Biologics in vasculitides: Where do we stand, where do we go from now?

Pazzola, Giulia; Muratore, Francesco; Pipitone, Nicolò; Salvarani, Carlo

doi:10.1016/j.lpm.2015.04.010

Cited by 5 publications

(2 citation statements)

References 121 publications

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“…Some case reports showed controversial data about MDS-related SIAD remission with biologics, such as a case of vasculitis associated with arthralgia that responded to TNF-α inhibitors (5-7)(10). Even though we reported on various SIADs, the biologics efficacy seems to be much lower in MDS-related disorders than non MDS-related SIADs (16). Intravenous immunoglobulins are widely used in the setting of various immune disorders, in particular as salvage therapy in immune thrombocytopenia, inflammatory myositis or adult Still's disease (17).…”

Section: Efficacy Of Different Biologics and Azacytidinementioning

confidence: 87%

Biologics in myelodysplastic syndrome-related systemic inflammatory and autoimmune diseases: French multicenter retrospective study of 29 patients

Mékinian

Dervin

Lapidus

et al. 2017

Autoimmunity Reviews

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Section: Efficacy Of Different Biologics and Azacytidinementioning

confidence: 87%

Biologics in myelodysplastic syndrome-related systemic inflammatory and autoimmune diseases: French multicenter retrospective study of 29 patients

Mékinian

Dervin

Lapidus

et al. 2017

Autoimmunity Reviews

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“…However, this approach has not been effective for large vessel vasculitis due to giant cell arteritis as demonstrated in randomized controlled trials or a prospective trial for granulomatosis with polyangiitis [31, 40]. Adalimumab stabilized our patient’s cerebral vasculitis without remarkably reversing progressive arteriopathy.…”

Section: Discussionmentioning

confidence: 91%

Tocilizumab reverses cerebral vasculopathy in a patient with homozygous SAMHD1 mutation

Henrickson

Wang

2017

Clin Rheumatol

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An auto-inflammatory syndrome consequent to SAMHD1 mutations involves cerebral vasculopathy characterized by multifocal stenosis and aneurysms within large arteries, moyamoya, chronic ischemia, and early-onset strokes (SAMS). While this condition involves the innate immune system, additional clinical features mimic systemic lupus erythematosus. Mutations in this gene can also cause a subset of the rare genetic condition Aicardi-Goutières syndrome. To date, no established therapy successfully prevents disease progression. We report a corticosteroid-dependent SAMS patient, a 19-year-old male of Old Order Amish ancestry, with diffuse cerebral arteriopathy identified through contrast brain magnetic resonance arteriography (MRA) and MRI. He received subcutaneous adalimumab every 2 weeks for 9 months with minimal response. Then, he started intravenous tocilizumab (6 mg/kg/dose) every 4 weeks. He sustained steadily normalizing cerebral vasculopathy and lab abnormalities resolved, allowing prednisone reduction. We conclude that the cerebral vasculopathy of the homozygous SAMHD1 mutation-mediated auto-inflammatory disease SAMS responded favorably to tocilizumab infusion therapy.

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Vasculitis research: Don’t slow down and plan for a life-time commitment

Pagnoux

Guillevin²

2015

La Presse Médicale

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Biologics in vasculitides: Where do we stand, where do we go from now?

Cited by 5 publications

References 121 publications

Biologics in myelodysplastic syndrome-related systemic inflammatory and autoimmune diseases: French multicenter retrospective study of 29 patients

Biologics in myelodysplastic syndrome-related systemic inflammatory and autoimmune diseases: French multicenter retrospective study of 29 patients

Tocilizumab reverses cerebral vasculopathy in a patient with homozygous SAMHD1 mutation

Vasculitis research: Don’t slow down and plan for a life-time commitment

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