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Purpose: Meaningful score differences (MSDs), as defined by recent FDA guidance, can improve the interpretation of outcome measure scores and score changes. Well-accepted methods for estimating MSDs typically rely on external anchor variables, but the applications of these methods are limited in children and adolescents with rheumatic diseases. This project explored multiple candidate anchors for the PROMIS ® Pediatric measures of Physical Activity, Fatigue, Pain Interference, and Mobility for children with juvenile idiopathic arthritis (JIA) or systemic lupus erythematosus (SLE)., Methods: Longitudinal data were extracted from the Childhood Arthritis and Rheumatology Research Alliance (CARRA) Registry. Candidate anchors included patient-reported domain-specific global impressions of change (GIC) along with other parent- and clinician-reported variables. Prior to MSD estimation, the quality of the anchors was assessed using a priori criteria (correlation ≥0.30, n≥10, <10% missing). Anchors meeting criteria were used to calculate MSDs., Results: Among 289 children with JIA and 47 with SLE, the GIC did not meet criteria inhalf of the scenarios. Other candidate anchors performed slightly better. The calculated MSDs varied by external anchor across measures, diagnoses, and direction of change (better vs worse)., Conclusions: Many of the candidate external anchoring variables did not meet pre-specified criteria for calculating MSDs. Even for those that did, the choice of anchoring variable had a strong impact on the estimated MSD value and were different from other published values. As in adults, establishing pediatric MSDs requires selection of high-quality anchors, as changes in the variables used as anchors can impact MSD values and any subsequent score interpretations., Competing Interests: Declarations. Conflict of interest: Dr. Zigler has received consultant honoraria from Emmes Corporation and travel support from the Rett Syndrome Research Trust. Dr. Randell receives support from the NIH and Lupus Foundation of America, and her spouse has financial relationships with Merck & Co and Biogen. Dr. Schanberg has research funding from BMS and PCORI. She serves on the DSMBs for Sanofi (Sarilumab) and UCB (certolizumab). Dr. von Scheven receives financial support from the Childhood Arthritis and Rheumatology Research Alliance for her role on Executive Committee. Dr. Reeve, in the past five years, has received consultant honoraria from Novartis Pharmaceuticals, the University of Florida, Northwestern University, Johns Hopkins University, and Regeneron Pharmaceuticals. Authors Dr. Weitzman, C.M. Mann, Z. Li, and A. Hernandez declare they have no financial interests., (© 2024. The Author(s).)

Purpose: This study aimed to determine whether the modified Screening to Brief Intervention (S2BI) and Brief Screener for Tobacco, Alcohol, and Drugs (BSTAD) that included e-cigarettes/vaping as examples could identify all nicotine and cannabis use or whether additional questions specifically about vaping are needed., Methods: Between July 2020 and February 2022, adolescents recruited from primary care or an outpatient adolescent substance use disorder program were randomized to complete S2BI or BSTAD followed by specific questions about vaping. Screen questions were updated to include "vaping" in the parenthetical examples of nicotine and cannabis use., Results: Data from 502 adolescent participants who completed both the S2BI or BSTAD and vaping questions were analyzed. The S2BI and BSTAD identified 92% (23/25) and 100% (16/16) of nicotine vaping and 100% (16/16) and 95.5% (21/22) of cannabis vaping, respectively., Discussion: The S2BI and BSTAD tools accurately identify nicotine and cannabis use without specific questions about vaping., (Copyright © 2024 Society for Adolescent Health and Medicine. All rights reserved.)

Importance: Efficient screening tools that effectively identify substance use disorders (SUDs) among youths are needed., Objective: To evaluate the psychometric properties of 3 brief substance use screening tools (Screening to Brief Intervention [S2BI]; Brief Screener for Tobacco, Alcohol, and Drugs [BSTAD]; and Tobacco, Alcohol, Prescription Medication, and Other Substances [TAPS]) with adolescents aged 12 to 17 years., Design, Setting, and Participants: This cross-sectional validation study was conducted from July 1, 2020, to February 28, 2022. Participants aged 12 to 17 years were recruited virtually and in person from 3 health care settings in Massachusetts: (1) an outpatient adolescent SUD treatment program at a pediatric hospital, (2) an adolescent medicine program at a community pediatric practice affiliated with an academic institution, and (3) 1 of 28 participating pediatric primary care practices. Participants were randomly assigned to complete 1 of the 3 electronic screening tools via self-administration, followed by a brief electronic assessment battery and a research assistant-administered diagnostic interview as the criterion standard measure for Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM-5) diagnoses of SUDs. Data were analyzed from May 31 to September 13, 2022., Main Outcomes and Measures: The main outcome was a DSM-5 diagnosis of tobacco/nicotine, alcohol, or cannabis use disorder as determined by the criterion standard World Mental Health Composite International Diagnostic Interview Substance Abuse Module. Classification accuracy of the 3 substance use screening tools was assessed by examining the agreement between the criterion, using sensitivity and specificity, based on cut points for each tool for use disorder, chosen a priori from previous studies., Results: This study included 798 adolescents, with a mean (SD) age of 14.6 (1.6) years. The majority of participants identified as female (415 [52.0%]) and were White (524 [65.7%]). High agreement between screening results and the criterion standard measure was observed, with area under the curve values ranging from 0.89 to 1 for nicotine, alcohol, and cannabis use disorders for each of the 3 screening tools., Conclusions and Relevance: These findings suggest that screening tools that use questions on past-year frequency of use are effective for identifying adolescents with SUDs. Future work could examine whether these tools have differing properties when used with different groups of adolescents in different settings.

Purpose: The COVID-19 pandemic required most pediatric rehabilitation programs to shift to a virtual delivery format without the benefits of evidence to support this transition. Our study explored families' experiences participating virtually in More Than Words , a program for parents of autistic children, with the goal of generating new evidence to inform both virtual service delivery and program development., Method: Twenty-one families who recently completed a virtual More Than Words program participated in a semistructured interview. The interviews were transcribed and analyzed in NVivo using a top-down deductive approach that referenced a modified Dynamic Knowledge Transfer Capacity model., Results: Six themes capturing families' experiences with different components of virtual service delivery were identified: (a) experiences participating from home, (b) accessing the More Than Words program, (c) delivery methods and program materials, (d) the speech-language pathologist-caregiver relationship, (e) new skills learned, and (f) virtual program engagement., Conclusions: Most participants had a positive experience in the virtual program. Suggested areas for improvement included the time and length of intervention sessions and increasing social connections with other families. Practice considerations related to the importance of childcare during group sessions and having another adult to support the videorecording of parent-child interactions. Clinical implications include suggestions for how clinicians can create a positive virtual experience for families., Supplemental Material: https://doi.org/10.23641/asha.22177601.

Hepatitis C virus (HCV) and human immunodeficiency virus (HIV) are often co-transmitted. Viral coinfection results in worse outcomes. Persons who inject drugs (PWIDs) face barriers to medical treatment, but HCV treatment is indicated and effective even with ongoing active drug use. We aimed to assess access to HCV care and treatment results in patients coinfected with HIV-HCV. This is a real-world retrospective single-center study of patients followed in the HIV clinic between 2002 and 2018. Linkage to care was defined as achieving care cascade steps: (1) hepatology clinic visit, (2) receiving prescription of anti-HCV treatment, and (3) documentation of sustained virologic response (SVR). Of 1660 patients with HIV, 254 with HIV-HCV coinfection were included. Only 39% of them achieved SVR. The rate limiting step was the engagement into hepatology care. Being a PWID was associated with ~50% reduced odds of achieving study outcomes, active drug use was associated with ~90% reduced odds. Older age was found to facilitate treatment success. Once treated, the rate of SVR was high in all populations. HCV is undertreated in coinfected young PWIDs. Further efforts should be directed to improve access to care in this marginalized population.

Objectives: This article reports on the experience of an outpatient adolescent substance use disorder treatment program transitioning to virtual care during the COVID-19 pandemic. We describe the processes used to make the transition, including development of a safety protocol for patients seen virtually, present clinical volume data before and after the transition, and we describe a range of patient experiences through 3 clinical vignettes., Methods: Using data from the electronic health record, we generated counts of the total number of scheduled and completed appointments between December 2019 and June 2020. We used simple proportions to calculate the completed visit rate., Results: Both the absolute number of scheduled appointment and the percent of appointments completed increased with the initiation of virtual care, supporting the acceptability of this modality. Several patients experienced clinical improvements in conjunction with greater engagement, though challenges were also noted., Conclusions: Virtual visits are a practical mode of treatment for adolescent substance use disorders, and more evidence is needed to understand the risks and benefits of this treatment modality., Competing Interests: The authors report no conflicts of interest., (Copyright © 2021 American Society of Addiction Medicine.)

Purpose: The objective of this study was to examine the effects of coaching by speech-language pathologists on educators' interactive shared book reading, children's participation in shared reading, and children's language development., Method: Thirty-two educators and small groups of preschoolers were randomly assigned to experimental and comparison groups. The experimental group (n = 15) received 4 in-service workshops plus 5 individualized coaching sessions. The comparison group received only the 4 workshops. Participants were video-recorded during a shared book reading activity with a small group of children at pretest and posttest. The video recordings were transcribed and coded to yield measures of conversations, educators' questions, and children's responses. The mean length of utterances of the children's responses was also calculated., Results: There were no significant Time × Group interaction effects for the number and length of shared reading conversations or for the number of participants in these conversations. However, significant Time × Group interactions were observed for the use of educators' experiential reasoning questions, children's experiential reasoning responses, and the mean length of utterances of children's responses., Conclusion: These results suggest that coaching increases educators' use of inferential questions, enhancing an interactive shared-reading strategy that had a direct impact on the children's quality and complexity of language.

Background: The body of literature on narratives of bilingual children with and without specific language impairment (SLI) is growing. However, little is known about the narrative abilities of bilingual preschool children with SLI and their patterns of growth., Aims: To determine the similarities and differences in narrative abilities between preschoolers with and without SLI who are either monolingual or bilingual at two time points., Methods & Procedures: Forty children completed a narrative retell task in English at two test points. The mean ages were 52 and 58 months at Times 1 and 2, respectively. We examined performance on measures of narrative macrostructure (narrative information) and microstructure (sentence length, number of different words, verb accuracy, first mentions) in monolingual and bilingual children with and without SLI. The bilingual children were from diverse first-language backgrounds and all spoke English most of the time., Outcomes & Results: A series of repeated-measures analyses of variance was used with language ability (typical development or SLI) and bilingual status (monolingual versus bilingual) as the between-subjects factors and time (Times 1 or 2) as the within-subjects factor. Results indicated a significant main effect of time for four measures (i.e., Information Score, lexical diversity, sentence length and verb accuracy). The between-subjects analyses indicated a significant difference between the typically developing children and the children with SLI in all measures and a significant difference between monolingual and bilingual children for verb accuracy only., Conclusions & Implications: This study showed that all four groups of children showed growth over a 6-month period and that bilingual children exposed predominantly to English in the home performed similarly to their monolingual peers in measures of narrative information, sentence length, number of different words and first mentions., (© 2015 Royal College of Speech and Language Therapists.)

Background and Aims: Secondary sclerosing cholangitis in critically ill patients (SSC-CIP) is a relatively new previously unrecognized entity which may lead to severe biliary disease with rapid progression to cirrhosis. We present for the first time a case series of patients with rapidly progressive SSC-CIP requiring aggressive intensive care treatment following major burn injury., Results: SSC-CIP was diagnosed in 4 consecutive patients hospitalized due to major burn injuries at our Intensive Care Unit (ICU). SSC-CIP was diagnosed when ERCP (n = 1) or MRCP (n = 3) demonstrated irregular intrahepatic bile ducts with multiple strictures and dilatations and, when a liver biopsy (n = 3) demonstrated severe cholestasis and bile duct damage. All patients were males; none of whom had pre-existing liver disease. Ages: 18-56 y. All patients suffered from severe (grade 2-3) burn injuries with total burn surface area ranging from 35 to 95%. Mean length of ICU hospitalization was 129.2 ± 53.0 days. All patients required mechanical ventilation (with a mean PEEP of 8.4 ± 2.1 cm H2O) and the administration of catecholamines for hemodynamic stabilization. All patients demonstrated severe cholestasis. Blood cultures and cultures from drained liver abscesses grew hospital acquired multiple resistant bacteria. Liver cirrhosis developed within 12 months. One patient underwent orthotopic liver transplantation. Two patients (50%) died. In conclusion, SSC-CIP following major burn injury is a rapidly progressive disease with a poor outcome. Liver cirrhosis developed rapidly. Awareness of this grave complication is needed for prompt diagnosis and considerations of a liver transplantation.

About 80% of hepatocellular carcinoma (HCC) is caused by hepatitis B virus (HBV) and/or hepatitis C virus (HCV) infections especially in the setting of established cirrhosis or advanced fibrosis, making HCC prevention a major goal of antiviral therapy. HCC tumors are highly complex and heterogeneous resulting from the aberrant function of multiple molecular pathways. The roles of HCV or HBV in promoting HCC development are still either directly or indirectly are still speculative, but the evidence for both effects is compelling. In patients with chronic hepatitis viral infection, cirrhosis is not a prerequisite for tumorigenesis., (Copyright © 2015 Elsevier Inc. All rights reserved.)

Purpose: The current study investigated the effects of coaching as part of an emergent literacy professional development program to increase early childhood educators' use of verbal references to print and phonological awareness during interactions with children., Method: Thirty-one educators and 4 children from each of their classrooms (N = 121) were randomly assigned to an experimental group (21 hr of in-service workshops plus 5 coaching sessions) and a comparison group (workshops alone). The in-service workshops included instruction on how to talk about print and phonological awareness during a post-story craft/writing activity. All educators were video-recorded during a 15-min craft/writing activity with a small group of preschoolers at pretest and posttest. All videotapes were transcribed and coded for verbal references to print and phonological awareness by the educators and children., Results: Although at posttest, there were no significant group differences in the educators' or the children's references to print as measured by rate per minute, both the educators and the children in the experimental group used a significantly higher rate per minute of references to phonological awareness relative to the comparison group., Conclusion: Professional development that included coaching with a speech-language pathologist enabled educators and children to engage in more phonological awareness talk during this activity.

Purpose: The purpose of this study was to investigate whether an emergent literacy professional development program enhanced educators' use of vocabulary-teaching strategies during shared reading with small groups of pre-schoolers., Method: Thirty-two pre-school educators and small groups of pre-schoolers from their classrooms were randomly assigned to experimental or comparison groups. The 15 educators in the experimental group received four in-service workshops as well as five individualized classroom coaching sessions. The comparison group received only the workshops. Each educator was video-recorded reading a storybook to a small group of pre-schoolers at pre-test and post-test. The videos were transcribed and coded to yield measures of the vocabulary-teaching strategies and children's vocabulary-related talk., Result: The findings revealed that the children in the experimental group engaged in significantly more vocabulary-related talk relative to the comparison group. A non-significant trend in the data indicated that educators in the experimental group used more vocabulary-teaching strategies at post-test. The educators' familiarity with children's authors and book titles at pre-test was a significant predictor of their outcomes., Conclusion: These findings suggest that an emergent literacy professional development program that includes coaching can enhance children's participation in vocabulary-related conversations with their educators.

Wilson's disease (WD) is an autosomal recessive inherited disorder of hepatic copper metabolism. WD can be present in different clinical conditions, with the most common ones being liver disease and neuropsychiatric disturbances. Most cases present symptoms at < 40 years of age. However, few reports exist in the literature on patients in whom the disease presented beyond this age. In this report, we present a case of late onset fulminant WD in a 58-year-old patient in whom the diagnosis was established clinically, by genetic analysis of the ATP7B gene disclosing rare mutations (G1099S and c.1707+3insT) as well as by high hepatic copper content. We also reviewed the relevant literature. The diagnosis of WD with late onset presentation is easily overlooked. The diagnostic features and the genetic background in patients with late onset WD are not different from those in patients with early onset WD, except for the age. Effective treatments for this disorder that can be fatal are available and will prevent or reverse many manifestations if the disease is discovered early.

A ciliated hepatic foregut cyst (CHFC) is a rare foregut developmental malformation usually diagnosed in adulthood. Five percent of reported cases of CHFC transform into squamous cell carcinoma. We report the presentation, evaluation, and surgical management of a symptomatic 45-year-old male found to have a 6.2 cm CHFC. Contrast tomography-guided fine-needle aspiration demonstrated columnar, ciliated epithelium consistent with the histologic diagnosis of CHFC. The intracystic levels of carbohydrate antigen (CA) 19-9 and carcinoembryonic antigen (CEA) were extremely high (978118 U/mL and 973 μg/L, respectively). Histologically, the wall of the cyst showed characteristic pseudopapillae lined with a ciliated stratified columnar epithelium, underlying smooth muscle, an outer fibrous layer and no atypia. Immunohistochemistry for CA19-9 and CEA was positive. This is the first case report of a CHFC in which levels of CA 19-9 and CEA were measured. Our findings suggest that a large sized multilocular cyst and elevated cyst CA19-9 and CEA levels do not exclude a CHFC from consideration in the diagnosis. CHFCs should be included in the differential diagnosis of hepatic lesions. Accurate diagnosis of a CHFC is necessary given its potential for malignant transformation, and surgical excision is recommended.

Purpose: This study examined the efficacy of a professional development program for early childhood educators that facilitated emergent literacy skills in preschoolers. The program, led by a speech-language pathologist, focused on teaching alphabet knowledge, print concepts, sound awareness, and decontextualized oral language within naturally occurring classroom interactions., Method: Twenty educators were randomly assigned to experimental and control groups. Educators each recruited 3 to 4 children from their classrooms to participate. The experimental group participated in 18 hr of group training and 3 individual coaching sessions with a speech-language pathologist. The effects of intervention were examined in 30 min of videotaped interaction, including storybook reading and a post-story writing activity., Results: At posttest, educators in the experimental group used a higher rate of utterances that included print/sound references and decontextualized language than the control group. Similarly, the children in the experimental group used a significantly higher rate of utterances that included print/sound references and decontextualized language compared to the control group., Conclusion: These findings suggest that professional development provided by a speech-language pathologist can yield short-term changes in the facilitation of emergent literacy skills in early childhood settings. Future research is needed to determine the impact of this program on the children's long-term development of conventional literacy skills.

Purpose: In working with children with language impairments, some clinical scholars and clinicians advocate using input that is simplified to the point of being ungrammatical (telegraphic input), while others advocate simplified but grammatical input. This article considers 2 types of external evidence on this topic., Method: First, a meta-analysis of relevant research, including intervention studies and processing studies, is reported. Next, 4 experts present their opinions., Results: Children in the majority of the intervention studies showed no difference in language comprehension based on type of input, although 1 study with very few children favored telegraphic input for language production. In the processing studies, which measured immediate comprehension, children from clinical populations responded inconsistently when listening to the 2 types of input. Children who had typical language, however, favored grammatical input in their responses. Regarding the experts' opinions, 2 suggest that telegraphic input is sometimes warranted; 1, who previously indirectly promoted its occasional use, no longer believes it should be used; and 1 provides reasons why telegraphic input should not be used and may even be harmful., Conclusions: Empirical findings and expert views are summarized as ways of informing parents of the weak evidence base regarding the best type of input.

Purpose: The purpose of this multiple case study was to describe outcomes for three parents and their children following participation in a social interactive model of language intervention. More specifically, this study sought to clarify if changes in children's participation, engagement, and initiation of social interaction could be observed following an 11-week intervention., Method: Three preschool children with autism spectrum disorders and their mothers participated in an 11-week intervention program. The intervention taught parents to follow the children's lead, promote children's participation in routines, and model language at the children's level. Outcome measures included estimates of parents' responsive language input, and measures of children's rate of communication, number of engagements in social interaction, and initiations., Results: The results indicated that all three mothers increased their responsive comments during play interactions and were rated as being more responsive on a rating scale. All three children evidenced positive outcomes in vocabulary and the number of engagements in social interaction. In addition, improvement was observed in social initiation skills for all three children., Conclusions: Increases in mothers' responsiveness and children's engagement in social interactions are consistent with the theoretical mechanisms of the social interaction model of language intervention. The results suggest that further randomized control trials of this intervention approach are warranted.

Purpose: The purpose of this study was to determine the feasibility of a 2-day in-service education program for (a) promoting the use of two emergent literacy strategies by early childhood educators and (b) increasing children's responses to these strategies., Method: Sixteen early childhood educators were randomly assigned to an experimental and a control group. The experimental in-service program sought to increase educators' use of abstract utterances and print references. Educators were videotaped with small groups of preschoolers during storybook reading and a post-story craft activity. Pretest and posttest videotapes were coded to yield rates of abstract language, verbal print references, and children's responses., Results: In comparison to the control group, educators in the experimental program used more abstract utterances that elicited talk about emotions and children's past experiences during storybook reading. They also used significantly more print references during a post-story craft activity. In addition, children in the experimental group responded more often with appropriate responses to abstract utterances and print references in comparison to children in the control group., Conclusion: A 2-day in-service education program resulted in short-term behavioral changes in educators' use of abstract language and print references. Suggestions for improving instruction include providing opportunities for classroom practice with feedback, modeling the use of strategies in classroom routines, and long-term mentoring of educators to promote retention of gains.

An exploratory study examined adults' questions to small groups of children to determine how questions influenced their response rate and complexity of response. Thirteen educators of toddlers and 13 educators of preschoolers were videotaped during free-play. Both groups of educators used an equivalent frequency of open-ended and closed questions, but the preschool educators used more topic-continuing questions. Consistent with their developmental level, preschoolers responded more frequently than toddlers. Toddlers demonstrated few effects of question type. In contrast, preschoolers used more multiword utterances following open-ended questions and topic-continuing questions. Implications for in-service education for staff of early childhood settings include increasing the use of both open-ended and topic-continuing questions.