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BMJ Paediatrics Open Jul 2023Neonatal care is commonly regionalised, meaning specialist services are only available at certain units. Consequently, infants with surgical conditions needing...
BACKGROUND
Neonatal care is commonly regionalised, meaning specialist services are only available at certain units. Consequently, infants with surgical conditions needing specialist care who are born in non-surgical centres require postnatal transfer. Best practice models advocate for colocated maternity and surgical services as the place of birth for infants with antenatally diagnosed congenital conditions to avoid postnatal transfers. We conducted a systematic review to explore the association between location of birth and short-term outcomes of babies with gastroschisis, congenital diaphragmatic hernia (CDH) and oesophageal atresia with or without tracheo-oesophageal fistula (TOF/OA).
METHODS
We searched MEDLINE, CINAHL, Web of Science and SCOPUS databases for studies from high income countries comparing outcomes for infants with gastroschisis, CDH or TOF/OA based on their place of delivery. Outcomes of interest included mortality, length of stay, age at first feed, comorbidities and duration of parenteral nutrition. We assessed study quality using the Newcastle-Ottawa Scale. We present a narrative synthesis of our findings.
RESULTS
Nineteen cohort studies compared outcomes of babies with one of gastroschisis, CDH or TOF/OA. Heterogeneity across the studies precluded meta-analysis. Eight studies carried out case-mix adjustments. Overall, we found conflicting evidence. There is limited evidence to suggest that birth in a maternity unit with a colocated surgical centre was associated with a reduction in mortality for CDH and decreased length of stay for gastroschisis.
CONCLUSIONS
There is little evidence to suggest that delivery in colocated maternity-surgical services may be associated with shortened length of stay and reduced mortality. Our findings are limited by significant heterogeneity, potential for bias and paucity of strong evidence. This supports the need for further research to investigate the impact of birth location on outcomes for babies with congenital surgical conditions and inform future design of neonatal care systems.
PROSPERO REGISTRATION NUMBER
CRD42022329090.
Topics: Infant; Infant, Newborn; Humans; Female; Pregnancy; Hernias, Diaphragmatic, Congenital; Gastroschisis; Cohort Studies; Esophageal Atresia; Tracheoesophageal Fistula
PubMed: 37474200
DOI: 10.1136/bmjpo-2023-002007 -
Journal of Pediatric Surgery Oct 2023Anastomotic stricture is a common postoperative complication of oesophageal atresia ± tracheoesophageal fistula (OA/TOF) repair. Acid gastro-oesophageal reflux... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Anastomotic stricture is a common postoperative complication of oesophageal atresia ± tracheoesophageal fistula (OA/TOF) repair. Acid gastro-oesophageal reflux disease (GORD) is considered to be a factor in stricture formation and acid suppression medication is recommended post-operatively in consensus guidance. We aimed to investigate whether patients who were treated prophylactically with acid suppression medication had a reduced incidence of strictures compared to those who did not receive it.
METHODS
A systematic review of studies was performed, searching multiple databases without language or date restrictions. Multiple reviewers independently assessed study eligibility and literature quality. The primary outcome was anastomotic stricture formation, with secondary outcomes of GORD, anastomotic leak, and oesophagitis. Meta-analysis was performed using a random effects model, and the results were expressed as an odds ratio (OR) with 95% confidence intervals (CI).
RESULTS
No randomised studies on the topic were identified. Twelve observational studies were included in the analysis with ten reporting the primary outcome. The quality assessment showed a high risk of bias in several papers, predominantly due to non-objective methods of assessment of oesophageal stricture and the non-prospective, non-randomised nature of the studies. Overall, 1395 patients were evaluated, of which 753 received acid suppression medication. Meta-analysis revealed a trend towards increased odds of anastomotic strictures in infants receiving prophylactic medication, but this was not statistically significant (OR 1.33; 95% CI 0.92, 1.92). No significant differences were found in secondary outcomes.
CONCLUSIONS
This meta-analysis found no evidence of a statistically significant link between the prophylactic prescribing of acid suppression medication and the risk of developing anastomotic stricture after OA repair. The literature in this area is limited to observational studies and a randomised controlled trial is recommended to explore this question.
LEVEL OF EVIDENCE
Level III.
Topics: Infant; Humans; Esophageal Atresia; Constriction, Pathologic; Tracheoesophageal Fistula; Esophageal Stenosis; Postoperative Complications; Gastroesophageal Reflux; Anastomosis, Surgical; Randomized Controlled Trials as Topic; Observational Studies as Topic
PubMed: 37355433
DOI: 10.1016/j.jpedsurg.2023.05.024 -
Endoscopic interventional therapies for tracheoesophageal fistulas in children: A systematic review.Frontiers in Pediatrics 2023An electronic literature search was performed using the keywords "tracheoesophageal fistula," "endoscopic," and "children" in the four major medical databases (Ovid,... (Review)
Review
METHODS
An electronic literature search was performed using the keywords "tracheoesophageal fistula," "endoscopic," and "children" in the four major medical databases (Ovid, Embase, PubMed, and Web of Science) right from inception to September 2022. All English language articles describing the endoscopic interventional therapies of TEF in children were reviewed. Two independent researchers screened eligible articles at the title and abstract level. Full texts of potentially relevant articles were then screened again, and reference lists were screened manually to identify additional studies. Relevant data were extracted and analyzed. A synthesis of the relevant data was presented in descriptive form because of the heterogeneity of the included articles. The Chi-Squared test was used with a significance level of 5% (< 0.05).
RESULTS
Among the 1,167 retrieved papers, a total of 46 studies describing 170 TEF patients with an age range of 0.3-175 months were included, including 11 cases of acquired tracheoesophageal fistula, 144 cases of recurrent tracheoesophageal fistula, and 15 cases of congenital tracheoesophageal fistula (H-type TEF). A total of 119 out of 170 fistulas were successfully blocked endoscopic techniques with an overall success rate of 70.0%, while 48 fistulas failed to close by endoscopic interventions, following which the procedure was converted to open surgery. No obviously severe intraoperative/postoperative complications occurred during the follow-up period, but only a mild esophageal stricture was noticed in six patients and grade II tracheal stenosis in one patient. Two patients died from causes unrelated to endoscopic procedures, with a mortality rate of approximately 1.2%. A comparative assessment of different endoscopic interventional techniques for TEF that detected endotracheal stenting was performed in six patients and one fistula was successfully blocked (16.7%). De-epithelialization alone was performed in 65 patients and the fistula healed in 47 of them (72.3%), with the mean number of successful treatments required being 2.3 times. Chemical sealant injection was administered in 33 patients and success was achieved in 21 (63.6%). The average requirement for endoscopic procedures was 1.5 times. De-epithelialization, in combination with chemical sealant injection, was performed in 62 patients, achieving the highest success rate of 77.4% (48 patients). Other treatment methods were performed in four patients and successfully treatment outcomes were reported in two of them (50.0%). The mean number of successful treatments required was four times, and a treatment was converted to surgery in one patient (25.0%). An assessment of different TEF types showed that 9 out of 15 congenital TEFs, 7 out of 11 acquired TEFs, and 103 out of 144 recurrent TEFs were successfully occluded. A comparison of the success rate across multiple groups showed a significant difference with a score of < 0.05, while there was no significant difference in the success rate of different TEF-type groups (> 0.05).
CONCLUSION
Endoscopic intervention is currently a preferred treatment modality for children with TEF because of its less-invasive nature, less complications, and high success rate. Among all interventional techniques, de-epithelialization, in combination with chemical sealant, has a higher success rate than other techniques. However, due to the limited number of cases reported for implementing many kinds of techniques, an ideal endoscopic interventional technique has yet to be devised, often necessitating more treatment applications and close follow-up.
PubMed: 36911034
DOI: 10.3389/fped.2023.1121803 -
Archives of Plastic Surgery Sep 2022Tracheoesophageal puncture (TEP) and voice prosthesis insertion following laryngectomy may fail to form an adequate seal. When spontaneous closure of the fistula tract...
Tracheoesophageal puncture (TEP) and voice prosthesis insertion following laryngectomy may fail to form an adequate seal. When spontaneous closure of the fistula tract does not occur after conservative measures, surgical closure is required. The purpose of this study was to summarize the available evidence on surgical methods for TEP site closure. A comprehensive search across PubMed, Web of Science, SCOPUS, and Cochrane was performed to identify studies describing surgical techniques, outcomes, and complications for TEP closure. We evaluated the rate of unsuccessful TEP closure after surgical management. A meta-analysis with a random-effect method was performed. Thirty-four studies reporting on 144 patients satisfied inclusion criteria. The overall incidence of an unsuccessful TEP surgical closure was 6% (95% confidence interval [CI] 1-13%). Subgroup analysis showed an unsuccessful TEP closure rate for silicone button of 8% (95% CI < 1-43%), 7% (95% CI < 1-34%) for dermal graft interposition, < 1% (95% CI < 1-37%) for radial forearm free flap, < 1% (95% CI < 1-52%) for ligation of the fistula, 17% (95% CI < 1-64%) for interposition of a deltopectoral flap, 9% (95% CI < 1-28%) for primary closure, and 2% (95% CI < 1-20%) for interposition of a sternocleidomastoid muscle flap. Critical assessment of the reconstructive modality should take into consideration previous history of surgery or radiotherapy. Nonirradiated fields and small defects may benefit from fistula excision and tracheal and esophageal multilayer closure. In cases of previous radiotherapy, local flaps or free tissue transfer yield high successful TEP closure rates. Depending on the defect size, sternocleidomastoid muscle flap or fasciocutaneous free flaps are optimal alternatives.
PubMed: 36159387
DOI: 10.1055/s-0042-1756347 -
Frontiers in Pediatrics 2022Esophageal atresia (EA) with tracheoesophageal fistula (TEF) is a common congenital anomaly. It is still unknown whether azygos vein preservation will increase the...
BACKGROUND
Esophageal atresia (EA) with tracheoesophageal fistula (TEF) is a common congenital anomaly. It is still unknown whether azygos vein preservation will increase the difficulty or time of operation and reduce the quality of anastomosis. Thus, we conducted this meta-analysis to explore the puzzle.
METHODS
Two researchers independently searched the databases. Randomized controlled trials were included if these studies applied thoracotomy to perform operations and compared the outcomes in patients with EA/TEF between azygos vein preservation groups and azygos vein ligation groups. The Jadad score was used to assess the quality of the included studies. Statistical heterogeneity was evaluated using the value. A fixed or random-effect model was applied regarding the value.
RESULTS
Four studies involving 286 patients were included. The pooled estimates indicated that preservation of the azygos vein decreased the incidence of anatomic leakage with a pooled risk ratio (RR) of 0.54 (95% CI 0.29-0.99, = 0.05) and mortality with an RR of 0.51 (95% CI 0.29-0.90; = 0.02). Preservation of the azygos vein might not require a longer operative time than ligation of the azygos vein.
CONCLUSIONS
This research certifies that preservation of the azygos vein is able to reduce the prevalence of anastomotic leakage and mortality.
PubMed: 35967577
DOI: 10.3389/fped.2022.965275 -
Frontiers in Pediatrics 2022Esophageal atresia is corrected surgically by anastomosing and recreating esophageal continuity. To allow the removal of excess fluid and air from the anastomosis, a...
BACKGROUND
Esophageal atresia is corrected surgically by anastomosing and recreating esophageal continuity. To allow the removal of excess fluid and air from the anastomosis, a prophylactic and temporary intraoperative chest tube (IOCT) has traditionally been placed in this area during surgery. However, whether the potential benefits of this prophylactic IOCT overweigh the potential harms is unclear.
OBJECTIVE
To assess the benefits and harms of using a prophylactic IOCT during primary surgical repair of esophageal atresia.
DATA SOURCES
We conducted a systematic review with a meta-analysis. We searched Cochrane Central Register of Controlled Trials (2021, Issue 12), MEDLINE Ovid, Embase Ovid, CINAHL, and Science Citation Index Expanded and Conference Proceedings Citation Index-(Web of Science). Search was performed from inception until December 3rd, 2021.
STUDY SELECTION
Randomized clinical trials (RCT) assessing the effect of a prophylactic IOCT during primary surgical repair of esophageal atresia and observational studies identified during our searches for RCT.
DATA EXTRACTION AND SYNTHESIS
Two independent reviewers screened studies and performed data extraction. The certainty of the evidence was assessed by GRADE and ROBINS-I.
PROSPERO REGISTRATION
A protocol for this review has been registered on PROSPERO (CRD42021257834).
RESULTS
We included three RCTs randomizing 162 neonates, all at overall "some risk of bias." The studies compared the placement of an IOCT vs. none. The meta-analysis did not identify any significant effect of profylacitic IOCT, as confidence intervals were compatible with no effect, but the analyses suggests that the placement of an IOCT might lead to an increase in all-cause mortality (RR 1.66, 95% CI 0.76-3.65; three trials), serious adverse events (RR 1.08, 95% CI 0.58-2.00; three trials), intervention-requiring pneumothorax (RR 1.65, 95% CI 0.28-9.50; two trials), and anastomosis leakage (RR 1.66, 95% CI 0.63-4.40). None of our included studies assessed esophageal stricture or pain. Certainty of evidence was very low for all outcomes.
CONCLUSIONS
Evidence from RCTs does not support the routine use of a prophylactic IOCT during primary surgical repair of esophageal atresia.
PubMed: 35372168
DOI: 10.3389/fped.2022.849992 -
Radiation Oncology (London, England) Mar 2022There is currently no standard treatment for locoregional recurrence of esophageal squamous cell carcinoma (ESCC) previously treated with radiotherapy. This study aimed... (Meta-Analysis)
Meta-Analysis
BACKGROUND
There is currently no standard treatment for locoregional recurrence of esophageal squamous cell carcinoma (ESCC) previously treated with radiotherapy. This study aimed to assess the efficacy and safety of re-irradiation for ESCC patients with locoregional recurrence.
METHODS
The PubMed, EmBase, and Cochrane library databases were systematically searched for eligible studies published before January 2021. The pooled effect estimates were calculated using the random effects model. Subgroup analyses were conducted to assess the treatment effectiveness of re-irradiation based on specific characteristics.
RESULTS
Nine retrospective studies including 573 ESCC patients with locoregional recurrence were selected. The pooled incidences of the 1-year, 2-year, 3-year, and 5-year survival for patients after re-irradiation were 59% (95% confidence interval [CI]: 35-83; P < 0.001), 25% (95% CI: 16-33; P < 0.001), 25% (95% CI: 4-45; P = 0.017), and 15% (95% CI: 2-27; P = 0.024), respectively. The rates of complete response and local re-recurrence after re-irradiation were 54% (95% CI: 21-88; P = 0.001) and 62% (95% CI: 55-70; P < 0.001), respectively. The median overall survival and local failure-free survival for patients after re-irradiation were 13.94 months (95% CI: 4.18-46.51; P < 0.001) and 11.01 months (95% CI: 5.99-20.22; P < 0.001), respectively. Grade ≥ 3 adverse events of esophageal perforation, tracheoesophageal fistula, and radiation pneumonitis were significantly more common after re-irradiation.
CONCLUSIONS
This study found that re-irradiation for ESCC patients with locoregional recurrence after previous radiotherapy was feasible. However, patients should be carefully observed in order to treat associated adverse events, including esophageal perforation, tracheoesophageal fistula, and radiation pneumonitis.
Topics: Esophageal Neoplasms; Esophageal Squamous Cell Carcinoma; Humans; Neoplasm Recurrence, Local; Re-Irradiation; Retrospective Studies
PubMed: 35346285
DOI: 10.1186/s13014-022-02019-0 -
Pediatric Surgery International May 2021H type tracheoesophageal fistula (H-TEF) is a rare congenital anomaly. Management may be complicated by late diagnosis and variation(s) in the therapeutic strategy. A...
BACKGROUND
H type tracheoesophageal fistula (H-TEF) is a rare congenital anomaly. Management may be complicated by late diagnosis and variation(s) in the therapeutic strategy. A systematic review of published studies explores the utility of diagnostic studies, operations and postoperative complications.
METHODS
Medline and PubMed database(s) were searched for ALL studies reporting H-TEF during 1997-2020. Using PRISMA methodology, manuscripts were screened for eligibility and reporting.
RESULTS
Forty-seven eligible studies were analysed. Primary diagnosis varied widely with surgeons performing oesophagography and trachea-bronchoscopy. Preoperative localisation techniques included fluoroscopy, guidewire placement and catheterisation. A cervical approach (209 of 272 cases), as well as thoracotomy, thoracoscopy and endoscopic fistula ligation, were all described. Morbidity included fistula recurrence (1.7%), leak (2%), tracheomalacia (3.4%) and respiratory sequelae (1%). The major adverse complication in all studies was vocal cord palsy secondary to laryngeal nerve injury (18.5%) yet strikingly few centres routinely reported undertaking vocal cord screening pre or postoperatively.
CONCLUSION
This study shows that paediatric surgeons record low volume activity with H type tracheoesophageal fistula. Variation(s) in clinical practice are widely evident. Laryngeal nerve injury and its subsequent management warrant special consideration. Care pathways may offset attendant morbidity and define 'best practice.'
Topics: Bronchoscopy; Female; Humans; Infant, Newborn; Male; Postoperative Complications; Postoperative Period; Recurrent Laryngeal Nerve Injuries; Retrospective Studies; Thoracoscopy; Thoracotomy; Trachea; Tracheoesophageal Fistula; Tracheomalacia
PubMed: 33474597
DOI: 10.1007/s00383-020-04853-3 -
BMC Pediatrics Sep 2019Congenital bronchopulmonary foregut malformation (CBPFM) is a rare congenital malformation involving both the digestive and respiratory system. Early diagnosis is...
BACKGROUND
Congenital bronchopulmonary foregut malformation (CBPFM) is a rare congenital malformation involving both the digestive and respiratory system. Early diagnosis is difficult, and delayed recognition may result in considerable complications. The aim of the study was to identify and analyze the clinical characteristics and radiological features of CBPFMs.
METHODS
A systematic review was conducted in accordance with PRISMA guidelines. PubMed, Ovid database, EMBASE were searched for relevant publications to identify all published case-reports of CBPFM since 1992. Data about the demography, clinical presentation, pathology, imaging features, treatment and prognosis were collected.
RESULTS
Sixty-one cases were included in our study. Cases were aged from 1 day to 59 years with the majority aged 3 years or younger. The most common type was group III (37.7%), followed by group II (29.5%)group I (27.9%) and group IV (4.9%). The presentations included respiratory distress (32.8%), cough/choking following food intake and other presentations associated respiratory infection. Thirty-eight cases (62.3%) were diagnosed by upper gastrointestinal series (UGI). Misdiagnosis was common. Eight cases (13.1%) of the included cases died.
CONCLUSIONS
Early recognition and extensive delineation of the anatomy of CBPFM are important to correct these anomalies successfully. UGI is the first choice to confirm the abnormal bronchus communicating with the esophagus. Resection of abnormal pulmonary tissue, lobe or even unilateral lung is preferred. Reconstruction procedures are feasible in selected patients.
Topics: Adolescent; Adult; Bronchi; Child; Child, Preschool; Dextrocardia; Esophageal Atresia; Esophagus; Female; Humans; Infant; Infant, Newborn; Lung; Male; Middle Aged; Pulmonary Artery; Stomach; Tracheoesophageal Fistula; Young Adult
PubMed: 31477056
DOI: 10.1186/s12887-019-1686-1 -
F1000Research 2018Oral hormone pregnancy tests (HPTs), such as Primodos, containing ethinylestradiol and high doses of norethisterone, were given to over a million women from 1958 to...
Oral hormone pregnancy tests (HPTs), such as Primodos, containing ethinylestradiol and high doses of norethisterone, were given to over a million women from 1958 to 1978, when Primodos was withdrawn from the market because of concerns about possible teratogenicity. We aimed to study the association between maternal exposure to oral HPTs and congenital malformations. We have performed a systematic review and meta-analysis of case-control and cohort studies that included data from pregnant women and were exposed to oral HPTs within the estimated first three months of pregnancy, if compared with a relevant control group. We used random-effects meta-analysis and assessed the quality of each study using the Newcastle-Ottawa Scale for non-randomized studies. We found 16 case control studies and 10 prospective cohort studies, together including 71 330 women, of whom 4,209 were exposed to HPTs. Exposure to oral HPTs was associated with a 40% increased risk of all congenital malformations: pooled odds ratio (OR) = 1.40 (95% CI 1.18 to 1.66; P<0.0001; I = 0%). Exposure to HPTs was associated with an increased risk of congenital heart malformations: pooled OR = 1.89 (95% CI 1.32 to 2.72; P = 0.0006; I =0%); nervous system malformations OR = 2.98 (95% CI 1.32 to 6.76; P = 0.0109 I = 78%); gastrointestinal malformations, OR = 4.50 (95% CI 0.63 to 32.20; P = 0.13; I = 54%); musculoskeletal malformations, OR = 2.24 (95% CI 1.23 to 4.08; P= 0.009; I = 0%); the VACTERL syndrome (Vertebral defects, Anal atresia, Cardiovascular anomalies, Tracheoesophageal fistula, Esophageal atresia, Renal anomalies, and Limb defects), OR = 7.47 (95% CI 2.92 to 19.07; P < 0.0001; I = 0%). This systematic review and meta-analysis shows that use of oral HPTs in pregnancy is associated with increased risks of congenital malformations.
PubMed: 30631442
DOI: 10.12688/f1000research.16758.2