<scp>COVID</scp>‐19 and lymphangioleiomyomatosis: Experience at a reference center and the potential impact of the use of <scp>mTOR</scp> inhibitors

Baldi, Bruno Guedes; Colares, Philippe de Figueiredo Braga; Kairalla, Ronaldo Adib; Oliveira, Martina Rodrigues de; Carvalho, Carlos Roberto Ribeiro

doi:10.1002/ajmg.a.61877

Cited by 7 publications

(9 citation statements)

References 5 publications

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“…10 In a small series from Brazil of six patients with lymphangioleiomyomatosis who received a diagnosis of COVID-19, half were using sirolimus, and all completely recovered from the infection. 5 In the present study, the use of mTOR inhibitors was not associated with worse outcomes, despite these patients having poorer lung function than untreated patients, which should provide reassurance to patients with lymphangioleiomyomatosis and clinicians.…”

Section: Discussionsupporting

confidence: 54%

“…[2][3][4] However, only a small number of patients with lymphangioleiomyomatosis and COVID-19 have been reported, and the outcomes and impact of mTOR inhibitors in lymphangioleiomyomatosis and COVID-19 infection are unclear. [5][6][7] We conducted an international study to evaluate the consequences of COVID-19 in patients with lymphangioleiomyomatosis and the impact of mTOR inhibitors on outcomes after COVID-19.…”

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confidence: 99%

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COVID-19 in Lymphangioleiomyomatosis

Baldi

Radzikowska²,

Cottin

et al. 2022

Chest

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Lymphangioleiomyomatosis is a rare low-grade neoplastic lung disease that occurs sporadically in women or in people with tuberous sclerosis complex. Mechanistic target of rapamycin (mTOR) inhibitors are used for patients with progressive or severe pulmonary impairment or extrapulmonary manifestations. 1 COVID-19 is associated with worse outcomes in patients with chronic pulmonary diseases. [2][3][4] However, only a small number of patients with lymphangioleiomyomatosis and COVID-19 have been reported, and the outcomes and impact of mTOR inhibitors in lymphangioleiomyomatosis and COVID-19 infection are unclear. [5][6][7] We conducted an international study to evaluate the consequences of COVID-19 in patients with lymphangioleiomyomatosis and the impact of mTOR inhibitors on outcomes after COVID-19. MethodsWe conducted a retrospective observational study that assessed patients with lymphangioleiomyomatosis receiving care at lymphangioleiomyomatosis clinics in Brazil, the United States, Europe, and Japan who contracted COVID-19 between December 1, 2019, and August 31, 2021. All local institutional ethical committees approved the study. Demographicand clinical features associated with lymphangioleiomyomatosis, risk factors for poor outcomes resulting from COVID-19, pulmonary function tests closest to COVID-19, use of mTOR inhibitors, symptoms, details if hospitalized, treatments received, and outcomes related to COVID-19 were obtained through medical records and telephone follow-up. Long COVID-19 was defined as persisting symptoms or new supplemental oxygen use at least 6 weeks after the onset of symptoms.

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Section: Discussionsupporting

confidence: 54%

mentioning

confidence: 99%

COVID-19 in Lymphangioleiomyomatosis

Baldi

Radzikowska²,

Cottin

et al. 2022

Chest

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“… 130 In agreement with this hypothesis, hospitalized patients with critical COVID-19 treated with NAC showed significant clinical improvement and reduction of inflammation markers, suggesting a beneficial effect of NAC in COVID-19, possibly mediated by mTOR inhibition. 131 Limited observational studies of patients with tuberous sclerosis complex (TSC) and/or lymphangioleiomyomatosis (LAM) on long-term mTOR inhibitors (sirolimus or everolimus) have not identified an increased risk of COVID-19 or poor outcomes of the infection 132 , 133 and clinical trials of mTOR inhibitors in COVID-19 are ongoing. However, whether activation of the mTOR pathway plays a role in SARS-CoV-2-infected patients with SLE remains to be elucidated.…”

Section: Convergent Immunopathogenic Mechanisms Of Covid-19 and Slementioning

confidence: 99%

COVID-19 in patients with systemic lupus erythematosus: lessons learned from the inflammatory disease

Fernández-Ruiz

Paredes

Niewold

2021

Translational Research

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As the world navigates the Coronavirus disease 2019 (COVID-19) pandemic, there is a growing need to assess its impact in patients with autoimmune rheumatic diseases, such as systemic lupus erythematosus (SLE). Patients with SLE are a unique population when considering the risk of contracting COVID-19 and infection outcomes. The use of systemic glucocorticoids and immunosuppressants, and underlying organ damage from SLE are potential susceptibility factors. Most patients with SLE have evidence of high type I IFN activity, which may theoretically act as an antiviral line of defense or contribute to the development of a deleterious hyperinflammatory response in COVID-19. Other immunopathogenic mechanisms of SLE may overlap with those described in COVID-19, and thus studies in SLE could provide some insight into immune responses occurring in severe cases of the viral infection. We reviewed the literature to date on COVID-19 in patients with SLE and provide an in-depth review of current research in the area, including immune pathway activation, epidemiology, clinical features, outcomes, and the psychosocial impact of the pandemic in those with autoimmune disease.

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“…Likewise, the use of mTOR inhibitors (e.g. Everolimus) in the treatment of tuberous sclerosis doesn’t seem related to a greater risk of infection neither to a more severe course of COVID-19 [ 83 ].…”

Section: Methodsmentioning

confidence: 99%

Eligibility criteria for pediatric patients who may benefit from anti SARS-CoV-2 monoclonal antibody therapy administration: an Italian inter-society consensus statement

et al. 2022

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The fast diffusion of the SARS-CoV-2 pandemic have called for an equally rapid evolution of the therapeutic options.The Human recombinant monoclonal antibodies (mAbs) have recently been approved by the Food and Drug Administration (FDA) and by the Italian Medicines Agency (AIFA) in subjects aged ≥12 with SARS-CoV-2 infection and specific risk factors.Currently the indications are specific for the use of two different mAbs combination: Bamlanivimab+Etesevimab (produced by Eli Lilly) and Casirivimab+Imdevimab (produced by Regeneron).These drugs have shown favorable effects in adult patients in the initial phase of infection, whereas to date few data are available on their use in children.AIFA criteria derived from the existing literature which reports an increased risk of severe COVID-19 in children with comorbidities. However, the studies analyzing the determinants for progression to severe disease are mainly monocentric, with limited numbers and reporting mostly generic risk categories.Thus, the Italian Society of Pediatrics invited its affiliated Scientific Societies to produce a Consensus document based on the revision of the criteria proposed by AIFA in light of the most recent literature and experts’ agreement.This Consensus tries to detail which patients actually have the risk to develop severe disease, analyzing the most common comorbidities in children, in order to detail the indications for mAbs administration and to guide the clinicians in identifying eligible patients.

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COVID‐19 and lymphangioleiomyomatosis: Experience at a reference center and the potential impact of the use of mTOR inhibitors

Cited by 7 publications

References 5 publications

COVID-19 in Lymphangioleiomyomatosis

COVID-19 in Lymphangioleiomyomatosis

COVID-19 in patients with systemic lupus erythematosus: lessons learned from the inflammatory disease

Eligibility criteria for pediatric patients who may benefit from anti SARS-CoV-2 monoclonal antibody therapy administration: an Italian inter-society consensus statement

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