Utility of thiol/disulphide homeostasis as a biomarker for acute appendicitis: a systematic review and meta-analysis.

The aim of this study was to analyze the role of thiol/disulfide homeostasis (TDH) parameters as an indicator of oxidative stress in acute appendicitis (AA). PubMed, EMBASE, Web of Science, and Scopus databases were systematically searched. Studies reporting on TDH in AA (both complicated and uncomplicated cases) were included. The comparator group were healthy controls. The TDH domain was compared between the groups using anti-oxidant parameters, namely native thiol and total thiol levels, and native thiol/total thiol ratio; and oxidant parameters, namely disulfide level, disulfide/native thiol ratio, and disulfide/total thiol ratio. The statistical analysis was performed using a random-effects model. The methodological quality of the studies was assessed utilizing the Newcastle-Ottawa scale. Eleven studies with a total of 926 subjects, comprising 457 patients with uncomplicated appendicitis, 147 with complicated appendicitis, and 322 healthy controls were included. Our study demonstrated significantly increased oxidative stress in AA as compared to healthy controls in all TDH parameters and significantly lower total thiol levels in complicated AA as compared to uncomplicated AA. Due to a poor methodological quality in five out of eleven studies, future prospective studies with adequate power are essential to validate these observations and refine the diagnostic approaches to AA.

Complication rate after gastrostomy placement in children can be reduced by simple surgical steps.

AIM: To evaluate if the incidence of postoperative complications after gastrostomy placement is correlated to perioperative parameters or patient characteristics. METHODS: In this prospective observational study, children <18 years of age planned to receive a gastrostomy at partaking clinics between 2014 and 2019 were invited. Pre-, peri- and postoperative variables were collected and followed up 3 months postoperatively. RESULTS: Five hundred and eighty-two patients were included (median age: 26 months, median weight: 10.8 kg), mainly laparoscopic (52.0%) and push-PEG (30.2%) technique used. The incidence of complications was lower in the group of patients receiving a gastrostomy tube that was 2 mm longer than the gastrostomy canal (p < 0.001-0.025), and a thickness of 12 Fr (p < 0.001-0.009). These findings were confirmed by multivariate analysis also including operative technique, age and weight. Patients with oncological disease had significantly higher incidence of pain and infection but the lowest incidence of granulomas (p < 0.001-0.01). CONCLUSION: This study indicates that a 12 Fr gastrostomy tube that is 2 mm longer than the gastrostomy canal is correlated with the lowest incidence of postoperative complications the first 3 months after surgery. Oncological patients had the lowest incidence of granulomas which probably is related to chemotherapy.

Persistent fecal incontinence into adulthood after repair of anorectal malformations.

PURPOSE: Persistent fecal incontinence beyond childhood is common in ARM patients. The aim of this study was to analyze a consecutive series of adult patients with persistent incontinence, establish the causes, and evaluate whether further treatment could be offered. METHODS: Forty-four adult ARM patients with reported incontinence were invited. Eighteen patients (11 males, median age 40.5 years, range 18-50 years) accepted and underwent clinical examination, rectoscopy, and 3D-ultrasound. Five had previously been treated with secondary surgery to improve continence. RESULTS: Seventeen of the 18 patients had abnormal findings at examination. Eight patients had obstruction of the reconstructed anus. Eleven patients had sacral deformities. Nine patients had a defect in the external anal sphincter and nine patients could not contract the sphincter on demand. Five patients had significant prolapse of mucosa. In one patient, the neoanus was totally misplaced, one patient had a rectovaginal fistula, and one patient had short bowel syndrome due to several small bowel resections. Ten patients were offered conservative and five surgical treatment. CONCLUSIONS: This case series of adults shows that a majority of the patients can be offered further treatment. This indicates a need for structured follow-up of ARM patients into adulthood.

Controlled outcome of Hirschsprung's disease beyond adolescence: a single center experience.

PURPOSE: The aim of this study was to assess the function and quality of life of Hirschsprung's Disease (HD) beyond adolescence and relate it to matched controls. METHODS: All 203 patients diagnosed with HD at our department from 1961 to 1995 were identified. 21 had died, 43 had unclear diagnosis and 16 could not be traced. The remaining 123 patients were sent bowel function and SF-36 quality of life questionnaires. 69 patients (mean age 37.8, range 22-58, 13 female) responded and were matched with 138 age and sex-matched controls. RESULTS: Function: HD-patients had significantly higher number of bowel movements per week, higher incidence of soiling, urgency, permanent stomas, use of laxatives, enemas and loperamide. HD-patients also scored significantly lower in their satisfaction with their bowel function. There was, however, no significant difference in Miller Incontinence score. QOL: HD-patients reported a significantly higher incidence of negative impact by their bowel function on daily life, social interaction and ability to go on vacation. There were no significant differences in SF-36-scores. CONCLUSIONS: Bowel function has a lifelong negative impact on the lives of patients with HD. This strongly indicates a need for structured follow-up beyond adolescence.

Outcome of laparoscopic versus open gastrostomy in children.

PURPOSE: Laparoscopic gastrostomy (LAPG) has gained popularity in children. The aim of this study was to compare the outcome of LAPG versus open gastrostomy (OG) in children with focus on complications, operative times and postoperative length of stay. METHODS: Retrospective study of children who had gastrostomies inserted at our tertiary Pediatric Surgery Center from 2000 until 2013. The indications for a gastrostomy were an anticipated need for enteral support for at least 6 months. Totally 243 children were included in the study, 83 with LAPG and 160 with OG. RESULTS: We found a significant difference in postoperative length of stay, 3 days in the LAPG group versus 4 days in the OG group but no difference in a sub-group analysis from 2010 to 2013 when both techniques were used. There was no difference in median operative time or complications rates. Granuloma was the dominating complication in both groups. CONCLUSION: These two feeding-access techniques are comparable regarding complications, operative times and postoperative length of stay. The choice of surgical method should be individualized based on the patient's characteristics and the experience of the surgeon. The favorable results with LAPG in adults are not necessarily transferable to children since there are physiological and anatomical differences.

Antroduodenal manometry findings in children with suspected pediatric intestinal pseudo-obstruction.

BACKGROUND: In 2018 diagnostic criteria for pediatric intestinal pseudo-obstruction (PIPO) were established. Neuromuscular dysfunction of the gastrointestinal tract is one of these, and often examined through antroduodenal manometry (ADM). There is little data on antroduodenal manometries in children. Our objectives were to retrospectively apply these criteria to children evaluated for suspected motility disorder, to reevaluate the ADM patterns and compare children who did and did not meet the PIPO criteria and also with healthy adults. METHODS: Children with a suspected gastrointestinal motility disorder previously investigated with 24-h 8-lead ADM were reevaluated by applying the 2018 ESPGHAN/NASPGHAN PIPO diagnostic criteria and the 2018 ANMS-NASPGHAN guidelines. ADM findings were compared between children who retrospectively fulfilled a PIPO diagnosis, children who did not, and a control group of healthy adults. KEY RESULTS: Of 34 children (age 7.9 (±5.1) years, 18 males), 12 retrospectively fulfilled the 2018 PIPO diagnostic criteria. Twenty-five children (10 in the PIPO group) had abnormal diagnostic findings on ADM, whereas 9 (2 in the PIPO group) had no such findings. A PIPO diagnosis implied a significantly higher degree of abnormal ADM patterns (2.33 vs. 1.23, p = 0.02). There were no major differences in quantitative ADM measurements between the groups except higher pressures in children. CONCLUSIONS AND INFERENCES: Children who retrospectively fulfilled a PIPO diagnosis had a significantly higher abundance of abnormal ADM findings compared with symptomatic children without PIPO and healthy adults. Our data indicate a need for set criteria for evaluation of ADM in children with suspected PIPO.

Role of Magnetic Compression Anastomosis in Long-Gap Esophageal Atresia: A Systematic Review.

Background: Magnetic compression anastomosis (MCA) is an alternative technique for patients with long-gap esophageal atresia (EA). It allows for preservation of the native esophagus. We aimed to systematically summarize the current literature on MCA in EA. Methods: Studies where neonates with EA were treated with MCA devices were included, while studies on esophageal stenosis were excluded. All clinical studies, including comparative studies, case series, and case reports, were eligible for inclusion. Methodological quality assessment was performed using a validated tool. Results: Twelve studies with a total of 42 patients were included in this review. There was a wide variation among these studies with regard to the time of initiation of MCA (1 day to 7 months), procedure time (13-320 minutes), and magnet characteristics (strength, size, and shape of the magnets used). The time to achieve anastomosis ranged from 1 to 12 days. Stricture at the anastomotic site was reported in almost all the patients, which required multiple endoscopic dilatations (median no. of dilatations/patient = 9.8). Stent placement for refractory stricture was required in 9 (21%) patients, and surgery for stricture was required in 6 (14%) patients. Long-term outcomes included esophageal dysmotility (n = 3) and recurrent pulmonary infections (n = 3) were reported in only four studies. Conclusion: As per the findings of this review, neonates with long-gap EA undergoing MCA would invariably require multiple sittings of endoscopic dilatations (median no. of dilatations/patient = 9.8). Also, there is a wide variation among the included studies in terms of the procedure of MCA. Future studies with a standardized procedure for achieving MCA are needed to determine additional outcomes in this fragile patient population.

Controlled long term outcome of pyloromyotomy for pyloric stenosis: No long-term adverse effect.

PURPOSE: Pyloromyotomy for pyloric stenosis is one of the more common surgical procedures performed on infants. The long-term effects of the procedure are however unclear. The purpose of this study was to study the effects into adult life, compare them with controls and to see if there is a need for structured follow up of patients. METHODS: Of the 101 patients operated for pyloric stenosis between 1972 and 1974 at our tertiary referral center 91 could be traced. They were all invited to participate in the study and were sent validated questionnaires (PAGI-SYM, GIQLI) as well as a study-specific questionnaire examining the use of antacid drugs, incidence of gastroscopy and abdominal surgery. Sixty patients responded (66%, mean age 45 years, 46 male) and were included. Thereafter, 600 age and sex-matched controls were sent the same questionnaires. 132 responded (22%, 90 male) and were includes as controls. RESULTS: No significant differences could be found in any of the examined parameters when looking at the whole material or the male patients. Female patients had higher PAGI-SYM-scores for post prandial fullness (mean 1.11 vs 0.43, P = 0.035) and heartburn (mean 0.59 vs 0.14, P = 0.043) when compared to controls. CONCLUSIONS: The present study shows that most patients operated for pyloric stenosis during infancy experience no negative effects into adulthood. The finding in the female patient group is interesting but is unlikely to have any clinical implications. The results from this study strongly implicate that there is no need for follow up of patients into adulthood. LEVEL OF EVIDENCE: Level III.

Percutaneous hernia repair in children: Safe to introduce.

BACKGROUND AND OBJECTIVE: In 2014 we introduced percutaneous internal ring suture as an alternative to open surgery for the treatment of inguinal hernia. This study aims to evaluate the introduction of the procedure at our institution. METHODS: In total, 100 consecutive patients operated with percutaneous internal ring suture were compared with 100 consecutive patients operated with open surgery. The patients were operated from August 2014 until November 2017. Patient demographics, clinical history, operative time, time in theater, and postoperative complications were extracted from charts. RESULTS: The mean operative time for percutaneous internal ring suture was 26.54 min and for open surgery 39.94 min, P < 0.0001. The total mean operative theater time for percutaneous internal ring suture was 108.95 min and for open surgery 118.4 min, P = 0.0343. During follow-up, two percutaneous internal ring sutures were operated for recurrent hernia. In the open surgery-group, three patients were operated for recurrent hernia, three for secondary testicular retention, and three for metachronous contralateral hernia. CONCLUSIONS: Even when established as a new technique, the percutaneous internal ring suture procedure is safe and results in shorter operative time and shorter theater time compared to open surgery.

Injectable bulking treatment of persistent fecal incontinence in adult patients after anorectal malformations.

BACKGROUND/PURPOSE: Injectable bulking therapy has emerged as a treatment for fecal incontinence (FI), however there are no studies including adult patients with anorectal malformations (ARM). This study aimed to evaluate non-animal stabilized hyaluronic acid with dextranomer (NASHA/Dx) for the treatment of adult ARM patients with persistent FI. METHODS: Seven adults with ARM and incontinence to loose stool at least once weekly and without rectal or mucosal prolapse were treated with anal NASHA/Dx injection. They were evaluated preoperatively, at 6 and 18 months with a bowel function questionnaire and a 2-week bowel diary as well as FIQL and SF-36 quality of life questionnaires. RESULTS: Before treatment, the mean number of incontinence episodes over 2 weeks was 20.7 (median 16, range 8-52). At 6 months, the corresponding figures were 5.3 (median 4, range 0-19, p = 0.018), and at 18 months the figures were 4.3 (median 2, range 1-20, p = 0.018). An improved physical function in SF-36 from 74.3 at baseline to 86.4 at 6 months was noted (p = 0.04). No serious adverse events occurred. CONCLUSIONS: NASHA/Dx is a promising treatment option for selected adult patients with persistent FI after ARM. Longer follow up of larger patient series and studies on patients in adolescence is needed. TYPE OF STUDY: Treatment Study. LEVEL OF EVIDENCE: Level IV.

Submucosal injection of stabilized nonanimal hyaluronic acid with dextranomer: a new treatment option for fecal incontinence.

PURPOSE: NASHA Dx gel has been used extensively for treatments in the field of urology. This study was performed to evaluate NASHA Dx gel as an injectable anal canal implant for the treatment of fecal incontinence. METHODS: Thirty-four patients (5 males, 29 females; median age, 61 years; range, 34 to 80) were injected with 4 x 1 ml of NASHA Dx gel, just above the dentate line in the submucosal layer. The primary end point was change in the number of incontinence episodes and a treatment response was defined as a 50 percent reduction compared with pretreatment. All patients were followed up at 3, 6, and 12 months. RESULTS: The median number of incontinence episodes during four weeks was 22 (range, 2 to 77) before treatment, at 6 months it was 9 (range, 0 to 46), and at 12 months it was 10 (range, 0 to 70, P = 0.004). Fifteen patients (44 percent) were responders at 6 months, compared with 19 (56 percent) at 12 months. No long-term side effects or serious adverse events were reported. CONCLUSIONS: Submucosal injection of NASHA Dx gel is an effective treatment for fecal incontinence. The effect is sustained for at least 12 months. The treatment is associated with low morbidity.

Long-Term Outcome after Dynamic Graciloplasty for Treatment of Persistent Fecal Incontinence in Patients with Anorectal Malformations.

PURPOSE: Dynamic graciloplasty (DGP) has been used to treat severe fecal incontinence since the 1980s. Previous studies have shown an inferior outcome in patients with anorectal malformations (ARMs). Our experience has been that DGP has been appreciated by ARM -patients. The objective of the study was to evaluate the long-term outcome of DGP in our patients with ARM compared with patients with other underlying conditions. MATERIALS AND METHODS: Twenty-three patients operated with DGP at our institution from 1996 to 2010 were sent validated bowel function and quality of life questionnaires. Eighteen of 23 responded. Seven had ARM and 11 had other etiologies of fecal incontinence. The mean follow-up time was 11.6 years (range, 5-17). RESULTS: Four of 7 of the patients with ARM and 8 of 11 of patients with other etiologies used their implants at follow-up. The Miller incontinence score was slightly higher for patients with ARMs, but they had less constipation and higher Fecal Incontinence Quality of Life (FIQL)- and 36-Item Short Form Health Survey (SF-36) scores. None of the differences were statistically significant. CONCLUSION: This study cannot confirm earlier reports in which DGP has an inferior outcome in patients with ARM. We therefore believe that the procedure should remain a treatment option for selected patients.

Outcome in adults with anorectal malformations in relation to modern classification - Which patients do we need to follow beyond childhood?

BACKGROUND/PURPOSE: Knowledge about the functional outcome in adults with anorectal malformations is essential to organize structured transition to adult care for this patient group. The aim of this study was to investigate the functional outcome and quality of life in adults with anorectal malformations characterized according to the Krickenbeck classification. METHODS: Of 256 patients diagnosed with anorectal malformations at our institution in 1961-1993, 203 patients could be traced and were invited to participate in the study. One hundred and thirty-six patients replied (67%) and were compared with one hundred and thirty-six population based sex and age-matched controls. Patients and controls were evaluated with both a validated questionnaire as well as a study-specific questionnaire to assess bowel function. SF-36 was used for quality of life. Outcome in nine incontinence-related parameters, 10 constipation-related, 6 urogenital function-related, and 13 quality of life parameters were assessed in the patients and compared to the outcome of controls as well as to the type of anorectal malformations according to the Krickenbeck classification. RESULTS: The ARM-patients had an inferior outcome (P<0.05) for all incontinence parameters, 8 of 10 parameters for constipation, 2 of 6 for urogenital function and 7 of 13 quality of life parameters. Patients with rectobulbar and vestibular fistulas had the worst statistical outcome but patients with cloaca and rectoprostatic/bladder-neck fistula had worse outcome in absolute numbers. Forty-four patients (32%) reported incontinence of stool at least once a week and 16 (12%) had a permanent colostomy. CONCLUSIONS: The functional outcome and quality of life in adults with anorectal malformations are closely related to the type of malformation. A large proportion of the patients have persistent fecal incontinence, constipation and sexual problems that have a negative influence on their quality of life. Structured multidisciplinary follow-up of adults with anorectal malformations by pediatric and colorectal surgeons, as well as urologists and gynecologists is therefore advocated.

Adult outcomes after surgery for Hirschsprung's disease: Evaluation of bowel function and quality of life.

BACKGROUND AND AIMS: Hirschsprung's disease (HSCR) is treated surgically. There is a risk for faecal incontinence and constipation postoperatively. The long-term bowel functional outcome in adults and quality of life are sparsely studied. The aim of this study was to assess bowel function and quality of life in patients who had undergone surgery for HSCR during childhood. METHODS: All patients treated between 1969 and 1994 at St. Göran's Children's Hospital in Stockholm were invited to participate in the study. After consent, the patients received questionnaires containing general questions, validated questions on bowel function, questions about urinary function, SF-36 health survey (SF-36) and the Gastrointestinal Quality of Life Index (GIQLI). Clinical data were extracted from the case records. Controls matched for sex and age were randomly selected from the National Swedish Population Register. RESULTS: 48 of 60 (80%) invited patients responded to the questionnaires. Nine patients were excluded since the HSCR diagnosis could not be confirmed. The median age of the included patients was 28 (20-43) years. Most patients had undergone Soave's operation (73.4%) and two patients had a stoma at the time of follow-up. The bowel function was impaired in the HSCR group compared to controls, especially problems with flatulence, need to strain at defecation and several defecations for emptying. Patients in the HSCR group also had significantly more problems with faecal incontinence than controls. Quality of life according to SF-36 did not differ significantly between patients and controls, but the GIQLI score showed a significantly worse outcome in the HSCR group compared to the controls. CONCLUSION: General quality of life in adults treated for HSCR during childhood is comparable to controls. However, they have impaired bowel function and gastrointestinal quality of life.

Posterior sagittal anorectoplasty results in better bowel function and quality of life in adulthood than pull-through procedures.

BACKGROUND/PURPOSE: The short-term outcome of posterior sagittal anorectoplasty (PSARP) procedure has been reported to be better than after abdominoperineal or abdominosacroperineal (AP) procedures. This study aimed to investigate the long-term functional outcome and quality of life after PSARP in adulthood and compare with the outcome after AP procedures. METHODS: Twenty-four patients operated with PSARP at the Department of Pediatric Surgery, Uppsala, Sweden, from 1984 to 1993 were identified. They were compared with 20 patients that underwent AP pull-through procedures from 1974 to 1983. The patients were sent validated bowel function and quality of life (SF-36) questionnaires. Sixteen PSARP (median age 21, five females) patients and fourteen AP patients (seven abdominosacroperineal and seven abdominoperineal pull-throughs, median age 32, seven females) responded and were included in the study. RESULTS: The median Miller incontinence score was 1 (range 0-13) in the PSARP group and 10 (range 3-16) in the pull-through group (P=0.0042). The use of underwear protection and oral loperamide was significantly less frequent in the PSARP group (P=0.0096 and 0.0021 respectively). The SF-36 scores of Vitality, Mental health and Mental Cluster Scale were higher in the PSARP group (P=0.0291, 0.0500, 0.0421 respectively). CONCLUSIONS: PSARP results in superior bowel function and better quality of life in adulthood compared with AP procedures for the repair of anorectal malformations.

Long-term outcome after free autogenous muscle transplantation for anal incontinence in children with anorectal malformations.

PURPOSE: Patients with high anorectal anomalies are often incontinent after reconstruction, particularly with the older forms of surgical treatment, that is, anorectal pull-through or Stephen's operations. In 1974, a new treatment for anal incontinence in children was introduced at the Akademiska Hospital: free autogenous muscle transplantation (FAMT) to the perirectal area. All the patients receiving FAMT were totally incontinent before the procedure and had no rectal sensitivity. The aim of this study was to evaluate the long-term functional outcome of this procedure. METHODS: Twenty-two patients (17 males) operated on with FAMT below the age of 15 years were identified through records. One of the patients had died, and 2 were not available for follow-up. The remaining 19 were sent a validated bowel function questionnaire, and 15 (78.9%) of 19 patients responded (12 males). These 15 patients were compared with 15 patients with the same sex, age, and a similar malformation from our patient database. RESULTS: At follow-up, after an average of 30 years postoperatively, 2 of 15 patients with FAMT had a stoma compared with 3 of 15 in the control group. The Miller incontinence score had a mean of 6.2 (median, 6; range, 0-15) in the FAMT group and 3.7 (median, 4; range, 0-12) in the control group. All patients in both groups could sense stool, and 11 of 13 patients in the FAMT group could distinguish between feces and flatus. CONCLUSIONS: The patients with FAMT had a slightly inferior anorectal function compared with the controls. Considering they were all totally incontinent before FAMT, we conclude that FAMT has an acceptable effect 30 years postoperatively. Therefore, we find that FAMT could be an alternative for anorectal malformation patients who are totally incontinent.