Results of the use of botulinum toxin (Botox) in the treatment of obstructive colopathy after surgical treatment of patients with Hirschsprung's disease

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Abstract

BACKGROUND: The article presents the experience of treating patients who underwent surgery for Hirschsprung's disease and suffer from obstructive colopathy caused by achalasia of the internal anal sphincter.

AIM: Rationale for the treatment tactics of obstructive colopathy caused by hypertonicity of the internal anal sphincter after "pull-through" surgery for Hirschsprung's disease, using botulinum toxin injections.

METHODS: The study included 15 patients, 9 of them boys, with an average age of 4 years at the time of the procedure. In 80% of cases (12 patients), the botulinum toxin injection procedure was performed more than once (2 children ― three times) with an interval of 6 months. The main complaints were constipation in all cases, colostomy in 60% of cases, bloating and flatulence in 80%, recurrent Hirschsprung-associated enterocolitis in 80%. The average maximum size of the colon according to irrigography was 80 mm.

RESULTS: The analysis showed that the dynamics after botulinum toxin administration showed statistically significant changes in the occurrence of constipation (p < 0.001) and fecal incontinence (p = 0.006) and the frequency of Hirschsprung-associated enterocolitis (p = 0.001). During the analysis of pressure changes in the anal canal before and after administration of Botox, statistically significant changes in pressure values were observed at rest, with voluntary contraction and with coughing (p < 0.001).

CONCLUSION: The study shows that the administration of botulinum toxin in anal sphincter achalasia in patients undergoing Hirschsprung's disease surgery significantly facilitates the manifestations of obstructive colopathy.

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About the authors

Avazjon A. Dekhkonboev

Republican Specialized Scientific and Practical Medical Center of Pediatrics

Email: vkholostova@yandex.ru
ORCID iD: 0009-0006-5515-1449

MD, Cand. Sci. (Medicine)

Uzbekistan, Tashkent

Akmaljon M. Akhmadjonov

Republican Specialized Scientific and Practical Medical Center of Pediatrics

Email: vkholostova@yandex.ru
ORCID iD: 0009-0002-4313-1070
Uzbekistan, Tashkent

Victoria V. Kholostova

Filatov N.F. Children's City Hospital; The Russian National Research Medical University named after N.I. Pirogov

Author for correspondence.
Email: vkholostova@yandex.ru
ORCID iD: 0000-0002-3463-9799
SPIN-code: 5853-8174

MD, PhD, Assistant Professor

Russian Federation, Moscow; Moscow

Alexey N. Smirnov

Filatov N.F. Children's City Hospital; The Russian National Research Medical University named after N.I. Pirogov

Email: smirnov-dgkb13@yandex.ru
ORCID iD: 0000-0002-8646-189X
SPIN-code: 6810-0334

MD, Dr. Sci. (Medicine), Professor

Russian Federation, Moscow; Moscow

Shavkatbek T. Bozorov

Andijan State Medical Institute

Email: vkholostova@yandex.ru
ORCID iD: 0000-0001-8576-2458

MD, Cand. Sci. (Medicine), Assistant Professor

Uzbekistan, Andijan

Adham A. Gafurov

Andijan State Medical Institute

Email: vkholostova@yandex.ru
ORCID iD: 0000-0002-4388-9116

MD, Dr. Sci. (Medicine), Professor

Uzbekistan, Andijan

Jasurbek Kh. Ibragimov

Andijan State Medical Institute

Email: vkholostova@yandex.ru
ORCID iD: 0000-0002-7270-2090

MD, Cand. Sci. (Medicine), Assistant Professor

Uzbekistan, Andijan

Khasanboy Z. Toshmatov

Andijan State Medical Institute

Email: vkholostova@yandex.ru
ORCID iD: 0009-0005-1417-9974

MD, Cand. Sci. (Medicine), Assistant Professor

Uzbekistan, Andijan

Sherzod O. Toshboyev

Andijan State Medical Institute

Email: vkholostova@yandex.ru
ORCID iD: 0000-0003-1824-1454

MD, Dr. Sci. (Medicine), Professor

Uzbekistan, Andijan

Namir A. Al-Masaht

Filatov N.F. Children's City Hospital; The Russian National Research Medical University named after N.I. Pirogov

Email: vkholostova@yandex.ru
ORCID iD: 0000-0002-8050-7244

MD, Cand. Sci. (Medicine), Assistant Professor

Russian Federation, Moscow; Moscow

Pavel M. Yarustovskiy

Filatov N.F. Children's City Hospital; The Russian National Research Medical University named after N.I. Pirogov

Email: vkholostova@yandex.ru
ORCID iD: 0000-0003-4613-9705
SPIN-code: 8637-1328

MD, Cand. Sci. (Medicine), Assistant Professor

Russian Federation, Moscow; Moscow

References

  1. Roorda D, Abeln ZA, Oosterlaan J, et al. Botulinum toxin injections after surgery for Hirschsprung disease: systematic review and meta-analysis. World J Gastroenterol. 2019;25(25):3268–3280. doi: 10.3748/wjg.v25.i25.3268
  2. Langer JC, Rollins MD, Levitt M, et al.; American Pediatric Surgical Association Hirschsprung Disease Interest Group. Guidelines for the management of postoperative obstructive symptoms in children with Hirschsprung disease. Pediatr Surg Int. 2017;33(5):523–526. doi: 10.1007/s00383-017-4066-7
  3. Bokova E, Prasade N, Janumpally S, et al. State of the art bowel management for pediatric colorectal problems: hirschsprung disease. Children (Basel). 2023;10(8):1418. doi: 10.3390/children10081418
  4. Pini Prato A, Faticato MG, Mazzola C, et al. Intrasphincteric Botox injections in Hirschsprung’s disease: indications and outcome in 64 procedures over a ten-year period. Minerva Pediatr (Torino). 2023;75(4):482–489. doi: 10.23736/S2724-5276.18.05238-6
  5. Encisco EM, Lim II, Velazco CS, et al. Hirschsprung-associated enterocolitis at a referral institution: a retrospective review. J Pediatr Surg. 2023;58(8):1578–1581. doi: 10.1016/j.jpedsurg.2023.04.008
  6. Church JT, Gadepalli SK, Talishinsky T, et al. Ultrasound-guided intrasphincteric botulinum toxin injection relieves obstructive defecation due to Hirschsprung’s disease and internal anal sphincter achalasia. J Pediatr Surg. 2017;52(1):74–78. doi: 10.1016/j.jpedsurg.2016.10.023
  7. Youn JK, Han JW, Oh C, et al. Botulinum toxin injection for internal anal sphincter achalasia after pull-through surgery in Hirschsprung disease. Medicine (Baltimore). 2019;98(45):e17855. doi: 10.1097/MD.0000000000017855
  8. Patrus B, Nasr A, Langer JC, Gerstle JT. Intrasphincteric botulinum toxin decreases the rate of hospitalization for postoperative obstructive symptoms in children with Hirschsprung disease. J Pediatr Surg. 2011;46(1):184–187. doi: 10.1016/j.jpedsurg.2010.09.089
  9. Evans-Barns HM, Swannjo JB, Trajanovska M, et al. Post-operative anorectal manometry in children with Hirschsprung disease: a systematic review. Neurogastroenterol Motil. 2022;34(8):e14311. doi: 10.1111/nmo.14311
  10. Trinh TH, Nguyen NM, Lam KT, et al. Anorectal manometry findings in relation with long-term functional outcomes of the patients operated on for Hirschsprung’s disease compared to the reference-based population. Pediatr Surg Int. 2023;39(1):131. doi: 10.1007/s00383-023-05402-4

Supplementary files

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2. Fig. 1. Dependence of malnutrition on stool consistency (a) and mesenteric lymphadenitis (b).

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3. Fig. 2. Analysis of the frequency of constipation before and after treatment.

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4. Fig. 3. Analysis of the frequency of fecal incontinence before and after treatment.

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5. Fig. 4. Analysis of the frequency of Hirschsprung-associated enterocolitis before and after treatment.

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6. Fig. 5. Patient H., 3 years old, Mowat–Wilson syndrome, underwent surgery at the age of 11 months for the rectosigmoid form of Hirschsprung's disease. Suffers from recurrent Hirschsprung-associated enterocolitis, constipation: X-ray review abdominal cavity with multiple fluid levels (a); irrigography: megacolon of the left half of the colon (b); incomplete emptying of the intestine, inflammatory changes of the mucous membrane of the cobblestone type (c). The same patient aged 5 years after double administration of botulinum toxin (d).

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7. Fig. 6. Patient D. underwent surgery for Hirschprung's disease at the age of 6 months of life. Irrigography at the age of 9 years (a): megacolon of the left half, "cobblestone pavement" pattern, clinical manifestations of chronic intestinal obstruction. The study was conducted 2 years later, at the age of 11, after twice administration of botulinum toxin (b): clinical improvement, normalization of stool, while the X-ray picture showed little dynamics.

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8. Fig. 7. Patient M., 3 years old, underwent radiological intervention 1.5 years ago for the rectosigmoid form of Hirschsprung's disease: the abdominal X-ray shows a picture of colonic obstruction (a); megacolon (b): the diameter of the colon is expanded to 10 cm throughout (after the introduction of botulinum toxin, there is a pronounced positive trend, however, 2 years later a month ago, the child died against the background of a lightning-fast course of toxic colitis).

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9. Fig. 8. Dynamics of changes in the average resting pressure in the anal canal before and after treatment.

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10. Fig. 9. Dynamics of changes in the average pressure in the anal canal during coughing before and after treatment.

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11. Fig. 10. Dynamics of changes in the average resting pressure during voluntary contraction of the anal canal before and after treatment.

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