RESUMEN
Background: Minimally invasive surfactant therapy (MIST), a method of surfactant delivery via a thin catheter during spontaneous breathing, is an increasingly popular alternative to intubation and surfactant administration. Recently, purpose-built catheters for MIST received regulatory approval in Canada and became available for use. However, procedural success and user experience with such catheters have not been described. Methods: This retrospective cohort study included neonates who received MIST using purpose-built catheters between January 1, 2021, and March 31, 2022. Two types of purpose-built catheters were used in this period - SurfCath™ and BLEScath™. Procedural success, number of attempts, and adverse events in neonates receiving MIST via the two catheters were compared using chi-square or Fisher's tests. User experience was described using an ease-of-use scale. Results: Thirty-seven neonates met eligibility criteria; 22 received MIST via SurfCath™, whereas 15 received MIST via BLEScath™. Success rates were 91% in SurfCath™ and 93% in BLEScath™ (P> 0.994). Failed attempts were lower in SurfCath™ (23%) in comparison to BLEScath™ (33%), but the difference was not statistically significant (P=0.708). Among operators, 90% found SurfCath™ very easy/relatively easy to use compared to 43% of users reflecting the same degree of use with BLEScath™ (P=.021). There was no difference in adverse events. Conclusion: This is the first study in Canada to report MIST with purpose-built catheters. Overall, the success rate was equally high with both catheters. Users subjectively reported higher ease of use with SurfCath™. Commercially available purpose-built catheters should facilitate universal adaptation of the MIST method.
RESUMEN
Non-immune hydrops is a prenatal finding which can occur due to an underlying genetic diagnosis such as common chromosomal aneuploidy (Trisomy 21, Turner syndrome etc.). It is extremely rare to have more than one genetic cause of hydrops fetalis in a single pregnancy. This report describes a dichorionic diamniotic pregnancy for a consanguineous couple where noninvasive prenatal testing was "high risk" for Trisomy 21. Family declined amniocentesis and opted for postnatal genetic testing. The pregnancy was later complicated with severe hydrops fetalis leading to demise for one of the twins, and a premature delivery of the other twin who had remarkable collodion not in keeping with Trisomy 21. Postnatal genetic investigations confirmed both Trisomy 21 and prenatal lethal Gaucher disease in the survivor twin. This case report highlights some of the prenatal diagnostic challenges for a consanguineous couple where a rare cause of fetal hydrops was concealed in a setting of a common chromosomal aneuploidy. The prompt postnatal diagnosis of perinatal lethal Gaucher disease, confirmed with undetectable glucocerebrosidase enzyme activity, assisted the family in the decision of providing palliative care for their infant who was quickly deteriorating. The importance of postnatal genetic evaluation and its impact on immediate patient management in an NICU setting is emphasized. This dual diagnosis was significant for the couple as it explained pervious pregnancy losses and has important future recurrence risk implications.
