Background Follow-up studies of infants born prematurely are essential to understand the long-term consequences of preterm birth and the efficacy of interventions delivered in the neonatal period. Retention of participants for follow-up studies, however, is challenging, with attrition rates of up to 70%. Our aim was to examine retention rates in two follow-up studies of prematurely born children and identify participant or study characteristics that were associated with higher attrition, and to discuss retention strategies with regard to the literature. Methods Data from children recruited at birth to one of two studies of prematurely born infants were assessed. The two studies were the United Kingdom Oscillation Study (UKOS, a randomised study comparing two modes of neonatal ventilation in infants born less than 29 weeks of gestational age (GA)), and an observational study examining the impact of viral lower respiratory tract infections in infancy in those born less than 36 weeks of GA (virus study). The UKOS participants, but not those in the virus study, had regularly been contacted throughout the follow-up period. UKOS subjects were followed up at 11 to 14 years of age and subjects in the virus study at 5–7 years of age. At follow up in both studies, pulmonary function and respiratory morbidity were assessed. Retention rates to follow-up in the two studies and baseline characteristics of those who were and were not retained were assessed. Results Retention was significantly higher in UKOS than the virus study (61% versus 35%, p < 0.0001). Subjects lost to UKOS follow up had greater deprivation scores ( p < 0.001), a greater likelihood of intrauterine tobacco exposure ( p = 0.001) and were more likely to be of non-white ethnicity ( p < 0.001). In the virus study, those lost to follow-up had higher birth weights ( p = 0.036) and were less likely to be oxygen dependent at hospital discharge ( p = 0.003) or be part of a multiple birth ( p = 0.048). Conclusions Higher retention was demonstrated when there was regular contact in the follow-up period. Both social factors and initial illness severity affected the retention into follow-up studies of prematurely born infants, though these factors were not consistent across the two studies.

Pulmonary rehabilitation (PR) is a highly effective intervention for COPD, though lack of enjoyment and preconceptions about exercise impact on uptake, completion and maintenance of physical activity post-PR. We piloted dance classes led by an experienced choreographer and a trained dancer as an exercise modality for 5 patients with COPD (61-76 years, 3 female, GOLD 2-3). The intervention, comprising 1 hour of stretching, posture, balance, coordination and strength exercises based on a variety of movement and dance techniques and styles at low to high intensity, was trialled in 1 session and refined over 2 further sessions. During the latter 2 sessions heart rate (HR) was recorded; dyspnoea and leg fatigue numerical rating scale (NRS) values taken at 4 points during the class; participants rated mood, sense of group cohesion and enjoyment.Peak HR reached 79-95% of predicted maximum, and was maintained above 70%max for median 26% of class duration. Individual participants' peak NRS values for dyspnoea ranged from 4-9, and for leg fatigue from 4-8. All participants showed improved mood (from median (range) 5 (4-7) to 7 (5-7)), sense of group cohesion (6 (3-6) to 7 (5-7)), deemed the sessions worthwhile (7 (6-7)), and expressed enjoyment (7 (6-7)) and a sense of achievement (6 (3-7)) on completion. Qualitative data indicated participants underestimated class duration by up to 15%, and that concentration on technique helped divert attention away from dyspnoea.Expert-led dance sessions may provide a suitable intensity of training stimulus to improve strength and fitness in individuals with COPD, with potential wider psychosocial benefits. A larger randomised trial is warranted to compare dance-based exercise to conventional PR.

Background X-linked myotubular myopathy (XLMTM) is a life-threatening congenital myopathy that, in most cases, is characterized by profound muscle weakness, respiratory failure, need for mechanical ventilation and gastrostomy feeding, and early death. Objective We aimed to characterize the neuromuscular, respiratory, and extramuscular burden of XLMTM in a prospective, longitudinal study. Methods Thirty-four participants <  4 years old with XLMTM and receiving ventilator support enrolled in INCEPTUS, a prospective, multicenter, non-interventional study. Disease-related adverse events, respiratory and motor function, feeding, secretions, and quality of life were assessed. Results During median (range) follow-up of 13.0 (0.5, 32.9) months, there were 3 deaths (aspiration pneumonia; cardiopulmonary failure; hepatic hemorrhage with peliosis) and 61 serious disease-related events in 20 (59%) participants, mostly respiratory (52 events, 18 participants). Most participants (80%) required permanent invasive ventilation (>16 hours/day); 20% required non-invasive support (6–16 hours/day). Median age at tracheostomy was 3.5 months (95% CI: 2.5, 9.0). Thirty-three participants (97%) required gastrostomy. Thirty-one (91%) participants had histories of hepatic disease and/or prospectively experienced related adverse events or laboratory or imaging abnormalities. CHOP INTEND scores ranged from 19–52 (mean: 35.1). Seven participants (21%) could sit unsupported for≥30 seconds (one later lost this ability); none could pull to stand or walk with or without support. These parameters remained static over time across the INCEPTUS cohort. Conclusions INCEPTUS confirmed high medical impact, static respiratory, motor and feeding difficulties, and early death in boys with XLMTM. Hepatobiliary disease was identified as an under-recognized comorbidity. There are currently no approved disease-modifying treatments.

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