Pediatric Gastroenterology, Hepatology & Nutrition

The Korean Society of Pediatric Gastroenterology, Hepatology and Nutrition (대한소아소화기영양학회)
권호 표지
DOI QR코드

DOI QR Code

Time is Gut. Approaching Intestinal Leiomyositis: Case Presentation and Literature Review

  • Received : 2023.12.31
  • Accepted : 2024.03.24
  • Published : 2024.07.15
Download PDF
⟨ Previous Next ⟩

Abstract

T-lymphocytic intestinal leiomyositis is a rare cause of "pediatric intestinal pseudo-obstructions." Diagnosis may be difficult and requires full-thickness bowel biopsies during laparotomy or laparoscopy with possible enterostomy. Currently, immunosuppressive therapy is the only available treatment. A delay in diagnosis and therapy may negatively affect the prognosis because of ongoing fibrotic alterations; therefore, early diagnosis and consequent treatment are crucial. This review summarizes the available information on the nosology, diagnostic steps, and treatment modalities. Here, we report the youngest case of enteric leiomyositis reported in the last two decades and analyze its management by reviewing previous cases.

Keywords

References

  1. Byrne WJ, Cipel L, Euler AR, Halpin TC, Ament ME. Chronic idiopathic intestinal pseudo-obstruction syndrome in children--clinical characteristics and prognosis. J Pediatr 1977;90:585-9. https://doi.org/10.1016/S0022-3476(77)80371-5
  2. Nezelof C, Vivien E, Bigel P, Nihoul-Fekete C, Arnaud-Battandier F, Bresson JL, et al. [Idiopathic myositis of the small intestine. An unusual cause of chronic intestinal pseudo-obstruction in children]. Arch Fr Pediatr 1985;42:823-8. French.
  3. Haas S, Bindl L, Fischer HP. Autoimmune enteric leiomyositis: a rare cause of chronic intestinal pseudo-obstruction with specific morphological features. Hum Pathol 2005;36:576-80. https://doi.org/10.1016/j.humpath.2005.01.005
  4. Arista-Nasr J, Gonzalez-Romo M, Keirns C, Larriva-Sahd J. Diffuse lymphoplasmacytic infiltration of the small intestine with damage to nerve plexus. A cause of intestinal pseudo-obstruction. Arch Pathol Lab Med 1993;117:812-9.
  5. Rudolph CD, Hyman PE, Altschuler SM, Christensen J, Colletti RB, Cucchiara S, et al. Diagnosis and treatment of chronic intestinal pseudo-obstruction in children: report of consensus workshop. J Pediatr Gastroenterol Nutr 1997;24:102-12.
  6. Dudley HA, Sinclair IS, McLaren IF, McNair TJ, Newsam JE. Intestinal pseudo-obstruction. J R Coll Surg Edinb 1958;3:206-17.
  7. Thapar N, Saliakellis E, Benninga MA, Borrelli O, Curry J, Faure C, et al. Paediatric intestinal pseudo-obstruction: evidence and consensus-based recommendations from an ESPGHAN-led expert group. J Pediatr Gastroenterol Nutr 2018;66:991-1019. https://doi.org/10.1097/MPG.0000000000001982
  8. Ruuska TH, Karikoski R, Smith VV, Milla PJ. Acquired myopathic intestinal pseudo-obstruction may be due to autoimmune enteric leiomyositis. Gastroenterology 2002;122:1133-9. https://doi.org/10.1053/gast.2002.92396
  9. Ginies JL, Francois H, Joseph MG, Champion G, Coupris L, Limal JM. A curable cause of chronic idiopathic intestinal pseudo-obstruction in children: idiopathic myositis of the small intestine. J Pediatr Gastroenterol Nutr 1996;23:426-9.
  10. Uchida K, Otake K, Inoue M, Koike Y, Matsushita K, Araki T, et al. Chronic intestinal pseudo-obstruction due to lymphocytic intestinal leiomyositis: case report and literature review. Intractable Rare Dis Res 2012;1:35-9. https://doi.org/10.5582/irdr.2012.v1.1.35
  11. Oton E, Moreira V, Redondo C, Lopez-San-Roman A, Foruny JR, Plaza G, et al. Chronic intestinal pseudo-obstruction due to lymphocytic leiomyositis: is there a place for immunomodulatory therapy? Gut 2005;54:1343-4. https://doi.org/10.1136/gut.2005.071811
  12. Knowles CH, De Giorgio R, Kapur RP, Bruder E, Farrugia G, Geboes K, et al. The London Classification of gastrointestinal neuromuscular pathology: report on behalf of the Gastro 2009 International Working Group. Gut 2010;59:882-7. https://doi.org/10.1136/gut.2009.200444
  13. Smith VV, Milla PJ. Histological phenotypes of enteric smooth muscle disease causing functional intestinal obstruction in childhood. Histopathology 1997;31:112-22. https://doi.org/10.1046/j.1365-2559.1997.2250839.x
  14. De Giorgio R, Sarnelli G, Corinaldesi R, Stanghellini V. Advances in our understanding of the pathology of chronic intestinal pseudo-obstruction. Gut 2004;53:1549-52. https://doi.org/10.1136/gut.2004.043968
  15. Mann SD, Debinski HS, Kamm MA. Clinical characteristics of chronic idiopathic intestinal pseudo-obstruction in adults. Gut 1997;41:675-81. https://doi.org/10.1136/gut.41.5.675
  16. Rigby SP, Schott JM, Bliss P, Higgens CS, Kamm MA. Dilated stomach and weak muscles. Lancet 2000;356:1898.
  17. McDonald GB, Schuffler MD, Kadin ME, Tytgat GN. Intestinal pseudoobstruction caused by diffuse lymphoid infiltration of the small intestine. Gastroenterology 1985;89:882-9. https://doi.org/10.1016/0016-5085(85)90587-6
  18. Martire D, Garnier S, Sagnol S, Bourret A, Marchal S, Chauvet N, et al. Phenotypic switch of smooth muscle cells in paediatric chronic intestinal pseudo-obstruction syndrome. J Cell Mol Med 2021;25:4028-39. https://doi.org/10.1111/jcmm.16367
  19. Muto M, Matsufuji H, Tomomasa T, Nakajima A, Kawahara H, Ida S, et al. Pediatric chronic intestinal pseudo-obstruction is a rare, serious, and intractable disease: a report of a nationwide survey in Japan. J Pediatr Surg 2014;49:1799-803. https://doi.org/10.1016/j.jpedsurg.2014.09.025
  20. Goulet O, Sauvat F, Jan D. Surgery for pediatric patients with chronic intestinal pseudo-obstruction syndrome. J Pediatr Gastroenterol Nutr 2005;41 Suppl 1:S66-8. https://doi.org/10.1097/01.scs.0000180312.55417.8e
  21. Di Lorenzo C. Pseudo-obstruction: current approaches. Gastroenterology 1999;116:980-7. https://doi.org/10.1016/S0016-5085(99)70082-X
  22. Valli PV, Pohl D, Fried M, Caduff R, Bauerfeind P. Diagnostic use of endoscopic full-thickness wall resection (eFTR)-a novel minimally invasive technique for colonic tissue sampling in patients with severe gastrointestinal motility disorders. Neurogastroenterol Motil 2018;30:e13153.
  23. Smith VV, Gregson N, Foggensteiner L, Neale G, Milla PJ. Acquired intestinal aganglionosis and circulating autoantibodies without neoplasia or other neural involvement. Gastroenterology 1997;112:1366-71. https://doi.org/10.1016/S0016-5085(97)70151-3
  24. Pakarinen MP, Kurvinen A, Koivusalo AI, Ruuska T, Makisalo H, Jalanko H, et al. Surgical treatment and outcomes of severe pediatric intestinal motility disorders requiring parenteral nutrition. J Pediatr Surg 2013;48:333-8. https://doi.org/10.1016/j.jpedsurg.2012.11.010
  25. Di Lorenzo C, Lucanto C, Flores AF, Idries S, Hyman PE. Effect of sequential erythromycin and octreotide on antroduodenal manometry. J Pediatr Gastroenterol Nutr 1999;29:293-6.
  26. Hashizume N, Yagi M, Ushijima K, Seki Y, Fukahori S, Muto M, et al. Pharmacotherapy for pediatric chronic intestinal pseudo-obstruction: nationwide survey in Japan. Pediatr Int 2017;59:467-72. https://doi.org/10.1111/ped.13201
  27. Pironi L, Goulet O, Buchman A, Messing B, Gabe S, Candusso M, et al. Outcome on home parenteral nutrition for benign intestinal failure: a review of the literature and benchmarking with the European prospective survey of ESPEN. Clin Nutr 2012;31:831-45. https://doi.org/10.1016/j.clnu.2012.05.004
  28. Ko D, Yang HB, Youn J, Kim HY. Clinical outcomes of pediatric chronic intestinal pseudo-obstruction. J Clin Med 2021;10:2376.
  29. Koletzko B, Goulet O, Hunt J, Krohn K, Shamir R; Parenteral Nutrition Guidelines Working Group. European Society for Clinical Nutrition and Metabolism; European Society of Paediatric Gastroenterology, Hepatology and Nutrition (ESPGHAN); European Society of Paediatric Research (ESPR). Guidelines on paediatric parenteral nutrition of the European Society of Paediatric Gastroenterology, Hepatology and Nutrition (ESPGHAN) and the European Society for Clinical Nutrition and Metabolism (ESPEN), supported by the European Society of Paediatric Research (ESPR). J Pediatr Gastroenterol Nutr 2005;41 Suppl 2:S1-87. https://doi.org/10.1002/j.1536-4801.2005.tb00011.x
  30. Bogdanos DP, Choudhuri K, Vergani D. Molecular mimicry and autoimmune liver disease: virtuous intentions, malign consequences. Liver 2001;21:225-32. https://doi.org/10.1034/j.1600-0676.2001.021004225.x
  31. Kagnoff MF, Paterson YJ, Kumar PJ, Kasarda DD, Carbone FR, Unsworth DJ, et al. Evidence for the role of a human intestinal adenovirus in the pathogenesis of coeliac disease. Gut 1987;28:995-1001. https://doi.org/10.1136/gut.28.8.995
  32. Stanghellini V, Cogliandro RF, De Giorgio R, Barbara G, Cremon C, Antonucci A, et al. Natural history of intestinal failure induced by chronic idiopathic intestinal pseudo-obstruction. Transplant Proc 2010;42:15-8. https://doi.org/10.1016/j.transproceed.2009.12.017