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J Thorac Cardiovasc Surg 2007;134:772-779
© 2007 The American Association for Thoracic Surgery

Surgery for Congenital Heart Disease

Two-year general and neurodevelopmental outcome after neonatal complex cardiac surgery in patients with deletion 22q11.2: A comparative study

Joseph Atallah, MDa, Ari R. Joffe, MDa,*, Charlene M.T. Robertson, MDa,c, Norma Leonard, MDb, Patricia M. Blakley, MD, PhDd, Alberto Nettel-Aguirre, PhDe,f, Reg S. Sauve, MDe, David B. Ross, MDg, Ivan M. Rebeyka, MDg the Western Canadian Complex Pediatric Therapies Project Follow-up Grouph

a Departments of Pediatrics
b Medical Genetics
g Surgery
c University of Alberta, Edmonton, Alberta, Canada; Neonatal and Infant Follow-up Clinic
d Glenrose Rehabilitation Hospital, Edmonton, Alberta, Canada; Department of Pediatrics
e University of Saskatchewan, Saskatoon, Saskatchewan, Canada; Department of Pediatrics
f University of Calgary, Calgary, Alberta, Canada; Biostatistician
h Research Methods Team, Faculty of Medicine, University of Calgary, Calgary, Alberta, Canada; D. Moddemann, Winnipeg, Manitoba, Canada; A. Ninan, Regina, Saskatchewan, Canada

Received for publication December 19, 2006; revisions received February 2, 2007; accepted for publication March 14, 2007.

* Address for reprints: Ari R. Joffe, MD, FRCP(C), Department of Pediatrics, 3A3.07 Walter C Mackenzie Center, 8440- 112 St, Edmonton, Alberta, Canada T6G 2B7 (Email: ajoffe{at}cha.ab.ca).

Objective: Neonatal complex cardiac surgery carries a significant risk for adverse neurodevelopmental outcome. We hypothesized this risk to be higher in patients with deletion 22q11.2.

Methods: From 1996 to 2004, neonates who had complex cardiac surgery at age 6 weeks or less had multisite, multidisciplinary health and neurodevelopmental outcomes (Bayley Scales of Infant Development II; mental and psychomotor developmental indices [MDI, PDI] as mean [SD] and delay [<70]) assessed at 18 to 24 months of age. All 16 patients with deletion 22q11.2 (group 1) were compared with 16 patients without deletion 22q11.2 (group 2) having undergone neonatal complex cardiac surgery at the same center and matched for cardiac lesion, socioeconomic status, and year of operation. Outcomes were compared by univariate and multivariate analyses.

Results: Heart lesions in each group consisted of 6 (37.5%) cases of interrupted aortic arch, 6 (37.5%) cases of truncus arteriosus, and 4 (25%) cases of tetralogy of Fallot. Outcomes were available for all survivors. Mortality was 3 (19%) and 1 (6%) in groups 1 and 2, respectively (P = .6). MDI and PDI scores were 66.1 (10.6) and 55.0 (9.4) for group 1 and 86.3 (14.6) and 82.3 (14.3) for group 2 (P < .001). Only deletion 22q11.2 was significant in stepwise multiple regression to predict MDI and PDI scores. Mental delay occurred in 8 (61.5%) of 13 in group 1 and 2 (13.8%) of 15 in group 2 (P = .016). Psychomotor delay occurred in 11 (84.6%) of 13 in group 1 and 1 (6.7%) of 15 in group 2 (P < .001).

Conclusion: Neonates affected by deletion 22q11.2 and having neonatal complex cardiac surgery have significantly worse neurodevelopmental outcome than do those without deletion 22q11.2.

Abbreviations and Acronyms CCS = complex cardiac surgery; CHD = congenital heart disease; del22q11.2 = deletion 22q11.2; IAA = interrupted aortic arch; IQ = intelligence quotient; MDI = mental developmental index; PA = pulmonary atresia; PDI = psychomotor developmental index; SD = standard deviation; TA = truncus arteriosus; TOF = tetralogy of Fallot; VSD = ventricular septal defect




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