Esophageal High-Resolution Manometry for Diagnosing the Severity of the Chronic Intestinal Pseudo-Obstruction: A Case Se
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ORIGINAL ARTICLE
Esophageal High‑Resolution Manometry for Diagnosing the Severity of the Chronic Intestinal Pseudo‑Obstruction: A Case Series Hiroki Sato1 · Kenya Kamimura1 · Hideaki Matsui2 · Takashi Owaki1 · Shinichi Morita1 · Yuto Tanaka1 · Natsuki Ishikawa1 · Yoshifumi Shimada3 · Junji Yokoyama1 · Toshifumi Wakai3 · Shuji Terai1 Received: 23 August 2020 / Accepted: 28 October 2020 © Springer Science+Business Media, LLC, part of Springer Nature 2020
Abstract Background Chronic intestinal pseudo-obstruction (CIPO) is a severe and refractory intestinal motility disorder. However, due to its rarity and difficult histological investigation, its pathophysiology has not been characterized. Aim Therefore, in this study, we aimed to determine the role of esophageal high-resolution manometry (HRM) in CIPO and the histological and clinical characteristics of the disease. Methods Patients with CIPO were analyzed for clinical characteristics; histological findings; and clinical courses after therapeutic intervention. In addition, HRM was performed to determine the esophageal involvement. Results Eleven patients were diagnosed with CIPO, and five required the long period of parenteral nutrition showing impaired esophageal motility including achalasia and absent contractility diagnosed with HRM. The four of these five cases showed acute onset of the CIPO following the triggering events of pregnancy, appendicitis, and surgery. In contrast, other six patients with normal or Jackhammer esophagus on HRM had moderate severity of CIPO with gradual onset. The histological analyses revealed that the loss of the intestinal neural ganglion cells and layers by inflammation, destruction, and atrophy are related to the severity of the clinical course of the disease and esophageal HRM findings of achalasia and absent contractility. Conclusions HRM may be useful to diagnose the severity of the clinical course and to determine the therapeutic options for CIPO. Keywords Chronic intestinal pseudo-obstruction · High-resolution manometry · Auerbach’s plexus · α-Synuclein · Achalasia
Introduction
Electronic supplementary material The online version of this article (https://doi.org/10.1007/s10620-020-06701-9) contains supplementary material, which is available to authorized users. * Kenya Kamimura kenya‑[email protected]‑u.ac.jp 1
Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, 1‑757 Asahimachi‑dori, Niigata 951‑8510, Japan
2
Department of Neuroscience of Disease, Brain Research Institute, Niigata University, Niigata 951‑8585, Japan
3
Division of Digestive and General Surgery, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 951‑8510, Japan
Chronic intestinal pseudo-obstruction (CIPO) is a rare and refractory motility disorder of the gastrointestinal (GI) tract. The annual incidence and prevalence of CIPO in Japan were estimated at 0.225 and 0.900 per 100,000, respectively [1]. In pathophysiology, GI tract passage is impaired, des
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