Evaluation of a Telehealth Parent Training Program for Parents of Children with Autism Spectrum Disorder and Sleep Difficulties

Many children with autism spectrum disorder (ASD) experience some form of sleep difficulty (e.g., delayed sleep onset, unwanted co-sleeping, prolonged or frequent night wakings). Although research supports parent-implemented behaviour-analytic sleep interventions to address sleep difficulties in children with ASD (e.g., Jin et al., 2013; Linnehan et al., 2022), more research is needed to determine how accurately parents implement behavioural sleep interventions and the effectiveness of parent training and coaching via telehealth. The present study used a concurrent multiple baseline across participants design to evaluate parents’ ability to implement their child’s behaviour-analytic sleep intervention (i.e., treatment fidelity) and a pre-/post-test design to evaluate parents’ ability to monitor and make decisions related to their child’s sleep (i.e., decision-making accuracy). Parent stress levels were evaluated pre- and post-intervention using the Parenting Stress Index 4th Edition Short Form (PSI-4-SF; Abidin, 2012). Child sleep-related outcomes (e.g., sleep onset delay, occurrences of sleep-interfering behaviours, and total sleep duration) were also monitored. Four parent-child dyads participated; mothers were the primary parent participants. Parents received behavioural skills training and nighttime coaching, via telehealth, over a 12-week intervention period. Overall, results indicate that parents’ treatment fidelity remained high throughout intervention (i.e., >80%). Further, parents’ decision-making accuracy increased from pre-test to post-test and remained at post-test levels during intervention. Two of four parents returned the PSI-4-SF. Results indicate that the intervention did not increase or decrease parent stress levels. Additionally, sleep onset delay decreased for two of four child participants. Occurrences of sleep-interfering behaviours remained variable for all child participants. Total sleep duration increased for two of four child participants. All three children who were co-sleeping at the start of the study were sleeping independently by the end of the study. All parents rated the sleep intervention as positive and acceptable. Strengths, limitations, and areas for future research are discussed

Spontaneous coronary artery dissection associated with incidental finding of left ventricular thrombus

Spontaneous Coronary Artery Dissection (SCAD) is one of the nonatherosclerotic causes of Acute Coronary Syndrome. It’s extremely rare for SCAD to present in an asymptomatic male, with incidental finding of Left Ventricular (LV) thrombus on echocardiogram. This report presents the case of a 36-year-old male with such an atypical presentation of Spontaneous Coronary Artery Dissection with Left Ventricular apical thrombus as a complication. The patient received successful medical management, with excellent clinical outcomes. This case highlights the importance of an early recognition and treatment strategy for both conditions using medical therapy

Transient Ischemic Attack, the Initial Presentation of Azygos to Pulmonary Vein Fistula

There are different sources of cerebral emboli, including cardiac embolism, extracranial arterial embolism, paradoxical embolism, trauma, and iatrogenic embolism. In rare cases, atypical sources should be ruled out. We are reporting a lady who presented with transient ischemic attack and had a fistula between the azygos to the pulmonary vein. (Level of Difficulty: Advanced.

Gadolinium-based coronary angiography in a patient with prior known anaphylaxis to iodine-based dye

Gadodiamide is a gadolinium-based chemical element that is considered safe and well tolerated in patients without renal dysfunction and is therefore routinely used as a contrast agent in magnetic resonance imaging. Although radio-opaque, it is not frequently used for coronary angiography due to its less than optimal image quality and prohibitive cost. Our center’s previous experience was less than satisfactory but the addition of a power injection system yielded good quality diagnostic images. We report a case of 63 years old male with a known history of severe, life-threatening anaphylactic reaction to previous iodinated dye presenting with persistent angina despite optimal medical therapy. Coronary and bypass graft angiography was performed using 24 cc of undiluted Gadodiamide (OMNISCAN) with a power injector (ACIST®) without any incidents or premedication with an interpretable angiogram

International Multicenter Study of Clinical Outcomes of Sinonasal Melanoma Shows Survival Benefit for Patients Treated with Immune Checkpoint Inhibitors and Potential Improvements to the Current TNM Staging System

OBJECTIVES: Sinonasal mucosal melanoma (SNMM) is an extremely rare and challenging sinonasal malignancy with a poor prognosis. Standard treatment involves complete surgical resection, but the role of adjuvant therapy remains unclear. Crucially, our understanding of its clinical presentation, course, and optimal treatment remains limited, and few advancements in improving its management have been made in the recent past. METHODS: We conducted an international multicenter retrospective analysis of 505 SNMM cases from 11 institutions across the United States, United Kingdom, Ireland, and continental Europe. Data on clinical presentation, diagnosis, treatment, and clinical outcomes were assessed. RESULTS: One-, three-, and five-year recurrence-free and overall survival were 61.4, 30.6, and 22.0%, and 77.6, 49.2, and 38.3%, respectively. Compared with disease confined to the nasal cavity, sinus involvement confers significantly worse survival; based on this, further stratifying the T3 stage was highly prognostic (p < 0.001) with implications for a potential modification to the current TNM staging system. There was a statistically significant survival benefit for patients who received adjuvant radiotherapy, compared with those who underwent surgery alone (hazard ratio [HR] = 0.74, 95% confidence interval [CI]: 0.57–0.96, p = 0.021). Immune checkpoint blockade for the management of recurrent or persistent disease, with or without distant metastasis, conferred longer survival (HR = 0.50, 95% CI: 0.25–1.00, p = 0.036). CONCLUSIONS: We present findings from the largest cohort of SNMM reported to date. We demonstrate the potential utility of further stratifying the T3 stage by sinus involvement and present promising data on the benefit of immune checkpoint inhibitors for recurrent, persistent, or metastatic disease with implications for future clinical trials in this field

International multicenter study of clinical outcomes of sinonasal melanoma shows survival benefit for patients treated with immune checkpoint inhibitors and potential improvements to the current TNM staging system

Objectives: Sinonasal mucosal melanoma (SNMM) is an extremely rare and challenging sinonasal malignancy with a poor prognosis. Standard treatment involves complete surgical resection, but the role of adjuvant therapy remains unclear. Crucially, our understanding of its clinical presentation, course, and optimal treatment remains limited, and few advancements in improving its management have been made in the recent past. Methods: We conducted an international multicenter retrospective analysis of 505 SNMM cases from 11 institutions across the United States, United Kingdom, Ireland, and continental Europe. Data on clinical presentation, diagnosis, treatment, and clinical outcomes were assessed. Results One-, three-, and five-year recurrence-free and overall survival were 61.4, 30.6, and 22.0%, and 77.6, 49.2, and 38.3%, respectively. Compared with disease confined to the nasal cavity, sinus involvement confers significantly worse survival; based on this, further stratifying the T3 stage was highly prognostic (p &lt; 0.001) with implications for a potential modification to the current TNM staging system. There was a statistically significant survival benefit for patients who received adjuvant radiotherapy, compared with those who underwent surgery alone (hazard ratio [HR] = 0.74, 95% confidence interval [CI]: 0.57-0.96, p = 0.021). Immune checkpoint blockade for the management of recurrent or persistent disease, with or without distant metastasis, conferred longer survival (HR = 0.50, 95% CI: 0.25-1.00, p = 0.036). Conclusions: We present findings from the largest cohort of SNMM reported to date. We demonstrate the potential utility of further stratifying the T3 stage by sinus involvement and present promising data on the benefit of immune checkpoint inhibitors for recurrent, persistent, or metastatic disease with implications for future clinical trials in this field

Clinical outcomes, Kadish-INSICA staging and therapeutic targeting of somatostatin receptor 2 in olfactory neuroblastoma

Introduction: olfactory neuroblastoma (ONB) is a rare cancer of the sinonasal region. We provide a comprehensive analysis of this malignancy with molecular and clinical trial data on a subset of our cohort to report on the potential efficacy of somatostatin receptor 2 (SSTR2)-targeting imaging and therapy.Methods: we conducted a retrospective analysis of 404 primary, locally recurrent, and metastatic olfactory neuroblastoma (ONB) patients from 12 institutions in the United States of America, United Kingdom and Europe. Clinicopathological characteristics and treatment approach were evaluated. SSTR2 expression, SSTR2-targeted imaging and the efficacy of peptide receptor radionuclide therapy [PRRT](177Lu-DOTATATE) were reported in a subset of our cohort (LUTHREE trial; NCT03454763).Results: dural infiltration at presentation was a significant predictor of overall survival (OS) and disease-free survival (DFS) in primary cases (n = 278). Kadish-Morita staging and Dulguerov T-stage both had limitations regarding their prognostic value. Multivariable survival analysis demonstrated improved outcomes with lower stage and receipt of adjuvant radiotherapy. Prophylactic neck irradiation significantly reduces the rate of nodal recurrence. 82.4% of the cohort were positive for SSTR2; treatment of three metastatic cases with SSTR2-targeted peptide-radionuclide receptor therapy (PRRT) in the LUTHREE trial was well-tolerated and resulted in stable disease (SD).Conclusions: this study presents pertinent clinical data from the largest dataset, to date, on ONB. We identify key prognostic markers and integrate these into an updated staging system, highlight the importance of adjuvant radiotherapy across all disease stages, the utility of prophylactic neck irradiation and the potential efficacy of targeting SSTR2 to manage disease.</p

Rationale, design, and baseline characteristics in Evaluation of LIXisenatide in Acute Coronary Syndrome, a long-term cardiovascular end point trial of lixisenatide versus placebo

BACKGROUND: Cardiovascular (CV) disease is the leading cause of morbidity and mortality in patients with type 2 diabetes mellitus (T2DM). Furthermore, patients with T2DM and acute coronary syndrome (ACS) have a particularly high risk of CV events. The glucagon-like peptide 1 receptor agonist, lixisenatide, improves glycemia, but its effects on CV events have not been thoroughly evaluated. METHODS: ELIXA (www.clinicaltrials.gov no. NCT01147250) is a randomized, double-blind, placebo-controlled, parallel-group, multicenter study of lixisenatide in patients with T2DM and a recent ACS event. The primary aim is to evaluate the effects of lixisenatide on CV morbidity and mortality in a population at high CV risk. The primary efficacy end point is a composite of time to CV death, nonfatal myocardial infarction, nonfatal stroke, or hospitalization for unstable angina. Data are systematically collected for safety outcomes, including hypoglycemia, pancreatitis, and malignancy. RESULTS: Enrollment began in July 2010 and ended in August 2013; 6,068 patients from 49 countries were randomized. Of these, 69% are men and 75% are white; at baseline, the mean ± SD age was 60.3 ± 9.7 years, body mass index was 30.2 ± 5.7 kg/m(2), and duration of T2DM was 9.3 ± 8.2 years. The qualifying ACS was a myocardial infarction in 83% and unstable angina in 17%. The study will continue until the positive adjudication of the protocol-specified number of primary CV events. CONCLUSION: ELIXA will be the first trial to report the safety and efficacy of a glucagon-like peptide 1 receptor agonist in people with T2DM and high CV event risk