Progressive multifocal leukoencephalopathy in patients with sarcoidosis

Lefaucheur, Romain; Jamilloux, Yvan; Ahtoy, Patrick; Bouwyn, Jean Paul; Maltête, David; Bourre, Bertrand; Néel, A.; Broussolle, C.; Hamidou, M.; Sève, P.

doi:10.1212/wnl.0000000000000902

Cited by 2 publications

(3 citation statements)

References 7 publications

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“…Risk factors for infection included extra-thoracic sarcoidosis and treatment with corticosteroids. [ 16 ] More recently, Lefaucheur et al [ 18 ] reported 10 cases of progressive multifocal encephalopathy associated with sarcoidosis and 20 observations from literature. The mean CD4 lymphocyte cell count was 235 ± 142 mm 3 ), 10 patients were naive of treatment, and 17 (57%) patients died.…”

Section: Discussionmentioning

confidence: 99%

Severe infections in sarcoidosis

Dureault

Chapelon

Biard³

et al. 2017

Medicine

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Sarcoidosis is associated with cell-mediated immunodeficiency and treatment of symptomatic sarcoidosis usually includes systemic immunosuppressants. Data relative to incidence, prognosis factors, and outcome of infections are scarce.Retrospective cohort study of 585 patients with biopsy proven sarcoidosis in a tertiary referral specialist clinic, with a nested case-control analysis. Twenty nine patients (4.9%) with severe infections were compared to 116 controls subjects with sarcoidosis, matched according to their gender, ethnicity, age at diagnosis, and treatment with corticosteroids.After a median follow-up of 8 years [range; 1–46], 38 severe infections [mycobacterial infections (n = 14), fungal infections (n = 10), bacterial (n = 8), viral (n = 3) and parasitic (n = 1)] were observed in 30 patients. The incidence of severe infections was 0.71% persons-year (CI 95% 0.5–0.98) and 0.43% persons-year (CI 95% 0.27–0.66). Patients with severe infection were more frequently of male gender (60% vs 46%) and were more likely treated by ≥ 3 immunosuppressive agents (OR = 3.8, IC 95% [1.5–9.64], P = .005) and by cyclophosphamide (OR = 5.55, IC 95% [1.9–16.1], P = .002), and with neurological (OR = 3.36 CI 95% [1.37–8.25], P = .008), or cardiac (OR = 2.65 CI 95% [1.09–6.43], P = .031) involvement of the sarcoidosis, compared to the controls. Two patients died within the 6 months following infection, due to progressive multifocal leucoencephalopathy (n = 1), and of peritonitis (n = 1).Severe infections are observed in 5.1% of our patients with sarcoidosis after a median follow-up of 8 years. Risk factors for severe infections included neurological or cardiac involvement of sarcoidosis, the use of immunosuppressive agents and mainly cyclophosphamide.

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Section: Discussionmentioning

confidence: 99%

Severe infections in sarcoidosis

Dureault

Chapelon

Biard³

et al. 2017

Medicine

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“…First attempts used IL‐2 to foster the reconstitution of CD4 + T cells. However, this strategy showed no clinical or biological efficacy 53 …”

Section: Restoration Of Immunity As a Treatment For Pmlmentioning

confidence: 97%

“…However, this strategy showed no clinical or biological efficacy. 53 IL-7 is a pivotal cytokine in T-cell biology. IL-7 stimulates thymic differentiation and homeostatic expansion of naïve peripheral T cells.…”

Section: Interleukinsmentioning

confidence: 99%

Advances in Treatment of Progressive Multifocal Leukoencephalopathy

Bernard‐Valnet

Koralnik

Pasquier

2021

Annals of Neurology

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Progressive multifocal encephalopathy (PML) is a severe demyelinating disease of the central nervous system (CNS) caused by JC virus (JCV), which occurs in immunocompromised individuals. Management of PML relies on restoration of immunity within the CNS. However, when this restoration cannot be readily achieved, PML has a grim prognosis. Innovative strategies have shown promise in promoting anti‐JCV immune responses, and include T‐cell adoptive transfer or immune checkpoint inhibitor therapies. Conversely, management of immune reconstitution inflammatory syndrome, particularly in iatrogenic PML, remains a major challenge. In this paper, we review recent development in the treatment of PML. ANN NEUROL 2021;90:865–873

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Progressive multifocal leukoencephalopathy in patients with sarcoidosis

Cited by 2 publications

References 7 publications

Severe infections in sarcoidosis

Severe infections in sarcoidosis

Advances in Treatment of Progressive Multifocal Leukoencephalopathy

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