Long-term Neurodevelopmental Impairment in Neonates Surgically Treated for Necrotizing Enterocolitis: Enterostomy Associated with a Worse Outcome

Abstract

Background: Necrotizing enterocolitis (NEC) patients often have neurodevelopmental impairment, but the long-term follow-up data is limited. We determined whether surgical factors were of prognostic value for long-term neurodevelopment in children surviving surgery for NEC (SNEC).

Patients and methods: SNEC patients born between 1996 and 2002 were tested for verbal (VIQ), performance (PIQ) and total (TIQ) intelligence using Wechsler's Intelligence Scale for Children, Third Edition, Dutch Version, and motor skills using the Movement Assessment Battery for Children (M-ABC). Neonatal and surgical data were obtained retrospectively to assess prognostic factors.

Results: 19 patients (12 boys), median age 9.9 years (range 6.2–13.1), gestational age 31.0 weeks (range 25.2–40), birth weight 1 250 g (range 780–3 175) were evaluated. Infants with an enterostomy (n=14) scored lower on intelligence than children with a primary anastomosis (n=5): VIQ 85±12 vs. 101±15, p=0.04; PIQ 79±13 vs. 92±11, p=0.06; TIQ 82±11 vs. 97±13, p=0.04. Motor skills were either suspect or clinically impaired in 74%. Clinical classification of results suggest more children in the enterostomy group had a performance rated as clinically impaired compared to the primary anastomosis group, although no statistical difference in M-ABC score was found. There were no differences between primary anastomosis and enterostomy patients with regard to gestational age, birth weight, comorbidities, preoperative Bell stage, residual small and large bowel lengths, inotropic medication, duration of ventilatory support, NICU and hospital stay, and physical exam data on follow-up. However, a selection bias could not be ruled out.

Conclusions: The results suggest that an enterostomy in SNEC patients could be associated with worse neurodevelopmental outcomes by the age of 6–13 years compared to a primary anastomosis, although the severity of illness was comparable between both groups. Further studies are needed to prevent selection bias and to elucidate the impact of abdominal surgical factors on neurodevelopmental outcome and the underlying pathophysiology.

Key words

necrotizing enterocolitis - neurodevelopmental impairment - primary anastomosis - enterostomy - gastrointestinal surgery

References

• 1 Ladd AP, Rescorla FJ, West KW. et al . Long-term follow-up after bowel resection for necrotizing enterocolitis: factors affecting outcome.  J Pediatr Surg. 1998l;  33 967-972
• 2 Lin PW, Stoll BJ. Necrotising enterocolitis.  Lancet. 2006;  368 1271-1283
• 3 Ricketts RR, Jerles ML. Neonatal necrotizing enterocolitis: experience with 100 consecutive surgical patients.  World J Surg. 1990;  14 600-605
• 4 Hofman FN, Bax NM, van der Zee DC. et al . Surgery for necrotising enterocolitis: primary anastomosis or enterostomy?.  Pediatr Surg Int. 2004;  20 481-483
• 5 Rees CM, Pierro A, Eaton S. Neurodevelopmental outcomes of neonates with medically and surgically treated necrotizing enterocolitis.  Arch Dis Child Fetal Neonatal Ed. 2007;  92 F193-F198
• 6 Hintz SR, Kendrick DE, Stoll BJ. et al . Neurodevelopmental and growth outcomes of extremely low birth weight infants after necrotizing enterocolitis.  Pediatrics. 2005;  115 696-703
• 7 Stevenson DK, Kerner JA, Malachowski N. et al . Late morbidity among survivors of necrotizing enterocolitis.  Pediatrics. 1980;  66 925-927
• 8 Mikkola K, Ritari N, Tommiska V. et al . Neurodevelopmental outcome at 5 years of age of a national cohort of extremely low birth weight infants who were born in 1996–1997.  Pediatrics. 2005;  116 1391-1400
• 9 Blakely ML, Tyson JE, Lally KP. et al . Laparotomy vs. peritoneal drainage for necrotizing enterocolitis or isolated intestinal perforation in extremely low birth weight infants: outcomes through 18 months adjusted age.  Pediatrics. 2006;  117 e680-e687
• 10 O’Brien F, Roth S, Stewart A. et al . The neurodevelopmental progress of infants less than 33 weeks into adolescence.  Arch Dis Child. 2004;  89 207-211
• 11 Mu SC, Lin CH, Chen YL. et al . Relationship between perinatal and neonatal indices and intelligence quotient in very low birth weight infants at the age of 6 or 8 years.  Pediatr Neonatol. 2008;  49 13-18
• 12 Walsh MC, Kliegman RM, Hack M. Severity of necrotizing enterocolitis: influence on outcome at 2 years of age.  Pediatrics. 1989;  84 808-814
• 13 Struijs MC, Diamond IR, de Silva N. et al . Establishing norms for intestinal length in children.  J Pediatr Surg. 2009;  44 933-938
• 14 Wechsler D, Schuttehatte M, Dekker P. et al .Wechsler Intelligence Scale for Children, Third Version: Handleiding en verantwoording. 1st ed. Amsterdam: Harcourt Assessment BV; 2001
• 15 Grégoire J. Comparison of 3 Short Forms of the Wechsler Intelligence Scale for Children – Third Edition (WISC-III). Eur Rev of Applied Psychology. 2000 50: 437-441
• 16 Smits-Engelsman B. Movement Assessment Battery for Children: Handleiding. 1st ed. Amsterdam: Harcourt Assessment BV; 1992
• 17 Walsh MC, Kliegman RM, Hack M. Severity of necrotizing enterocolitis: influence on outcome at 2 years of age.  Pediatrics. 1989;  84 808-814
• 18 Koster van Groos GAS. Beknopte handleiding bij de diagnostische criteria van de DSM-IV-TR. 3rd ed. Amsterdam: Harcourt Book Publishers; 2007: 89
• 19 Merritt TA, Pillers D, Prows SL. Early NICU discharge of very low birth weight infants: a critical review and analysis.  Semin Neonatol. 2003;  8 95-115
• 20 Schulzke SM, Deshpande GC, Patole SK. Neurodevelopmental outcomes of very low-birth-weight infants with necrotizing enterocolitis: a systematic review of observational studies.  Arch Pediatr Adolesc Med. 2007;  161 583-590
• 21 Gessler P, Schmitt B, Pretre R. et al . Inflammatory response and neurodevelopmental outcome after open-heart surgery in children.  Pediatr Cardiol. 2009;  30 301-305
• 22 Dror DK, Allen LH. Effect of vitamin B12 deficiency on neurodevelopment in infants: current knowledge and possible mechanisms.  Nutr Rev. 2008;  66 250-255
• 23 Chalouhi C, Faesch S, Anthoine-Milhomme MC. et al . Neurological consequences of vitamin B12 deficiency and its treatment.  Pediatr Emerg Care. 2008;  24 538-541
• 24 Bos AF, Martijn A, van Asperen RM. et al . Qualitative assessment of general movements in high-risk preterm infants with chronic lung disease requiring dexamethasone therapy.  J Pediatr. 1998;  132 300-306
• 25 Cole CR, Hansen NI, Higgins RD. et al . Very low birth weight preterm infants with surgical short bowel syndrome: incidence, morbidity and mortality, and growth outcomes at 18 to 22 months.  Pediatrics. 2008;  122 e573-e582
• 26 Jayaprakash A, Creed T, Stewart L. et al . Should we monitor vitamin B12 levels in patients who have had end-ileostomy for inflammatory bowel.  Int J Colorectal Dis. 2004;  19 316-318
• 27 Nightingale JMD. Management of patients with a short bowel.  World J Gastroenterol. 2001;  7 741-751
• 28 Novak EM, Dyer RA, Innis SM. High dietary omega-6 fatty acids contribute to reduced docosahexaenoic acid in the developing brain and inhibit secondary neurite growth.  Brain Res. 2008;  1237 136-145
• 29 Innis SM. Perinatal biochemistry and physiology of long-chain polyunsaturated fatty acids.  J Pediatr. 2003;  143 S1-S8
• 30 Aguayo P, Fraser JD, Sharp S. et al . Stomal complications in the newborn with necrotizing enterocolitis.  J Surg Res. 2009;  157 275-278
• 31 Fasoli L, Turi RA, Spitz L. et al . Necrotizing enterocolitis: extent of disease and surgical treatment.  J Pediatr Surg. 1999;  34 1096-1099

Correspondence