Ashok Panigrahy1, Vincent Lee2, Rafael Ceschin3, Giulio Zuccoli2, Nancy Beluk2, Omar Khalifa4, Jodie K Votava-Smith5, Mark DeBrunner6, Ricardo Munoz7, Yuliya Domnina7, Victor Morell8, Peter Wearden8, Joan Sanchez De Toledo7, William Devine4, Maliha Zahid4, Cecilia W Lo4. 1. Department of Pediatric Radiology, Childrens Hospital of Pittsburgh of University of Pittsburgh Medical Center and University of Pittsburgh School of Medicine, Pittsburgh, PA; Department of Biomedical Informatics, University of Pittsburgh, Pittsburgh, PA. Electronic address: panigrahya@upmc.edu. 2. Department of Pediatric Radiology, Childrens Hospital of Pittsburgh of University of Pittsburgh Medical Center and University of Pittsburgh School of Medicine, Pittsburgh, PA. 3. Department of Pediatric Radiology, Childrens Hospital of Pittsburgh of University of Pittsburgh Medical Center and University of Pittsburgh School of Medicine, Pittsburgh, PA; Department of Biomedical Informatics, University of Pittsburgh, Pittsburgh, PA. 4. Department of Developmental Biology, University of Pittsburgh School of Medicine, Pittsburgh, PA. 5. Department of Pediatrics, Division of Cardiology, Childrens Hospital of Los Angeles, Los Angeles, CA. 6. Division of Pediatric Cardiology, Department of Pediatrics, University of Pittsburgh School of Medicine, Pittsburgh, PA. 7. Cardiac Intensive Care Division, University of Pittsburgh School of Medicine, Pittsburgh, PA. 8. Division of Pediatric Cardiothoracic Surgery, Department of Surgery, University of Pittsburgh School of Medicine, Pittsburgh, PA.
Abstract
OBJECTIVE: To test for associations between abnormal respiratory ciliary motion (CM) and brain abnormalities in infants with congenital heart disease (CHD) STUDY DESIGN: We recruited 35 infants with CHD preoperatively and performed nasal tissue biopsy to assess respiratory CM by videomicroscopy. Cranial ultrasound scan and brain magnetic resonance imaging were obtained pre- and/or postoperatively and systematically reviewed for brain abnormalities. Segmentation was used to quantitate cerebrospinal fluid and regional brain volumes. Perinatal and perioperative clinical variables were collected. RESULTS: A total of 10 (28.5%) patients with CHD had abnormal CM. Abnormal CM was not associated with brain injury but was correlated with increased extraaxial cerebrospinal fluid volume (P < .001), delayed brain maturation (P < .05), and a spectrum of subtle dysplasia including the hippocampus (P < .0078) and olfactory bulb (P < .034). Abnormal CM was associated with higher composite dysplasia score (P < .001), and both were correlated with elevated preoperative serum lactate (P < .001). CONCLUSIONS: Abnormal respiratory CM in infants with CHD is associated with a spectrum of brain dysplasia. These findings suggest that ciliary defects may play a role in brain dysplasia in patients with CHD and have the potential to prognosticate neurodevelopmental risks.
OBJECTIVE: To test for associations between abnormal respiratory ciliary motion (CM) and brain abnormalities in infants with congenital heart disease (CHD) STUDY DESIGN: We recruited 35 infants with CHD preoperatively and performed nasal tissue biopsy to assess respiratory CM by videomicroscopy. Cranial ultrasound scan and brain magnetic resonance imaging were obtained pre- and/or postoperatively and systematically reviewed for brain abnormalities. Segmentation was used to quantitate cerebrospinal fluid and regional brain volumes. Perinatal and perioperative clinical variables were collected. RESULTS: A total of 10 (28.5%) patients with CHD had abnormal CM. Abnormal CM was not associated with brain injury but was correlated with increased extraaxial cerebrospinal fluid volume (P < .001), delayed brain maturation (P < .05), and a spectrum of subtle dysplasia including the hippocampus (P < .0078) and olfactory bulb (P < .034). Abnormal CM was associated with higher composite dysplasia score (P < .001), and both were correlated with elevated preoperative serum lactate (P < .001). CONCLUSIONS:Abnormal respiratory CM in infants with CHD is associated with a spectrum of brain dysplasia. These findings suggest that ciliary defects may play a role in brain dysplasia in patients with CHD and have the potential to prognosticate neurodevelopmental risks.
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