Characterization of Biologic Discontinuation Among Pediatric Patients with Crohn's Disease.

BACKGROUND & AIMS: Biologic therapies may effectively treat Crohn's disease (CD), and pediatric patients who discontinue multiple biologics risk exhausting treatment options. The frequency and context of biologic discontinuation have not been well-characterized. We aimed to determine patterns of biologic use, discontinuation, and evaluation in pediatric patients with CD. METHODS: Pediatric patients with CD at 7 U.S. centers (2010-2020) were identified. Prospective ImproveCareNow registry data were supplemented with medical record abstraction. Biologics included monoclonal antibody and small molecule medications. Therapeutic drug monitoring (TDM) was considered induction if <14 weeks after biologic start, proactive if later during quiescent disease, and reactive during active disease. RESULTS: Of 823 patients included (median age, 13.0 years; 40% female), 86% started biologics (78% infliximab, 21% adalimumab, <1% others). Twenty-six percent used concomitant immunomodulators for ≥12 months. Most (85%) measured TDM including 47% induction, 69% proactive, and 24% reactive. Twenty-nine percent discontinued their first biologic after median 793 days because of inefficacy (34%), anti-drug antibodies (8%), adverse events (8%), or non-adherence (12%). If inefficacy, 86% underwent pre-discontinuation evaluation. If infliximab or adalimumab inefficacy and TDM was done, 62% had levels <10 µg/mL. Proactive TDM and concomitant immunomodulators were associated with 60% and 32% reduced biologic discontinuation. CONCLUSIONS: Most children with CD are treated with biologics; 25%-37% discontinue biologics, resulting in 1 in 12 using >2 biologics during pediatric care. Half of patients discontinued biologics without trial of high-dose therapy and 14% without any evaluation. Concomitant immunomodulator use and proactive TDM decreased risk of biologic discontinuation. Strategies are needed to preserve biologic efficacy and prevent biologic discontinuation.

The 988 suicide hotline-Lifeline or letdown? A pre-post policy analysis.

Suicide has emerged as an urgent threat in recent years as COVID-19 impaired the health and economic wellbeing of millions of Americans. According to the Centers for Disease Control and Prevention, the impact of COVID-19 and the ongoing opioid epidemic has "taken a mental, emotional, physical, and economic toll on individuals, families, and communities," increasing the need for innovative solutions to prevent suicide on a national scale. The National Suicide Hotline Designation Act of 2020 established 988 as the universal telephone number for suicide prevention and represents a key federal intervention to address this crisis. However, research on 9-8-8's effectiveness is limited, given the Act's recent enactment and implementation at the federal and state levels. This policy analysis investigates how and to what extent the mental health crisis system in Georgia has improved since the implementation of the 2020 Act as well as the implications of state law on population-level mental health outcomes. Georgia is used as a nationally representative case study for two reasons: (1) Georgia had a robust statewide suicide hotline prior to 2020, providing solid infrastructure on which federal expansion of a suicide hotline number could be built, and (2) the conflicting characteristics of Georgia's mental health system represent several different pockets of the U.S., allowing this analysis to apply to a broad range of states and locales. The paper draws on takeaways from Georgia to propose state and national policy recommendations for equitable interventions to prevent and respond to this form of violence.

Variation in the Desire for Cleft Revision Surgery among Children, Caregivers, and Surgeons.

BACKGROUND: Although revision surgery is part of the reconstructive process for children with cleft lip and/or cleft palate, the indications for revision vary, and the extent to which surgeons and families agree on appearance is unclear. The authors sought to understand the extent to which children with cleft lip and/or cleft palate, surgeons, caregivers, and control observers agree on satisfaction with appearance and the desire for revision. METHODS: Children with cleft lip and/or cleft palate (n = 100) and their caregivers (n = 100) were surveyed regarding satisfaction with appearance using the Cleft Evaluation Profile. Surgeons (n = 10) and control observers (n = 10) rated photographs of these children using questions analogous to the Cleft Evaluation Profile. General linear model repeated measures analysis of variance were used to detect significant differences between raters, with an alpha of 0.05. RESULTS: The children reported greater satisfaction with appearance across all domains compared with surgeons (nose, 7.77 versus 5.51, p < 0.001; lip, 7.94 versus 5.90, p < 0.001; maxilla, 8.16 versus 6.56, p < 0.001) and general observers (nose, 7.78 versus 6.00, p < 0.001; lip, 7.80 versus 6.12, p < 0.001; maxilla, 8.16 versus 7.40, p < 0.001). Children and caregivers expressed similar degrees of satisfaction with appearance of the lip (5.48 ± 1.69 versus 5.6 ± 1.49, p > 0.5) and maxilla (6.08 ± 1.1 versus 5.8 ± 1.2, p = 0.07). There was no significant relationship between children and surgeons in terms of the desire for revision surgery (p = 0.44). CONCLUSIONS: All groups expressed differing levels of satisfaction with cleft-specific aspects of appearance. Importantly, children were more satisfied than all other groups. Care must be taken to evaluate perceptions of all stakeholders before moving forward with cleft revision surgery.

Caregiver-Reported Outcomes and Barriers to Care among Patients with Cleft Lip and Palate.

BACKGROUND: For children with cleft lip and/or palate, access to care is vital for optimizing speech, appearance, and psychosocial outcomes. The authors posited that inadequate access to care negatively impacts outcomes in this population. METHODS: Sixty caregivers of children with cleft lip and palate were surveyed to assess perceived barriers using the validated Barriers to Care questionnaire. The questionnaire includes 39 items divided into five subscales, with higher scores indicating fewer barriers. Caregiver-reported outcomes were assessed using the Cleft Evaluation Profile, which captures cleft-specific appearance- and speech-related outcomes. Higher scores correspond to less satisfactory outcomes. Desire for revision surgery was assessed as a binary outcome among caregivers. Multivariable regression was used to evaluate the relationship of barriers to care, caregiver-reported outcomes, and desire for revision, adjusting for clinical and demographic covariates. RESULTS: Sixty percent of caregivers perceived barriers to care, and caregivers who reported poorer access to care described poorer cleft-related outcomes (r = 0.19, p = 0.024). Caregivers with poorer skills (r = 0.17, p = 0.037), expectations (r = 0.17, p = 0.045), and pragmatics (r = 0.18, p = 0.026) subscale scores were associated with worse Cleft Evaluation Profile scores. Barriers were also negatively associated with aesthetic item scores (r = 0.11, p = 0.025). Finally, caregivers reporting fewer barriers were 21.2 percent less likely to express interest in revision surgery. CONCLUSIONS: Barriers to care were associated with poorer appearance-related outcomes and increased interest in revision among caregivers of cleft patients. Enhancing access to care is critical in order to effectively meet goals of care for these families.

Provider perspectives on employment for people with serious mental illness.

BACKGROUND: Persons with serious mental illnesses (SMI) can lead productive lives, and the majority want to work. Mental health providers can play an important role in helping their clients gain and maintain employment. AIMS: The Provider Survey was developed to shed additional light on providers' views toward employment and recovery, and the utilization of Individual Placement and Support (IPS) supported employment (SE) services for people with SMI. METHODS: A total of 1,306 providers of the State of Connecticut participated in the survey. Four main questions were addressed in the survey: what do providers do, what do they view as most important regarding employment, what are their views when it comes to what promotes recovery and what barriers do providers face in attempting to refer their clients to IPS services. RESULTS: Referring clients to additional supports was rated as the most important aspect of what providers do; encouragement was rated as the most important component to enable clients in gaining and maintaining employment; agency, belonging and medical care were rated as most important in promoting recovery; and expectations that clients would be discriminated against at work was the most important barrier to referring clients for SE. Also, employment and finances were seen as the least important factors in promoting the recovery of people with SMI. CONCLUSION: This survey suggests that one reason that more clients may not be referred to IPS programs is that clinicians do not view employment or financial self-sufficiency as important factors in recovery, further compounding the historical view that these persons are unable to, and uninterested in, working. Such findings call for a provider education and training campaign to highlight the fact that most persons with SMI - like most persons in general - do want to work and, with supports, most are capable of doing so.

Speech Perceptions and Health-Related Quality of Life Among Children With Cleft Lip and Palate.

BACKGROUND: The association between perception of speech and health-related quality of life (HRQOL) among patients with cleft palate is not well understood. The purpose of this study was to determine: the agreement between patient and parent perception of speech, the correlation between patient/parent speech perception and objective analysis by a speech-language pathologist (SLP), and the relationship between objective speech analysis and HRQOL among children with cleft lip with or without palate (CLCP). METHODS: The authors surveyed 108 CLCP patients who received treatment at a large tertiary medical center from 2013 to 2014. Patients and parents were queried regarding their difficulty with speech, and an SLP performed perceptual speech analysis with each patient. Patient-reported survey instruments were used to assess anxiety, depression, anger, peer relationships, stigma, and overall psychosocial health. The authors assessed the agreement between patients and SLP analysis as well as association between speech and HRQOL. RESULTS: Patient and parent-reported speech quality demonstrated moderate agreement regarding the quality of the child's speech (r = 0.46-0.64). Parent and patient speech perception was not well associated with SLP analysis (V = 0.06-0.30). Patient speech perception was correlated with depression (P = 0.03), while SLP analysis was correlated with anger (P = 0.03, P = 0.004), depression (P = 0.007), and difficulty with peer relationships (P = 0.02). CONCLUSIONS: Patients and parents have different perceptions of the quality of the child's speech, and their ratings differ from SLP perceptual speech analysis. Both patient speech perception and SLP analysis are correlated with important aspects of quality of life, and should be considered when evaluating children with CLCP.

The Feasibility and Validity of PROMIS: A Novel Measure of Quality of Life among Children with Cleft Lip and Palate.

BACKGROUND: Health-related quality of life is inconsistently captured among children with cleft lip and palate. The Patient-Reported Outcomes Measurement Information System (PROMIS) captures health-related quality of life, with the added benefit of comparability across clinical conditions. In this study, the authors define the validity and feasibility of PROMIS among children with clefts. METHODS: Children with cleft lip and palate who were at least 5 years old and able to complete instruments independently were eligible for inclusion (n = 93). Children completed PROMIS anxiety, depression, and peer relationship item banks as short forms or computerized adaptive tests. Participants also completed the Pediatric Quality of Life Inventory. Construct validity was measured by Spearman correlations between PROMIS and the Pediatric Quality of Life Inventory controlling for race, sex, age, and income. Feasibility was measured using instrument completion time, reading level, and floor/ceiling effects. RESULTS: PROMIS computerized adaptive tests (peer relationship, r = 0.49; depression, r = -0.56; and anxiety, r = -0.36) and short forms (peer relationship, r = 0.65; depression, r = -0.54; and anxiety, r = -0.56) demonstrated moderate correlation with the Pediatric Quality of Life Inventory. Computerized adaptive tests had fewer floor (0 percent versus 0 percent) and ceiling (8.6 to 19.3 percent versus 21.8 to 41.9 percent) effects than short forms, and demonstrated better readability. Computerized adaptive tests required more time than short forms (peer relationship, 0.84 ± 0.67 versus 1.3 ± 0.92; depression, 0.52 ± 0.38 versus 1.1 ± 0.73; and anxiety, 0.53 ± 0.23 versus 1.1 ± 0.62; p = 0.001), as each computerized adaptive test included on average four more questions. CONCLUSIONS: PROMIS correlates well with the Pediatric Quality of Life Inventory and demonstrates similar accuracy, with better readability and efficiency. Use of PROMIS will improve our ability to compare children with cleft lip and palate to diverse populations and clinical conditions. CLINICAL QUESTION/LEVEL OF EVIDENCE: Diagnostic, II.

Health-Related Quality of Life and the Desire for Revision Surgery Among Children With Cleft Lip and Palate.

PURPOSE: Children with cleft lip with or without palate (CLCP) require multiple reconstructive procedures, however, little is known about their desire for surgical revision. The purpose of this study was to examine the relationship between health-related quality of life (HRQOL) and the desire for revision. METHODS: The authors surveyed children with CLCP (n = 71) and their caregivers regarding general and cleft-specific HRQOL and the desire for revision surgery. The authors used logistic regression models to evaluate the relationship between HRQOL and the desire for revision stratified by age, and determined the level of agreement between caregivers and children. RESULTS: In this cohort, 54.9% of children desired revision, primarily of the nose (n = 23), lip (n = 20), and dentoalveolar structures (n = 19). Children 11 years or older were more likely to desire revision than younger children (OR 3.39, 95% CI [1.19, 9.67], P <0.05). Children who reported poorer HRQOL with respect to appearance (OR 2.31, 95% CI [1.25-4.29], P = 0.008), social development (OR 0.91, 95% CI [0.84-0.99], P = 0.02), and communication (OR 0.94, 95% CI [0.89-0.99], P = 0.02) were significantly more likely to desire revision than children who reported more positive HRQOL. Caregivers' and children's desires for revision were only modestly correlated (r = 0.41). CONCLUSIONS: Children with CLCP who report poorer HRQOL are more likely to desire revision than children with higher HRQOL; these differences are further magnified among older children. Given the modest correlation between patient and caregiver goals for revision, it is important to evaluate both perspectives when considering revision surgery.