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Short bowel syndrome (SBS) is a malabsorptive state occuring as a result of surgical resection or congenital disease of a significant portion of the small intestine . The amount of resection or remaining bowel generally dictates the degree of malabsorption and consequentely the need for specialized enteral nutrition or parenteral nutrition (PN). Intestinal failure in the context of SBS is defined as a dependence on PN to maintain minimal energy and fluid requirement for growth in children. Common causes of SBS in infants and children include necrotizing enterocolitis, midgut volvulus, intestinal atresia, and gastroschisis. Early identification of patients at risk for long-term PN dependency is the first step toward avoiding severe complications. Close monitoring of nutritional status, steady and early introduction of enteral nutrition, and aggressive prevention, diagnosis, and treatment of infections such as central venous catheter sepsis and bacterial overgrowth can significantly improve the prognosis. Intestinal transplantation is an emerging treatment that may be considered when intestinal failure is irreversible and children are experiencing serious complications related to TPN administration.
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PMID:Overview of pediatric short bowel syndrome. 1866 16

Most surgeons agree that complex neonatal ovarian cysts, regardless of size, warrant operative intervention. Management of simple cysts >4 cm is still controversial, although many favor intervention because of the increased risk of torsion. Whereas laparoscopic cyst resection is favored by some, others prefer less invasive percutaneous needle aspiration. We present a newborn infant who was admitted with sepsis and respiratory failure after home delivery. Ultrasound done on day 8 to check for umbilical venous line placement incidentally showed a simple cyst measuring 3.6 x 5.9 x 6.9 cm that was presumed to be of ovarian origin. Percutaneous needle aspiration was atraumatic and revealed serous fluid, with a high estradiol level. Four days later, surgery was indicated for clinical deterioration with suspected hemorrhage into the cyst. We found a midgut volvulus with extensive necrosis secondary to a jejunal duplication cyst. Ovaries were normal and there was no evidence of malrotation. Postoperatively, after discussion with the parents, support was withdrawn and the child died. We should not rely solely on ultrasonographic features and fluid characteristics to diagnose a large neonatal abdominal cyst, but rather confirm the diagnosis with laparoscopy.
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PMID:Lethal outcome after percutaneous aspiration of a presumed ovarian cyst in a neonate. 1934 4

Irreversible intestinal failure in children is predominantly caused by surgical conditions such as volvulus, necrotizing enterocolitis, and gastroschisis. Functional intestinal failure from motility disorders such as intestinal pseudo-obstruction or enterocyte dysfunction with microvillus inclusion disease also may require intestine replacement. Approved indications for intestinal transplantation include liver dysfunction, loss of major venous access, frequent central line-related sepsis, and recurrent episodes of severe dehydration despite intravenous fluid management. Surgical options include transplantation of the isolated intestine, combined liver-intestine transplantation, or multivisceral transplantation of the stomach, duodenum, pancreas, and small bowel (with or without the liver). Immunosuppression for intestinal transplantation is based on tacrolimus therapy, often with induction immunosuppression using antilymphocyte antibodies (eg, antithymocyte antibody and alemtuzumab). Experience at centers of excellence demonstrates 1- and 5-year patient survival rates of 95% and 77%, respectively, with ongoing investigations focusing on lowering long-term causes of graft loss such as chronic rejection.
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PMID:Current perspectives on pediatric intestinal transplantation. 1946 23

Internal herniation, defined as a protrusion of the bowel through a peritoneal or mesenteric aperture into a compartment of the abdominal cavity, is a potentially serious complication following Roux-en-Y gastric bypass. In view of the high risk of strangulation and volvulus with possible progression to necrotic bowel, sepsis and multiple organ failure, early diagnosis and prompt surgical intervention are crucial to minimise morbidity and mortality rates. As little is known about internal herniation, pathogenesis, consequences, clinical presentation and diagnostic approach are discussed in this article. A review of the literature and the consideration of three cases make it clear that diagnosis is often delayed due to a vague, non-specific clinical presentation and the frequently negative radiological investigations. Displacement of the superior mesenteric artery (SMA) with crowding, stretching and engorgement of its visceral branches, mesenteric swirl and a clustered appearance of small bowel loops are CT findings with a high specificity but low sensitivity. A high degree of vigilance, communication between radiologists and surgeons and a low threshold for surgical exploration is therefore necessary.
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PMID:Internal herniation after Roux-en-Y gastric bypass: case reports and a review of the literature. 1994 98

3 patients, 2 women aged 41 and 47 and one man aged 75 years, presented with abdominal pain and distension. In 2 patients the diagnosis 'sigmoid volvulus' was reached following plain abdominal X-ray. Both patients underwent sigmoidal resection with primary anastomosis after endoscopic deflation. The third patient proved to have a caecal volvulus on emergency laparotomy and underwent ileocaecal resection. In 2/3 of the cases diagnosis can be made by history, physical examination and conventional X-ray. Delay in the diagnosis increases the risk of peritonitis and death due to ischaemia and perforation of the colon. In the absence of peritoneal tenderness, signs of ischaemia or sepsis, the initial treatment consists of endoscopic deflation, which is successful in 68-78% of cases. Resection of the sigmoid colon is recommended a few days after endoscopic decompression in order to prevent recurrence. Caecal volvulus is not suitable for treatment with endoscopic deflation and should be treated with ileocaecal resection.
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PMID:[Colonic obstruction due to volvulus of the sigmoid colon or caecum]. 2029 24

Acute esophageal necrosis (AEN), commonly referred to as "black esophagus", is a rare clinical entity arising from a combination of ischemic insult seen in hemodynamic compromise and low-flow states, corrosive injury from gastric contents in the setting of esophago-gastroparesis and gastric outlet obstruction, and decreased function of mucosal barrier systems and reparative mechanisms present in malnourished and debilitated physical states. AEN may arise in the setting of multiorgan dysfunction, hypoperfusion, vasculopathy, sepsis, diabetic ketoacidosis, alcohol intoxication, gastric volvulus, traumatic transection of the thoracic aorta, thromboembolic phenomena, and malignancy. Clinical presentation is remarkable for upper gastrointestinal bleeding. Notable symptoms may include epigastric/abdominal pain, vomiting, dysphagia, fever, nausea, and syncope. Associated laboratory findings may reflect anemia and leukocytosis. The hallmark of this syndrome is the development of diffuse circumferential black mucosal discoloration in the distal esophagus that may extend proximally to involve variable length of the organ. Classic "black esophagus" abruptly stops at the gastroesophageal junction. Biopsy is recommended but not required for the diagnosis. Histologically, necrotic debris, absence of viable squamous epithelium, and necrosis of esophageal mucosa, with possible involvement of submucosa and muscularis propria, are present. Classification of the disease spectrum is best described by a staging system. Treatment is directed at correcting coexisting clinical conditions, restoring hemodynamic stability, nil-per-os restriction, supportive red blood cell transfusion, and intravenous acid suppression with proton pump inhibitors. Complications include perforation with mediastinal infection/abscess, esophageal stricture and stenosis, superinfection, and death. A high mortality of 32% seen in the setting of AEN syndrome is usually related to the underlying medical co-morbidities and diseases.
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PMID:Black esophagus: acute esophageal necrosis syndrome. 2061 76

Cecal volvulus is an uncommon cause of acute bowel obstruction in adults. The mechanism is torsion of the enlarged, poorly-fixed or hypermobile cecum. Patients with this condition may display highly variable clinical presentations, ranging from intermittent, self-limiting abdominal discomfort to acute abdominal pain associated with intestinal strangulation and sepsis. The treatment needs to be individualized for each case, but surgical management is required in almost every case. In the presence of gangrene or perforation of the cecum, resection and primary ileocolic anastomosis is recommended. However, in non-complicated cases detorsion and cecopexy are adequate. The authors report one case of cecal volvulus in a 55-year-old women treated with cecopexy that complicated with septic jaundice.
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PMID:Cecum volvulus complicated by septic jaundice. 2134 Feb 64

To identify causes of death (COD) in propositi with Cornelia de Lange syndrome (CdLS) at various ages, and to develop guidelines to improve management and avoid morbidity and mortality, we retrospectively reviewed a total of 426 propositi with confirmed clinical diagnoses of CdLS in our database who died in a 41-year period between 1966 and 2007. Of these, 295 had an identifiable COD reported to us. Clinical, laboratory, and complete autopsy data were completed on 41, of which 38 were obtainable, an additional 19 had autopsies that only documented the COD, and 45 propositi had surgical, imaging, or terminal event clinical documentation of their COD. Proband ages ranged from fetuses (21-40 weeks gestation) to 61 years. A literature review was undertaken to identify all reported causes of death in CdLS individuals. In our cohort of 295 propositi with a known COD, respiratory causes including aspiration/reflux and pneumonias were the most common primary causes (31%), followed by gastrointestinal disease, including obstruction/volvulus (19%). Congenital anomalies accounted for 15% of deaths and included congenital diaphragmatic hernia and congenital heart defects. Acquired cardiac disease accounted for 3% of deaths. Neurological causes and accidents each accounted for 8%, sepsis for 4%, cancer for 2%, renal disease for 1.7%, and other causes, 9% of deaths. We also present 21 representative clinical cases for illustration. This comprehensive review has identified important etiologies contributing to the morbidity and mortality in this population that will provide for an improved understanding of clinical complications, and management for children and adults with CdLS.
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PMID:Causes of death and autopsy findings in a large study cohort of individuals with Cornelia de Lange syndrome and review of the literature. 2206 64

We report three infants who presented with acute gastric volvulus and recovered initially after de-torsion, but later presented with sequelae due to ischemia of gastroesophageal junction, stomach and gastroduodenal junction. The first two infants could not be fed orally or by gastrostomy tube because of microgastria and stricture of the lower esophagus and gastroduodenal junction, and were managed on jejunostomy feeds, while the third child was managed on gastrostomy feeds till the gastric substitution surgery. The first case was treated nonsurgically with repeated dilatations, but ultimately succumbed to sepsis and malnutrition. In the second child, attempted dilatation resulted in esophageal perforation and she was reconstructed using ileocecal segment as a substitute for stomach and lower esophagus, and has done well. The third child was managed surgically by the Hunt Lawrence J pouch as stomach substitute and has also done well.
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PMID:Acute gastric volvulus: Late-onset ischemic consequences and their management. 2212 14

A diverticulum is a bulging sack in any portion of the gastrointestinal tract. The most common site for the formation of diverticula is the large intestine. Small intestine diverticular disease is much less common than colonic diverticular disease. The most common symptom is non-specific epigastric pain and a bloating sensation. Major complications include diverticulitis, gastrointestinal bleeding, acute perforation, pancreatic or biliary (in the case of duodenal diverticula) disease, intestinal obstruction, intestinal perforation, localized abscess, malabsorption, anemia, volvulus and bacterial overgrowth. We describe the clinical case of a 65-year-old female patient with a diagnosis on hospital admittance of acute appendicitis and a intraoperative finding of diverticular disease of the small intestine, accompanied by complications such as intestinal perforation, bleeding and abdominal sepsis. This was surgically treated with intestinal resection and ileostomy and a subsequent re-intervention comprising perforation of the ileostomy and stomal remodeling. The patient remained hospitalized for approximately 1 month with antibiotics and local surgical wound healing, as well as changes in her diet with food supplements and metabolic control. She showed a favorable clinical evolution and was dismissed from the hospital to her home. We include here a discussion on trends in medical and surgical aspects as well as early handling or appropriate management to reduce the risk of fatal complications.
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PMID:Diverticular disease of the small bowel. 2318 48


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