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Clonazepam Treatment of Pathologic Aerophagia in Children with Mental Retardation

  • Lee, Ga-Hyun (Department of Pediatrics, Keimyung University School of Medicine) ;
  • Jang, Hyo-Jeong (Department of Pediatrics, Seoul National University College of Medicine) ;
  • Hwang, Jin-Bok (Department of Pediatrics, Keimyung University School of Medicine)
  • Received : 2014.08.27
  • Accepted : 2014.09.29
  • Published : 2014.12.30

Abstract

Purpose: Pathologic aerophagia (PA) may lead to bowel perforation or volvulus in mentally retarded patients. The authors investigated the effects of clonazepam on the management of PA in children with severe to profound mental retardation (MR). Methods: This study was undertaken as a retrospective case analysis of 21 PA patients with MR who were followed for over 12 months and diagnosed as having PA. Patients were assigned to two management groups, that is, to a clonazepam randomized open-labeled, treatment group or a reassurance group. The following were recorded and analyzed; age, response, remission rate to clonazepam treatment, and the side effect of clonazepam. It was defined positive response (response+) as being symptom-free for a whole week within 1 month of commencing treatment and remission(+) as being symptom-free for a whole month within 6 months of treatment. Results: The average age of the 21 PA children with MR was 10 years and 13 patients were female. Symptom duration before diagnosis of PA was 7 months. Clinical features of the clonazepam-trial group (n=11) and the reassurance group (n=10) were non-significantly different. Response(+) was achieved by 2 patients (18.2%) in the clonazepamtrial group and by no patient in the reassurance group. Remission(+) was achieved by 6 patients (54.5%) in the clonazepam-trial group and by one patient (10%) in the reassurance group (p=0.040). Conclusion: When PA children with MR with severe bowel distention are considered for surgical treatment to prevent acute abdomen, a trial of clonazepam could be recommended.

Keywords

References

  1. Hwang JB, Choi WJ, Kim JS, Lee SY, Jung CH, Lee YH, et al. Clinical features of pathologic childhood aerophagia: early recognition and essential diagnostic criteria. J Pediatr Gastroenterol Nutr 2005;41:612-6. https://doi.org/10.1097/01.mpg.0000179856.68968.e0
  2. Lekkas CN, Lentino W. Symptom-producing interposition of the colon. Clinical syndrome in mentally deficient adults. JAMA 1978;240:747-50. https://doi.org/10.1001/jama.1978.03290080037020
  3. Trillis F Jr, Gauderer MW, Ponsky JL, Morrison SC. Transverse colon volvulus in a child with pathologic aerophagia. J Pediatr Surg 1986;21:966-8. https://doi.org/10.1016/S0022-3468(86)80108-7
  4. van der Kolk MB, Bender MH, Goris RJ. Acute abdomen in mentally retarded patients: role of aerophagia. Report of nine cases. Eur J Surg 1999;165:507-11. https://doi.org/10.1080/110241599750006802
  5. Komuro H, Matoba K, Kaneko M. Laparoscopic gastropexy for chronic gastric volvulus complicated by pathologic aerophagia in a boy. Pediatr Int 2005;47:701-3. https://doi.org/10.1111/j.1442-200x.2005.02128.x
  6. Basaran UN, Inan M, Aksu B, Ceylan T. Colon perforation due to pathologic aerophagia in an intellectually disabled child. J Paediatr Child Health 2007;43:710-2. https://doi.org/10.1111/j.1440-1754.2007.01190.x
  7. Lee EJ, Song AR, Choi E, Hwang JB, Oh HJ, Lee YH. A case of pathologic aerophagia with mental retardation managed by percutaneous endoscopic gastrostomy. Korean J Pediatr Gastroenterol Nutr 2000;3:93-7.
  8. Fukuzawa H, Urushihara N, Fukumoto K, Sugiyama A, Mitsunaga M, Watanabe K, et al. Esophagogastric separation and abdominal esophagostomy via jejunal interposition: a new operation for extreme forms of pathologic aerophagia. J Pediatr Surg 2011;46:2035-7. https://doi.org/10.1016/j.jpedsurg.2011.05.024
  9. Hwang JB, Kim JS, Ahn BH, Jung CH, Lee YH, Kam S. Clonazepam treatment of pathologic childhood aerophagia with psychological stresses. J Korean Med Sci 2007;22:205-8. https://doi.org/10.3346/jkms.2007.22.2.205
  10. Frye RE, Hait EJ. Air swallowing caused recurrent ileus in Tourette's syndrome. Pediatrics 2006;117:e1249-52. https://doi.org/10.1542/peds.2005-2914
  11. Weil RS, Cavanna AE, Willoughby JM, Robertson MM. Air swallowing as a tic. J Psychosom Res 2008;65:497-500. https://doi.org/10.1016/j.jpsychores.2008.04.001
  12. Holburn CS. Aerophagia: an uncommon form of self-injury. Am J Ment Defic 1986;91:201-3.
  13. Hemmink GJ, Weusten BL, Bredenoord AJ, Timmer R, Smout AJ. Aerophagia: excessive air swallowing demonstrated by esophageal impedance monitoring. Clin Gastroenterol Hepatol 2009;7:1127-9. https://doi.org/10.1016/j.cgh.2009.06.029
  14. Halb C, Pomerleau M, Faure C. Multichannel intraesophageal impedance pattern of children with aerophagia. Neurogastroenterol Motil 2014;26:1010-4. https://doi.org/10.1111/nmo.12355
  15. Blondeau K, Boecxstaens V, Rommel N, Farre R, Depeyper S, Holvoet L, et al. Baclofen improves symptoms and reduces postprandial flow events in patients with rumination and supragastric belching. Clin Gastroenterol Hepatol 2012;10:379-84. https://doi.org/10.1016/j.cgh.2011.10.042
  16. Singer HS, Wendlandt J, Krieger M, Giuliano J. Baclofen treatment in Tourette syndrome: a double-blind, placebo-controlled, crossover trial. Neurology 2001;56:599-604. https://doi.org/10.1212/WNL.56.5.599
  17. Gonce M, Barbeau A. Seven cases of Gilles de la Tourette's syndrome: partial relief with clonazepam: a pilot study. Can J Neurol Sci 1977;4:279-83. https://doi.org/10.1017/S0317167100025129
  18. Merikangas JR, Merikangas KR, Kopp U, Hanin I. Blood choline and response to clonazepam and haloperidol in Tourette's syndrome. Acta Psychiatr Scand 1985;72:395-9. https://doi.org/10.1111/j.1600-0447.1985.tb02628.x
  19. Goetz CG. Clonidine and clonazepam in Tourette syndrome. Adv Neurol 1992;58:245-51.
  20. Steingard RJ, Goldberg M, Lee D, DeMaso DR. Adjunctive clonazepam treatment of tic symptoms in children with comorbid tic disorders and ADHD. J Am Acad Child Adolesc Psychiatry 1994;33:394-9. https://doi.org/10.1097/00004583-199403000-00014
  21. Singer HS. Neurobiology of Tourette syndrome. Neurol Clin 1997;15:357-79. https://doi.org/10.1016/S0733-8619(05)70318-2

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