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PubMed2001

Jejunoileal atresia and associated malformations: correlation with the timing of in utero insult.

PURPOSE: Duodenal atresia is associated with a higher incidence of associated congenital malformations than jejunoileal atresia, supporting the hypothesis that the duodenal obstruction occurs early in fetal life. In this study, the authors analyzed the incidence of major associated malformations in jejunal atresia (JA) and ileal atresia (IA) to determine if there is a positive correlation between the proximity of the intestinal atresia and the association of other major anomalies. METHODS: Records of all patients with jejunoileal atresias treated at the authors' institution between 1980 and 1997 were examined. RESULTS: There were 83 patients with jejunoileal atresias, 38 with JA, and 45 with IA. Sixteen (42%) of the JA patients had an associated major congenital malformation, whereas only 1 (2%) of the IA patients had an associated malformation. A single atresia was found in 18 (47%) of JA patients and 41 (91%) of IA patients. Twenty (53%) of the JA patients had either multiple or apple-peel atresia. Thirteen patients (16%) died, 11 with JA, and 2 with IA. Of the 11 patients with JA who died, 6 had multiple atresias, 4 had cystic fibrosis, and 1 had small bowel volvulus. CONCLUSION: The higher incidence of associated major congenital extraintestinal malformations in JA compared with IA patients suggests that some cases of JA may arise from a malformative process.

Abnormalities, Multiple↗
PubMedDate not supplied

Ninety-five cases of intestinal transplantation at the University of Miami.

Intestinal failure requiring total parenteral nutrition (TPN) is associated with significant morbidity and mortality. Intestinal transplantation can be a lifesaving option for patients with intestinal failure who develop serious TPN-related complications. The aim of this study was to evaluate survival, surgical technique, and patient care in patients treated with intestinal transplantation. We reviewed data collected from 95 consecutive intestinal transplants performed between December 1994 and November 2000 at the University of Miami. Fifty-four of the patients undergoing intestinal transplantation were children and 41 were adults. The series includes 49 male and 46 female patients. The causes of intestinal failure included mesenteric venous thrombosis (n = 12), necrotizing enterocolitis (n = 11), gastroschisis (n = 11), midgut volvulus (n = 9), desmoid tumor (n = 8), intestinal atresia (n = 6), trauma (n = 5), Hirschsprung's disease (n = 5), Crohn's disease (n = 5), intestinal pseudoobstruction (n = 4), and others (n = 19). The procedures performed included 27 isolated intestine transplants, 28 combined liver and intestine transplants, and 40 multivisceral transplants. Since 1998, we have been using daclizumab (Zenepax) for induction of immunosuppression and zoom videoendoscopy for graft surveillance. We began to use intense cytomegalovirus prophylaxis and systemic drainage of the portal vein. The 1-year patient survival rates for isolated intestinal, liver and intestinal, and multivisceral transplantations were 75%, 40%, and 48%, respectively. Since 1998, the 1-year patient and graft survival rates for isolated intestinal transplants have been 84% and 72%, respectively. The causes of death were as follows: sepsis after rejection (n = 14), respiratory failure (n = 8), sepsis (n = 6), multiple organ failure (n = 4), arterial graft infection (n = 3), aspergillosis (n = 2), post-transplantation lymphoproliferative disease (n = 2), intracranial hemorrhage (n = 2), and fungemia, chronic rejection, graft vs. host disease, necrotizing enterocolitis, pancreatitis, pulmonary embolism, and viral encephalitis (n = 1 case of each). Intestinal transplantation can be a lifesaving alternative for patients with intestinal failure. The prognosis after intestinal transplantation is better when it is performed before the onset of liver failure. Rejection monitoring with zoom videoendoscopy and new immunosuppressive therapy with sirolimus, daclizumab, and campath-1H have contributed to the improvement in patient survival.

Adolescent↗
PubMed2001

Analysis of patients with longitudinal intestinal lengthening procedure referred for intestinal transplantation.

BACKGROUND/PURPOSE: Longitudinal intestinal lengthening procedures (LILP) in patients with short gut syndrome (SGS) enhances small intestinal peristalsis and decreases bacterial overgrowth without reducing absorptive surface. Therefore, patients theoretically may be easily weaned off TPN. The aim of this study was to evaluate the impact of failed LILP in SGS patients referred for intestinal transplantation. METHODS: Twenty-seven (11%) of 230 children with SGS and total parenteral nutrition (TPN) dependency evaluated for intestinal transplantation at our institution had undergone LILP. This was performed at a mean age of 1.7 years (range, 1 day to 14.7 years); the mean age at the time of evaluation was 3.3 years (range, 0.4 to 17 years). Two patients underwent LILP immediately after birth. The principle diagnoses producing SGS were gastroschisis (n = 8), intestinal atresia (n = 11), neonatal volvulus (n = 7) and necrotizing enterocolitis (n = 1). Before LILP, the mean length of intestine was 32 cm (range, 8 to 70 cm). Fifteen (56%) patients had jaundice at the time of evaluation. RESULTS: All but one child were considered candidates for intestinal transplantation. The mean intestinal length achieved after LILP was 48 cm (range, 16 to 100). The mean follow-up from the date of LILP was 876 days (range, 109 to 4,109 days). After LILP, only 9 (33%) patients increased their caloric intake through the enteral route by > or =50%, and only 1 patient could be weaned off TPN. In the patients with liver dysfunction at the time of LILP, none recovered. Most of the patients had multiple episodes of sepsis after LILP. Fourteen (52%) of 27 patients underwent intestinal transplantation, 7 combined with a liver allograft because of TPN-induced end-stage liver disease. Six of the transplanted patients are alive and TPN free. Of the remaining 13 (48%) nontransplanted patients, 9 patients died. The main cause of death was TPN-induced liver failure. Three patients are on partial TPN, and only 1 patient was weaned off TPN. The presence of an ileocecal valve did not impact on outcome. Surprisingly, patients with > or =50% of colon at the time of LILP had poorer survival than those with less. Twelve (44%) of 27 patients had surgical complications, and in both patients with LILP performed in the neonatal period it failed immediately with acute complications. There were no differences in patient survival rate for patients with SGS without LILP (n = 203) and those with LILP (n = 27). CONCLUSION: Based on patients with unsuccessful LILP referred for intestinal transplantation, we believe this procedure should be avoided in the neonatal period, in those patients with liver dysfunction, and when intestinal length is <50 cm.

Adolescent↗
PubMed1996

Clinical significance of prenatal ultrasonographic intestinal dilatation in fetuses with gastroschisis.

OBJECTIVE: Our purpose was to evaluate the clinical significance of intestinal dilatation detected by prenatal ultrasonographic examination in fetuses with gastroschisis. STUDY DESIGN: A retrospective chart review was performed of all patients cared for at Los Angeles County/University of Southern California Women's and Children's Hospital with the prenatal diagnosis of gastroschisis over a 7-year period (1988 through 1995). Patients were divided into two groups on the basis of the presence or absence of ultrasonographically measured fetal bowel diameter of > or = 17 mm. Neonatal outcomes of the two groups were compared. RESULTS: Twenty-one patients met the entry criteria during the study period. Fetuses with maximal bowel diameter of > or = 17 mm did not have a longer time to full oral feeding, a longer initial hospital stay, or a greater need for bowel resection when compared with fetuses with a bowel diameter < 17 mm. Two newborns underwent bowel resection because of intestinal atresia. Prenatal ultrasonographic examination failed to show significant bowel dilatation in either infant. CONCLUSION: Our data suggest that prenatal evidence of intestinal dilatation in fetuses with gastroschisis does not predict immediate neonatal outcome. Thus this finding is not an appropriate indication for preterm delivery in the absence of other evidence of fetal compromise.

Adult↗
PubMed1986

[Colonic atresia].

Nine cases of colonic atresia, treated in the Departament of Pediatric Surgery of "La Paz" Pediatric Hospital, are reported. They account for 11% of all our intestinal atresias. We review the different etiologic and pathologic theories and classificate our group according to the LOUW and HAYS method. In two cases their digestive anomalies and in one renal anomalies were found to be associated mortality was 11%.

Colon↗
PubMedDate not supplied

Neonatal ileoileocolic intussusception associated with ileal polyp: report of one case.

Neonatal intussusception is an uncommon disease. We report a case of neonatal ileoileocolic intussusception led by an ileal polyp in a female neonate. The patient presented with irritable crying, bilious vomiting and frank bloody stool on the 26th day of life. On physical examination, a mobile abdominal mass was palpated. Abdominal sonography demonstrated a long segment intussusception; associated with a low echogenic mass. At laparotomy, ileoileocolic intussusception led by an ileal polyp was found. Pathology confirmed the diagnosis of polyp. Because intestinal obstruction is the primary manifestation, neonatal intussusception is initially indistinguishable from obstructions due to other reasons like intestinal atresia, congenital bands, necrotizing enterocolitis or midgut volvulus. Our experience showed that although uncommon, intussusception should be considered in the differential diagnosis of intestinal obstruction during the newborn period.

Female↗
PubMed1987

Congenital segmental dilatation of small intestine with oesophageal atresia and duodenal atresia in a premature infant.

In a premature infant documented to have oesophageal atresia laparotomy revealed the additional findings of segmental dilatation of the small intestine as well as duodenal atresia, the coincidental occurrence of which is extremely rare. Resection of the dilated segment was performed with end-to-end one-layer anastomosis and side-to-side duodeno-duodenostomy and a feeding gastrostomy was created. Three months later end-to-end oesophago-oesophagostomy was successfully performed via a right thoracotomy. During the 8 years since then the child's growth and development have been normal.

Dilatation, Pathologic↗
PubMed1997

[Perinatal surgery and perinatal treatment].

Recent results of neonatal surgery in Japan are presented. Nowadays, babies born with esophageal atresia, diaphragmatic and abdominal wall defects (gastroschisis and amphalocele) require special intensive surgical care after delivery, while those with imperforate anus, hirschoprung's disease and intestinal atresia will follow a relatively smooth postoperative course. Prenatal surgery or treatment may be necessary for some fetuses with congenital diaphragmatic hernia and congenital cystic adenomatoid malformation of the lung. Experiences at the authors institutions were reported, and recent results at institutions in the united states are introduced.

Congenital Abnormalities↗
PubMed1988

[Surgical managements of massive involvement of small bowel and of short gut syndrome].

Despite the recent advent of total parenteral nutrition (TPN), the long-term survival of the patients with short gut syndrome is not satisfactory with complications of catheter-related sepsis and cholestasis. The causes of short gut syndrome in pediatric surgery are multiple intestinal atresias, necrotizing enterocolitis (NEC), and midgut volvulus. For the multiple atresias, multiple anastomosis without resection has been successfully undertaken to avoid developing short gut syndrome for the last 17 years. For the massive involvement of the intestines due to NEC or volvulus, high jejunostomy and peritoneal drainage with TPN support have been tried for the last two patients and successfully weaned from the TPN within two months after closure of jejunostomy. However, in case 2 malabsorption has been persistent presumably due to a severe degree of mucosal damage occurring in the remaining intestines. Whether this eventually gets back to the normal or not is unknown. For the patients with short gut syndrome, small bowel reversal procedure was successfully done and now doing well 10 years after surgery. This paper reports details of these 3 cases.

Enterocolitis, Pseudomembranous↗
PubMed1980

The bulbous bowel segment: a sign of congenital small bowel obstruction.

A study of 29 neonates with intestinal obstruction has shown that a massively dilated, bulbous terminal segment of bowel is characteristic of neonatal infestinal obstruction of congenital origin. The sign is an important diagnostic indicator but its absence does not exclude intestinal atresia.

Dilatation, Pathologic↗
PubMed1995

Gastrointestinal manometry findings in a case with dilated small bowel and disturbed transit treated successfully with bowel plication.

We report the manometric findings in a case of dilated small bowel and disturbed transit successfully treated with plication of the dilated small bowel. The female newborn infant required total parenteral nutrition following an operation for small bowel atresia. X-ray showed a dilated proximal small bowel. Jejunal manometry showed normal phase 3 migration but persistently low-amplitude contractions in the dilated segment. After plication of the dilated intestine, symptoms of bowel obstruction disappeared. A second manometry two weeks after the operation showed contractions with normal amplitude. These findings indicate that: (1) disturbed transit in the dilated intestine proximal to small intestinal atresia is associated with persistently low contraction amplitude, and (2) the amplitude can be increased by the plication of the dilated loop.

Digestive System↗
PubMed2000

Imperforate anus in Feingold syndrome.

A father and daughter had the characteristic findings of Feingold syndrome including microcephaly, short palpebral fissures, brachydactyly with clinodactyly of fifth fingers, and bilateral syndactyly of second to third and fourth to fifth toes. The infant presented with long-gap esophageal atresia without fistula (type A). Her father, who had short stature and learning disabilities, had congenital imperforate anus with a recto-vesical fistula. This is the first report of distal intestinal atresia in Feingold syndrome.

Abnormalities, Multiple↗
PubMed2004

Intraoperative colonic lavage in infants using a Replogle tube.

On-table colonic lavage for preparing obstructed bowel prior to anastomosis or stoma formation is now routine in adults, but it is seldom used in paediatric practice. We describe a simple technique for intraoperative colonic lavage using inexpensive medical devices that are also readily available and disposable, a Replogle tube, a fine-bore sucker, and a 20-ml syringe. We have used this technique in infants with intestinal obstruction due to anorectal malformations, intestinal atresias, and post-NEC strictures, to good effect.

Anus, Imperforate↗
PubMed1989

Malrotation of the intestines in children: the effect of age on presentation and therapy.

Because of the devastating consequences of midgut volvulus as a result of malrotation, we reviewed the charts of 70 consecutive children to define the spectrum of presentation. Although 27 patients (39%) had presenting symptoms within the first ten days of life, 35 (50%) were older than 2 months of age. In general, the older children had a longer course of vague, antecedent symptoms such as intermittent, nonbilious vomiting and chronic abdominal pain. Associated congenital anomalies were common, with 32 patients (46%) presenting with 56 anomalies, the most prevalent of which were intestinal atresia, imperforate anus, duodenal web, and cardiac and orthopedic anomalies. Upper gastrointestinal (GI) series revealed the diagnosis in 29 cases (41%), as did contrast enema in 24 (34%). It is important to note that volvulus, intestinal gangrene, and mortality occurred regardless of age or chronicity of symptoms. Fifteen patients (21%) were discovered serendipitously while being evaluated and treated for seemingly unrelated conditions. No morbidity of mortality occurred in those patients who underwent subsequent semielective Ladd's procedure. The majority of morbidity and all seven mortalities occurred in patients with volvulus and intestinal necrosis. This study emphasizes the need for consideration of Ladd's procedure for children of all ages. In addition, due to the broad range of initial symptoms, a high index of suspicion is required in evaluating children with possible malrotation. Because it remains impossible to predict which patients will have catastrophic complications (based on age or type of presentation), we urge that even incidentally discovered patients with intestinal malrotation undergo Ladd's procedure.

Abnormalities, Multiple↗
PubMed1992

Tracheoesophageal fistula, gastrointestinal abnormalities, hypospadias, and prenatal growth deficiency.

We studied 2 sibs, born to consanguineous parents, who presented with an MCA pattern which includes low birthweight, tracheoesophageal fistula, duodenal atresia, extrahepatic biliary atresia, hypoplastic pancreas, and hypospadias. This constellation of congenital anomalies appears to be a previously unreported autosomal recessive syndrome. A computerized search of the data files of the Spanish Collaborative Study of Congenital Malformations (ECEMC) identified 3 other unrelated infants with intestinal atresias, hypospadias, and low birth weight. These cases may represent a milder expression of the same syndrome.

Abnormalities, Multiple↗
PubMed1992

Prenatal diagnosis of intestinal obstruction due to ileal atresia.

A case of intestinal obstruction due to ileal atresia where the diagnosis was made prenatally by ultrasound is presented. Close monitoring of the fetus was done ultrasonographically to look for any evidence of meconium peritonitis. The baby was delivered preterm but weighed 3.3 kg. Laparotomy and enterostomy was done and the baby is currently well.

Female↗