Cases reported "Short Bowel Syndrome"

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1/18. intestinal absorption and permeability in paediatric short-bowel syndrome: a pilot study.

    BACKGROUND: Sugar absorption tests are an effective, noninvasive way to assess intestinal permeability. The role of intestinal barrier integrity in complications and outcome of short-bowel syndrome is not known. The purpose of the study was to evaluate whether such tests provide information on the status of intestinal mucosa of these patients. methods: Six children with short-bowel syndrome--median age, 12 months, and median small bowel length at birth, 30 cm--had a sugar test with 3-o-methyl-D-glucose, D-xylose, D-rhamnose, and melibiose approximately 2 months after operation. The melibiose/L-rhamnose ratio was used as an index of permeability, and percentages of 3-o-methyl-D-glucose and D-xylose absorbed were used as indices of absorption. parenteral nutrition requirement, bowel length, liver disease, recent sepsis, and bacterial overgrowth were recorded. RESULTS: Three patients had increased permeability, and all of them had had a recent episode of sepsis and severe liver disease. All subjects had malabsorption of 3-o-methyl-D-glucose, and five of six had malabsorption of D-xylose and L-rhamnose. The absorption of 3-o-methyl-D-glucose correlated with bowel length (r2 = 0.78; P = 0.04), whereas the absorption of D-xylose correlated with parenteral requirement (r2 = 0.66; P = 0.04) at that time. CONCLUSIONS: Increased permeability was observed in three of six patients with short-bowel syndrome associated with a recent episode of sepsis and severe liver disease. Other indices of malabsorption correlated significantly with different clinical features of the disease. A prospective larger scale study in a homogeneous population is indicated to assess at multiple points during the disease course whether the test can be helpful in the management of these patients.
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2/18. short bowel syndrome.

    short bowel syndrome is an uncommon disease that results from extensive intestinal resection. Short bowel patients develop severe malabsorption of macronutrients, micronutrients, electrolytes and water, and pose difficult management problems. This report describes a typical patient with the short bowel syndrome and how each component of the malabsorption syndrome is managed to maintain nutritional, electrolyte, and water balance. In practice, some short bowel patients become dependent on parenteral nutrition for life, while others become independent with time due to intestinal adaptation and can be managed on oral intake and supplementations. Short bowel patients are at risk of developing gallstones, oxalate kidney stones and, rarely, d-lactic acidosis, and the pathophysiology of these disease processes is outlined. A minority of short bowel patients may ultimately require intestinal transplantation due to irreversible complications, and the current status of this intervention is reviewed. Finally, growth factors that stimulate intestinal growth and, thus, enhance absorptive capacity, are currently being identified and may eventually be introduced in the treatment of these patients.
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3/18. Conjugated bile acid replacement therapy reduces urinary oxalate excretion in short bowel syndrome.

    BACKGROUND: Patients with short bowel syndrome (SBS) have steatorrhea, in part because of bile acid malabsorption that causes decreased bile acid secretion into the duodenum and consequent fat maldigestion. In SBS patients with colon in continuity, luminal calcium forms calcium fatty acid soaps rather than precipitating as insoluble calcium oxalate. Soluble oxalate is hyperabsorbed by the colon leading to hyperoxaluria and an increased risk for renal calcium oxalate stones and deposits. The authors hypothesized that oral ingestion of conjugated bile acids would increase fat absorption and thereby decrease calcium fatty acid soap formation and oxalate hyperabsorption. methods: The effect of conjugated bile acid replacement therapy (9 g/d) on fecal fat excretion and urine oxalate excretion was measured in an appropriate patient, utilizing the metabolic balance technique. The effects of chronic bile acid replacement therapy on oxalate excretion and nutritional status also were measured in a 3-month outpatient study. RESULTS: Natural conjugated bile acid replacement therapy reduced fecal fat excretion from 119 to 79 g/d (Delta40 g/d), and urinary oxalate excretion from 87 to 64 mg/d (966 to 710 micromol/d; Delta23 mg/d). Cholylsarcosine, a synthetic conjugated bile acid, had similar but less powerful effects. During a 3-month outpatient trial of natural conjugated bile acids (9 g/d), urine oxalate decreased to normal levels (27 mg/d) in association with weight gain, decreased hunger, and decreased hyperphagia. CONCLUSION: Conjugated bile acid replacement therapy reduced fecal fat excretion, reduced urinary oxalate excretion, and improved nutritional status in a patient with SBS with colon in continuity, hyperoxaluria, and oxalate nephrolithiasis.
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4/18. Conjugated bile acid replacement therapy in short bowel syndrome patients with a residual colon.

    AIM: To test the efficacy of cholylsarcosine (synthetic conjugated bile acid) and ox bile extracts (mixture of natural conjugated bile acids) on fat absorption, diarrhea, and nutritional state in four short bowel syndrome (SBS) patients with a residual colon not requiring parenteral alimentation. methods: The effect of cholylsarcosine (2 g/meal) on steatorrhea and diarrhea was examined in short-term balance studies with a constant fat intake in all four patients. The effect of continuous cholylsarcosine ingestion on nutritional state was assessed by changes in body weight in three patients. In two patients, the effects of cholylsarcosine were compared with those of ox bile extracts. Because of the low incidence rate of SBS this is not a controlled study. RESULTS: In balance studies, cholylsarcosine increased fat absorption from 65.5 to 94.5 g/day (a 44 % increment), an energy gain of 261 kcal/d. Fecal weight increased by 26 %. In two patients natural conjugated bile acids also reduced steatorrhea, but greatly increased diarrhea. As outpatients consuming an unrestricted diet and ingesting cholylsarcosine, three patients gained weight at an average rate of 0.9 kg/week without worsening of diarrheal symptoms. CONCLUSIONS: Cholylsarcosine is efficacious and safe for enhancing fat absorption and nutritional status in short bowel syndrome patients with residual colon. Natural conjugated bile acids improve steatorrhea to a smaller extent and greatly worsen diarrhea.
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5/18. 53-years-old with mental status changes.

    D-lactic acidosis, a complication of short bowel syndrome, presents with a variety of neurological symptoms and metabolic acidosis. Treatment is hydration, replacement of nutritional deficiency replacement, and selective antibiotics. Prevention entails complex carbohydrate diet and vitamin and mineral supplements.
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6/18. Living-related small bowel transplantation: two cases experiences.

    We have recently performed living-related small bowel transplantation for 2 patients. The first patient was a 14-year-old boy with total parenteral nutrition (TPN)-dependent short-bowel syndrome associated with hypoganglionosis of the entire intestine. He received a bowel graft from his 43-year-old mother. The second patient was a 27-year-old woman, who had massive enterectomy due to volvulus and developed vitamin deficiencies and severe metabolic disorders as a result of long-term TPN. She underwent living-related bowel transplantation from her 57-year-old mother. Blood types were ABO identical, cytotoxic cross matches were negative, and cytomegalovirus statuses were positive-to-positive in both cases. Up to one third of the donor bowel was harvested from the donor distal ileum more than 30 cm away from the ileocecal valve. The graft vessels were connected to infrarenal aorta, and inferior vena cava. The immunosuppressive regimen consisted of daclizumab, tacrolimus, and steroid. The graft surveillance was accomplished using zoom endoscopy and mucosal biopsy. The first patient developed progressive acute cellular rejection (ACR) on the 9th postoperative day (POD)-9 requiring OKT-3 therapy, which was effective. Two months after transplantation, he was weaned from TPN, tolerating oral intake with a fully functioning graft. The second patient experienced no episode of ACR and was weaned off TPN on POD-29 with a functioning graft. Her metabolic disorder dramatically improved after bowel transplantation. Both donors had no complication and were discharged from the hospital on POD-10. Living-related bowel transplantation is an extreme option of treatment for patients with short-bowel syndrome.
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7/18. Recurrent D-lactic acidosis in a child with short bowel syndrome.

    D-lactic acidosis is a rare complication in children with short bowel syndrome. It results from fermentation of dietary carbohydrate by luminal bacteria in the small bowel caused by bacterial overgrowth. We present the case of a 14-year-old boy who had been diagnosed with short bowel syndrome from surgical treatment of midgut volvulus five years previously. His nutritional status was maintained by total parenteral nutrition and enteral feeding as tolerated. During hospitalization, episodic confusion and hyperpnea developed. The investigation showed severe metabolic acidosis with serum bicarbonate of 9 mmol/L and a wide anion gap. The serum D-lactic acid was 11.21 mmol/L. There was no evidence of renal or hepatic failure. Therefore, D-lactic acidosis from enteral carbohydrate overload was diagnosed. The treatment was correction of metabolic acidosis by sodium bicarbonate infusion and carbohydrate restriction. The results of the therapy were satisfactory. Early detection and appropriate treatment is necessary to avoid morbidity and mortality following this complication of short bowel syndrome.
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keywords = nutritional status, status
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8/18. Artificial sphincter with colonic reservoir for severe anal incontinence because of imperforate anus and short-bowel syndrome: report of a case.

    Anal sphincter replacement is a treatment option for severe anal incontinence that is not amenable to direct repair. We describe the unusual case of a 25-year-old male who has been successfully treated by implantable artificial sphincter for severe anal incontinence caused by imperforate anus and concomitant short-bowel syndrome. In early life, the patient underwent several surgical procedures, including a perineal colostomy for a high imperforate anus associated with a rectourethral fistula. At aged 21 years, he underwent a subtotal small-bowel resection and a restorative jejunotransverse anastomosis for acute intestinal necrosis related to intestinal malrotation with volvulus. The length of remnant jejunum was 90 cm. Consequently. the patient became permanently incontinent and required nutritional therapy. An artificial sphincter, after the creation of colonic reservoir, was implanted around the pulled-through colon. At two-year follow-up after implantation, despite short remnant bowel, the patient was fully continent without medication. Normal nutritional status was maintained under 100 percent oral nutrition. The patient's quality of life improved dramatically. He has returned to work and had no major restriction in his level of social functioning. Our case illustrates for the first time the original concept of artificial anal artificial sphincter implantation around a pulled-through colon, which constitutes a different situation from disease with rectum in place.
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keywords = nutritional status, status
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9/18. Presentation of dapsone-induced methemoglobinemia in a patient status post small bowel transplant.

    methemoglobinemia, an uncommon hemoglobinopathy, affects oxygen transport, causing tissue hypoxia. In the perioperative period, methemoglobinemia is often overlooked as a cause of low oxygen saturation, often mistaken for the more common causes of hypoxia, such as atelectasis, pulmonary edema, or pulmonary embolus, among other causes of respiratory failure. Most cases of methemoglobinemia in the perioperative period are precipitated by local anesthetics, especially benzocaine. dapsone, a sulfone antibiotic, is used for prophylaxis against pneumocystis carinii pneumonia in immunosuppressed patients. It is commonly used in organ transplant patients, especially those intolerant to sulfa. With the expansion of organ transplantation and improved long-term survival of these patients, presentation of status post organ transplant patients for surgery will inevitably increase. We report a case of dapsone-induced methemoglobinemia observed in a status post small bowel transplant patient in the postanesthesia care unit.
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10/18. Treatment of short gut syndrome with early living related small bowel transplantation.

    AIM: To investigate the results of treating short bowel syndrome with an early living related small bowel transplantation (SBT). methods: A 17-year-old boy with a 20-cm-long residual intestine due to necrotic volvulus received an early living related SBT from his mother. Donor-specific blood transfusion was performed for 8 weeks before transplantation, each time for 50 mL every week. cytomegalovirus status in both donor and recipient was negative. A 160-cm distal ileal segment was removed from the donor. The graft ilecolic artery and vein were anastomosed to the recipient's infrarenal aorta and caval vein. The proximal end of the graft was anastomosed end-to-end to the residual recipient jejunum; the distal anastomosis, between the distal end of the graft and transverse colon. An ileostomy was also performed. immunosuppression, infection prophylaxis, and antithrombotic and nutrition support were given postoperatively. RESULTS: The donor had an uneventful recovery. No technical complications were observed. The recipient was alive and well at 31 weeks after the operation. No graft rejection or infection was observed. He was off TPN 8 weeks after the operation and took low-fat food. The D-xylose test in the recipient was almost normal. CONCLUSIONS: Early living related small intestine transplantation is a good treatment for short bowel syndrome.
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