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At least 361 records · Page 20Linked to original sources
PubMed2005

Fibroblast growth factor-10 serves a regulatory role in duodenal development.

PURPOSE: Duodenal obstruction occurs in 1 of 6000 live births and requires urgent surgical intervention. Duodenal atresia previously has been ascribed to a developmental failure of luminal recanalization; however, the cause of duodenal atresia remains incompletely understood. Although familial intestinal atresias have been described and syndromic associations are known, no specific genetic link has been established. Fibroblast growth factor-10 (Fgf10) is a known regulatory molecule relevant to mesenchymal-epithelial interactions, and mice deficient in Fgf10 demonstrate congenital anomalies in several organ systems including the gastrointestinal tract. The authors hypothesized that Fgf10 could serve a regulatory role in establishing normal duodenal development. METHODS: Wild-type mice with beta-galactosidase under the control of the Fgf10 promoter were harvested from timed-pregnancy mothers. The expression of Fgf10 in the duodenum during development was evaluated by developing the embryos in X-Gal solution. Wild-type and mutant Fgf10(-/-) embryos were harvested from timed-pregnancy mothers at 18.5 days postconception (near term) and were analyzed for duodenal morphology (Institutional Animal Care and Use Committee-approved protocol 32-02). Photomicrographs were reviewed. RESULTS: Fibroblast growth factor-10 is active in the duodenum at a late stage of development. The Fgf10(-/-) mutants demonstrate duodenal atresia with a variable phenotype similar to clinical findings. The duodenum fails to develop luminal continuity and has proximal dilation. The phenotype occurs in an autosomal recessive pattern with incomplete penetrance (38%). CONCLUSIONS: Fibroblast growth factor-10 serves as a regulator in normal duodenal growth and development. Its deletion leads to duodenal atresia and challenges traditionally accepted theories of pathogenesis. This novel, genetically mediated duodenal malformation reflects an animal model that will allow further evaluation of the pathogenesis of this surgically correctable disease. By studying the mechanism of Fgf10 function in foregut development, the authors hope to better understand these anomalies and to explore possible therapeutic alternatives.

Animals↗
PubMed1979

Preoperative condition of neonates who have ultimately died.

In a retrospective study the case sheets of 42 neonates are examined. They were infants with oesophageal atresias, small and large intestinal atresias, exomphalos and gastroschisis, diaphragmatic hernia, intestinal obstruction as well as two children with a ruptured spleen secondary to birth trauma. Various laboratory investigations such as the pH, pCO2, standard bicarbonate and serum electrolytes as well as the child's weight on admission and, if possible, the birth weight were evaluated. There was no definite relationship between the preoperative laboratory values and the chances of survival of surgical neonates except if the values were extreme. Infections and septicaemia were the most important causes of postoperative complications.

Congenital Abnormalities↗
PubMed2000

A simple method of intraoperative confirmation of intestinal patency.

Routine intraoperative rectal temperature monitoring may serve in addition as a means of distal intestinal patency confirmation. A simple method, which is of immense importance especially when operating on infants and small children after NEC or intestinal atresia, is described.

Body Temperature↗
PubMedDate not supplied

[Effect of obstructive malformations on the gastrointestinal tract on bilirubin levels in newborn infants].

The authors have reviewed their cases of neonatal obstructive malformations of the intestinal tract and analyzed the incidence of association with indirect hyperbilirubinemia. 39 patients in four years are considered. In 23 of them there is a bilirubin level greater than 5 mg/100 ml. A great number of high bilirubin levels (greater than 15 mg/100 ml) is associated with volvulus, malrotations and intestinal atresias. Bilirubin levels between 11 and 15 mg/100 ml are often connected with meconium ileus while ano-rectal atresias have a low bilirubin level (less than 5 mg/100 ml). The reported data allow to conclude that the jaundice is an early, but unfortunately, not specific symptom of congenital intestinal obstruction. The entero-hepatic circulation seems to be an important cause of the neonatal hyperbilirubinemia.

Anal Canal↗
PubMed1983

The distended neonate.

A prospective study, followed by a retrospective audit, was made of the radiographs and clinical features of 225 consecutive neonates with abdominal distension presenting during a period of 4 years. More than one major cause for the distension was found in a third of cases. Excluding cases of ano-rectal atresia, diagnostic difficulties were encountered in 31% of cases and, occasionally, the diagnosis was revised several times as investigations proceeded. Neuhauser's sign of 'bubbly' meconium is unreliable, being found in cases of meconium ileus, ileal atresia, Hirschsprung's disease and necrotising enterocolitis. Concomitant small-bowel atresia should be suspected in all cases of meconium ileus; intestinal malrotation should be considered in association with duodenal and intestinal atresias. In the absence of an obvious anatomical defect, it is difficult to differentiate between the various causes of functional obstruction by radiological means alone and all such infants should be referred to a specialised centre for intensive investigation and treatment. Very often, the skills of a neonatologist, paediatric surgeon and, especially, an experienced pathologist will be required in addition to those of the radiologist.

Anal Canal↗
PubMed1998

Hereditary multiple atresias of the gastrointestinal tract: report of a case and review of the literature.

Hereditary multiple atresia of the gastrointestinal tract is an extremely rare subgroup of intestinal atresia. The aim of this study was to report a new case, to review the literature, and to describe the unique features of this malformation. A computer-generated list of articles on this subject was obtained, and all articles relative to this malformation were reviewed. Thirty-five other well-documented cases were found in the literature. Hereditary multiple atresias have several unique features: (1) the abdominal x-ray shows signs of gastric or duodenal atresia combined with typical large rounded or oval homogeneous calcifications in the abdominal cavity, (2) intraoperatively widespread atresias (exclusively type I and II) extending mostly from stomach to rectum are found, (3) cystic dilatation of the bile ducts can be present in cases with both complete pyloric and duodenal or proximal jejunal atresia, (4) the pathogenesis is still speculative; a combined immunodeficiency should be excluded, and (5) a fatal outcome is the rule.

Abnormalities, Multiple↗
PubMed1995

Jejunoileal atresia in neonates.

BACKGROUND: Jejunoileal atresia is the most common intestinal atresia and the most frequent cause of neonatal intestinal obstruction. The prognosis depends on several important factors. The patients reported were collected for evaluation of the factors contributing to their prognoses. METHODS: In the ten-year period from October 1982 to October 1992, all jejunoileal atresia patients who received treatment in this hospital were entered in this study. Possible factors contributing to prognosis, such as prematurity, low birth weight, apple peel atresia, anastomotic leakage, sepsis, short bowel syndrome were evaluated using Mann-Whitney U-test. RESULTS: Twenty-four patients of jejunoileal atresia had been managed in our hospital in 10 years period. Their ages ranged from one to thirteen days old (mean 3.9 days). The numbers and types of atresia were as follow: type I (n = 4), type II (n = 4), type IIIa (n = 10), type IIIb (n = 5), type IV (n = 1). All patients received surgical correction. Nine complications were found in eight patients, which included four anastomotic leakage, three wound infections and two intestinal obstructions. There were three mortalities from sepsis. Total parenteral nutrition was applied to 14 patients (58.3%). The mean hospital stay was 45.1 days, and the survival rate was 87.5%. Low birth weight and anastomotic leakage contributed to prolonged hospital stays (p < 0.05). CONCLUSIONS: Jejunoileal atresia is a common neonatal surgical condition. The prognosis is generally good, except when a patient presented with factors, such as, low birth weight or anastomotic leakage, which prolonged hospital stays. Moreover, sepsis is the only important factor contributing to mortality.

Female↗
PubMedDate not supplied

Malformations reported in chorionic villus sampling exposed children: a review and analytic synthesis of the literature.

PURPOSE: To determine whether the frequency of vascular disruption defects, other than limb defects, is increased in reports of chorionic villus sampling (CVS) exposed children compared with an unexposed population. METHODS: Only studies that reported the total number of CVS-exposed pregnancies and details of pregnancy outcome, including all the malformations, were included. Twenty-five articles met these criteria. RESULTS: The frequencies of gastroschisis, intestinal atresias, and clubfoot were significantly increased among the CVS-exposed infants as compared with the baseline unexposed population. The frequencies of other vascular disruption defects, including Poland sequence, amniotic band sequence, and cleft lip/cleft palate, were not increased. CONCLUSION: CVS-exposed children have an increased frequency of intestinal atresia, gastroschisis, and clubfoot compared with the nonexposed population. The fact that an increased frequency of other defects attributed to vascular disruption was not found may be due to under-ascertainment, misclassification, or "lumping" of the defects identified in previous studies.

Chorionic Villi Sampling↗
PubMed1982

An experimental study of the effect of an intestinal perforation at various developmental stages.

The effect of an experimentally induced intestinal perforation was studied in chick embryos. In 5 different groups of embryos a perforation was induced at 5 different developmental stages, while an equal number of sham operations was carried out for each stage. The results demonstrate that the effect of an intestinal perforation is determined by the developmental stage at which this perforation occurs. There was an inverse relationship between the developmental stage at which the perforation was induced and the remaining traces of the perforation site at the end of incubation. A perforation induced at an earlier developmental stage was more likely to lead to intestinal atresia coupled with only microscopic signs of meconium peritonitis. The severity of the adhesions increased concurrently with the developmental stage.

Animals↗
PubMed2001

Hirschsprung's disease, associated rare congenital anomalies.

OBJECTIVE: Hirschsprung's disease may be associated with a number of congenital anomalies of which Down's syndrome and intestinal atresias are commonly encountered. The study aimed to assess the impact of rare associated anomalies on the diagnosis and management of Hirschsprung's disease. METHODS: A retrospective review of the clinical presentation, diagnosis and outcome of thirty five consecutive newly diagnosed cases of Hirschsprung's disease encountered over two years was performed. RESULTS: Besides Down's syndrome (two), intestinal atresia (one) and pigmentary ocular defects (two), three rare anomalies (Occipital meningocele, Calcific meconium cyst with anal stenosis, Malrotation) were encountered in four of thirty five cases. The clinical features, radiologic anatomy and gross morphology of the bowel were unconventional and the diagnosis was supported by intraoperative acetylcholinesterase staining of biopsies. Though the diagnosis was relatively delayed in these cases, the outcome has been comparable to the rest. The dilemma in their diagnosis and management and their possible pathoembryology is discussed. CONCLUSION: Awareness of such associations and a specific investigative protocol is imperative for timely diagnosis and minimal morbidity in complex presentations of Hirschsprung's disease.

Abnormalities, Multiple↗
PubMed1992

Colon atresia and stenosis in Zimbabwe: case reports and a review of the literature.

Two neonates with colon atresia and one with colon stenosis presented in Harare over a six month period. The first patient was treated with a local resection and primary anastomosis for a type II colon atresia. The second had an excision of obstructing septum, widening coloplasty and cecostomy for a type I colon atresia. The third patient had a sigmoido-rectoplasty and cecostomy for a sigmoid colon stenosis. The third patient also had a cystic duplication of the small bowel. No other anomalies were noted. All three patients survived and were developing normally at six months post-operatively. A review of medical records at Harare and Parirenyatwa Hospitals revealed no other case of colon atresia in the last 10 years. The incidence by site of gastro-intestinal atresias in Zimbabwe is consistent with other reports.

Colon↗
PubMed2003

Fetal small bowel obstruction: report of two cases.

Fetal small bowel obstruction is usually diagnosed by sonography in the late second or early third trimester. We report two such cases of different etiology: a case of meconium ileus due to cystic fibrosis and a case of intestinal atresia. The only sonographic finding that allowed differentiation between them was the echogenic bowel in the case of cystic fibrosis.

Adult↗
PubMed2000

The contemporary outcome of gastroschisis.

BACKGROUND: The aim of this study was to evaluate the contemporary outcome in the management of gastroschisis. METHODS: A retrospective analysis was conducted of 91 babies admitted over a 7-year period to a single neonatal surgical unit with a diagnosis of gastroschisis. RESULTS: An antenatal diagnosis was made in 89 (98%) cases. Surgical intervention occurred in 90 babies, at a mean of 5 hours (range, 0.5 to 17) postdelivery. In 72 (80%) cases, primary closure of the abdominal defect was achieved, with a silo fashioned in the remaining 18 (20%). One child died before abdominal closure. The median time to full oral feeding was 30 days (range, 5 to 160 days), and to discharge was 42 days (range, 11 to 183 days). Those children who required a silo, took longer to feed (P =.008) and stayed longer in the hospital (P =.021). The 8 (8.8%) children with an intestinal atresia, required significantly more operative procedures (P =.0001) and took significantly longer to achieve full oral feeding (P =.04), but the presence of an atresia was not an independent risk factor for mortality. There were 7 deaths (7.7%), 3 within the first 7 days. Of the deaths, 5 (71%) were caused by overwhelming sepsis. CONCLUSIONS: The contemporary mortality rate from gastroschisis is less than 8%, and minimizing septic complications would contribute significantly to reducing this. Strategies designed to improve morbidity must focus on optimizing management of those factors associated with a prolonged recovery, namely intestinal atresia, prematurity, and the use of a silo.

Birth Weight↗
PubMed1980

Apple peel small bowel. A review of twelve cases.

This is a review of 12 cases with apple peel small bowel with agenesis of the dorsal mesentery and intestinal atresia, 11 of them jejunal and 1 duodenal in type who were treated at the Clinica Infantil "La Paz" Madrid over a period of ten years (1969--1979). The associated anomalies, the surgical treatment and the cause of death as well as the etiology and pathogenesis of this malformation are discussed.

Duodenum↗
PubMed2002

Embryogenesis of adriamycin-induced hindgut atresia in rats.

It was proposed that the pathogenesis of multiple intestinal atresias (MIA) in human fetuses is a consequence of malformative processes of the gastrointestinal tract rather than an ischemic process. Recently, MIA has been described in adriamycin-exposed rat fetuses. The aim of this study was to describe the embryogenesis of hindgut atresia (HA) in the adriamycin animal model. Timed-pregnant Sprague-Dawley rats were injected with adriamycin on days 6-9 of gestation. Embryos were removed on different gestational days during organogenesis and histologic sections were examined and compared with control specimens. In experimental embryos, HA was seen on day 13; however, the lumen was patent on day 12. HA was associated with abnormal vascular anatomy that was obvious on days 12 and 13. It is concluded that HA in adriamycin-exposed embryos occurs at the beginning of organogenesis. Although it was associated with an obvious vascular anomaly, further studies are required to find out whether it is ischemic in origin.

Animals↗
PubMed2006

Submucosal plexus of dilatated gut disappears after ligation in chicken embryos: preliminary results.

INTRODUCTION: Dilatation and impaired function of the gut is a condition often seen in newborns with bowel obstruction caused by intestinal atresia. In a previous experimental study in chicken embryos, we established a model to study ultrastructural changes during the development of the enteric nervous system after small bowel ligation. The aim of this study is to investigate the changes of the enteric nervous system (ENS) after gut ligation. METHODS: 56 chicken embryos were investigated. In the operation group fertilized eggs and the allantoic membrane were opened and the small bowel was ligated on embryonal day (ED) 11. The controls were sham-operated. The gut was prepared and harvested for analysis on ED 11, 12, 13, 14, 15, 16, 17 and 18. Silver staining or staining of the specimens for acetylcholinesterase (AchE) was performed. RESULTS: A marked dilatation of the bowel was observed three days after operation (ED 14). The submucosal (PSM) and myenteric plexus (PM) appeared normal at this time, however silver staining showed rarification of the neuronal axonal network between the myenteric and submucosal plexus. Later, on ED 16 an additional rarification of the submucosal plexus was also seen in the operation group using AchE staining, compared to the controls. DISCUSSION: The data suggest that distension of the gut hinders normal development of the ENS in the gut ligation model of chicken embryos. The changes were observed sequentially, starting with rarification of the axonal network between the PM and PSM. Future studies will be required to show whether the changes of the ENS are reversible.

Animals↗