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©The Author(s) 2019.
World J Gastroenterol. Jul 7, 2019; 25(25): 3268-3280
Published online Jul 7, 2019. doi: 10.3748/wjg.v25.i25.3268
Published online Jul 7, 2019. doi: 10.3748/wjg.v25.i25.3268
Table 1 Study characteristics of included studies
| Study | Study design | Inclusion period | Sample size, n | Male (%) | Syndromal patients (%) | Total follow-up in months | Number of BT injections needed per patient | Age at first BT injection (yr) | Type BT | Mean dose (IU/injection) | Guidance at BT injection | Definition of outcomes | Improvement in obstructive symptoms < 1 mo | Prolonged improvement in obstructive symtpoms | Improvement of enterocolitis | Complications/adverse effects | Decrease in mean resting pressure on anorectal manometry (mmHG) |
| Basson (2014) | Retrospective | 2010-2014 | 11 | 67 | NR | 12-72 | 1pt: 1BTI5pt: 2BTI4pt: 3BTI1pt: 4BTI | 5 | Dysport | 200 | Palpation | Successful-Improvement-FailedFavorable: successful/improvement | 10/11 (91%) | 5/11 (45%) | NR | 1 (transient soiling) | NR |
| Chumpitazi (2008) | Retrospective | 1998-2007 | 30 | 80 | 10 | 41.2 ± 4.9 | 2.7 ± 0.2 | 5 | Botox | NR | Palpation | Poor-Fair-Good-Excellent Favorable: Excellent/good | 27/30 (90%) | 11/30 (37%) | NR | 8 (7 transients soiling, 1 anal pain) | NR |
| Chumpitazi (2011) | Retrospective | 1998-2016 | 37 | 80 | 23 | 41.4 ± 4.5 | 2.8 ± 0.3 | NR | Botox | 100 | NR | Poor-Fair-Good-Excellent Favorable: Excellent/good | 33/37 (90%) | NA | NR | NR | NR |
| Church (2016) | Retrospective | 2010-2015 | 30 | NR | NR | 20 | 87% in total:With US: 2Without US: 1 | 3.1 | NR | 40 | US-guided | Improvement of symptoms | NA | NR | 3/4 (75%) | NR | NR |
| Han-Geurts (2014) | Retrospective | 2002-2013 | 33 | 79 | 0 | 7.3 yr (1-24) | 2 (1-5) | 3.6 | Dysport | 200 | NR | Poor-Fair-Good-Excellent Favorable: Excellent/good | 25/33 (76%) | 17/33 (52%) | 7/19 (37%) | 2 | NR |
| Hemanshoo Thakkar (2017) | Retrospective | 2002-2014 | 6 | NR | NR | 6 yr (1-12) | 3 | Dysport | 200 | US-guided | Short-term: postoperative complications < 30 dLong-term: Rintala Bowel Function Score (BFS) | 1/6 (17%) | NR | NR | NR | NR | |
| Hosseini (2008) | Prospective | 2002-2006 | 16 | 62 | NR | 8 pt: 1-3 | NR | Dysport | NR | NR | Improvement in Constipation score (good/recurrence/non-responders) | 14/16 (88%) | 8/16 (50%) | NR | NR | NR | |
| Jiang (2009) | Prospective | 2000-2008 | 23 | 65 | NR | 12 | NR | Botox | 120 | Palpation | Poor-Moderate-Excellent Favorable: Moderate/Excellent | NR | 21/23 (91%) | NR | 9 (anal pain) | 8-30 | |
| Koisuvalo (2009) | Retrospective | 2005-2008 | 16 * | 62 | 12.5 | 19 (3-43) | 2 (1-4) | 4 | NR | 100 | NR | No effect-Little effect-Significant effect-Symptoms dissapearedFavorable: Significant effect/symptoms dissapeared | 10/16 (63%) | 3/8 (38%) | 1/4 (25%) | 4 (increased soiling) | 28-31 (2 w); 8-24 (8 m) |
| Langer (1997) | Prospective | NR | 4 | 50 | 25 | 3 pt: 1, 1 pt 2 | 6 | Botox | NR | Palpation | Improvement of obstructive symptoms, presence of incontinence | 3/4 (75%) | 1/4 (25%) | NR | 1 (transient incontinence) | NR | |
| Langer (2004) | Retrospective | NR | 14 | NR | NR | 24 | 4 pt: 1, 9pt: 1-4 | 4 | NR | 150 | NR | Improvement of obstructive symptoms, presence of incontinence | 9/14 (64%) | 4/14 (29%) | NR | NR | NR |
| Minkes (2000) | Prospective | NR | 18 | 78 | NR | 8: 1; 10: 2-6 | NR | Botox | 60 | Palpation | No response-Significant response | 12/18 (67%) | 5/18 (28%) | NR | 4 (transient incontinence) | 35-37 | |
| Patrus (2010) | Retrospective | 1998-2008 | 22 | 78 | 5 | 5.0 ± 2.9 yr (0-10) | 2 (1-23) | 8.4 | NR | 120 | NR | Improvement of obstructive symptoms, presence of incontinence | 18/22 (81%) | 6/22 (27%) | NR | 0 | NR |
| Wester (2015) | Retrospective | 2007-2014 | 18 | 83 | 33 | 3, 8 yr (0, 1-8, 3) | 3 (1-13) | 2.4 | Botox | 100 | Palpation | Good-Insufficient | NR | 13/18 (72%) | NR | NR | NR |
Table 2 Main findings and risk of bias analysis
| Effect | No. of studies | Event rate (95%CI) | Heterogeneity (I2) | Significant predictors | Eggers intercept | Fail safe, n |
| Improvement of obstructive symptoms | 14 | ER = 0.66 95%CI: 0.55-0.75a | 49.5% | None | -0.42 | 43 |
| Decreasing enterocolitis incidence | 3 | ER = 0.58, 95%CI: 0.27-0.84b | 71.0% | NA | 3.27 | 0 |
| Adverse effects | 9 | ER = 0.17, 95%CI: 0.10-0.29c | 52.1% | NA | -2.78 b | 101 |
Table 3 Quality of included studies
| Study | Selection | Comparibility | Outcome | Total |
| Basson (2014) | 2 | 0 | 3 | Poor |
| Chumpitazi (2008) | 3 | 0 | 3 | Poor |
| Chumpitazi (2011) | 3 | 0 | 3 | Poor |
| Church (2016) | 2 | 0 | 2 | Poor |
| Han-Geurts (2014) | 2 | 0 | 3 | Poor |
| Hemanshoo Takkar (2017) | 3 | 0 | 3 | Poor |
| Hosseini (2008) | 3 | 0 | 2 | Poor |
| Jiang (2009) | 3 | 0 | 3 | Poor |
| Koisuvalo (2009) | 3 | 0 | 3 | Poor |
| Langer (1997) | 3 | 0 | 3 | Poor |
| Langer (2004) | 3 | 0 | 2 | Poor |
| Minkes (2000) | 3 | 0 | 2 | Poor |
| Patrus (2010) | 3 | 0 | 3 | Poor |
| Wester (2015) | 3 | 0 | 3 | Poor |
- Citation: Roorda D, Abeln ZA, Oosterlaan J, van Heurn LW, Derikx JP. Botulinum toxin injections after surgery for Hirschsprung disease: Systematic review and meta-analysis. World J Gastroenterol 2019; 25(25): 3268-3280
- URL: https://www.wjgnet.com/1007-9327/full/v25/i25/3268.htm
- DOI: https://dx.doi.org/10.3748/wjg.v25.i25.3268