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Zhao Q, Sun X, Liu K, Peng Y, Jin D, Shen W, Wang R. Correlation between capsule endoscopy classification and CT lymphangiography of primary intestinal lymphangiectasia. Clin Radiol 2023; 78:219-226. [PMID: 36509551 DOI: 10.1016/j.crad.2022.10.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/15/2022] [Revised: 09/21/2022] [Accepted: 10/05/2022] [Indexed: 12/13/2022]
Abstract
AIM To investigate the correlation between capsule endoscopy (CE) classification of primary intestinal lymphangiectasia (PIL) and computed tomography (CT) lymphangiography (CTL). MATERIALS AND METHODS A total of 52 patients with diagnosed PIL were enrolled. All patients were examined using CTL and small intestinal CE before surgery. CE assessments included the morphology, scope, colour, and size of lesions. CTL assessments included intestinal wall, lymphatic vessel dilatation, lymph fluid reflux, and lymphatic fistula. Patients were divided into three groups according to type diagnosed by CE, and the CTL characteristics were analysed among the groups. RESULTS CE showed 15 patients with type I, 27 with II, and 10 with type III. Intestinal wall thickening was observed in 15 type I, 21 type II, and seven type III. Pericardial effusion was observed in only three type I patients; the difference among types was statistically significant (p=0.02). Abnormal contrast agent distribution in the intestinal wall and mesentery was observed in 15 type II patients, and the difference was significantly greater than that of types I and III (p=0.02). Abnormal contrast agent distribution in the abdominal cavity was observed in 12 type II, and the difference was statistically significant (p=0.03). CONCLUSION The CE PIL classification reflects the extent and scope of intestinal mucosa lesions; CTL more systematically demonstrates abnormal lymphatic vessels or reflux, and its manifestations of PIL are related to the CE classification. The combination of CTL with CE is useful for accurately evaluating PIL, and provides guidance for preoperative assessment and treatment management of PIL patients.
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Affiliation(s)
- Q Zhao
- Department of Radiology, Peking University Ninth School of Clinical Medicine, Beijing Shijitan Hospital, Capital Medical University, Beijing, China
| | - X Sun
- Department of Radiology, Peking University Ninth School of Clinical Medicine, Beijing Shijitan Hospital, Capital Medical University, Beijing, China
| | - K Liu
- Department of Gastroenterology, Peking University Ninth School of Clinical Medicine, Beijing Shijitan Hospital, Capital Medical University, Beijing, China
| | - Y Peng
- Beijing Jiaotong University, China
| | - D Jin
- Peking University Third Hospital, China
| | - W Shen
- Department of Lymph Surgery, Peking University Ninth School of Clinical Medicine, Beijing Shijitan Hospital, Capital Medical University, Beijing, China
| | - R Wang
- Department of Radiology, Peking University Ninth School of Clinical Medicine, Beijing Shijitan Hospital, Capital Medical University, Beijing, China.
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Meng MM, Liu KL, Xue XY, Hao K, Dong J, Yu CK, Liu H, Wang CH, Su H, Lin W, Jiang GJ, Wei N, Wang RG, Shen WB, Wu J. Endoscopic classification and pathological features of primary intestinal lymphangiectasia. World J Gastroenterol 2022; 28:2482-2493. [PMID: 35979262 PMCID: PMC9258282 DOI: 10.3748/wjg.v28.i22.2482] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/02/2021] [Revised: 12/09/2021] [Accepted: 04/28/2022] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND The appearance of the intestinal mucosa during endoscopy varies among patients with primary intestinal lymphangiectasia (PIL). AIM To classify the endoscopic features of the intestinal mucosa in PIL under endoscopy, combine the patients' imaging and pathological characteristics of the patients, and explain their causes. METHODS We retrospectively analyzed the endoscopic images of 123 patients with PIL who were treated at the hospital between January 1, 2007 and December 31, 2018. We compared and analyzed all endoscopic images, classified them into four types according to the endoscopic features of the intestinal mucosa, and analyzed the post-lymphographic computed tomography (PLCT) and pathological characteristics of each type. RESULTS According to the endoscopic features of PIL in 123 patients observed during endoscopy, they were classified into four types: nodular-type, granular-type, vesicular-type, and edematous-type. PLCT showed diffuse thickening of the small intestinal wall, and no contrast agent was seen in the small intestinal wall and mesentery in the patients with nodular and granular types. Contrast agent was scattered in the small intestinal wall and mesentery in the patients with vesicular and edematous types. Analysis of the small intestinal mucosal pathology revealed that nodular-type and granular-type lymphangiectasia involved the small intestine mucosa in four layers, whereas ectasia of the vesicular- and edematous-type lymphatic vessels largely involved the lamina propria mucosae, submucosae, and muscular layers. CONCLUSION Endoscopic classification, combined with the patients' clinical manifestations and pathological examination results, is significant and very useful to clinicians when scoping patients with suspected PIL.
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Affiliation(s)
- Ming-Ming Meng
- Department of Gastroenterology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Kui-Liang Liu
- Department of Gastroenterology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Xin-Ying Xue
- Department of Respiratory and Critical Care Medicine, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Kun Hao
- Departments of Lymphatic Surgery, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Jian Dong
- Departments of Radiology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Chun-Kai Yu
- Departments of Pathology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Hong Liu
- Department of Gastroenterology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Cang-Hai Wang
- Department of Gastroenterology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Hui Su
- Department of Gastroenterology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Wu Lin
- Department of Gastroenterology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Guo-Jun Jiang
- Department of Gastroenterology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Nan Wei
- Department of Gastroenterology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Ren-Gui Wang
- Departments of Radiology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Wen-Bin Shen
- Departments of Lymphatic Surgery, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
| | - Jing Wu
- Department of Gastroenterology, Beijing Shijitan Hospital, Capital Medical University, Beijing 100038, China
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Yasuda T, Sakurazawa N, Kuge K, Omori J, Arai H, Kakinuma D, Watanabe M, Suzuki H, Iwakiri K, Yoshida H. Protein-losing enteropathy caused by a jejunal ulcer after an internal hernia in Petersen's space: A case report. World J Clin Cases 2022; 10:323-330. [PMID: 35071535 PMCID: PMC8727264 DOI: 10.12998/wjcc.v10.i1.323] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/21/2021] [Revised: 07/29/2021] [Accepted: 11/29/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND The incidence of internal hernias has recently increased in concordance with the popularization of laparoscopic surgery. Of particular concern are internal hernias occurring in Petersen's space, a space that is surgically created after treatment for gastric cancer and obesity. These hernias cause devastating sequelae, such as massive intestinal necrosis, fatal Roux limb necrosis, and superior mesenteric vein thrombus. In addition, protein-losing enteropathy (PLE) is a rare syndrome involving gastrointestinal protein loss, although its relationship with internal Petersen’s hernias remains unknown.
CASE SUMMARY A 75-year-old man with a history of laparotomy for early gastric cancer developed Petersen's hernia 1 year and 5 mo after surgery. He was successfully treated by reducing the incarcerated small intestine and closure of Petersen’s defect without resection of the small intestine. Approximately 3 mo after his surgery for Petersen’s hernia, he developed bilateral leg edema and hypoalbuminemia. He was diagnosed with PLE with an alpha-1 antitrypsin clearance of 733 mL/24 h. Double-balloon enteroscopy revealed extensive jejunal ulceration as the etiology, and it facilitated minimum bowel resection. Pathological analysis showed extensive jejunal ulceration and collagen hyperplasia with nonspecific inflammation of all layers without lymphangiectasia, lymphoma, or vascular abnormalities. His postoperative course was unremarkable, and his bilateral leg edema and hypoalbuminemia improved after 1 mo. There was no relapse over the 5-year follow-up period.
CONCLUSION PLE and extensive jejunal ulceration may occur after Petersen's hernia. Double-balloon enteroscopy helps identify and resect these lesions.
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Affiliation(s)
- Tomohiko Yasuda
- Department of Gastrointestinal Surgery, Nippon Medical School Chiba Hokusoh Hospital, Chiba 270-1694, Japan
| | - Nobuyuki Sakurazawa
- Department of Gastrointestinal and Hepato-Biliary-Pancreatic Surgery, Nippon Medical School, Tokyo 113-8603, Japan
| | - Komei Kuge
- Department of Gastrointestinal Surgery, Nippon Medical School Chiba Hokusoh Hospital, Chiba 270-1694, Japan
| | - Jun Omori
- Department of Gastroenterology, Nippon Medical School, Tokyo 113-8603, Japan
| | - Hiroki Arai
- Department of Gastrointestinal Surgery, Nippon Medical School Chiba Hokusoh Hospital, Chiba 270-1694, Japan
| | - Daisuke Kakinuma
- Department of Gastrointestinal Surgery, Nippon Medical School Chiba Hokusoh Hospital, Chiba 270-1694, Japan
| | - Masanori Watanabe
- Department of Gastrointestinal Surgery, Nippon Medical School Chiba Hokusoh Hospital, Chiba 270-1694, Japan
| | - Hideyuki Suzuki
- Department of Gastrointestinal Surgery, Nippon Medical School Chiba Hokusoh Hospital, Chiba 270-1694, Japan
| | - Katsuhiko Iwakiri
- Department of Gastroenterology, Nippon Medical School, Tokyo 113-8603, Japan
| | - Hiroshi Yoshida
- Department of Gastrointestinal and Hepato-Biliary-Pancreatic Surgery, Nippon Medical School, Tokyo 113-8603, Japan
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de Carvalho JF, Lerner A, Gonçalves CM, Shoenfeld Y. Sjögren syndrome associated with protein-losing enteropathy: case-based review. Clin Rheumatol 2021; 40:2491-2497. [PMID: 33145631 DOI: 10.1007/s10067-020-05487-5] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/28/2020] [Revised: 10/25/2020] [Accepted: 10/27/2020] [Indexed: 10/23/2022]
Abstract
The association between Sjögren's syndrome (SS) and protein-losing enteropathy (PLE) was scarcly reported. To analyze the clinical, therapeutic, and outcome characteristics of patients with SS and PLE and also to delineate the potential mechanisms and pathways connecting the gut to SS targeted organ's pathology. Systematic screening was conducted using PubMed/MEDLINE, LILACS, SciELO, Web of Science, and Cochrane, dating 1980 to 2020. SS and PLE were the key words. Eighteen patients with SS and PLE were summarized. The patient's ages ranged between 20 and 88 years, and only 4 were males. Primary SS was observed in most cases. Anti-Ro was detected in 100% of the cases while anti-La was reported in 64% of them. The clinical manifestations were protein loss, edema of the lower limbs, pleural effusion, ascites, facial edema, anasarca, diarrhea, and weight loss. Among these clinical manifestations, edema of the lower limbs was the most severe. Albumin concentration was 0.9-3.4 g/dL which increased to 2.8-4.3 g/dL after treatment. Small bowel biopsy was performed in all of the cases. Concerning the therapy, all the patients received systemic glucocorticoids. All of them improved. The period of onset of improvement ranged from 3 weeks to 36 months (an average of 3 months). The early diagnosis and appropriate therapy of PLE in patients with anti-Ro positive SS and who present edema, anasarca, or hypoalbuminemia is vital for a beneficial outcome. An excellent clinical improvement in all the cases was observed when treated early enough by cortico-therapy, thus preventing patient's deterioration, complications, and reducing morbidity and potential mortality.
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Affiliation(s)
- Jozélio Freire de Carvalho
- Institute for Health Sciences from Federal University of Bahia, Rua das Violetas, 42, ap. 502, Pituba, Salvador, Bahia, Brazil.
| | - Aaron Lerner
- Chaim Sheba Medical Center, The Zabludowicz Research Center for Autoimmune Diseases, Tel Hashomer, Israel
| | | | - Yehuda Shoenfeld
- Chaim Sheba Medical Center, The Zabludowicz Research Center for Autoimmune Diseases, Tel Hashomer, Israel
- Saint Petersburg State University, St. Petersburg, Russia
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5
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Abstract
PURPOSE OF REVIEW The present review offers its readers a practical overview of protein-losing enteropathy, particularly with regard to diagnostic and therapeutic approaches. The aim is to support clinicians in their daily practice with a practical tool to deal with protein-losing enteropathy. RECENT FINDINGS The literature covering protein-losing enteropathy does not appear to be quite recent and also guidelines are scanty. The main innovations during the last decade probably regard the introduction of enteroscopic techniques in the diagnostic flowchart. The use of video-capsule and device-assisted enteroscopy has enabled the direct exploration of the small bowel and the identification of the damage causing the loss of proteins from the gastrointestinal tract. Other innovations are to do with the therapies of the disorder underlying protein-losing enteropathy, although the support with nutritional supplementation are the direct remedies to tackle the protein loss. SUMMARY Protein-losing enteropathy represents an important clinical aspect of different gastrointestinal and extra-intestinal diseases. An established flowchart is still unavailable, but the use of enteroscopy has deeply changed the modern diagnostic approach. Nutritional support and therapy of the underlying disease are pivotal to patients' management.
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Cappell MS, Edhi A, Amin M. Case report of primary intestinal lymphangiectasia diagnosed in an octogenarian by ileal intubation and by push enteroscopy after missed diagnosis by standard colonoscopy and EGD. Medicine (Baltimore) 2018; 97:e9649. [PMID: 29505002 PMCID: PMC5779771 DOI: 10.1097/md.0000000000009649] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/27/2017] [Accepted: 12/27/2017] [Indexed: 12/17/2022] Open
Abstract
RATIONALE Primary intestinal lymphangiectasia (PIL) is a rare, presumably congenital lesion that is usually diagnosed in patients < 3 years old, is rarely first diagnosed in adulthood, and when first diagnosed in adulthood typically presents with symptoms for many years. Although PIL is often identified by endoscopic abnormalities, it must be emphasized that the jejunoileum/distal duodenum must be intubated for diagnosis because the lesions are present in these regions. This work demonstrates that 1)-PIL can occur in an octogenarian; 2)-shows that the characteristic endoscopic findings are not found at colonoscopy without terminal ileal intubation; and 3)-may be missed at standard EGD without distal duodenal intubation. DIAGNOSES A patient initially presented at age 83 with symptoms of watery diarrhea, abdominal distention, 5-Kg-weight-gain, and weakness for one month, and had typical clinical findings of PIL including chylous ascites, pleural effusions, bilateral pitting leg edema, hypoalbuminemia, borderline lymphopenia, hypovitaminosis-D, and hypocalcemia. Protein-losing-enteropathy was demonstrated by positive stool tests for alpha-1-antitrypsin. Standard colonoscopy revealed no significant lesions, but terminal ileal intubation during colonoscopy demonstrated creamy-white, punctate, mucosal lesions in terminal ileum, characteristic of lymphangiectasia. EGD with intubation to mid-descending duodenum revealed no significant lesions, but subsequent enteroscopy demonstrated lesions in distal duodenum/proximal jejunum similar to those in terminal ileum characteristic of lymphangiectasia. Histopathologic analysis of lesions of terminal ileum/distal duodenum demonstrated dilated mucosal vessels, confirmed as lymphatic vessels by immunohistochemistry. PIL was diagnosed after excluding secondary causes of intestinal lymphangiectasia. INTERVENTIONS/OUTCOMES Patient placed on standard PIL diet: oral supplements of medium-chain triglycerides, a high protein diet, supplements of fat-soluble vitamins, and avoiding long-chain fatty acids, with marked clinical improvement. LESSONS This work shows that: 1)-standard EGD and colonoscopy may miss characteristic lesions of PIL, 2)-enteroscopy or terminal ileal intubation at colonoscopy may be required for the diagnosis because lesions are typically located in distal duodenum/jejunoileum; and 3)-PIL can first present in the very elderly even with symptoms of short duration.
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Affiliation(s)
- Mitchell S. Cappell
- Division of Gastroenterology & Hepatology, Department of Medicine, William Beaumont Hospital
- Division of Gastroenterology & Hepatology, Department of Medicine, Oakland University William Beaumont School of Medicine
| | - Ahmed Edhi
- Division of Gastroenterology & Hepatology, Department of Medicine, William Beaumont Hospital
| | - Mitual Amin
- Department of Pathology, William Beaumont Hospital
- Department of Pathology, Oakland University William Beaumont School of Medicine, Royal Oak, MI, USA
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7
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Levitt DG, Levitt MD. Protein losing enteropathy: comprehensive review of the mechanistic association with clinical and subclinical disease states. Clin Exp Gastroenterol 2017; 10:147-168. [PMID: 28761367 PMCID: PMC5522668 DOI: 10.2147/ceg.s136803] [Citation(s) in RCA: 96] [Impact Index Per Article: 12.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022] Open
Abstract
Protein losing enteropathy (PLE) has been associated with more than 60 different conditions, including nearly all gastrointestinal diseases (Crohn’s disease, celiac, Whipple’s, intestinal infections, and so on) and a large number of non-gut conditions (cardiac and liver disease, lupus, sarcoidosis, and so on). This review presents the first attempt to quantitatively understand the magnitude of the PLE in relation to the associated pathology for three different disease categories: 1) increased lymphatic pressure (e.g., lymphangiectasis); 2) diseases with mucosal erosions (e.g., Crohn’s disease); and 3) diseases without mucosal erosions (e.g., celiac disease). The PLE with lymphangiectasis results from rupture of the mucosal lymphatics, with retrograde drainage of systemic lymph into the intestinal lumen with the resultant loss of CD4 T cells, which is diagnostic. Mucosal erosion PLE results from macroscopic breakdown of the mucosal barrier, with the epithelial capillaries becoming the rate-limiting factor in albumin loss. The equation derived to describe the relationship between the reduction in serum albumin (CP) and PLE indicates that gastrointestinal albumin clearance must increase by at least 17 times normal to reduce the CP by half. The strengths and limitations of the two quantitative measures of PLE (51Cr-albumin or α1-antitrypsin [αAT] clearance) are reviewed. αAT provides a simple quantitative diagnostic test that is probably underused clinically. The strong, unexplained correlation between minor decreases in CP and subsequent mortality in seemingly healthy individuals raises the question of whether subclinical PLE could account for the decreased CP and, if so, could the mechanism responsible for PLE play a role in the increased mortality? A large-scale study correlating αAT clearance with serum albumin concentrations will be required in order to determine the role of PLE in the regulation of the serum albumin concentration of seemingly healthy subjects.
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Affiliation(s)
- David G Levitt
- Department of Integrative Biology and Physiology, University of Minnesota
| | - Michael D Levitt
- Research Service, Veterans Affairs Medical Center, Minneapolis, MN, USA
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Yamamoto H, Ogata H, Matsumoto T, Ohmiya N, Ohtsuka K, Watanabe K, Yano T, Matsui T, Higuchi K, Nakamura T, Fujimoto K. Clinical Practice Guideline for Enteroscopy. Dig Endosc 2017; 29:519-546. [PMID: 28370422 DOI: 10.1111/den.12883] [Citation(s) in RCA: 115] [Impact Index Per Article: 14.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
Management of small bowel diseases has evolved since the advent of capsule endoscopy (CE) and balloon-assisted enteroscopy (BAE). One of the most common indications for enteroscopy is obscure gastrointestinal bleeding (OGIB), followed by small bowel stenosis, tumors, and inflammatory bowel disease. Although enteroscopes have been regarded as useful tools, correct guidelines are required to ensure that we manipulate these enteroscopes safely and efficiently in clinical practice. Herein, the Japanese Gastroenterological Endoscopy Society has developed 'Clinical Practice Guidelines for Enteroscopy' in collaboration with the Japanese Society of Gastroenterology, the Japanese Gastroenterological Association, and the Japanese Association for Capsule Endoscopy. These guidelines are based on the evidence available until now, but small bowel endoscopy is a relatively new technology, so the guidelines include recommendations based on a consensus reached among experts when the evidence has not been considered sufficient. These guidelines were not designed to be disease-based, but focus on how we should use small bowel CE and BAE in everyday clinical practice.
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Affiliation(s)
| | - Haruhiko Ogata
- Japan Gastroenterological Endoscopy Society
- Japanese Society of Gastroenterology
| | - Takayuki Matsumoto
- Japan Gastroenterological Endoscopy Society
- Japanese Gastroenterological Association
| | - Naoki Ohmiya
- Japan Gastroenterological Endoscopy Society
- Japanese Association for Capsule Endoscopy
| | - Kazuo Ohtsuka
- Japan Gastroenterological Endoscopy Society
- Japanese Gastroenterological Association
| | - Kenji Watanabe
- Japanese Society of Gastroenterology
- Japanese Association for Capsule Endoscopy
| | - Tomonori Yano
- Japan Gastroenterological Endoscopy Society
- Japanese Association for Capsule Endoscopy
| | - Toshiyuki Matsui
- Japan Gastroenterological Endoscopy Society
- Japanese Gastroenterological Association
| | - Kazuhide Higuchi
- Japan Gastroenterological Endoscopy Society
- Japanese Society of Gastroenterology
| | - Tetsuya Nakamura
- Japan Gastroenterological Endoscopy Society
- Japanese Society of Gastroenterology
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Ohmiya N, Horiguchi N, Tahara T, Nagasaka M, Nakagawa Y, Shibata T, Tsukamoto T, Kuroda M. In vivo characterization of abnormalities in small-bowel diseases using probe-based confocal laser endomicroscopy. Endosc Int Open 2017; 5:E547-E558. [PMID: 28670610 PMCID: PMC5482742 DOI: 10.1055/s-0043-106184] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/08/2016] [Accepted: 02/13/2017] [Indexed: 12/22/2022] Open
Abstract
BACKGROUND AND STUDY AIMS Probe-based confocal laser endomicroscopy (pCLE) enables real-time optical biopsy. Little is known about pCLE imaging deep inside the small bowel, therefore the aim of this study was to determine its usefulness. PATIENTS AND METHODS Between April 2014 and January 2016, we performed 38 pCLE examinations during double-balloon enteroscopy with intravenous fluorescein in 37 patients with: tumors (n = 10), vascular disorders (n = 6), inflammatory diseases and drug injuries (n = 13), other disorders (n = 4), and normal findings (n = 4). We measured the calibers of capillary and lymphatic vessels at 15 different sites and compared the calibers between pCLE images and histopathology. We also compared pCLE findings with pathologic diagnosis. RESULTS The inner diameters of capillary vessels beneath the epithelium and in the middle of villi were 16.2 ± 3.0 µm and 14.5 ± 3.1 µm, respectively, in the pCLE images, but these were not consistent with formalin-fixed paraffin-embedded histology. In tumors, larger capillary vessels were observed in aggressive malignant lymphoma and metastasis, and a "soccer ball-like pattern" constituting homogenous dark cells packed with polygonal, narrower capillary vessels was characteristic in pCLE images of a malignant lymphoma follicle. Hereditary hemorrhagic telangiectasia and angiodysplasia contained anastomosis of capillary vessels of different calibers. In IgA vasculitis, segmental capillary strictures were observed. Intestinal lymphangiectasia with protein-losing enteropathy contained a reticular network of lymphatic vessels and dilated lymphatic ducts accompanied by numerous cell gaps. pCLE findings corresponded to pathologic diagnosis in 32 examinations (91 %). CONCLUSIONS pCLE is useful for in vivo analysis of abnormalities of the capillary and lymphatic vessels and epithelium, and for diagnosis in various small-bowel diseases.
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Affiliation(s)
- Naoki Ohmiya
- Department of Gastroenterology, Fujita Health University School of Medicine, Toyoake, Aichi, Japan,Corresponding author Naoki Ohmiya, MD PhD Department of GastroenterologyFujita Health University School of Medicine1-98 Kutsukake-choToyoakeAichi 470-1192Japan+81-562-938300
| | - Noriyuki Horiguchi
- Department of Gastroenterology, Fujita Health University School of Medicine, Toyoake, Aichi, Japan
| | - Tomomitsu Tahara
- Department of Gastroenterology, Fujita Health University School of Medicine, Toyoake, Aichi, Japan
| | - Mitsuo Nagasaka
- Department of Gastroenterology, Fujita Health University School of Medicine, Toyoake, Aichi, Japan
| | - Yoshihito Nakagawa
- Department of Gastroenterology, Fujita Health University School of Medicine, Toyoake, Aichi, Japan
| | - Tomoyuki Shibata
- Department of Gastroenterology, Fujita Health University School of Medicine, Toyoake, Aichi, Japan
| | - Tetsuya Tsukamoto
- Department of Diagnostic Pathology I, Fujita Health University School of Medicine, Toyoake, Aichi, Japan
| | - Makoto Kuroda
- Department of Diagnostic Pathology I, Fujita Health University School of Medicine, Toyoake, Aichi, Japan
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10
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Abstract
Protein-losing enteropathy (PLE) is a rare complication of intestinal diseases. Its main manifestation is hypoproteinemic edema. The diagnosis of PLE is based on the verification of protein loss into the intestinal lumen, by determining fecal α1-antitrypsin concentration and clearance. The localization of the affected colonic segment is clarified using radiologic and endoscopic techniques. The mainstay of treatment for PLE is a fat-free diet enriched with medium-chain triglycerides. Surgical resection of the affected segment of the colon may be the treatment of choice for severe hypoproteinemia resistant to drug therapy.
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Affiliation(s)
- A I Parfenov
- Moscow Clinical Research and Practical Center, Moscow Healthcare Department, Moscow, Russia
| | - L M Krums
- Moscow Clinical Research and Practical Center, Moscow Healthcare Department, Moscow, Russia
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11
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Nakamura M, Hirooka Y, Watanabe O, Yamamura T, Funasaka K, Ohno E, Miyahara R, Kawashima H, Shimoyama Y, Goto H. Evaluation of the mRNA and Protein Expressions of Nutritional Biomarkers in the Gastrointestinal Mucosa of Patients with Small Intestinal Disorders. Intern Med 2016; 55:2145-2152. [PMID: 27522989 DOI: 10.2169/internalmedicine.55.6781] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/06/2022] Open
Abstract
Objective The objectives of this study were to investigate the mRNA and protein expression of biomarkers related to absorption in the small intestinal mucosa of humans and determine the relationships between small intestinal diseases and nutrition. Methods The study subjects consisted of patients scheduled to undergo double-balloon endoscopy (DBE) or total colonoscopy for suspected gastrointestinal disorder in a clinical practice. Biopsies were taken from apparently normal mucosa in the visible areas of 6 parts of the intestines from the duodenum to the colon. The mRNA expression of specific biomarkers (SGLT1, SGLT5, GIP, GLP, LAT1, LAT2, and NPC1L1) in the mucosa was compared among three patient groups: Inflammation, Tumor, and Control. Results Sixty-six patients participated in this study. Both routes of DBE were performed in 20 patients, in whom biopsy samples were obtained from the mucosa for all sections. There were no remarkable differences in the mRNA expression levels among the 3 groups. However, SGLT1, GIP, GLP, and NPC1L1 exhibited specific distribution patterns. The expression levels of GIP and NPC1L1 were highest in the upper jejunum, but were extremely low in the terminal ileum and colon. A comparison of the mRNA expression profile in each intestinal section revealed that the SGLT1 mRNA expression in the Tumor group and the GIP mRNA expression in the Inflammation group were significantly higher than the corresponding levels in the Control group in the upper jejunum. Conclusion The gastrointestinal mucosa of patients with small bowel diseases can maintain proper nutrient absorption, except in the upper jejunum.
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Affiliation(s)
- Masanao Nakamura
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine, Japan
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Lee SJ, Song HJ, Boo SJ, Na SY, Kim HU, Hyun CL. Primary intestinal lymphangiectasia with generalized warts. World J Gastroenterol 2015; 21:8467-8472. [PMID: 26217101 PMCID: PMC4507119 DOI: 10.3748/wjg.v21.i27.8467] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/22/2015] [Revised: 02/25/2015] [Accepted: 04/17/2015] [Indexed: 02/06/2023] Open
Abstract
Primary intestinal lymphangiectasia (PIL) is a rare protein-losing enteropathy with lymphatic leakage into the small intestine. Dilated lymphatics in the small intestinal wall and mesentery are observed in this disease. Laboratory tests of PIL patients revealed hypoalbuminemia, lymphocytopenia, hypogammaglobulinemia and increased stool α-1 antitrypsin clearance. Cell-mediated immunodeficiency is also present in PIL patients because of loss of lymphocytes. As a result, the patients are vulnerable to chronic viral infection and lymphoma. However, cases of PIL with chronic viral infection, such as human papilloma virus-induced warts, are rarely reported. We report a rare case of PIL with generalized warts in a 36-year-old male patient. PIL was diagnosed by capsule endoscopy and colonoscopic biopsy with histological tissue confirmation. Generalized warts were observed on the head, chest, abdomen, back, anus, and upper and lower extremities, including the hands and feet of the patient.
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Araki A, Tsuchiya K, Watanabe M. Advances in balloon endoscopes. Clin J Gastroenterol 2015; 7:189-99. [PMID: 26183736 DOI: 10.1007/s12328-014-0485-3] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/19/2014] [Accepted: 03/25/2014] [Indexed: 01/10/2023]
Abstract
In September 2003, a double-balloon endoscope (DBE) composed of balloons attached to a scope and an overtube was released in Japan prior to becoming available in other parts of the world. The DBE was developed by Dr. Yamamoto (1), and 5 different types of scopes with different uses have already been marketed. In April 2007, a single-balloon small intestinal endoscope was released with a balloon attached only to the overtube as a subsequent model. This article presents a detailed account of the development of these scopes up to the present time.
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Affiliation(s)
- Akihiro Araki
- Department of Gastroenterology and Hepatology, Graduate School, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo, 113-8510, Japan,
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Ohmiya N, Nakamura M, Yamamura T, Yamada K, Nagura A, Yoshimura T, Hirooka Y, Hirata I, Goto H. Classification of intestinal lymphangiectasia with protein-losing enteropathy: white villi type and non-white villi type. Digestion 2015; 90:155-66. [PMID: 25278259 DOI: 10.1159/000365987] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/04/2014] [Accepted: 07/18/2014] [Indexed: 02/04/2023]
Abstract
BACKGROUND/AIMS We classified intestinal lymphangiectasia (IL) into two categories, the white and non-white villi types, and evaluated their clinical characteristics and therapeutic responses. METHODS Of the 988 patients who underwent double-balloon enteroscopy, 14 consecutive patients (7 men and 7 women, median age at onset 34 years) were enrolled with immunohistochemically confirmed IL with protein-losing enteropathy. RESULTS Enteroscopically the white villi type (n = 8) showed white plaques and white-tipped villi were scattered in the small bowel, while non-white villi type (n = 6) showed that apparently normal but under more detailed observation, low and round villi with a normal color were diffused. The serum albumin levels and fecal α1-antitrypsin clearance before treatment were significantly worse in the non-white villi type (p = 0.017 and 0.039, respectively), whereas the serum immunoglobulin A and M levels were significantly lower in the white villi type (p = 0.010 and 0.046, respectively). At gastroscopy, a non-cirrhotic snakeskin appearance was significantly observed in the non-white villi type (p = 0.015). The corticosteroid response was better in the non-white villi type (p = 0.015). CONCLUSION Two distinct subgroups were found in IL. This classification was useful in pathophysiological clustering and in predicting the therapeutic response.
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Affiliation(s)
- Naoki Ohmiya
- Department of Gastroenterology, Fujita Health University School of Medicine, Toyoake, Japan
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Amiot A. Gastro-entéropathies exsudatives. Rev Med Interne 2015; 36:467-73. [DOI: 10.1016/j.revmed.2014.12.001] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/08/2014] [Revised: 11/20/2014] [Accepted: 12/03/2014] [Indexed: 12/13/2022]
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Takada J, Araki H, Kubota M, Ibuka T, Shiraki M, Shimizu M, Moriwaki H. Protein-losing enteropathy cured by resection of adenomatous goiter: report of a case. Clin J Gastroenterol 2015; 8:120-5. [PMID: 25845937 DOI: 10.1007/s12328-015-0565-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/17/2015] [Accepted: 03/24/2015] [Indexed: 10/23/2022]
Abstract
A 51-year-old Japanese woman presented to our hospital with systemic edema and general fatigue. Her serum albumin level was very low (1.5 g/dL). Technetium-99 m-human serum albumin ((99m)Tc-HSA) scintigraphy showed albumin leakage from the upper small bowel. Magnetic resonance lymphangiography showed dilated lymphatic vessels in the chest, whereas double-balloon enteroscopy (DBE) showed white villi and chyle leakage in the deeper part of the duodenal mucosa. A duodenal mucosa biopsy specimen revealed lymphangiectasia. She was diagnosed with protein-losing enteropathy (PLE). Treatment with a fat-restricted diet and tranexamic acid--previously reported to be effective against PLE--was attempted, but was ineffective. A thyroid tumor was simultaneously detected in her left neck, and was found to extend to the mediastinum on computed tomography. The tumor (size, >5 cm) was resected, and a pathological diagnosis of adenomatous goiter was made. The patient's serum albumin level increased to normal levels within 1 month postoperatively. After 6 months, (99m)Tc-HSA scintigraphy showed no albumin leakage from the gastrointestinal tract, and disappearance of white villi and chyle leakage on DBE. No lymphangiectasia was noted in the biopsy specimen. Adenomatous goiter was thus considered the cause of the PLE, possibly through lymph flow obstruction in the mediastinum.
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Affiliation(s)
- Jun Takada
- Department of Gastroenterology, Gifu University Graduate School of Medicine, 1-1 Yanagido, Gifu, 501-1194, Japan,
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Chen Z, Li MT, Xu D, Yang H, Li J, Zhao JL, Zhang HH, Han SM, Xu T, Zeng XF. Protein-losing enteropathy in systemic lupus erythematosus: 12 years experience from a Chinese academic center. PLoS One 2014; 9:e114684. [PMID: 25490025 PMCID: PMC4260872 DOI: 10.1371/journal.pone.0114684] [Citation(s) in RCA: 24] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/29/2014] [Accepted: 11/12/2014] [Indexed: 01/08/2023] Open
Abstract
Objective Protein-losing enteropathy (PLE) is a complication in some systemic lupus erythematosus (SLE) patients that is often misdiagnosed. With this study, we provide insight into clinical characteristics, laboratory characteristics, diagnostic tests, risk factors, treatment, and prognosis of the disease. Methods A retrospective, case-control study was performed in 44 patients with SLE-related PLE (PLE group) and 88 patients with active SLE (control group) admitted to our care from January 2000−January 2012. Risk factors for SLE-related PLE were examined, and we analyzed the accuracy of single and combined laboratory characteristics in discriminating SLE-related PLE from active SLE. Serum albumin and C3 levels were measured as outcome during and after treatment with corticosteroids and immunosuppressive agents. Results The PLE group had lower mean serum albumin and 24-hour urine protein levels, higher mean total plasma cholesterol levels, and greater frequencies of anti-SSA and SSB seropositivity compared with the control group. Anti-SSA seropositivity, hypoalbuminemia, and hypercholesterolemia were independent risk factors for SLE-related PLE. The simultaneous presence of serum albumin (<22 g/l) and 24-hour urine protein (<0.8 g/24 h) had high specificity, positive predictive value, negative predictive value, and positive likelihood ratio, a low negative likelihood ratio and no significant reduction in sensitivity. High dosage of glucocorticosteroid combined with cyclophosphomide were mostly prescribed for SLE-related PLE. Conclusion SLE-related PLE should be considered when an SLE patient presents with generalized edema, anti-SSA antibody seropositivity, hypercholesterolemia, severe hypoalbuminemia, and low 24-hour urine protein levels. Aggressive treatment for lupus might improve prognosis.
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Affiliation(s)
- Zhen Chen
- Department of Rheumatology, Peking Union Medical College Hospital, Chinese Academy of Medical Science & Peking Union Medical College, Beijing, China, and Key Laboratory of Rheumatology and Clinical Immunology, Ministry of Education, Beijing, China
| | - Meng-Tao Li
- Department of Rheumatology, Peking Union Medical College Hospital, Chinese Academy of Medical Science & Peking Union Medical College, Beijing, China, and Key Laboratory of Rheumatology and Clinical Immunology, Ministry of Education, Beijing, China
| | - Dong Xu
- Department of Rheumatology, Peking Union Medical College Hospital, Chinese Academy of Medical Science & Peking Union Medical College, Beijing, China, and Key Laboratory of Rheumatology and Clinical Immunology, Ministry of Education, Beijing, China
| | - Hong Yang
- Department of Gastroenterology, Peking Union Medical College Hospital, Chinese Academy of Medical Science & Peking Union Medical College, Beijing, China
| | - Jing Li
- Department of Rheumatology, Peking Union Medical College Hospital, Chinese Academy of Medical Science & Peking Union Medical College, Beijing, China, and Key Laboratory of Rheumatology and Clinical Immunology, Ministry of Education, Beijing, China
| | - Jiu-Liang Zhao
- Department of Rheumatology, Peking Union Medical College Hospital, Chinese Academy of Medical Science & Peking Union Medical College, Beijing, China, and Key Laboratory of Rheumatology and Clinical Immunology, Ministry of Education, Beijing, China
| | - Heng-Hui Zhang
- Peking University People’s Hospital, Peking University Hepatology Institute, Beijing Key Laboratory of Hepatitis C and Immunotherapy for Liver Diseases, Beijing, China
| | - Shao-Mei Han
- Department of Epidemiology and Statistics, Chinese Academy of Medical Science & Peking Union Medical College, Beijing, China
| | - Tao Xu
- Department of Epidemiology and Statistics, Chinese Academy of Medical Science & Peking Union Medical College, Beijing, China
| | - Xiao-Feng Zeng
- Department of Rheumatology, Peking Union Medical College Hospital, Chinese Academy of Medical Science & Peking Union Medical College, Beijing, China, and Key Laboratory of Rheumatology and Clinical Immunology, Ministry of Education, Beijing, China
- * E-mail:
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Dabos KJ, Koulaouzidis A. Portal hypertensive enteropathy, occult bleeding, and capsule endoscopy: where do we go from here? Dig Dis Sci 2014; 59:899-901. [PMID: 24652110 DOI: 10.1007/s10620-014-3103-9] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/20/2023]
Affiliation(s)
- Konstantinos J Dabos
- Endoscopy Unit, Centre for Liver and Digestive Disorders, The Royal Infirmary of Edinburgh, 51 Little France Crescent, Edinburgh, UK
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Aoud SE, Frikha F, Snoussi M, Ben Salah R, Bahloul Z. Entéropathies exsudatives : à propos de cinq observations et revue de la littérature. ACTA ACUST UNITED AC 2014. [DOI: 10.1007/s12157-014-0516-1] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/25/2022]
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