Alone-Together: Shelves as Intergenerational Maps of Sense-Laden, Relational, Multimodal Pedagogies

Engaging with the concept of sensory shelfies, this essay exhibits how children and adults move across and between sounds, images and objects to make meaning and to tell stories. We glance beyond boundaries and imagine the notion of the shelf as an ongoing mapping of self where layers of experience enmesh and superimpose, and where our sense of self unfolds in the in-between, liminal spaces. These twelve shelves multimodally depict the porosity and fissures that opened up as we moved fluidly between online-offline spaces alone-together. Putting into practice an experimental and speculative approach to our research (Truman et al, 2020; Springgay and Truman, 2018), we argue for these methods as pedagogies that engage with the dynamic complexity of spaces of self. The point of departure for this researc

Alone-together:intergenerational mapping of digital and analogue spaces of self

The project featured in this article experiments with mapping methods as part of a research-creation approach to exploring spaces, times, and movements within materialisations of self. Bringing together adults and children across two cities during lockdown, the project problematises a stance on ‘learning loss’ during the pandemic and instead focuses on the potential of the experiential blurriness of analogue and digital spaces. Rather than seeking to control and structure online learning – thereby denying and limiting its possibilities, explorations, and senses of self – three researchers set out on a speculative approach that acknowledges the dynamic complexity of physical and virtual ways of knowing and being. The article discusses the affordances and challenges that the methodology offers and concludes with the broader implications of this research for reimagined post-pandemic pedagogies. In the end, we advocate for mapping as a way of generously creating spaces and activating meaning-making in diverse learning contexts

COVID-19 and X-linked agammaglobulinemia (XLA) - insights from a monogenic antibody deficiency

Purpose of review The clinical outcomes from COVID-19 in monogenic causes of predominant antibody deficiency have pivotal implications for our understanding of the antiviral contribution of humoral immunity. This review summarizes the lessons learned from COVID-19 infection in X-linked agammaglobulinemia (XLA) due to genetic defects in Bruton's tyrosine kinase (BTK). Recent findings Key molecular pathways underlying the development of severe COVID-19 are emerging, highlighting the possible contribution of BTK to hyperinflammation. SARS-CoV-2 specific T-cell responses and complement activation appear insufficient to achieve viral clearance in some B-cell deficient individuals. Whilst appearing efficacious in this group, use of convalescent plasma has been recently associated with the evolution of viral escape variants. Early data suggests individuals with XLA can mount a viral-specific T-cell vaccine response, however, the clinical significance of this is still emerging. Summary In contrast to reports made early in the pandemic, we show XLA patients remain susceptible to severe disease. Persistent infection was common and is likely to carry a significant symptom burden and risk of novel variant evolution. COVID-19 infection in this vulnerable, antibody deficient group due to genetic, therapeutic or disease causes may require prompt and specific intervention for both patient and societal benefit

Haematopoietic stem cell transplant for norovirus-induced intestinal failure in X-linked agammaglobulinemia

Since the first clinical description in 1952, immunoglobulin replacement therapy remains the mainstay of treatment of patients with X-linked agammaglobulinemia (XLA). However, this therapy only replaces IgG isotype and does not compensate for the loss of Bruton tyrosine kinase in non-B-lymphocytes. Patients may still therefore develop complications despite current standard of care. Here, we describe an XLA patient with persistent chronic norovirus infection, refractory to treatment and causing intestinal failure. The patient underwent haematopoietic stem cell transplantation, curing XLA and allowed clearance of norovirus prior to humoral immunoreconstitution, suggesting non-humoral immunodeficiency in these patients

Dimethyl fumarate in patients admitted to hospital with COVID-19 (RECOVERY): a randomised, controlled, open-label, platform trial

Dimethyl fumarate (DMF) inhibits inflammasome-mediated inflammation and has been proposed as a treatment for patients hospitalised with COVID-19. This randomised, controlled, open-label platform trial (Randomised Evaluation of COVID-19 Therapy [RECOVERY]), is assessing multiple treatments in patients hospitalised for COVID-19 (NCT04381936, ISRCTN50189673). In this assessment of DMF performed at 27 UK hospitals, adults were randomly allocated (1:1) to either usual standard of care alone or usual standard of care plus DMF. The primary outcome was clinical status on day 5 measured on a seven-point ordinal scale. Secondary outcomes were time to sustained improvement in clinical status, time to discharge, day 5 peripheral blood oxygenation, day 5 C-reactive protein, and improvement in day 10 clinical status. Between 2 March 2021 and 18 November 2021, 713 patients were enroled in the DMF evaluation, of whom 356 were randomly allocated to receive usual care plus DMF, and 357 to usual care alone. 95% of patients received corticosteroids as part of routine care. There was no evidence of a beneficial effect of DMF on clinical status at day 5 (common odds ratio of unfavourable outcome 1.12; 95% CI 0.86-1.47; p = 0.40). There was no significant effect of DMF on any secondary outcome

The United Kingdom Primary Immune Deficiency (UKPID) registry 2012 to 2017

This is the second report of the United Kingdom Primary Immunodeficiency (UKPID) registry. The registry will be a decade old in 2018 and, as of August 2017, had recruited 4758 patients encompassing 97% of immunology centres within the United Kingdom. This represents a doubling of recruitment into the registry since we reported on 2229 patients included in our first report of 2013. Minimum PID prevalence in the United Kingdom is currently 5·90/100 000 and an average incidence of PID between 1980 and 2000 of 7·6 cases per 100 000 UK live births. Data are presented on the frequency of diseases recorded, disease prevalence, diagnostic delay and treatment modality, including haematopoietic stem cell transplantation (HSCT) and gene therapy. The registry provides valuable information to clinicians, researchers, service commissioners and industry alike on PID within the United Kingdom, which may not otherwise be available without the existence of a well‐established registry