Christopher S. Greeley

Report of the Tennessee Task Force on Screening Newborn Infants for Critical Congenital Heart Disease

A member of the Tennessee state legislature recently proposed a bill that would mandate all newborn infants to undergo pulse oximetry screening for the purpose of identifying those with critical structural heart disease before discharge home. The Tennessee Task Force on Screening Newborn Infants for Critical Congenital Heart Defects was convened on September 29, 2005. This group reviewed the current medical literature on this topic, as well as data obtained from the Tennessee Department of Health, and debated the merits and potential detriments of a statewide screening program. The estimated incidence of critical congenital heart disease is 170 in 100000 live births, and of those, 60 in 100000 infants have ductal-dependent left-sided obstructive lesions with the potential of presentation by shock or death if the diagnosis is missed. Of the latter group, the diagnosis is missed in ∼9 in 100 000 by fetal ultrasound assessment and discharge examination and might be identified by a screening program. Identification of the missed diagnosis in these infants before discharge could spare many of them death or neurologic sequelae. Four major studies using pulse oximetry screening were analyzed, and when data were restricted to critical left-sided obstructive lesions, sensitivity values of 0% to 50% and false-positive rates of between 0.01% and 12% were found in asymptomatic populations. Because of this variability and other considerations, a meaningful cost/benefit analysis could not be performed. It was the consensus of the task force to provide a recommendation to the legislature that mandatory screening not be implemented at this time. In addition, we determined that a very large, prospective, perhaps multistate study is needed to define the sensitivity and false-positive rates of lower-limb pulse oximetry screening in the asymptomatic newborn population and that there needs to be continued partnering between the medical community, parents, and local, state, and national governments in decisions regarding mandated medical care.

Fractures at Diagnosis in Infants and Children With Osteogenesis Imperfecta

Background: In infants and children with fractures from an unclear cause, osteogenesis imperfecta (OI) is often included as a potential etiology. In infants and children with OI there exists a gap in the published literature regarding the fracture pattern seen at the time of diagnosis. As an additional aid to the diagnosis of OI, we sought to characterize the fracture patterns in infants and children at the time of their diagnosis. Methods: We performed a retrospective chart review of a series of infants and children under 18 years of age who have the diagnosis of OI (any type) from a single institution. Results: We identified 68 infants and children with OI: 23 (34%) type 1, 1 (2%) type 2, 17 (25%) type 3, 24 (35%) type 4, and 3 (4%) unknown type. A family history of OI was present in 46% of children. Forty-nine (72.0%) patients were diagnosed solely on clinical characteristics, without genetic or fibroblast confirmation. Rib fractures were noted in 21% of the subjects with none being identified during infancy. The number of fractures identified at diagnosis ranged from 1 to >37 with 7 (10%) having more than 2 fractures. All subjects with more than 2 fractures were diagnosed prenatally or in the immediate newborn period. Seventeen (25%) infants were diagnosed after 1 week of age but before 12 months of age. None of these infants had either rib fractures or more than 1 fracture at the time of diagnosis. Conclusions: The majority of children diagnosed with OI are diagnosed by clinical features alone. The fracture pattern at the time of diagnosis in OI is variable with 10% having more than 2 fractures. The diagnosis of OI was made in utero or at delivery in 43% of children. Multiple rib fractures in an infant would be an unexpected finding in OI. Level of Evidence: Level III.

Lynøe et al. – #theRestoftheStory

Lynøe et al. is an adjunct to a larger report by the Swedish Agency For Health Technology Assessment And Assessment Of Social Services (‘SBU’) entitled ‘Traumatic shaking – The role of the triad in medical investigations of suspected traumatic shaking’ (1). Lynøe et al. purport to rigorously, objectively and transparently examine the evidence-based medical literature on shaken baby syndrome (SBS) under well-established guidelines of systematic reviews (2). And, having done so, it concludes that there is ‘very low’ scientific evidence supporting the premise that the ‘triad’ implies that an infant has been violently shaken, and ‘low’ scientific evidence supporting the assumption that shaking an infant causes the ‘triad’ (3). Despite a number of high-quality systematic reviews on the diagnosis of abusive head trauma (AHT), another rigorous review of the published literature would be a welcome addition to the growing body of literature on the condition. Unfortunately, there are significant concerns about the methodology, objectivity and transparency of this study.

Maltreatment of Children Under Age 2 With Specific Birth Defects: A Population-Based Study.

BACKGROUND AND OBJECTIVES: Children with disabilities are at an increased risk for maltreatment. However, the risk of maltreatment is unknown for children with specific types of birth defects. This study was conducted to determine whether the risk and predictors of maltreatment differ between children with and without 3 birth defects: Down syndrome, cleft lip with/without cleft palate, and spina bifida. METHODS: This population-based study of substantiated childhood maltreatment was conducted in Texas from 2002 to 2011. Linked data were used to describe the risk and types of maltreatment that occurred before age 2 years in children with and without specific birth defects. Poisson regression was used to identify predictors of maltreatment and assess differences in those predictors between children with and without these specific birth defects. RESULTS: The risk of maltreatment (any type) in children with cleft lip with/without cleft palate and spina bifida was increased by 40% and 58%, respectively, compared with children with no birth defects. The risk of any maltreatment was similar between children with Down syndrome and unaffected children. Across birth defect groups, the risk of medical neglect was 3 to 6 times higher than in the unaffected group. Child-, family-, and neighborhood-level factors predicted maltreatment in children with and without birth defects. CONCLUSIONS: The overall risk of substantiated maltreatment was significantly higher for some but not all birth defect groups. The factors associated with increased risk were similar across groups. Enhancement of existing maltreatment prevention and early intervention programs may be effective mechanisms to provide at-risk families additional support.

The Future of Child Maltreatment Prevention

I THE UNITED States there are an estimated 900 000 children per year who are victims of child maltreatment.1 The majority of them are victims of neglect. Over the past 3 decades, there has been considerable attention to better defining our understanding of the etiology of child maltreatment. Initial work focused on sociological and psychodynamic models of child maltreatment. These models laid the ground work for the current ecological model of child maltreatment.2 The ecological model endorses a complex set of forces that create an environment which places the child at greater risk for becoming a victim of maltreatment. Although historically “bad” or “sick” people abused children, current models view children as embedded in layers and layers of varying positive and negative spheres of influences. These influences change throughout different periods in the life of a child. Each of these forces can be either stressors (risks) or supports (protectors) of the child. The limited understanding of the complexity of risks has resulted in a large variety of approaches to child maltreatment prevention. Strategies that have had the most rigorous evaluation, and strongest evidence, involve in-home visitation with either a nurse3 or a social worker4 and in-hospital parental education and engagement.5 Other promising prevention strategies include parental education about normal infant crying6 and infant soothing techniques,7 parent support programs,8 and community-based family engagement.9 Sadly, many interventions have limited evidence of their effectiveness, and few are subject to rigorous evaluation. This trend is changing. The past decade has seen the rise of evidence-based practice in the child maltreatment– prevention field,10 but randomized trials are still quite rare. This issue of Pediatrics includes the report of a cluster-randomization trial of an intervention that could bring child maltreatment prevention to the pediatric office.11 Dubowitz et al11 help move the field forward by presenting the Safe Environment for Every Kid (SEEK) model. The SEEK model consists of (1) specially trained (resident) physicians, (2) additional parental resource information, (3) administration of a screening questionnaire, and (4) a dedicated study social worker. The simplicity of the intervention is laudable. The authors report that the SEEK model resulted in fewer child protective service reports in the study population. Although the trial involved large numbers (558 parents completed the study), there was a 23% drop-out rate. Because only parents who completed the protocol were analyzed, the higher-risk parents may have selectively dropped out. Because this was a cluster trial, it is unclear if this design may have introduced bias into the final sample.12 Despite this limitation, the magnitude of the effect is quite striking. Because this trial was performed in a pediatric resident continuity clinic, the authors are correct to endorse replication of the model in different practice scenarios before it is widely implemented. There will be no single silver-bullet program that will “fix the problem of child abuse.” The future of child maltreatment prevention will involve evaluating which mix of programs at what dosage, for what duration, and at which time in a child’s life achieves the greatest impact. It will also be important to evaluate how these programs are blended into communities with policies that are optimized for children and families, which will necessitate rigorous, theory-based, prospective, multicommunity studies. A barrier to these efforts is the limited regional or national coordination for child maltreatment–prevention efforts. Effective, sustainable child maltreatment–prevention strategies will require a shift in thinking from child maltreatment as a disease to thinking that child maltreatment is a symptom of a larger family, community, or societal disease. Other symptoms of the larger societal disease include 9 million children without health insurance,13 25% of 9th-graders not finishing high school,14 12.6 million US households with food insecurity,15 and at least 13 million children living in poverty.16 We need not only to know the physical health of the children we see but also the health of the community in which they live. The underlying, perhaps unstated, goal of child maltreatment–prevention initiatives is a significant improvement in the lives of children, which is a shift from child maltreatment prevention to child well-being promotion.17 We need to use the ameliorative tools we have at our disposal (home visitation, in-hospital education, and now, perhaps, in-office engagement) together in ways to affect larger transformative changes to the social norms.18 Sustainable prevention initiatives will move beyond simply more or better “programs” (ie, a larger “army of therapists”). Sustainable prevention initiatives involve leveraging current, and perhaps future,

Shaken baby syndrome and forensic pathology

The controversy [1] that has surrounded abusive head trauma (AHT) for the past decade is, at its core, fabricated. The premise that the vigorous shaking of an infant is dangerous to that infant is agreed upon by all but the most adamant critics. The complex features of AHT are often disparagingly distilling simply to ‘‘The Triad’’; a term devoid of any real clinical meaning and not used at all in practice. There are two primary drivers of the current debate over AHT. Firstly, there are alternative hypotheses that have seemingly been developed primarily for use in court, as opposed to a clinical or scientific need. These theories serve to create confusion in the courtroom and distract attention from AHT as a cause of injury or death. Secondly, the medical literature is misused in ways to create an illusion of evidence to support these novel and tangential theories. This misuse results in a mélange of support for otherwise speculative theories that are often unrecognizable to clinicians caring for children and creating confusion for families. False controversy

The Evolution of the Child Maltreatment Literature

* Abbreviations: AHT — : abusive head trauma The first descriptions of the injuries associated with child abuse are commonly attributed to the French pathologist Auguste Ambroise Tardieu.1,2 His “Etude medico-legale sur les sevices et mauvais traitements exerces sur des enfants” (Forensic Study on Cruelty and the Ill Treatment of Children) in 18603 described findings of child fatalities from “acts of cruelty and ill treatment, of which young children fall victim from their parents, their schoolmasters, and all who exert over these children some degree of authority.”2 John Caffey reintroduced the study of child abuse in 19464 when he described 6 infants with chronic subdural hematomas in whom he identified long bone fractures from an “obscure” traumatic origin. Fredrick Silverman, a junior associate of Caffey’s,5 collaborated with C. Henry Kempe and colleagues in … Address correspondence to Christopher Greeley, MD, MSB 2.104, 6431 Fannin St, University of Texas Health Science Center at Houston, Houston, TX 77030. E-mail: christopher.s.greeley{at}uth.tmc.edu

The Scope and Trends of Pediatric Hospitalizations in Texas, 2004–2010

OBJECTIVE To examine demographics and trends of financial cost and prominent diseases/conditions resulting in inpatient hospitalizations for infants, children, and adolescents in Texas between 2004 and 2010. METHODS Longitudinal retrospective cross-sectional study using the Texas Hospital Inpatient Discharge Database, including all pediatric hospitalizations in the state of Texas, 2004 to 2010. RESULTS Texas has an average of 591 571 pediatric hospitalizations per year. Birth was the most common reason for hospitalization, representing 64% of all pediatric hospitalization annually in Texas. Respiratory illnesses were the most common discharge diagnosis for hospitalized children ages 1 month to 9 years and demonstrated a 2% decrease over the study period. The rate of hospitalizations for digestive conditions and childbirth also demonstrated a decrease over this time frame: 4.7% and 3.0%, respectively. The rate of mental illness diagnoses increased 2.5% over the time frame and represented the most common discharge diagnosis for children aged 10 to 14. Childbirth was the most common reason for hospitalization for adolescents aged 15 to 17 years. There was no increase in total cost of pediatric hospitalizations over the time period under study. CONCLUSIONS After birth, respiratory illnesses represent the most common reason for hospitalization for children (between 1 month and 10 years of age) in Texas. Mental health conditions and childbirth represent the most common reason for hospitalization for young adolescents (10-14 years) and older adolescents (15-17 years), respectively.

Potential Opportunities for Prevention or Earlier Diagnosis of Child Physical Abuse in the Inpatient Setting

OBJECTIVES To compare rates of previous inpatient visits among children hospitalized with child physical abuse (CPA) with controls as well as between individual abuse types. METHODS In this study, we used the Pediatric Health Information System administrative database of 44 childrens hospitals. Children <6 years of age hospitalized with CPA between January 1, 2011, and September 30, 2015, were identified by discharge codes and propensity matched to accidental injury controls. Rates for previous visit types were calculated per 10 000 months of life. χ2 and Poisson regression were used to compare proportions and rates. RESULTS There were 5425 children hospitalized for CPA. Of abuse and accident cases, 13.1% and 13.2% had a previous inpatient visit, respectively. At previous visits, abused children had higher rates of fractures (rate ratio [RR] = 3.0 times; P = .018), head injuries (RR = 3.5 times; P = .005), symptoms concerning for occult abusive head trauma (AHT) (eg, isolated vomiting, seizures, brief resolved unexplained events) (RR = 1.4 times; P = .054), and perinatal conditions (eg, prematurity) (RR = 1.3 times; P = .014) compared with controls. Head injuries and symptoms concerning for occult AHT also more frequently preceded cases of AHT compared with other types of abuse (both P < .001). CONCLUSIONS Infants hospitalized with perinatal-related conditions, symptoms concerning for occult AHT, and injuries are inpatient populations who may benefit from abuse prevention efforts and/or risk assessments. Head injuries and symptoms concerning for occult AHT (eg, isolated vomiting, seizures, and brief resolved unexplained events) may represent missed opportunities to diagnose AHT in the inpatient setting; however, this requires further study.

Diagnosis in Infants and Children With Osteogenesis Imperfecta.

In reply: We are grateful for Drs Shelmerdine, Arthurs, and Calder’s interest in our article “Fractures at diagnosis in infants and children with osteogenesis imperfecta.” We would like to address the concerns raised by the authors in their letter,2 as we believe that they arise from a misunderstanding of the intent and methodology of our study. First, Dr Shelmerdine and colleagues criticize our study as both not having a control group and not undertaking “genetic testing and confessionals” on all subjects. We believe that this criticism is unwarranted and stems from a misunderstanding of the purpose of our study. As stated in our paper, the study was intended to be purely descriptive in nature. We stated that the purpose of the study was to “characterize the fracture patterns and clinical information relied upon by clinicians at the time of diagnosis in a sample of infants and children with OI.”1 While we hypothesized that the pattern difference would exist, we were careful not to state that our study was designed to demonstrate that. As fracture patterns at the time of diagnosis of osteogenesis imperfecta (OI) in children had not been described prior, we felt, and still do, that there is value to begin to identify if a true overlap of fracture pattern does indeed exist. Our data, while limited to a retrospective chart review, did not demonstrate any similarity to fracture patterns often described in infant victims of abuse. We recognize that our statement “infants with multiple rib fractures are unlikely to be suffering from undiagnosed OI” may seem generous to Dr Shelmerdine and colleagues. Given that our study is the only one to report fracture patterns in infants at the time of diagnosis of OI in the literature and that no new data have emerged in the 2 years since publication, that conclusion remains unchallenged. As noted by Dr Shelmerdine and colleagues, we did identify 22% of the subjects did indeed have rib fractures, with 13/15 presenting antenatally or in the immediate newborn period. None of our subjects developed rib fractures in infancy (28d to 12mo of life). As we noted in our response to Dr Paterson’s letter,3,4 birth is a profoundly traumatic experience and it remains unlikely that an infant with a predisposition to fractures would be born without skeletal injury, but then readily fracture as an infant. Along these lines, Dr Shelmerdine and colleagues express concern that our study will be used to “to refute an alternative diagnosis in a child with multiple rib injuries.”2 While the judicial process can torture published data beyond recognition, we were careful not to imply that OI is not a serious consideration when evaluating a child with seemingly unexplained fracture. OI is a meaningful consideration in all infants and children with seemingly unexplained fractures. The purpose of the study was to begin to explore how similar, at the time of the diagnosis of OI, these conditions really were. The first step, in our study, is to describe the fracture pattern at the time of OI diagnosis. In that context, we are unclear what Dr Shelmerdine and colleagues meant regarding alternative diagnoses in children with multiple rib injuries. If there exist data demonstrating that children at the time of diagnosis with OI can present (outside of the immediate newborn period) with multiple rib fractures, we are unaware of it. Second, Dr Shelmerdine and colleagues indicate that OI and physical abuse of a child are not mutually exclusive. We agree. They further interpret our paper as implying that we were stating that we could indicate whether a fracture was intentional or accidental based upon radiographic imaging. We regret that we may have given Dr Shelmerdine and colleagues that impression, but are unclear where in the manuscript that was implied. Again, as a descriptive study of fracture patterns in children at the time of diagnosis of OI, we in no way were exploring causes or mechanism of injury: accidental or inflicted. Lastly, Dr Shelmerdine and colleagues also are critical of our paper in not presenting radiographic imaging. We agree and as we noted such in our manuscript. We urged caution in overinterpreting our data stating “we likely have undercounted the number of fractures because as a record review we did not review the radiographs individually and some subjects may have not had complete skeletal assessments at the time of diagnosis.”1 We are grateful for Drs Shelmerdine, Arthurs, and Calder’s interest in our paper and appreciate their thoughtful comments.