included 23 studies looking for effectiveness of a screening programme and 11 studies comparing various outcomes of different screening strategies. A trend favoring universal ultrasound screening was observed.
CONCLUSION
The literature supports universal ultrasound screening and has proved its cost-effectiveness. However, considering the logistic and financial challenges in our country, immediate implementation of universal ultrasound screening seems impractical. In the absence of any current guidelines for screening for DDH in India, we suggest professional organizations involved in the care of children and public health policy-makers to come together to develop national screening guidelines for DDH.
PubMed: 35003532
DOI: 10.1007/s43465-021-00409-2 -
Otology & Neurotology : Official... Dec 2014The management options for otologic symptoms (i.e., hearing loss, otorrhea) in patients with fibrous dysplasia of the temporal bone (FDTB) include either observation or... (Review)
Review
OBJECTIVES
The management options for otologic symptoms (i.e., hearing loss, otorrhea) in patients with fibrous dysplasia of the temporal bone (FDTB) include either observation or otologic surgery. The objective of this review is to describe the hearing outcomes in patients with FDTB to compare surgical intervention and conservative management to determine an evidence-based approach to patient management.
DATA SOURCES
Cochrane Central Register of Controlled Trials (1995-April 5, 2013), MEDLINE (January 1948-April 5, 2013), EMBASE (January 1974-April 5, 2013), conference proceedings, and the reference lists of articles.
STUDY SELECTION
Studies describing interventions or no intervention in patients with FDTB were included. Studies failing to report hearing outcomes were excluded.
DATA EXTRACTION
Two authors independently reviewed titles and abstracts, read full-text articles, assessed quality, and extracted data. The Preferred Reporting Items for Systematic Review and Meta-Analyses (PRISMA) guidelines were followed.
DATA SYNTHESIS
Seventy-two studies involving 215 patients were included. Thirty-one studies (n = 49 patients), including three case series and 28 case reports, had sufficient information for analysis of hearing outcomes (Level 4 evidence). The surgical intervention showing the best hearing outcomes was canaloplasty, with improvement in 84% of patients (n = 16 patients) and stable hearing thresholds in 18% (n = 3 patients).
CONCLUSION
The literature lacks consistent reporting of hearing outcomes after otologic surgery in FDTB. Level 4 evidence suggests that an improvement in hearing thresholds is achievable in most patients when the disease involves the external ear canal. A management algorithm is proposed using the available evidence.
Topics: Fibrous Dysplasia of Bone; Hearing Loss; Humans; Temporal Bone; Treatment Outcome
PubMed: 25280053
DOI: 10.1097/MAO.0000000000000602 -
Pediatric Pulmonology Nov 2022In adults, permissive hypercapnia reduces mortality and ventilation duration. However, in preterm infants, the findings from past research regarding the efficacy and... (Meta-Analysis)
Meta-Analysis Review
CONTEXT
In adults, permissive hypercapnia reduces mortality and ventilation duration. However, in preterm infants, the findings from past research regarding the efficacy and safety of permissive hypercapnia are controversial.
OBJECTIVE
To evaluate the efficacy and safety of permissive hypercapnia versus normocapnia in preterm infants on mechanical ventilation.
DATA SOURCES
MEDLINE, EMBASE, CENTRAL, and CINAHL STUDY SELECTION: Published randomized controlled trials (RCTs), non-RCTs, interrupted time series, cohort studies, case-control studies, and controlled before-and-after studies were included.
DATA EXTRACTION
Two reviewers independently screened the title, abstract, and full text, extracted data, assessed the risk of bias, and evaluated certainty of evidence (CoE) according to the Grading of Recommendations Assessment and Development and Evaluation approach. A meta-analysis of RCTs was performed using the random-effects model.
RESULTS
Four RCTs (693 infants) and one cohort study (371 infants) were included. No significant differences existed between the permissive hypercapnia and normocapnia groups for bronchopulmonary dysplasia (BPD) (risk ratio [RR], 0.94; 95% confidence interval [CI], 0.74-1.18; very low CoE) and a composite outcome of death or BPD (RR, 1.05; 95% CI, 0.90-1.23; very low CoE). Permissive hypercapnia may increase necrotizing enterocolitis (RR, 1.69; 95% CI, 0.98-2.91; very low CoE), but the null or trivial effect cannot be excluded. No significant differences existed between the two groups for any other outcome assessed (very low-to-low CoE).
LIMITATIONS
The sample sizes were less than the optimal sizes for all outcomes assessed, indicating the need for further trials.
CONCLUSIONS
Permissive hypercapnia did not have any significant benefit or harm in preterm infants.
Topics: Bronchopulmonary Dysplasia; Enterocolitis, Necrotizing; Humans; Hypercapnia; Infant; Infant, Newborn; Infant, Premature; Respiration, Artificial
PubMed: 35945674
DOI: 10.1002/ppul.26108 -
Quality of Life Research : An... Jul 2023To synthesize evidence on the impact of bronchopulmonary dysplasia (BPD) on the Quality of Life (QoL) of affected individuals from three perspectives: (i) QoL of... (Review)
Review
PURPOSE
To synthesize evidence on the impact of bronchopulmonary dysplasia (BPD) on the Quality of Life (QoL) of affected individuals from three perspectives: (i) QoL of caregiver; (ii) caregiver's perception of BPD patient's QoL; and (iii) BPD patient's self-reported QoL.
METHODS
Quantitative studies (case-control, cohort, and case series) on the QoL of BPD patients or their caregivers were considered. We conducted a systematic literature search of 6 databases (PubMed, Embase, World of Science, CINAHL, PsycINFO, and Chinese National Knowledge Infrastructure) for relevant studies. All databases were searched from the date of inception of the databases to 31 March 2022. Populations of interest were caregivers with preterm babies with BPD, or children/adults who were born premature and diagnosed with BPD. The main outcome measures were total and subdomain QoL scores, and factors affecting QoL.
RESULTS
A total of 1078 articles were found; 10 were eligible for analysis, which included 247 caregivers and 1632 patients with BPD. The QoL of patients differed by domains-some were poorer or similar, but none of the QoL domains was better than QoL of healthy controls. Poor sleep and acute care needs of BPD patients negatively affected caregiver's QoL, while increasing illness acuity negatively affected the QoL of BPD patients. The QoL of BPD patients and their caregivers was most adversely affected during the immediate post-discharge period and tended to improve with time. The physical QoL of BPD patients was similar to that of preterm babies without BPD when assessed during late childhood and early adulthood.
CONCLUSION
QoL assessment should be performed as an outcome measure and incorporated in the care plan for BPD patients and their caregivers. Systematic Review Registration PROSPERO CRD42021292253.
Topics: Infant, Newborn; Infant; Adult; Humans; Child; Bronchopulmonary Dysplasia; Quality of Life; Caregivers; Aftercare; Patient Discharge
PubMed: 36484947
DOI: 10.1007/s11136-022-03311-y -
PloS One 2016There is much debate surrounding the use of inhaled bronchodilators and corticosteroids for infants with bronchopulmonary dysplasia (BPD). (Review)
Review
BACKGROUND
There is much debate surrounding the use of inhaled bronchodilators and corticosteroids for infants with bronchopulmonary dysplasia (BPD).
OBJECTIVE
The objective of this systematic review was to identify strengths and knowledge gaps in the literature regarding inhaled therapies in BPD and guide future research to improve long-termoutcomes.
METHODS
The databases of Academic Search Complete, CINAHL, PUBMED/MEDLINE, and Scopus were searched for studies that evaluated both acute and long-term clinical outcomes related to the delivery and therapeutic efficacy of inhaled beta-agonists, anticholinergics and corticosteroids in infants with developing and/or established BPD.
RESULTS
Of 181 articles, 22 met inclusion criteria for review. Five evaluated beta-agonist therapies (n = 84, weighted gestational age (GA) of 27.1(26-30) weeks, weighted birth weight (BW) of 974(843-1310) grams, weighted post menstrual age (PMA) of 34.8(28-39) weeks, and weighted age of 53(15-86) days old at the time of evaluation). Fourteen evaluated inhaled corticosteroids (n = 2383, GA 26.2(26-29) weeks, weighted BW of 853(760-1114) grams, weighted PMA of 27.0(26-31) weeks, and weighted age of 6(0-45) days old at time of evaluation). Three evaluated combination therapies (n = 198, weighted GA of 27.8(27-29) weeks, weighted BW of 1057(898-1247) grams, weighted PMA of 30.7(29-45) weeks, and age 20(10-111) days old at time of evaluation).
CONCLUSION
Whether inhaled bronchodilators and inhaled corticosteroids improve long-term outcomes in BPD remains unclear. Literature regarding these therapies mostly addresses evolving BPD. There appears to be heterogeneity in treatment responses, and may be related to varying modes of administration. Further research is needed to evaluate inhaled therapies in infants with severe BPD. Such investigations should focus on appropriate definitions of disease and subject selection, timing of therapies, and new drugs, devices and delivery methods as compared to traditional methods across all modalities of respiratory support, in addition to the assessment of long-term outcomes of initial responders.
Topics: Administration, Inhalation; Adrenal Cortex Hormones; Bronchodilator Agents; Bronchopulmonary Dysplasia; Humans; Infant, Newborn
PubMed: 26840339
DOI: 10.1371/journal.pone.0148188 -
Frontiers in Pediatrics 2019Bronchopulmonary dysplasia (BPD) is the most common complication of very preterm birth and can lead to lifelong health consequences. Optimal nutrition is a cornerstone...
Bronchopulmonary dysplasia (BPD) is the most common complication of very preterm birth and can lead to lifelong health consequences. Optimal nutrition is a cornerstone in the prevention and treatment of BPD. In very preterm infants, mother's own milk (MOM) feeding is associated with lower risks of necrotizing enterocolitis, retinopathy of prematurity, and sepsis. Although several studies have shown that MOM may protect against BPD, a systematic analysis of the evidence has not been performed to date. A comprehensive literature search was conducted using PubMed/MEDLINE and EMBASE, from their inception to 1 December 2017. Longitudinal studies comparing the incidence of BPD in preterm infants fed with exclusive MOM, MOM supplemented with preterm formula (PF), and/or exclusively fed with PF were selected. A random-effects model was used to calculate the Mantel Haenszel risk ratio (RR) and 95% confidence interval (CI). Fifteen studies met the inclusion criteria (4,984 infants, 1,416 BPD cases). Use of exclusive MOM feedings was associated with a significant reduction in the risk of BPD (RR 0.74, 95% CI 0.57-0.96, 5 studies). In contrast, meta-analysis could not demonstrate a significant effect on BPD risk when infants fed with more than 50% MOM were compared with infants fed with <50% MOM (RR 0.98, 95% CI 0.77-1.23, 10 studies) or when infants fed with MOM supplemented with PF were compared with infants fed with exclusive PF (RR 1.00, 95% CI 0.78-1.27, 6 studies). Meta-regression showed that differences in gestational age were a significant confounder of the effect of MOM. To our knowledge, this is the first systematic review and meta-analysis that specifically evaluates the role of MOM on BPD. Our data indicate that MOM may reduce the incidence of BPD when used as an exclusive diet, but this result needs to be interpreted with caution. We did not find the same difference in analyses with other dosages of MOM. Further studies adequately powered to detect changes in BPD rates and that adjust for confounders are needed to confirm the beneficial effects of MOM on BPD.
PubMed: 31275904
DOI: 10.3389/fped.2019.00224 -
Dento Maxillo Facial Radiology May 2009To evaluate the principal features of fibrous dysplasia (FD) by systematic review (SR) and to compare their frequencies between four global groups. (Review)
Review
OBJECTIVES
To evaluate the principal features of fibrous dysplasia (FD) by systematic review (SR) and to compare their frequencies between four global groups.
METHODS
The databases searched were the PubMed interface of Medline and LILACS. Only those reports of FD which occurred in a series in the reporting authors' caseload were considered. All cases were confirmed fibro-osseous lesions histopathologically. The SR-included series must also have included radiographs.
RESULTS
Of the 106 reports considered (including the Hong Kong report), 31 reports and a total of 788 cases were included in the SR. 11 SR-included series were in languages other than English. FD affected both genders equally, but was 50% more prevalent in the maxilla. The mean age at first presentation was 24 years. The decade with the greatest frequency was the second, in which males accounted for 63%. The main symptom in 90% of all SR-included cases was swelling (including deformation of the jaws). Not one SR-included case directly involved the ocular apparatus. All cases displayed buccolingual expansion; all mandibular cases exhibited downward displacement of the lower border of the mandible and almost all maxillary cases involved the maxillary antrum. Only 35% of reports included follow-up; 18% of cases recurred or were reactivated. Not one case displayed sarcomatous change.
CONCLUSIONS
Long-term follow-up of large series that would have revealed the long-term outcomes of FD was lacking. This is necessary because many cases do not burn out at the end of adolescence, as expected of a hamartoma, but are reactivated.
Topics: Facial Bones; Fibrous Dysplasia of Bone; Humans
PubMed: 19372108
DOI: 10.1259/dmfr/16645318 -
Surgical Endoscopy Apr 2021This systematic review and meta-analysis aims to evaluate efficacy and safety of endoscopic treatment for the non-polypoid dysplasia in patients with long-standing IBD. (Meta-Analysis)
Meta-Analysis
BACKGROUND
This systematic review and meta-analysis aims to evaluate efficacy and safety of endoscopic treatment for the non-polypoid dysplasia in patients with long-standing IBD.
METHODS
Medline, Embase, Cochrane, and clinicaltrials.gov registry were comprehensively searched. Pooled estimates of curative, R0, en-bloc resection rates, CRC, metachronous dysplasia, and local recurrence rates were calculated. Subgroup analysis according to areas, lesion size, endoscopic resection techniques, and grades of dysplasia were conducted. Data synthesis was completed in R using the package "meta".
RESULTS
Of the 973 studies initially identified, 7 met the inclusion/exclusion criteria. These were all single-arm cohorts and included a total of 202 patients with IBD and non-polypoid dysplasia. The combined R0 and en-bloc resection rate were 0.70 (95% CI 0.55-0.81) and 0.86 (95% CI 0.65-0.95), respectively, with a recurrence rate of 0.08 (95% CI 0.05-0.13). CRC and metachronous dysplasia incidences were pooled as 32.53 (95% CI 12.21-86.67) and 90.24 (95% CI 44.91-181.33) per 1000 patient years.
CONCLUSIONS
Non-polypoid dysplasia associated with IBD can be resected endoscopically, especially by ESD. However, these patients have higher CRC and metachronous dysplasia incidence rates than patients with polypoid dysplasia, indicating a closer endoscopic surveillance.
Topics: Aged; Endoscopic Mucosal Resection; Humans; Inflammatory Bowel Diseases; Middle Aged
PubMed: 33523273
DOI: 10.1007/s00464-020-08225-9