BackgroundDespite improvements in the treatment of esophageal atresia with tracheoesophageal fistula (TEF) in recent decades, complications still exist. The rate of fistula recanalization after surgical repair is ~5% in large cohorts. However, there is controversy regarding the gold standard of treatment. This research aimed to evaluate the efficacy of treatment of pediatric patients with isolated (H-type) and recurrent TEF in our clinic.MethodsWe retrospectively analyzed 12 patients (7 boys, 5 girls) aged 35 days–14.6 years. The median age of our patients was 632 days [95% CI (confident interval) 120.1 to 2118.7]; the mean birth weight was 2713 g (95% CI 2258 to 3169; median: 2763 g); and the mean gestational age was 37.1 weeks (95% CI 35.4 to 38.8; median: 37 weeks). All patients were managed for isolated or recurrent TEF between January 1, 2015 and December 31, 2020 using endoscopy (laser de-epithelialization).ResultsLaser de-epithelialization alone was effective in 8 of 12 patients (66.67%), with a mean number of de-epithelializations of 2.25 (range: 1–4). After one attempt at de-epithelialization, success was achieved in only two patients (n=16.67%). The mortality rate was 0%. The median follow-up for patients who received endoscopic treatment exclusively (n=8) was 3.7 years (95% CI 1.38 to 4.87) after the last stage of de-epithelialization.ConclusionFlexible endoscopy is an alternative treatment to open surgical repair of isolated and recurrent TEF in children. The effectiveness of endoscopic laser de-epithelialization alone with subsequent fistula obliteration was 66.7%, with a median follow-up of 3.7 years.
BACKGROUND
Blue rubber bleb nevus syndrome (BRBNS) is a rare vascular disease, difficult to diagnose and choose a treatment method, especially in young children. There are several limiting factors to the use of enteroscopy for diagnostics and treatment in pediatric patients, in general. The literature on BRBNS cases is limited and presents various therapeutic approaches.
CASE SUMMARY
We present here a case of BRBNS involving a 4-year-old female, whose intestinal venous lesions were successfully treated by endoscopic sclerotherapy and aethoxysklerol foam. Skin lesions, typical for BRBNS, appeared on the 8
th
d of the child’s life and their number increased over the next several months. The child also experienced episodes of critical decrease in hemoglobin level (by as much as 52 g/L) for several years, requiring iron supplementation and several blood transfusions. Video capsule endoscopy revealed numerous vascular formations in the small bowel. The combined findings of gastrointestinal venous formations and skin lesions prompted BRBNS diagnosis. Single-balloon enteroscopy was used to perform sclerotherapy, with aethoxysklerol foam. A positive effect was observed within 19 mo of follow-up. We continue to monitor the patient’s hemoglobin level, every 2 wk, and it has remained satisfactory (> 120 g/L).
CONCLUSION
Endoscopic sclerotherapy can be effective in the clinical management of gastrointestinal manifestations of BRBNS in young children.
scite is a Brooklyn-based organization that helps researchers better discover and understand research articles through Smart Citations–citations that display the context of the citation and describe whether the article provides supporting or contrasting evidence. scite is used by students and researchers from around the world and is funded in part by the National Science Foundation and the National Institute on Drug Abuse of the National Institutes of Health.