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There is limited understanding of attrition (premature treatment withdrawal and non-completion) from pediatric chronic pain services. This narrative review aimed to summarize attrition prevalence from face-to-face pediatric outpatient chronic pain interventions, identify associated factors and develop a theoretical model to account for attrition in this setting. A comprehensive search of the published literature revealed massive variability (0-100%) in the reported attrition rates from pediatric chronic pain interventions that varied in type and format (individual vs. group, single discipline vs. interdisciplinary, psychological only vs. multiple combined interventions, of different durations). The factors associated with attrition from pediatric chronic pain programs varied between the studies: some have assessed patient sex, psychological and other comorbidities, avoidance strategies, missed schooling, family composition/tensions, caregiver catastrophizing, scheduling, caregiver leave and clinic access. A theoretical model is presented depicting youth, caregiver and service factors that may impact attrition from pediatric chronic pain interventions. Where available, literature is drawn from the pediatric chronic pain context, but also from adult chronic pain and pediatric weight management fields. The implications for research and clinical practice are discussed, including improved reporting, patient screening and targeted supports to promote intervention completion. This review contributes to a better understanding of attrition, which is crucial for optimizing pediatric chronic pain service outcomes.
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Paediatric Palliative Care Ambulance Plans ('Plans') are used by New South Wales Ambulance (Australia) to support the care needs of children with life-limiting conditions. We aimed to describe the population of children with Plans and provide details regarding Plan completion, paramedic responses during ambulance callouts, and correspondence between Plan recommendations and paramedic responses. Plans lodged in January 2017-December 2019 were retrospectively coded for demographic information, completeness and care preferences. Associated paramedic callout notes (January 2018-December 2019) were coded for paramedic responses. Of 141 Plans retrieved, 38 (41.3% of those providing suggested medications) suggested medication use outside general paramedic scope of practice. Of 199 associated ambulance callouts, reasons for callout included symptom management, planned transfer, death notification and end-of-life care. Over two-thirds of callouts (n = 135, 67.8%) occurred after-hours. Most paramedic callouts (n = 124, 62.3%), excluding planned transfers, resulted in children being transported. Paramedic interventions corresponded with interventions suggested in Plans. However, only 24 (25.3%) of paramedic callout notes documented Plans being sighted. This study provided detailed information about children with palliative care needs for whom Plans were being used, the nature of these Plans and associated paramedic callouts. However, it is not known how paramedics were influenced by Plans.
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OBJECTIVES: This retrospective, cross-sectional study investigated the nature and extent of burden experienced by caregivers of children and adolescents with chronic pain, and factors associated with increased caregiver burden. METHODS: The Pediatric Electronic Persistent Pain Outcomes Collaboration database provided prospectively collected data from 1929 families attending 9 pediatric chronic pain services across Australia and New Zealand. Data included demographic information, responses to child pain and functioning measures, caregiver work impairment, and psychosocial functioning. RESULTS: Caregivers of children with chronic pain reported work impairment associated with their child's pain (mean: 15% ± SD 25 absenteeism; 38% ± SD 29 productivity lost), significantly worse than published international population norms (large-scale community survey data), most other caregiver samples of adults and children with other chronic conditions, and adult samples with various pain conditions. Caregivers reported considerable burden in multiple psychosocial functioning domains, particularly leisure functioning, pain-related catastrophizing, and adverse parenting behaviors (with greater pain-related avoidance). Caregiver psychosocial burden was significantly associated with child psychosocial functioning (ß = -0.308, P < 0.01), school absenteeism (ß = 0.161, P < 0.01), physical disability (ß = 0.096, P < 0.05), and pain duration (ß = 0.084, P < 0.05), but not pain intensity. Caregiver work productivity loss was significantly associated with school absenteeism (ß = 0.290, P < 0.01), child physical disability (ß = 0.148, P < 0.01), child health care utilization (ß = 0.118, P < 0.05), and worst pain intensity (ß = 0.101, P < 0.05). DISCUSSION: These results highlight the significant and varied impacts experienced by caregivers of children with chronic pain. This work is novel in reporting significant work impairment and confirms psychosocial burden in a larger sample than previous studies.
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Dolor Crónico , Adulto , Adolescente , Humanos , Niño , Estudios Retrospectivos , Carga del Cuidador , Estudios Transversales , Cuidadores/psicología , Electrónica , Calidad de VidaRESUMEN
OBJECTIVE: To investigate, via systematic review and meta-analysis, caregiver sociodemographic and biopsychosocial factors associated with anxiety, depression, posttraumatic stress symptoms, and stress of caregivers in the pediatric chronic pain context. METHODS: EMBASE, Medline, and PsycINFO databases were searched from their inception to the search date (April 4, 2022). Studies were included if they examined caregivers of youth with chronic pain, were published in a peer-reviewed journal and assessed at least one quantitative association between relevant variables. Qualitative and intervention studies were excluded. A total of 3,052 articles were screened. Risk of bias was assessed using the JBI Checklist for analytical cross-sectional studies. Meta-analyses were conducted using robust variance estimation for associations reported in at least three studies, as well as a narrative synthesis of the evidence. RESULTS: Fourteen studies assessing 1,908 caregivers were included in this review. Meta-analytic results showed a positive pooled correlation coefficient between caregiver catastrophizing about their child's pain and caregiver anxiety (r = 0.51; 95% CI: 0.35-0.65; p<.01) and depression (r = 0.45; 95% CI: 0.29-0.58; p < .01). Self-blame and helplessness were related to increased caregiver anxiety (r = 0.55; 95% CI: 0.40-0.67; p<.01), but not depression. No significant relationship was found for pain-promoting behavior and anxiety or depression. The qualitative synthesis of all other eligible studies showed associations between relevant psychological burden variables and various caregiver factors, which were mainly psychosocial. CONCLUSION: Results should be interpreted with caution due to the small number of studies. Further research is needed to get a better understanding of these relationships and to examine the causal direction of effects.
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Cuidadores , Dolor Crónico , Adolescente , Humanos , Niño , Cuidadores/psicología , Dolor Crónico/psicología , Estudios Transversales , Ansiedad , Trastornos de AnsiedadRESUMEN
A proportion of youth with chronic pain do not respond to interdisciplinary pain management and may require transition to adult pain services. This study sought to characterize a cohort of patients referred to pediatric pain services who subsequently required referral to an adult pain service. We compared this transition group with pediatric patients eligible by age to transition but who did not transition to adult services. We sought to identify factors predicting the need to transition to adult pain services. This retrospective study utilized linkage data from the adult electronic Persistent Pain Outcomes Collaboration (ePPOC) and the pediatric (PaedePPOC) data repositories. The transition group experienced significantly higher pain intensity and disability, lower quality of life, and higher health care utilization relative to the comparison group. Parents of the transition group reported greater distress, catastrophizing, and helplessness relative to parents in the comparison group. Three factors significantly predicted transition: compensation status (OR = 4.21 (1.185-15)), daily anti-inflammatory medication use (OR = 2 (1.028-3.9)), and older age at referral (OR = 1.6 (1.3-2.17)). This study demonstrated that patients referred to pediatric pain services who subsequently need transition to adult services are a uniquely disabled and vulnerable group beyond comparative peers. Clinical applications for transition-specific care are discussed.
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This twin family study first aimed to investigate the evidence for genetic factors predicting the risk of lifetime prevalence of non-specific low back pain of at least three months duration (LBP (life)) and one-month current prevalence of thoracolumbar back pain (TLBP (current)) using a study of children, adolescents, and their first-degree relatives. Secondly, the study aimed to identify associations between pain in the back with pain in other regions and also with other conditions of interest. Randomly selected families (n = 2479) with child or adolescent twin pairs and their biological parents and first siblings were approached by Twins Research Australia. There were 651 complete twin pairs aged 6-20 years (response 26%). Casewise concordance, correlation, and odds ratios were compared for monozygous (MZ) and dizygous (DZ) pairs to enable inference about the potential existence of genetic vulnerability. Multivariable random effects logistic regression was used to estimate associations between LBP (life) or TLBP (current) as an outcome with the potentially relevant condition as predictors. The MZ pairs were more similar than the DZ pairs for each of the back pain conditions (all p values < 0.02). Both back pain conditions were associated with pain in multiple sites and with primary pain and other conditions using the combined twin and sibling sample (n = 1382). Data were consistent with the existence of genetic influences on the pain measures under the equal environments assumption of the classic twin model and associations with both categories of back pain were consistent with primary pain conditions and syndromes of childhood and adolescence which has research and clinical implications.
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BACKGROUND: Paediatric chronic pain adversely impacts the child's functioning, health-related quality of life and development. However, there is a need for a holistic assessment of parental impacts of caring for a child with chronic pain. This qualitative study aimed to investigate the possible psychosocial, functional and work impacts of caring for a child with chronic pain on parents, including any positive effects of the experience. METHODS: Ten parents (eight mothers and two fathers), whose child attended the Sydney Children's Hospital Interdisciplinary Chronic and Complex Pain Clinic, participated in semi-structured interviews exploring the impact of caring for a child with chronic pain. Interviews were audio-recorded, transcribed, coded (with good inter-coder agreement) and analysed using thematic analysis. RESULTS: The parental experience of caring for a child with chronic pain was encapsulated by four overarching themes: (1) 'the constant and all-consuming nature of pain' - parents described the unpredictable, yet constant nature of chronic pain, contributing to wide-reaching impacts in various areas of their life; (2) 'dealing with uncertainty' - their experience was commonly characterized by a sense of uncertainty, stress, hopelessness and fear; (3) 'importance of support and self-care' - strong support networks and prioritizing self-care were crucial in alleviating the negative effects of paediatric chronic pain; and (4) 'a revitalized and optimistic view on life and relationships' - some parents identified unique and positive effects, such as stronger relationships, personal growth and a reformed view on life. CONCLUSIONS: This study provided rich data on the various impacts of caring for a child with chronic pain, highlighting the need for the development of holistic, family-centred interventions addressing both child and parental functioning.
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Dolor Crónico , Femenino , Niño , Humanos , Calidad de Vida/psicología , Padres/psicología , Madres/psicología , Miedo , Investigación CualitativaRESUMEN
Parents play a key role in providing children with health-related information and emotional support. This communication occurs both in their homes and in pediatric healthcare environments, such as hospitals, outpatient clinics, and primary care offices. Often, this occurs within situations entailing heightened stress for both the parent and the child. There is considerable research within the communication literature regarding the nature of both verbal and nonverbal communication, along with the way in which these communication modalities are either similar (i.e., congruent) or dissimilar (i.e., incongruent) to one another. However, less is known about communication congruency/incongruency, specifically in parent-child relationships, or within healthcare environments. In this narrative review, we explore the concept of verbal and nonverbal communication incongruence, specifically within the context of parent-child communication in a pediatric healthcare setting. We present an overview of verbal and nonverbal communication and propose the Communication Incongruence Model to encapsulate how verbal and nonverbal communication streams are used and synthesized by parents and children. We discuss the nature and possible reasons for parental communication incongruence within pediatric settings, along with the consequences of incongruent communication. Finally, we suggest a number of hypotheses derived from the model that can be tested empirically and used to guide future research directions and influence potential clinical applications.
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Background: Commonly applied diagnostic criteria for growing pains (GP) have evolved without determination by an authoritative representative body. GP and restless legs syndrome (RLS) share anatomical, distributional, temporal, and other clinical features and are associated in individuals over time, in families, and in population samples. In this study, we tested the hypothesis that GP, diagnosed by widely used criteria, is confounded by cases of painful restless legs syndrome (RLS-Painful). Methods: A twin family study of genetic influence and associations of GP using questionnaires was administered by Twins Research Australia. Twins (3-18 years; monozygous 503, dizygous 513), their oldest siblings, mothers, and fathers were randomly selected from the twin registry. Family members completed the questionnaires assessing lifetime prevalence of GP by commonly applied criteria and covariates including the history of iron deficiency and pediatric pain disorders. A GP-Specific phenotype was defined as GP without urge to move the legs. We determined similarities in twin pairs for the GP and GP-Specific phenotypes, family associations, and estimated familial and individual-specific associations for each phenotype. Results: Lifetime prevalence was one-third lower for GP-Specific than for GP among the twin and family members. Monozygous twin pairs were more similar than dizygous twin pairs for GP and for the derived GP-Specific phenotype by three methods, consistent with genetic influence. There were familial associations, but the essential evidence for genetic influence was the twin-cotwin data. GP was associated, in multivariable analyses, with migraine, headaches, recurrent abdominal pain, and iron deficiency, while GP-Specific associations were limited to migraine and headaches. Conclusions: GP is hybrid, one-third of cases having symptoms and associations of RLS, necessarily RLS-Painful. GP-Specific (without symptoms and associations of RLS) could have a genetic etiology. We propose new criteria to facilitate etiological and therapeutic research.
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AIMS: The extent to which maternal transmission of primary dysmenorrhoea is genetically determined in adolescents and young women has yet to be determined. We aimed to assess heritability and associations relevant to primary pain syndromes using a twin family study. METHODS: Participants were young menstruating female twins, and their oldest sisters and mothers, whose families were registered with Twins Research Australia and previously participated in a twin family study of primary paediatric pain disorders. Questionnaire packs were mailed, assessing current maximum and average menstrual pain intensity, current pain interference with activities and retrospective dysmenorrhea secondary symptoms. RESULTS: The sample comprised 206 twin individuals (57 monozygous (MZ) and 46 dizygous (DZ) pairs) aged 10-22 years, eldest siblings (n = 38) aged 13-28 years and mothers (n = 101) aged 32-61 years. The estimated regression coefficient of the relationship between mother-daughter and twin-sibling dyads indicated significant associations for the measures of dysmenorrhea and supported heritability. Adjusted for age, the within twin-pair correlation for MZ twins was generally more than twice that of DZ twins. Heritability estimates were maximal pain intensity 0.67 (P = 3.8 × 10-11 ), average pain intensity 0.63 (P = 3.7 × 10-10 ), pain interference 0.57 (P = 1.8 × 10-8 ) and retrospective symptoms 0.57 (P = 1.8 × 10-8 ). Twin individuals with a lifetime (three-month) history of iron deficiency and those with painless restless legs syndrome (RLS) were significantly more likely to have more intense pain associated with menstruation. CONCLUSION: Primary dysmenorrhea in adolescents and young women was shown to be relatively strongly genetically influenced and associated especially with a history of iron deficiency and painless RLS which have potential therapeutic implications.
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Dismenorrea , Gemelos Dicigóticos , Adolescente , Niño , Dismenorrea/epidemiología , Dismenorrea/genética , Femenino , Humanos , Madres , Estudios Retrospectivos , Gemelos Dicigóticos/genética , Gemelos Monocigóticos/genéticaRESUMEN
OBJECTIVE: Living with a child with a life-limiting condition (LLC), for which there is no hope of cure and premature death is expected, places much stress on a family unit. Familial communication has the potential to serve as a buffer when children are faced with stressful situations. The overall aim of the study was to learn more about illness-related communication between parents and well-siblings, giving particular consideration to the amount of illness-related communication, and sibling satisfaction with familial communication. METHODS: Participants included 48 well-siblings (aged 6-21 years) of children with LLCs and their parents. Parents and well-siblings independently completed validated measures of familial communication and sibling functioning. Parents also provided demographic information and completed a questionnaire assessing amount of illness-related information provided to well-siblings. RESULTS: Parents reported that 47.8% of well-siblings never or rarely initiated conversations about their sibling's illness. Moreover, 52.2% of well-siblings never or rarely spoke about death. Amount of illness-related communication between parents and well-siblings was most strongly predicted by parental resilience and well-sibling age. Parents engaged in significantly more illness-related communication with girls than boys (t(44)=-2.28, p = .028). Well-siblings (p < .01) and parents (p < .05) rated satisfaction with familial communication significantly higher than published norms. The only significant predictor of well-sibling satisfaction with familial communication was greater familial cohesion. Family communication variables were not significantly correlated with measures of sibling functioning (all p's>.05). CONCLUSIONS: This study provides new information regarding parent and well-sibling communication in families who have a child with a LLC.
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Padres , Hermanos , Niño , Comunicación , Familia , Femenino , Humanos , Masculino , Encuestas y CuestionariosRESUMEN
BACKGROUND: Living with a child who has a life-limiting condition (LLC) is likely to have a major impact on all family members. There is a need to have a clearer understanding of the nature and extent of this impact on parents and well-siblings. The current study aimed to investigate the psychosocial functioning of well-siblings and parents living with a child with an LLC. Further, the study aimed to assess the resilience resources of both well-siblings and parents, giving consideration to how these relate to psychosocial functioning. METHODS: Participants included 48 well-siblings (6-21 years) and 42 parents of children with LLCs. Parents and well-siblings independently completed validated measures of child and adult functioning and personal resilience. Parents provided demographic information about the patient and family. RESULTS: The emotional, social and school functioning of well-siblings in the current study was found to be significantly poorer than published norms (all p's < .01). Parental self-reported depression, anxiety and stress scores were also all significantly poorer than published norms (all p's < .01). There was negligible agreement between well-sibling self-reported functioning and parental proxy-report of the well-siblings functioning (all r's < .126, all p's > .464). Sibling self-reported resilience was positively correlated with each of the measures of psychosocial functioning (all r's > .318, p's < .05). Parental resilience was significantly negatively correlated with depressive symptoms (r = -.369, p < .05) and anxiety symptoms (r = -.473, p < .01) but not stress scores (r = -.074, p = .644). CONCLUSION: Family members living with a child who has an LLC were found to have significantly poorer psychosocial functioning than published norms. Although one cannot infer a causal direction from the current study, greater self-reported well-sibling and parental resilience were associated with aspects of better self-reported psychosocial functioning. Future studies should assess the impact of psychosocial interventions aimed at enhancing the resilience and functioning of both well-siblings and parents.
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Padres , Hermanos , Adulto , Niño , Familia , Humanos , Padres/psicología , Funcionamiento Psicosocial , Autoinforme , Hermanos/psicologíaRESUMEN
Early planning and knowing which factors to consider when planning the location of death (LOD) of a palliative child, may help minimize the burden of hasty decision-making in the future, and may provide families with a sense of control. The current paper reviewed which factors were associated with pediatric LOD and further considered some emerging factors that should are important to better facilitate integrative planning. Three overarching areas of consideration related to pediatric LOD planning were identified including health service factors, familial factors and patient factors. Multiple sub-factor considerations are presented. Further, the paper presents a conceptual model of the factors found to be related to pediatric LOD planning. The limitations that exist with rigorously and empirically studying pediatric LOD preferences are apparent from the dearth of knowledge seen in the field. However, future studies should continue to examine such factors more closely to better understand the nuanced implications.
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Cuidados Paliativos , Cuidado Terminal , Niño , Familia , HumanosRESUMEN
Any painful or medical experience that we face is viewed from the lens with which we understand and view the experience [...].
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OBJECTIVES: Although there are many benefits of short-stay hospital admissions for high volume, pediatric surgical procedures, this model of care places greater responsibility on parents for the management of children's pain. This study aimed to document the trajectory of child pain outcomes and a range of parent-reported functional outcomes following discharge from a short-stay surgical admission. Moreover, we aimed to document the trajectory of parental perceived personal coping resources. Second, we assessed whether parental dispositional factors, assessed before hospital discharge, predicted the child's pain intensity and parent-reported functional recovery. METHODS: Participants included children (aged 4 to 14 y) admitted for a short-stay tonsillectomy or appendectomy, and their parents. Parents completed a questionnaire before discharge from hospital. Demographic and surgical information was recorded from medical records. Following discharge, daily assessments of pain and functioning were carried out over a 10-day period using iPods or mobile phones. Predischarge and postdischarge data were obtained for 55 child and parent dyads. RESULTS: Pain intensity scores returned to low levels (2/10 or less) by day 5 for appendectomy and day 10 for tonsillectomy. Parents' perceived personal coping resources increased more slowly following tonsillectomy than appendectomy. Controlling for time since surgery and parental coping resources, parental pain-related catastrophizing was a significant predictor of child pain and functional recovery. DISCUSSION: Short-stay surgery results in parents facing considerable burden in managing their child's pain and functional impairment over a 10-day period. The potential value of screening for parental pain-related catastrophizing before discharge from hospital warrants further consideration and may enable identification of children likely to experience poorer recovery.
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Cuidados Posteriores , Alta del Paciente , Adaptación Psicológica , Niño , Humanos , Dolor , PadresRESUMEN
OBJECTIVE: This study assessed the feasibility of administering a multidimensional, self-report pain assessment protocol to children in an inpatient, acute pain context, and sought insight into the interrelationships between sensory, affective, and evaluative pain dimensions. METHODS: A total of 132 children (5 to 16 y) experiencing acute pain were recruited from acute pain ward rounds or the short-stay surgical unit. A multidimensional self-report assessment protocol was administered, assessing pain intensity, pain-related affect, bother, perceived unfairness, and pain expectations (for tomorrow and in 1 wk). Duration of protocol administration was assessed and ease of administration was rated. Pain-related behaviors were rated using the Face, Legs, Activity, Cry, and Consolability (FLACC) Scale. RESULTS: The duration of protocol administration was <2.5 minutes, on average, for all age groups. Median ease of protocol administration was 7/10 for 5- to 7-year-olds and 8/10 for older age groups. Pain-related bother was higher for 14- to 16-year-olds, relative to younger age groups, and significantly correlated with perceived unfairness (r=0.59, P<0.01), intensity (r=0.76, P<0.01), and affect (r=0.33, P<0.05). For younger age groups, bother was significantly positively correlated only with pain intensity (rs=0.59 to 0.79, Ps<0.01) and affect (rs=0.4 to 0.71, Ps <0.05). A stepwise multiple regression analysis found multidimensional self-reported information (especially pain intensity and perceived unfairness), accounted for significant additional amount of variance, beyond that explained by age, pain duration, and observed pain behavior. DISCUSSION: Sensory, affective, and evaluative aspects of children's clinical, acute pain experience may be assessed using self-report tools, which provide unique and valuable information about their pain experience.
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Dolor Agudo , Dolor Agudo/diagnóstico , Adolescente , Niño , Preescolar , Humanos , Pacientes Internos , Dimensión del Dolor , Autoinforme , Encuestas y CuestionariosRESUMEN
The primary pain disorders of childhood are highly prevalent but have infrequently been studied collectively. Genetic influences have been suggested to be causally implicated. Surveys were sent to 3909 Australian twin families, assessing the lifetime prevalence of growing pains, migraine, headache, recurrent abdominal pain, low back pain, and persistent pain (not otherwise specified) in pediatric twins and their immediate family members. Comparisons between monozygous (MZ) and dizygous (DZ) twin pair correlations, concordances and odds ratios were performed to assess the contribution of additive genetic influences. Random-effects logistic regression modelling was used to evaluate relationships between twin individuals and their co-twins, mothers, fathers and oldest siblings with the subject conditions. Twin analyses of responses from 1016 families revealed significant influence of additive genetic effects on the presence of growing pains, migraine, and recurrent abdominal pain. The analyses for headache, low back pain, and persistent pain overall did not conclusively demonstrate that genetic influences were implicated more than shared environmental factors. Regression analyses demonstrated varying levels of significance in relationships between family members and twin individuals for the tested conditions, with strongest support for genetic influences in growing pains and migraine. These data, together with previously published association analyses, suggest common causal influences including genes.
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Little is known about self-report pain intensity scales best suited for young children. We tested the ability of preschool children to use two simplified scales (concrete ordinal and faces). Three- to 5-year-olds (n = 123) were asked to make binary discriminations ('less' vs 'more' pain) between response options using the Simplified Faces Pain Scale and Simplified Concrete Ordinal Scale and to complete a seriation task. Eighty participants were also asked to use the Simplified Concrete Ordinal Scale, with modified verbal anchors, to rate the loudness of tones and to assess practice effects. Binary discrimination accuracy and seriation ability improved with age. When using the Simplified Concrete Ordinal Scale to rate the loudness of tones, even the 3-year-olds performed significantly better than chance, and performance was better in 4- and 5-year-olds. Little evidence supported the ability of 3-year-olds to use either of the simplified tools in the pain context. The 4-year-olds demonstrated greater accuracy in using the Simplified Concrete Ordinal Scale than the Simplified Faces Pain Scale, suggesting that the Simplified Concrete Ordinal Scale may be more appropriate for this age group.
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Dolor , Preescolar , Humanos , Dimensión del Dolor , AutoinformeRESUMEN
We aimed to evaluate the utility of clinical somatosensory testing (SST), an office adaptation of laboratory quantitative sensory testing, in a biopsychosocial assessment of a pediatric chronic somatic pain sample (N = 98, 65 females, 7-18 years). Stimulus-response tests were applied at pain regions and intra-subject control sites to cutaneous stimuli (simple and dynamic touch, punctate pressure and cool) and deep pressure stimuli (using a handheld pressure algometer, and, in a subset, manually inflated cuff). Validated psychological, pain-related and functional measures were administered. Cutaneous allodynia, usually regional, was elicited by at least one stimulus in 81% of cases, most frequently by punctate pressure. Central sensitization, using a composite measure of deep pressure pain threshold and temporal summation of pain, was implied in the majority (59.2%) and associated with worse sleep impairment and psychological functioning. In regression analyses, depressive symptoms were the only significant predictor of pain intensity. Functional interference was statistically predicted by deep pressure pain threshold and depressive symptoms. Manually inflated cuff algometry had comparable sensitivity to handheld pressure algometry for deep pressure pain threshold but not temporal summation of pain. SST complemented standard biopsychosocial assessment of pediatric chronic pain; use of SST may facilitate the understanding of disordered neurobiology.
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OBJECTIVE: This study was designed to investigate painless and painful subsets of pediatric restless legs syndrome (RLS) for genetic influence and for associations with iron deficiency and common pediatric pain disorders. METHODS: In a twin family study, twins (3-18 years) and their oldest siblings, mothers and fathers completed questionnaires, assessing lifetime prevalence of RLS using current criteria, as well as history of iron deficiency and pediatric pain disorders. Subsets were categorized as RLS-Painless or RLS-Painful. Within twin pair analyses were conducted to assess familial and potential genetic effects for the defined subsets. Penalized maximum likelihood logistic regression was used to test familial associations. Random-effects logistic regression modeling was used in the total pediatric sample to investigate univariate and multivariate associations with the subsets. RESULTS: Data were available for 2033 twin individuals (1007 monozygous (MZ), 1026 dizygous (DZ); 51.7% female), 688 siblings, 1013 mothers and 921 fathers. Odds ratios, correlations and casewise concordance were significantly higher in MZ than in DZ twins only for RLS-Painful. RLS-Painless, though familial (co-twin and mother), was not genetically influenced, but was independently associated with female sex (OR 0.52, p = 0.003), iron deficiency (OR 4.20, p < 0.001) and with persistent pain disorders (OR 2.28, p = 0.02). RLS-Painful was familial and was probably genetically influenced; was independently associated with non-migraine headaches (OR 2.70, p = 0.02) and recurrent abdominal pain (OR 2.07, p = 0.04). CONCLUSIONS: Pediatric RLS was heterogeneous and was categorized into contrasting painless and painful phenotypes. RLS-Painless was associated with iron deficiency while RLS-Painful accounted for the heritability of RLS.