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PubMed1999

Hepatopathy in two infants with short-bowel syndrome and cytomegalovirus infection.

In children with short-bowel syndrome and the need for long-term parenteral nutrition, hepatic dysfunction is a multifactorial phenomenon that has not been completely understood. Alterations in gut motility lead to intraluminal stasis which is thought to be a major etiologic factor for bacterial overgrowth and subsequent cholestasis, especially when the ileocecal valve is absent. We report on two infants with short-bowel syndrome caused by gastroschisis and intestinal atresia. The intestinal lengths after resection were 18 and 55 cm. Long-term parenteral nutrition (PN) was obligatory due to intestinal shortness in the first patient and dilatation of the preatretic bowel segment with ineffective peristalsis in the second patient. Despite multiple trials of enteral nutrition and medical therapy for gut decontamination and stimulation of bowel motility, hepatopathy developed in both patients in a similar period of time and to about the same degree. At the age of 4 and 6 weeks, respectively, increasing bilirubin values were measured. Deterioration of liver function and thrombocytopenia at the age of 3 to 4 months led to the diagnosis of acute cytomegalovirus (CMV) infection. Treatment with ganciclovir followed. Both patients died of acute liver failure at the age of 7 and 9 months, respectively. Additional hepatic injury secondary to CMV infection might have contributed to the rapid deterioration of liver disease. Screening for further hepatotoxic factors, especially infectious etiologies, is therefore recommended in children with short-bowel syndrome. Liver transplantation should be considered early in cases of progressive hepatic dysfunction.

Cytomegalovirus Infections↗
PubMed1990

Fetal abdominal abnormalities associated with genetic syndromes.

Sonographic technology has given the prenatal diagnostician the ability to accurately visualize fetal malformations. This article demonstrates the currently available capability of applying sonographically derived information concerning fetal dysmorphology to our knowledge of genetic disorders and syndromes. As a result, there can be a significant improvement in the management and counseling of these patients. After an initial discussion of normal embryology, principles of abdominal ultrasonography are discussed and applied to the following anomalies: omphalocele, gastroschisis, diaphragmatic hernia, duodenal atresia, other intestinal atresias and stenoses, renal anomalies, and obstructive uropathies.

Abdomen↗
PubMed1996

Does the amniotic fluid protein absorption contribute significantly to the fetal weight?

OBJECTIVE: This study was carried out to evaluate the significance of amniotic fluid protein ingestion and absorption on fetal growth. METHODOLOGY: Neonates with small bowel atresia during a 30 year period were studied retrospectively. RESULTS: There were 56 patients enlisted, 17 with duodenal atresia, 18 with jejunal atresia and 21 with ileal atresia. The percentage of mothers with polyhydramnios and the percentage of premature babies decreases as the intestinal atresia becomes more distal. The mean gestational age and the mean birthweight increase as the intestinal atresia becomes more distal. On the other hand, the percentage of the neonates with birthweight below the 50th and the 10th percentiles do not differ significantly as the intestinal atresia becomes more distal. CONCLUSIONS: It appears that the variation of birthweights in babies with different levels of small bowel atresia may be due to the difference in gestation caused by polyhydramnios. The effect of amniotic fluid protein absorption on fetal bodyweight could not be demonstrated clinically in this study.

Analysis of Variance↗
PubMed1981

The effect of experimentally induced intestinal perforation at an early developmental stage.

In chick embryos we studied the effect on intestinal development of an experimentally induced perforation and a vascular lesion performed at an early developmental stage. The results show that an intestinal perforation will heal, but may lead to intestinal atresia with microscopic signs of meconium peritonitis. Conversely, a vascular lesion induced at an early stage of development does not lead to intestinal atresia, while a vascular lesion performed at a late stage of development does result in intestinal atresia, but without any signs of meconium peritonitis.

Animals↗
PubMed1996

[The colonic atresia: an uncommon cause of neonatal intestinal obstruction].

The intestinal atresia is a common cause of neonatal bowel obstruction, but the colonic atresia is an uncommon cause of neonatal intestinal obstruction. We present a newborn with congenital colon atresia who underwent laparotomy, revealing a colonic atresia type III. We recommend resection of the dilated proximal colon with primary anastomosis end to end if the patient is not perforated or in bad general conditions.

Anastomosis, Surgical↗
PubMed1999

Outcome analysis for gastroschisis.

BACKGROUND/PURPOSE: Several factors are reportedly associated with an adverse outcome in gastroschisis, including mode of delivery, in utero diagnosis, type of closure, concurrent anomalies, intestinal atresia, and necrotizing enterocolitis (NEC). Since 1969, the authors have treated 185 patients who had gastroschisis. The authors analyzed their database to identify variables associated with increased morbidity and mortality. METHODS: A retrospective study of all patients with gastroschisis treated at our institution in the last 30 years was performed. The characteristics of survivors and nonsurvivors were compared. A logistic regression analysis was performed, with survival as the dependent variable, and the following parameters as independent variables: in utero diagnosis, mode of delivery, gestational age and birth weight, era of repair, type of closure, presence of other associated anomalies, intestinal atresia, and development of necrotizing enterocolitis. Further logistic regression analysis was performed, with various indicators of morbidity as dependent variables. These included development of sepsis, bowel obstruction, and complications related to the closure or to the silo. No attempt at long-term follow-up was made. RESULTS: A total of 185 infants with gastroschisis were treated at our institution from 1969 to 1999. Mean gestational age was 36.6 weeks, and the mean birth weight was 2,501 g. A total of 21 infants had intestinal atresia. NEC developed in 8 infants. Six infants had other serious anomalies. The overall survival rate was 91%. Survival improved in last 2 decades (94%). There were no differences in gestational age, birth weight and mode of delivery, method of closure, or presence of intestinal atresia between survivors and nonsurvivors. Only the era of repair (P = .002), presence of necrotizing enterocolitis (P = .044), and presence of other major anomalies (P < .001) correlated with mortality in the logistic regression analysis. Sepsis, bowel obstruction, and closure complications accounted for most of the morbidity. Analysis of these three morbidity factors identified low gestational age (P = .038) and development of necrotizing enterocolitis (P = .020) as independent predictors of sepsis. Closure complications were only associated with lower birth weight (P = .006). No predictors of bowel obstruction were identified. CONCLUSIONS: Mode of delivery, method of closure, birth weight and gestational age, and the presence of intestinal atresia do not appear to correlate with survival in infants with gastroschisis. Only the presence of another major anomaly, the era of repair, and the development of necrotizing enterocolitis were associated with increased mortality. Degree of prematurity and development of enterocolitis were associated with an increased incidence of septic complications. Low birth weight was a marker for closure complications. Type of delivery (vaginal or cesarean section) had no influence on either morbidity or mortality.

Birth Weight↗
PubMed1994

Spinal anesthesia for primary repair of gastroschisis: a new and safe technique for selected patients.

Spinal anesthesia has been described for infants and premature babies undergoing minor operative procedures. The advantages of shorter operating time, avoidance of intubation, and shorter hospital stay have made this the gold standard for premature and other high-risk infants requiring minor procedures. However, little is known about this technique for major interventions in newborns and preterm infants. Recently, four infants born with gastroschisis underwent repair under spinal anesthesia. Two had accompanying intestinal atresia (one with a prenatal perforation and pan-hypopituitarism), and two had intact gastrointestinal systems. The gestational ages were 39, 33, 36, and 36 weeks, respectively. All had primary closure of the defect; one had no repair of the atresia because the bowel was thick and matted with a significant peel, and the defect was not identified. In the second case with atresia, necrosis and perforation of a localized segment of intestine was identified proximal to the intestinal atresia, and was exteriorized with the primary repair. When they arrived in the operating room, all four infants were breathing spontaneously, on room air, after appropriate fluid resuscitation. All underwent spinal anesthesia, which was the only agent used for the operation. The operative time was 45, 25, 30, and 25 minutes, respectively (mean, 31.25 minutes). The duration of anesthesia was 170 to 230 minutes (mean, 205 minutes). All infants were returned to the neonatal intensive care unit on room air and breathing spontaneously. One was given morphine postoperatively and suffered significant respiratory depression, requiring intubation. It appears that spinal anesthesia is safe and effective for major operative procedures in high-risk infants. (ABSTRACT TRUNCATED AT 250 WORDS)

Abdominal Muscles↗
PubMed1999

Pyloric atresia associated with duodenal and jejunal atresia and duplication.

A case of congenital pyloric atresia associated with duodenal atresia, jejunal atresia, and a duplication is described. A review of the literature revealed nine previously reported cases of congenital pyloric atresia and multiple intestinal atresias, but this is the first report of such an association and a duplication.

Cysts↗
PubMed2003

Classification, epidemiology and aetiology.

Intestinal failure and its most important cause, short-bowel syndrome (SBS), are rare clinical entities leading to a vast complex of symptoms and complications with significant morbidity and mortality. Both conditions occur as the result of a massive reduction in enteral nutrient absorptive capacity. Disease manifestation is based on aetiological and anatomical characteristics such as remaining intestinal length and the presence of a functionally intact colon. Congenital and perinatal conditions, for example, intestinal atresia, necrotizing enterocolitis (NEC) and intestinal volvulus are the most important causes in children. The aetiology in adults is based on diseases inducing loss of intestinal function or loss of intestinal surface area after extensive surgical resections. The most frequent causes are mesenteric infarction, radiation enteritis and Crohn's disease. Knowledge of the epidemiology of intestinal failure and SBS is limited, being mainly based on the extrapolated figures of home parenteral nutrition centres and single-centre studies. At present, the incidence of SBS is estimated to be 2-5 per million.

Adult↗
PubMed1980

Necrotizing enterocolitis following operation in the neonatal period.

Necrotizing enterocolitis (NEC) usually occurs in low birth weight infants who have had perinatal stress, and the mortality remains significant. There are a few reports of NEC in the postoperative period, especially in young infants. Nine neonates developed NEC following operations and form the basis of this report. The interval between operation and the diagnosis of NEC varied from 3 days to 4 mo. The surgical lesions included one case each of esophageal atresia, tetralogy of Fallot, supralevator rectal atresia with rectourethral fistula, and multiple intestinal atresias. Three babies had gastroschisis and two had "apple peel" intestinal atresia. Only 3 of the 9 survived. The usual clinical findings of NEC, abdominal distention, bile stained gastric residuals and diarrhea (with or without blood), can occur in the postoperative period without NEC and are, therefore, not reliable diagnostic signs. Significant changes in the clinical course of these babies occurred from 7 hr to 5 days before the diagnosis was established. In these patients the roentgen findings that established the diagnosis of NEC included intestinal ileus, pneumatosis intestinalis, and portal vein gas. Pneumatosis intestinalis and portal vein gas were the most reliable diagnostic signs, but appeared relatively late in the course of the disease. In one case pneumatosis was seen only in retrospect. None of the patients had definite pneumoperitoneum. Awareness of NEC as a potential postoperative complication may result in early recognition, treatment and survival.

Congenital Abnormalities↗
PubMed2006

Intrauterine hemorrhage from an umbilical cord ulcer associated with fetal duodenal atresia: a case report.

In the recent years, the association between congenital intestinal atresia and umbilical cord ulceration has been demonstrated; however, this complication is rarely encountered. To the best of our knowledge, only 14 cases on this association have been published previously. We encountered a case of acute intrauterine hemorrhage from an umbilical cord ulcer in a fetus with duodenal atresia that resulted in early neonatal death. There is little information available on the association between congenital intestinal atresia and umbilical cord ulceration. Greater understanding may alter the obstetric management. Although the prevention of sudden perinatal death is considered to be difficult, frequent fetal monitoring and detailed ultrasonographic examinations may facilitate the early detection of this condition, thereby enabling the rescue of the affected fetuses. Further investigation is required to establish the management protocol.

Adult↗
PubMed2002

Prolonged parenteral nutrition after neonatal gastrointestinal surgery. A Danish experience.

INTRODUCTION: Long-term treatment with parenteral nutrition (PN) may be essential for survival in infants after neonatal gastrointestinal surgery. It seemed well indicated in a population-based study to estimate the need for long-term PN and to characterize the infants that received TPN with regard to diagnosis and clinical course. METHODOLOGY: This study reviews the clinical course of infants with gastrointestinal disease (gastroschisis, intestinal atresia, omphalocele, volvulus, Hirschsprung's disease and necrotizing enterocolitis) with a prolonged need for parenteral nutrition in the Western part of Denmark over a period of 11 1/2 years. RESULTS: A total of 21 patients with need for PN for 55 days or more due to gastrointestinal disease were registered with a cumulative hospital stay of 4462 days. The study showed a low incidence of long-term PN due to gastro-intestinal surgical illness with an incidence of approx 6.1 per 10(5) births per year. Gastroschisis and intestinal atresia were the most common primary diagnoses. Two groups of patients were identified: 1) six infants with short gut syndrome caused by major resections or atresia of the small intestine (less than 50 cm intestine) and 2) a larger group (n = 15) with no or only minor resection of the small bowel. The former group had a significantly longer hospital stay and a longer need for PN. The incidence of short bowel syndrome including six infants was 1.7 per 10(5) births per year. CONCLUSIONS: Prolonged need for PN in infants operated for gastrointestinal disease was registered at a low frequency in the Western part of Denmark, most frequently caused by gastroschisis and intestinal atresia. The clinical course was characterized by repetitive problems of catheter replacements and septic episodes. The mortality due to conditions related to the gastrointestinal disease was 14% (3/21).

Digestive System Surgical Procedures↗