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Abstract Background Hybrid models that integrate both in-person and remote health services are increasingly recognized as a promising approach. Nevertheless, research that defines and characterizes these models in children and young people is scarce and essential for establishing guidelines for implementation of hybrid allied health services. This scoping review evaluates four key aspects of hybrid allied health services in children and young people: 1. definitions, 2. service characteristics, 3. outcome measures, and 4. results of hybrid allied health services. Methods Six databases were searched: Medline (Ovid), Embase, CINHAL, Psycinfo, Cochrane CENTRAL, and Web of Science. Of the 9,868 studies potentially meeting the inclusion criteria, 49 studies focused on children and young people. Following full-text review, n = 21 studies were included. Results Terminology used for hybrid allied health services varied across studies which targeted diverse clinical populations and varied in study design, type and frequency of remote and in-person treatments. Over 75% of cases used custom-written software, limiting scalability. All interventions started in-person, possibly to establish a therapeutic alliance and solve technological issues. Most hybrid allied health services (67%) were in mental health, while only a minority involved physical, occupational or speech therapy. The most common outcomes were feasibility and satisfaction, but tools used to measure them were inconsistent. Although 57% of studies demonstrated effectiveness of hybrid allied health services, none measured cost-effectiveness. Discussion Despite the potential of hybrid allied health services for children and young people, the literature remains at a preliminary stage. Standardization of definitions and outcome measures, and clearer reporting of service characteristics and results would likely promote consolidation of hybrid allied health services in children and young people into clinical practice.

Abstract Background Microbial methane oxidation, methanotrophy, plays a crucial role in mitigating the release of the potent greenhouse gas methane from aquatic systems. While aerobic methanotrophy is a well-established process in oxygen-rich environments, emerging evidence suggests their activity in hypoxic conditions. However, the adaptability of these methanotrophs to such environments has remained poorly understood. Here, we explored the genetic adaptability of aerobic methanotrophs to hypoxia in the methanogenic sediments of Lake Kinneret (LK). These LK methanogenic sediments, situated below the oxidic and sulfidic zones, were previously characterized by methane oxidation coupled with iron reduction via the involvement of aerobic methanotrophs. Results In order to explore the adaptation of the methanotrophs to hypoxia, we conducted two experiments using LK sediments as inoculum: (i) an aerobic "classical" methanotrophic enrichment with ambient air employing DNA stable isotope probing (DNA-SIP) and (ii) hypoxic methanotrophic enrichment with repeated spiking of 1% oxygen. Analysis of 16S rRNA gene amplicons revealed the enrichment of Methylococcales methanotrophs, being up to a third of the enriched community. Methylobacter, Methylogaea, and Methylomonas were prominent in the aerobic experiment, while hypoxic conditions enriched primarily Methylomonas. Using metagenomics sequencing of DNA extracted from these experiments, we curated five Methylococcales metagenome-assembled genomes (MAGs) and evaluated the genetic basis for their survival in hypoxic environments. A comparative analysis with an additional 62 Methylococcales genomes from various environments highlighted several core genetic adaptations to hypoxia found in most examined Methylococcales genomes, including high-affinity cytochrome oxidases, oxygen-binding proteins, fermentation-based methane oxidation, motility, and glycogen use. We also found that some Methylococcales, including LK Methylococcales, may denitrify, while metals and humic substances may also serve as electron acceptors alternative to oxygen. Outer membrane multi-heme cytochromes and riboflavin were identified as potential mediators for the utilization of metals and humic material. These diverse mechanisms suggest the ability of methanotrophs to thrive in ecological niches previously thought inhospitable for their growth. Conclusions Our study sheds light on the ability of enriched Methylococcales methanotrophs from methanogenic LK sediments to survive under hypoxia. Genomic analysis revealed a spectrum of genetic capabilities, potentially enabling these methanotrophs to function. The identified mechanisms, such as those enabling the use of alternative electron acceptors, expand our understanding of methanotroph resilience in diverse ecological settings. These findings contribute to the broader knowledge of microbial methane oxidation and have implications for understanding and potential contribution methanotrophs may have in mitigating methane emissions in various environmental conditions.

Damian Chi Hong Siu, Liat Gafni-Lachter Boston University, Sargent College of Health and Rehabilitation Sciences, Boston, MA, USACorrespondence: Damian Chi Hong Siu, Graduate, Post-professional Occupational Therapy Doctorate, Boston University, Sargent College of Health and Rehabilitation Sciences, 635 Commonwealth Ave, Boston, MA, 02215, USA, Email damiansiu@gmail.comAbstract: Chronic obstructive pulmonary disease (COPD) is a preventable yet widespread and profoundly debilitating respiratory condition, exerting substantial personal and global health ramifications alongside significant economic implications. The first objective of this literature review was to identify reviews the barriers to optimal COPD care, categorizing them into personal patient factors, professional awareness and knowledge, patient-professional relationships, and healthcare service models, including access to care that significantly impacts the quality of COPD management. The second objective was to introduce three approaches for enhancing COPD care outcomes: Self-Management Educational Programs, Health Qigong, and Telehealth service provision, each demonstrating positive effects on COPD patients’ health status. These evidence-based interventions offer promising avenues for enhancing COPD care and patient outcomes. Integrating these approaches into comprehensive COPD management strategies holds potential for improving the well-being and quality of life of individuals living with this chronic condition.Keywords: pulmonary disease, chronic obstructive, health qigong, telehealth, self-management education

Jansen metaphyseal chondrodysplasia (JMC) is an ultra-rare disorder caused by germline heterozygous PTHR1 variants resulting in constitutive activation of parathyroid hormone type 1 receptor. A description of ocular manifestations of the disease is lacking. Six patients with JMC underwent a detailed ophthalmic evaluation, spectral-domain optical coherence tomography (OCT), visual field testing, and craniofacial CT scans. Five of 6 patients had good visual acuity. All patients had widely spaced eyes; 5/6 had downslanted palpebral fissures. One patient had proptosis, and another had bilateral ptosis. Two patients had incomplete closure of the eyelids (lagophthalmos), one had a history of progressive right facial nerve palsy with profuse epiphora, while the second had advanced optic nerve atrophy with corresponding retinal nerve fiber layer (RNFL) thinning on OCT and significant bilateral optic canal narrowing on CT scan. Additionally, this patient also had central visual field defects and abnormal color vision. A third patient had normal visual acuity, subtle temporal pallor of the optic nerve head, normal average RNFL, but decreased temporal RNFL and retinal ganglion cell layer analysis (GCA) on OCT. GCA was decreased in 4/6 patients indicating a subclinical optic nerve atrophic process. None of the patients had glaucoma or high myopia. These data represent the first comprehensive report of ophthalmic findings in JMC. Patients with JMC have significant eye findings associated with optic canal narrowing due to extensive skull base dysplastic bone overgrowth that appear to be more prevalent and pronounced with age. Progressive optic neuropathy from optic canal narrowing may be a feature of JMC, and OCT GCA can serve as a useful biomarker for progression in the setting of optic canal narrowing. We suggest that patients with JMC should undergo regular ophthalmic examination including color vision, OCT, visual field testing, orbital, and craniofacial imaging. Lay Summary: Jansen metaphyseal chondrodysplasia (JMC) is an ultra-rare disorder caused by overactivity of the parathyroid hormone type 1 receptor, a receptor that is important for regulating bone development. Some patients report eye problems, but these have not been well-described. We therefore performed extensive eye examinations in 6 individuals with JMC. One had progressive right-sided facial paralysis associated with eye-tearing and dryness. A second individual had decreased central vision. Using optical coherence tomography (OCT), a method which takes detailed pictures of the retina, we found thinning of the retina's nerve layers (RNFL). Computed tomography (CT) of the skull showed narrowing of the bony canal through which the optic nerve passes. A third individual had normal vision but RNFL thinning just on the sides. Analysis of ganglion cells, the nerve cells carrying signals from the retina to the brain, showed localized deterioration of the optic nerve; skull CT also revealed optic canal narrowing. These findings suggest that JMC is associated with progressive damage to the optic nerve. Thus, patients with JMC would benefit from regular eye exams including OCT and visual field testing, in addition to routine imaging. Graphical Abstract [ABSTRACT FROM AUTHOR]

Hyperphosphatemic familial tumoral calcinosis (HFTC) is a rare disorder caused by deficient FGF23 signaling and resultant ectopic calcification. Here, we systematically characterized and quantified macro- and micro-calcification in a HFTC cohort using CT and 18F-sodium fluoride PET/CT (18F-NaF PET/CT). Fourier-transform infrared (FTIR) spectroscopy was performed on 4 phenotypically different calcifications from a patient with HFTC, showing the dominant component to be hydroxyapatite. Eleven patients with HFTC were studied with CT and/or 18F-NaF PET/CT. Qualitative review was done to describe the spectrum of imaging findings on both modalities. CT-based measures of volume (eg, total calcific burden and lesion volume) and density (Hounsfield units) were quantified and compared to PET-based measures of mineralization activity (eg, mean standardized uptake values—SUVs). Microcalcification scores were calculated for the vasculature of 6 patients using 18F-NaF PET/CT and visualized on a standardized vascular atlas. Ectopic calcifications were present in 82% of patients, predominantly near joints and the distal extremities. Considerable heterogeneity was observed in total calcific burden per patient (823.0 ± 670.1 cm3, n = 9) and lesion volume (282.5 ± 414.8 cm3, n = 27). The largest lesions were found at the hips and shoulders. 18F-NaF PET offered the ability to differentiate active vs quiescent calcifications. Calcifications were also noted in multiple anatomic locations, including brain parenchyma (50%). Vascular calcification was seen in the abdominal aorta, carotid, and coronaries in 50%, 73%, and 50%, respectively. 18F-NaF-avid, but CT-negative calcification was seen in a 17-year-old patient, implicating early onset vascular calcification. This first systematic assessment of calcifications in a cohort of patients with HFTC has identified the early onset, prevalence, and extent of calcification. It supports 18F-NaF PET/CT as a clinical tool for distinguishing between active and inactive calcification, informing disease progression, and quantification of ectopic and vascular disease burden. Lay Summary: Hyperphosphatemic familial tumoral calcinosis (HFTC) is a rare disorder in which patients develop sometimes large debilitating calcifications of soft tissues and blood vessels. It is caused by deficient fibroblast growth factor-23 that leads to high phosphate levels, which contributes to the calcifications. The calcifications and manifestations of this disorder have not been well characterized. We determined the mineral composition of the calcifications to be hydroxyapatite. Capitalizing on the fact fluoride can be integrated into hydroxyapatite, we used radiolabeled sodium fluoride PET/CT scans (18F-NaF PET/CT) to characterize and quantify the calcifications in 11 patients. Eighty-two percent of the patients had calcifications, with the largest located at the hips and shoulders. Micro-calcifications were found in the blood vessels of most patients, including children. The technique also enabled us to differentiate between active vs stable calcifications. This first systematic assessment of calcifications in patients with HFTC showed the utility of 18F-NaF PET/CT as a tool to identify and quantify calcifications, as well as distinguish between active and stable calcifications. This approach will inform disease progression and may prove useful for measuring response to treatment. Graphical Abstract [ABSTRACT FROM AUTHOR]