Kim Y, Kwak J, Jung S, Lee S, Jang HN, Cho HS, Chang SH, Kim HJ. Oral cyclophosphamide-induced posterior reversible encephalopathy syndrome in a patient with ANCA-associated vasculitis: A case report. World J Clin Cases 2021; 9(21): 6130-6137 [PMID: 34368335 DOI: 10.12998/wjcc.v9.i21.6130]
Corresponding Author of This Article
Hyun-Jung Kim, MD, PhD, Professor, Internal Medicine, Gyeongsang National University Hospital, 79 Gangnam-ro, Jinju 52727, South Korea. kimhjyh@gnu.ac.kr
Research Domain of This Article
Urology & Nephrology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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: http://creativecommons.org/licenses/by-nc/4.0/
World J Clin Cases. Jul 26, 2021; 9(21): 6130-6137 Published online Jul 26, 2021. doi: 10.12998/wjcc.v9.i21.6130
Oral cyclophosphamide-induced posterior reversible encephalopathy syndrome in a patient with ANCA-associated vasculitis: A case report
Yire Kim, Jihye Kwak, Sehyun Jung, Seunghye Lee, Ha Nee Jang, Hyun Seop Cho, Se-Ho Chang, Hyun-Jung Kim
Yire Kim, Jihye Kwak, Sehyun Jung, Seunghye Lee, Ha Nee Jang, Hyun Seop Cho, Se-Ho Chang, Hyun-Jung Kim, Internal Medicine, Gyeongsang National University Hospital, Jinju 52727, South Korea
Ha Nee Jang, Hyun Seop Cho, Se-Ho Chang, Hyun-Jung Kim, Institute of Health Sciences, Gyeongsang National University, Jinju 52727, South Korea
Se-Ho Chang, Hyun-Jung Kim, Internal Medicine, College of Medicine, Gyeongsang National University, Jinju 52727, South Korea
Author contributions: Kim Y, Kwak J, Cho HS and Kim HJ collected data, reviewed the literature and contributed to manuscript drafting; Jung S, Lee S, Jang HN and Chang SH reviewed the literature and contributed to manuscript drafting; Kim Y analyzed and interpreted the imaging findings; Kim Y and Kim HJ were responsible for the revision of the manuscript for important intellectual content; All authors have read and approve the final manuscript.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
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: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Hyun-Jung Kim, MD, PhD, Professor, Internal Medicine, Gyeongsang National University Hospital, 79 Gangnam-ro, Jinju 52727, South Korea. kimhjyh@gnu.ac.kr
Received: March 29, 2021 Peer-review started: March 29, 2021 First decision: April 28, 2021 Revised: April 30, 2021 Accepted: May 20, 2021 Article in press: May 20, 2021 Published online: July 26, 2021
Abstract
BACKGROUND
Posterior reversible encephalopathy syndrome (PRES) manifests many neurological symptoms with typical features on neuroimaging studies and has various risk factors. Cyclophosphamide is one of the therapeutic agents for anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis. Cyclophosphamide as the sole cause of PRES has been reported in only a few cases. Herein, we report a unique case of early-onset oral cyclophosphamide-induced PRES in a patient with ANCA-associated vasculitis.
CASE SUMMARY
A 73-year-old man was transferred to our hospital for sepsis due to acute cholangitis. He had already received hemodialysis for two weeks due to septic acute kidney injury. His azotemia was not improved after sepsis resolved and perinuclear-ANCA was positive. Kidney biopsy showed crescentic glomerulonephritis. Alveolar hemorrhage was observed on bronchoscopy. He was initially treated with intravenous methylprednisolone and plasma exchange for one week. And then, two days after adding oral cyclophosphamide, the patient developed generalized tonic-clonic seizures. We diagnosed PRES by Brain magnetic resonance imaging (MRI) and electroencephalography. Seizures were controlled with fosphenytoin 750 mg. Cyclophosphamide was suspected to be the cause of PRES and withdrawal. His mentality was recovered after seven days and brain MRI showed normal state after two weeks.
CONCLUSION
The present case shows the possibility of PRES induction due to short-term use of oral cyclophosphamide therapy. Physicians should carefully monitor neurologic symptoms after oral cyclophosphamide administration in elderly patients with underlying diseases like sepsis, renal failure and ANCA-associated vasculitis.
Core Tip: Posterior reversible encephalopathy syndrome (PRES) is a neurological disease that can occur suddenly during the clinical course of various disorders. Since the clinical course of PRES is reversible, it is important to diagnose it quickly and correct the cause. Cyclophosphamide has recently been reported as one of the causes of PRES. This is the first report that PRES occurred at the early onset of oral cyclophosphamide therapy. Physicians should carefully monitor neurologic symptoms after oral cyclophosphamide administration in elderly patients with underlying diseases like sepsis, renal failure, and anti-neutrophil cytoplasmic antibody-associated vasculitis.
