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Long-Term Outcome After Neonatal Meconium Obstruction

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Long-Term Outcome After Neonatal Meconium Obstruction Long-term Outcome After Neonatal Meconium Obstruction Julie R. Fuchs, MD, and Jacob C. Langer, MD ABSTRACT. Objective. It is unclear whether children meconium ileus and those undergoing resection or enter- with cystic fibrosis (CF) who present with neonatal ostomy. Patients with meconium obstruction who do not meconium ileus have a different long-term outcome from have CF have an excellent long-term prognosis. This those presenting later in childhood with pulmonary com- information will be useful in counseling the families of plications or failure to thrive. We examined a cohort of infants presenting with neonatal meconium obstruction. patients with meconium ileus, and compared their long- Pediatrics 1998;101(4). URL: http://www.pediatrics.org/ term outcome with children who had CF without meco- cgi/content/full/101/4/e7; cystic fibrosis, meconium ileus, nium ileus and neonates who had meconium obstruction meconium plug syndrome. without CF (meconium plug syndrome). Study Design. Comparative study using retrospective and follow-up interview data. ABBREVIATION. CF, cystic fibrosis. Patients. Group 1 consisted of 35 surviving CF pa- tients who presented with meconium ileus between 1966 econium obstruction in the neonate is a and 1992. Two control groups were also studied: 35 age- spectrum of disease that includes meco- and sex-matched CF patients without meconium ileus 1 (group 2), and 12 infants presenting with meconium plug Mnium ileus and meconium plug syndrome. syndrome during the same time period (group 3). Meconium ileus is characterized by extremely viscid, Outcome Measures. Pulmonary, gastrointestinal, nu- protein-rich inspissated meconium causing terminal tritional, and functional status were reviewed, and sur- ileal obstruction, and accounts for approximately gical complications were recorded. 20% of neonatal intestinal obstructions.2 Although Results. Mean follow-up was 12.6 6 6, 12.6 6 6, and meconium ileus can rarely occur in otherwise normal 9.3 6 4 years in groups 1, 2, and 3, respectively. Patients children,3 the majority of patients with meconium without CF (group 3) demonstrated better growth and ileus have cystic fibrosis (CF). Meconium ileus is the functional status, and had a lower incidence of pulmo- presenting symptom in 10% to 15% of these patients.4 nary and gastrointestinal problems. Although the pres- ence of meconium ileus among CF patients was associ- Meconium plug syndrome is more commonly seen ated with an earlier diagnosis, there were no significant in premature infants, is characterized by colonic ob- differences between groups 1 and 2 with respect to hepa- struction, and is only rarely associated with CF. tobiliary, nutritional, functional, or respiratory status. Approximately half of neonates with meconium Meconium ileus was associated with a higher risk of ileus present with uncomplicated intestinal obstruc- meconium ileus equivalent (20% vs 6%), although this tion. In many of these children, the obstruction can difference was not statistically significant. Long-term be relieved using a water-soluble contrast enema, surgical complications (adhesive small bowel obstruc- and surgery is reserved for those who fail nonopera- tion and blind loop syndrome) were seen in 27% of tive management. Patients presenting with compli- children with meconium ileus; there were no long-term cated meconium ileus, including volvulus, atresia, surgical complications in groups 2 or 3, because these gangrene, perforation, and meconium peritonitis al- infants did not have any neonatal surgical procedures. 5 Children presenting with complicated meconium ileus ways require operative intervention. A variety of had a higher rate of long-term surgical complications procedures have been described, including enterot- than those with uncomplicated meconium ileus (36% vs omy and lavage, placement of a T-tube, resection 17%), and those managed with resection or enterostomy with primary anastomosis, and various types of en- had more complications than those treated by enterot- terostomies, including the Bishop-Koop ileostomy, omy and lavage (33% vs 0%). Mikulicz double-barreled enterostomy, Santulli Conclusions. Long-term outcome is similar in CF pa- proximal chimney enterostomy, and the double en- tients who present with meconium ileus and those who terostomy (proximal enterostomy and mucus fistu- do not, except for a slightly higher incidence of meco- la).6 In contrast, virtually all infants with meconium nium ileus equivalent, and a significantly higher rate of surgical complications. The risk of surgical complica- plug syndrome are cured of their obstruction after a tions is highest in those presenting with complicated water-soluble contrast enema. Survival of neonates with meconium ileus has im- proved over the last few decades because of im- From the Department of Surgery, Washington University School of Medi- proved surgical technique, neonatal intensive care, cine, St Louis, Missouri. nutritional support, and medical management of the Received for publication Jun 27, 1997; accepted Jan 5, 1998. pulmonary complications of CF.7 In addition, it is Address correspondence to: Jacob C. Langer, MD, Division of Pediatric increasingly being recognized that CF is a heteroge- Surgery, St Louis Children’s Hospital, Room 5W12, One Childrens Place, St Louis, MO 63110. neous disease. The characteristics and severity of PEDIATRICS (ISSN 0031 4005). Copyright © 1998 by the American Acad- gastrointestinal and pulmonary complications vary emy of Pediatrics. considerably among patients in long-term follow-up http://www.pediatrics.org/cgi/content/full/101/4/Downloaded from www.aappublications.org/newse7 by guestPEDIATRICS on September Vol. 26, 2021 101 No. 4 April 1998 1of6 studies.8 It would be helpful for both families and nium plug syndrome, 3 had died from complications clinicians if it were possible to predict the likelihood of prematurity, 4 did not have confirmed meconium of long-term problems at the time of diagnosis. This plug syndrome, and 9 patients could not be located study was designed to determine whether neonates for long-term follow-up, leaving 12 patients for anal- with CF who present with meconium obstruction ysis in group 3. have a different long-term outcome than those who Characteristics of the three groups are shown in do not. To control for the effect of CF itself on out- Table 1. Gestational age at birth was significantly come, we also sought to determine long-term out- higher in group 2 compared to group 1. Although the come in patients presenting with neonatal meconium same trend was present for birth weight, the differ- obstruction who did not have CF. ence was not statistically significant. Children in group 1 were significantly younger when the diag- MATERIAL AND METHODS nosis of CF was made than those in group 2. In group 1, meconium ileus was uncomplicated in Charts of all newborns presenting with meconium ileus be- 5 5 tween 1966 and 1992 were reviewed (group 1). Two control 60% (n 21) and complicated in 40% (n 14). The groups were also studied: CF patients without meconium ileus complicated cases included patients with proximal (group 2), and infants presenting with meconium plug syndrome volvulus (n 5 7), jejunal atresia (n 5 6), ileal atresia (group 3). Group 2 was generated by randomly matching each of (n 5 2), perforation (n 5 7), meconium peritonitis the meconium ileus patients by sex and age to a patient being 5 5 followed in the CF clinic who had not presented with meconium (n 7), and ascites (n 2). Some cases had multiple ileus. Group 3 included infants with a diagnosis of meconium complications. In group 1, 33 of 35 patients had CF plug syndrome presenting between 1966 and 1992. For the pur- diagnosed by sweat chloride, and the other 2 had poses of this study, obstruction was defined as meconium ileus if meconium ileus with normal sweat chloride levels; 1 the meconium obstruction was ileal, and as meconium plug syn- drome if the obstruction was primarily colonic. of these presented with complicated meconium ileus, For each patient, the hospital and clinic chart (if available) were and the other was uncomplicated. Sweat chlorides reviewed. Information gleaned included sex, date of birth, gesta- were elevated in all group 2 patients and in none of tional age, birth weight, associated anomalies, initial presentation, the group 3 patients. and radiologic or surgical procedure. All patients or families were Of the 21 patients with uncomplicated meconium also contacted by telephone, and all families contacted agreed to participate in the study. Gastrointestinal complaints and abdom- ileus, 9 (43%) were successfully treated by contrast inal symptomatology were discussed in detail, with specific ques- enema alone, and 12 (57%) required surgical inter- tions regarding meconium ileus equivalent, recurrent diarrhea, vention [intestinal resection with Mikulicz ileostomy constipation, abdominal pain, rectal prolapse, and gastrointestinal (n 5 2), intestinal resection and Bishop-Koop ileos- bleeding. Meconium ileus equivalent was strictly defined for our tomy (n 5 3), enterotomy with N-acetylcysteine or purposes as partial or complete intestinal obstruction with evi- 5 dence of impacted feces in the right colon or terminal ileum on saline lavage (n 4), resection with primary anasto- radiographic examination, necessitating treatment by either an mosis (n 5 2), and milking of meconium into the enema or intestinal lavage solution by mouth. Diarrhea, constipa- colon without enterostomy (n 5 1)]. There was one
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