Chronic Graft-versus-host Disease Presenting with Multiple Punctate Intracranial Lesions on Contrast-enhanced Magnetic Resonance Imaging

Terada, Makoto; Nakamagoe, Kiyotaka; Obara, Naoshi; Ogawa, Shinichi; Sakamoto, Noriaki; Sato, Taiki; Nohara, Seitaro; Tamaoka, Akira

doi:10.2169/internalmedicine.56.7329

Cited by 10 publications

(8 citation statements)

References 23 publications

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“…The two cases reported here showed negative imaging, which made the diagnosis more difficult. The most common histological feature was the infiltration of CD3-positive T cell-dominant inflammatory cells in the perivascular space or within the vessel wall, whereas only scattered infiltrates were observed in the brain parenchyma[ 19 ]. Most of these inflammatory cells were CD8-positive cytotoxic T cells[ 20 ].…”

Section: Discussionmentioning

confidence: 99%

“…The MRI of leukoencephalopathy involves symmetric, high-intensity lesions in the white matter on T2-weighted imaging and fluid-attenuated inversion recovery. Punctate and curvilinear gadolinium enhancement can be seen along the path of the perforating medullary arteries[ 11 , 19 ]. We know that leukoencephalopathy can also result from many immunosuppressants, radiation therapy, and opportunistic infections after HSCT[ 5 ].…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Noninvasive tools based on immune biomarkers for the diagnosis of central nervous system graft-vs-host disease: Two case reports and a review of the literature

Lyu

Hao

et al. 2021

WJCC

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BACKGROUND Central nervous system graft- vs -host disease (CNS-GVHD) is a rare cause of CNS disorders after allogeneic hematopoietic stem cell transplantation. Currently, establishing a diagnosis of CNS-GVHD is challenging because the diagnostic criteria and diagnostic methods are not well defined and many confounding factors need to be ruled out. CASE SUMMARY Here, we present two patients with CNS-GVHD. Both patients with a history of acute GVHD or chronic GVHD developed neurological symptoms that could not be explained by other causes, and had abnormal cerebrospinal fluid (CSF) studies as determined by CSF and blood immune biomarker examinations, suggestive of suspected CNS-GVHD. Due to the lack of specific magnetic resonance imaging abnormalities and the rapid clinical deterioration of the patients, we did not attempt to perform a brain biopsy, but prompted the initiation of empirical immunosuppressive therapy. In view of the rapid and favorable response to local and systematic immunosuppressive treatment and the aforementioned neurologic manifestations together with CSF abnormalities and other negative findings, a final diagnosis of CNS-GVHD was made. CONCLUSION CSF and blood immune biomarker examinations facilitated the diagnosis of CNS-GVHD, which are particularly suitable for patients who are critically ill and require urgent treatment and for those who are unsuitable for invasive diagnostic procedures.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Noninvasive tools based on immune biomarkers for the diagnosis of central nervous system graft-vs-host disease: Two case reports and a review of the literature

Lyu

Hao

et al. 2021

WJCC

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“…Secondary vasculitis due to chronic graft versus host disease after haematopoietic stem cell transplantation may also show military enhancement on brain MRI. Terada et al [27] and Sostak et al [32] described a brain miliary enhancement along the path of the PVS likely due to the small vessel disruption, either by the direct injury of the endothelial cells or by angiocentric infiltrates composed of various combinations of T lymphocytes, B lymphocytes and histiocytes.…”

Section: Primary and Secondary Cns Vasculitismentioning

confidence: 99%

“…The two CNS vasculitis described with brain miliary enhancement at MRI are primary angiitis of the central nervous system (PACNS) and secondary CNS vasculitis due to chronic graft versus host disease [26,27].…”

Section: Primary and Secondary Cns Vasculitismentioning

confidence: 99%

Brain miliary enhancement

et al. 2020

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Purpose Miliary enhancement refers to the presence of multiple small, monomorphic, enhancing foci on T1-weighted postcontrast MRI images. In the absence of a clear clinical presentation, a broad differential diagnosis may result in invasive procedures and possibly brain biopsy for diagnostic purposes. Methods An extensive review of the literature is provided for diseases that may present with miliary enhancement on T1weighted brain MR images. Additional disease-specific findings, both clinical and radiological, are summarized and categorized by the presence or absence of perivascular space involvement. Results Miliary pattern of enhancement may be due to a variety of underlying causes, including inflammatory, infectious, nutritional or neoplastic processes. The recognition of disease spread along the perivascular spaces in addition to the detection or exclusion of disease-specific features on MRI images, such as leptomeningeal enhancement, presence of haemorrhagic lesions, spinal cord involvement and specific localisation or systemic involvement, allows to narrow the potential differential diagnoses. Conclusion A systematic approach to disease-specific findings from both clinical and radiological perspectives might facilitate diagnostic work-up, and recognition of disease spread along the perivascular spaces may help narrowing down differential diagnoses and may help to minimize the use of invasive diagnostic procedures.

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“…Common targets are the skin, gastrointestinal tract and liver, especially in the acute form of GVHD. Progression to chronic form may theoretically affect any organ and is characterized by clinical manifestations resembling those observed in autoimmune disorders (Jagasia et al, 2015, Pechey et al, 2015, Terada et al, 2017. Few cases of GVHD of the central nervous system (CNS-GVHD) have been reported so far (Ruggiu et al, 2017).…”

Section: Introductionmentioning

confidence: 99%

Neuronal surface antibody-mediated encephalopathy as manifestation of chronic graft-versus-host disease after allogeneic hematopoietic stem cell transplantation

Pirotte

Forte

Lutteri

et al. 2018

Journal of Neuroimmunology

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Although it remained controversial for a long time, central nervous system (CNS) involvement of graft-versus-host disease (GVHD) is now becoming recognized as a real nosological entity. Previous case reports have suggested heterogeneous clinical presentations and it is not excluded that the whole spectrum of manifestations has not yet been fully described. Here, we report the case of a 58-year-old man with chronic GVHD who developed a rapidly ingravescent encephalopathy. There was no evidence for CNS immune-mediated lesions on conventional imaging nor for cellular infiltration in the cerebrospinal fluid. Serum analyses revealed the presence of anti-neuronal antibodies directed against anti-contactin-associated protein 2 (anti-Caspr2), a protein associated with voltage-gated potassium neuronal channels. Functional imaging with 2-deoxy-2-[fluorine-18] fluoro- d-glucose integrated with computed tomography (18F-FDG PET-CT) demonstrated diffuse cortical and subcortical hypometabolism. The patient was treated with a combination of immunosuppressive agents (corticosteroids, cyclophosphamide and rituximab) and progressively recovered normal neurocognitive functions. Taken together, these data suggest that CNS-GVHD may manifest as a reversible antibody-mediated functional encephalopathy. This report suggests for the first time the interest of screening for anti-neuronal antibodies and functional imaging with brain 18F-FDG PET-CT in diagnosing this severe complication of allogeneic hematopoietic cell transplantation (alloHSCT).

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Chronic Graft-versus-host Disease Presenting with Multiple Punctate Intracranial Lesions on Contrast-enhanced Magnetic Resonance Imaging

Cited by 10 publications

References 23 publications

Noninvasive tools based on immune biomarkers for the diagnosis of central nervous system graft-vs-host disease: Two case reports and a review of the literature

Noninvasive tools based on immune biomarkers for the diagnosis of central nervous system graft-vs-host disease: Two case reports and a review of the literature

Brain miliary enhancement

Neuronal surface antibody-mediated encephalopathy as manifestation of chronic graft-versus-host disease after allogeneic hematopoietic stem cell transplantation

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