Year 2021 / Volume 113 / Number 11
Original
The diagnostic value of capsule endoscopy in children with intestinal lymphangiectasia

765-769

DOI: 10.17235/reed.2021.7682/2020

Jie Wu, Zhiheng Huang, Min Ji, Zhinong Jiang, Yuhuan Wang, Zifei Tang, Ying Huang,

Abstract
Background: intestinal lymphangiectasia is an unusual cause of protein-losing enteropathy due to either congenital malformation or obstruction of the intestinal lymphatics. However, few reports have investigated the use of video capsule endoscopy in children with intestinal lymphangiectasia. This study was performed to evaluate the diagnostic value of video capsule endoscopy for pediatric intestinal lymphangiectasia. Methods: in this retrospective study, all patients who underwent video capsule endoscopy between January 2014 and July 2020 were included. Clinical information and video capsule endoscopy data were analyzed. Results: twelve children were enrolled, 7 males and 5 females, with an age at disease onset of 4.5 (range: 3.2-9.3) years and a disease duration of 12.0 (range: 1.3-30.0) months. The most common symptoms were hypoproteinemia (10, 83.3 %), diarrhea (7, 58.3 %), edema (6, 50.0 %), and abdominal pain (3, 25.0 %). Eight patients had low lymphocyte counts, whereas 10 had reduced serum albumin levels (23.2 ± 5.8 g/L). Video capsule endoscopy revealed an overall white snowy appearance due to the presence of whitish, swollen villi in all patients. Regarding the macroscopic lesions of lymphangiectasia, 7 cases involved the entire small bowel from the duodenum to the ileocecal valve, while 5 cases involved part of the small bowel. All patients were treated with medium-chain triglyceride diets, and albumin infusions were administered to 10 patients; sirolimus treatment was administered to 3 patients. At the last follow-up, 5 patients still had hypoalbuminemia and one patient had died of intestinal lymphoma. Conclusion: video capsule endoscopy is useful for the diagnosis of intestinal lymphangiectasia and should be applied as a valuable and less invasive examination to confirm or establish a diagnosis.
Share Button
New comment
Comments
No comments for this article
References
1. Vignes S, Bellanger J. Primary intestinal lymphangiectasia (Waldmann's disease). Orphanet J Rare Dis 2008;3:5.
2. Oh TG, Chung JW, Kim HM, et al. Primary intestinal lymphangiectasia diagnosed by capsule endoscopy and double balloon enteroscopy. World J Gastrointest Endosc 2011;3(11):235-40.
3. Glukhovsky A, Jacob H. The development and application of wireless capsule endoscopy. Int J Med Robot 2004;1(1):114-23.
4. Chamouard P, Nehme-Schuster H, Simler J-M, et al. Videocapsule endoscopy is useful for the diagnosis of intestinal lymphangiectasia. Dig Liver Dis 2006;38(9):699-703.
5. Wu J, Huang Z, Wang Y, et al. Clinical features of capsule endoscopy in 825 children: A single-center, retrospective cohort study. Medicine (Baltimore) 2020;99(43):e22864.
6. Rivet C, Lapalus MG, Dumortier J, et al. Use of capsule endoscopy in children with primary intestinal lymphangiectasia. Gastrointest Endosc 2006;64(4):649-50.
7. Wu J, Huang Z, Wu H, et al. The diagnostic value of video capsule endoscopy for Meckel's diverticulum in children. Rev Esp Enferm Dig 2020;112(6):429-33.
8. Prasad D, Srivastava A, Tambe A, et al. Clinical Profile, Response to Therapy, and Outcome of Children with Primary Intestinal Lymphangiectasia. Dig Dis 2019;37(6):458-66.
9. Arguelles-Arias F, Donat E, Fernández-Urien I, et al. Guideline for wireless capsule endoscopy in children and adolescents: A consensus document by the SEGHNP (Spanish Society for Pediatric Gastroenterology, Hepatology, and Nutrition) and the SEPD (Spanish Society for Digestive Diseases). Rev Esp Enferm Dig 2015;107(12):714-31.
10. Antao B, Bishop J, Shawis R, et al. Clinical application and diagnostic yield of wireless capsule endoscopy in children. J Laparoendosc Adv Surg Tech A 2007;17(3):364-70.
11. van der Reijden SM, van Wijk MP, Jacobs MAJM, et al. Video Capsule Endoscopy to Diagnose Primary Intestinal Lymphangiectasia in a 14-Month-Old Child. J Pediatr Gastroenterol Nutr 2017;64(6):e161.
12. Ohmiya N, Nakamura M, Yamamura T, et al. Classification of intestinal lymphangiectasia with protein-losing enteropathy: white villi type and non-white villi type. Digestion 2014;90(3):155-66.
13. Ersoy O, Akin E, Demirezer A, et al. Evaluation of primary intestinal lymphangiectasia by capsule endoscopy. Endoscopy 2013;45(Suppl 2 UCTN):E61-2.
14. Patel KV, Goel RM, Wong T. Diffuse large B-cell lymphoma recurrence complicating primary intestinal lymphangiectasia. Clin Gastroenterol Hepatol 2013;11(12):e86-7.
15. Ozen A, Comrie WA, Ardy RC, et al. CD55 Deficiency, Early-Onset Protein-Losing Enteropathy, and Thrombosis. N Engl J Med 2017;377(1):52-61.
16. Hoshina T, Kusuhara K, Saito M, et al. Cytomegalovirus-associated protein-losing enteropathy resulting from lymphangiectasia in an immunocompetent child. Jpn J Infect Dis 2009;62(3):236-8.
17. Troskot R, Jurčić D, Bilić A, et al. How to treat an extensive form of primary intestinal lymphangiectasia? World J Gastroenterol 2015;21(23):7320-5.
18. Li S, Liu X, He Y, et al. Nutritional therapy and effect assessment of infants with primary intestinal lymphangiectasia: Case reports. Medicine (Baltimore) 2017;96(51):e9240.
Related articles

Letter

Pneumatosis intestinalis in a pediatric patient

DOI: 10.17235/reed.2021.8482/2021

Digestive Diseases Image

Endoscopic forceps removal for complicated magnetic beads impaction

DOI: 10.17235/reed.2021.7731/2020

Digestive Diseases Image

Jejunal schwannoma detected by video capsule endoscopy

DOI: 10.17235/reed.2020.6474/2019

Letter to the Editor

An unusual etiology of abdominal pain in children

DOI: 10.17235/reed.2016.4168/2015

Citation tools
Wu J, Huang Z, Ji M, Jiang Z, Wang Y, Tang Z, et all. The diagnostic value of capsule endoscopy in children with intestinal lymphangiectasia. 7682/2020


Download to a citation manager

Download the citation for this article by clicking on one of the following citation managers:

Metrics
This article has received 1320 visits.
This article has been downloaded 276 times.

Statistics from Dimensions


Statistics from Plum Analytics

Publication history

Received: 24/11/2020

Accepted: 07/03/2021

Online First: 18/03/2021

Published: 05/11/2021

Article revision time: 93 days

Article Online First time: 114 days

Article editing time: 346 days


Share
This article hasn't been rated yet.
Reader rating:
Valora este artículo:




Asociación Española de Ecografía Digestiva Sociedad Española de Endoscopia Digestiva Sociedad Española de Patología Digestiva
The Spanish Journal of Gastroenterology is the official organ of the Sociedad Española de Patología Digestiva, the Sociedad Española de Endoscopia Digestiva and the Asociación Española de Ecografía Digestiva
Cookie policy Privacy Policy Legal Notice © Copyright 2023 y Creative Commons. The Spanish Journal of Gastroenterology