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Survival Benefit and Persistent Hospitalization Burden After Ventricular Septal Defect Closure in Infants with
Hiroyuki Nagao1, Takamichi Uchiyama2, Shintaro Nemoto3,4
1Department of Pediatrics, Takatsuki General Hospital, Osaka, Japan.
Insights
Ventricular septal defect (VSD) closure improves survival in infants with trisomy 18. However, survivors face significant hospitalizations due to non-cardiac issues, underscoring the need for ongoing multidisciplinary care.
Area of Science:
- Pediatric Cardiology
- Genetics
- Congenital Heart Disease
Background:
- Infants with trisomy 18 often have complex congenital heart defects, including ventricular septal defects (VSDs).
- The management of VSDs in this population presents significant challenges regarding long-term outcomes.
Purpose of the Study:
- To evaluate the impact of VSD closure on long-term survival and hospitalization in infants with trisomy 18.
- To identify factors associated with hospitalization rates post-intervention.
Main Methods:
- Retrospective study of 33 infants with trisomy 18 and VSD (2002-2025).
- Comparison of three management groups: staged VSD closure post-pulmonary artery banding (PAB), PAB alone, and non-surgical management.
- Kaplan-Meier survival analysis and negative binomial regression for hospitalization rates.
Main Results:
- VSD closure was associated with significantly improved long-term survival (log-rank p=0.009).
- Survivors experienced frequent unexpected hospitalizations, primarily due to respiratory and digestive complications.
- Digestive complications showed a trend towards increased admissions (p=0.09) after multivariable adjustment.
Conclusions:
- VSD closure enhances survival for infants with trisomy 18.
- A significant burden of hospitalization persists due to non-cardiac comorbidities, necessitating comprehensive, lifelong multidisciplinary care.
- Detailed parental counseling on the long-term medical needs is crucial.
Abstract:
This retrospective study evaluated the impact of ventricular septal defect (VSD) closure on long-term survival and hospitalization status in infants with trisomy 18. Thirty-three infants with trisomy 18 and VSD (2002-2025) were categorized into three groups: staged VSD closure following pulmonary artery banding (PAB) (n = 7), PAB alone (n = 3), or non-surgical management (n = 23). Median follow-up durations (interquartile range) were 124.6 (75.8-153.1), 17.8 (11.4-18.1), and 17.1 (2.7-59.3) months, respectively. Kaplan-Meier analysis demonstrated significantly better survival in the VSD closure group (log-rank p = 0.009). However, these survivors required frequent unexpected hospitalizations for non-cardiac comorbidities. In univariate negative binomial regression, respiratory and digestive complications were significantly associated with higher unexpected admission rates (incidence rate ratio [IRR] = 1.07, 95% confidence interval [CI]: 1.02-1.12; and IRR = 1.24, 95% CI: 1.08-1.41, respectively; p < 0.01). After multivariable adjustment, only digestive complications showed a notable, though non-significant, trend (IRR = 1.15, 95% CI: 0.98-1.36, p = 0.09). Notably, VSD closure was associated with a higher admission rate in univariate analysis (IRR = 1.95, 95% CI: 0.95-4.00, p = 0.07), but this association disappeared after multivariable adjustment. VSD closure was associated with improved survival in infants with trisomy 18, but a substantial hospitalization burden persisted due to non-cardiac comorbidities. These findings highlight the importance of lifelong multidisciplinary care and detailed parental counseling regarding the expected long-term medical burden following VSD closure.
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