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Anorectal Malformations: Long-term outcome and aspects of secondary treatment
Uppsala University, Disciplinary Domain of Medicine and Pharmacy, Faculty of Medicine, Department of Surgical Sciences.
2015 (English)Doctoral thesis, comprehensive summary (Other academic)
Abstract [en]

Faecal incontinence (FI) is defined as the inability to control bowel movements. The causes of FI are many and diverse. One of the more uncommon reasons for FI is Anorectal Malformations (ARMs). An ARM is a congenital anomaly that affects somewhere between 1/2500 and 1/5000 live born babies. Many ARM patients have persistent FI. Several different procedures have been utilised to address this issue. This thesis aims to evaluate (1) the long-term outcome in adulthood of ARMs in relation to the modern Krickenbeck classification, and (2) scope for treating FI with transanal injection with dextranomer in non-animal stabilised hyaluronic acid (NASHA/Dx), in patients both with and without ARMs.

All patients treated for ARMs in Uppsala up to 1993 were invited to participate in a questionnaire study of quality of life and function. The study included 136 patients and compared them with 136 age- and sex-matched controls. The Krickenbeck classification was found to predict functional outcome, and ARM patients had more problems with incontinence and obstipation, as well as inferior Quality of Life (QoL), compared with controls. 

Thirty-six patients with FI, owing to causes other than ARMs, were treated with transanal submucous injection of NASHA/Dx. The patients were monitored for two years after treatment. Significant reductions in both their incontinence score and the number of their incontinence episodes were achieved.  A significant improvement in QoL was observed in patients who had at least a 75% reduction in incontinence episodes. No serious complications occurred.

A prospective study of transanal injection of NASHA/Dx was conducted on seven patients with persistent FI after ARMs. After six months a significant reduction in the number of incontinence episodes was obtained. A significant improvement in QoL was also found. No serious complications occurred.

In conclusion, adult patients with ARMs have inferior outcome of anorectal function and QoL compared with controls. NASHA/Dx is effective and appears to be safe in treating FI in general. This effect seems to be the same in selected patients with persistent FI after ARMs.

Place, publisher, year, edition, pages
Uppsala: Acta Universitatis Upsaliensis, 2015. , 109 p.
Series
Digital Comprehensive Summaries of Uppsala Dissertations from the Faculty of Medicine, ISSN 1651-6206 ; 1065
Keyword [en]
anorectal malformation, faecal incontinence, long-term outcome, Krickenbeck, NASHA/Dx
National Category
Surgery
Identifiers
URN: urn:nbn:se:uu:diva-241243ISBN: 978-91-554-9140-6 (print)OAI: oai:DiVA.org:uu-241243DiVA: diva2:777993
Public defence
2015-02-27, Rosénsalen, Entrance 95/96, ground floor, Uppsala University Children’s Hospital, Uppsala, 13:15 (English)
Opponent
Supervisors
Available from: 2015-02-05 Created: 2015-01-09 Last updated: 2015-03-09
List of papers
1. Outcome in adults with anorectal malformations in relation to modern classification – Which patients do we need to follow beyond childhood?
Open this publication in new window or tab >>Outcome in adults with anorectal malformations in relation to modern classification – Which patients do we need to follow beyond childhood?
2017 (English)In: Journal of Pediatric Surgery, ISSN 0022-3468, E-ISSN 1531-5037, Vol. 52, no 3, 463-468 p.Article in journal (Refereed) Published
Abstract [en]

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.

Keyword
Anorectal malformations, Long term outcome, Functional outcome, Sexual outcome, Quality of life, Transition to adult care
National Category
Surgery
Research subject
Pediatric Surgery
Identifiers
urn:nbn:se:uu:diva-241240 (URN)10.1016/j.jpedsurg.2016.10.051 (DOI)000397964100019 ()27894765 (PubMedID)
Available from: 2015-01-09 Created: 2015-01-09 Last updated: 2017-04-26Bibliographically approved
2. Submucosal injection of stabilized nonanimal hyaluronic acid with dextranomer: a new treatment option for fecal incontinence
Open this publication in new window or tab >>Submucosal injection of stabilized nonanimal hyaluronic acid with dextranomer: a new treatment option for fecal incontinence
Show others...
2009 (English)In: Diseases of the Colon & Rectum, ISSN 0012-3706, E-ISSN 1530-0358, Vol. 52, no 6, 1101-1106 p.Article in journal (Refereed) Published
Abstract [en]

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.

National Category
Medical and Health Sciences
Identifiers
urn:nbn:se:uu:diva-113092 (URN)10.1007/DCR.0b013e31819f5cbf (DOI)000273644400010 ()19581853 (PubMedID)
Available from: 2010-01-25 Created: 2010-01-25 Last updated: 2017-12-12Bibliographically approved
3. Efficacy and quality of life 2 years after treatment for faecal incontinence with injectable bulking agents
Open this publication in new window or tab >>Efficacy and quality of life 2 years after treatment for faecal incontinence with injectable bulking agents
2013 (English)In: Techniques in Coloproctology, ISSN 1123-6337, E-ISSN 1128-045X, Vol. 17, no 4, 389-395 p.Article in journal (Refereed) Published
Abstract [en]

BACKGROUND:

Stabilized non-animal hyaluronic acid/dextranomer (NASHA® Dx) gel as injectable bulking therapy has been shown to decrease symptoms of faecal incontinence, but the durability of treatment and effects and influence on quality of life (QoL) is not known. The aim of this study was to assess the effects on continence and QoL and to evaluate the relationship between QoL and efficacy up to 2 years after treatment.

METHODS:

Thirty-four patients (5 males, mean age 61, range 34-80) were injected with 4 × 1 ml NASHA Dx in the submucosal layer. The patients were followed for 2 years with registration of incontinence episodes, bowel function and QoL questionnaires.

RESULTS:

Twenty-six patients reported sustained improvement after 24 months. The median number of incontinence episodes before treatment was 22 and decreased to 10 at 12 months (P = 0.0004) and to 7 at 24 months (P = 0.0026). The corresponding Miller incontinence scores were 14, 11 (P = 0.0078) and 10.5 (P = 0.0003), respectively. There was a clear correlation between the decrease in the number of leak episodes and the increase in the SF-36 Physical Function score but only patients with more than 75 % improvement in the number of incontinence episodes had a significant improvement in QoL at 24 months.

CONCLUSIONS:

Anorectal injection of NASHA Dx gel induces improvement of incontinence symptoms for at least 2 years. The treatment has a potential to improve QoL. A 75 % decrease in incontinence episodes may be a more accurate threshold to indicate a successful incontinence treatment than the more commonly used 50 %.

National Category
Medical and Health Sciences
Identifiers
urn:nbn:se:uu:diva-196656 (URN)10.1007/s10151-012-0949-8 (DOI)000321915700010 ()23224913 (PubMedID)
Available from: 2013-03-12 Created: 2013-03-12 Last updated: 2017-12-06Bibliographically approved
4. Injectable bulking treatment of persistent faecal incontinence after anorectal malformations. A preliminary report.
Open this publication in new window or tab >>Injectable bulking treatment of persistent faecal incontinence after anorectal malformations. A preliminary report.
(English)Manuscript (preprint) (Other academic)
Abstract [en]

Background/Purpose

During the last decades injectable bulking therapy has emerged as a promising treatment option for fecal incontinence (FI). Very little has been published on the use of injectable bulking therapy on patients with persistent FI after 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 adult patients with persistent FI after ARM were treated with perianal submucuos injection of NASHA/Dx. They were evaluated preoperatively and at three and six months after treatment with a validated bowel function questionnaire and a two-week bowel diary as well as FIQL and SF-36 quality of life questionnaires.

Results

Before treatment mean Miller incontinence score was 13,7 and the mean number of incontinence episodes over two weeks was 20,7. At 6 months after treatment the corresponding figures were 10,7 (P=0,1088) and 5,3 (P=0,0180) respectively. No significant changes could be found in either FIQL or SF-36 scores. No serious adverse events occurred during the study.

Conclusions

NASHA/Dx is a promising treatment option for selected adult patients with persistent FI after ARM. Long-term follow up of larger patient series as well as studies on patients in adolescence are needed.

 

National Category
Surgery
Identifiers
urn:nbn:se:uu:diva-241241 (URN)
Available from: 2015-01-09 Created: 2015-01-09 Last updated: 2015-03-09

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