Cases of the month: three children with abdominal complaints.
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Abdominal Pain↗
Biomedical subjects
H Lindahl
Publications and source records attributed to H Lindahl.
Segmental colonic motility in patients with anorectal malformations.
BACKGROUND: Constipation is one of the most important functional sequelae in patients with anorectal malformations. The cause of this motility disorder is unknown. AIM: The purpose of this study was to assess total colonic transit time (TCT) and segmental colonic transit time (SCT) in patients with anorectal malformations. METHOD: Ninety patients with anorectal malformations (40 low and 50 high; median age, 7 years; range, 3 to 13) and twenty-five healthy children (median age, 8 years; range, 3 to 14 years) underwent measurement of TCT and SCT by the saturation technique. Ten radiopaque markers were ingested daily for 6 days followed by administration of a single abdominal x-ray on day 7. TCT in days was calculated by dividing the number of retained markers in the whole colon by the daily intake. SCT in four colonic segments (right, transverse, left, rectosigmoid) was described as a percentage of TCT (markers in one segment versus total number of retained markers). In high anomalies the degree of rectosigmoid dilatation was assessed by contrast enemas taken before closure of the stoma and later during follow-up. RESULTS: TCT was significantly (P < .03) prolonged in patients with anorectal anomalies (median high, 2.1 days; low, 1.9 days versus 1.3 in healthy subjects). In patients with high anomalies right SCT was prolonged when compared with low anomalies and healthy subjects (median high, 24% versus low, 10% and normal subjects, 10%; P < .01). The impairment was more severe in patients with very high anomalies (P < .005). Patients with a low anomaly had prolonged rectosigmoid SCT (median low, 65% versus high, 43% and normal subjects, 49%; P < .05). Prolonged right colonic SCT and TCT correlated with symptomatic constipation in patients with high anomalies (P < .05) but not with those who had low anomalies. Impaired overall functional outcome correlated with prolonged right colonic SCT in patients with high anomalies and with prolonged rectosigmoid SCT in patients with low anomalies. There was no correlation between the degree of rectosigmoid dilatation and SCT or TCT. CONCLUSION: Patients with anorectal malformations have abnormal colonic motility. The type of motility disorder in low anomalies is rectosigmoid hypomotility. In patients with high anomalies the motility disturbance is more generalized. The overall functional outcome was strongly related to the degree of these motility disorders.
Adolescent↗
Long-term pulmonary sequelae in survivors of congenital diaphragmatic defects.
Between 1948 and 1980, 107 of 164 patients survived after repair of a congenital diaphragmatic defect. Sixty of the survivors (mean age, 29.6 years; SD, 9.0 years) underwent clinical examination, chest radiography, spirometry, and diffusing capacity measurement 11 to 41 years after the diaphragmatic repair. Twenty-seven of the 60 had body plethysmography, xenon 133 radiospirometry, and a test of bronchial hyperreactivity. Subjective physical performance was below average for eight patients (13%), seven patients (12%) had asthma, and four patients (7%) reported increased susceptibility to respiratory infections. Thirty-one patients (52%) had ventilatory impairment, which was obstructive in nine (15%), restrictive in seven (12%), and obstructive and restrictive in 15 patients (25%). Nine (35%) of the 26 patients tested had bronchial hyperreactivity. The presence of ventilatory impairment and bronchial hyperreactivity correlated with the initial clinical severity of the affliction. Chest asymmetry (29 patients; 48%) and scoliosis (16 patients; 27%) were more common among patients with ventilatory impairment than among those with normal spirometric findings. Ventilatory impairment and thoracic deformities are common in adults with repaired diaphragmatic defects. Surveillance should begin in infancy and continue into adulthood.
Adolescent↗
Chest wall and spinal deformities in adults with congenital diaphragmatic defects.
Between 1948 and 1980, 107 of 164 patients survived after repair of congenital diaphragmatic defects. To study chest wall and spinal deformities among these patients, 60 survivors (mean age, 29.6 +/- 9.0 years) underwent clinical examination, chest and spinal radiography, spirometry, and diffusing capacity measurements. Twenty-seven of these patients also had body pletysmography, xenon 133 radiospirometry, and a test for bronchial hyperreactivity. Chest asymmetry was present in 29 patients (48%). Eleven patients (18%) had pectus excavatum, and one had a pectus carinatum deformity. Anterior asymmetry, pectus deformities, and a flat chest were more common among the patients who initially had a large diaphragmatic defect. Two patients underwent anterior thoracoplasty because of their deformities. Sixteen patients (27%) had significant scoliosis (Cobb angle > or = 10 degrees). Scoliosis was more common in patients with ventilatory impairment than in those with normal lung function. One patient required spinal stabilization, one with severe kyphoscoliosis remains under observation, and a patient with moderate scoliosis was treated with a spinal brace. The results of this study indicate that chest wall deformities and scoliosis are common among adults with repaired congenital diaphragmatic defects. In most patients the deformity is mild, but some will require surgery. Surveillance until adulthood is appropriate.
Adolescent↗
Restorative proctocolectomy for ulcerative colitis in children--is the J-pouch better than straight pull-through?
Proctocolectomy and ileoanal anastomosis (IAA), with or without an ileal pouch, is the preferred method for the surgical treatment of ulcerative colitis (UC) in children. The authors compared the surgical complications and functional outcome for patients with UC who had proctocolectomy and straight IAA with those of patients who had proctocolectomy and IAA with a J-pouch. Twenty-two children (aged 7 to 15 years) with intractable UC had an IAA, 11 by straight ileoanal anastomosis (SIAA) and 11 with a J-pouch (JIAA). The median clinical and endoscopic follow-up period for SIAA patients was 4 years (range, 3.0 to 6.5 years); for JIAA patients it was 2 years (range, 1 to 3 years). The frequency of daytime and nighttime defecation was significantly higher (P < .03) for patients with SIAA at 3, 6, and 12 months postoperatively. At the last follow-up visit, the mean frequency of defecation for patients with SIAA was 6.1 +/- 2.5/d; that for patients with JIAA was 3.3 +/- 0.5/d (P < .03). Six of 10 patients with SIAA but none with JIAA had nighttime bowel evacuations. None of the patients had daytime soiling; 3 with SIAA and 2 with JIAA had slight soiling during sleep. There was no significant difference in the incidence of early (SIAA 27% v JIAA 45%) and late (SIAA 54% v JIAA 27%) surgical complications between the two groups. Endoscopically confirmed symptomatic pouchitis during the first postoperative year was found in 4 of the 11 (36%) patients with SIAA and in 2 of the 11 (18%) with JIAA (difference not significant). The early and long-term functional results of JIAA are superior to those of SIAA. There is no difference in the incidence of surgical complications and pouchitis between JIAA and SIAA patients.
Adolescent↗
Long-term gastrointestinal morbidity in patients with congenital diaphragmatic defects.
Recent reports indicate a significant incidence of gastroesophageal reflux (GER) and other nonpulmonary problems after the repair of congenital diaphragmatic defects. Reports of follow-up through adulthood are few and based on a small number of patients. From 1948 to 1982, 107 of 164 patients (65%) treated at the authors' institution survived after repair of congenital diaphragmatic hernia or eventration. Sixty of the 107 survivors (56%) (mean age, 29.6 years; SD, 9.0 years) were interviewed and examined clinically. Forty-one of the sixty (68%) underwent upper gastrointestinal endoscopy. Early postoperative GER was recorded for 11 of the 60 patients (18%). Two of them underwent fundoplication because of an esophageal stricture. At the time of the follow-up study, 38 of the 60 (63%) reported symptoms suggestive of GER. Endoscopic or histological GER (esophagitis, Barrett's esophagus) was present in 22 of 41 patients (54%). No significant correlation between the initial severity of the diaphragmatic defect or neonatal postoperative problems and the late GER could be verified. Intestinal obstruction requiring hospital admission occurred in 12 of the 60 patients (20%), eight of whom had surgical treatment between 1 month and 20 years after repair of the diaphragmatic defect. GER and intestinal obstruction are common among patients who have undergone repair of a congenital diaphragmatic defect. Investigations for GER should be performed routinely during the follow-up of these patients.
Adolescent↗
H-type anorectal malformations: incidence and clinical characteristics.
To establish the incidence of H-type anorectal malformations (ARM), the hospital records of 629 patients with ARM were studied. Patients who had a congenital rectourogenital connection and an external anal opening in a normal or ectopic position were considered to have an H-type malformation. Twenty (3.2%; 14 females, 6 males) of the 629 patients studied had an H-type ARM. Major associated anomalies were found in 60% of these patients. Thirteen patients (12 females, 1 male) had a normally placed anus; the H fistula was low rectovestibular in 10, rectovaginal in 2, and rectourethral in 1. Seven patients had an ectopic anal opening. Three males had a rectourethral H fistula; in two of these there was a double fistula. Two males had a rectovesical H fistula. Of the two females with ectopic anal openings, one had a high rectovaginal H fistula and the other had a low fistula. In only one case was the diagnosis of H fistula made immediately after birth. Three patients died of severe cardiac abnormalities during the neonatal period or early infancy. Primary perineal repair or a limited posterior surgical anorectoplasty (PSARP) was used in 15 cases; formal PSARP was used for the other two. Four patients had between one and 4 recurrences; all but one of these fistulas were repaired subsequently. Long term (median follow-up period, 12 years; range, 4 to 38 years), 12 patients had good bowel function and no faecal soiling; four of these had constipation that was manageable with laxatives. Two patients (aged 10 and 12 years) had daily soiling. Two others were too young to evaluate, and one (with severe mental retardation) has a permanent colostomy.
Adolescent↗
[Diagnosis of an abdominal mass after a bicycling accident].
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Bezoars↗
Long-term complications in cases of isolated esophageal atresia treated with esophageal anastomosis.
Long-term follow-up examination with esophagogastroduodenoscopy was performed on seven patients who had undergone successful delayed anastomosis for isolated esophageal atresia. The follow-up period ranged from 1.2 to 11.3 years (mean, 5.3). All patients had undergone fundoplication because of symptomatic gastroesophageal reflux (GER). Three anastomotic strictures had to be resected. At the time of the last follow-up examination, the subjective results were excellent for five patients and good for two. The last endoscopy showed macroscopic esophagitis in three and normal mucosa in four. The fundoplication was partly disrupted in two patients. In three patients the fundoplication was competent but partly intrathoracic. Histological examination showed moderate esophagitis in one, mild esophagitis in one, and normal mucosa in five patients; however, four patients were on continuous medication for esophagitis. In conclusion, the subjective results of patients with isolated esophageal atresia treated with esophageal anastomosis are good. However, long-term complications caused by GER are common in these patients. Therefore, active search and treatment of reflux is necessary for these patients.
Anastomosis, Surgical↗
Is normal bowel function possible after repair of intermediate and high anorectal malformations?
The bowel function of 46 patients who had undergone internal sphincter-saving posterior sagittal anorectoplasty (PSARP) was evaluated by a questionnaire 3 to 10 years after the closure of the colostomy. The bowel function was assessed by a multivariate scoring method. All patients had also been followed up clinically and manometrically. Seventy healthy children with a similar age and sex distribution were used as controls. Sixteen (35%) patients had bowel function scores that were within the range of the scores of healthy children (mean score of healthy children +/- SD). Another 16 (35%) patients with intermediate scores had a clinically good continence. Fourteen (30%) patients with low scores had a clinically fair or poor functional result; 3 of them required enemas because of severe constipation and 11 used protective aids or had to change underwear frequently because of daily soiling. There was a correlation between a good continence outcome and the presence of a functional internal sphincter and high anorectal resting pressure. Absence of a functional internal sphincter, severe sacral anomalies, and constipation were associated with poor functional results. It is concluded that normal bowel function after internal sphincter-saving PSARP may be expected in a significant proportion of children with high or intermediate anorectal anomalies.
Anal Canal↗
Total parenteral nutrition-associated cholestasis in surgical neonates may be reversed by intravenous cholecystokinin: a preliminary report.
Neonatal infants who require total parenteral nutrition (TPN) after major operations are susceptible to total parenteral nutrition-associated cholestasis (TPNAC). A therapeutic dilemma ensues if cholestasis does not resolve after the institution of full enteral nutrition. The authors report the reversal of TPN-associated cholestasis by intravenous cholecystokinin in eight infants who had undergone major surgery during the neonatal period. The indications for surgery were necrotizing enterocolitis in three patients, midgut volvulus in one, gastroschisis in one, diaphragmatic hernia in one, necrosis of the stomach in one, and cardiac anomaly in one. Four of the infants were premature. Median duration of TPN was 25 days (range, 20 to 150 days). Seven patients were weaned from TPN before treatment with cholecystokinin was instituted Mean duration of pretreatment full enteral nutrition in these seven patients was 35 days (range, 20 to 55 days). One girl with short gut syndrome tolerated only 10% of her caloric needs via the enteral route. All patients had alcoholic stools, conjugated hyperbilirubinemia, no excretion of Technetium-labeled HIDA to the biliary tree or duodenum (six patients), and significantly elevated liver enzyme values. In three patients, echography showed biliary sludge or stones in the gall bladder. Porcine cholecystokinin (2 IDU/kg) was administered intravenously for 3 to 5 days. If the stool color did not normalize, cholecystokinin injections were repeated using a larger dose (4 IDU/kg). In seven patients, including the girl with short gut syndrome, the clinical jaundice and conjugated hyperbilirubinemia completely resolved within 1 to six weeks. No biliary sludge or stones were seen in the posttreatment echography in any of the patients.(ABSTRACT TRUNCATED AT 250 WORDS)
Cholecystokinin↗
Prenatal muscle relaxation in congenital diaphragmatic hernia.
An infant with congenital diaphragmatic hernia was given 2 mg vecuronium bromide intramuscularly in utero 40 min before vaginal delivery at 40 weeks gestation. At birth the infant had complete muscle relaxation, which facilitated decompression of the bowel and surgical correction. Prenatal muscle relaxation may improve the care of infants with congenital diaphragmatic hernia.
Adult↗
Oesophageal atresia--need for life-long follow-up?
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Adolescent↗
Barrett's oesophagus and perforation of gastric tube ulceration into the pericardium: a late complication after reconstruction of oesophageal atresia.
15 years after replacement of atretic segment with a gastric tube, perforation of an intrathoracic gastric tube ulcer into the pericardium, oesophago-pericardial fistula and severe mediastinitis developed in a 17-year-old male after reconstruction of oesophageal atresia using a retrosternal Heimlich-tube with a cervical oesophagogastric anastomosis. For six years he suffered from reflux oesophagitis and had Barrett's metaplasia in the cervical oesophagus. The patient had previously had one unsuccessful attempt to correct the oesophageal atresia at the age of two years using transverse colon. The two stage treatment included subtotal oesophagectomy, pericardiotomy, cervical oesophagostomy, gastrostomy, mediastinal and pericardial irrigation with antibiotics. After healing of the mediastinitis, the continuity of the alimentary tract was restored by using an isoperistaltic subcutaneous ileocolic segment. After a follow-up of five years the patient is well, without any oesophageal symptoms.
Adolescent↗
Does disordered upper gastrointestinal motility predispose to Helicobacter pylori colonization of the stomach in children?
Helicobacter pylori is an important factor in the pathogenesis of chronic gastritis and gastroduodenal ulcer disease. However, the basic causal mechanisms of H pylori colonization on the gastric mucosa are still unclear. The authors evaluated the prevalence of H pylori colonization in 266 children who underwent upper gastrointestinal endoscopy during a 12-month period. The indications for endoscopy were follow-up of esophagitis related to gastroesophageal reflux (n = 17), suspicion of gastroesophageal reflux (n = 51), abdominal pain (n = 28), vomiting (n = 30), follow-up of esophageal atresia (n = 46) and duodenal atresia (n = 28), inflammatory bowel disease (n = 28), and miscellaneous (n = 38). The methods used to detect H pylori colonization were histology and the rapid urease test. H pylori colonization was demonstrated in 31 (11.6%) of the 266 patients. In two patient groups, a high prevalence of colonization was identified. In patients with an operated duodenal atresia, 36% (10 of 28) had H pylori on the gastric mucosa. The organism was demonstrated on the gastric mucosa in 47% (8 of 17) of the patients with gastroesophageal reflux-related esophagitis; five of the eight patients had neurological impairment. In the other patient groups, the prevalence of H pylori infection ranged from 2% to 14%. The present study suggests that, in children, the disturbed esophagogastroduodenal motility, which is commonly associated with gastroesophageal reflux and duodenal atresia, predisposes to H pylori infection.
Adolescent↗
Fecal continence and quality of life for adult patients with an operated high or intermediate anorectal malformation.
Fecal continence and quality of life were evaluated by a questionnaire completed by 33 patients (mean age, 35 years; 25 males, 8 females) who had undergone anorectal reconstruction for a high or intermediate anorectal anomaly between 1946 and 1962. The malformation was classified as high in 30 cases and as intermediate in three. The primary reconstruction was abdominoperineal in 23 patients and direct perineal in 10. Ten patients had undergone secondary sphincter reconstructions. Fecal-continence was assessed by the scoring system described by Holschneider. Thirty-five healthy people with a similar age and sex distribution were used as controls. Only 6 patients (18%), including all three with an intermediate anomaly, had good fecal continence. None had completely normal bowel habits. Eighteen patients (54%) had a fair continence outcome. Seven patients (21%) had complete fecal incontinence. A permanent colostomy had been performed in two patients. Urinary incontinence was reported by 11 patients (33%). Ten patients (30%) had difficulties with sexual functions. Social problems related to defective continence were found in 28 patients (85%). All controls had good fecal continence; 80% had completely normal bowel habits. The authors conclude that most adults with high anorectal malformations who have undergone abdominoperineal or direct perineal repair suffer from severely defective fecal continence and have poor quality of life.
Adult↗
Surgical treatment of intractable cholestasis associated with total parenteral nutrition in premature infants.
We report surgical reversal of intractable total parenteral nutrition (TPN)-associated cholestasis refractory to conservative treatment in 9 premature infants. Indications for TPN were poorly tolerated enteral feedings in all patients. Five patients had undergone gastrointestinal operations; in addition, 7 of the 9 patients had had bacterial sepsis. The median duration of TPN was 28 days (range, 20 to 50 days). The median duration of preoperative full enteral nutrition after weaning from TPN was 34 days (range, 16 to 95 days). All patients had progressive conjugated hyperbilirubinemia, no excretion of Tc-labeled HIDA to the biliary tree and duodenum, and markedly elevated liver enzyme values. Intraoperative cholangiography showed normal biliary anatomy in all cases; in addition, 2 patients had gallbladder stones. Bile was hyperviscous in all patients and contained biliary sludge in 4. The biliary tree was irrigated and the liver biopsied in all patients. The gallbladder was removed from 2 patients who had stones in the gallbladder. Liver histology was consistent with TPN-associated cholestasis in all cases, and in addition, 4 cases showed significant destruction of intrahepatic bile ducts. One patient died 2 weeks postoperatively from intracerebral hemorrhage. Jaundice completely resolved in other patients within 2 weeks. HIDA-biligraphy performed 1 to 2 months postoperatively showed normal excretion of the radioactive marker to the biliary tree and duodenum in all cases. The functional abnormality in bile excretion and bile duct motility in TPN-associated cholestasis may be reversed by irrigation of the biliary tree. Surgical intervention should be considered when cholestasis is progressive and refractory to medical management.
Cholestasis↗
Anorectal function in adults operated for a benign sacrococcygeal teratoma.
Fecal continence and quality of life were evaluated by a questionnaire in 26 adult patients (mean age, 30 years; 6 men, 20 women) who had undergone surgery for a benign sacrococcygeal teratoma in infancy. The fecal continence was assessed by a score described by Holschneider. Twenty-six healthy people with a similar age and sex distribution were used as controls. Good fecal continence was reported by 88% of the patients; however, only 27% had completely normal bowel habits. Some fecal soiling was present in 27% of the patients. No correlation between the severity of aberrations in anorectal functions and the degree of intrapelvic extension of the tumor was found. Social problems related to deficient anorectal function were reported by 27% of the patients. Other health problems including urinary incontinence were reported by 50% of the patients. All the controls had good fecal continence; 77% of them had completely normal bowel habits. The present study shows that at the adult age, a significant proportion of patients who have undergone surgery for sacrococcygeal teratoma suffer from deficient anorectal function and a diminished quality of life.
Adult↗