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PubMed1990

[Results of biofeedback on incontinence caused by anorectal atresia].

Eight patients with anorectal incontinence because of anorectal atresia, aged between seven and seventeen years, following surgical treatment with Romualdi-Soave procedure in seven cases and with PSARP in one case, have received biofeedback training to improve their continence. Clinical evaluation has been performed with a new numeric punctuation method that allows a quantitative and precise appraisal of incontinence (Incontinence Punctuation: IP). From a clinical point of view, biofeedback training has increased 4.16 points IP in patients in whom the method has been effective. It has prolonged the mean duration of voluntary contraction in 5.9 sec. and elevated in 13.6 mm. Hg. the maximal pressure contraction. In two patients, clinical evolution and manometry measurements indicated surgical treatment and PSARP was performed, improving IP in 5.5 points. We considered biofeedback training, when indicated, an adequate and innocuous technique to achieve an important clinical continence improvement in these patients.

Adolescent↗
PubMed1996

A familial disorder with duodenal atresia and tetralogy of Fallot.

We report on two sibs with tetralogy of Fallot (TOF) and duodenal atresia (DA). The first child, a 6-year-old girl, had a right facial palsy in addition to the TOF and DA. Her brother, age 10 months, was born with bilateral microtia without facial palsy. The children are the product of an apparently non-consanguineous union between clinically normal parents. The pertinent family history includes a paternal aunt with TOF and a cleft lip and palate who died in childhood and another paternal aunt with a supernumerary thumb. This family has anomalies found in several syndromes, but does not meet the diagnostic criteria for any of them. The genetic basis for this condition remains unknown, but the pattern of inheritance is likely either autosomal recessive, or autosomal dominant with variable expression and reduced penetrance. The pathogenesis is unknown, but either a disturbance in neural crest cell migration or familial predisposition to vascular disruption might explain this pattern of malformations.

Child↗
PubMed1988

Congenital atresia and stenosis of the duodenum: the impact of a prenatal diagnosis.

This review was undertaken to determine the effects of prenatal diagnosis, on the basis of ultrasonographic evidence, of a duodenal obstruction on the care and morbidity of the newborn infant. The records of 46 cases of either atresia or stenosis of the small bowel in infants treated during the last six years in a tertiary perinatal center were reviewed retrospectively. A prenatal fetal diagnosis of obstruction was made in 13 patients (12 obstructions located in the duodenum and one in the jejunum). The mean gestational age at prenatal diagnosis was 33.7 weeks (range 27 to 37 weeks). The course of those infants with the prenatal diagnosis made by ultrasound was compared with that of those infants in whom the diagnosis was established after the onset of symptoms of obstruction appeared. Such a prenatal diagnosis resulted in earlier surgical intervention (1.8 versus 3.9 days). Metabolic complications secondary to repeated vomiting were found to be less frequent. The expectant mother in whom such a fetal prenatal diagnosis has been made can be transported to a tertiary level perinatal center where neonatal and specialized surgical expertise are available. After birth, the infant can undergo immediate operation without being separated from its mother.

Abnormalities, Multiple↗
PubMed2004

Choledochal cyst and duodenal atresia: a rare combination of malformations.

A rare case of congenital duodenal atresia (DA) associated with a choledochal cyst (CC) is reported. At 38 weeks of gestation, a 1,610-g girl was born by cesarean section with a prenatal diagnosis of congenital DA. After the disease was confirmed by radiographs, she underwent a duodenoduodenostomy for complete separation of the duodenum with an annular pancreas. Thirty-two months after the initial operation, she developed upper abdominal pain and acholic stools. Abdominal ultrasonography demonstrated a CC and dilated intrahepatic bile ducts. Magnetic resonance cholangiopancreatography showed an anomalous arrangement of the choledochus and the main pancreatic duct. A diffusely dilated extrahepatic bile duct was resected, and a hepaticoduodenostomy was performed after a cholecystectomy. The patient was discharged without complications. We could not find a similar case report in the English literature. Although it is not reported that there is a close relation of DA and CC in embryologic development, the presence of this combination should be considered.

Abnormalities, Multiple↗
PubMed2002

Choledochal cyst and duodenal atresia: a rare combination.

A rare case of congenital duodenal atresia (DA) associated with a choledochal cyst (CC) is reported. At 38 weeks of gestation, a 1,610-g girl was born by cesarean section with a prenatal diagnosis of DA. After the disorder was confirmed by X-ray, she underwent a duodenoduodenostomy for a complete separation of the duodenum with an annular pancreas. Thirty-two months after the initial operation, she developed upper abdominal pain and acholic stools. Abdominal ultrasonography demonstrated a CC and dilatated intrahepatic bile ducts. Magnetic resonance cholangiopancreatography showed an anomalous arrangement of the choledochus and main pancreatic duct. A diffusely dilatated extrahepatic bile duct was resected, and a hepaticoduodenostomy was performed after cholecystectomy. The patient was discharged without complications. We could not find a similar case report in the English literature. Although it is not reported that there is a close relationship in embryologic development of DA and CC, one should be aware of the possibility of this combination.

Child, Preschool↗
PubMed1989

Late gastrointestinal bleeding and protein loss after distal small-bowel resection in infancy.

Four children who underwent extensive small-bowel resection in infancy developed recurrent iron deficiency anemia due to gastrointestinal bleeding between 4 and 12 years later. The initial resections were required for multiple ileal atresia (n = 2) and gastroschisis (n = 2). Three patients have had melena and one had persistently guaiac-positive stools. Three patients had protein-losing enteropathy, and in one there was persistent hypoalbuminemia. Colonoscopy identified circumferential ulcerative lesions at the surgical anastomosis (n = 2) and at laparotomy another patient had well-defined linear ulcers close to the surgical anastomosis. Histology demonstrated focal ulceration with chronic inflammation, but did not show granulomata, crypt abscesses, or malignancy. Multiple imaging procedures and gastroduodenoscopy failed to identify an alternative bleeding source. Medical therapy including iron, antacids, sucralfate, H2 antagonists, and cholestyramine was ineffective. Two patients have undergone anastomotic resection. One experienced symptomatic recurrence 4 months after surgery. Repeat colonoscopy found ulceration at the new anastomosis with similar histology. The other patient remains asymptomatic 7 months postsurgery. Recurrent gastrointestinal hemorrhage due to anastomotic ulceration, of unknown etiology, appears to be a late complication of small-bowel resection in infancy.

Abdominal Muscles↗
PubMed2000

Apoptosis in murine duodenum during embryonic development.

Duodenum is thought to go through a solid-core stage followed by recanalization during its development. This study investigates the role of apoptosis in normal duodenal development, especially during widening of the lumen, and hence, the possible role of apoptosis in duodenal atresia (DA). Twenty-four time-mated Sprague-Dawley rats were killed from day 13 to day 20 of gestation. Duodenums of 3 fetuses were chosen randomly from each rat and processed. Apoptosis was determined by the terminal deoxytransferase-mediated biotin dUTP nick-end labeling (TUNEL) technique (ApopTag). Apoptosis count and cross-sectional areas were measured with an image analyzer (MetaMorph). The number of apoptotic cells per unit area duodenum peaked on day 15 for the mucosal/submucosal layer and on day 14 for the muscular/mesenchymal layer. The maximal number of apoptotic cells per cross-section of duodenum was between 7 and 8. The cross-sectional areas of the duodenal wall and lumen increased exponentially between day 17 and day 19 while duodenal-wall thickness remained relatively constant throughout duodenal development. The localization, timing, and intensity of apoptosis do not suggest that apoptosis is responsible for the widening of the duodenal lumen; enlargement of the lumen is related to the increase in duodenal circumference. Apoptosis thus may not be involved in the pathogenesis of DA.

Animals↗
PubMed1995

Prospective evaluation of the absorptive capacity of the bowel after major and minor resections in the neonate.

The outcome after major bowel resection in the neonatal period depends primarily on the time needed for bowel adaptation. A prospective study was begun in neonates after small bowel resection to evaluate the absorptive capacity of the bowel and growth parameters as a result of adaptation of the bowel. Twenty-four neonates who underwent bowel resection were included. The underlying diagnoses were necrotising enterocolitis (12), jejunal atresia (3), meconium peritonitis (3), and other (6). During the study, a standardized treatment with respect to nutrition was followed. At predetermined times, enterostomy fluid or faeces were collected for analysis of carbohydrate content, fat content, and fatty acids, together with a xylose test and a hydrogen breath test. Growth parameters included weight and height. The 24 patients were divided into two groups. Group A consisted of eight patients with short bowel syndrome (defined as loss of more than 50% of the original small bowel length related to gestational age). The mean remaining small bowel length in this group was 34.0% (24% to 42%). Group B consisted of 16 patients who had only minor bowel resections. Retarded growth was observed in four group A patients. Low levels of carbohydrate absorption were found 2 and 4 weeks after the initial operation in group A, and 2 weeks after the initial operation in group B. Low levels of fat absorption were found 4 and 8 weeks after the initial operation in group A. The coefficient of absorption of the different fatty acids showed complete absorption of caprylic acid.(ABSTRACT TRUNCATED AT 250 WORDS)

Case-Control Studies↗
PubMed2005

Annular pancreas in two consecutive siblings: an extremely rare case.

Annular pancreas is the rare congenital anomaly where the pancreas forms a full or incomplete ring around the second segment of the duodenum, causing various degrees of stenosis or atresia. It is estimated that it appears in 1 out of 12 000-15 000 births of living neonates and until now, in the literature, only 6 cases have been reported among individuals of the same family. We present the case of two siblings, a boy and a girl, with annular pancreas from consecutive pregnancies of the same couple. Both neonates had a prenatal diagnosis of duodenal obstruction and they underwent duodenoduodenal, proximal transverse to distal longitudinal anastomosis. Furthermore, the girl had a mobile ascending colon. Their postoperative condition was perfect. The case we are reporting is an addition to the other 6 cases of familial presentation of annular pancreas and is similar to one of them. In these families, a total of 16 persons present this congenital anomaly while 14 are seemingly healthy. Twelve of the affected persons are female and 4 male. In conclusion, it can be stated that female individuals seem to have a greater propensity to transmit the disease to their descendants, compared to males, suggesting the possible action of an autosomal recessive sex-influenced gene. The recording of such rare family cases should be encouraged, in order to fully recognize a possible type of inherited transmission.

Anastomosis, Surgical↗
PubMed1988

Duodenal atresia: late follow-up.

In this study, 41 randomly chosen patients aged 15 to 35 years (mean 22 years) were carefully examined. As primary operations there were 13 membrane excisions, five duodenoduodenostomies, 22 duodenojejunostomies, and one gastrojejunostomy. Twenty-eight patients were symptom-free, ten admitted some discomfort, three had major pains, including one with a history of duodenal ulcer. Reoperation for adhesion ileus had been performed in six patients, in the early postoperative phase in one instance. At late follow-up barium meals (N = 41) showed completely normal findings in two cases only, hiatal hernia in two, gastritis in three, duodenogastric reflux in 12, slight dilation of the duodenum with good emptying and no reflux in 16, a huge duodenal sac in nine, diminished peristalsis in eight, delayed emptying in five, slight luminal narrowing in three, duodenal diverticuli in nine, bezoars in two, and a polyp in the duodenum of one patient. Ultrasound (N = 35) revealed a gallbladder septum in one patient and a dilated common bile duct in another; in one subject the gallbladder was not visualized satisfactorily. Isotope biligraphy (N = 15) showed biliary reflux to the stomach in 12 cases. Endoscopy (N = 20) findings were: esophagitis (1), hiatal hernia (2), gastric mucosa in the lower esophagus (2), biliary reflux (9), gastritis (7), gastric polyps (2), dilated duodenum of variable degree (19), diminished peristalsis (4), marked retention (2), abnormal papilla (3), diverticuli (4), and a persistent membrane (1). Histology showed superficial gastritis in three patients. E coli was cultured from the duodenal juice in five patients and Candida found in two.(ABSTRACT TRUNCATED AT 250 WORDS)

Adolescent↗
PubMedDate not supplied

Duodenal atresia and stenosis: reassessment of treatment and outcome based on antenatal diagnosis, pathologic variance, and long-term follow-up.

Duodenal atresia and stenosis was observed in 103 infants and children from 1972 to 1991. There were 59 girls and 44 boys. Atresia was noted in 79 instances and stenosis in 24. Maternal hydramnios was detected in 33 cases, 46 babies were premature, and 31 had Down's syndrome. Fifty-four infants had significant associated anomalies including 35 with cardiac defects. Diagnosis was achieved by prenatal ultrasound examination in 14 cases, observation of a double-bubble sign on abdominal radiograph in 73, and contrast studies in 30 infants including 24 with stenosis. At operation annular pancreas was noted in 37 cases, malrotation in 37 cases, anterior portal vein in 4, and a second web in 3. Surgical treatment included duodenoduodenostomy in 85, duodenotomy and web excision in 8, and duodenojejunostomy in 10. Operative survival was 95%. Deaths were related to complex cardiac defects. Despite antenatal diagnosis, prompt intervention, and apparent early surgical success (95% survival), late deaths (5%) and late complications including motility disorders, megaduodenum, gastroesophageal reflux, duodenal-gastric reflux, gastritis, peptic ulcer disease, blind loop syndrome, and biliary-pancreatic conditions may be observed months to years after management during the neonatal period. Modifications in surgical technique including early tapering duodenoplasty may be useful, and close long-term follow-up is an essential component of patient care.

Anastomosis, Surgical↗
PubMed2005

Novel procedures for enhancing high jejunal atresia repair: bilateral side-plication and plication before anastomosis.

In high jejunal atresia (HJA), discrepancy between the diameters of the proximal and distal jejunum can often be significant and cause major anastomotic complications. We developed 2 novel procedures to enhance conventional jejuno-jejunostomy (JJ): bilateral side-plication during JJ (BSP): plication of both lateral aspects of the dilated proximal jejunum, and jejunal plication before JJ (pre-P): plication of the jejunum proximal to the planned JJ site along the anti-mesenteric line. We used BSP in a neonate with a membranous stenosis 5 cm distal to the duodeno-jejunal flexure, and pre-P in 2 neonates, both with HJA and hugely dilated proximal jejunum. By using BSP, the jejunum could be plicated symmetrically near the closed jejunotomy, allowing the caliber change at the closed jejunotomy to be smooth without any kinking. If simple anti-mesenteric plication had been performed instead, proximal-to-distal kinking caused by closing the jejunotomy site transversely after the membrane was excised could have worsened. By using pre-P, the caliber change across the JJ could be made smooth and symmetric. If plication had been performed along the anti-mesenteric line after completing the JJ, the proximal jejunum near the JJ would have become asymmetrical causing the JJ to become kinked. Postoperatively, there were no JJ-related complications, and all subjects are well after a mean follow-up period of 3.0 years. Our novel procedures are simple and appear to enhance JJ by controlling kinking at the JJ site.

Dilatation, Pathologic↗
PubMed1998

Jejunoileal atresia: a 27-year experience.

PURPOSE: In this study, the authors review cases of jejunoileal atresia (JIA) to evaluate their surgical treatment strategy. METHODS: Eighty-eight neonates who underwent surgical repair for JIA were divided into four groups for the type of lesion: group 1, membranous (n = 23), group II, interrupted (n = 49), group III, multiple (n = 9), and group IV, apple-peel (n = 7). Group I patients were treated with membranectomy or bowel resection and anastomosis, group II with resection of the dilated bowel and one anastomosis, group III with two to six multiple anastomoses to preserve bowel length, and group IV with minimal bowel resection and bowel anastomosis. During surgery a uniform protocol was used to minimize bowel resection and to perform an end-to-end single layer anastomosis using either Halsted horizontal mattress or conventional interrupted sutures. Mortality, morbidity, days for functional recovery, and central venous nutrition (CVN) were included in the review. RESULTS: Of 88 patients, three died of causes unrelated to operation for JIA. Nine patients underwent an additional laparotomy for leakage (n = 4) and obstruction (n = 5). Oral feeding was allowed on day 5.4+/-4.3 and full caloric intake via the enteric route on day 12.5+/-10.0. Twenty-one patients required CVN for 32.4+/-19.1 days. None required a long-term treatment for the short bowel syndrome. CONCLUSION: This study concludes that efforts to preserve bowel length are laudable to avoid the short bowel syndrome and that an end-to-end single layer anastomosis contributes to early recovery of bowel function.

Anastomosis, Surgical↗
PubMed1998

Increased frequency of cystic fibrosis among infants with jejunoileal atresia.

There appears to be an increased frequency of cystic fibrosis (CF) among infants with jejunoileal atresia (JIA). However, the figures vary widely, and no population-based data exist. The purpose of this study was to quantitate the magnitude of the association between JIA and CF in Atlanta using population-based data from 1968 to 1995. Case subjects included all infants with isolated JIA born during 1968-1995 to mothers residing in the five-county metropolitan Atlanta area at the time of birth. To ascertain cases, we reviewed records of the Metropolitan Atlanta Congenital Defects Program (MACDP), a population-based birth defects registry. Caucasian JIA cases were cross-referenced with patients in the CF registry at the Egleston Cystic Fibrosis Center at Emory University to more completely ascertain the diagnosis of CF among JIA cases. During 1968-1995, MACDP ascertained a total of 94 isolated JIA cases, for a birth prevalence of 1.8/10,000 live births. Among the cases, 38 were Caucasian, 52 were African-American, and 4 were of Asian or Hispanic ethnicity. Four of the 38 Caucasian JIA cases (11%) also had CF. The expected number of JIA cases with CF is 0.019 based on the estimated population incidence of 1/ 2,000 for CF. The observed to expected (O/E) ratio of Caucasian JIA cases with CF is greater than 210 (P<0.0001). Caucasian infants with JIA have more than 210 times the risk for CF compared with Caucasian infants in the general population. The results of this study have implications for the management of infants born with JIA and genetic counseling for families with affected infants.

Academic Medical Centers↗
PubMed1996

Adults born with high anorectal atresia--how do they manage?

PURPOSE: We are interested in the way patients, who underwent surgery for high anorectal atresia, control their defecation. Considering that some patients, despite newer operative techniques, always will suffer from minor or major soiling we attempted to find some guidelines for postoperative support for future patients. METHOD: Fifty-eight patients (median age, 26 (range, 18.1-56.9) years) were personally interviewed. RESULTS: Regulating defecation is done in five different modes: 16 patients have stools after urge, 15 control their stools mainly by going to the toilet at regular times, 18 perform bowel-irrigations or use enemas, 2 have loss of feces continuously, and 7 patients have an ileostomy or colostomy. More than one-half of patients influence their defecation by diet. Of the patients with anal defecation, 6 never soil, 39 sometimes soil small amounts, and 6 often soil seriously. Eighteen patients occasionally suffer from constipation. There is no mode of defecation regulation outstanding in preventing soiling or constipation. However, patients who do not regulate defecation somehow suffer from serious soiling. Most patients are content with their level of cleanliness. CONCLUSION: Irrespective of the mode of defecation regulation, many patients soil sometimes small amounts and a few often soil seriously. In view of the fact that most patients had to find the current control of defecation regulation by themselves rather late and lacked professional support, it is questionable whether the chosen mode of defecation regulation is the most optimal mode for each patient. We assume that a stepwise protocol under professional support, starting by the most natural mode of defecation, will improve defecation regulation in a more efficient way (earlier and better).

Activities of Daily Living↗