COMPLICATIONS AFTER TRANSABDOMINAL SOAVE’S PROCEDURE IN CHILDREN WITH HIRSCHSPRUNG’S DISEASE

Shahnam ASKARPOUR Mehran PEYVASTEH Mohammad Hossein IMANIPOUR Hazhir JAVAHERIZADEH Saeed HESAM About the authors

ABSTRACT

Background:

Hirschsprung’s disease is a congenital disorder that causes functional obstruction of large bowel.

Aim:

To evaluate complication and bowel function score of children with Hirschsprung’s disease who underwent transabdominal Soave’s procedure.

Methods:

In this study all the children with Hirschsprung’s disease who underwent transabdominal Soave procedure were evaluated regarding bowel function and complication of trans-abdominal Soave’s procedure.

Results:

Were enrolled 160 children. Enterocolitis and constipation were seen in 15% of the cases. Fecal incontinency was the least frequent study which was seen in 1% of the children.

Conclusion:

Constipation and enterocolitis was the most frequent complication following transabdominal Soave technique.

HEADINGS:
Constipatio; Hirschsprung diseas; Enterocoliti

RESUMO

Racional:

A doença de Hirschsprung é um distúrbio congênito que causa obstrução funcional do intestino grosso.

Objetivo:

Avaliar as complicações e o escore de função intestinal de crianças com a doença submetidas ao procedimento transabdominal de Soave.

Métodos:

Neste estudo, todas as crianças com doença de Hirschsprung submetidas ao procedimento transabdominal de Soave foram avaliadas quanto à função intestinal e complicação do procedimento.

Resultados:

Foram incluídas 160 crianças. Enterocolite e constipação foram observadas em 15% dos casos. A incontinência fecal foi menos frequente e observada em 1% das crianças.

Conclusão:

Obstipação e enterocolite foram as complicações mais frequentes após a técnica de Soave transabdominal em crianças.

DESCRITORES:
Constipação intestina; Enterocolit; Doença de Hirschsprung

INTRODUCTION

Hirschsprung’s disease is a congenital disorder that causes functional obstruction of large bowel. It´s incidence is estimated in 1:5000 live birth with a male predominance11 Amiel J, Lyonnet S. Hirschsprung disease, associated syndromes, and genetics: a review. J Med Genet 2001;38:729-39.,22 Archibong AE. Pattern of aganglionic megacolon in Calabar, Nigeria. S Afr Med J 2002,92:642-644.. Diagnosis is done using anorectal manometry, barium enema33 de Lorijn F, Boeckxstaens GE, Benninga MA. Symptomatology, pathophysiology, diagnostic work-up, and treatment of Hirschsprung disease in infancy and childhood. Curr Gastroenterol Rep 2007;9:245-53. and rectal biopsy. Niramis et al1212 Niramis R, Watanatittan S, Anuntkosol M, et al. Quality of life of patients with Hirschsprung's disease at 5 - 20 years post pull-through operations. Eur J Pediatr Surg 2008;18:38-43. with patients who underwent pull-through procedure, found enterocolitis as the most common post-surgical complication. For Little et al.99 Little D, Snyder C. Early and late complications following operative repair of Hirschsprung's disease. Hirschsprung's Disease and Allied Disorders: Springer; 2008. p. 375-85 enterocolitis was the most common post-operative complication followed by constipation and bowel obstruction. In the study by Shakya et al1616 Shakya VC, Agrawal CS, Adhikary S. Initial experience with Soave's transabdominal pull-through: an observational study. Int J Surg 2010;8:225-8., constipation was seen in 11.7% of children who underwent transabdominal Soave’s pull-through procedure. In the literature review by Rintala et al1515 Rintala RJ, Pakarinen MP. Long-term outcomes of Hirschsprung's disease. Semin Pediatr Surg 2012;21:336-43., fecal incontinency and constipation were the most post-operative complication of Hirschsprung’s disease. Bowel function was lower than normal population.

The aim of this study was to evaluate complications and bowel function score in children with Hirschsprung’s disease who underwent transabdominal Soave’s procedure.

METHODS

This study was approved by Ethical Committee of the University (IRAJUMS.REC.1395.364). It was approved by Research Affair of Ahvaz Jundishapur University of Medical Sciences.

All children who underwent trans-abdominal Soave’s procedure were included. Patients with Down syndrome and total colonic involvement were excluded.

Qualitative clinical scoring was used for assessment of bowel function which was proposed by Holschneider44 Holschneider AM. Elektromanometrie des Enddarmes. 2nd ed. München, Wien, Baltimore. There is no need for physical examination. According to these criteria, 14 points means excellent bowel function Score interpretation is shown in Figure 1.

FIGURE 1
Functional score for clinical evaluation

RESULTS

In this study, 160 children with Hirschsprung disease who underwent Soave’s procedure were included. Results of postoperative complications are in Table 1 and show constipation (n=24,15%) and enterocolitis (n=24, 15%) being the more frequent complications after trans-abdominal Soave’s procedure. The least one was fecal incontinency which was seen in 1% (n=2) of the cases. The patients´ score is seen in Table 3.

TABLE 1
Complications following transabdominal Soave’s procedure

Initially were included 163 children who underwent surgery during two years. Among them, two who had Down syndrome and one total aganglionosis were excluded. So, the total enrollment was 160 children. There were 108 (67.5%) male and 52 (32.5%) female. Most of them (n=96, 60%) were diagnosed when they had less than one month of age (Table 2). As seen in Table 2, most of the patients underwent procedure at the ages <1 month.

TABLE 2
Age distribution at diagnosis

TABLE 3
Evaluation of patients according to bowel function score

DISCUSSION

Most of our cases were diagnosed and underwent surgery in the neonatal period, contrary to the results published by Mabula et al1010 Mabula JB, Kayange NM, Manyama M, et al. Hirschsprung's disease in children: a five year experience at a Univerity teaching hospital in northwestern Tanzania. BMC Res Notes 2014,7:410 referring only 5.5% in this condition. In the developed countries, more than 90% of the cases were in the neonatal period. So, our findings are consistent with developed countries, as discussed by Archibong22 Archibong AE. Pattern of aganglionic megacolon in Calabar, Nigeria. S Afr Med J 2002,92:642-644..

In this study 67.5% of the patients were boys and 32.5% girls. De Lor gin et al33 de Lorijn F, Boeckxstaens GE, Benninga MA. Symptomatology, pathophysiology, diagnostic work-up, and treatment of Hirschsprung disease in infancy and childhood. Curr Gastroenterol Rep 2007;9:245-53. and Martucciello1111 Martucciello G. Hirschsprung's disease, one of the most difficult diagnoses in pediatric surgery: a review of the problems from clinical practice to the bench. Eur J Pediatr Surg 2008;18:140-9., the number of boys/girls were reported about 4/1 which, so higher than here.

Enterocolitis and constipation were the most common postoperative complication as also referred by other authors77 Khazdouz M, Sezavar M, Imani B, et al. Clinical outcome and bowel function after surgical treatment in Hirschsprung's disease. Afr J Paediatr Surg 2015;12:143-7.,1010 Mabula JB, Kayange NM, Manyama M, et al. Hirschsprung's disease in children: a five year experience at a Univerity teaching hospital in northwestern Tanzania. BMC Res Notes 2014,7:410. The rate of enterocolitis in this study was higher than the one reported by Parahita et al.1313 Parahita IG, Makhmudi A, Gunadi. Comparison of Hirschsprung-associated enterocolitis following Soave and Duhamel procedures. J Pediatr Surg 2017. and Huang et al also mentioned enterocolitis as the earliest postoperative complication (28.73%) being fecal incontinency (20.99%) also frequent55 Huang W-K, Li X-L, Zhang J, et al. Prevalence, risk factors, and prognosis of postoperative complications after surgery for Hirschsprung Disease. J Gastrointest Surg 2018;22:335-343.. Constipation can be caused by high anal resting pressure and a weak rectal peristalsis as described by Keshtgar et al66 Keshtgar AS, Ward HC, Clayden GS, et al. Investigations for incontinence and constipation after surgery for Hirschsprung's disease in children. Pediatr Surg Int 2003;19:4-8..

Fecal incontinency was reported in 1% of our cases, differently to the ones reported by Niramis et al1212 Niramis R, Watanatittan S, Anuntkosol M, et al. Quality of life of patients with Hirschsprung's disease at 5 - 20 years post pull-through operations. Eur J Pediatr Surg 2008;18:38-43. in 15.6% with the procedure44 Holschneider AM. Elektromanometrie des Enddarmes. 2nd ed. München, Wien, Baltimore. Possibly, poor surgical technique could be the contributing factor for fecal incontinency 7.

Constipation was seen in 15% of children in this study. It has different results in the literature, as Niramis et al12 that reported it´s presence in 8.5%, lower than in our study.

The same divergence can be seen with the anastomotic stricture. In our sample it occurred in 7% different from Niramis et al data with 17.1%1212 Niramis R, Watanatittan S, Anuntkosol M, et al. Quality of life of patients with Hirschsprung's disease at 5 - 20 years post pull-through operations. Eur J Pediatr Surg 2008;18:38-43..

The main limitations of this study were being in a single center and relatively in short follow-up. Another multicenter study with longer follow-up is recommended to obtain more reliable results.

CONCLUSION

Constipation and fecal incontinency were the most frequent complication following transabdominal Soave’s procedure in follow-up of two years

ACKNOWLEDGMENT

The data used in this manuscript was from general physician thesis of Mohmmad Hossein Imanipour(GP95108).

REFERENCES

  • 1
    Amiel J, Lyonnet S. Hirschsprung disease, associated syndromes, and genetics: a review. J Med Genet 2001;38:729-39.
  • 2
    Archibong AE. Pattern of aganglionic megacolon in Calabar, Nigeria. S Afr Med J 2002,92:642-644.
  • 3
    de Lorijn F, Boeckxstaens GE, Benninga MA. Symptomatology, pathophysiology, diagnostic work-up, and treatment of Hirschsprung disease in infancy and childhood. Curr Gastroenterol Rep 2007;9:245-53.
  • 4
    Holschneider AM. Elektromanometrie des Enddarmes. 2nd ed. München, Wien, Baltimore
  • 5
    Huang W-K, Li X-L, Zhang J, et al. Prevalence, risk factors, and prognosis of postoperative complications after surgery for Hirschsprung Disease. J Gastrointest Surg 2018;22:335-343.
  • 6
    Keshtgar AS, Ward HC, Clayden GS, et al. Investigations for incontinence and constipation after surgery for Hirschsprung's disease in children. Pediatr Surg Int 2003;19:4-8.
  • 7
    Khazdouz M, Sezavar M, Imani B, et al. Clinical outcome and bowel function after surgical treatment in Hirschsprung's disease. Afr J Paediatr Surg 2015;12:143-7.
  • 8
    Levitt MA, Martin CA, Olesevich M, et al. J Pediatr Surg 2009;44:271-7.
  • 9
    Little D, Snyder C. Early and late complications following operative repair of Hirschsprung's disease. Hirschsprung's Disease and Allied Disorders: Springer; 2008. p. 375-85
  • 10
    Mabula JB, Kayange NM, Manyama M, et al. Hirschsprung's disease in children: a five year experience at a Univerity teaching hospital in northwestern Tanzania. BMC Res Notes 2014,7:410
  • 11
    Martucciello G. Hirschsprung's disease, one of the most difficult diagnoses in pediatric surgery: a review of the problems from clinical practice to the bench. Eur J Pediatr Surg 2008;18:140-9.
  • 12
    Niramis R, Watanatittan S, Anuntkosol M, et al. Quality of life of patients with Hirschsprung's disease at 5 - 20 years post pull-through operations. Eur J Pediatr Surg 2008;18:38-43.
  • 13
    Parahita IG, Makhmudi A, Gunadi. Comparison of Hirschsprung-associated enterocolitis following Soave and Duhamel procedures. J Pediatr Surg 2017.
  • 14
    Peyvasteh M, Askarpour S, Ostadian N, et al. Diagnostic accuracy of barium enema findings in Hirschsprung's disease. Arq Bras Cir Dig 2016;29:155-158.
  • 15
    Rintala RJ, Pakarinen MP. Long-term outcomes of Hirschsprung's disease. Semin Pediatr Surg 2012;21:336-43.
  • 16
    Shakya VC, Agrawal CS, Adhikary S. Initial experience with Soave's transabdominal pull-through: an observational study. Int J Surg 2010;8:225-8.
  • 17
    Wetherill C, Sutcliffe J. Hirschsprung disease and anorectal malformation. Early human development 2014;90:927-932.

  • Financial source:

    none

Publication Dates

  • Publication in this collection
    2019

History

  • Received
    17 July 2018
  • Accepted
    16 Oct 2018
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