Genome-assisted prediction of complex  traits is an ingredient of “Genomic Selection,” a paradigm adopted successfully in animals and plants of agricultural importance. The approach has impacted the timing of selection decisions, and it has delivered improvements in the quality of predictions (“accuracy”) relative to what can be attained by the use of pedigrees and phenotypes. It has enhanced the rate of response to genetic selection and spectacularly so in dairy cattle, at least as suggested by genome-based estimates of genetic change. Researchers have spent much effort in developing and adapting prediction machines, and the author will focus on this matter, with mild excursions into tangential issues. The material is organized into nine sections and, since the author was to opine, it represents a set of personal views, rather than a review of literature, made retrospectively.1. Deconstruction of “genetic architecture”:Molecular genetics and biochemistry confirm that the theory of quantitative genetics provides just a linear  approximation to complexity with little (if any) mechanistic value. The intricate interactions and feedbacks inherent in biological systems cannot be captured by simple linear regressions, even if highly-dimensional regression models are fitted to the data. The effective dimension of a model cannot exceed the sample size. For example a model with 5 million parameters run with a sample size of 500 does not provide meaningful estimates of more than 500 distinct estimable functions of parameters: individual site effects are not likelihood-identified. The view that quantitative genomics can unravel the “genetic architecture” of complex traits by providing an inventory of allelic frequencies and allelic substitution effects, or by a decomposition of variance  is equivalent to stating that tons of bricks, steel, and glass can represent Zaha Hadid's new Beijing airport or Frank's Gehry's Guggenheim Museum at Bilbao. The author often refraines from using the buzz term “genetic architecture” and favores “statistical architecture” instead.2. Crumbs are not bread:The QTL paradigm [superseded by zillions of genome-wide association study (GWAS) in human genetics] has had a minor impact on agricultural practices , with few exceptions. GWAS with single-marker regression is also insufficient because it accounts for little genetic variation , apart from ignoring interactions as stated above. Although a more complex model may improve learning, the author has not seen reports where variable selection methods and members of the Bayesian alphabet capture signals much more effectively than a simple GWAS run with large samples, as in human genetics consortia. Shrinkage methods are typically “vector optimized” (with ridge regression notoriously so), and the borrowing of information facilitated by proper priors tends to make signals similar to each other. Bayesian variable selection (BVS) with spike-slab distributions may be more powerful, but signals from large-effect variants are strengthened at the expense of mitigating small effects. In BVS or LASSO, the “richer get richer and the poorer get poorer” whereas ridge regression is more “social democrat,” making effect-size estimates similar to each other. If a pizza for 500 persons is divided into 5 million unexpected guests, each will end up getting a crumb. The perception of the author is that advances in the resolution and causality of small-effect variants via GWAS and genome-enabled prediction have been marginal, at least in agriculture.3. Corroboration vs. induction:The main contributions of quantitative genetics (genomics) have been in description, prediction, and decision, e.g., selection choices, inbreeding management, and optimum contribution theory, as opposed to inference. In predictive approaches, genomic heritability or correlations take the role of “regularization knobs”  for constructing prediction machines. The objective is to make statements about yet-to-be-observed phenotypes based on some training data. Predictions can be calibrated empirically, but inferences cannot. How can one say that an estimate of an entelechy, such as heritability is bad or good? Following Descartes: “I cannot be observed, therefore, I do not exist.” According to Encyclopedia Britannica, for Descartes to prove that heritability exists, one must assume it does. For prediction, “there is no need for that hypothesis” as often attributed to Laplace.4. Occam's razor resurrected:A less recognized but important ingredient of the study on genomic selection of Meuwissen et al. .r et al. , are user et al.  in assesIt is futile to have information-rich training samples but unrepresentative and ridiculously small testing sets, as large variation among outcomes of similar prediction exercises is to be expected. Small testing sets and failure to replicate cross-validation in some studies have produced results where models accommodating dominance and epistasis appear as delivering a somewhat better performance than additive prediction models. Such results may be “false positives” reflecting chance, rather than signal.6. And the Oscar goes to…:A simple method such as genomic best linear unbiased predictor (GBLUP) may tell something about the state of nature and perform adequately. An involved procedure, such as a deep neural network (DNN), may tell nothing and yet produce spectacular results, although it has failed miserably in some studies. Like all neural networks, a DNN is regarded as “universal approximator.” An ongoing meta-analysis of hundreds of studies made in INIA, Spain (disclaimer: I will be a coauthor) places reproducing kernel Hilbert spaces regression (RKHS); e.g., Wahba  for routine use, but there can be exceptions. GBLUP is a “good thing” as pointed out in the early '90s, and we have known for a while that it is not only a special case of RKHS, but also a maximum penalized likelihood estimator, a linear neural network, and that it has a Bayesian interpretation. It has been extended to cross-sectional, multi-trait, longitudinal situations and has been “robustified.” Importantly, software developed mainly at the University of Georgia by Misztal allows crunching millions of predicted genomic breeding values. GBLUP with mild tweeks will probably remain the technology (term used deliberately) of choice for genetic evaluation of selection candidates. The science of genome-enabled prediction has arrived at a reasonable destination, but the voyage will continue, and new data will bring challenges.7. Help needed:Despite an abundance of chips enabling large-scale genotyping, training samples are seldom drawn at random, thus unrepresentative. This situation constitutes a selection process that is often not considered in predictive models. Animal breeders have been widely influenced by the “selection bias” study of Henderson  to be an unbiased predictor, but it is identical to ridge regression in some settings. However, ridge regression is a biased estimator. Is this a Dr. Jekyll Mr. Hide issue or some statistical bipolarity? The answer is that prediction and estimation unbiasedness have different definitions! Say you own a plant or a bull called “Charlie,” a fixed entity with identity . You are not interested in learning the average of a (very) large sample of potential Charlies; rather, you seek the breeding value of the Charlie you have. If ridge regression is used to estimate the breeding value of Charlie, Dr. Jekyll says there is estimation bias, but Mr. Hide states that there would be none. The latter is wrong (in the bias sense) with respect to Charlie, but not with respect to an average of potential Charlies, some of which will be AA, some Aa, and some aa. All good members of the Bayesian alphabet including GBLUP, with its appealing Bayesian interpretation  continues being crucial in practice, but it has become academically non glamorous at these times of massive genotyping, epigenotyping, proteomics, metabolomics, and (fine) phenotyping. It is important not to lose perspective as, otherwise, breeders will get inebriated with a cocktail “on apps.” Another issue of (some) concern is that the current emphasis on “big data,” “massive computing,” and “visualization” may diminish basic science education, as it appears that current thesis students start crunching numbers before they know genomics, or the meaning of a probability distribution, or attain an elementary knowledge of randomization or causality. Foundational theory and concepts should continue being taught. Otherwise, the field may drown in a technology-induced maelstrom, and critical or even visionary perspectives may end up playing a role that becomes secondary to that of a beautiful visualization or, even worse, to a machine.W. G. Hill (“Bill”) noted in a 2010 study discussing from Lush to Genomics: “Opinions we can debate.” I look forward to that conversation (Hill, DG prepared the first draft of the opinion, provided critical comments, typed the definitive version, and checked the spelling. The author contributed to the article and approved the submitted version.The author declares that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. The handling editor declared a past collaboration with the author DG.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


More recently, however, greater attention has been focused on differences based on family income. Under either measure, the achievement gap is large and persistent, with serious economic, social, and ethical consequences.A large body of research finds that gaps in achievement appear well before children enter primary school , that is, center‐based, predominantly classroom‐style programs serving preschool children.Age at which children begin receiving services. Just about all programs begin by age four, with many starting at age three, and some starting before that.Ancillary service(s) provided. Some programs provide parenting skills training, some provide medical/dental services, some both, as well as other services.Eligibility. Some programs target particular groups of children (such as “at risk” children or those from families below a specified income threshold), some are neighborhood oriented, and others are available universally, regardless of income or other disadvantage.Curriculum used (if any). Some programs use a particular curriculum , some do not specify what, if any, curriculum they use (only that each center/classroom follow one), and others implement state or locally developed curricula.Focus of the program. Some programs primarily target children's cognitive skills, some target socioemotional skills, and some focus on “whole‐child” development, or others some combination or variant thereof.Follow‐on services. Some programs provide services through the 1st year (or later) of primary school.Intensity. Some programs are full‐day, some are half‐day, and others are somewhere in between.Length/duration. Some programs are 1‐year (or more) in length, some are about 9–10 months long , and, in the past, others, such as the first version of Head Start, were for shorter periods (like the summer).Service setting. Most programs are operated are in private, nonprofit centers, but many are in public schools.Source of funding. Most are state/local programs, often supported through federal funds, some are federal programs, and others are demonstration projects run by universities or other nongovernmental agencies.Teacher credentials. Some programs require that teachers have bachelor's degrees, some require associate's degrees, some require a certificate in ECE or development, some require a combination of these, and others require none.There is, however, no set or uniform model for an ECE intervention, although most provide some form of educational programming directly to preschool children. The following are some of the key variations:Even among Head Start programs , the variation among these and other dimensions is large. Some Head Start programs are full‐day, but most are part‐day. Some are one‐academic‐year long ; others are longer. About half of Head Start programs use the Creative Curriculum,This variation does not appear to be unique to Head Start nor to the U.S. experience. For example, the UK's Sure Start programs provide substantial autonomy to local programs and, according to the National Evaluation of Sure Start researchers, “This has resulted in the emergence of great diversity in Sure Start local programmes”  are the indispensable foundation for social, emotional, and language skills that lead to later success. Indeed, the terms “critical periods” and “sensitive periods” are often used when discussing child development in the first 3–5 years of life  of the intervention is not sufficient to overcome the family and environmental factors that also influence children's plasticity of learning.1.3.1Nearly all ECE programs attempt to increase the “school readiness” of children. Although school readiness is defined in various ways, “experts agree that readiness is a multifaceted concept that goes beyond academic and cognitive skills” ,Social and emotional development,Language and literacy,Cognition, andPerceptual, motor, and physical development.The Head Start Early Learning Outcomes Framework . In order for this theory to be validated, children's immediate cognitive gains are/should be sustained (and built upon) as they age.Sleeper effects, wherein immediate cognitive gains seem to dissipate but are, in actuality “lying dormant until some process of maturational change or environmental triggering [stimulates] their reawakening during the later school years” (p. 33) . Because this model is based on unobservable changes in the child, its presence is only deduced by the child's later achievement or situation, which makes the causal link difficult to establish.School competence theory , wherein initial improvements in cognitive skills, although fading over time, lead to reductions in grade retention and referral to special education in the first few years of school, resulting in more positive academic expectations and achievement  that links program participation to a reduction in the achievement gap. Instead, it generally looks for positive changes in various measures without consideration of the specific causal mechanism that might explain them.1.4all research on a subject, from all sources, including literature not found in peer‐reviewed journals; (c) screen studies using a set of predetermined inclusion/exclusion criteria; and (d) evaluate the quality of the studies they include and assess the validity of their findings (see Appendix A for a full list of the reviews we considered).Many papers and articles attempt to summarize  research on ECE programs, although there is substantial variation in the degree to which they systematically search for, screen, review, synthesize, and evaluate the extant literature. We found and reviewed more than 40 of these research reviews, of which 13 both examined ECE effectiveness and could be considered systematic reviews; that is, they (a) employ an explicit search strategy; (b) seek to identify The others that we found either do not appear to be systematic reviews or do not investigate the effectiveness of participation in an ECE program, in which case, as we describe below, they typically compare the effectiveness of one programmatic variation to another.Among those that we do not consider to be systematic reviews is a 2017 Brookings Institution volume produced by a blue‐ribbon panel of ECE experts that reviewed the evidence of prekindergarten (pre‐K) programs  drawing on programmatic differences identified during study coding to examine what program characteristics appear to be the most effective. Often, however, due to either resource limitations or other reasons, researchers narrow the focus on the review in one way or another. Some, as in the case of a systematic review by D'Onise, Lynch, Sawyer, and McDermott , narrow Other reviews (of the 40), which we did not include because they do not examine studies with a no service control or comparison group, take a comparative effectiveness approach, reviewing and synthesizing studies that compare children who receive one variation of a program to those receiving another . This approach is commonly used to identify the most effective curricula. Nguyen's  meta‐anaOthers have attempted to identify whether ECE programs that include a “parenting education” component have a larger effect on children's cognitive and preacademic outcomes .First, those reviews with meta‐analyses typically report one average effect size for a particular domain, combining effect sizes for a wide range of outcome measures , and/or interventions, which threatens the validity of the review. As Sharpe  argues, We think, therefore, that the better approach is to use more narrowly constructed domains, as described by the What Works Clearinghouse .In addition, but less importantly, these reviews are not up‐to‐date and typically do not provide a full picture of ECE effectiveness. Of the 13 reviews mentioned above, only one  have been completed, the findings must be collected piecemeal, with each based on analyses using different sets of studies with varying methods for identifying and screening research and for evaluating the quality of the included studies .1.4.2Unfortunately, there is sharp disagreement among experts (and policy advocates) about the effectiveness of ECE programs.ECE supporters argue that early intervention can compensate for the conditions that, at school entry, put disadvantaged children behind on various developmental dimensions—with the gap increasing as school becomes more demanding. On a selective basis, they point to various studies  that show lasting gains in educational attainment, employment, and earnings, and substantial reductions in out‐of‐wedlock births and criminal behavior.Naysayers argue that the ECE intervention is not strong enough to counter other forces in the child's life (such as family and neighborhood) and that these other factors are much more important to the child's cognitive and socioemotional development. They point to studies  that show no or small effects or whose effects seem to disappear in a few years (“fade out”).There is a certain selectivity in the way past studies are used to argue whether ECE programs “work,” rather than more balanced assessment of whether they can narrow the achievement gap to a socially significant degree. For example, Elango et al.  claim thWise policy in this area calls for as full an accounting as possible of what is known about the effectiveness of ECE programs, including how they are administered, if they are cost effective, whether one model is more effective than another, and which model works best for whom.Setting our project apart from other studies will be its emphasis on reconciling the capacities—and needs—of parents with the ability of ECE programs and other social institutions to help provide what children need for optimal social and cognitive development. We expect the findings to recognize the reciprocal importance of parent–child  relationships. Thus, we will also review the findings of other programs that seek similar outcomes, for example, programs like Parents as Teachers or Nurse–Family Partnership that seek to improve children's cognitive outcomes through a change in parental knowledge, capacity, and/or behavior.Moreover, our review will attempt to fill a void in the ECE literature by providing an up‐to‐date synthesis of ECE's effects on a wide range of outcome measures, across cognitive, socioemotional, behavioral, and health domains  from early childhood through adulthood. We expect to take great care to avoid combing dissimilar effects, thus reducing the “apples and oranges” threat to the validity of our findings.Because our report will be prepared under the auspices and rigorous standards of the respected Campbell Collaboration and with the guidance of a blue‐ribbon advisory board—Jacob Klerman, Rebecca Maynard, Michael Puma, and Matthew Stagner —we have high hopes that it will have broad policy influence.In the current political climate, the role of the federal government in early childhood programing has become uncertain. But even if the federal government is less active in this area than previously, program expansion at the state and local level will likely continue. Although many would prefer bolder federal action, state and local action could present a greater opportunity for experimentation and innovation. In fact, many of the evaluations of the programs to be included in this review were funded and administered by organizations other than the federal government, including state and local governments, nonprofit organizations, and academic institutions.Therefore, besides national policymakers and researchers, we will present our findings in a way that is useable by state and local planners, emphasizing the applicability of findings to particular populations, program settings, and geographical locations while also considering the applicability of decades‐old findings to present day conditions. Available evidence, for example, points to different approaches for children from severely disadvantaged households as well as for children from homes where English is the second language.Rather than using such findings as evidence that ECE programs “work,” we hope to use them as guidelines for how programs could be structured for greater effectiveness in particular situations. In doing so, we will attempt to reconcile early learning and development objectives with child care as a means for helping parents to work in the paid economy.In addition to reaching out to federal and state officials, we would use our long‐standing ties to the early childhood community to disseminate our findings.2This project seeks to answer questions about the effectiveness of early childhood programs in meeting their important goals, paying careful attention to the effectiveness of program variations .(1)How effective are existing ECE programs when it comes to improving the cognitive, socioemotional, behavioral, and health outcomes of low‐income children? And what does the research say about these effects in the short, intermediate, and long term?(2)What program variations are more effective than others? This inquiry comprises many more narrowly focused programmatic questions that we will attempt to answer using moderator analyses:Do programs that provide ancillary services to parents/caregivers produce, on average, larger effects than those that provide only direct instruction to children?What is the optimal program length , duration , and intensity  of an ECE program?Do low‐income students benefit more from participation in programs that primarily target and provide services to these students or from programs that are offered to students regardless of income or other disadvantage ?Does the level of teacher credential  influence the magnitude of effect sizes? And, if so, how?How does the magnitude of effect sizes of U.S. programs compare to those in other countries?How does the magnitude of effect sizes of demonstration projects  compare to public programs ?How has the magnitude of effect sizes changed since 1960?Do programs that employ a curriculum produce larger effects sizes? And, if so, which curricula appear more effective?Do programs that offer follow‐on services produce, on average, larger effects sizes than those that do not?Is program focus  associated with differences in magnitude of effect sizes?How does the magnitude of effect sizes change in relation to class size and/or student‐teacher ratios?This review will be guided by two overarching questions:33.1The body of evaluative literature of ECE programs is substantial and diverse, covering more than half a century and programs ranging from small demonstration projects to large‐scale public programs such as Head Start. Over that time, a wide range of research methods have been used to evaluate ECE programs, including randomized controlled trials, matched comparison group studies, and, more recently, regression discontinuity designs (RDDs) and econometric analyses using large, national datasets.Typically, these research studies attempt to estimate the effect of ECE participation on one or more child development domains, which are cognitive , socioemotional, behavioral, and health outcomes, as well as later outcomes such as crime, teen pregnancy, earnings, and use of or reliance on welfare assistance. Some studies provide longitudinal assessment of these outcomes, tracking the same children beginning at program enrollment, through elementary school, and up to and including adulthood. Others measure immediate effects only or use survey data to estimate long‐term effects  without attempting to measure earlier outcomes.The High/Scope Perry Preschool Project , which operated in Ypsilanti, MI between 1962 and 1967, sought to help “children who were at high risk of school failure to successfully develop the intellectual abilities needed to succeed in school and thus graduate from high school”  participate in other ECE programs, including other Head Start centers, which poses the potential for both substitution and crossover bias.Oklahoma's pre‐K program, a publicly funded universal  preschool program, was established in 1998 and currently serves about 70% of all eligible 4‐year‐olds. In 2003, William Gormley, Ted Gayer, Deborah Phillips, and Brittany Dawson used a regression‐discontinuity design and a sample of more than 2,800 children to compare the test scores of children in the Tulsa Public School District who had attended pre‐K with those who were about to enter pre‐K, reporting separately findings for all racial and ethnic groups . Programs will not be excluded if they also provide ancillary services .Eligible programs need not provide curriculum‐based instruction, be staffed by certified teachers, nor operate for a particular minimum number of hours per day. We will, however, collect information on these program characteristics during coding and, provided we have enough studies, use them as moderators in our analysis. We expect to use these moderators to learn about the plasticity of children's learning and what program variations might improve their skills and achievement.Studies that use student outcomes to measure or compare the effectiveness of teachers or curriculum will not be included in our meta‐analysis. These studies, however, might contain evidence that is relevant to our review, and we will, therefore, make note of these studies so that we might use them to interpret our findings.3.2.2Eligible studies should investigate the effect of an applicable intervention that serves disadvantaged children from low‐income families sometime from birth to age five (with age breaks). We define “disadvantaged” or “low income” as below twice the official U.S. poverty line . Our preliminary research, however, found that many studies use these terms without providing a definition, or that the programs targeted children using factors other than income.We have found that the following terms are compatible and used frequently in the literature: “eligible for free or reduced price lunch” (or “FRPL”), “low income,” “low socioeconomic status,” “poor,” and “at risk.” Studies that use these terms (or slight variations) to define the participants will be eligible for inclusion. We will base this decision on a combination of both the study author's description of the program as well as information reported about the characteristics of the enrolled students. We will capture information about the composition of the sample during study coding and use this information in moderator analyses.When studies evaluate universal programs that serve children from all SES backgrounds, we will only include them if effects for low‐income children are reported separately. When reported, however, these effects are usually the result of subgroup analyses, which are subject to issues related to sample size and statistical power. We will not, therefore, combine the results from universal programs and targeted programs but, instead, will report their results separately. We will also collect information on the percent of enrollees in these universal programs that are from disadvantaged families and, perhaps, use this as a moderator in our analysis.We will exclude studies that target children with severe developmental disabilities, learning disabilities, or chronic medical disorders.3.2.3Eligible studies should either report at least one immediate (or “short‐term”) outcome measure or be a follow‐on to a study that reported one .Cognitive, such as school performance; language, reading, and math skills; grades; and school performance;Health, such as health status, height and weight , obesity, and receipt of dental care; andSocioemotional, such as activity level, aggression, attention, activity level, internalizing and externalizing behaviors, organization/impulse control, sociability, and suspension/expulsion;In addition, the outcome measure(s) reported should fall under one (or more) of three broad categories:More precise outcome domains are described under “synthesis procedures and statistical analysis” below.3.2.4Eligible studies should investigate the effectiveness of participation in ECE programs compared to nonparticipation  and report primary research findings  with at least one eligible outcome measure.Insufficient Causal Validity. We think this is the best approach because it should provide a more precise justification for the decision to include or exclude each study.We do not intend to use study design alone as a criterion to make inclusion/exclusion decisions; rather, our plan is to rigorously review each eligible study for threats to causal validity, as described below  in English. In addition, these studies should have been conducted in the United States or, because of their social and economic conditions, and research and evaluation infrastructures, a Western European country, as defined by the World Bank , or one of the following English‐speaking countries: Australia, Canada, or New Zealand.3.2.61.Is the study reported or available in English?2.Was the study published after 1960?3.Does the study report primary research findings ?4.Is the study an outcome or impact evaluation and does the study report at least one cognitive, socioemotional, behavioral, or health outcome or impactfor children?5.Does the study attempt to attribute these outcomes or impacts to children's participation in what appears to be a preschool‐type program?6.Did the program provide educational instruction directly to children?7.Were children selected for participation because of a severe developmental disability, learning disability, developmental delay, or chronic medical disorder?8.Is there another apparent reason  for excluding this study?9.Is the study eligible for inclusion at this stage?We expect to apply our inclusion criteria in two stages. In the first, the title and abstract for each reference will be screened using the following set of general questions:κ for the reviewers' responses to question eight. If inter‐rater reliability meets or exceeds 0.80, the remaining references will be split in half with one half assigned to each reviewer. If κ falls below 0.80, discrepancies will be resolved by a third reviewer. In this case, another sample of references will be double screened and inter‐rater reliability reassessed (this iterative process will continue until satisfactory inter‐rater reliability is achieved).Given the large number of expected results, we will have two reviewers independently double screen the first 15% of references in order to establish inter‐rater reliability. Reliability will be assessed by calculating Cohen's κ of 0.80 seems impractical after a number of iterations, we would double screen all remaining studies and exclude only those for which both reviewers agree are not relevant.Alternatively, if a In order to screen studies efficiently, we will immediately exclude a study when it fails to meet any one criterion, even if the exclusion happens after the first question of the screening tool. Studies that do not meet the criteria in stage one will be excluded from the review. In the event that there is insufficient information to answer one (or more) of the screening questions, the question will be marked as “cannot tell” and the study will remain eligible for the next stage of screening .1.Was the study conducted in the United States or one of the following countries?a.Australiab.Austriac.Belgiumd.Canadae.Denmarkf.Finlandg.Franceh.Germany i.Greecej.Icelandk.Italyl.Luxembourgm.New Zealandn.Norwayo.Portugalp.Spainq.Swedenr.Switzerlands.The Netherlandst.UK1.Did the intervention target children sometime after birth to age five? 2.Are participants described as, or are effects reported separately for children described as, the following? .a.At‐riskb.Disadvantagedc.Low‐incomed.Low socioeconomic statuse.Poorf.FRPL eligible3.Did the program provide educational instruction to children on topics such as math, language, and science as a primary component of the intervention? .4.Did the program provide only supplemental instruction or tutorial services to children?5.Does the study use student outcomes to evaluate the effectiveness of teacher credentials (or other characteristics), professional development programs for teachers, pedagogical approaches, or curriculum? .6.Did the intervention occur primarily in a center (public or private), faith‐based setting (church or other religious institution), or public school? .7.Did the program operate for at least one academic year (approximately 180 days)? .8.Does the study report at least one immediate outcome or is it a follow‐on to one that did so?9.Does the study employ either a comparison (or control) group, comparison time series, or control variable?10.Is the study eligible for inclusion at this stage?The studies that remain eligible after stage one will be subjected to a full text screening using the following set of more focused questions:κ will be calculated to measure inter‐rater reliability using the responses to question ten. If κ is satisfactory (0.8 or greater), the remaining references will be equally distributed among the reviewers. If not, a third reviewer will resolve the discrepancies and the process will repeat until satisfactory reliability is achieved. Or, if the desired κ does not seem attainable, we will double screen the remaining studies and only exclude studies that both reviewers agree should be removed.As with the first stage, the first 15% of the references will be independently double screened and Cohen's In the event that there is insufficient information with which to answer one (or more) of the screening questions, the answer will be marked as “cannot tell” and the study will remain eligible for inclusion. Studies that meet the criteria in both stages will be assessed for risk of bias (see below).If it is later determined (during the risk of bias assessment or full coding) that a study was improperly screened, it may be excluded from the analysis, and an explanation will be provided in the review.3.3We will attempt to find as many English‐language studies of ECE programs as possible, including gray literature. To do so, we expect to search numerous electronic databases, the websites of research and advocacy organizations and government agencies, and a number of international trial registers, as suggested by the Campbell Collaboration. In addition, we will conduct hand searches of high‐priority journals and review the references of published systematic reviews and meta‐analyses.We will use Zotero to manage our references and, when all searches are complete, to remove duplicates.3.3.1host, a collection of approximately one hundred indices and databases. We will individually search each of the following databases using the EBSCOhost interface (this is the only interface available to us through the University of Maryland libraries with which to search these databases):Academic Search CompleteAcademic Search PremierChicano DatabaseCriminal Justice AbstractseBook CollectionEBSCO eClassics CollectionEconLitEducation Index Retrospective: 1929–1983Education SourceEducation Resource Information Center (ERIC)Family and Society Studies WorldwideFamily Studies AbstractsHistorical Abstracts with Full TextNational Criminal Justice Reference Service AbstractsOpen DissertationsPrimary SearchProfessional Development CollectionPsycARTICLESPsychology and Behavioral Sciences CollectionPsychINFORace Relations AbstractsSocial Work AbstractsSocINDEX with Full TextTeacher Reference CenterThe Nation ArchiveThe National Review ArchiveThe New Republic ArchiveUrban Studies AbstractThe bulk of our electronic database searches will be conducted using EBSCO(preschool OR pre‐school OR prekindergarten OR pre‐kindergarten OR pre‐K OR preK OR “Head Start” OR “early childhood education” OR “early childhood care and education” OR “early N5 education” OR “nursery school” OR ECCE OR ECE) AND  AND  AND (child*) AND Entries in these databases and indices are well catalogued and include reference information and an abstract for each. Thus, we have designed the following search phrase, which concatenates keywords using the boolean operators “OR,” and “AND” to search only the abstracts for each entry:We will not use date or language of publication as search limiters; rather, we will screen studies for date and language relevance during stage one of our inclusion criteria.host search, we plan to search individually the following databases:JSTOROpenGreyWeb of Science Core Collection: Citation IndexesWorldCatIn addition to our EBSCOhost, except that we will apply the search phrase to the abstracts, titles, and keywords (referred collectively to as “topics” in Web of Science) of the references they contain instead of the abstracts only.For Web of Science, we will use a similar procedure to which we will use for EBSCO(preschool OR prekindergarten OR “nursery school”) AND (poor OR “low income” OR disadvantage*) AND (effect* OR outcome* OR impact*) AND  AND (experiment OR quasi‐experiment OR quazi‐experiment OR random*) NOT (autis*)The search tools of the other three, however, are either limited to shorter search phrases or do not allow for  the search term to be applied only to abstracts, and thus we will use the following, shorter boolean search phrase when searching them:If a search returns no results, we will search again using only the keywords contained in the first set of parentheses in the longer search term.3.3.2Abt AssociatesAdministration for Children and Families' National Research Conference on Early ChildhoodAmerican Educational Research AssociationAmerican Institutes for ResearchCenter for Education PolicyCenter for Research and Reform in EducationCenter for Research in Educational PolicyEvidence for Policy and Practice Information and Coordinating Centre (EPPI‐Centre)Chapin HallDanish Clearinghouse for Educational ResearchEducation Endowment FoundationEuropean Early Childhood Education Research AssociationFrank Porter Graham Child Development InstituteMathematica Policy ResearchMDRCNational Center for Education ResearchNational Education AssociationNational Association for the Education of Young ChildrenNational Institute for Early Education ResearchNBER Working paper SeriesOECDPeabody Research InstituteRAND CorporationRegional Educational LaboratoriesU.S. Department of Health and Human ServicesWestatWhat Works ClearinghouseWorldbankOur search will include reviewing the websites of research firms, advocacy groups, institutions of higher learning, and government agencies for technical reports, white papers, and conference proceedings. We will search the following websites:https://10times.com/childhood-education)Annual International Conference on Childhood Education (https://www.openingmindsusa.org/)Early Childhood Care and Education International Conference https://interprofessional.ubc.ca/initiatives/earlyyears2020/)Early Years Conference National Association for the Education of Young Children (NAEYC) Annual Conference National Head Start Conference (http://nrcec.net/)National Research Conference on Early Childhood (https://en.ecceconference.com/)Opening Minds Early Childhood Education Conference . Our preliminary searches yielded more than 100,000 results, which is far more than we can realistically screen, and thus we will limit the number of results to those contained in the first 20 pages .3.3.3We will conduct handsearches of 10–15 high‐priority journals. The journals to be searched will be identified using the results from our database searches and based on their availability at the University of Maryland's libraries. Those journals available at the library and containing the greatest number of references returned in our results will be considered high‐priority.In conducting our handsearches, we will review the table of contents of each volume since 1960, record in Zotero any studies that match our search criteria, and apply our inclusion criteria on‐the‐spot (rather than adding to our reference database and applying at a later date).3.3.4During the preparation of our title registration and the description of prior reviews (see Appendixes A through C), as well as subsequent grant writing activities, we established a list of previous systematic reviews, meta‐analyses, and literature reviews conducted in the area of ECE. We will use the references of these reviews to identify any studies that may have been missed during the first two stages of the search. Any studies that may be related to our topic will be included in our reference list for future screening.3.3.5The Campbell Collaboration provides a list of 22 trial registers. We will review these registers for any ongoing, concluding, or future evaluations of ECE programs. While these studies may not be completed in time to be included in this review, they may be useful in future updates.3.4Meets Standards without Reservations or Meets Standards with Reservations (see “Risk of Bias” below) will undergo full coding. Two reviewers will apply the coding instrument to these studies (a copy of the coding instrument can be found in Appendix E). The coding instrument is designed to extract the following information:Study characteristics ;Intervention characteristics ;Participant characteristics ; andOutcome measures .Studies that have met our inclusion criteria and either When coding for outcome measures, we will attempt to ascertain whether the effects reported are intent‐to‐treat or treatment‐on‐the‐treated estimates and if the measure has face validity.κ, intraclass correlation) to measure inter‐rater reliability. If inter‐rater reliability is not satisfactory  the reviewers will meet and resolve discrepancies by consensus. Another sample of studies will be independently double coded and inter‐rater reliability will be reassessed. When inter‐rater reliability is deemed satisfactory, the remaining studies will be divided in half and each reviewer will be assigned one.We anticipate that we will have too many studies to allow for double coding of each study, and therefore we expect to establish a sample of studies (approximately 15%) that will be independently double coded. Where possible, we will use the appropriate test   rating standards, and it draws from a number of additional sources, that is, the Our rating tool comprises two standards, each composed of two or more criteria, meant to examine broadly the degree of causal validity in each study. These standards evaluate the extent to which the methods used by the researchers appear to account for threats to causal validity that may be the result of selection bias or attrition (as defined below).Meets Standard without Reservations or Does Not Meet Standard, with the exception of the selection standard, which can also be rated as Meets Standard with Reservations.As described below, each standard will be rated according to the extent to which the study satisfies the set of criteria that comprise it. Accordingly, the standards will be rated (borrowing language from the WWC) as either Strong Causal Validity, if the study meets each of the standards without reservations. The only studies eligible for this rating, therefore, would be properly conducted randomized control trials . Studies that evaluate and/or compare multiple variations of a specific program  would be eligible for this rating, provided that the participants were randomly assigned and that there is a nonprogram group that did not receive services from the program.Sufficient Causal Validity, if the study satisfies the selection standard (but does not use random assignment or if randomization was not performed correctly) and it meets the attrition standards .Insufficient Causal Validity, if the study fails to meet any of the standards.When each standard has been rated, an overall study rating will be assigned as follows:One caveat to these rating rules is in the case of studies that use an age cutoff RDD. These studies typically use strict age cutoffs to assign children to either a program or nonprogram group. When limiting the comparison to children just above and just below the cutoff, the assignment is treated “as if” randomization has occurred. These studies, however, may have too few children near the cutoff to generate precise estimates, and thus the researchers may include children further from it to compensate. Presumably, the further children are from the cutoff, the more dissimilar they are, which threatens the causal validity of the findings. Indeed, Thistlewaite and Campbell  argue thSufficient Causal Validity rating as outlined above.Given these two limitations, we will include age cutoff RDDs, but consider them separately, provided they meet the attrition standard of the Insufficient Causal Validity, because, if included in our analysis, their findings, perhaps attributable to one or more of the threats to causal validity, might bias our results. Second, with the remaining studies, we will use the study rating as a moderator in our analysis. Provided we have enough studies, we plan to conduct sensitivity analyses in order to decide whether the results from studies with Strong Causal Validity should be combined with those of Sufficient Causal Validity. We anticipate that the results from these analyses will suggest that there is a statistically significant correlation between study rating and effect size and that we would therefore conclude that these two groups should be analyzed separately.We expect to use these overall study ratings to reduce the risk of bias in our analysis in two ways. First, we will exclude any studies that are rated as Assignment to condition. This criterion evaluates whether participants were randomly assigned to either a program or a nonprogram group, and it is only satisfied when the assignment was, in fact, random. A study does not meet this criterion if a combination of random assignment and matching was used, if randomization was improperly conducted , or if, after randomization, the resulting groups were dissimilar on baseline characteristics and, therefore, required a statistical adjustment (see below).Baseline equivalence. This criterion evaluates the extent to which the groups being compared are similar as measured using baseline measures, including pretest scores and demographic characteristics. It is satisfied if the study meets the baseline equivalence requirement set forth by the WWC. Accordingly, we will calculate an effect size to measure the variation between the groups on pretest scores and for each baseline characteristic reported. We will consider groups to be sufficiently similar when the effect sizes are ≤0.05SD. When an effect size is between 0.05SD and 0.25SD, we will require that the study authors have conducted and reported a statistical adjustment . Effect sizes >0.25SD will be considered unequal, regardless of any adjustments made, and will, therefore, not satisfy the baseline equivalence criterion.The selection standard only applies to studies that use a comparison group, and it comprises two criteria:We will, however, consider the following methods as sufficient to obviate the baseline equivalence criterion: propensity score matching, fixed effects, and difference‐in‐differences.Meets Standard without Reservations, if both criteria are satisfied;Meets Standard with Reservations, if the selection to condition criterion is not met but the baseline equivalence criterion is satisfied; orDoes Not Meet Standard, if the baseline equivalence criterion is not satisfied.The selection standard will be rated as follows:Reporting of attrition information. This criterion evaluates whether attrition information is reported for all groups in the study, and thus it is satisfied only when this information is included in the study.Study attrition. This criterion evaluates the degree of attrition in each study, and it is satisfied if the level of attrition does not exceed the WWC's attrition guidelines for ECE interventions .Substitution bias. This moderator evaluates the extent to which the nonprogram group receives services that are similar to those received by the program group (but not from the program).Crossover bias. This moderator evaluates the extent to which those assigned to the nonprogram group end up in the program group and receive its services (or vice versa). Using a rating system similar to the one employed by Duncan and Magnuson ; and high, if there is evidence of interaction between the groups and the groups are of similar aged children and/or if the likelihood that the interaction had a significant effect on the nonprogram children is high.This moderator evaluates, overall, the extent to which members of the nonprogram group receive the intervention (“crossover bias”), something similar (“substitution bias”), and are affected by it (“diffusion of treatment” and “contagion”) (see below for how each of these types of contamination will be rated). We will rate this threat as follows: low, if the threat is low for each of the four types of contamination; medium, if the threat is medium for only one or two of the types and is low for the others; and high, if the threat is high for any one of the four types or if the threat is medium for more than two.This moderator evaluates both the quality of the program and the degree to which the program was implemented with fidelity. Regrettably, we do not expect to be able to rigorously assess implementation for each program, so if the study authors do not comment on implementation then we will answer the question as “cannot tell.” Otherwise we will rely on study authors to comment on whether the program was implemented with fidelity. We realize that this is not optimal, but we think that it deserves consideration when a study reports an implementation issue.Like Wilson et al. , we willSecond, to reduce the threat of outcome reporting bias, in the event that we cannot calculate an effect size for a reported outcome, we will discuss the finding, especially as it relates to our results, and provide a list of any such outcomes in a table. According to Wilson et al. , these oIn addition, to reduce the risk of location bias, we will search for and include as many relevant English‐language studies as possible, conducted, as mentioned above in the United States, Western Europe, Australia, Canada, and New Zealand.We realize that these approaches do not account for potential biases related to the language of publication or time to publication, and they only partially account for possible bias related to publication status, selective reporting of outcomes, and location of the study. We believe, however, that the threat from these potential biases is small and our approach is sufficient to balance the need for addressing these minor threats and the limited resources available to do so.3.63.6.1We will use R to calculate effect sizes  for the outcomes reported in the study findings, to combine effect sizes, and to perform moderator analyses.3.6.2d and Hedges' g estimates tend to be biased upwards, the latter provides a more precise measure of effect when dealing with small sample sizes and when the SDs of the groups in the study are dissimilar , Hedges' g should produce a more conservative and accurate estimate under these conditions, and thus we plan to use it as our primary index of effect. This should produce a more conservative and more accurate estimate under these conditions. Furthermore, this is a common approach in meta‐analyses of ECE programs, and the preferred option in WWC reviews  measured as a continuous variable, including reading and/or math test scores. For these, we expect to use the standardized mean difference as our index of effect, and although both Cohen's r Ellis, . Since mr Ellis,  had smalg, we will use the formula proposed by Hedges , we will use the odds ratio as our measure of effect. Occasionally, studies may vary in how they measure certain outcomes, sometimes measuring a given outcome construct as dichotomous and sometimes as continuous. In these situations, we will convert the dichotomous effect size first to Cohen's n et al. .3.6.3SDs, and sample sizes) is not included in a study, we will not impute missing values (except for sample sizes when sufficient information is reported), but, instead, contact the study authors to request the missing data. If we are unable to obtain the necessary statistic, we will not include the result in our analysis. We would, however, discuss the finding in our report, especially as it compares to our results. In the event that an outcome is reported as an effect size in another form , we will perform the necessary transformation using the appropriate conversion formula as described in Borenstein et al. (d and from Cohen's d to Hedges' g).Although we expect missing data to be an issue in only a small percentage of studies, when the necessary information , we will include the findings in our review, but not in the meta‐analysis, and they would be mentioned in our discussion of results as either supporting or contrasting evidence.p‐value for each effect size.We are not aware of any evaluations of ECE that use a cluster‐randomized design; however, should one be included in our analysis, we would calculate its effect size using the WWC's guidelines for doing so. In addition, we would apply the clustering correction described by the WWC to adjust the standard error and 3.6.4t distribution, any result of 2.0 or greater is considered large (about two SDs), and hence we would review the study for any apparent plausible explanation , and visually inspect them for apparent outliers. For each identified outlier, we will calculate the sample‐adjusted meta‐analytic deviancy (SAMD) statistic developed and described by Huffcut and Arthur  to measu3.6.5As mentioned earlier, our review is a comprehensive look at the effectiveness of ECE programs across a broad array of outcome measures and ages. To reduce the risk of comparing apples and oranges, we will group outcome measures by age at measurement (using brackets) and then by outcome domain.We expect to use 10 age brackets: (a) infant/toddler, 0–2 years old; (b) preschool, 3–4 years old; (c) kindergarten, 5–6 years old; (d) first grade, 6–7 years old; (e) second grade, 7–8 years old; (f) third through fifth grade, 8–11 years old; (g) middle school, 11–14 years old; (h) high school, 14–18 years old; (i) young adult, 18–25 years old; and (j) adult, 26 years old and over (we will reconsider these age brackets as we identify studies during the review).Cognitive MathematicsSocial‐emotional developmentLanguage developmentAlphabeticsFluencyComprehensionGeneral reading achievementAcademicPlacement in special educationGrade retentionGrades/GPALong‐term outcome measures Educational attainmentCrimeIncomeHealth, including life expectancy and health behaviorsWe have identified the following domains and subdomains in which we intend to group effect sizes: , and the degree to which the outcomes appear to be sufficiently similar.When a domain comprises subdomains, we will consider combining the effect sizes across the subdomains to generate an overall domain average effect size. The decision to do so will depend on a number of factors, including the number of effect sizes, the type of effect sizes and whether they can be combined  at a particular age, we will average the effect sizes using the inverse‐variance weight function , we will calculate the average effect size across these studies for the common outcomes and use this average in our analysis.We will then calculate, as is typical practice, the average across‐study effect size for each domain (and subdomain) using random‐effects inverse‐variance weights and will report 95% confidence intervals for all mean effect sizes.We will consider using robust variance estimation (RVE) if we ultimately perform a meta‐regression and have a sufficient number of studies to apply the method. Hedges, Tipton, and Johnson  caution 3.6.8Q test to determine whether the level of heterogeneity is statistically significant and the I2 statistic to describe the percentage of variation across studies that is due to heterogeneity rather than chance ;Duration/intensity of the program ;Characteristics of the population served ;Study design used ; andCharacteristics of the study .Provided we have enough studies and that the information within the studies permits, we will examine the following moderators for their influence on effect sizes either through subgroup analyses using random‐effects assumptions or by using meta‐regression, as detailed by Higgins and Thompson :CharactWe expect that the number of studies that provide effect sizes for a given domain and age bracket will be small, and therefore our ability to conduct these moderator analyses may be limited.3.6.10Strong Causal Validity to those rates as Sufficient Causal Validity in order to decide whether they should be combined. In addition, we will explore whether and to what extent our decision to Winsorize outlier effect sizes may have affected our findings.We will conduct sensitivity analyses to compare the results of studies rated as Procedures and Standards Handbook. We will, however, explore other methods for interpreting our results, including the empirical benchmarks proposed by Hill et al. . Next, we would organize these codes, first by grouping and then combining them into “descriptive themes.” Last, we would compare the descriptive themes from each of the studies in order to identify patterns and draw conclusions .The analytical themes identified using thematic synthesis can be used to supplement gaps in our review that cannot be filled with quantitative analysis. Kluve et al.  and PetrContent: D. B., D. C., and J. S.Systematic review methods: D. B., D. C., and J. S.Statistical analysis: D. B., D. C., and J. S.Information retrieval: D. B., D. C., and J. S.We received $49,000 from the Jacobs Foundation, $38,080 from an anonymous donor, and support from the University System of Maryland Foundation.http://www.welfareacademy.org/pubs/early_education/index.shtml).We have not been involved with the evaluating of early childhood education programs or in developing a previous systematic review, but we have written a series of assessments of the evaluations of individual early childhood education programs that we have published on our website (Douglas Besharov is responsible for updating the review. Searches for new primary research will be conducted every other year and updates to the review will be made to reflect new studies accordingly.Supporting informationClick here for additional data file.


Major self-mutilation, defined as self-inflicted physical harm without suicidal intent, can be a catastrophic complication of schizoaffective disorder. Oedipism and self-amputation are two sequelae seen in schizoaffective patients. Oedipism is a type of self-mutilation where an individual inflicts an ocular injury to oneself, often leading to blindness. Self-amputation, another complication seen in those with schizoaffective disorder, is defined as the act of deliberately removing healthy limbs. This case report discusses a 39-year-old Ukrainian-American male with a history of schizoaffective disorder who displayed both oedipism and self-amputation behavior of varying extremities. The patient’s plan of care was established once an extensive history was obtained and medical records were consolidated. This report contributes to the literature on rare cases of oedipism and self-amputation in patients diagnosed with schizoaffective disorder, depressed type. Intentional self-injurious or self-mutilating behavior is a perplexing clinical phenomenon, which is defined by Walsh and Rosen as “deliberate, non-life-threatening, self-effected bodily harm or disfigurement of a socially unacceptable nature” . The morIn rare cases, patients may exhibit acts of major self-mutilation, inflicting bodily harm to oneself without suicidal intent, as was seen in 11.3% of patients in one study . OedipisIn this report, we present the case of a 39-year-old white Ukrainian male who had exhibited several episodes of oedipism and self-mutilation.A 39-year-old white Ukrainian-American male lawyer from Kyiv, Ukraine, presented with a chief complaint, which he stated as follows: “I feel original and it empowers me.” He was admitted to the Coney Island Hospital due to a left-arm laceration that he had inflicted upon himself in an attempt to amputate his arm with an electric saw. He had felt the pain to be unbearable, and hence the patient had called an ambulance. A mental status examination was conducted, and it yielded the following findings. The patient’s general appearance at the time of admission was mildly anxious and guarded, and he looked older than his age due to frontal balding. He described his mood as “okay” with a coherent linear goal-directed thought process and content. At that time, the patient presented as guarded, depressed, exhibited poor eye contact, and showed signs of anhedonia such as social withdrawal and negative feelings towards himself. He denied any suicidal or homicidal ideation at the time, and the remainder of the mental status examination was unremarkable.The patient stated that his recent attempt to inflict self-harm had been due to his feeling lonely, hopeless, and depressed secondary to a diagnosis of the human immunodeficiency virus (HIV). He indicated that he drank alcohol to help him cope. The patient appeared uneasy when discussing his HIV diagnosis. It was determined that the incident had occurred in the context of severe major depressive disorder with psychotic features and under the influence of alcohol.His medications upon admission, listed in Table After 11 weeks of treatment at the Coney Island Hospital, he was transferred to the South Beach Psychiatric Center for further stabilization. Past medical and psychiatric historyThe patient had a psychiatric history of schizoaffective disorder, medical history of HIV and hepatitis C (treated during 2008-09), hypothyroidism, right-eye blindness, and below-knee left amputation in 2016 secondary to self-mutilation. His history was remarkable for multiple major self-mutilating behaviors. This had begun in 2014, during a visit to the Netherlands, where he had exposed himself to freezing temperatures that had resulted in frostbite, subsequently leading to the amputation of his left fingers. Within the same year, he had traveled back to Ukraine and had stabbed himself in the right eye with a knife, which had resulted in permanent blindness. During his stay in Ukraine, he had committed two more acts of major self-mutilation; in 2016, he had put his left leg on a train track, which had resulted in a below-knee amputation, and later in 2018, he had dragged his right leg outside a moving tram-train, which had led to the loss of his phalanges. Later that year, he had moved back to the United States, at which point he had attempted to self-amputate his leg by laying on a train track. However, he had been unsuccessful due to local law enforcement intervention. His last attempt at self-mutilation before he had been indefinitely hospitalized had been in December 2018, when he had purchased an electric saw with the intention to amputate his left arm. He had been unsuccessful and called the ambulance due to the pain.The patient had a history of two documented psychotic breakdowns; one in 2009 and the other in 2012. His delusions included paranoid auditory and visual hallucinations with Capgras delusions, which is characterized by a belief in which a familiar person or object is replaced by an imposter. He believed his family members were aliens, that streetlamps were communicating with him, and he was being followed by the government. Upon the patient’s transfer to South Beach Psychiatric Center, his past hospitalization history was revealed, which is summarized in Figure Ultimately, during the hospitalization at Coney Island, he was diagnosed with schizoaffective disorder, depressed type, and he continued to receive clozapine and lithium. He was then transferred to the South Beach Psychiatric Center. Social historyThe patient described his childhood as a happy one; he said had grown up in a supportive home and denied any history of sexual, physical, or emotional trauma. He reported having been raised Roman Catholic but had not practiced his religion and nor had he undergone any significant religious or spiritual experiences during his adult life. He reported that studying philosophy was the “closest thing to spirituality” in his life. His father had died in 2001, at which time he had left for London to pursue a master’s degree in Law, after which he had gone to Columbia in 2004 to attend law school.The patient stated that he had worked as a clerk “reviewing documents” from 2005 to 2013, indicating that he could not find a job as a lawyer due to his poor English language skills. He denied ever practicing as a lawyer and admitted to being unemployed since 2013. He endorsed non-compliance with his work schedule and recurrent problems with his manager, which had subsequently resulted in the termination of his employment. He stated that it was during that time that his depressive symptoms had begun to worsen.He had gotten divorced in 2013 and continued to have an estranged relationship with his ex-wife. It was noted the ex-wife continuously interjected herself into the patient’s healthcare with weekly visits and 10-15 weekly phone calls. The patient expressed his resentment towards her; she refused his visitation rights with his 10-year-old son, whom he had not seen since the age of two.Collateral information from the patient's motherAs per the patient’s mother, she had not observed any self-harming or suicidal behavior in the patient as a child. She endorsed that he had contracted HIV and hepatitis C during his teenage years, from sharing needles and heroin use at the age of 18. She stated that his older brother felt “embarrassed of his diagnosis” and did not keep in touch with him. She reported a series of bizarre behaviors beginning in 2014; he had exhausted all his savings and flown back to his home country, Ukraine, without informing his family and had appeared at his mother’s house in Kyiv before leaving again to “travel the world in 2014.” She observed that he had looked like a “robot” with “glassy eyes.” She stated that while in Ukraine, he had sporadically disappeared into the wilderness and forests without telling her. She endorsed weekly contact whereby he would call her to say, “I am OK. Bye. Talk to you in a week.” Four years later, in February of 2018, he had called her saying, “I am not in a good state, pick me up.” She described his appearance at that time as “very yellow/jaundiced” and he had been missing his left leg below the knee. This had been the first time that she had been made aware of his physical changes, and she had scheduled a psychiatric appointment for him. She had found him a hotel to stay in that night; however, later in the evening, she had found herself on the other end of a phone call with the police who had stated that they had pulled him out from beneath the tram that he had jumped in front of, resulting in the amputation of his right toes. The patient endorsed that this had been a suicide attempt and he had drunk vodka heavily before the attempt to ease the pain.AssessmentAt the South Beach Psychiatric Center, apart from the aforementioned mental status examination findings, the patient initially denied having a significant substance use history, despite his blood alcohol level being 114 upon admission . The patient reported feeling chronically depressed since 2008 (when he had been diagnosed with HIV and hepatitis C). He stated the discovery had made him feel “hopeless” and not worth living.When questioned about his major self-mutilation behaviors, he indicated that he had not been attempting suicide; he denied having delusions or hallucinations during those episodes. Instead, he said he had been attempting to understand “his threshold for pain.” He did admit to one prior episode of suicidal attempt and stated that the subsequent episodes of self-harm had been intentional and said that they had been attempts to “just to injure myself.” He reported loneliness as a contributing factor to his depressed mood. He continuously minimized the seriousness of his self-inflicted injuries, stating “it’s just no big deal, I was depressed, so I did it.” According to the patient, he believed that self-mutilation made him feel “empowered.” He stated that he often abused alcohol to numb some of the pain while committing these acts of self-mutilation.While establishing the patient's care, the biological, psychological, and social factors that potentially affected his condition were analyzed, which is summarized in Table At the unit, the patient exhibited good behavioral control and did not need any pro re nata (PRN) or stat medications. However, he interacted only minimally with staff and peers. He was observed spending a substantial time alone and was reading most of the time.DiagnosisThe patient had an extensive history of paranoid ideation with perceived threats, Capgras delusions, ideas of reference with attribution of salience to irrelevant stimuli, with auditory and visual hallucinations, and bizarre instances of self-injury. The positive symptoms were episodic in nature and were not elicited during his admission. The negative symptoms included social isolation, alogia, avolition, and flat affect. Due to the patient’s psychotic features, depressed mood, and chronic alcohol dependence, it was concluded that the final diagnosis was schizoaffective disorder, depressed type, and substance abuse disorder related to alcohol. Differential diagnosesThe differentials included schizoaffective disorder, depressed type; schizophrenia, undifferentiated; major depressive disorder with psychotic features; personality disorder (cluster B) and schizoid vs schizotypal; body integrity identity disorder; and substance or medication-induced psychotic disorder. Despite the initial focus on depressive symptoms, severe primary psychotic disorder rose high on the differential given the patient’s extensive history of paranoid ideation with perceived threats, Capgras delusions, ideas of reference with attribution of salience to irrelevant stimuli, auditory and visual hallucinations, and bizarre instances of self-injury. A detailed summary of the patient’s differential diagnoses is presented in Table TreatmentThe patient was started on naltrexone 50 mg/day to manage his substance abuse ; this was done to ensure that he maintained his sobriety, which would make him less inclined to self-mutilate since he had a history of such behaviors while intoxicated. Clozapine 400 mg/day was also added to the patient's treatment regimen for his psychosis and to reduce self-harming behaviors . The patOnce the patient was stabilized, he was transferred to a community assisted program in Brooklyn, where he would have around-the-clock supervision, aid with medication management, as well as individual and group therapy. He would continue to receive services through HIV/AIDS Services Administration. Additionally, the patient’s family was educated on the ways to manage the patient’s residual symptoms in the community. He is a Social Security Disability recipient and will receive benefits to support his livelihood in the post-discharge environment.Schizoaffective disorder affects 0.3% of the population; self-mutilating behavior can be a characteristic of schizoaffective disorder . OedipisOne case study highlighted oedipism being triggered by delusional ideas of influence, possession, erotomania, and persecution . Sexual Various theories have been postulated regarding the etiology of self-mutilation ,16. EarlIn addition to the psychological theories behind self-mutilating behaviors, several biological theories have been proposed as well . SeveralBesides the neurobiological bases of self-mutilation, several psychiatric disorders have also been identified . In addiThough the literature describes sexual themes related to oedipism as a result of homosexuality, there was insufficient information about his sexual orientation and his level of comfort with his sexuality to determine if this may have played a contributory role in his oedipism.It is difficult to speculate on the driving force that had led this patient to self-mutilation. It could be postulated that because the patient experienced symptoms of psychosis concurrently with the episodes of self-mutilation, the act of self-mutilation may have been carried out with the intent to increase the secretion of endorphins and dopamine, which are typically lacking in psychotic patients . Though The use of alcohol or drugs, including LSD, cocaine, cannabis, and amphetamines, has been implicated in the amplification of psychotic episodes leading to self-mutilation . The patWe discussed the case of a patient with a history of schizoaffective disorder, depressed type, with major self-mutilation behavior. The patient’s self-mutilation episodes included oedipism and several self-amputations that had occurred over a period of four years. Self-mutilation in patients with psychiatric conditions often involves the genitalia and not the eyes or extremities as seen in this patient. A patient self-inflicting harm, amputation, and even blindness, is scarcely seen in clinical practice. When reviewing the literature, it is evident that each case is unique: from the etiology of each episode of self-mutilation to the ensuing treatment that must address the respective underlying pathology. The literature documents the heterogeneity of etiologies underpinning episodes of self-harm, especially self-amputation and oedipism. This case report contributes to the currently scarce body of literature on patients exhibiting oedipism, self-amputation, and self-mutilation partly due to an underlying psychiatric condition of schizoaffective disorder. Further studies are required to substantiate or rule out any neurobiological and neuroanatomical basis of the self-mutilating behavior. Also, further psychological analyses are warranted to solidify or rule out the psychological basis of self-mutilation.


To understand grieves and struggles of family caregivers providing care for bedridden elderly patients affected by chronic degenerative diseases.This cross-sectional study was developed following the guidelines of the clinical-qualitative method. The sample was composed of 10 female family caregivers of bedridden elderly patients affected by chronic degenerative diseases in a city in the interior of Minas Gerais, Brazil. The sample size was determined by data saturation. The instruments used included semi-structured interviews and a field diary. The audio-recorded interviews were transcribed verbatim and submitted to content analysis. The field diary provided contributions to the organization of categories, conferring a more accurate context.The participants experienced two types of grief, one for the loss of a “healthy family member” and the other for the “announced death” of this person. Additionally, the participants faced two main struggles: overcoming (objective and subjective) fatigue and becoming fully capable of performing their roles.The family caregivers of bedridden elderly patients affected by chronic degenerative diseases experience grieves and struggles that should be taken into account from the beginning of the care process through mental health actions intended to meet their needs. The expression “family caregiver” refers to someone who provides unpaid and usually intuitive assistance to a sick family member, promoting his/her wellbeing.,2 Becaus,. The second axis included 10 questions addressing the care tasks . The third axis included 6 questions that investigated how the family caregiver performed his/her roles . Finally, the fourth axis encompassed 7 questions addressing the family caregivers’ self-care .A semi-structured interview script was used to encourage the participants to verbally express their feelings and thoughts regarding the topic under study. A field After the participants signed a free and informed consent form, the interviews were held in person, according to the participants’ convenient date and time, between October and December 2018. Considering the guidelines for clinical-qualitative studies, interviews preferably take place in the natural setting of care. Hence, dThese aspects were somewhat heterogeneous because participants 1 and 2 opted to be interviewed in the patients’ bedroom, while the remaining participants chose other rooms. Participants 3 and 4 chose to be accompanied by formal caregivers, participants 5 and 7 chose to be accompanied by their sisters, and participant 8 chose her granddaughter. There were no verbal or non-verbal expressions that embarrassed the participants. To reduce eventual biases, the companions also received clarification regarding the target population. Additionally, when the author responsible for data collecting was alone with the participants, she offered the possibility of an additional interview without any companions if the participants desired to review or detail any of the information provided. None of the participants requested an additional interview.The interviews were audio-recorded and lasted 40 minutes on average, and only one interview was conducted with each of the participants. The author responsible for data collection took note in the field diary of observations and reflections regarding the participants’ non-verbal behavior and the home environment. The field diary was filled out immediately after the interviews to avoid potential embarrassment. Data collection was based on current ethical guidelines regulating research with human subjects in Brazil (Resolution CNS No. 466/2012). The study was approved by the Institutional Review Board at the university to which the authors are affiliated (Opinion Report CEP No. 2.798.042).corpus of analysis was mainly composed of verbatim transcriptions of the audio-recorded interviews. This material was submitted to content analysis, as proposed by Bardin. and how long they are providing care (M=8.3 years). This information is presented in n=6) and presented complications and sequelae of neurological diseases, especially Alzheimer’s (n=6).Most participants were married, presented a low level of education, and were the daughters of the elderly patient receiving care. Some disparities were found regarding the participants’ age Report 1: Another situation observed in the research field, in another participant’s home, support this line of reasoning, which led to the following observation recorded on the field diary: “The scene of illness is wide open at the house’s door. You can see from the street the bedridden patient in his bedroom. From the outside you can see that there is someone ill at home”. Report 2 refers to this situation and clarifies that the house was rearranged to confer more comfort to the patient. The patient started occupying one room at the house’s entrance, which used to be a living room. His wife’s bedroom remained in the same place, at the back of the house.It used to be a living room [...], so we installed it [a larger window] there, arranged a single bed for him [...] and used that [a recently built access ramp] to take him outside. (Participant 10) Report 2: It is worth noting that, as showed by Reports 3, 4, and 5, the participants occasionally expressed grief for the patients’ progressive worsening clinical conditions, and as a consequence, sensed his/her finitude. Therefore, it seems reasonable to state that the results concerning the first category reveal that the participants also experienced grief for the “announced death”, though more discreetly, than the grief for the loss of a “healthy family member”. He [patient] is quiet, speechless, doesn’t talk, doesn’t walk [...] He doesn’t know anyone who enters there; he’s on oxygen [...]. (Participant 3)Report 3: He [patient] used to swallow well, used to eat very well, now he doesn’t want anything. (Participant 5) Report 4: Because he still walked when he got sick. He was able to walk before he fell out of bed. So, it’s over for three years now. (Participant 4)Report 5: The second category showed that the participants’ experiences as family caregivers were permeated by two significant struggles. One of which refers to the need to overcome the fatigue caused by their functions. The author responsible for data collection noted that most of the participants showed signs of exhaustion. The following entry in the field diary refers to a specific case: “During the interview, the participant kept her eyes sad and showed signs of tiredness”. Apparently, the participants’ fatigue resulted from two reasons. As shown by Reports 6, 7, and 8, one of which was an objective reason and concerned the care tasks under their responsibility.You get really tired [...]. Sometimes you have to wake up in the middle of the night [to care for the patient]. I put my mobile to wake me up, so I go and take a look at her, you know, to see if she’s choking, you have to keep an eye on that. I sleep beside her. We sleep in the same room. I divorced my husband five years ago, and since then, I sleep with her. (Participant 1)Report 6:: In my free time, I just want to rest. I’m usually tired [for providing care to the patient] in my free time. (Participant 2)Report 7It’s tiresome, you know? [to provide care]. Sometimes, you spend the night without sleep a wink. It’s a struggle. (Participant 6)Report 8: The second reason for the participants’ tiredness, as we inferred, was subjective and related to the complex and gradual process of reconstructing their personal and professional identities, determined by the complete abandonment of their previous social roles, with which they apparently became involved after they became family caregivers. They suggested that their lives were completely changed, and they needed to reinvent themselves after it. Reports 9, 10, and 11 are emblematic in this sense.I didn’t live here, I lived abroad, in another city, another country. But then my mother needed me to come back, and I did. It was a radical change. (Participant 9)Report 9: I was an assistant secretary. I got on leave in September, and it’s been two years. I got on leave to help and take care of my mother, I got on leave ahead of time, you know?(Participant 7)Report 10:I used to do handcrafts, now I don’t anymore... (Participant 8)Report 11: However, this process of identity reformulation has positive aspects as well, considering that most of the participants developed personal resources that started to be used to favor adaptation to the new context caused by the illness of the elderly patient to whom they provide care. As a consequence, they experienced a comforting sense of accomplishment. Some of them clearly stated that their role “is not easy” but “it’s gratifying”.To conclude the second category, we point out that the participants apparently fought another fight, attempting to acquire the skills and competencies necessary to play their family caregiver role, mainly to deal with critical situations, as shown in Report 12. After all, these situations clearly revealed the participants’ limitations - mainly originated from a lack of technical training - regarding the performance of care tasks, consequently raising insecurities. However, as noted by Report 13, what made this struggle particularly challenging, at least for some of the participants, was a fear that the person receiving care would, at some point, require the use of intracorporeal devices.We are not perfect [...] I do my best when she feels sick [...], but I feel insecure. Sometimes I freak out when she [patient] feels sick. (Participant 6)Report 12: I’m afraid that she might need a tube because I have no practice. A breathing tube… Because they [physicians] say that it may get to this point, you know?(Participant 1)Report 13:First, we need to clarify that the participants’ profile is in line with the profile of family caregivers reported by other studies conducted in Brazil concerning sex, kinship, and educational level.,22 RegarIn this sense, as defended by other authors, the resuAccording to the participants’ reports, the grief for an “announced death” suggests anticipatory grief, because it concerns a situation in which death is very likely; however, one cannot estimate when it will actually occur. This is per se, but also from the process of reconstructing their personal and professional identities; a process initiated when they became family caregivers. However, we note that this reconstruction process also entails a positive facet despite the burdensome negative aspects. The family caregivers composing other studies’ samples,(This study also shows that the participants had to deal with the fatigue caused by the role they played in their routine lives, which, in our view, accrued from both the care provided samples,,21 also Furthermore, the participants endeavored to acquire the skills and competencies necessary to perform care tasks, representing part of their struggles. One integrative review suggestsTherefore, the conclusion is that as family caregivers of bedridden elderly patients affected by chronic degenerative diseases, the participants experienced two types of grief, one for the loss of a “healthy family member” and another for the “announced death” of this patient. Note that the first grief stood out and encompassed the loss of the place the patient previously occupied in the family. Additionally, the participants also faced two main struggles: overcoming their objective and subjective fatigue and becoming skillful to perform their roles. These results support the practice of health workers as they reinforce the importance of implementing mental health actions for family caregivers since the beginning of the care process and highlight their need for a support network within and outside the family.This study’s results also pose some questions that deserve to be further explored in the future. Specifically, multi-center, longitudinal studies are recommended to identify potential variations in how anticipatory grief is manifested during the care process. Note that this study has some limitations determined by its cross-sectional nature, in which a phenomenon was described - the experience of family caregivers performing their role within the home context - as spontaneously manifested at a given point in time. Another limitation refers to the fact that the participants were recruited from a single public home care service.