Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jun 26, 2022; 10(18): 6234-6240
Published online Jun 26, 2022. doi: 10.12998/wjcc.v10.i18.6234
Primary intestinal lymphangiectasia presenting as limb convulsions: A case report
Yun Cao, Xiao-Hong Feng, Hai-Xiang Ni
Yun Cao, Xiao-Hong Feng, Hai-Xiang Ni, Department of Endocrinology, The First Affiliated Hospital of Zhejiang Chinese Medical University (Zhejiang Provincial Hospital of Traditional Chinese Medicine), Hangzhou 310006, Zhejiang Province, China
Author contributions: Cao Y and Feng XH contributed to literature review and manuscript drafting; Cao Y and Ni HX contributed to patient management and data analysis; and all authors approved the final article and assured all the questions regarding the accuracy of the article.
Informed consent statement: The patient consented to his case being published anonymously.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest to disclose.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Xiao-Hong Feng, MD, Associate Professor, Department of Endocrinology, The First Affiliated Hospital of Zhejiang Chinese Medical University (Zhejiang Provincial Hospital of Traditional Chinese Medicine), No. 54 Youdian Road, Hangzhou 310006, Zhejiang Province, China. feng_xiaohong2021@163.com
Received: November 26, 2021
Peer-review started: November 26, 2021
First decision: January 24, 2022
Revised: March 15, 2022
Accepted: April 29, 2022
Article in press: April 29, 2022
Published online: June 26, 2022
Processing time: 202 Days and 12.1 Hours
Abstract
BACKGROUND

Primary intestinal lymphangiectasia (PIL) is a rare protein-losing enteropathy characterized by abnormally dilated lymphatic structures, resulting in leakage of lymph (rich in protein, lymphocytes, and fat) from the intestinal mucosal and submucosal layers and thus hypoproteinemia, lymphopenia, hypolipidemia, and pleural effusion.

CASE SUMMARY

A 19-year-old Chinese male patient complained of recurrent limb convulsions for the last 1 year. Laboratory investigations revealed low levels of calcium and magnesium along with hypoproteinemia and high parathyroid hormone levels, whereas gastroscopy exhibited chronic non-atrophic gastritis and duodenal lymphatic dilatation. Subsequent gastric biopsy showed moderate chronic inflammatory cell infiltration distributed around a small mucosal patch in the descending duodenum followed by lymphatic dilatation in the mucosal lamina propria, which was later diagnosed as PIL. The following appropriate medium-chain triglycerides nutritional support significantly improved the patient’s symptoms.

CONCLUSION

Since several diseases mimic the clinical symptoms displayed by PIL, like limb convulsions, low calcium and magnesium, and loss of plasma proteins, it is imperative to conduct a detailed analysis to avoid any misdiagnosis while pinpointing the correct clinical diagnosis and simultaneously ruling out other clinical aspects in the reported cases without any past disease history. A careful assessment should always be made to ensure an accurate diagnosis in a timely manner so that the patient can be delivered quality health services for a positive health outcome.

Keywords: Protein-losing enteropathy; Primary intestinal lymphangiectasia; Limb convulsions; Adult; Case report

Core Tip: In this case report, a 19-year-old Chinese male patient complained of recurrent limb convulsions for 1 year. Laboratory investigations revealed low levels of calcium and magnesium along with hypoproteinemia and high parathyroid hormone levels, but the patient had no limbs edema, which is rear in primary intestinal lymphangiectasia cases. Differential diagnosis is difficult in such case. Careful analysis and examination results finally enabled the patient to receive effective treatment after a definite diagnosis.