Parental caregiving for a child with a life-limiting condition (LLC) is complex physical and mental work. The impact of this caregiving on parents’ physical health is unknown.OBJECTIVES
(1) To review existing evidence on the physical health of parents caring for a child with a LLC and (2) to determine how physical health of parents is measured.DATA SOURCES
Medline, Embase, PsycINFO, and Cumulative Index of Nursing and Allied Health Literature were searched.STUDY SELECTION
Peer-reviewed articles were included if they reported primary data on the physical health of a caregiver of a child with a LLC. Studies were excluded if they described only the caregiver’s mental health or if the caregivers were bereaved at the time of data collection.DATA EXTRACTION
Of 69 335 unique citations, 81 studies were included in the review.RESULTS
Caregiver health was negatively impacted in 84% of studies. Pain and sleep disturbance were the most common problems. Ways of measuring the physical health of caregiver varied widely. We found an absence of in-depth explorations of the social and economic contexts, which could potentially mitigate the impact of caregiving. Furthermore, we find health interventions tailored to this group remain largely unexplored.LIMITATIONS
Studies were heterogenous in methodology, making comparisons of results across studies difficult.CONCLUSIONS
These findings support the need for improving access to interventions aimed at improving physical health in this population. The rate of health-seeking behaviors, preventive health care access and screening for health conditions is understudied and represent important directions for further research.
Transgender adolescents (TGAs) exhibit disproportionate levels of mental health problems compared with cisgender adolescents (CGAs), but psychosocial processes underlying mental health disparities among TGAs remain understudied. We examined self-reported childhood abuse among TGAs compared with CGAs and risk for abuse within subgroups of TGAs in a nationwide sample of US adolescents.METHODS
Adolescents aged 14 to 18 completed a cross-sectional online survey (n = 1836, including 1055 TGAs, 340 heterosexual CGAs, and 433 sexual minority CGAs). Participants reported gender assigned at birth and current gender identity (categorized as the following: cisgender males, cisgender females, transgender males, transgender females, nonbinary adolescents assigned female at birth, nonbinary adolescents assigned male at birth, and questioning gender identity). Lifetime reports of psychological, physical, and sexual abuse were measured.RESULTS
Seventy-three percent of TGAs reported psychological abuse, 39% reported physical abuse, and 19% reported sexual abuse. Compared with heterosexual CGAs, TGAs had higher odds of psychological abuse (odds ratio [OR] = 1.84), physical abuse (OR = 1.61), and sexual abuse (OR = 2.04). Within separate subgroup analyses, transgender males and nonbinary adolescents assigned female at birth had higher odds of reporting psychological abuse than CGAs.CONCLUSIONS
In a nationwide online sample of US adolescents, TGAs had elevated rates of psychological, physical, and sexual abuse compared with heterosexual CGAs. Risk for psychological abuse was highest among TGAs assigned female at birth. In the future, researchers should examine how more frequent experiences of abuse during childhood could contribute to disproportionate mental health problems observed within this population.
Treating respiratory distress in newborns is expensive. We compared the cost-effectiveness of 2 common noninvasive therapies, nasal continuous positive airway pressure (CPAP) and nasal high-flow (nHF), for newborn infants cared for in nontertiary special care nurseries.METHODS
The economic evaluation was planned alongside a randomized control trial conducted in 9 Australian special care nurseries. Costs were considered from a hospital perspective until infants were 12 months of age. A total of 754 infants with respiratory distress, born ≥31 weeks’ gestation and with birth weight ≥1200 g, <24 hours old, requiring noninvasive respiratory support and/or supplemental oxygen for >1 hour were recruited during 2015–2017. Inpatient costing records were obtained for 753 infants, of whom 676 were included in the per-protocol analysis. Two scenarios were considered: (1) CPAP versus nHF, with infants in the nHF group having "rescue" CPAP backup available (trial scenario); and (2) CPAP versus nHF, as sole primary support (hypothetical scenario). Effectiveness outcomes were rate of endotracheal intubation and transfer to a tertiary-level NICU.RESULTS
As sole primary support, CPAP is more effective and on average cheaper, and thus is superior. However, nHF with back-up CPAP produced equivalent cost and effectiveness results, and there is no reason to make a decision between the 2 treatments on the basis of the cost or effectiveness outcomes.CONCLUSIONS
Nontertiary special care nurseries choosing to use only 1 of the modes should choose CPAP. In units with both modes available, using nHF as first-line therapy may be acceptable if there is back-up CPAP.
To estimate associations between gestational age (GA) and teacher-reported academic outcomes at age 9 years among children born at term (37–41 weeks).METHODS
A secondary data analysis of 1405 children participating in a national US birth cohort study was conducted. At age 9 years, students were evaluated by their teachers in the areas of mathematics, science and social studies, and language and literacy. Unadjusted and adjusted logistic regression models of associations between GA and teacher-reported academic outcomes were estimated and neonatal morbidities were explored as potential pathways.RESULTS
A continuous measure of GA in weeks was significantly associated with above-average rankings in all areas. The associations were similar across outcomes (eg, mathematics [odds ratio (OR): 1.13; confidence interval (CI): 1.02–1.25], science and social studies [OR: 1.13; CI: 1.01–1.26], and language and literacy [OR: 1.16; CI: 1.05–1.28]) in a model that adjusted for child sex, maternal characteristics, and obstetric risk factors and delivery complications. Other specifications indicate a positive association between late term (41 weeks) and mathematics and a negative association between early term (37–38 weeks) and language and literacy, compared with term (39–40). The associations did not appear to operate through neonatal morbidity.CONCLUSIONS
The findings highlight the importance of GA, even at term. Whereas current guidelines suggest waiting until at least 39 weeks to deliver when possible, our findings add to a small group of studies suggesting that GA through 41 weeks is associated with improvements in some educational outcomes.
The mental health of parents of children with medical complexity (CMC) is poorly understood, yet it drives child and family health outcomes. For parents of CMC, compared with parents of noncomplex children with special health care needs (CSHCN) and children without special health care needs (non-CSHCN), we examined self-reported mental health, knowledge of community sources for help, and emotional support.METHODS
Using parent-reported data from the combined 2016–2017 National Survey of Children’s Health, we divided the population into 3 groups: households with CMC, noncomplex CSHCN, and non-CSHCN. We compared these groups regarding the following: (1) parents’ risks for poor or fair mental health and knowledge of where to go for community help and (2) parent-reported sources of emotional support.RESULTS
Of 63 955 588 parent-child dyads (weighted from a sample of 65 204), parents of CMC had greater adjusted odds of reporting poor or fair mental health compared with parents of noncomplex CSHCN (adjusted odds ratio [aOR] 2.0; 95% confidence interval [CI] 1.1–3.8) and non-CSHCN (aOR 4.6; 95% CI 2.5–8.6). Parents of CMC had greater odds of not knowing where to find community help compared with parents of noncomplex CSHCN (aOR 2.1; 95% CI 1.4–3.1) and non-CSHCN (aOR 2.9; 95% CI 2.0–4.3). However, parents of CMC were most likely to report receiving emotional support from health care providers and advocacy groups (P < .001).CONCLUSIONS
Among all parents, those with CMC were at the highest risk to report suboptimal mental health. They more often reported that they do not know where to find community help, but they did say that they receive emotional support from health care providers and advocacy groups. Future researchers should identify ways to directly support the emotional wellness of parents of CMC.
Supplemental Nutrition Assistance Program (SNAP) benefits are designed to buffer families from food insecurity, but studies suggest that most benefits are used by midmonth. In this study, we examined whether the home food environment varies across the SNAP benefits cycle among participating families .METHODS
Participants in this mixed-methods study were 30 SNAP participants who were primary caregivers of a child ages 4–10 years. The home food environment was measured 1 week before SNAP benefit replenishment and again within 1 week after replenishment by using the Home Food Inventory. Household food insecurity was assessed by using the US Department of Agriculture Household Food Security Survey. Wilcoxon rank tests were used to evaluate changes in median Home Food Inventory subscales and food insecurity pre- to post-replenishment. Qualitative interviews with participating caregivers were conducted to explore contextual factors influencing the home food environment across the benefits cycle.RESULTS
Participants had significantly fewer types of vegetables (median: 7.0 vs 8.5, median difference 1.73, 95% confidence interval: 0.5–2.5, P = .03) and higher food insecurity pre- versus post-replenishment (median: 4.0 vs 2.0, median difference 1, 95% confidence interval: 0.1–1.5, P = .03). Caregivers described employing a variety of intentional strategies to reduce cyclic variation in food availability.CONCLUSIONS
Findings suggest that there is relatively limited cyclic variation in the home food environment among families participating in SNAP. This may be explained by a number of assistance programs and behavioral strategies caregivers used to make food last and buffer against scarcity. Future research should evaluate the relationship between the degree of home food environment changes and child health outcomes.
We sought to estimate the prevalence of oral health problems and receipt of preventive oral health (POH) services among children and youth with special health care needs (CYSHCN) and investigate associations with child- and family-level characteristics.METHODS
We used pooled data from the 2016–2018 National Survey of Children’s Health. The analytic sample was limited to children 1 to 17 years old, including 23 099 CYSHCN and 75 612 children without special health care needs (non-CYSHCN). Parent- and caregiver-reported measures of oral health problems were fair or poor teeth condition, decayed teeth and cavities, toothaches, and bleeding gums. POH services were preventive dental visits, cleanings, tooth brushing and oral health care instructions, fluoride, and sealants. Bivariate and multivariable logistic regression analyses were conducted.RESULTS
A higher proportion of CYSHCN than non-CYSHCN received a preventive dental visit in the past year (84% vs 78%, P < .0001). Similar patterns were found for the specific preventive services examined. However, CYSHCN had higher rates of oral health problems compared with non-CYSHCN. For example, decayed teeth and cavities were reported in 16% of CYSHCN versus 11% in non-CYSHCN (P < .0001). In adjusted analyses, several factors were significantly associated with decreased prevalence of receipt of POH services among CYSHCN, including younger or older age, lower household education, non-English language, lack of health insurance, lack of a medical home, and worse condition of teeth.CONCLUSIONS
CYSHCN have higher rates of POH service use yet worse oral health status than non-CYSHCN. Ensuring appropriate use of POH services among CYSHCN is critical to the reduction of oral health problems.
To compare the effectiveness of care management combined with a patient portal versus a portal alone for communication among children with attention-deficit/hyperactivity disorder (ADHD).METHODS
Randomized controlled trial conducted at 11 primary care practices. Children aged 5 to 12 years old with ADHD were randomly assigned to care management + portal or portal alone. The portal included parent-reported treatment preferences and goals, medication side effects, and parent- and teacher-reported ADHD symptom scales. Care managers provided education to families; communicated quarterly with parents, teachers, and clinicians; and coordinated care. The main outcome, changes in the Vanderbilt Parent Rating Scale (VPRS) score as a measure of ADHD symptoms, was assessed using intention-to-treat analysis.RESULTS
A total of 303 eligible children (69% male; 46% Black) were randomly assigned, and 273 (90%) completed the study. During the 9-month study, parents in the care management + portal arm communicated inconsistently with care managers (mean 2.2; range 0–6) but similarly used the portal (mean 2.3 vs 2.2) as parents in the portal alone arm. In multivariate models, VPRS scores decreased over time (Adjusted β = –.015; 95% confidence interval –0.023 to –0.07) in both groups, but there were no intervention-by-time effects (Adjusted β = .000; 95% confidence interval –0.011 to 0.012) between groups. Children who received ≥2 care management sessions had greater reductions in VPRS scores than those with fewer sessions.CONCLUSIONS
Results did not provide evidence that care management combined with a patient portal was different from portal use alone among children with ADHD. Both groups demonstrated similar reductions in ADHD symptoms. Those families with greater care management engagement demonstrated greater reductions than those with less engagement.
Secondary oral vancomycin prophylaxis (OVP) has been used in adults with a history of Clostridioides difficile infection (CDI) while receiving systemic antibiotics to prevent CDI recurrence. However, this practice has not been studied in pediatric patients. The objective of this study was to assess the utility of secondary OVP in pediatric patients with previous CDI who received subsequent antibiotic exposure.METHODS
A multicampus, retrospective cohort evaluation was conducted among patients aged ≤18 years with any history of clinical CDI and receiving systemic antibiotics in a subsequent encounter from 2013–2019. Patients who received concomitant OVP with antibiotics were compared with unexposed patients. The primary outcome was CDI recurrence within 8 weeks after antibiotic exposure. Infection with vancomycin-resistant enterococci and risk factors for CDI recurrence were assessed.RESULTS
A total of 148 patients were screened, of which 30 and 44 patients received OVP and no OVP, respectively. Patients who received OVP had greater antibiotic use and hospital lengths of stay. The incidence of CDI recurrence within 8 weeks of antibiotic exposure was significantly lower in patients who received OVP (3% vs 25%; P = .02) despite this group having notably more risk factors for recurrence. There were no vancomycin-resistant enterococci infections in any patients within either group. After adjustment in a multivariable analysis, secondary OVP was associated with less risk of recurrence (odds ratio, 0.10; 95% confidence interval, 0.01–0.86; P = .04).CONCLUSIONS
Secondary OVP while receiving systemic antibiotics reduces the risk of recurrent CDI in pediatric patients with a history of CDI.
To longitudinally examine the nature of moral distress (MoD) experienced by clinicians caring for extremely low gestational age neonates.METHODS
Neonatologists, medical trainees, and nurses were surveyed at regular intervals on their experience of MoD and their preferred level of care in relation to 99 neonates born <28 weeks’ gestational age managed from birth until discharge or death in 2 tertiary NICUs. Clinicians reporting significant distress (≥6 of 10 on Wocial’s Moral Distress Thermometer) were asked to provide open-ended responses on why they experienced MoD. Descriptive statistics were used to analyze frequency and intensity of MoD across different clinician characteristics. Open-ended responses were analyzed by using mixed methods.RESULTS
Over 18 months, 4593 of 5332 surveys (86% response rate) were collected. MoD was reported on 687 (15%) survey occasions; 91% of neonates elicited MoD during their hospitalization. In their open-ended answers, clinicians invoked 5 main themes to explain their distress: (1) infant-centered reasons (83%), including illness severity, predicted outcomes, and disproportionate care; (2) management plans (26%); (3) family-centered reasons (19%); (4) parental decision-making (16%); and (5) provider-centered reasons (15%). MoD was strongly associated with the perception of "parents wanting too much." Neonatologists experienced less distress and were more likely than nurses and trainees to align preferred levels of care with family wishes.CONCLUSIONS
The majority of preterm infants will generate some MoD; however, it is rarely shared and of a sustained nature. The main constraint reported by clinicians was "parents wanting too much," leading to disproportionate care.
Hospitalizations for ambulatory care sensitive conditions (ACSCs) are thought to be avoidable with high-quality outpatient care. Morbidity related to ACSCs has been associated with socioeconomic contextual factors, which do not necessarily capture the complex pathways through which a child’s environment impacts health outcomes. Our primary objective was to test the association between a multidimensional measure of neighborhood-level child opportunity and pediatric hospitalization rates for ACSCs across 2 metropolitan areas.METHODS
This was a retrospective population-based analysis of ACSC hospitalizations within the Kansas City and Cincinnati metropolitan areas from 2013 to 2018. Census tracts were included if located in a county where Children’s Mercy Kansas City or Cincinnati Children’s Hospital Medical Center had >80% market share of hospitalizations for children <18 years. Our predictor was child opportunity as defined by a composite index, the Child Opportunity Index 2.0. Our outcome was hospitalization rates for 8 ACSCs.RESULTS
We included 604 943 children within 628 census tracts. There were 26 977 total ACSC hospitalizations (46 hospitalizations per 1000 children; 95% confidence interval [CI]: 45.4–46.5). The hospitalization rate for all ACSCs revealed a stepwise reduction from 79.9 per 1000 children (95% CI: 78.1–81.7) in very low opportunity tracts to 31.2 per 1000 children (95% CI: 30.5–32.0) in very high opportunity tracts (P < .001). This trend was observed across cities and diagnoses.CONCLUSIONS
Links between ACSC hospitalizations and child opportunity extend across metropolitan areas. Targeting interventions to lower-opportunity neighborhoods and enacting policies that equitably bolster opportunity may improve child health outcomes, reduce inequities, and decrease health care costs.
Children who receive more responsive care during their early childhood tend to exhibit stronger cognitive development, mental well-being, and physical health across their life course.OBJECTIVE:
Determine how to design effective responsivity training programs for caregivers.DATA SOURCES:
We searched seven electronic databases through October 2020.STUDY SELECTION:
Randomized trials (k = 120) of programs training parents of children ages 0 to 6 to be more responsive.DATA EXTRACTION:
Two reviewers independently extracted data. Data were pooled by using random-effects pairwise and network meta-analyses.RESULTS:
Programs had, on average, a medium effect (d = 0.56; 95% confidence interval [CI]: 0.47 to 0.65). The most effective programs included didactic teaching and opportunities for parents to observe models, practice skills, and receive feedback (d = 1.07; 95% CI: 0.37 to 1.77), or all these instructional methods in addition to reflection (d = 0.86; 95% CI: 0.64 to 1.09). Programs that had participants observe examples of responsivity (d = 0.70; 95% CI: 0.57 to 0.83), used researchers as facilitators (d = 0.89; 95% CI: 0.66 to 1.12), assigned homework (d = 0.85; 95% CI: 0.66 to 1.02), and had a narrow scope (d = 0.72; 95% CI: 0.57 to 0.87) were more effective than those that did not.LIMITATIONS:
Most samples included only mothers from Western countries and lacked follow-up data.CONCLUSIONS:
Having parents observe examples of responsive caregiving and complete home-practice in short, focused programs may be an effective, scalable approach to enhancing responsivity in the general population and reducing inequalities in child development.
Children with isolated unilateral multicystic dysplastic kidney (MCDK) or congenital solitary kidney (CSK) undergo serial renal ultrasonography with variable frequency until they are transitioned to adult care. A growing body of literature suggests the value of frequent ultrasonography in this population is limited, providing no benefit to overall outcomes. Despite emerging evidence, ultrasound remains overused, resulting in avoidable health care expenditures and unnecessary use of resources. With our initiative, we aimed to improve quality of care by reducing avoidable ultrasounds in these children.METHODS
This was a single-center, prospective, interrupted time series of children <18 years with ultrasound-confirmed isolated unilateral MCDK or CSK in the outpatient nephrology clinic to evaluate the effect of a decision-making algorithm on the proportion of children receiving an avoidable ultrasound. An algorithm depicting a consensus, evidence-based protocol for managing pediatric MCDK or CSK was refined through content expert feedback and usability testing to standardize frequency of ultrasonography. Ultrasounds were deemed necessary after birth, at 6 months, and at 2, 5, 10, and 15 years. Differences pre- and postintervention were determined by using a U chart and t and F tests for significance.RESULTS
The algorithm resulted in a 47% reduction (P < .001) in the proportion of avoidable ultrasounds ordered in children with MCDK and CSK. This reduction was sustainable over a 6-month period and would result in at least $46 000 annual savings.CONCLUSIONS
Introduction of a clinical decision-making algorithm was associated with a reduction in avoidable ultrasound testing. Improving adherence across providers may allow for an even more pronounced reduction.
Summary measures are used to quantify a hospital’s quality of care by combining multiple metrics into a single score. We used Baby-MONITOR, a summary quality measure for NICUs, to evaluate quality by race and ethnicity across and within NICUs in the United States.METHODS
Vermont Oxford Network members contributed data from 2015 to 2019 on infants from 25 to 29 weeks’ gestation or of 401 to 1500 g birth weight who were inborn or transferred to the reporting hospital within 28 days of birth. Nine Baby-MONITOR measures were individually risk adjusted, standardized, equally weighted, and averaged to derive scores for African American, Hispanic, Asian American, and American Indian infants, compared with white infants.RESULTS
This prospective cohort included 169 400 infants at 737 hospitals. Across NICUs, Hispanic and Asian American infants had higher Baby-MONITOR summary scores, compared with those of white infants. African American and American Indian infants scored lower on process measures, and all 4 minority groups scored higher on outcome measures. Within NICUs, the mean summary scores for African American, Hispanic, and Asian American NICU subsets were higher, compared with those of white infants in the same NICU. American Indian summary NICU scores were not different, on average.CONCLUSIONS
With Baby-MONITOR, we identified differences in NICU quality by race and ethnicity. However, the summary score masked within-measure quality gaps that raise unanswered questions about the relationships between race and ethnicity and processes and outcomes of care.
Evidence to guide treatment of pediatric medium-chain acyl-coenzyme A dehydrogenase (MCAD) deficiency and phenylketonuria (PKU) is fragmented because of large variability in outcome selection and measurement. Our goal was to develop core outcome sets (COSs) for these diseases to facilitate meaningful future evidence generation and enhance the capacity to compare and synthesize findings across studies.METHODS
Parents and/or caregivers, health professionals, and health policy advisors completed a Delphi survey and participated in a consensus workshop to select core outcomes from candidate lists of outcomes for MCAD deficiency and PKU. Delphi participants rated the importance of outcomes on a nine-point scale (1–3: not important, 4–6: important but not critical, 7–9: critical). Candidate outcomes were progressively narrowed down over 3 survey rounds. At the workshop, participants evaluated the remaining candidate outcomes using an adapted nominal technique, open discussion, and voting. After the workshop, we finalized the COSs and recommended measurement instruments for each outcome.RESULTS
There were 85, 61, and 53 participants across 3 Delphi rounds, respectively. The candidate core outcome lists were narrowed down to 20 outcomes per disease to be discussed at the consensus workshop. Voting by 18 workshop participants led to COSs composed of 8 and 9 outcomes for MCAD deficiency and PKU, respectively, with measurement recommendations.CONCLUSIONS
These are the first known pediatric COSs for MCAD deficiency and PKU. Adoption in future studies will help to ensure best use of limited research resources to ultimately improve care for children with these rare diseases.
The Preschool Obstructive Sleep Apnea Tonsillectomy and Adenoidectomy study is a prospective randomized controlled study of children aged 3 to 5 years. This follow-up evaluated postoperative outcomes 24 months after randomization.METHODS
Baseline, 12-month, and 24-month assessments included intellectual ability, polysomnography, audiology, a pediatric sleep questionnaire, the parent rating scale of the Behavior Assessment System for Children, and the Behavior Rating Inventory of Executive Functioning.RESULTS
In total, 117 (55% male) of 190 children, 61.6% of those initially randomly assigned, attended 24-month follow-up; 62 of 99 were assigned T/A within 2 months (eT/A); and 55 of 91 were assigned to T/A after the 12-month follow-up (T/A12). Intellectual ability, our primary outcome, did not differ according to the timing of T/A. Exploratory analyses revealed changes in both groups after T/A, including fewer children having day sleeps (eT/A from baseline 97% to 11%, T/A12 from 36% at 12 months to 9%), improved symptom scores (eT/A 0.62 to 0.25, T/A12 0.61 to 0.26; P < .001), improved behavior T-scores (eT/A 71.0 to 59.9, T/A12 63.6 to 50.5; P < .001), and improved polysomnography (obstructive apnea-hypopnea index eT/A 1.9 to 0.3 per hour, T/A12 1.3 to 0.3; P < .001). The eT/A group revealed temporary postoperative improvement of Woodcock-Johnson III subscales (sound blending and incomplete word scores) and behavioral withdrawal.CONCLUSIONS
T/A for mild obstructive sleep apnea led to large improvements in sleep and behavior in preschool-aged children, regardless of the timing of surgery.
There are no effective interventions to prevent hospital admissions in infants with bronchiolitis. The American Academy of Pediatrics recommends against routine bronchodilator use for bronchiolitis. The objective of this study was to characterize trends in and outcomes associated with the use of bronchodilators for bronchiolitis.METHODS
This is a multicenter retrospective study of infants <12 months of age with bronchiolitis from 49 children’s hospitals from 2010 to 2018. The primary outcomes were rates of hospital admissions, ICU admissions, emergency department (ED) return visits after initial ED discharge, noninvasive ventilation, and invasive ventilation. Multivariable logistic regression was used to evaluate the rates of outcomes among hospitals with high and low early use of bronchodilators (on day of presentation).RESULTS
A total of 446 696 ED visits of infants with bronchiolitis were included. Bronchodilator use, hospital admissions, and ED return visits decreased between 2010 and 2018 (all P < .001). ICU admissions and invasive and noninvasive ventilation increased over the study period (all P < .001). Hospital-level early bronchodilator use (hospitals with high versus low use) was not associated with differences in patient-level hospital admissions, ICU admissions, ED return visits, noninvasive ventilation, or invasive ventilation (all P > .05).CONCLUSIONS
In a large study of infants at children’s hospitals, bronchodilator therapy decreased significantly from 2010 to 2018. Hospital-level early bronchodilator use was not associated with a reduction in any outcomes. This study supports the current American Academy of Pediatrics recommendation to limit routine use of bronchodilators in infants with bronchiolitis.
Primary Sjögren syndrome is an autoimmune disease characterized by inflammation of the salivary and lacrimal exocrine glands but can also present with systemic extraglandular manifestations, including pulmonary disease. Commonly described pulmonary manifestations of Sjögren syndrome include airway disease, interstitial lung disease, pulmonary arterial hypertension, and lymphoproliferative disorders. However, diffuse alveolar hemorrhage as a sequela of Sjögren syndrome has rarely been described in the adult literature and has never been described in a child. Here we report the case of an 11-year-old girl who presented with diffuse alveolar hemorrhage and was diagnosed with childhood-onset Sjögren syndrome who otherwise lacked typical clinical features, such as sicca symptoms, at the time of presentation. She was successfully treated with corticosteroids and rituximab, with sustained pulmonary remission 1 year post diagnosis. Our case highlights the heterogenous presentation of Sjögren syndrome in the pediatric population and the need for increased awareness among pediatric providers to recognize potential systemic manifestations of this disease to avoid delayed diagnosis.