Acute hemorrhagic leukoencephalitis with a positive transfusion-related antibody screening test

Article information

J Neurocrit Care. 2024;17(2):102-103
Publication date (electronic) : 2024 December 11
doi : https://doi.org/10.18700/jnc.240027
Department of Neurology, Hallym University College of Medicine, Chuncheon, Korea
Corresponding authors: Chulho Kim, MD, PhD Department of Neurology, Hallym University College of Medicine, 1 Hallimdaehak-gil, Chuncheon 24252, Korea Tel: +82-33-240-5255, Fax: +82-33-255-1338 E-mail: gumdol52@naver.com
Received 2024 August 9; Revised 2024 November 1; Accepted 2024 November 21.

A 65-year-old female patient visited the emergency department with a confusional mentality. The symptoms of the patient were initially suspected to be due to an overdose of her usual antipsychotics, and we planned to instantly discontinue her medications. However, her hemoglobin was observed to be 5.5 g/dL. Although the transfusion-related antibody screening test was positive, the patient received 1 point for packed red blood cells because of severe anemia. The same antibody screening test performed 10 years previously yielded negative results. She returned to the emergency department a day later with recurrent seizures. Brain magnetic resonance imaging (MRI) showed multiple patches of high signal intensity in the fluid-attenuated inversion recovery image and multiple leptomeningeal enhancements on contrast-enhanced T1-weighted imaging (Fig. 1). Cerebrospinal fluid analysis showed no pleocytosis, but the consciousness of the patient deteriorated to semicoma within 3 days. Follow-up brain MRI showed multiple diffusion-restrictive lesions with diffuse punctuated hemorrhages and extensive mass effect (Fig. 2). Posterior reversible encephalopathy syndrome (PRES) was considered as a differential diagnosis, but concomitant massive edema, diffusion-restrictive lesions, and extensive hemorrhage were more consistent with acute hemorrhagic leukoencephalitis (AHLE) than PRES. In addition, recent medications were observed to be unrelated, and laboratory tests, including autoimmune diseases, infectious diseases, metabolic diseases, and thrombosis abnormalities, including coronavirus disease 2019, were all negative. Despite extensive, intensive care with 5 days of steroid pulse therapy, the patient progressed to a coma and died of multiorgan failure. AHLE is a severe form of acute demyelinating encephalomyelitis [1,2]. Herein, we report a case of MRI-suspected AHLE that was associated with an immunologic response to blood transfusion in a patient with a positive antibody screening test.

Fig. 1.

Initial magnetic resonance imaging of the patients. Initial fluid-attenuated inversion recovery image (A) showed multiple scattered white matter lesions (arrows) but no specific findings in the diffusion-weighted image (B) and gradient echo sequence (C).

Fig. 2.

Follow-up magnetic resonance imaging of the patient. Widespread white matter hyperintensity with extensive cerebral edema (A) was observed in follow-up fluid-attenuated inversion recovery images. Multiple diffusion-restrictive lesions (B, E) and numerous microhemorrhages (arrowheads) were observed in follow-up diffusion-weighted imaging and gradient recalled echo images (C, D). Apparent diffusion coefficient map imaging confirmed a low signal intensity lesion (F) suggestive of cytotoxic edema.

Notes

Ethics statement

This study was approved by the Institutional Review Board (IRB)/Ethics Committee of Chuncheon Sacred Heart Hospital (No. 2024-07-008), and written informed consent was waived from the IRB.

Conflict of interest

Chulho Kim is an editorial board member of the journal, but she was not involved in the peer reviewer selection, evaluation, or decision process of this article. No other potential conflict of interest relevant to this article was reported.

Author contributions

Conceptualization: all authors. Data curation: all authors. Supervision: CK, JHS. Validation: JC, SHL. Visualization: JC. Writing–original draft: JC, CK. Writing–review and editing: all authors. All authors read and agreed to the published version of the manuscript.

References

1. Gibbs WN, Kreidie MA, Kim RC, Hasso AN. Acute hemorrhagic leukoencephalitis: neuroimaging features and neuropathologic diagnosis. J Comput Assist Tomogr 2005;29:689–93.
2. Grzonka P, Scholz MC, De Marchis GM, Tisljar K, Rüegg S, Marsch S, et al. Acute hemorrhagic leukoencephalitis: a case and systematic review of the literature. Front Neurol 2020;11:899.

Article information Continued

Fig. 1.

Initial magnetic resonance imaging of the patients. Initial fluid-attenuated inversion recovery image (A) showed multiple scattered white matter lesions (arrows) but no specific findings in the diffusion-weighted image (B) and gradient echo sequence (C).

Fig. 2.

Follow-up magnetic resonance imaging of the patient. Widespread white matter hyperintensity with extensive cerebral edema (A) was observed in follow-up fluid-attenuated inversion recovery images. Multiple diffusion-restrictive lesions (B, E) and numerous microhemorrhages (arrowheads) were observed in follow-up diffusion-weighted imaging and gradient recalled echo images (C, D). Apparent diffusion coefficient map imaging confirmed a low signal intensity lesion (F) suggestive of cytotoxic edema.