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PubMed2004

Ileocaecal valve atresia: our surgical approach.

BACKGROUND: Atresia of ileocaecal valve is the rarest type of intestinal atresia; there are very few reports on it in the literature and all cases described to date were treated with ileocolic resection. METHODS: We present a rare case of ileocaecal valve atresia, the third in the literature, in which a "different" technical approach was employed as an alternative to the usual surgical technique (ileocolic resection). The described case came to our attention in a patient presenting with complicated intestinal perforation in whom we performed an ileal stoma after an unsuccessful conservative approach. Approximately four months later we operated her to close the stoma: during this procedure we discovered the colic valve atresia and treated it by creating a "new ileocaecal valve" and carrying out an appendectomy. After a 7-year follow-up no complications have occurred and the girl enjoys very good health. CONCLUSIONS: We conclude that performing a plasty of Bauhin's valve is a valid alternative to intestinal resection in such cases of atresia even if, according to some authors, the resection of the ileocaecal valve was not associated with increased morbidity and mortality (3).

Digestive System Surgical Procedures↗

PubMed1987

Familial multiple jejunal atresia with malrotation.

Two siblings with multiple jejunal atresia and malrotation of the midgut are reported. In the first, incomplete rotation of the gut was associated with two jejunal atresias and an intraluminal diaphragm. In the second, there was complete failure of rotation of the gut with two jejunal atresias, one of which had a V-shaped mesenteric defect. I am unaware of any previous reports of familial multi level small intestinal atresias with malrotation. Possible reasons for this association are discussed.

Female↗

PubMed1997

Additional congenital anomalies in babies with gut atresia or stenosis: when to investigate, and which investigation.

A wide variety of additional congenital anomalies occur in babies born with a gut atresia or stenosis. The specific pattern of anomalies depends on the location of the atresia. The serious nature of many of them makes perioperative diagnosis imperative. Eighty-six babies born with pure oesophageal atresia (OA), duodenal atresia (DA) or stenosis, or jejuno-ileal atresia (JIA) have been studied. These, combined with over 2,000 cases in the literature, have been used to develop a protocol to optimally investigate babies with gut atresia for associated anomalies. The authors recommend routinely obtaining anterio-posterior and lateral chest and abdominal radiographs for babies with pure OA, DA and intestinal atresia, making sure the entire spine can be visualised. Cardiac and renal ultrasonography (US) should be routine in all babies with pure OA or DA. A micturating cystourethrogram should be done in those babies with abnormal urinary tract US or an associated anorectal anomaly. A sweat test should be obtained in babies with JIA, and a rectal biopsy should be taken in babies with the combination of Down's syndrome and DA to exclude Hirschsprung's disease.

Abnormalities, Multiple↗

PubMed1993

Congenital abnormalities of internal organs and body cavities.

Many of these malformations are reported sporadically, but a few are common, and several have important clinical implications. One example is schistosomus reflexus, which is always a challenge for veteran clinicians and often baffling to inexperienced obstetricians. The recent finding that early palpation of the amniotic vesicle can cause intestinal atresia in calves is extremely significant for dairy practitioners. Finally, there is the ethical question in breeding animals--repairing defects that are thought to be genetic, such as atresia ani, scrotal hernia, and umbilical hernia.

Animals↗

PubMed2006

Atresia of the ileocecal junction with agenesis of the ileocecal valve and vermiform appendix: report of a case.

Intestinal atresia involving the ileocecal region is a very rare intestinal malformation, and the presence or absence of the ileocecal valve influences its surgical management. We report the case of a male newborn with a provisional diagnosis of distal ileal atresia, in whom laparotomy revealed that the entire ileocecal region was atretic with an absent ileocecal valve and appendix vermiformis. We resected the dilated terminal ileum together with the atretic segment and performed an ileocolic anastomosis between the terminal ileum and the transverse microcolon without valve reconstruction. When last seen, 8 months after the operation, the baby was developing normally. Ileocolic anastomosis without valve replacement appears to be sufficient if an ileocecal valve is completely absent and only a short segment of the terminal ileum is lost.

Abnormalities, Multiple↗

PubMed1984

[Importance of infection following laparotomy in childhood].

Laparotomy was performed on 579 children at the University Clinic of Paediatric Surgery in Mainz from 1.1.1975 to 31.12.1982. The children were up to 15 years of age; appendicitis or inguinal and umbilical hernia cases were not included. Postoperative sepsis occurred in 74 patients (12.8% of all children with laparotomy); in 51 cases positive bacteriological findings were seen besides the clinical and clinicochemical ones. Sepsis morbidity was particularly high in children who had not yet completed their first year of life (postoperative sepsis occurring in approximately every fourth infant); among the disease patterns, the following were particularly prominent: Defects of the abdominal wall (23 out of 50 children developed postoperative sepsis); intestinal atresia (18 out of 59 children); intestinal perforation (11 out of 39 children). In addition, sepsis morbidity was enhanced after relaparotomies. Gram-negative bacteria were most frequent among the 51 patients with bacteriologically positive findings; these bacteria consisted mostly of representatives of the group of enterogenous pathogens. These groups of bacteria were also the most frequently occurring pathogens in mixed and secondary infections. 33 out of 74 children with postoperative sepsis died. The mortality rate was 68% in prematurely born infants compared with mature newborns. Lethality was highest among children with congenital defects of the abdominal wall and intestinal perforations. Among the patients with bacteriologically positive findings the lethality was particularly high with multiple attacks of sepsis, in case of septitides caused by multiple pathogens, by Candida albicans and after relaparotomy.

Adolescent↗

PubMedDate not supplied

[Clinical analysis of congenital abdominal wall defects--omphalocele and gastroschisis].

INTRODUCTION: This is a retrospective analysis of therapeutic approaches and treatment outcomes of congenital abdominal wall defects (omphalocele and gastroschisis) in a five year period. The aim of this study was to identify factors which can affect the prognosis of future therapeutic procedures. MATERIAL AND METHODS: We evaluated 13 children, 7 with omphalocele (2 female/5 male; mean birth weight of 2862 g; mean gestational age of 37 weeks), and 6 patients with gastroschisis (2 female/4 male; mean birth weight of 2640 g; mean gestational age of 36/2 weeks). All patients were treated at the Clinic of Pediatric Surgery from 1999 to 2003. RESULTS: In this study, thirteen cases of congenital abdominal wall defects (omphalocele and gastroschisis) were retrospectively investigated. All patients underwent prenatal ultrasound. Omphalocele was prenatally detected in 42.8% of fetuses, and gastroschisis in 16.7%. Coexisting anomalies were present in 57.1% of patients with omphalocele and in 16.7% of newborns with gastroschisis. Three patients with omphalocele were treated operatively, and four only conservatively. The abdominal wall of patients with gastroschisis was primarily closed in three patients. Two patients required a staged abdominal wall closure. One patient with gastroschisis and intestinal atresia underwent primary closure after partial intestinal resection and enterostomy. Based on these responses, a management protocol (algorithm) was recommended. The most common postoperative complication, in 7 cases of omphalocele, was mechanical ileus (n = 1), whereas among patients with gastroschisis the commonest were mechanical ileus (n = 1) and intestinal perforation (n = 1). The mortality of patients with omphalocele was 52% and with gastroschisis 66.7%. CONCLUSION: A strategy designed to optimize antenatal and neonatal factors is expected to increase the survival rate of patients with abdominal wall defects.

Abnormalities, Multiple↗

PubMed1983

Management of the newborn with gastrointestinal anomalies and tracheoesophageal fistula.

A standard approach to the otherwise healthy newborn with esophageal atresia and tracheoesophageal fistula has been well established. A significant number of these infants have additional gastrointestinal anomalies that necessitate alteration of the usual operative management. Over 2 1/2 years, nine such newborns have been managed. There were 17 additional gastrointestinal abnormalities, the most frequent being an imperforate anus (5), malrotation (5), and intestinal atresia (4). Management consisted of initial formal laparotomy, thoracotomy, and central line placement. In four infants these procedures were performed at a single sitting. Eight infants survived (89 percent); the single death resulted from intracerebral hemorrhage. Detection of coexisting gastrointestinal anomalies is mandatory in the management of the newborn with esophageal atresia. Standard local gastrostomy and subsequent thoracotomy may delay recognition of these associated anomalies, increase morbidity, and complicate subsequent correction. Preliminary abdominal exploration with correction, diversion, or both followed by thoracotomy, either immediate or staged, is indicated.

Abnormalities, Multiple↗

PubMed1998

Prenatal and neonatal intussusception.

Intussusception found in the 1st month of life is rare and usually discussed as one entity, neonatal intussusception, but in fact, includes the intussusceptions occurring both prenatally and neonatally, of which the clinical presentations and results are different. Four full-term babies with prenatal intussusception presenting as intestinal atresia (IA) and three premature babies with neonatal intussusception mimicking necrotizing enterocolitis (NEC) are presented. Prenatal intussusception, as one of the causes of IA produces prominent signs of intestinal obstruction immediately after birth. Preoperative evaluation usually fails to yield a definitive diagnosis, but surgery is usually performed in time and is successful. In neonatal intussusception, full-term infants usually have a pathological lead point and the colon is almost always involved. A barium enema is thus useful in diagnosis. Premature babies, on the other hand, rarely have a colonic component, and the clinical features are insidious and similar to NEC. This results in diagnostic confusion that may lead to a dangerous delay in appropriate surgical correction. A high level of suspicion about this condition in cases diagnosed presumptively with NEC is important. Serial abdominal sonograms may be helpful in the early diagnosis of neonatal intussusception.

Fatal Outcome↗

PubMedDate not supplied

Congenital pyloric atresia: the spectrum.

Congenital pyloric atresia (CPA) is a very rare malformation. It can occur as an isolated lesion or in association with other genetically determined conditions such as epidermolysis bullosa or aplasia cutis congenital, or form part of the hereditary multiple intestinal atresias syndrome. Five newborns with CPA representing the spectrum are presented. The clinical features, diagnosis, and outcome are also discussed.

Female↗

PubMed2000

Variability in the phenotypic expression of fryns syndrome: A report of two sibships.

We report on two sibships with four fetuses of 12, 15, 17, and 20 weeks of gestation, respectively, and 1 preterm baby of 31 weeks of gestation affected by a multiple congenital disorder with manifestation suggestive of Fryns syndrome. In addition to the characteristic malformation pattern in Fryns syndrome, they presented with fetal hydrops, cystic hygroma, and multiple pterygias, allowing prenatal ultrasound diagnosis as early as in the 11th week of gestation. The two affected fetuses of family 1 showed severe craniofacial anomalies with bilateral cleft lip and palate, acral hypoplasia, postaxial oligodactyly, persistent truncus arteriosus, and interrupted aortic arch, asplenia sequence, and complex central nervous system midline malformations. In family 2 with three affected sibs, ear anomalies with atresia of the auditory canals, postaxial hexadactyly, intestinal atresias, callosal defects, and eye colobomas were the most outstanding features. On the basis of the present findings and former reports, the inter- and intrafamiliar phenotypic variability in Fryns syndrome, possible pathogenetic mechanisms, and the value of prenatal diagnosis are discussed. In the pathogenetic discussion, a special emphasis is put on the neural crest cell developmental field.

Abnormalities, Multiple↗

PubMed1998

Staged silo repair of gastroschisis with preservation of the umbilical cord.

BACKGROUND: The optimal surgical approach for gastroschisis remains controversial, although primary closure after vigorous stretching of the abdominal wall and decompression of the intestinal contents is currently preferred. METHODS: Between 1984 and 1997, 24 newborns with gastroschisis were treated at Saitama Children's Medical Center. The average gestational age was 37.3 weeks, and the average birth weight was 2,285 g. One patient had the associated anomaly of intestinal atresia and short bowel. Rupture of the intestines during delivery was noted in one patient. The authors applied their nonaggressive staged repair using a prosthetic silo with preservation of the umbilical cord in 20 of the 24 cases (83.3%). Primary closure with preservation of the umbilical cord was performed in the remaining four cases (16.7%). In these patients, the gastroschisis was mild. RESULTS: In the 20 cases treated by staged repair, the average interval between the first and second operation was 9.8 days. Mechanical ventilation was not required in 16 of 20 (80%) patients treated by staged repair, or in two of four (50%) patients treated by primary repair. The number of days to the first feeding averaged 14.6 days in 23 cases, excluding the patient with short bowel syndrome who required continuous total parenteral nutrition (TPN). TPN through a central venous catheter was required in 3 of 23 patients (13.0%). The overall average hospital stay was 55.1 days. Survival was 24 of 24 or 100%. Complications included perforation of the intestines, gastric bleeding, ventral hernia, and wound infection. No infections were associated with the prosthetic silo. All of the patients had a satisfactory cosmetic outcome. Recent advances in neonatal intensive care, including antibiotic therapy, reduced the possibility of infection. CONCLUSIONS: This staged repair of gastroschisis was simple and safe, neither requiring experienced surgical judgment nor complicated postoperative management, and achieved satisfactory results. Furthermore, preservation of the umbilical cord provided an improved cosmetic appearance.

Abdominal Muscles↗

PubMed1996

A decade of gastroschisis in the era of antenatal ultrasound.

BACKGROUND: Gastroschisis is an uncommon condition in which viscera protrude at the base of the umbilical cord. To investigate the possible relationships between antenatal ultrasound findings, patient demographics, smoking, alcohol consumption and this condition, 21 causes are reviewed. METHODS: The medical records and antenatal ultrasounds of 21 children diagnosed with gastroschisis at the Adelaide Children's Hospital between 1 January 1985 and 31 December 1992 were reviewed. RESULTS: Antenatal ultrasound was employed in 15 cases, and the diagnosis was accurately made in 13 (86.7%) of these. There were 17 live births, two elective terminations and two pre-term abortions. Seven of the 21 cases had associated anomalies. The anomalies included five atresias, a ventricular septal defect (VSD), and a dislocated gall-bladder. Postoperative complications (which included one death) occurred in seven of the 17 patients. Bowel dilatation or thickening was first detected on five ultrasound examinations performed before 21 weeks' gestation, and four ultrasounds after 21 weeks. The nine cases with bowel changes on ultrasound were associated with a high atresia rate and a longer hospital stay, but not with an increased complication rate. Maternal race, parity, and alcohol consumption were not associated with increased risk of fetal gastroschisis. All mothers were under 27 years of age. There were nine mothers who smoked during pregnancy and a disproportionate number of mothers who lived outside the metropolitan area with gastroschisis-affected offspring. CONCLUSIONS: Bowel changes seen on antenatal ultrasound increase the chances of intestinal atresia and longer hospitalization. Smoking during pregnancy may be associated with an increased risk of gastroschisis.

Abdominal Muscles↗

PubMed2002

Omphalocele: beyond the size issue.

A small omphalocele was diagnosed and followed up prenatally. Ultrasonography examinations and clinical examination at birth were all consistent with an isolated, small omphalocele. Immediate surgical exploration at birth found an associated type I intestinal atresia. This rare association and the need for immediate postnatal management of omphalocele are discussed, and the literature is reviewed.

Adult↗

PubMed1986

Additional anomalies in Hirschsprung's disease: an analysis based on the nationwide survey in Japan.

A total number of 1628 cases with Hirschsprung's disease were collected in a nationwide survey; 181 of the cases had additional anomalies. The overall incidence of additional anomalies was 11.1% (181/1628). Down's syndrome and cardiac anomalies occurred in 2.9% and 2.5%, respectively: A greater incidence than seen in the general population. The longer the aganglionic segment, the higher the incidence of additional anomalies. Megacystis and megaureter were less common. Intestinal atresia and stenosis were seen in 15 patients, the rate being 0.9%. The overall mortality in patients with additional anomalies was higher at 17.7% (32/181) than in those without.

Abnormalities, Multiple↗

PubMed1999

Vanishing gut in infants with gastroschisis.

Infants born with gastroschisis (GS) in association with intestinal atresia are well described, however, atresia to the extent where none or very little of the midgut remains has rarely been reported. In this paper we present one infant born with a GS defect in combination with total loss of the midgut and another with a tight GS defect with atresia of the proximal and distal midgut at the regions where the bowel had gone through the abdominal wall. On examining these and other case reports in the literature, we realised that there is a GS spectrum, at one end with simple, localised atresia with the rest of the gut exhibiting minimal vascular compromise and at the other a very small or no defect with absence of the entire midgut.

Fatal Outcome↗

PubMed2005

The basic biology of apoptosis and its implications for pediatric surgery.

Apoptosis, or programmed cell death, is an evolutionarily conserved and highly regulated process of nonfunctional cell death. Through this process, the body disposes of unwanted cells by self-destruction: it is our final defense against damaged cells. In the last decades, many of the essential pathways that control this phenomenon have been elucidated. Apoptosis plays an important role in developmental processes, as well as in cellular homeostasis. This process is known to be accelerated or diminished in many pathologic states. Therefore the understanding of apoptotic regulation has significant clinical ramifications. This article reviews the basic understanding of programmed cell death with respect to areas of interest to pediatric surgeons, including: Hirschsprung disease, intestinal atresias, testicular disorders, short bowel syndrome, ischemia-reperfusion injury and pediatric oncology. Pro or antiapoptotic interventions may become a future target for cell and organ protection in patients suffering from these diseases.

Apoptosis↗

PubMed1975

Experimental congenital malformations.

Traditional approaches to human and experimental teratology are briefly described, with roles of pathologists indicated. Some approaches to experimental teratology which pathologists might use are then described and illustrated. These include identification of subclasses of malformation types, study of the chronologic sequence of maldevelopment, study of embryo death, and examination of tumor-malformation relationships. The malformations used to illustrate these approaches are cleft palate, anencephaly, hydrocephalus, and intestinal atresia. The final section deals with general methodology in experimental teratology; a listing of books dealing with experimental teratology is included.

Abnormalities, Drug-Induced↗