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PubMed2000

Jejuno-ileal atresia in identical twins: report of a case.

Small intestinal atresia occurring in twins is extremely rare. The aims of this study are to report a case of identical twins with different types of jejunal atresia, to review six other pairs of twins, and discuss the etiology. Our case was the seventh in which both twins were diagnosed to have jejuno-ileal atresia. Furthermore, they are also only the sixth and seventh patients known to survive. All the other published cases involved identical twins except one. Three pairs had different types of atresia and four pairs did not have any other anomalies. In addition, to date there have been no further indications of any other members of these families being affected with intestinal atresia. Therefore, we conclude that most such cases might be due to environmental influences during gestation.

Anastomosis, Surgical↗
PubMedDate not supplied

Multiple congenital septal atresias of the intestine: histomorphologic and pathogenetic implications.

Congenital atresias of the gastrointestinal tract are usually single and divided into three forms on a morphologic basis. The septal or diaphragmatic type (type I) is the least common. We report an infant with multiple type I atresias involving both the small and large intestine and describe the unique histologic features of the septa: fragmentation of muscularis mucosa, multiple septal cysts lined by columnar epithelium, circular and longitudinal muscular layers, and absence of inflammation. This infant had a sibling who died with multiple intestinal atresias of the septal type. The histologic features suggest that type I gastrointestinal atresias may be due to failure of complete recanalization rather than result from healing of vascular of inflammatory events.

Abnormalities, Multiple↗
PubMed1997

Improved long-term outcome for patients with jejunoileal apple peel atresia.

BACKGROUND/PURPOSE: Although apple peel intestinal atresia is a rare lesion associated with significant morbidity and high mortality, the authors have seen no deaths since 1983. Similar success has rarely been reported, and there are no reports of long-term follow-up. This study examines the short-term and long-term complications and outcome for these children, critiques our evolution in care, and gives current recommendations for therapy. METHODS: A retrospective review of 12 patients over 11 years was conducted. Perinatal history and operative and perioperative management were examined and end results and complications using different management plans compared. Long-term outcome was determined through clinic follow-up. RESULTS: Mean follow-up was 5.1 years. Children had a mean 61.4 cm of total small bowel. Seven patients underwent a primary anastomosis and five had enterostomies. The proximal jejunum was tapered, plicated, resected or left intact. Eleven children required gastrostomy tubes. All children required total parenteral nutrition. Full enteral feeding was achieved in all children, but three required gastrostomy supplementation. Three patients who had enterostomies suffered bowel obstruction, two with dilated, dysmotile proximal jejunum required subsequent tapering. Eight children maintained a growth curve between the 5th and 50th percentile. None have short bowel physiology, and all have achieved acceptable bowel function. CONCLUSIONS: (1) Total parenteral nutrition is essential for initial nutritional management. (2) Use of an enterostomy leads to an increased incidence of complications. (3) The dilated proximal bowel should be resected, tapered, or plicated, and a primary anastomosis should be performed. (4) Gastrostomy tubes are necessary for initial management. (5) Early morbidity is common, though excellent long-term outcome and normal growth and development are expected.

Anastomosis, Surgical↗
PubMed2000

Neonatal intestinal obstruction in Zaria, Nigeria.

OBJECTIVE: To study the aetiology, morbidity and mortality of neonatal intestinal obstruction. DESIGN: A retrospective study. SETTING: Ahmadu Bello University Teaching Hospital, Zaria, Nigeria. SUBJECTS: One hundred and fifty one neonates (< or = 28 days) undergoing surgery for mechanical intestinal obstruction. RESULTS: The male/female ratio was 3:1 and median age at presentation was four days (range five hours-28 days). Anorectal malformation was the commonest cause, 104 (68.9%), 86.5% of which were high anomalies and 13.5% low; the median age at presentation was three days. Fifty two per cent of colostomies for the high anomalies were performed using general anaesthesia and 48% local anaesthetic, but there was an increasing use of local anaesthesia over the years. Hirschsprung's disease accounted for 11 (7.3%) of the cases, representing 20% of all patients presenting with Hirschsprung's disease to this hospital; the median age was six days and in two patients the caecum and sigmoid colon respectively had perforated; nine patients had colostomy, one caecostomy and one ileostomy (total colonic aganglionosis). Eleven (7.3%) patients had incarcerated or strangulated ingunial hernia (ten) and congenital ventral hernia (one); the hernias were repaired in all patients and three required intestinal resection for gangrene, two of which had ipsilateral testicular gangrene, necessitating orchidectomy. Intestinal atresia was the fourth common cause of obstruction ten (6.7%), eight of which were jejunoileal atresias and two duodenal and the median age was seven days; one atresia was associated with Hirschsprung's disease and had ileostomy, all other jejunoileal atresias were resected and duodenoduodenostomy was performed for the duodenal atresias. Other less common causes of neonatal intestinal obstruction were incarcerated exomphalos, malrotation, hypertrophic pyloric stenosis, annular pancreas, and idiopathic ileal volvulus and meconium ileus respectively. Postoperative complications occurred in sixteen of 95 patients (16.8%) including colostomy or ileostomy complications 11, wound infection three and anastomotic dehiscence (two). The overall mortality was 21.1%, 70% from overwhelming infection and 30% respiratory embarrassment; the mortality from the various conditions were Hirschsprung's disease 43%, intestinal atresia 40%, incarcerated exomphalos 40%, anorectal malformation 18.5% and the only patient with volvulus died. CONCLUSION: The morbidity and mortality of neonatal intestinal obstruction in this hospital has improved over previous years due largely to meticulous resuscitation before surgery but the problems of late presentation and poor neonatal intensive care facilities persist. The findings are at variance with those in developed countries.

Female↗
PubMedDate not supplied

Duodenal atresia, biliary atresia, and intestinal infarct in truncal aplasia cutis congenita.

A newborn boy with aplasia cutis congenita had biliary atresia, distal duodenal atresia, and a severe infarct of the intestine resulting in complete absence of the entire midgut. The boy died due to biliary atresia and severe short gut syndrome approximately 3 weeks after birth. While the association of duodenal atresia with aplasia cutis congenita has been described, the findings of biliary atresia and midgut atresia in association with aplasia cutis congenita have not been described previously.

Adult↗
PubMed2000

The influence of bowel atresia in gastroschisis on fetal growth, cardiotocograph abnormalities and amniotic fluid staining.

OBJECTIVES: To investigate the relationship between intestinal atresia and fetal growth, intrapartum cardiotocograph abnormalities and staining of the amniotic fluid in gastroschisis pregnancies. DESIGN: Observational study of 115 gastroschisis pregnancies conceived between 1980 and 1996. SETTING: South West Region of England. MAIN OUTCOME MEASURES: Birthweight, intrapartum cardiotocograph abnormality, staining of amniotic fluid and Apgar score. RESULTS: There was no statistically significant difference between the Apgar scores or the frequency of amniotic fluid staining of gastroschisis cases with or without atresia. Patent bowel gastroschisis was associated significantly with more cardiotocograph abnormalities (61% vs 26%, P = 0.026) and reduced growth (P = 0.02), when compared with cases with intestinal atresia. CONCLUSIONS: Bile vomiting may be an important cause of amniotic fluid staining. Bowel atresia protects against the increased incidence of cardiotocograph and growth abnormality found in cases of gastroschisis which may reflect their aetiology.

Amniotic Fluid↗
PubMed2001

Multiple gastrointestinal atresias result from disturbed morphogenesis.

Multiple gastrointestinal atresias (MGA) have been reported to account for 6% to 32% of all intestinal atresias. Controversy exists regarding the pathogenesis. Many investigators believe MGA to be the result of multiple ischemic infarctions of the intestinal tract. However, some have suggested that MGA results from a malformative process early in fetal life. Prenatal exposure to adriamycin in a rat model has been reported to lead to a spectrum of tracheoesophageal and associated malformations of the gastrointestinal tract, including intestinal atresias, identical to these observed in humans. The aim of this study was to determine the incidence and histopathologic findings of MGA in order to understand the pathogenesis. Timed-pregnant Sprague-Dawley rats were injected with adriamycin (1.75 mg/kg) in nine different gestational-day protocols. MGA was only seen in those rats who received adriamycin on gestational days 7, 8, and 9. The litters were recovered on day 21 by cesarean section. The digestive tracts (DT) of the fetuses were harvested for macroscopic and microscopic examination. Ten rats who received adriamycin on gestational days 7, 8, and 9 produced 87 newborns; 1 was damaged during dissection. DT anomalies occurred in 80 (93%) of the 86 newborns; 94% of these demonstrated MGA. There was a very high incidence of associated anomalies in newborns with MGA. Histologically, the blind-ending atresias showed different degrees of villous hyperplasia with or without intraluminal material. This is the first report demonstrating a high rate of occurrence of MGA in the adriamycin rat model. The injection of adriamycin early in gestation, the high incidence of associated malformations, and the anatomic and histologic findings in MGA indicate that MGA is a result of a malformative rather than an ischemic process.

Animals↗
PubMed1981

Histochemical studies of experimental fetal intestinal obstruction.

Experimental intestinal atresia can be produced by mesenteric disruption in fetal lambs. In previous reports, a detailed histochemical study of the bowel in this atresia model demonstrated: (1) hyperplasia of ganglion cells in the dilated proximal segment, (2) involutional changes in the area of maximal distension, (3) decreased to absent adenosine triphosphatase (ATP-ase) production in the area of the atresia, (4) gradual increase of ATP-ase production to normal proximally, and (5) greater reduction of ATP-ase production along the antimesenteric border compared to the mesenteric border. In the present study, a model of fetal intestinal obstruction by simple ligation of the bowel has been created to observe the effects of pure obstruction of the lumen of the fetal bowel without the possible ischemic effects of any vascular interruption. Studies with this model reveal: (1) hyperplasia of ganglion cells in the dilated proximal segment, and (2) decreased ATP-ase production proximal to the obstruction, but (3) no involutional changes in the area of maximal distension. These findings show a pattern of disturbance of bowel morphology and function caused by obstruction of the fetal bowel that is similar to but less severe than that seen with intestinal atresia.

Acetylcholinesterase↗
PubMed1986

The importance of mesenteric vascular insufficiency in meconium peritonitis.

The etiology of bowel perforations leading to meconium peritonitis was investigated in an attempt to explain the occurrence of such perforations in the absence of primary intestinal obstruction. Bowel specimens from 22 patients who had presented with meconium peritonitis during a 15-year period, as well as specimens from five patients with intestinal atresia without associated meconium peritonitis, were re-evaluated microscopically. An extensive review of the literature concerning meconium peritonitis, involving 1,084 patients, was compared with the results of a retrospective study of 69 patients from The Netherlands. In approximately 50 per cent of these cases there was no detectable primary cause of the bowel perforation. The pathologic findings, in combination with earlier experimental studies, indicate that vascular insufficiency may result in bowel wall perforation without prior intestinal obstruction. It is suggested that temporary decrease of mesenteric blood flow could lead to intestinal atresia and/or meconium peritonitis.

Birth Weight↗
PubMed1990

Severe combined immunodeficiency syndrome associated with autosomal recessive familial multiple gastrointestinal atresias: study of a family.

Hereditary multiple atresias involving the gastrointestinal tract from pylorus to rectum are the most unusual form of intestinal atresia; the type of inheritance was suggested to be autosomal recessive. The inheritance of the severe combined immunodeficiency syndrome can be autosomal recessive or X-linked. We report on 3 sibs with multiple-level intestinal atresias. One sib had severe combined immunodeficiency syndrome and clinical histories of the other 2 sibs strongly suggested a congenital immunodeficiency syndrome. The parents of those children were healthy and nonconsanguineous. To our knowledge, this is the first report of the association of multiple gastrointestinal atresias and immunodeficiency which appears to have an autosomal recessive pattern of transmission. Our family report suggests that, in the presence of multiple gastrointestinal atresias, attention should be given to possible associated immunological disorders.

Abnormalities, Multiple↗
PubMed1981

[Atresia of the jejunum, ileum and colon; a retrospective study].

The results are described of a retrospective study of 142 patients with atresias of the jejunum, ileum and colon in the Netherlands. It is pointed out that pregnancy had often been complicated by polyhydramnios. This may lead to an early diagnosis in the prenatal period. Whenever a neonate presents with bilious vomiting or distension of the abdomen, closer examination for intestinal atresia should take place. The production of meconium does not rule out the diagnosis of intestinal atresia.

Adolescent↗
PubMed1997

Gastrointestinal myoelectric activity in a child with gastroschisis and ileal atresia.

Gastroschisis is frequently associated with intestinal atresia and alterations in gastrointestinal function. The authors studied gastric and small bowel myoelectric activity in a child who had a complex course and prolonged inability to tolerate oral intake after staged repair of gastroschisis and an associated ileal atresia. The child remained unable to tolerate oral intake after repair of the atresia and was reexplored 3 months later to rule out a partial small bowel obstruction, with simultaneous placement of serosal electrodes on the stomach and proximal small bowel. Persistent gastric dysrhythmias were observed postoperatively, and the child was unable to tolerate gastrostomy tube feedings. Abnormalities were also seen in small bowel motility, including retrograde propagation of activity fronts of the migrating myoelectric complex. However, the intestine converted to a fed myoelectric pattern with tube feedings, and the child was subsequently able to tolerate feedings via a tube placed directly into the small bowel. The authors conclude that myoelectric recordings via implanted electrodes are safe and feasible in children, and may give information regarding underlying motility alterations. The ultimate clinical role of myoelectric recordings in treating children with suspected motility disorders will require further study.

Abdominal Muscles↗