A Case of Multiple Sclerosis—Like Relapsing Remitting Encephalomyelitis Following Allogeneic Hematopoietic Stem Cell Transplantation and a Review of the Published Literature

Das, Joyutpal; Gill, Atta Munawar; Lo, Christine; Chan-Lam, Natalie; Price, Siân; Wharton, Stephen B.; Jessop, Helen; Sharrack, Basil; Snowden, John A.

doi:10.3389/fimmu.2020.00668

Cited by 8 publications

(9 citation statements)

References 26 publications

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“…Though neurologic complications following HSCT are relatively common (9%–14%), 4 IMDD are rare. It is a diagnosis made after exclusion of more common conditions with overlapping features: infection, treatment‐related toxicity, and disease relapse.…”

Section: Discussionmentioning

confidence: 99%

“…It is a diagnosis made after exclusion of more common conditions with overlapping features: infection, treatment‐related toxicity, and disease relapse. Currently, there are about 20 case reports of CNS demyelination following HSCT in literature, all in adult patients 4 . In them, the median interval between HSCT and onset of symptoms was 1 year (range 0.1–8 years).…”

Section: Discussionmentioning

confidence: 99%

“…There are 79 reported cases of TD in children till date and none in the posttransplant setting 2 . Some characteristic features that can be noted in these cases are rapid progression of symptoms over a short time period (<1 week), 4 involvement of multiple CNS sites, 5 white matter predilection, smaller mass effect than expected when considering the lesion size, and rapid improvement in MRI appearance with steroid 10 . Most commonly, these lesions are well circumscribed enhancing supratentorial lesions with a predilection for the frontal and parietal lobes.…”

Section: Discussionmentioning

confidence: 99%

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Tumefactive demyelination in pediatrics: An unusual late neurological complication of hematopoietic stem cell transplant

Haldorai

Kamila

Gupta

et al. 2021

Pediatric Blood & Cancer

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Immune‐mediated demyelination is a rare posttransplant complication. Here, we report an 8.5‐year‐old boy who developed left hemiparesis, 18 months post matched sibling donor hematopoietic stem cell transplant (HSCT) for relapsed acute myeloid leukemia and was diagnosed to have tumefactive demyelination. The diagnosis was established based on clinical and radiological features. The complete resolution of the lesions with steroids further established the immune‐mediated pathophysiology.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Tumefactive demyelination in pediatrics: An unusual late neurological complication of hematopoietic stem cell transplant

Haldorai

Kamila

Gupta

et al. 2021

Pediatric Blood & Cancer

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“…Patients with CNS-GVHD were reported to respond to high dose corticosteroids, intravenous immunoglobulin treatments, immunosuppressive medications including methotrexate and etoposide ( 38 ). Chronic CNS-GVHD is a late complication of allo-HCT and clinical manifestations may include myasthenia gravis, myositis, demyelination, angiitis ( 39 , 40 ). Patients can also present with stroke-like episodes, lacunar syndromes, multiple sclerosis-like presentations or encephalitis ( 30 ).…”

Section: Human Studies On Cns-gvhdmentioning

confidence: 99%

Acute Graft-Versus-Host Disease, Infections, Vascular Events and Drug Toxicities Affecting the Central Nervous System

Vinnakota

Zeiser

2021

Front. Immunol.

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Allogeneic hematopoietic cell transplantation (allo-HCT) is a curative therapy for patients with hematological malignancies. Acute Graft versus host diseases (GVHD) is a major immune mediated side effect of allo-HCT that can affect the central nervous system (CNS) in addition to post-allo-HCT vascular events, drug toxicity or infections. Here we summarize and discuss recent preclinical data on the CNS as a target of acute GVHD and the known mechanisms contributing to neurotoxicity with a focus on microglia and T cells. We also discuss open questions in the field and place the findings made in mouse models in a clinical context. While in mice the neurological deficits can be assessed in a controlled fashion, in patients the etiology of the CNS damage is difficult to attribute to acute GVHD versus infections, vascular events, and drug-induced toxicity. Ultimately, we discuss novel therapies for GVHD of the CNS. Our understanding of the biological mechanisms that lead to neurotoxicity after allo-HCT increased over the last decade. This review provides insights into CNS manifestations of GVHD versus other etiologies of CNS damage in mice and patients.

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“…Central nervous system (CNS) complications can occur in 9%–15% of patients after allogeneic hematopoietic stem cell transplantations (allo-HSCT) ( Schmidt-Hieber et al., 2016 ; Das et al., 2020 ) with the common causes being mainly the posterior reversible encephalopathy syndrome (PRES), transplant-related thrombotic microangiopathies (TA-TMA), CNS graft versus host disease (CNS-GVHD), CNS infiltration of malignant disease, CNS infections and non-specific neurological symptoms ( Chaudhary et al., 2017 ; Maffini et al., 2017 ; Balaguer-Rosello et al., 2019 ). Furthermore, the clinical manifestations of CNS complications are non-specific, with most of them being disturbances of consciousness, convulsions, headaches, fever, and epilepsy.…”

Section: Introductionmentioning

confidence: 99%

Metagenomic Next Generation Sequencing in the Detection of Pathogens in Cerebrospinal Fluid of Patients After Alternative Donor Transplantation: A Feasibility Analysis

Zhang

Zhou

Gui

et al. 2021

Front. Cell. Infect. Microbiol.

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Central nervous system (CNS) complications can occur in 9%–15% of patients after allogeneic hematopoietic stem cell transplantation (allo-HSCT). The clinical manifestations of the CNS complications are non-specific, with most of them being disturbances of consciousness, convulsions, headaches, fever, and epilepsy, making it difficult to infer the cause of the complications based on clinical manifestations. We retrospectively analyzed the sensitivity and feasibility of metagenomic next generation sequencing (mNGS) in the diagnosis of CNS infections after allo-HSCT. Lumbar punctures were performed on 20 patients with CNS symptoms after receiving alternative donor HSCT(AD-HSCT) at the Affiliated Cancer Hospital of Zhengzhou University from February 2019 to December 2020, and their cerebrospinal fluid (CSF) was collected. The mNGS technique was used to detect pathogens in the CSF. Routine CSF testing, biochemical analyses, G experiments, GM experiments, ink staining, acid-fast staining, and bacterial cultures were carried out, and quantitative PCR (qPCR) tests were used to detect cytomegalovirus (CMV), Epstein-Barr virus (EBV), BK polyomavirus (BKPyV), and human alphaherpesvirus (HHV). A total of 29 tests were performed with 21 of them being positive. Of the five negative patients, three were diagnosed with a posterior reversible encephalopathy syndrome, one as having transplantation-associated thrombotic microangiopathy, and one with transient seizure caused by hypertension. Fifteen patients tested positive, of which four had single infections and eleven had mixed infections. Five cases of fungal infections, six cases of bacterial infections, and 13 cases of viral infections were detected. Among the 13 cases of viral infections, ten cases were CMV(HHV-5); three were BKPyV; two were Torque teno virus (TTV); Two were HHV-1,two were EBV(HHV4), and one each of HpyV5 and HHV-6B. Thirteen patients tested positive for virus while the qPCR detection method of 6 identical specimens were below the minimum detection limit(<1×103 U/ml). The mNGS technique is highly sensitive, and it can be used to diagnose CNS infections after allo-HSCT.

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A Case of Multiple Sclerosis—Like Relapsing Remitting Encephalomyelitis Following Allogeneic Hematopoietic Stem Cell Transplantation and a Review of the Published Literature

Cited by 8 publications

References 26 publications

Tumefactive demyelination in pediatrics: An unusual late neurological complication of hematopoietic stem cell transplant

Tumefactive demyelination in pediatrics: An unusual late neurological complication of hematopoietic stem cell transplant

Acute Graft-Versus-Host Disease, Infections, Vascular Events and Drug Toxicities Affecting the Central Nervous System

Metagenomic Next Generation Sequencing in the Detection of Pathogens in Cerebrospinal Fluid of Patients After Alternative Donor Transplantation: A Feasibility Analysis

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