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OBJECTIVE: The Hydrocephalus Clinical Research Network (HCRN) implemented a perioperative infection prevention bundle for all CSF shunt surgeries in 2007 that included the relatively unproven technique of intrathecal instillation of the broad-spectrum antibiotics vancomycin and gentamicin into the shunt. In the meantime, the field debated the use of antibiotic-impregnated catheter (AIC) shunt tubing using clindamycin and rifampin, an increasingly widespread, but expensive and controversial, technique. It is unknown whether there were changes in infecting organisms associated with the use of these techniques during CSF shunt surgery at the hospital level. Key comparison periods include during the use of intrathecal antibiotics (period 1 from June 1, 2007, to December 31, 2011, at HCRN hospitals) and AIC (period 2 from January 1, 2012, to December 31, 2015, at HCRN as well as increasing over time at non-HCRN hospitals) and only standard use of routine prophylactic antibiotics (period 1 at non-HCRN hospitals). The aim of this study was to examine rates of CSF shunt surgery-related infections from 2007 to 2012 at the hospital level, including HCRN and non-HCRN hospitals, with a focus on infections with gram-negative organisms. METHODS: The authors conducted a retrospective observational cohort study at 6 children's hospitals with enrollment from 2007 to 2012 and surveillance through 2015. Bimonthly rates of shunt surgery-related infections were summarized to produce an overall hospital-specific time series, as well as by HCRN/non-HCRN status. An interrupted time series analysis was performed to assess the impact of change in HCRN perioperative infection prevention bundle on overall bimonthly infection rates. Quarterly rates of gram-negative shunt surgery-related infections were summarized to produce an overall hospital-specific time series. RESULTS: The overall bimonthly CSF shunt infection rate over time did not change significantly from 2007 to 2012. There was no difference in the trajectory of infection rates between HCRN and non-HCRN hospitals during the entire study period. No change in distributions of gram-negative organism infections was observed in hospitals from 2007 to 2015. CONCLUSIONS: There were no differences observed in hospital-level infection rates for low-risk patients undergoing CSF shunt surgery. This included analyses based on participation in the HCRN network, given their regular use of intrathecal antibiotics in period 1 and a focus on gram-negative infections with increasing adoption of AICs in period 2.
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BACKGROUND: Cerebrospinal fluid (CSF) shunts allow children with hydrocephalus to survive and avoid brain injury (J Neurosurg 107:345-57, 2007; Childs Nerv Syst 12:192-9, 1996). The Hydrocephalus Clinical Research Network implemented non-randomized quality improvement protocols that were shown to decrease infection rates compared to pre-operative prophylactic intravenous antibiotics alone (standard care): initially with intrathecal (IT) antibiotics between 2007-2009 (J Neurosurg Pediatr 8:22-9, 2011), followed by antibiotic impregnated catheters (AIC) in 2012-2013 (J Neurosurg Pediatr 17:391-6, 2016). No large scale studies have compared infection prevention between the techniques in children. Our objectives were to compare the risk of infection following the use of IT antibiotics, AIC, and standard care during low-risk CSF shunt surgery (i.e., initial CSF shunt placement and revisions) in children. METHODS: A retrospective observational cohort study at 6 tertiary care children's hospitals was conducted using Pediatric Health Information System + (PHIS +) data augmented with manual chart review. The study population included children ≤ 18 years who underwent initial shunt placement between 01/2007 and 12/2012. Infection and subsequent CSF shunt surgery data were collected through 12/2015. Propensity score adjustment for regression analysis was developed based on site, procedure type, and year; surgeon was treated as a random effect. RESULTS: A total of 1723 children underwent initial shunt placement between 2007-2012, with 1371 subsequent shunt revisions and 138 shunt infections. Propensity adjusted regression demonstrated no statistically significant difference in odds of shunt infection between IT antibiotics (OR 1.22, 95% CI 0.82-1.81, p = 0.3) and AICs (OR 0.91, 95% CI 0.56-1.49, p = 0.7) compared to standard care. CONCLUSION: In a large, observational multicenter cohort, IT antibiotics and AICs do not confer a statistically significant risk reduction compared to standard care for pediatric patients undergoing low-risk (i.e., initial or revision) shunt surgeries.
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Antibacterianos , Profilaxis Antibiótica , Derivaciones del Líquido Cefalorraquídeo , Humanos , Derivaciones del Líquido Cefalorraquídeo/efectos adversos , Antibacterianos/administración & dosificación , Estudios Retrospectivos , Niño , Masculino , Preescolar , Femenino , Lactante , Profilaxis Antibiótica/métodos , Adolescente , Inyecciones Espinales , Hidrocefalia/cirugía , Catéteres de Permanencia/efectos adversos , Infección de la Herida Quirúrgica/prevención & control , Infecciones Relacionadas con Catéteres/prevención & control , CatéteresRESUMEN
OBJECTIVE: The objective of this study was to describe trends in the utilization of infection prevention techniques (standard care, intrathecal [IT] antibiotics, antibiotic-impregnated catheters [AICs], and combination of IT antibiotics and AICs) among participating hospitals over time. METHODS: This retrospective cohort study at six large children's hospitals between 2007 and 2015 included children ≤ 18 years of age who underwent initial shunt placement between 2007 and 2012. Pediatric Health Information System + (PHIS+) data were augmented with chart review data for all shunt surgeries that occurred prior to the first shunt infection. The Pearson chi-square test was used to test for differences in outcomes. RESULTS: In total, 1723 eligible children had initial shunt placement between 2007 and 2012, with 3094 shunt surgeries through 2015. Differences were noted between hospitals in gestational age, etiology of hydrocephalus, and race and ethnicity, but not sex, weight at surgery, and previous surgeries. Utilization of infection prevention techniques varied across participating hospitals. Hydrocephalus Clinical Research Network hospitals used more IT antibiotics in 2007-2011; after 2012, increasing adoption of AICs was observed in most hospitals. CONCLUSIONS: A consistent trend of decreasing IT antibiotic use and increased AIC utilization was observed after 2012, except for hospital B, which consistently used AICs.
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Antibacterianos , Hidrocefalia , Niño , Humanos , Estados Unidos/epidemiología , Lactante , Estudios Retrospectivos , Antibacterianos/uso terapéutico , Derivaciones del Líquido Cefalorraquídeo/efectos adversos , Catéteres , Hidrocefalia/cirugía , Hidrocefalia/tratamiento farmacológicoRESUMEN
BACKGROUND: Infection prevention techniques used during cerebrospinal fluid (CSF) shunt surgery include: (1) standard perioperative intravenous antibiotics, (2) intrathecal (IT) antibiotics, (3) antibiotic-impregnated catheter (AIC) shunt tubing, or (4) Both IT and AIC. These techniques have not been assessed against one another for their impact on the infecting organisms and patterns of antimicrobial resistance. METHODS: We performed a retrospective longitudinal observational cohort study of children with initial CSF shunt placement between January 2007 and December 2012 at 6 US hospitals. Data were collected electronically from the Pediatric Health Information Systems+ (PHIS+) database, and augmented with standardized chart review. Only subjects with positive CSF cultures were included in this study. RESULTS: Of 1,723 children whose initial shunt placement occurred during the study period, 196 (11%) developed infection, with 157 (80%) having positive CSF cultures. Of these 157 subjects, 69 (44%) received standard care, 28 (18%) received AIC, 55 (35%) received IT antibiotics, and 5 (3%) received Both at the preceding surgery. The most common organisms involved in monomicrobial infections were Staphylococcus aureus (38, 24%), coagulase-negative staphylococci (36, 23%), and Cutibacterium acnes (6, 4%). Compared with standard care, the other infection prevention techniques were not significantly associated with changes to infecting organisms; AIC was associated with decreased odds of methicillin resistance among coagulase-negative staphylococci. CONCLUSIONS: Because no association was found between infection prevention technique and infecting organisms when compared to standard care, other considerations such as tolerability, availability, and cost should inform decisions about infection prevention during CSF shunt placement surgery.
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Antibacterianos , Coagulasa , Humanos , Niño , Antibacterianos/uso terapéutico , Estudios Retrospectivos , Catéteres , Derivaciones del Líquido Cefalorraquídeo/efectos adversosRESUMEN
OBJECTIVE: We compare cytokine profiles at the time of initial CSF shunt placement between children who required no subsequent shunt revision surgeries and children requiring repeated CSF shunt revision surgeries for CSF shunt failure. We also describe the cytokine profiles across surgical episodes for children who undergo multiple subsequent revision surgeries. METHODS: This pilot study was nested within an ongoing prospective multicenter study collecting CSF samples and clinical data at the time of CSF shunt surgeries since August 2014. We selected cases where CSF was available for children who underwent an initial CSF shunt placement and had no subsequent shunt revision surgeries during >=24 months of follow-up (n = 7); as well as children who underwent an initial CSF shunt placement and then required repeated CSF shunt revision surgeries (n = 3). Levels of 92 human cytokines were measured using the Olink immunoassay and 41 human cytokines were measured using Luminex based bead array on CSF obtained at the time of each child's initial CSF shunt placement and were displayed in heat maps. RESULTS: Qualitatively similar profiles for the majority of cytokines were observed among the patients in each group in both Olink and Luminex assays. Lower levels of MCP-3, CASP-8, CD5, CXCL9, CXCL11, eotaxin, IFN-γ, IL-13, IP-10, and OSM at the time of initial surgery were noted in the children who went on to require multiple surgeries. Pro- and anti-inflammatory cytokines were selected a priori and shown across subsequent revision surgeries for the 3 patients. Cytokine patterns differed between patients, but within a given patient pro-inflammatory and anti-inflammatory cytokines acted in a parallel fashion, with the exception of IL-4. CONCLUSIONS: Heat maps of cytokine levels at the time of initial CSF shunt placement for each child undergoing only a single initial CSF shunt placement and for each child undergoing repeat CSF shunt revision surgeries demonstrated qualitatively similar profiles for the majority of cytokines. Lower levels of MCP-3, CASP-8, CD5, CXCL9, CXCL11, eotaxin, IFN-γ, IL-13, IP-10, and OSM at the time of initial surgery were noted in the children who went on to require multiple surgeries. Better stratification by patient age, etiology, and mechanism of failure is needed to develop a deeper understanding of the mechanism of inflammation in the development of hydrocephalus and response to shunting in children.
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Citocinas , Interleucina-13 , Humanos , Niño , Lactante , Reoperación , Estudios Prospectivos , Quimiocina CXCL10 , Proyectos Piloto , Estudios RetrospectivosRESUMEN
OBJECTIVE: To characterize the microbiota of the cerebrospinal fluid (CSF) from children with hydrocephalus at the time of initial surgical intervention. STUDY DESIGN: CSF was obtained at initial surgical intervention. One aliquot was stored in skim milk-tryptone-glucose-glycerol (STGG) medium and the second was unprocessed; both were then stored at -70°C. Bacterial growth for CSF samples stored in STGG were subsequently characterized using aerobic and anaerobic culture on blood agar and MALDI-TOF sequencing. All unprocessed CSF samples underwent 16S quantitative polymerase chain reaction (qPCR) sequencing, and a subset underwent standard clinical microbiological culture. CSF with culture growth (either after storage in STGG or standard clinical) were further analyzed using whole-genome amplification sequencing (WGAS). RESULTS: 11/66 (17%) samples stored in STGG and 1/36 (3%) that underwent standard clinical microbiological culture demonstrated bacterial growth. Of the organisms present, 8 were common skin flora and 4 were potential pathogens; only 1 was also qPCR positive. WGAS findings and STGG culture findings were concordant for only 1 sample, identifying Staphylococcus epidermidis. No significant difference in time to second surgical intervention was observed between the STGG culture-positive and negative groups. CONCLUSION(S): Using high sensitivity methods, we detected the presence of bacteria in a subset of CSF samples at the time of first surgery. Therefore, the true presence of bacteria in CSF of children with hydrocephalus cannot be ruled out, though our findings may suggest these bacteria are contaminants or false positives of the detection methods. Regardless of origin, the detection of microbiota in the CSF of these children may not have any clinical significance.
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Bacterias , Hidrocefalia , Humanos , Niño , Bacterias/genética , Medios de Cultivo , Secuenciación Completa del Genoma , Hidrocefalia/cirugía , Líquido CefalorraquídeoRESUMEN
Advances in both laboratory and computational components of high-throughput 16S amplicon sequencing (16S HTS) have markedly increased its sensitivity and specificity. Additionally, these refinements have better delineated the limits of sensitivity, and contributions of contamination to these limits, for 16S HTS that are particularly relevant for samples with low bacterial loads, such as human cerebrospinal fluid (CSF). The objectives of this work were to (i) optimize the performance of 16S HTS in CSF samples with low bacterial loads by defining and addressing potential sources of error, and (ii) perform refined 16S HTS on CSF samples from children diagnosed with bacterial meningitis and compare results with those from microbiological cultures. Several bench and computational approaches were taken to address potential sources of error for low bacterial load samples. We compared DNA yields and sequencing results after applying three different DNA extraction approaches to an artificially constructed mock-bacterial community. We also compared two postsequencing computational contaminant removal strategies, decontam R and full contaminant sequence removal. All three extraction techniques followed by decontam R yielded similar results for the mock community. We then applied these methods to 22 CSF samples from children diagnosed with meningitis, which has low bacterial loads relative to other clinical infection samples. The refined 16S HTS pipelines identified the cultured bacterial genus as the dominant organism for only 3 of these samples. We found that all three DNA extraction techniques followed by decontam R generated similar DNA yields for mock communities at the low bacterial loads representative of CSF samples. However, the limits of detection imposed by reagent contaminants and methodologic bias precluded the accurate detection of bacteria in CSF from children with culture-confirmed meningitis using these approaches, despite rigorous controls and sophisticated computational approaches. Although we did not find current DNA-based diagnostics to be useful for pediatric meningitis samples, the utility of these methods for CSF shunt infection remains undefined. Future advances in sample processing methods to minimize or eliminate contamination will be required to improve the sensitivity and specificity of these methods for pediatric meningitis. IMPORTANCE Advances in both laboratory and computational components of high-throughput 16S amplicon sequencing (16S HTS) have markedly increased its sensitivity and specificity. These refinements have better delineated the limits of sensitivity, and contributions of contamination to these limits, for 16S HTS that are particularly relevant for samples with low bacterial loads such as human cerebrospinal fluid (CSF). The objectives of this work were to (i) optimize the performance of 16S HTS in CSF samples by defining and addressing potential sources of error, and (ii) perform refined 16S HTS on CSF samples from children diagnosed with bacterial meningitis and compare results with those from microbiological cultures. We found that the limits of detection imposed by reagent contaminants and methodologic bias precluded the accurate detection of bacteria in CSF from children with culture-confirmed meningitis using these approaches, despite rigorous controls and sophisticated computational approaches.
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Meningitis Bacterianas , Microbiota , Humanos , Niño , ARN Ribosómico 16S/genética , Genes de ARNr , Reacción en Cadena de la Polimerasa/métodos , Meningitis Bacterianas/diagnóstico , Meningitis Bacterianas/microbiología , Bacterias/genética , ADN Bacteriano/genética , Líquido Cefalorraquídeo/microbiología , Secuenciación de Nucleótidos de Alto RendimientoRESUMEN
OBJECTIVES: Large (De Serres stage [IV-V]) head and neck lymphatic malformations (HNLMs) often have multiple, high-risk, invasive treatments (ITs) to address functional compromise. Logically reducing HNLM ITs should reduce treatment risk. We tested whether delaying HNLM ITs reduces total IT number. MATERIALS: Consecutive HNLM patients (n = 199) between 2010 and 2017, aged 0-18 years. METHODS: ITs (surgery or sclerotherapy) were offered for persistent or dysfunction causing HNLMs. Treatment effectiveness categorized by IT number: optimal (0-1), acceptable (2-5), or suboptimal (>5). Clinical data were summarized, and outcome associations tested (χ2 ). Relative risk (RR) with a Poisson working model tested whether HNLM observation or IT delay (>6 months post-diagnosis) predicts treatment success (i.e., ≤1 IT). RESULTS: Median age at HNLM diagnosis was 1.3 months (interquartile range [IQR] 0-45 m) with 107/199(54%) male. HNLM were stage I-III (174 [88%]), IV-V (25 [13%]). Initial treatment was observation (70 [35%]), invasive (129 [65%]). Treatment outcomes were optimal (137 [69%]), acceptable (36 [18%]), and suboptimal (26 [13%]). Suboptimal outcome associations: EXIT procedure, stage IV-V, oral location, and tracheotomy (p < 0.001). Stage I-III HNLMs were initially observed compared with stage I-III having ITs within 6 months of HNLM diagnosis, had a 82% lower relative treatment failure risk ([i.e., >1 IT], RR = 0.09, 95% CI 0.02-0.36, p < 0.001). Stage I-III HNLMs with non-delayed ITs had reduced treatment failure risk compared with IV-V (RR = 0.47, 95% CI 0.33-0.66, p < 0.001). CONCLUSION: Observation and delayed IT in stage I-III HNLM ("Grade 1") is safe and reduces IT (i.e., ≤1 IT). Stage IV-V HNLMs ("Grade 2") with early IT have a greater risk of multiple ITs. LEVEL OF EVIDENCE: 4 Laryngoscope, 133:956-962, 2023.
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Cabeza , Anomalías Linfáticas , Humanos , Masculino , Lactante , Femenino , Cuello , Anomalías Linfáticas/cirugía , Resultado del Tratamiento , Escleroterapia/métodosRESUMEN
OBJECTIVES: To describe the hearing thresholds of children under 3 years of age with cleft palate with or without cleft lip. METHODS: This retrospective study from a tertiary care children's hospital included children with cleft palate ± cleft lip born between January 01, 2008, and December 31, 2015. All patients who had the initial palate repair at our institution and at least one behavioral audiogram were included. Behavioral audiograms collected either in the sound field or under insert earphones showed results from the better hearing ear. We collected demographic information, cleft type, parent report of newborn hearing screen, syndrome diagnosis, comorbid conditions, and dates of tympanostomy tube placement and palatoplasty. The main outcome measure was hearing status as measured by behavioral audiograms, and, for a subset of patients, brainstem auditory evoked responses (BAER). Results were grouped by test type (behavioral vs BAER) and characteristics at time of testing (pre- or post-palatoplasty, with or without tympanostomy tube placement) to evaluate the range of severity of hearing loss. RESULTS: Three hundred and sixty patients were included in the cohort. The cohort had two groups: Group 1 included 37 patients who had BAER results prior to surgery and behavioral audiogram results post-surgery; Group 2 included 322 patients who had behavioral audiograms pre-surgery (n = 206) and/or post-surgery (n = 215). The median age of patients at time of BAER in Group 1 was 3.00 months [IQR: 1.00,3.75]; pre-surgery BAER results showed mild (45.9%, n = 17) or moderate hearing loss (29.7%, n = 11) by this age. Patients in Group 2 with pre-surgery behavioral audiograms had a median age of 12.00 months [IQR: 11.00, 14.00] at time of audiogram. Most patients had a mild (33.5%, n = 69) or moderate hearing loss (31.1%, n = 64). Post-surgery, patients had a median age of 13.00 months [IQR: 11.00, 15.00] at time of hearing assessment and typically had normal hearing (86.0%, n = 185). In most patients, hearing improved after palate repair with concurrent tympanostomy tube placement. CONCLUSIONS: Most patients with cleft palate ± cleft lip had evidence of mild or moderate hearing loss prior to cleft palate repair, and hearing loss was evident from early infancy. Hearing improved after concurrent tympanostomy tube placement and palatoplasty. Further research is needed to understand the clinical significance of untreated transient hearing loss during the first year of life in this patient population.
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Labio Leporino , Fisura del Paladar , Pérdida Auditiva , Niño , Preescolar , Labio Leporino/cirugía , Fisura del Paladar/complicaciones , Fisura del Paladar/diagnóstico , Fisura del Paladar/cirugía , Audición , Pérdida Auditiva/diagnóstico , Pérdida Auditiva/etiología , Pérdida Auditiva/cirugía , Humanos , Lactante , Recién Nacido , Ventilación del Oído Medio , Estudios RetrospectivosRESUMEN
Understanding the etiology of cerebrospinal fluid (CSF) shunt infections and reinfections requires detailed characterization of associated microorganisms. Traditionally, identification of bacteria present in the CSF has relied on culture methods, but recent studies have used high throughput sequencing of 16S rRNA genes. Here we evaluated the method of shotgun DNA sequencing for its potential to provide additional genomic information. CSF samples were collected from 3 patients near the beginning and end of each of 2 infection episodes. Extracted total DNA was sequenced by: (1) whole genome amplification followed by shotgun sequencing (WGA) and (2) high-throughput sequencing of the 16S rRNA V4 region (16S). Taxonomic assignments of sequences from WGA and 16S were compared with one another and with conventional microbiological cultures. While classification of bacteria was consistent among the 3 approaches, WGA provided additional insights into sample microbiological composition, such as showing relative abundances of microbial versus human DNA, identifying samples of questionable quality, and detecting significant viral load in some samples. One sample yielded sufficient non-human reads to allow assembly of a high-quality Staphylococcus epidermidis genome, denoted CLIMB1, which we characterized in terms of its MLST profile, gene complement (including putative antimicrobial resistance genes), and similarity to other annotated S. epidermidis genomes. Our results demonstrate that WGA directly applied to CSF is a valuable tool for the identification and genomic characterization of dominant microorganisms in CSF shunt infections, which can facilitate molecular approaches for the development of better diagnostic and treatment methods.
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Microbiota , Derivaciones del Líquido Cefalorraquídeo , Secuenciación de Nucleótidos de Alto Rendimiento , Humanos , Tipificación de Secuencias Multilocus , ARN Ribosómico 16S/genética , Análisis de Secuencia de ADNRESUMEN
OBJECTIVES: Head and neck lymphatic malformations (HNLM) are caused by gain-of-function somatic mutations in PIK3CA. Acetylsalicylic acid (ASA/aspirin) is thought to limit growth in PIK3CA-mutated neoplasms through PI3K pathway suppression. We sought to determine if ASA could be beneficial for HNLM. METHODS: Retrospective case series of patients (0-18 years) offered ASA (3-5 mg/kg/day) for HNLM treatment (2010-2018). Clinical and treatment characteristics, patient-reported symptom improvement, medication tolerance, compliance, and complications were recorded. Treatment response was determined by change in patient/caregiver-reported symptoms, or HNLM size [complete (resolved), partial (decreased), or stable]. RESULTS: Fifty-three patients were offered ASA, 23 (43%) accepted (median age 10 years, IQR 6-14). Compared to patients who declined, patients receiving ASA were more likely to have extensive malformations: ex-utero intrapartum treatment procedure, bilateral malformations, oral cavity location, ≥2 invasive treatments, or tracheotomy (p < 0.05). All patients with tissue available had PIK3CA mutations (13/23). Treatment indications included oral pain/blebs (12, 52%), recurrent pain/swelling (6, 26%), or sudden/persistent swelling (5, 22%). Treatment plan was commonly one 81 mg tablet daily (19, 83%) for 3-12 months (8, 42%). Therapeutic adherence was reported by 18 patients (78%). Symptoms improved in 18 patients [78%; decreased pain (9, 39%) and swelling (8, 35%)]. Treatment resulted in partial (14, 61%) or complete response (4, 17%). Three patients developed oral bleb bleeding, which resolved with medication discontinuation. CONCLUSION: ASA seems to be a well-tolerated, low-risk medication for HNLM treatment. This pilot study suggests that it often improves symptoms and reduces HNLM size. Further prospective, randomized studies are warranted to comprehensively assess indications, safety, and efficacy. LEVEL OF EVIDENCE: Level 4.
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Anomalías Linfáticas , Fosfatidilinositol 3-Quinasas , Aspirina/uso terapéutico , Niño , Humanos , Anomalías Linfáticas/tratamiento farmacológico , Anomalías Linfáticas/genética , Proyectos Piloto , Estudios RetrospectivosRESUMEN
OBJECTIVES/HYPOTHESIS: Language barriers may impact family experience, which is a key measure of healthcare quality. We compared family satisfaction between Spanish-speaking families (SSF) and English-speaking families (ESF) in pediatric otolaryngology. STUDY DESIGN: Retrospective cohort study. METHODS: Responses from the Family Experience Survey (FES), a hospital quality benchmarking survey, were analyzed from 2017 to 2019 at one academic pediatric otolaryngology practice. Question responses were compared between SSF versus ESF using mixed effect logistic regression models, adjusting for patient age, medical complexity, and insurance. RESULTS: A total of 4,964 FES survey responses were included (14% SSF). In multivariate analysis adjusting for age, medical complexity, and insurance, SSF were 1.7 times more likely than ESF to rate their provider with the highest rating (i.e. 9-10/10; 95% confidence interval [CI] 1.24-2.22). However, SSF were less likely than ESF to provide the highest rating on many individual aspects of care, including whether providers explained things intelligibly (odds ratio [OR] 0.43, 95% CI 0.25-0.74), listened carefully (OR 0.36, 95% CI 0.28-0.47), knew their medical child's history (OR 0.53, 95% CI 0.44-0.64), provided understandable information (OR 0.36, 95% CI 0.16-0.83), spent sufficient time with them (OR 0.38, 95% CI 0.31-0.48), allowed them to discuss their questions (OR 0.57, 95% CI 0.47-0.70), or had enough input in their children's' care (OR 0.46, 95% CI 0.26-0.80). CONCLUSIONS: In a large cohort of pediatric otolaryngology patients, SSF rated many individual aspects of their child's care less positively compared to ESF, despite rating their provider highly. Further research is needed to explore the reasons for these differences and how they can be improved. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:E2393-E2401, 2021.
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Atención Ambulatoria/estadística & datos numéricos , Dominio Limitado del Inglés , Otolaringología/estadística & datos numéricos , Pediatría/estadística & datos numéricos , Satisfacción Personal , Adolescente , Atención Ambulatoria/organización & administración , Niño , Preescolar , Femenino , Hispánicos o Latinos/estadística & datos numéricos , Humanos , Masculino , Otolaringología/organización & administración , Pediatría/organización & administración , Calidad de la Atención de Salud/estadística & datos numéricos , Estudios Retrospectivos , Encuestas y Cuestionarios/estadística & datos numéricosRESUMEN
BACKGROUND: Nearly 20% of patients with cerebrospinal fluid (CSF) shunt infection develop reinfection. It is unclear whether reinfections are caused by an organism previously present or are independent infection events. OBJECTIVE: We used bacterial culture and high throughput sequencing (HTS) of 16S ribosomal RNA (rRNA) genes to identify bacteria present in serial CSF samples obtained from children who failed CSF shunt infection treatment. We hypothesized that organisms that persist in CSF despite treatment would be detected upon reinfection. DESIGN/METHODS: Serial CSF samples were obtained from 6 patients, 5 with 2 infections and 1 with 3 infections; the study was limited to those for which CSF samples were available from the end of infection and beginning of reinfection. Amplicons of the 16S rRNA gene V4 region were sequenced. Taxonomic assignments of V4 sequences were compared with bacterial species identified in culture. RESULTS: Seven infection dyads averaging 13.5 samples per infection were analyzed. A median of 8 taxa [interquartile range (IQR) 5-10] were observed in the first samples from reinfection using HTS. Conventional culture correlated with high abundance of an organism by HTS in all but 1 infection. In 6 of 7 infection dyads, organisms identified by culture at reinfection were detected by HTS of culture-negative samples at the end of the previous infection. The median Chao-Jaccard abundance-based similarity index for matched infection pairs at end of infection and beginning of reinfection was 0.57 (IQR 0.07-0.87) compared to that for unmatched pairs of 0.40 (IQR 0.10-0.60) [p = 0.46]. CONCLUSION(S): HTS results were generally consistent with culture-based methods in CSF shunt infection and reinfection, and may detect organisms missed by culture at the end of infection treatment but detected by culture at reinfection. However, the CSF microbiota did not correlate more closely within patients at the end of infection and beginning of reinfection than between any two unrelated infections. We cannot reject the hypothesis that sequential infections were independent.
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Bacterias/aislamiento & purificación , Derivaciones del Líquido Cefalorraquídeo/efectos adversos , Líquido Cefalorraquídeo/microbiología , Staphylococcus aureus/aislamiento & purificación , Adolescente , Niño , Secuenciación de Nucleótidos de Alto Rendimiento , Humanos , Hidrocefalia/cirugía , ARN Ribosómico 16S , ReinfecciónRESUMEN
AIM: To evaluate the efficacy, safety and tolerability of a glucagon-like peptide-1 receptor agonist (GLP-1 RA) in patients with hypothalamic obesity (HO). MATERIALS AND METHODS: A two-arm, randomized, multicentre, double-blind, placebo-controlled trial was conducted in 10- to 25-year-olds with hypothalamic injury following intracranial tumour and HO. Participants were randomized to once-weekly subcutaneous injections of a GLP-1 RA exenatide 2 mg (ExQW) or placebo for 36 weeks. The primary efficacy endpoint was 36-week % change in body mass index (BMI). Secondary outcomes included change in body composition (by dual energy x-ray absorptiometry). RESULTS: Forty-two participants were randomized to ExQW (n = 23) or placebo (n = 19). Participants were 5 ± 2 years (mean ± SD) postdiagnosis and development of HO (BMI 37.3 ± 7.1 kg/m2 ). In intention-to-treat analysis, the effect of 36-week ExQW vs. placebo on % Δ BMI was not significant (estimated treatment difference -1.7 ± 1.8%, 95% CI -4.1 to 0.6%, P = .40); however, total body fat mass was reduced (estimated treatment difference -3.1 ± 1.4 kg, 95% CI -5.7 to -0.4 kg, P = .02). There was a significant reduction in waist circumference (estimated effect of treatment -3.5 [95% CI -5.5 to -1.6] cm, P = .004). All patients treated with placebo increased % of adipose tissue, while 50% treated with ExQW had reductions (P < .001). Mean HbA1c, glucose tolerance and serum lipids did not change significantly with therapy. ExQW was well tolerated. The most frequent adverse events were transient gastrointestinal disturbances (ExQW vs. placebo: nausea 6/23 vs. 3/18, vomiting 4/23 vs. 4/18 and diarrhoea 7/23 vs. 3/18). CONCLUSIONS: GLP-1 RAs are a promising and safe treatment to improve or stabilize HO in children and young adults.
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Diabetes Mellitus Tipo 2 , Receptor del Péptido 1 Similar al Glucagón , Adolescente , Adulto , Niño , Método Doble Ciego , Exenatida , Péptidos Similares al Glucagón , Hemoglobina Glucada , Humanos , Hipoglucemiantes , Obesidad/complicaciones , Obesidad/tratamiento farmacológico , Resultado del Tratamiento , Adulto JovenRESUMEN
OBJECTIVES: An increasing number of treatment modalities for lymphatic malformations are being described, complicating therapeutic decisions. Understanding lymphatic malformation natural history is essential. We describe management of head and neck lymphatic malformations where decisions primarily addressed lesion-induced functional compromise (ie, breathing, swallowing) to identify factors associated with invasive treatment and active observation. We hypothesize that non-function threatening malformations can be observed. STUDY DESIGN: Retrospective case series. METHODS: Retrospective case series of consecutive head and neck lymphatic malformation patients (2000-2017) with over 2 years of follow-up. Patient characteristics were summarized and associations with invasive treatment (surgery or sclerotherapy) tested using Fisher's exact. In observed patients, factors associated with spontaneous regression were assessed with Fisher's exact test. RESULTS: Of 191 patients, 101 (53%) were male, 97 (51%) Caucasian, and 98 (51.3%) younger than 3 months. Malformations were de Serres I-III 167 (87%), or IV-V 24 (12%), and commonly located in the neck (101, 53%), or oral cavity (36, 19%). Initial treatments included observation (65, 34%) or invasive treatments such as primary surgery (80, 42%), staged surgery (25, 13%), or primary sclerotherapy (9, 5%). Of 65 initially observed malformations, 8 (12%) subsequently had invasive treatment, 36 (58%) had spontaneous regression, and 21 (32%) elected for no invasive therapy. Spontaneous regression was associated with location in the lateral neck (P = .003) and macrocystic malformations (P = .017). CONCLUSION: Head and neck lymphatic malformation treatment selection can be individualized after stratifying by stage, presence of functional compromise, and consideration of natural history. Recognizing the spectrum of severity is essential in evaluating efficacy of emerging treatments, as selected malformations may respond to observation. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:1392-1397, 2021.
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Cabeza/anomalías , Anomalías Linfáticas/terapia , Cuello/anomalías , Espera Vigilante , Preescolar , Toma de Decisiones Clínicas , Bases de Datos Factuales , Femenino , Humanos , Lactante , Anomalías Linfáticas/patología , Masculino , Procedimientos Quirúrgicos Otorrinolaringológicos/estadística & datos numéricos , Estudios Prospectivos , Estudios Retrospectivos , Escleroterapia/estadística & datos numéricos , Resultado del TratamientoRESUMEN
BACKGROUND: Attention-deficit/hyperactivity disorder (ADHD) is present in 25%-50% of parents of children with ADHD, compromising parenting and child behavioral treatment. Efforts to treat multiplex ADHD families have not compared behavioral parenting interventions to parent psychopharmacology without confounds of other treatments. This report describes a pilot early intervention study directly comparing parent lisdexamfetamine dimesylate (LDX) to behavioral parent training (BPT) in families in which the mother had currently untreated ADHD and the young child displayed ADHD symptoms. METHODS: Mothers with ADHD (N = 35) of 4- to 8-year-old stimulant-naive children (N = 35) were randomly assigned to an 8-week trial of LDX (starting at 20 mg/d and titrated to a maximum of 70 mg/d) or BPT. Outcomes included multi-method, multi-informant measures of (1) maternal ADHD symptoms (Conners' Adult ADHD Rating Scales) and impairment (Clinical Global Impressions-Severity of Illness scale [CGI-S] and CGI-Improvement scale [CGI-I]), (2) parenting (Alabama Parenting Questionnaire [APQ] and Dyadic Parent-Child Interaction Coding System, Fourth Edition), and (3) child ADHD symptoms (Conners Parent Rating Scale Revised-Short Form and Conners Early Childhood Scale) and impairment (CGI-S, CGI-I, and Child Impairment Rating Scale). RESULTS: At 8 weeks, both treatments improved mothers' self-reported emotion regulation and mothers' functioning on the CGI, but only LDX improved mothers' self-reported core ADHD symptoms. LDX was associated with improvement in parents' perception of their own ADHD symptoms (Conners Inattention [P < .0001] and ADHD Index scores [P < .0001]) and their child's ADHD symptoms (P = .009). Fifty-six percent of the mothers treated with LDX (n = 10) were "much" or "very much" improved with regard to their adult ADHD based on the CGI-I scores versus 6% of mothers receiving BPT (n = 1; P = .003). BPT improved parenting on self-reported positive parenting (P = .007), inconsistent discipline (P > .0001), and corporal punishment (P = .001), while LDX improved reported inconsistent discipline (P = .001) and corporal punishment (P = .04) on the APQ, consistent with prior research. In contrast to parental LDX, which did not improve observed parenting, BPT was associated with increased positive parenting during child-directed play (P = .0002) and clean-up (P = .04) and less negative parenting (P = .04) during child-directed play. Six percent of children (n = 1) whose mothers were randomized to LDX (n = 18) were "much" or "very much" improved on the CGI-I compared to 35% (n = 16) of those treated with BPT (P = .04). CONCLUSIONS: LDX and BPT each had unique effects on maternal ADHD symptoms and parenting, but modest effects on at-risk children. In general, LDX was more effective at treating mothers' core ADHD symptoms, but both LDX and BPT improved mothers' emotion regulation, and BPT resulted in more consistent effects on parenting measures via both maternal report and direct observation. As most children remained significantly impaired after 8 weeks of unimodal treatment, combination treatment and/or longer treatment duration may be necessary to improve functioning of multiplex ADHD families. TRIAL REGISTRATION: ClinicalTrials.gov identifier: NCT01816074â.
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Trastorno por Déficit de Atención con Hiperactividad/terapia , Terapia Conductista/métodos , Estimulantes del Sistema Nervioso Central/uso terapéutico , Dimesilato de Lisdexanfetamina/uso terapéutico , Madres/psicología , Responsabilidad Parental , Adulto , Trastorno por Déficit de Atención con Hiperactividad/tratamiento farmacológico , Niño , Preescolar , Femenino , Humanos , Responsabilidad Parental/psicología , Escalas de Valoración Psiquiátrica , Resultado del TratamientoRESUMEN
OBJECTIVES: To evaluate the physical and psychosocial domains of health-related quality of life among children during the first year following community-acquired septic shock, and explore factors associated with poor physical and psychosocial health-related quality of life outcomes. DESIGN: Secondary analysis of the Life After Pediatric Sepsis Evaluation. SETTING: Twelve academic PICUs in the United States. PATIENTS: Children greater than or equal to 1 month and less than 18 years old who were perceived to be without severe developmental disability by their family caregiver at baseline and who survived hospitalization for community-acquired septic shock. INTERVENTIONS: Family caregivers completed the Pediatric Quality of Life Inventory for children 2-18 years old or the Pediatric Quality of Life Inventory Infant Scales for children less than 2 years old at baseline (reflecting preadmission status), day 7, and months 1, 3, 6, and 12 following PICU admission. Higher Pediatric Quality of Life Inventory Physical and Psychosocial Health Summary Scores indicate better health-related quality of life. MEASUREMENTS AND MAIN RESULTS: Of 204 children, 58 (28.2%) had a complex chronic comorbid condition. Children with complex chronic comorbid conditions had lower baseline physical health-related quality of life (62.7 ± 22.6 vs 84.1 ± 19.7; p < 0.001) and psychosocial health-related quality of life (68.4 ± 14.1 vs 81.2 ± 15.3; p < 0.001) than reference norms, whereas children without such conditions had baseline scores similar to reference norms. Children with complex chronic comorbid conditions recovered to their baseline health-related quality of life, whereas children without such conditions did not (physical health-related quality of life 75.3 ± 23.7 vs 83.2 ± 20.1; p = 0.008 and psychosocial health-related quality of life 74.5 ± 18.7 vs 80.5 ± 17.9; p = 0.006). Age less than 2 years was independently associated with higher month 12 physical health-related quality of life, and abnormal neurologic examination and neurologic injury suspected by a healthcare provider during the PICU course were independently associated with lower month 12 physical health-related quality of life. Treatment of increased intracranial pressure and medical device use at month 1 were independently associated with lower month 12 psychosocial health-related quality of life. CONCLUSIONS: Physical and psychosocial health-related quality of life were reduced among children during the first year following community-acquired septic shock compared with reference norms, although many recovered to baseline. Risk factors for poor health-related quality of life included neurologic complications during the hospitalization and dependence on a medical device 1 month postadmission.
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Sepsis , Choque Séptico , Adolescente , Cuidadores , Niño , Preescolar , Humanos , Lactante , Calidad de Vida , Factores de RiesgoRESUMEN
[This corrects the article DOI: 10.3389/fneur.2019.00623.].
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OBJECTIVE: To describe the Trach Safe Initiative and assess its impact on unanticipated tracheostomy-related mortality in outpatient tracheostomy-dependent children (TDC). METHODS: An interdisciplinary team including parents and providers designed the initiative with quality improvement methods. Three practice changes were prioritized: (1) surveillance airway endoscopy prior to hospital discharge from tracheostomy placement, (2) education for community-based nurses on TDC-focused emergency airway management, and (3) routine assessment of airway events for TDC in clinic. The primary outcome was annual unanticipated mortality after hospital discharge from tracheostomy placement before and after the initiative. RESULTS: In the 5 years before and after the initiative, 131 children and 155 children underwent tracheostomy placement, respectively. At the end of the study period, the institution sustained Trach Safe practices: (1) surveillance bronchoscopies increased from 104 to 429 bronchoscopies, (2) the course trained 209 community-based nurses, and (3) the survey was used in 488 home ventilator clinic visits to identify near-miss airway events. Prior to the initiative, 9 deaths were unanticipated. After Trach Safe implementation, 1 death was unanticipated. Control chart analysis demonstrates significant special-cause variation in reduced unanticipated mortality. DISCUSSION: We describe a system shift in reduced unanticipated mortality for TDC through 3 major practice changes of the Trach Safe Initiative. IMPLICATION FOR PRACTICE: Death in a child with a tracheostomy tube at home may represent modifiable tracheostomy-related airway events. Using Trach Safe practices, we address multiple facets to improve safety of TDC out of the hospital.
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Cuidados Posteriores/normas , Seguridad del Paciente/normas , Mejoramiento de la Calidad , Traqueostomía/mortalidad , Preescolar , Femenino , Humanos , Lactante , Masculino , Traqueostomía/efectos adversosRESUMEN
OBJECTIVES: A companion article reports the trajectory of long-term mortality and significant health-related quality of life disability among children encountering septic shock. In this article, the investigators examine critical illness factors associated with these adverse outcomes. DESIGN: Prospective, cohort-outcome study, conducted 2013-2017. SETTING: Twelve United States academic PICUs. PATIENTS: Critically ill children, 1 month to 18 years, with community-acquired septic shock requiring vasoactive-inotropic support. INTERVENTIONS: Illness severity, organ dysfunction, and resource utilization data were collected during PICU admission. Change from baseline health-related quality of life at the month 3 follow-up was assessed by parent proxy-report employing the Pediatric Quality of Life Inventory or the Stein-Jessop Functional Status Scale. MEASUREMENTS AND MAIN RESULTS: In univariable modeling, critical illness variables associated with death and/or persistent, serious health-related quality of life deterioration were candidates for multivariable modeling using Bayesian information criterion. The most clinically relevant multivariable models were selected among models with near-optimal statistical fit. Three months following septic shock, 346 of 389 subjects (88.9%) were alive and 43 of 389 had died (11.1%); 203 of 389 (52.2%) had completed paired health-related quality of life surveys. Pediatric Risk of Mortality, cumulative Pediatric Logistic Organ Dysfunction scores, PICU and hospital durations of stay, maximum and cumulative vasoactive-inotropic scores, duration of mechanical ventilation, need for renal replacement therapy, extracorporeal life support or cardiopulmonary resuscitation, and appearance of pathologic neurologic signs were associated with adverse outcomes in univariable models. In multivariable regression analysis (odds ratio [95% CI]), summation of daily Pediatric Logistic Organ Dysfunction scores, 1.01/per point (1.01-1.02), p < 0.001; highest vasoactive-inotropic score, 1.02/per point (1.00-1.04), p = 0.003; and any acute pathologic neurologic sign/event, 5.04 (2.15-12.01), p < 0.001 were independently associated with death or persistent, serious deterioration of health-related quality of life at month 3. CONCLUSIONS AND RELEVANCE: Biologically plausible factors related to sepsis-associated critical illness organ dysfunction and its treatment were associated with poor outcomes at month 3 follow-up among children encountering septic shock.