Hydroxychloroquine-Induced Toxic Myopathy Causing Diaphragmatic Weakness and Lung Collapse Requiring Prolonged Mechanical Ventilation.

Abstract

A 42-year-old woman with juvenile idiopathic arthritis was treated with anakinra, corticosteroids, and hydroxychloroquine when she developed chronic hypoxic respiratory myopathy. She was admitted to the intensive care unit for acute hypercapnic respiratory failure and required prolonged intubation, subsequent tracheostomy, and long-term ventilatory support due to multiple failed spontaneous breathing trials after discontinuation of anakinra and steroids. Muscle biopsy revealed type II fiber atrophy with the accumulation of autophagosomes and vacuoles presenting as curvilinear bodies, elevated MHC class I antigen expression, and infiltration by CD68+ macrophages and CD8+ T cells. Type II fiber atrophy was attributed to corticosteroid use and curvilinear bodies due to blockade of autophagy by hydroxychloroquine. After hydroxychloroquine was discontinued, the patient recovered to her prehospitalization baseline.

• Authors: Julie Pillittere, Sundus Mian, Timothy E Richardson, Andras Perl
• Journal: Journal of investigative medicine high impact case reports (J Investig Med High Impact Case Rep) 2020 Jan-Dec Vol. 8 Pg. 2324709620950113 ISSN: 2324-7096 [Electronic] United States
• PMID: 32787461 (Publication Type: Case Reports, Journal Article)

Chemical References

• Adrenal Cortex Hormones
• Antirheumatic Agents
• Interleukin 1 Receptor Antagonist Protein
• Hydroxychloroquine

Topics

• Adrenal Cortex Hormones
• Adult
• Antirheumatic Agents (adverse effects)
• Female
• Humans
• Hydroxychloroquine (adverse effects)
• Interleukin 1 Receptor Antagonist Protein
• Muscle Weakness (chemically induced)
• Muscular Diseases (chemically induced, complications, pathology)
• Quadriceps Muscle (pathology)
• Respiration, Artificial
• Respiratory Insufficiency (etiology, therapy)
• Tomography, X-Ray Computed
• Tracheostomy