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J Thorac Cardiovasc Surg 2009;138:374-381
© 2009 The American Association for Thoracic Surgery

Congenital Heart Disease

Subtle hemorrhagic brain injury is associated with neurodevelopmental impairment in infants with repaired congenital heart disease

Janet S. Soul, MD, CMa,f,*, Richard L. Robertson, MDb,g, David Wypij, PhDc,h,k, David C. Bellinger, PhD, MSca,f, Karen J. Visconti, PhDe, Adré J. du Plessis, MBChB, MPHa,f, Barry D. Kussman, MBBChd,i, Lisa A. Scoppettuolo, MSc,k, Frank Pigula, MDe,j, Richard A. Jonas, MDe,j, Jane W. Newburger, MD, MPHc,h

a Department of Neurology, at Children's Hospital Boston, Boston, Mass
b Department of Radiology, at Children's Hospital Boston, Boston, Mass
c Department of Cardiology, at Children's Hospital Boston, Boston, Mass
d Department of Anesthesiology, at Children's Hospital Boston, Boston, Mass
e Department of Cardiovascular Surgery, at Children's Hospital Boston, Boston, Mass
f Department of Neurology, at Harvard Medical School, Boston, Mass
g Department of Radiology, at Harvard Medical School, Boston, Mass
h Department of Pediatrics, at Harvard Medical School, Boston, Mass
i Department of Anesthesia, at Harvard Medical School, Boston, Mass
j Department of Surgery, at Harvard Medical School, Boston, Mass
k Department of Biostatistics, Harvard School of Public Health, Boston, Mass

Received for publication August 1, 2008; revisions received November 13, 2008; accepted for publication February 9, 2009.

* Address for reprints: Janet S. Soul, MD, CM, Department of Neurology, Children's Hospital Boston, 300 Longwood Avenue, Boston, MA 02115. (Email: janet.soul{at}childrens.harvard.edu).

Objective: Perioperative stroke and periventricular leukomalacia have been reported to occur commonly in infants with congenital heart disease. We aimed to determine the incidence and type of brain injury in infants undergoing 2-ventricle repair in infancy and to determine risk factors associated with such injury.

Methods: Forty-eight infants enrolled in a trial comparing 2 different hematocrits during surgical repair of congenital heart disease underwent brain magnetic resonance imaging scans and neurodevelopmental testing at 1 year of age.

Results: Eighteen (38%) of our subjects had tiny foci of hemosiderin by susceptibility imaging, without evidence of abnormalities in corresponding regions on conventional magnetic resonance imaging sequences. Subjects with foci of hemosiderin had a significantly lower Psychomotor Developmental Index at 1 year of age (79.6 ± 16.5, mean ± standard deviation) compared with subjects without these foci (89.5 ± 15.3; P = .04). Older age at surgery and diagnostic group were significantly associated with the presence of hemosiderin foci. Only 1 subject had a small stroke (2%), and 2 subjects had periventricular leukomalacia (4%).

Conclusion: Foci of hemosiderin without radiologic evidence of ischemic brain injury are an abnormality associated with adverse neurodevelopmental outcome not previously described in magnetic resonance imaging studies of children with surgically repaired congenital heart disease. The association of hemosiderin foci with older age at surgery and cardiac diagnosis, and not with risk factors associated with brain injury, in previous studies suggests that the cause and pathogenesis of this abnormality are different from ischemic brain lesions reported previously.

Abbreviations and Acronyms BAS = balloon atrial septostomy; BSID-II = Bayley Scales of Infant Development II; CHD = congenital heart disease; MDI = Mental Development Index; MPGR = multiplanar gradient recalled; PDI = Psychomotor Development Index; PVL = periventricular leukomalacia; TGA = D-transposition of the great arteries; TOF = tetralogy of Fallot; VSD = ventricular septal defect






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