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1.
Dysphagia ; 2024 Mar 19.
Artigo em Inglês | MEDLINE | ID: mdl-38503935

RESUMO

Feeding/swallowing and airway protection are complex functions, essential for survival, and continue to evolve throughout the lifetime. Medical and surgical advances across the globe have improved the long-term survival of medically complex children at the cost of increasing comorbidities, including dysfunctional swallowing (dysphagia). Dysphagia is prominent in children with histories of preterm birth, neurologic and neuromuscular diagnoses, developmental delays, and aerodigestive disorders; and is associated with medical, health, and neurodevelopmental problems; and long-term socioeconomic, caregiver, health system, and social burdens. Despite these survival and population trends, data on global prevalence of childhood dysphagia and associated burdens are limited, and practice variations are common. This article reviews current global population and resource-dependent influences on current trends for children with dysphagia, disparities in the availability and access to specialized multidisciplinary care, and potential impacts on burdens. A patient example will illustrate some questions to be considered and decision-making options in relation to age and development, availability and accessibility to resources, as well as diverse cultures and family values. Precise recognition of feeding/swallowing disorders and follow-up intervention are enhanced by awareness and knowledge of global disparities in resources. Initiatives are needed, which address geographic and economic barriers to providing optimal care to children with dysphagia.

2.
Am J Perinatol ; 40(6): 672-679, 2023 04.
Artigo em Inglês | MEDLINE | ID: mdl-34058764

RESUMO

OBJECTIVE: The study aimed to identify factors that impact timing of gastrostomy placement/removal and Nissen fundoplication (NF) in infants with bronchopulmonary dysplasia (BPD). STUDY DESIGN: Clinical data were reviewed retrospectively from patients recruited from the Johns Hopkins Bronchopulmonary Dysplasia Clinic (January 1, 2014-December 31, 2018). RESULTS: Patients with gastrostomy tubes (GTs) placed in the neonatal intensive care unit (NICU) were older at discharge (p < 0.001) and less likely to have abnormal upper gastrointestinal series findings (p = 0.005) than those with GTs placed after NICU discharge. Patients with NF had lower mean gestational ages (p = 0.011), longer NICU stays (p = 0.019), more frequent home ventilation requirements (p = 0.005), and greater likelihood of pulmonary hypertension (p = 0.032) compared with those without. Median age of GT removal was 61.6 months. Patients with GTs were weaned from supplemental oxygen and/or home ventilation before GT removal (p < 0.001). CONCLUSION: Patients with GT/NF were more medically complex than those with GT alone. Patients were more likely to be weaned from home respiratory support before GT removal. KEY POINTS: · Patients with GT/NF were more medically complex than those with GT alone.. · Patients were more likely to be weaned from home respiratory support before GT removal.. · Patients with GTs placed in NICU were older at discharge and less likely to have abnormal upper gastrointestinal series result..


Assuntos
Displasia Broncopulmonar , Gastrostomia , Recém-Nascido , Lactente , Humanos , Criança , Pré-Escolar , Fundoplicatura , Displasia Broncopulmonar/cirurgia , Estudos Retrospectivos , Alta do Paciente
3.
Dysphagia ; 37(5): 1226-1237, 2022 10.
Artigo em Inglês | MEDLINE | ID: mdl-34779911

RESUMO

The purpose of this study was to translate and adapt the Feeding/Swallowing Impact Survey (FS-IS) into Brazilian Portuguese and provide a validated instrument for caregivers of children with feeding/swallowing disorders. This cross-cultural study involved initial translation, synthesis of translations, back-translation, Committee of Experts, and pre-test. The sample consisted of 95 primary caregivers of children with feeding/swallowing disorders classified by Pediatric Dysphagia Evaluation Protocol (PDEP) in mild (n = 9), moderate-severe (n = 40), or profound (n = 46) dysphagia. Reliability and evidence of validity based on test content, response processes, internal structure and the relations to other variables were investigated. Internal consistency, test-retest, exploratory and confirmatory factor analysis were performed, in addition to the correlation with PedsQL™ Family Impact Module (PedsQLTMFIM). The pre-test participants did not report any difficulties in understanding the translated version. The Brazilian Portuguese version of FS-IS (Pt-Br-FS-IS) presented Cronbach's Alpha of 0.83, Exploratory Factor Analysis verified that the instrument would not be unifactorial (KMO = 0.74 and Bartlett's sphericity test p < 0.001) and Confirmatory Factor Analysis confirmed the original model in three subscales with χ2/df = 1.23, CFI = 0.92, TLI = 0.90, RMSEA (90% CI) 0.049 (0.011-0.073) adjustment indexes and the ICC was excellent in all subscales and total score. The correlation with PedsQL™FIM was significant in the total score and subscales. This study successfully translated and cross-culturally adapted the FS-IS instrument to the Brazilian Portuguese language and the investigation of its reliability and validity evidence suggests that the Pt-Br-FS-IS is a reliable and valid tool to measure the impact of feeding/swallowing disorders on the quality of life of caregivers of affected children.


Assuntos
Transtornos de Deglutição , Brasil , Criança , Deglutição , Transtornos de Deglutição/diagnóstico , Transtornos de Deglutição/etiologia , Humanos , Idioma , Psicometria/métodos , Qualidade de Vida , Reprodutibilidade dos Testes , Inquéritos e Questionários , Traduções
4.
Dysphagia ; 35(1): 90-98, 2020 02.
Artigo em Inglês | MEDLINE | ID: mdl-30955137

RESUMO

AIM: This investigation tested the construct validity of the first standardized assessment tool, the BaByVFSS Impairment Profile, (BaByVFSSImP©), developed for the quantification of swallowing observations made from videofluoroscopic swallow studies (VFSS) in bottle-fed babies. METHOD: Construct validity of the measures was tested using descriptive methods and confirmatory factor analysis (CFA) of swallowing scores obtained from a cohort of bottle-fed babies (median age 3 months 1 day, interquartile range 1 month 4 days-7 months 4 days) sequentially referred for VFSS based on clinical signs, symptoms, or risk factors associated with dysphagia and/or aspiration. Main outcome measures were emergence of functional domains derived from swallowing component impairment scores. RESULTS: Confirmatory factor analysis resulted in 21 significant components (factor loadings ≥ 0.5) grouping into five functional domains labeled for common contribution to overall swallowing function. The tool was organized into the BaByVFSSImP. Clinical relevance was explored using correlational analyses between domain scores, maximum penetration/aspiration scores, feeding status, and caregiver burden. INTERPRETATION: Quantification of physiologic swallowing impairment captured by BaByVFSSImP holds promise for identification of physiologically based targets for intervention, clinical decisions regarding enteral feeding, and tracking the trajectory of swallowing impairment throughout development in young children.


Assuntos
Cinerradiografia/normas , Transtornos de Deglutição/diagnóstico , Aspiração Respiratória/diagnóstico , Alimentação com Mamadeira , Cinerradiografia/métodos , Deglutição , Análise Fatorial , Comportamento Alimentar/fisiologia , Feminino , Humanos , Lactente , Masculino , Reprodutibilidade dos Testes
5.
Paediatr Respir Rev ; 32: 23-27, 2019 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-31005455

RESUMO

BACKGROUND: Aspiration can cause acute symptoms and chronic lung disease in the developing lung. However, the source of aspiration in infants is often unclear, making the choice of intervention difficult. OBJECTIVE: To quantify the source, amount and duration of lung aspiration in infants using gamma scintigraphy. METHODS: Two infants with clinical evidence of gastroesophageal reflux and oropharyngeal dysphagia swallowed formula radiolabeled with 99mtechnetium on Visit 1. Radiolabeled-formula was instilled by nasogastric tube on Visit 2. Lung aspiration was quantified over four hours and expressed as percent of total radioactivity administered. RESULTS: Aspiration was greatest with swallowing, compared to instillation, peaking between 2.0% and 2.4% within 30 min and between 0.40% and 0.65% within 20 min, respectively. Radioactivity remained above zero four hours after either administration. CONCLUSIONS: Quantification of the source, amount and duration of lung aspiration in infants is feasible using gamma scintigraphy. The impact of aspiration accrual on clinical care deserves further investigation.


Assuntos
Transtornos de Deglutição/diagnóstico por imagem , Deglutição , Refluxo Gastroesofágico/diagnóstico por imagem , Aspiração Respiratória de Conteúdos Gástricos/diagnóstico por imagem , Aspiração Respiratória/diagnóstico por imagem , Humanos , Lactente , Intubação Gastrointestinal , Masculino , Cintilografia/métodos , Compostos Radiofarmacêuticos , Coloide de Enxofre Marcado com Tecnécio Tc 99m
6.
Dysphagia ; 33(1): 76-82, 2018 02.
Artigo em Inglês | MEDLINE | ID: mdl-28894942

RESUMO

The incidence of feeding/swallowing impairments (deglutition disorders) in young children is rising and poses serious acute and long-term health consequences. Accurate detection and prompt intervention can lessen the impact of dysphagia-induced sequelae. Videofluoroscopic Swallow Studies (VFSSs) are used to make critical decisions for medically fragile children despite procedural variability and the lack of agreed upon measures for interpreting and reporting results. This investigation represents the first steps in the development of a novel tool for the quantification of oropharyngeal swallow physiology from full-length VFSS examinations in bottle-fed children. The Modified Barium Swallow Impairment Profile MBSImP™© served as the conceptual assessment model for development of components and operational score variants to characterize distinguishable VFSS observations. Twenty-four components of swallowing physiology were validated via expert consensus. Training materials included a library of 94 digitized video images comprised of distinct score variants for each component. Materials were disseminated to seven speech-language pathologists (SLPs) who participated in didactic and self-training sessions, and rated components. All SLPs achieved ≥80% reliability criterion after completing two or three training sessions. Agreement for 17 (71%) components was achieved after two sessions. Nutritive sucking/oral and airway-related components were most difficult to distinguish. Three sessions were required for 2 (33%) of the sucking/oral components and 4 (57%) of the airway-related components. These findings support the feasibility to standardize training and reliably score swallowing physiology using precise definitions and unambiguous visual images, and represent preliminary steps towards content validity and reliability of a standardized VFSS tool for bottle-fed children.


Assuntos
Alimentação com Mamadeira , Transtornos de Deglutição/diagnóstico , Deglutição/fisiologia , Criança , Transtornos de Deglutição/prevenção & controle , Fluoroscopia , Humanos , Orofaringe , Reprodutibilidade dos Testes , Gravação em Vídeo
7.
Semin Speech Lang ; 38(2): 135-146, 2017 04.
Artigo em Inglês | MEDLINE | ID: mdl-28324903

RESUMO

Speech-language pathologists (SLPs) have fulfilled primary roles in the evaluation and management of children with feeding/swallowing disorders for more than five decades. The increased incidence and prevalence of newborns, infants, and children with feeding and swallowing disorders has resulted in increased use of instrumental swallowing evaluations. The videofluoroscopic swallow study and fiberoptic endoscopic evaluation of swallowing are the two most commonly used swallowing assessments by SLPs, with ultrasound used less frequently. This article focuses on updates over the past decade in the procedures and utility of instrumental assessments of swallowing function, and identifies future directions that may enable us to meet the needs of the children who are in our care to attain functional outcomes.


Assuntos
Transtornos de Deglutição/diagnóstico , Transtornos de Alimentação na Infância/diagnóstico , Avaliação das Necessidades/tendências , Patologia da Fala e Linguagem/tendências , Criança , Pré-Escolar , Estudos Transversais , Transtornos de Deglutição/epidemiologia , Transtornos de Deglutição/terapia , Endoscopia/instrumentação , Endoscopia/tendências , Transtornos de Alimentação na Infância/epidemiologia , Transtornos de Alimentação na Infância/terapia , Fluoroscopia/instrumentação , Fluoroscopia/tendências , Previsões , Humanos , Lactente , Recém-Nascido , Patologia da Fala e Linguagem/instrumentação , Gravação em Vídeo/instrumentação , Gravação em Vídeo/tendências
8.
Dev Med Child Neurol ; 58(10): 1069-75, 2016 10.
Artigo em Inglês | MEDLINE | ID: mdl-27214374

RESUMO

AIM: This cross-sectional investigation aimed to assess the value of non-invasive measures of temporal respiratory-swallow coupling in individuals with ataxic swallowing. METHOD: Twenty participants (11 males, 9 females; range 9-21y) with ataxia telangiectasia were presented with water and pudding boluses. Their 193 swallows were compared with 2200 swallows from 82 age-matched healthy controls. The two components of airway protection during swallowing that were analyzed were: direction of peri-deglutitive airflow and duration of deglutitive inhibition of respiratory airflow (DIORA). RESULTS: Safe expiratory patterns of peri-deglutitive airflow occurred significantly less often in participants with ataxia telangiectasia than in age-matched control participants (younger p<0.015 and older p<0.001). The frequency of an expiratory pattern of peri-deglutitive airflow increased with age in participants in the comparison group (p=0.006), but not in those with ataxia telangiectasia (p=0.234). With age, mean duration of DIORA decreased in controls (p<0.001) but was unchanged in participants with ataxia telangiectasia (p=0.164). INTERPRETATION: Non-invasive quantitative measures of respiratory-swallow coupling capture temporal relationships that plausibly contribute to airway compromise from dysphagia. Changes in respiratory-swallow coupling observed with advancing age in control participants were not seen in participants with ataxia telangiectasia. Measures of perturbations may herald swallowing problems prior to development of pulmonary and nutritional sequelae.


Assuntos
Ataxia Telangiectasia/complicações , Transtornos de Deglutição/diagnóstico , Ventilação Pulmonar/fisiologia , Respiração , Adolescente , Adulto , Criança , Estudos Transversais , Transtornos de Deglutição/etiologia , Transtornos de Deglutição/fisiopatologia , Feminino , Humanos , Masculino , Adulto Jovem
9.
Dysphagia ; 31(2): 154-9, 2016 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-26590570

RESUMO

Use of high-flow oxygen via nasal cannula (HFO2-NC) is increasingly common in intensive care unit (ICU) settings. Despite the critical interface between respiration and swallowing, and the high acuity of patients in ICUs, the impact of HFO2-NC on feeding and swallowing is unknown. The present prospective, single-center, cohort study investigated the impact of HFO2-NC use on oral alimentation in neonatal and adult ICU patients. Oral alimentation status was evaluated in 100 consecutive ICU inpatients (50 neonatal and 50 adult) requiring HFO2-NC. Participant characteristics, respiratory support, successful initiation of oral feeding in neonates, and successful resumption of oral feeding in adults were recorded. Seventeen of 50 (34 %) neonates requiring HFO2-NC were deemed developmentally and medically appropriate by the neonatologist and nursing to begin oral alimentation. All 17 (100 %) were successful with initiation of oral feedings. Thirty-three of 50 (66 %) continued nil per os due to prematurity or medical conditions precluding oral alimentation at time of data collection. Thirty-nine of 50 (78 %) adults requiring HFO2-NC were deemed medically appropriate by the intensivist and nursing to resume oral alimentation (n = 34) or with a functional swallow without aspiration on FEES (n = 5). All 39 (100 %) resumed oral alimentation successfully. Eleven of 50 (22 %) continued nil per os due to severe respiratory issues precluding both swallow testing and oral alimentation at time of data collection. All developmentally and medically appropriate neonatal and adult patients requiring HFO2-NC were successful with either the introduction or resumption of oral alimentation. Patients requiring HFO2-NC who are identified as having feeding or swallowing issues should be referred for swallowing evaluations using the same criteria as patients who do not require HFO2-NC, as it is not the use of HFO2-NC but rather patient-specific determinants of feeding and swallowing readiness and their underlying medical conditions that impact readiness for oral alimentation status.


Assuntos
Cânula/efeitos adversos , Transtornos de Deglutição/etiologia , Deglutição/fisiologia , Nutrição Enteral/métodos , Oxigenoterapia/efeitos adversos , Adulto , Idoso , Idoso de 80 Anos ou mais , Transtornos de Deglutição/fisiopatologia , Feminino , Humanos , Lactente , Recém-Nascido , Unidades de Terapia Intensiva , Masculino , Pessoa de Meia-Idade , Oxigenoterapia/instrumentação , Estudos Prospectivos
10.
Dysphagia ; 31(4): 505-10, 2016 08.
Artigo em Inglês | MEDLINE | ID: mdl-27048206

RESUMO

The increasing incidence of pediatric dysphagia has raised questions about how to identify children at risk for aspiration. Multiple investigative imaging modalities are considered in diagnostic algorithms, since dysphagia may involve any or all phases of swallowing. Although upper gastrointestinal (UGI) series and videofluoroscopic swallow study (VFSS) are common procedures, the utility of UGI for detection of aspiration and the impact of oropharyngeal imaging during UGI on radiation exposure have not been well described. We hypothesized that diagnosis of aspiration on UGI would be predictive of aspiration on VFSS and screening swallows during UGI would increase radiation exposure. A retrospective review was completed of bottle-fed children undergoing UGI series within 1 month before/after standardized VFSS. UGI was imaged at 3 frames per second (fps) pulsed and VFSS at 30 fps continuous fluoroscopy. Cumulative radiation dose (CD) and dose area product (DAP) were recorded. VFSS and UGI were performed in 49 patients; however, only 21 (43 %) had documentation of swallow function on an UGI series. All children with aspiration on UGI demonstrated thin liquid aspiration on VFSS; however, 53 % without aspiration on UGI aspirated on VFSS. CD for UGI with versus without swallowing documentation was significantly higher (median = 0.33 vs. 0.21 mGy, p = 0.02) but within variability ranges reported for fluoroscopy equipment. DAP was not significantly higher for UGI with documentation of swallowing compared to without documentation of swallowing (median = 4.11 vs. 3.02 µGy cm(2), p = 0.09). UGI findings are specific but not sensitive markers for aspiration on VFSS. Imaging of swallowing on UGI may have an appreciable increase on radiation exposure.


Assuntos
Alimentação com Mamadeira , Transtornos de Deglutição/diagnóstico por imagem , Fluoroscopia/métodos , Orofaringe/diagnóstico por imagem , Radiografia/métodos , Aspiração Respiratória/diagnóstico por imagem , Pré-Escolar , Cinerradiografia/métodos , Deglutição/fisiologia , Feminino , Humanos , Lactente , Recém-Nascido , Masculino , Orofaringe/fisiopatologia , Exposição à Radiação/análise , Reprodutibilidade dos Testes , Estudos Retrospectivos , Sensibilidade e Especificidade
11.
Semin Speech Lang ; 37(4): 298-309, 2016 11.
Artigo em Inglês | MEDLINE | ID: mdl-27701706

RESUMO

Speech-language pathologists (SLPs) have played primary roles in the evaluation and management of children with feeding/swallowing disorders for more than five decades. Medical, surgical, and technological advances have improved the survival of young fragile infants and children, many of whom will present with feeding/swallowing problems. Regardless of their underlying etiologies, many of these children are at risk for aspiration-induced lung disease, undernutrition or malnutrition, developmental deficits, and stressful interactions with their caregivers. Unfortunately, our understanding of the physiology/pathophysiology of swallowing and its maturation, the development of standardized and efficacious evaluation and therapy tools, and identification of functional outcomes have not kept pace with our ability to identify children who are at increased risk for dysphagia and the associated sequelae. Given this paucity of evidence to guide practice in pediatrics, clinicians rely upon a combination of data extrapolated from adults with dysphagia, anecdotal reports, and institution-specific guidelines. This article focuses on updates in population demographics and advances in evaluation and treatment over the past decade and identifies future directions that may enable us to meet the needs of the children who are in our care to attain functional outcomes.


Assuntos
Transtornos de Deglutição , Deglutição , Criança , Humanos
12.
Dysphagia ; 29(6): 671-7, 2014 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-25159316

RESUMO

The impact of caring for children with deglutition disorders is poorly understood and tools to measure the unique concerns of these caregivers are lacking. The aims of this investigation were to develop and validate The Feeding/Swallowing Impact Survey (FS-IS) as an instrument designed to measure and improve understanding of caregiver issues. Demographic, economic, and dysphagic data were provided by the primary caregivers of 164 children (median age: 14 months, male: 78, female: 86) presenting for initial outpatient feeding/swallowing evaluations. Caregivers completed the PEDS-QL™ Family Impact Module (PEDS-QL™ FIM) and the FS-IS. A principal component analysis was conducted on the FS-IS to identify appropriate subscales. Concurrent validity was assessed by examining correlations between the FS-IS and PEDS-QL™ FIM. Caring for children with feeding/swallowing problems adversely impacted the Health-Related Quality of Life (HRQoL) of their caregivers. The FS-IS had a strong 3-factor solution to indicate 3 subscales: Daily Activities, Worry, and Feeding Difficulties. All three subscales and total score of the FS-IS correlated with PEDS-QL™ FIM. The FS-IS was validated as an instrument that may help clinicians detect specific factors that influence caregiver HRQoL, identify caregivers who might benefit from additional support, and ultimately improve the care of their children with feeding/swallowing disorders.


Assuntos
Cuidadores , Transtornos de Deglutição/terapia , Cuidadores/psicologia , Cuidadores/estatística & dados numéricos , Efeitos Psicossociais da Doença , Estudos Transversais , Transtornos de Deglutição/diagnóstico , Feminino , Humanos , Lactente , Masculino , Qualidade de Vida/psicologia , Reprodutibilidade dos Testes , Inquéritos e Questionários
13.
Perspect ASHA Spec Interest Groups ; 7(2): 543-552, 2022 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-36276931

RESUMO

Purpose: High-Flow Nasal Cannula (HFNC) has become an increasingly common means of noninvasive respiratory support in pediatrics and is being used in infants and children with respiratory distress both inside and outside of the intensive care units. Despite the widespread use of HFNC, there remains a paucity of data on optimal flow rates and its impact on morbidity, mortality, and desired outcomes. Given the scarcity of information in these critical areas, it is not surprising that guidelines for initiation of oral feeding do not exist. This review article will review HFNC mechanisms of action, its use in specific populations and settings, and finally what is known about initiation of feeding during this therapy. Conclusions: The practice of withholding oral feeding solely, because of HFNC, is not supported in the literature at the time of this writing, but in the absence of safety data from clinical trials, clinicians should proceed with caution and consider patient-specific factors while making decisions about oral feeding. Well-controlled prospective clinical trials are needed for development of best practice clinical guidelines and attainment of optimal outcomes.

15.
J Multidiscip Healthc ; 14: 1637-1644, 2021.
Artigo em Inglês | MEDLINE | ID: mdl-34234451

RESUMO

Ataxia telangiectasia (A-T) is a rare autosomal recessive disease caused by mutations in the ataxia telangiectasia mutated (ATM) gene. In the absence of a family history, the diagnosis of A-T is usually not made until the child is older and symptomatic. Classic A-T is characterized by a constellation of clinical symptoms including progressive ataxia, oculocutaneous telangiectasias and sinopulmonary disease and is usually associated with absence of ATM protein. Other laboratory features associated with A-T include elevated serum levels of alpha-fetoprotein (AFP) and increased chromosomal breakage with in vitro exposure to ionizing radiation. Sinopulmonary symptoms can occur to varying degrees across the lifespan. Some children will also have hypogammaglobulinemia and impaired antibody responses requiring supplemental gamma globulin. People with hypomorphic ATM mutations are often considered to have mild A-T with onset of ataxia and neurological progression occurring later in life with less impairment of the immune system. The risk of malignancy, however, is significantly increased in people with either classic or mild A-T. While hematological malignancies are most common in the first two decades of life, solid organ malignancies become increasingly common during young adulthood. Deterioration of neurologic function with age is associated with dysphagia with aspiration, growth faltering, loss of ambulation and decline in pulmonary function, morbidities that contribute to shortened life expectancy and decreased quality of life. Premature death is often due to malignancies or chronic respiratory insufficiency. A-T is currently managed with supportive care and symptomatic treatment. Current clinical trials, however, represent progress and hope towards disease-modifying therapies for A-T.

16.
Orphanet J Rare Dis ; 16(1): 123, 2021 03 10.
Artigo em Inglês | MEDLINE | ID: mdl-33691726

RESUMO

BACKGROUND: Ataxia telangiectasia (A-T) is a DNA repair disorder that affects multiple body systems. Neurological problems and immunodeficiency are two important features of this disease. At this time, two main severity groups are defined in A-T: classic (the more severe form) and mild. Poor growth is a common problem in classic A-T. An objective of this study was to develop growth references for classic A-T. Another objective was to compare growth patterns in classic A-T and mild A-T with each other and with the general population, using the CDC growth references. A final objective was to examine the effects of chronic infection on height. RESULTS: We found that classic A-T patients were smaller overall, and suffered from height and weight faltering that continued throughout childhood and adolescence. When compared to the CDC growth references, the median heights and weights for both male and female patients eventually fell to or below the 3rd centile on the CDC charts. Height faltering was more pronounced in females. Birthweight was lower in the classic A-T group compared to mild A-T and the general population, whereas birth length was not. Finally, we investigated height and BMI faltering in relation to number of infections and found no association. CONCLUSIONS: Classic A-T appears to affect growth in utero. Although children appear to grow well in very early life, faltering begins early, and is unrelenting.


Assuntos
Ataxia Telangiectasia , Adolescente , Estatura , Peso Corporal , Criança , Feminino , Transtornos do Crescimento , Humanos , Masculino
17.
Laryngoscope ; 131(6): 1223-1228, 2021 06.
Artigo em Inglês | MEDLINE | ID: mdl-32770795

RESUMO

OBJECTIVE: Liquid thickeners are commonly recommended in individuals with dysphagia and recurrent aspiration as a strategy for pneumonia prevention. The goal of this study was to examine the effects of small amounts of aspirated liquid thickener on the lungs. STUDY DESIGN: Animal model. Prospective small animal clinical trial. METHODS: Adult Sprague Dawley rats (n = 19) were divided into two groups and underwent three intratracheal instillations of either xanthan gum-based nectar-thick water (0.1-0.25 mL/kg) or water-only control over the course of 8 days. Blood was collected from a peripheral vein on days 1 and 8 and submitted for complete blood count (CBC) analysis. Rats were euthanized 10 days after the last instillation, and the lungs were harvested. Histopathology was conducted on lung specimens by a blinded licensed veterinary pathologist and scored for evidence of lung injury and pneumonia. RESULTS: Fifteen animals (8 nectar-thickener group, 7 control group) survived until the endpoint of the study (day 18). Serum CBC did not show abnormalities at any timepoint in either group. Histological evidence of lung inflammation and edema were significantly greater in the nectar-thick group compared to controls (P < .05). Signs of inflammation included aggregates of foamy macrophages, expansion of bronchiolar lymphoid tissue, and large numbers of eosinophilic intraalveolar crystals. Histiocytic and neutrophilic pneumonia was noted in one animal that received thickened liquids. CONCLUSION: Recurrent aspiration of small amounts of thickened water resulted in significant pulmonary inflammation in a murine model of aspiration. Results of this study support the need for further investigation of liquid thickener safety and its efficacy in reducing the pulmonary complications of swallowing disorders. LEVEL OF EVIDENCE: NA Laryngoscope, 131:1223-1228, 2021.


Assuntos
Transtornos de Deglutição/terapia , Lesão Pulmonar/induzido quimicamente , Pneumonia Aspirativa/prevenção & controle , Polissacarídeos Bacterianos/farmacologia , Água/farmacologia , Animais , Deglutição/efeitos dos fármacos , Transtornos de Deglutição/complicações , Modelos Animais de Doenças , Inflamação , Pulmão/efeitos dos fármacos , Pneumonia Aspirativa/etiologia , Estudos Prospectivos , Ratos , Ratos Sprague-Dawley , Recidiva , Viscosidade
18.
J Pediatr ; 156(4): 682-4.e1, 2010 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-20171651

RESUMO

Serum interleukin (IL)-8 levels were measured in 50 patients with ataxia telangiectasia (A-T) and 22 without A-T. In a cross-sectional study, the geometric mean of IL-8 level was significantly higher in the patients with A-T (P <.0001). Elevated serum IL-8 levels in patients with A-T suggest that systemic inflammation may contribute to the disease phenotype.


Assuntos
Ataxia Telangiectasia/sangue , Interleucina-8/sangue , Adolescente , Adulto , Ataxia Telangiectasia/imunologia , Biomarcadores/sangue , Proteína C-Reativa/metabolismo , Relação CD4-CD8 , Criança , Pré-Escolar , Estudos Transversais , Progressão da Doença , Feminino , Seguimentos , Humanos , Imunoensaio , Masculino , Pessoa de Meia-Idade , Fenótipo , Prognóstico , Estudos Retrospectivos , Adulto Jovem , alfa-Fetoproteínas/metabolismo
20.
Am J Speech Lang Pathol ; 29(2S): 956-966, 2020 07 10.
Artigo em Inglês | MEDLINE | ID: mdl-32650658

RESUMO

Purpose Pediatric feeding disorders (PFDs) present as a complex clinical challenge because of the heterogeneous underlying etiologies and their impact on health, safety, growth, and psychosocial development. A multidisciplinary team approach is essential for accurate diagnosis and prompt interventions to lessen the burdens associated with PFDs. The role of the speech-language pathologist (SLP) as a member of the multidisciplinary team will be highlighted. Method This clinical focus article reviews the definition of PFDs and pertinent literature on factors that contribute to the development of PFDs, the accurate diagnosis, and current interventions for infants and children. As part of the multidisciplinary team, the SLP has an integral role in determining whether a child cannot or will not eat and working with the team to identify and carryout appropriate interventions. Collaboration between SLPs and psychologists/behavioral specialists in conjunction with the parents/caregivers as part of the multidisciplinary team is essential to the advancement of therapeutic goals. Conclusions Due to their complex nature, the successful management of PFDs is only possible with the care and expertise of a multidisciplinary team, which includes parents/caregivers. SLPs are important members of these multidisciplinary teams and provide valuable input for the accurate identification and effective remediation of PFDs.


Assuntos
Transtornos da Comunicação , Transtornos da Alimentação e da Ingestão de Alimentos , Patologia da Fala e Linguagem , Criança , Transtornos da Alimentação e da Ingestão de Alimentos/diagnóstico , Transtornos da Alimentação e da Ingestão de Alimentos/terapia , Humanos , Lactente , Patologistas , Fala
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