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Z Gastroenterol 2021; 59(04): 326-330
DOI: 10.1055/a-1310-4500
Kasuistik

Postinfectious T-lymphocytic enteral leiomyositis as a rare cause of chronic intestinal pseudoobstruction

Postinfektiöse T-lymphozytäre enterale Leiomyositis als seltene Ursache für eine chronische intestinale Pseudoobstruktion
Bismark Osei Sarfo
1   Schön Klinik Hamburg-Eilbek, Hamburg, Germany
,
Hakan Kopdag
1   Schön Klinik Hamburg-Eilbek, Hamburg, Germany
,
Marian-Christopher Pott 
2   Inselspital Universitätsspital Bern, Switzerland
,
Lars Stiedenroth
1   Schön Klinik Hamburg-Eilbek, Hamburg, Germany
,
Ulf Nahrstedt
3   General Surgery, Schön Klinik Hamburg-Eilbek, Hamburg, Germany
,
Hansjörg Schäfer
4   Pathology, Universitätsklinikum Hamburg-Eppendorf, Hamburg, Germany
,
Götz von Wichert
5   Innere Medizin, Schön Klinik Hamburg Eilbek, Hamburg, Germany
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Abstract

T-lymphocytic enteral leiomyositis (T-lel) is a rare disorder causing chronic intestinal pseudo-obstruction (CIPO), with cases predominantly being reported in the field of veterinary and pediatric medicine. Here, we present a case of T-lel-associated CIPO in an adult female, who initially presented with a paralytic ileus 2 weeks after a common gastroenteritis. The histological diagnosis was established through full-thickness bowel biopsy, exhibiting a dense lymphocytic infiltrate in the lamina muscularis of the intestinal wall. This case shows that T-lel can be a cause of chronic intestinal pseudo-obstruction not only in children but also in adults. A subsequent induction of an immunosuppressive therapy with steroids, azathioprine, and ultimately TNF-alpha-inhibiting antibodies led to a slow recovery and stable disease.

Zusammenfassung

Die T-lymphozytäre enterale Leiomyositis (T-lel) ist eine seltene Erkrankung, die zu einer chronischen intestinalen Pseudoobstruktion (CIPO) führen kann. Die meisten dieser seltenen Fälle sind bei Kindern und in der Veterinärmedizin beschrieben. Hier berichten wir über den Fall einer 42-jährigen Patientin, die sich 2 Wochen nach gastroenterischen Beschwerden mit der Klinik eines paralytischen Ileus vorstellte. Nachdem eine weitreichende Diagnostik mittels laboranalytischer, endoskopischer und radiologischer Verfahren keine wegweisenden Befunde erbrachte, erfolgte die Durchführung einer Dünndarm-Vollwandbiopsie, woraufhin histologisch die Diagnose einer T-lymphozytären Leiomyositis gestellt werden konnte. Eine immunsuppressive Therapie mit initial Prednisolon und Azathioprin wurde aufgrund unzureichenden klinischen Ansprechens auf Adalimumab umgestellt, woraufhin letztlich eine Stabilisierung der Erkrankung und eine Verbesserung der Symptomatik erreicht werden konnten.

Key words

gastrointestinal motility - malassimilation - infectious colitis

Schlüsselwörter

gastrointestinale Motilität - Malassimilation - infektiöse Colitis


Publication History

Received: 23 June 2020

Accepted: 08 November 2020

Article published online:
12 April 2021

© 2021. Thieme. All rights reserved.

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  • Literatur

  • 1 Ruuska TH, Karikoski R, Smith VV. et al Acquired myopathic intestinal pseudo-obstruction may be due to autoimmune enteric leiomyositis. Gastroenterology 2002; 122: 1133-1139 . doi:10.1053/gast.2002.92396
    CrossrefPubMedGoogle Scholar
  • 2 Nezelof C, Vivien E, Bigel P. et al [Idiopathic myositis of the small intestine. An unusual cause of chronic intestinal pseudo-obstruction in children]. Archives francaises de pediatrie 1985; 42: 823-828
    PubMedGoogle Scholar
  • 3 Smith VV, Milla PJ. Histological phenotypes of enteric smooth muscle disease causing functional intestinal obstruction in childhood. Histopathology 1997; 31: 112-122 . doi:10.1046/j.1365-2559.1997.2250839.x
    CrossrefPubMedGoogle Scholar
  • 4 Haas S, Bindl L, Fischer HP. Autoimmune enteric leiomyositis: a rare cause of chronic intestinal pseudo-obstruction with specific morphological features. Human pathology 2005; 36: 576-580 . doi:10.1016/j.humpath.2005.01.005
    CrossrefPubMedGoogle Scholar
  • 5 Oton E, Moreira V, Redondo C. et al Chronic intestinal pseudo-obstruction due to lymphocytic leiomyositis: is there a place for immunomodulatory therapy?. Gut 2005; 54: 1343-1344 . doi:10.1136/gut.2005.071811
    CrossrefPubMedGoogle Scholar
  • 6 Uchida K, Otake K, Inoue M. et al Chronic intestinal pseudo-obstruction due to lymphocytic intestinal leiomyositis: Case report and literature review. Intractable & rare diseases research 2012; 1: 35-39 . doi:10.5582/irdr.2012.v1.1.35
    PubMedGoogle Scholar
  • 7 Keller J, Layer P. [Motility disorders of the small intestine]. Der Internist 2015; 56: 631-637 . doi:10.1007/s00108-014-3606-7
    CrossrefPubMedGoogle Scholar
  • 8 De Giorgio R, Cogliandro RF, Barbara G. et al Chronic intestinal pseudo-obstruction: clinical features, diagnosis, and therapy. Gastroenterology clinics of North America 2011; 40: 787-807 . doi:10.1016/j.gtc.2011.09.005
    CrossrefPubMedGoogle Scholar
  • 9 Zenzeri L, Tambucci R, Quitadamo P. et al Update on chronic intestinal pseudo-obstruction. Current opinion in gastroenterology 2020; 36: 230-237 . doi:10.1097/MOG.0000000000000630
    CrossrefPubMedGoogle Scholar
  • 10 Coulie B, Camilleri M. Intestinal pseudo-obstruction. Annual review of medicine 1999; 50: 37-55 . doi:10.1146/annurev.med.50.1.37
    CrossrefPubMedGoogle Scholar
  • 11 Di Lorenzo C. Pseudo-obstruction: current approaches. Gastroenterology 1999; 116: 980-987 . doi:10.1016/s0016-5085(99)70082-x
    CrossrefPubMedGoogle Scholar
  • 12 Downes TJ, Cheruvu MS, Karunaratne TB. et al Pathophysiology, Diagnosis, and Management of Chronic Intestinal Pseudo-Obstruction. Journal of clinical gastroenterology 2018; 52: 477-489 . doi:10.1097/MCG.0000000000001047
    CrossrefPubMedGoogle Scholar
  • 13 Antonucci A, Fronzoni L, Cogliandro L. et al Chronic intestinal pseudo-obstruction. World journal of gastroenterology 2008; 14: 2953-2961 . doi:10.3748/wjg.14.2953
    CrossrefPubMedGoogle Scholar
  • 14 Gabbard SL, Lacy BE. Chronic intestinal pseudo-obstruction. Nutrition in clinical practice: official publication of the American Society for Parenteral and Enteral Nutrition 2013; 28: 307-316 . doi:10.1177/0884533613485904
    CrossrefPubMedGoogle Scholar
  • 15 Sutton DH, Harrell SP, Wo JM. Diagnosis and management of adult patients with chronic intestinal pseudoobstruction. Nutrition in clinical practice: official publication of the American Society for Parenteral and Enteral Nutrition 2006; 21: 16-22 . doi:10.1177/011542650602100116
    CrossrefPubMedGoogle Scholar
  • 16 Thapar N, Saliakellis E, Benninga MA. et al Paediatric Intestinal Pseudo-obstruction: Evidence and Consensus-based Recommendations From an ESPGHAN-Led Expert Group. Journal of pediatric gastroenterology and nutrition 2018; 66: 991-1019 . doi:10.1097/MPG.0000000000001982
    CrossrefPubMedGoogle Scholar
  • 17 Selgrad M, De Giorgio R, Fini L. et al JC virus infects the enteric glia of patients with chronic idiopathic intestinal pseudo-obstruction. Gut 2009; 58: 25-32 . doi:10.1136/gut.2008.152512
    CrossrefPubMedGoogle Scholar
  • 18 Stanghellini V, Cogliandro RF, De Giorgio R. et al Natural history of chronic idiopathic intestinal pseudo-obstruction in adults: a single center study. Clinical gastroenterology and hepatology: the official clinical practice journal of the American Gastroenterological Association 2005; 3: 449-458 . doi:10.1016/s1542-3565(04)00675-5
    CrossrefPubMedGoogle Scholar
  • 19 Zacuto AC, Pesavento PA, Hill S. et al Intestinal Leiomyositis: A Cause of Chronic Intestinal Pseudo-Obstruction in 6 Dogs. Journal of veterinary internal medicine 2016; 30: 132-140 . doi:10.1111/jvim.13652
    CrossrefPubMedGoogle Scholar