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Double Hit of Hydroxichloroquine and Amiodarone Induced Renal Phospholipidosis in a Patient with Monoclonal Gammopathy and Sclerodermiform Syndrome: A Case Report and Review of the Literature

Authors :
José C. De la Flor
Pablo Rodríguez-Doyágüez
Daniel Villa
Rocío Zamora
Francisco Díaz
Source :
Medical Sciences, Vol 12, Iss 2, p 25 (2024)
Publication Year :
2024
Publisher :
MDPI AG, 2024.

Abstract

Phospholipidosis is a rare disorder which consists of an excessive intracellular accumulation of phospholipids and the appearance of zebra bodies or lamellar bodies when looking at them using electron microscopy. This disease is associated with certain genetic diseases or is secondary to drugs or toxins. Drug-induced phospholipidosis encompasses many types of pharmaceuticals, most notably chloroquine, amiodarone or ciprofloxacin. Clinically and histologically, renal involvement can be highly variable, with the diagnosis not being made until the zebra bodies are seen under an electron microscope. These findings may require genetic testing to discount Fabry disease, as its histological findings are indistinguishable. Most of the chemicals responsible are cationic amphiphilic drugs, and several mechanisms have been hypothesized for the formation of zebra bodies and their pathogenic significance. However, the relationship between drug toxicity and phospholipid accumulation, zebra bodies and organ dysfunction remains enigmatic, as do the renal consequences of drug withdrawal. We present, to our knowledge, the first case report of acute renal injury with a monoclonal gammopathy of renal significance, lesions, and sclerodermiform syndrome, with zebra bodies that were associated with the initiation of a hydroxychloroquine and amiodarone treatment, as an example of drug-induced-phospholipidosis.

Details

Language :
English
ISSN :
20763271
Volume :
12
Issue :
2
Database :
Directory of Open Access Journals
Journal :
Medical Sciences
Publication Type :
Academic Journal
Accession number :
edsdoj.4fafe11e9f61441fb16a986a180dcf14
Document Type :
article
Full Text :
https://doi.org/10.3390/medsci12020025