RESUMEN
BACKGROUND: Parenteral plant sterols (PSs) are considered hepatotoxic; however, liver PSs and their associations with liver injury in patients with intestinal failure (IF) have not been reported. MATERIALS AND METHODS: We analyzed liver and serum PS (avenasterol, campesterol, sitosterol, and stigmasterol) concentrations and ratios to cholesterol and their associations with biochemical and histologic liver damage in children with IF during (n = 7) parenteral nutrition (PN) and after weaning off it (n = 9), including vegetable oil-based lipid emulsions. RESULTS: Liver avenasterol, sitosterol, and total PS concentrations and cholesterol ratios were 2.4-fold to 5.6-fold higher in PN-dependent patients ( P < .05). Parenteral PS delivery reflected liver avenasterol and sitosterol ratios to cholesterol ( r = 0.83-0.89, P = .02-.04), while serum and liver total PS levels were positively interrelated ( r = 0.98, P < .01). Any liver histopathology was equally common while portal inflammation more frequent (57 vs 0%, P = .02) in PN-dependent patients. All liver PS fractions correlated positively with histologic portal inflammation ( r = 0.53-0.66, P < .05), and their total concentration was significantly ( P = .01) higher among patients with versus without portal inflammation. In PN-dependent patients, liver fibrosis and any histopathology correlated with liver campesterol and stigmasterol levels ( r = 0.79-0.87, P ≤ .03). CONCLUSION: Among children with IF, parenteral PSs accumulate in the liver, reflect their increased serum levels, and relate with biochemical liver injury, portal inflammation, and liver fibrosis, thus supporting their role in promoting liver damage.
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Inflamación/sangre , Enfermedades Intestinales/terapia , Hígado/efectos de los fármacos , Nutrición Parenteral/efectos adversos , Fitosteroles/efectos adversos , Fitosteroles/sangre , Alanina Transaminasa/sangre , Aspartato Aminotransferasas/sangre , Enfermedad Hepática Inducida por Sustancias y Drogas/sangre , Niño , Preescolar , Colesterol/análogos & derivados , Colesterol/sangre , Femenino , Humanos , Lactante , Enfermedades Intestinales/complicaciones , Hígado/fisiopatología , Masculino , Aceites de Plantas/administración & dosificación , Aceites de Plantas/efectos adversos , Vena Porta/patología , Sitoesteroles/sangre , Estigmasterol/sangre , Triglicéridos/sangre , gamma-Glutamiltransferasa/sangreRESUMEN
AIMS: To compare anorectal manometry (AM) in patients with different types of anorectal malformations (ARMs) in relation to functional outcomes. METHODS: A single-institution, cross-sectional study. After ethical approval, all patients ≥7years old treated for anterior anus (AA), perineal fistula (PF), vestibular fistula (VF), or rectourethral fistula (RUF) from 1983 onwards were invited to answer the Rintala bowel function score (BFS) questionnaire and to attend anorectal manometry (AM). Patients with mild ARMs (AA females and PF males) had been treated with minimally invasive perineal procedures. Females with VF/PF and males with RUF had undergone internal-sphincter saving sagittal repairs. RESULTS: 55 of 132 respondents (42%; median age 12 (7-29) years; 42% male) underwent AM. Patients with mild ARMs displayed good anorectal function after minimally invasive treatments. The median anal resting and squeeze pressures among patients with mild ARMs (60 cm H2O and 116 cm H2O respectively) were significantly higher than among patients with more severe ARMs (50 cm H2O, and 80cm H2O respectively; p≤0.002). The rectoanal inhibitory reflex was preserved in 100% of mild ARMs and 83% of patients with more severe malformations after IAS-saving sagittal repair. The functional outcome was poor in 4/5 patients with an absent RAIR (BFS≤11 or antegrade continence enema-dependence). Rectal sensation correlated significantly with the BFS. CONCLUSIONS: Our findings support the appropriateness of our minimally invasive approaches to the management of mild ARMs, and IAS-saving anatomical repairs for patients with more severe malformations. LEVEL OF EVIDENCE: III.
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Malformaciones Anorrectales/fisiopatología , Manometría , Procedimientos Quirúrgicos Mínimamente Invasivos , Fístula Rectal/cirugía , Enfermedades Uretrales/cirugía , Fístula Urinaria/cirugía , Adolescente , Adulto , Anciano , Canal Anal/anomalías , Canal Anal/cirugía , Malformaciones Anorrectales/cirugía , Niño , Estudios Transversales , Incontinencia Fecal/fisiopatología , Femenino , Humanos , Masculino , Recto/cirugía , Encuestas y Cuestionarios , Adulto JovenRESUMEN
OBJECTIVE The goal of this study was to determine the significance of spinal cord anomalies (SCAs) in patients with anorectal malformations (ARMs) by comparing the outcomes for bowel function, lower urinary tract symptoms (LUTS), and lower-limb neurological abnormalities to these outcomes in patients with similar ARMs and a normal spinal cord. METHODS The spinal cord MRI records of female patients treated for vestibular and perineal fistula (VF/PF) and male patients with rectourethral fistula (RUF) at a single center between 1983 and 2006 were reviewed. Bowel function and LUTS were assessed by questionnaire. Patients with extensive sacral anomalies or meningomyelocele were excluded. RESULTS Of 89 patients (median age 15 years, range 5-29 years), MRI was available in 90% (n = 80; 40 male patients with RUF), and 80% of patients returned the questionnaire (n = 64; 31 male patients with RUF). Spinal cord anomalies were found in 34%, comprising a filum terminale lipoma in 30%, low conus medullaris in 10%, and thoracolumbar syrinx in 6%. Bowel functional outcomes between patients with SCAs (n = 23) and those with a normal spinal cord (n = 41) were not significantly different for soiling (70% vs 63%), fecal accidents (43% vs 34%), and constipation (57% vs 39%; p = not significant for all). The LUTS, including urge (65% vs 54%), urge incontinence (39% vs 24%), stress incontinence (17% vs 22%), and straining (32% vs 29%) were also comparable between groups (p = not significant for all). No patients developed lower-limb neurological abnormalities. CONCLUSIONS The results suggest that the long-term functional outcomes for patients with SCAs who had VF/PF and RUF may not differ significantly from patients with the same type of ARMs and a normal spinal cord. The results favor a conservative approach to their management in the absence of abnormal neurological findings in the lower limbs.
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Anomalías Múltiples/fisiopatología , Malformaciones Anorrectales/fisiopatología , Médula Espinal/anomalías , Anomalías Múltiples/epidemiología , Anomalías Múltiples/terapia , Adolescente , Adulto , Malformaciones Anorrectales/complicaciones , Malformaciones Anorrectales/epidemiología , Malformaciones Anorrectales/terapia , Niño , Preescolar , Estudios Transversales , Manejo de la Enfermedad , Femenino , Estudios de Seguimiento , Humanos , Imagen por Resonancia Magnética , Masculino , Prevalencia , Fístula Rectal/complicaciones , Fístula Rectal/epidemiología , Fístula Rectal/fisiopatología , Fístula Rectal/terapia , Médula Espinal/diagnóstico por imagen , Resultado del Tratamiento , Adulto JovenRESUMEN
BACKGROUND/PURPOSE: Posterior sagittal anorectoplasty (PSARP) has become the standard surgical treatment for males rectourethral fistula (RUF) at most surgical centers worldwide. We aimed to define the long-term bowel functional outcomes following PSARP for RUF at our institution between 1983 and 2006, with comparison to age- and gender-matched controls. METHODS: Patients were invited to answer a detailed, previously validated Bowel Function Score (BFS) questionnaire by post. Respondents were matched by age and gender to three controls from the general population who had answered identical questionnaires. Case records were reviewed retrospectively for operative details. Ethical approval was obtained. Social continence was defined as soiling or fecal accidents <1/week and no requirement for changes of underwear or protective aids. RESULTS: Of 34 (79%) respondents (median age 19 (range, 4-29) years), 74% had voluntary bowel movements (VBMs), 24% were reliant on anterograde continence enema (ACE) washouts, and 1 patient had a colostomy. Impairment of bowel function was significantly higher in all aspects of fecal control among patients than controls (p<0.001). A statistically significant decline in fecal accidents and soiling was observed with age (p ≤ 0.03). Thirty-one percent of patients with VBMs had constipation managed with diet or laxatives (vs 2% of controls, p=0.0002). Of patients with VBMs followed up for > 12 years (n=20), 50% were completely continent (vs 73% of controls; p=NS). Overall, 76% of respondents were socially continent with or without artificial means in the form of ACE washouts. By BFS score, 39% had a good functional outcome, 27% had a moderate outcome, 9% had a clearly poor score and 24% were living with an ACE. CONCLUSIONS: Our results suggest that in the long-term, functional symptoms remain highly prevalent among patients treated for RUF with PSARP. However, the majority can be expected to achieve social continence, although for some this will require intervention with ACE bowel management. Approximately one third may report VBMs and complete continence.
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Defecación/fisiología , Predicción , Procedimientos de Cirugía Plástica/métodos , Fístula Rectal/cirugía , Recto/cirugía , Enfermedades Uretrales/cirugía , Fístula Urinaria/cirugía , Adolescente , Adulto , Niño , Preescolar , Colostomía/métodos , Estudios Transversales , Femenino , Estudios de Seguimiento , Humanos , Masculino , Fístula Rectal/fisiopatología , Recto/fisiopatología , Estudios Retrospectivos , Encuestas y Cuestionarios , Factores de Tiempo , Adulto JovenRESUMEN
PURPOSE: Rectopexy is a simple treatment of persisting complete rectal prolapse (RP) or related functional disorders in children. The results of rectopexy have been encouraging with few complications. We describe the postoperative complications and outcome of rectopexy in our institution from 2002 to 2013. METHODS: Ethical committee accepted the study. Hospital records of 27 successive patients (16 males), median age 7.2 (range 2.8-17) years, who underwent rectopexy (25 laparoscopic, 2 open) were reviewed. Indication for rectopexy included RP (n = 24), solitary rectal ulcer with enterocele (n = 2) and rectocele (n = 1). Nine patients (39 %) were healthy. In the remaining 14 patients, RP was secondary to anorectal malformation (n = 2), bladder exstrophy (n = 1), sacrococcygeal teratoma (n = 1) and myelomeningocele (n = 1) or associated with mental retardation (n = 8) and Asperger's syndrome (n = 1). Five (18 %) patients had constipation. Unexpected postoperative events and complications were rated by Clavien-Dindo classification (Grades I-V). RESULTS: Seventeen (61 %) patients had postoperative complications (Grade I n = 5, II n = 2 and III n = 7). Readmission was required in 11 (41 %) and reoperation, endoscopy or other surgical procedure in 9 (33 %) patients. Complications included severe faecal obstruction (n = 2), constipation (n = 3), faecal soiling (n = 1) urinary retention (n = 2), enuresis (n = 1), infection (n = 2), residual mucosal prolapse (n = 5), discomfort at defecation (n = 1) and recurrent RP (n = 2). Reoperations included sigmoid resection with re-rectopexy (n = 1), resection of mucosal prolapse (n = 1), suprapubic urinary catheter (n = 2), evacuation of faecal impaction (n = 2), colonoscopy (n = 3), appendicostomy for antegrade continence enema (n = 1). Mental retardation or behavioural disorder increased the risk of postoperative faecal obstruction and constipation RR = 84 (95 % CI 4.3-1600), p = 0.0035. After median follow-up of 4.1 (range 0.6-11) years RP or related condition was cured in 26 patients. Constipation and faecal soiling require management in a total of seven patients. CONCLUSIONS: Long-term results of rectopexy were good. Postoperative complications from mild to moderate grade were unexpectedly frequent. Preoperative neurobehavioural disorder and constipation increase the risk of postoperative problems and should be mentioned in patient counselling.
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Procedimientos Quirúrgicos del Sistema Digestivo/métodos , Procedimientos de Cirugía Plástica/métodos , Complicaciones Posoperatorias , Prolapso Rectal/cirugía , Recto/cirugía , Adolescente , Niño , Preescolar , Defecación , Femenino , Estudios de Seguimiento , Humanos , Masculino , Prolapso Rectal/fisiopatología , Resultado del TratamientoRESUMEN
AIM: The aim of this study was to characterize outcomes of children with severe intestinal motility disorders (IMD) requiring parenteral nutrition (PN). METHODS: Twenty consecutive children with primary IMD requiring long-term PN between 1984 and 2010 were included. Median (interquartile range) follow-up was 13.1 (5.2-20.1) years. Treatment, PN dependence, growth, nutritional status, liver function, and survival were assessed. RESULTS: Underlying etiology included chronic intestinal pseudo obstruction (CIPO; n=8) and Hirschsprung disease with extensive aganglionosis (n=12). CIPO and aganglionosis patients had 100 (86-100%) and 29 (19-40%) of age-adjusted small bowel length remaining, respectively. In order to facilitate enteral tolerance and avoid PN-associated liver disease, short aganglionic segment (40 cm) was left in situ in four of five cases, with aganglionosis extending to duodenojejunal flexure combined with Ziegler myectomy-myotomy in two. Six of seven children with aganglionosis extending into mid small intestine underwent staged jejunoanal pull-through. Feeding/venting gastrostomies (n=13) or jejunostomies were commonly employed. Probability of PN dependence owing to IMD was markedly increased in relation to short bowel syndrome (70 versus 19% at 5 years, P<0.0001). Two (10%) patients developed end-stage liver disease. A total of 11 (55%) patients (5 CIPO and 6 aganglionosis) weaned off PN after 8.2 years (1.8-17 years), including two patients after intestinal transplantation (ITx). Two children died before ITx-era giving overall survival of 90%. Survivors had well-preserved liver function, growth, and nutritional status. CONCLUSIONS: Despite high PN dependence, long-term survival is achievable in the majority of children with IMD requiring PN. A wide repertory of surgical options including ITx is required for optimal outcomes.
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Motilidad Gastrointestinal , Enfermedades Intestinales/cirugía , Nutrición Parenteral , Adolescente , Niño , Preescolar , Femenino , Humanos , Enfermedades Intestinales/terapia , Masculino , Índice de Severidad de la Enfermedad , Resultado del Tratamiento , Adulto JovenRESUMEN
OBJECTIVES: We prospectively evaluated incidence of prolonged (>28 days) parenteral nutrition (PN), associated complications, and significance of parenteral plant sterols (PS) in neonatal intestinal failure-associated liver disease (IFALD) compared with children. METHODS: We recruited 28 neonates (mean age 50 days, range 28-126) and 11 children (6.9 y, 2.1-16.6) in all of Finland. Patients underwent repeated measurements of serum cholesterol, noncholesterol sterols, including PS, cholestanol and cholesterol precursors, and liver biochemistry during and 1 month after discontinuation of PN. Healthy matched neonates (n=10) and children (n=22) served as controls. RESULTS: IFALD occurred more frequently among neonates (63%) than children (27%; P<0.05). Ratios of serum PS, including stigmasterol, sitosterol, avenasterol, and campesterol, and total PS were increased among neonates compared with healthy controls and children on PN by 2- to 22- and 2- to 5-fold (P<0.005), respectively. Neonates with IFALD had significantly higher ratios of serum PS and cholestanol compared with neonates without IFALD (P<0.05). Total duration of PN associated with serum cholestanol, stigmasterol, avenasterol, alanine aminotransferase, and aspartate aminotransferase (r=0.472-0.636, P<0.05). Cholestanol and individual serum PS, excluding campesterol, reflected direct bilirubin (r=0.529-0.688, P<0.05). IFALD persisted after discontinuation of PN in 25% of neonates with 4.2- and 2.2-times higher ratios of serum stigmasterol and cholestanol compared with neonates without IFALD (P<0.05). CONCLUSIONS: Frequent occurrence of IFALD among neonates on PN displays an association to duration of PN and markedly increased serum PS, especially stigmasterol, in comparison to healthy neonates and children on PN. Striking accumulation of parenteral PS may contribute to IFALD among neonates.
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Factores de Edad , Colestanol/sangre , Enfermedades Intestinales/complicaciones , Hepatopatías/etiología , Nutrición Parenteral/efectos adversos , Fitosteroles/sangre , Aceites de Plantas/efectos adversos , Adolescente , Alanina Transaminasa/sangre , Aspartato Aminotransferasas/sangre , Bilirrubina/sangre , Niño , Preescolar , Colesterol/análogos & derivados , Colesterol/sangre , Grasas de la Dieta/efectos adversos , Grasas de la Dieta/sangre , Emulsiones Grasas Intravenosas/efectos adversos , Emulsiones Grasas Intravenosas/química , Femenino , Finlandia/epidemiología , Humanos , Lactante , Recién Nacido , Enfermedades Intestinales/sangre , Enfermedades Intestinales/terapia , Hepatopatías/sangre , Hepatopatías/epidemiología , Masculino , Aceite de Oliva , Nutrición Parenteral/métodos , Aceites de Plantas/química , Prevalencia , Estudios Prospectivos , Aceite de Soja/efectos adversos , Aceite de Soja/química , Estigmasterol/sangreRESUMEN
BACKGROUND AND OBJECTIVE: Plant sterols (PS) in parenteral nutrition (PN) may contribute to intestinal failure-associated liver disease. We investigated interrelations between serum PS, liver function and histology, cholesterol metabolism, and characteristics of PN. PATIENTS AND METHODS: Eleven patients with intestinal failure (mean age 6.3 years) receiving long-term PN were studied prospectively (mean 254 days) and underwent repeated measurements of serum lipids, noncholesterol sterols, including PS, and liver enzymes. PS contents of PN were analyzed. Liver biopsy was obtained in 8 patients. Twenty healthy children (mean age 5.7 years) served as controls. RESULTS: Median percentage of parenteral energy of total daily energy (PN%) was 48%, including 0.9 g · kg(-1) · day(-1) of lipids. Respective amounts of PN sitosterol, campesterol, avenasterol, and stigmasterol were 683, 71, 57, and 45 µg · kg(-1) · day(-1). Median serum concentrations of sitosterol (48 vs 7.5 µmol/L, P < 0.001), avenasterol (2.9 vs 1.9, P < 0.01), stigmasterol (1.9 vs 1.2, P < 0.005), but not that of campesterol (9.8 vs 12, P = 0.22), were increased among patients in relation to controls, and correlated with PN% (r = 0.81-0.88, P < 0.005), but not with PN fat. Serum cholesterol precursors were higher in patients than in controls. Serum liver enzymes remained close to normal range. Glutamyl transferase correlated with serum PS (r = 0.61-0.62, P < 0.05). Liver fibrosis in 5 patients reflected increased serum PS (r = 0.55-0.60, P = 0.16-0.12). CONCLUSIONS: Serum PS moderately increase during olive oil-based PN, and correlate positively with PN% and glutamyl transferase. Despite well-preserved liver function, histology often revealed significant liver damage.
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Colesterol/análogos & derivados , Intestinos/patología , Lípidos/sangre , Hígado/patología , Nutrición Parenteral/efectos adversos , Fitosteroles/sangre , Sitoesteroles/sangre , Adolescente , Biomarcadores/sangre , Estudios de Casos y Controles , Niño , Preescolar , Colestasis/patología , Colesterol/sangre , Femenino , Estudios de Seguimiento , Humanos , Lactante , Metabolismo de los Lípidos/efectos de los fármacos , Hígado/enzimología , Fallo Hepático/metabolismo , Masculino , Aceite de Oliva , Aceites de Plantas/administración & dosificación , Estudios ProspectivosRESUMEN
AIMS: To establish the accuracy of anorectal manometry (ARM) with reference to operative rectal biopsy in the investigation of children under 1 year of age with defaecation difficulties. MATERIALS AND METHODS: The records of all infants who underwent ARM and operative rectal biopsy at our centre between 1994 and 2007 were reviewed. ARM was performed by a consultant paediatric surgeon in each and under ketamine anaesthesia, as operative rectal biopsy was taken. RESULTS: There were 81 patients (49 males, 32 females). The median age at investigation was 2 months (range 0.1-11 months). Thirty-three patients (41%) had Hirschprung's disease. No patient with a normal recto-anal inhibitory reflex (RAIR) had Hirschprung's disease (100% negative predictive value). No patient with Hirschprung's disease exhibited a reflex (100% sensitivity). The specificity and positive predictive value of ARM for Hirschprung's disease were 83% and 80%, respectively. Bowel habit normalised in 88% of patients with a reflex within 1 year. CONCLUSIONS: In children under 1 year of age, Hirschprung's disease is very unlikely in the presence of a RAIR. The specificity and positive predictive value of ARM for the diagnosis of Hirschprung's disease are inferior to those of rectal suction biopsy, and therefore, it cannot be recommended for use as a sole diagnostic tool for this disease. ARM may be a useful investigation in patients where the histologic specimen is inadequate and/or functional constipation is the most likely diagnosis. If RAIR is present, rectal biopsy may not be required.
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Canal Anal/fisiopatología , Enfermedades del Recto/diagnóstico , Enfermedades del Recto/patología , Recto/fisiopatología , Recto/cirugía , Canal Anal/patología , Biopsia , Enema , Femenino , Humanos , Lactante , Recién Nacido , Masculino , Manometría , Enfermedades del Recto/fisiopatología , Recto/patología , Reflejo , Resultado del TratamientoRESUMEN
BACKGROUND: The aetiology of fecal incontinence in children has traditionally been attributed to idiopathic constipation, structural defects or neuropsychiatric disorders. We describe a new subgroup of otherwise healthy children who have fecal soiling without any underlying cause for the incontinence. METHODS: The hospital records of children with fecal incontinence were screened to detect patients without any history, signs or symptoms of constipation or an organic, neurological or psychiatric cause for the incontinence. Anorectal manometry findings were compared with those of age-matched children with idiopathic constipation and soiling. RESULTS: Eight boys and 5 girls were identified. The median age at diagnosis was 7.9 years. Soiling had lasted median of 4.1 years, occurred at least every other day in 9, at least once a week in 2 and occasionally in 2 and required change of underwear or use of protective pads. Abdominal x-ray and barium enema showed normal findings. Sacral x-ray and/or MRI of the spinal cord showed normal bony spine and spinal cord. Five children had coexisting night and/or daytime wetting. Impaired rectal sensation was the only identifiable abnormality that was detected. The median volume required for the first sensation was 45 mL (range, 15-100 mL; normal, <15 mL). Anorectal manometry alone was unable to differentiate patients with functional fecal soiling from those with idiopathic constipation associated soiling. The median follow-up time after the diagnosis was 9.1 months. Treatment of fecal soiling consisted of education, dietary modification or stimulatory laxatives to establish regular toileting routines. Treatment improved fecal continence in 6 out of 8 cases with follow-up longer than 6 months. CONCLUSIONS: There is a small subgroup of children with fecal soiling who are otherwise healthy without constipation or any other underlying cause for the incontinence. These children seem to have isolated impairment of rectal sensation. In most, the prognosis is good with conservative treatment.
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Incontinencia Fecal/etiología , Incontinencia Fecal/terapia , Recto/inervación , Sensación/fisiología , Control de Esfínteres , Adolescente , Catárticos/uso terapéutico , Niño , Preescolar , Estreñimiento/complicaciones , Estreñimiento/epidemiología , Femenino , Humanos , Masculino , Manometría , Presión , Factores de Tiempo , Resultado del TratamientoRESUMEN
BACKGROUND/PURPOSE: Small bowel transplantation impairs enteric function and causes malabsorption of cholesterol and bile acids. Growth hormone stimulates intestinal absorptive function. The authors hypothesized that long-term growth hormone therapy could improve absorption of bile acids and cholesterol after autotransplantation of the jejunoileum. METHODS: Sixteen pigs with similar food, cholesterol, and fat intake underwent either sham laparotomy or a model of jejunoileal autotransplantation, including extrinsic autonomic denervation, lymphatic interruption, and in situ cold ischemia. Five randomly chosen autotransplanted animals received daily growth hormone treatment for 8 weeks. Serum lipids, absorption, and excretion of cholesterol, bile acids, and fat were determined after 8 weeks. Mucosal morphometrics, proliferation, and enzyme activities were determined. Plasma cholesterol precursors and plant sterols, respective markers of cholesterol synthesis and absorption, were measured after 2 and 8 weeks. RESULTS: After jejunoileal autotransplantation, growth hormone treatment significantly increased body weight gain, cholesterol absorption efficiency from 45.1% to 62.1%, plasma campesterol to cholesterol proportions, and biliary secretion of cholesterol. With or without growth hormone treatment, autotransplantation significantly increased fecal bile acid excretion, plasma cholesterol precursors, fecal bacterially modified neutral sterols, mucosal thickness of the ileum (but not jejunum), and intestinal transit time when compared with sham-operated animals. Crypt cell proliferation, mucosal enzyme activities, and microvilli showed no differences between the groups. CONCLUSIONS: These findings suggest that growth hormone treatment selectively improves cholesterol, but not bile acid absorption, after autotransplantation of the jejunoileum.