BACKGROUND: Severe motor and intellectual disabilities (SMID) involve intense physical and intellectual disabilities resulting in extreme dependence. Studies investigating the association between the experience of intera...BACKGROUND: Severe motor and intellectual disabilities (SMID) involve intense physical and intellectual disabilities resulting in extreme dependence. Studies investigating the association between the experience of interacting with individuals with SMID and nurses' positive mental well-being are lacking. The comprehensive analysis of factors associated with the positive mental well-being of nurses who interact with individuals with SMID may enhance the nurses' quality of life and contribute to improving the quality of care provided to individuals with SMID. METHODS: We conducted a two-month cross-sectional online survey among 263 nurses at a university hospital in 2023. Happiness and resilience were measured using the Subjective Happiness Scale (SHS) and the short version of the Resilience Scale (RS-14), respectively. Multiple regression analysis was conducted to investigate the relationship between the interaction experience with individuals with SMID and the SHS and RS-14 scores, alongside other factors possibly influencing positive mental well-being. RESULTS: Regarding subjective happiness, no factor related to individuals with SMID was significantly associated with the SHS scores. However, regarding resilience, the number of years spent engaging with them was positively associated with the RS-14 scores [regression coefficient (B), 2.401; 95 % confidence interval (CI), 0.073 to 4.728; p = 0.043]. CONCLUSION: Interaction experiences with individuals with SMID were not associated with subjective happiness; however, they were associated with resilience. Our findings suggest the necessity for nurses to establish sustainable relationships with individuals with SMID.
INTRODUCTION: The human cerebellum has large hemispheres (HS) and develops slowly over the pre- and postnatal periods. Several morphological studies have reported regional differences during the fetal period. This study...INTRODUCTION: The human cerebellum has large hemispheres (HS) and develops slowly over the pre- and postnatal periods. Several morphological studies have reported regional differences during the fetal period. This study aimed to investigate these differences by evaluating serial brain sections. MATERIALS AND METHODS: Ten cerebellar samples aged 21-43 postmenstrual weeks (PW) were analyzed. Horizontal serial sections were cut and stained using the Klüver-Barrera method. After microscopic observations, a volumetric analysis was performed for the cerebellar cortex and medulla among the HS, vermis (VM), and flocculus (FL). RESULTS: The lamina dissecans (LD) was more prominent and lasted longer in the HS than in the VM and FL. The maturation of Purkinje cells and the accumulation of granule cells in the internal granular layer started earlier in the latter than in the former. Cortical differentiation was most delayed in the inferior semilunar and tonsillar lobules. Volumetric analysis showed that volume increased more in the HS than in the VM and FL after midgestation, and that it increased more intensively in the cortex than in the medulla in each region. CONCLUSIONS: The human cerebellar cortex may develop through three stages in fetal period: (1) three-layers stage (before 20 PW); (2) five-layers stage (20-30 PW); (3) four-layers stage (after 30 PW). Particularly, the second stage is characterized by a transient appearance of the LD, which is most evident in the HS. Regional differences should be carefully considered when interpreting morphogenetic data.
OBJECTIVE: Persistent developmental stuttering (PDS), also known as childhood-onset speech fluency disorder, is characterized by involuntary disruptions in normal speech fluency, such as sound repetitions, prolongations,...OBJECTIVE: Persistent developmental stuttering (PDS), also known as childhood-onset speech fluency disorder, is characterized by involuntary disruptions in normal speech fluency, such as sound repetitions, prolongations, and silent pauses. Although structural neuroimaging techniques, particularly voxel-based morphometry (VBM), have been widely used to explore brain abnormalities in PDS, the precise brain regions predominantly affected remain unclear. This study aimed to investigate grey matter (GM) and white matter (WM) volumetric differences in adults with PDS compared to fluent speakers. METHODS: Magnetic resonance imaging (MRI) scans were obtained from 15 adults with PDS and 15 fluent control participants matched for age, sex, education, and hand preference. Whole-brain VBM analysis was conducted to assess GM and WM volume differences between the two groups. RESULTS: Compared to fluent speakers, adults with PDS demonstrated reduced WM volume in the cerebellum. GM alterations included increased volumes in the right postcentral gyrus and the left middle temporal gyrus (MTG), while decreased volumes were observed in the left superior frontal gyrus (SFG), bilateral paracentral lobule, right cuneus, and right cerebellum. CONCLUSION: These findings provide evidence of both grey and white matter abnormalities in adults with PDS, highlighting the involvement of specific cortical and subcortical regions. The results support the significance of structural brain differences in understanding the neurobiological basis of persistent stuttering.
Duchenne muscular dystrophy (DMD) is an inherited progressive muscle disease that is caused by variants in the DMD gene. The development of therapies for DMD that promote dystrophin protein production or ameliorate dystr...Duchenne muscular dystrophy (DMD) is an inherited progressive muscle disease that is caused by variants in the DMD gene. The development of therapies for DMD that promote dystrophin protein production or ameliorate dystrophin deficiency-induced pathology is currently underway. Therapies that promote dystrophin production are known as disease-modifying therapies, and include exon-skipping therapy using antisense oligonucleotides (AS-oligo). This therapy suppresses the function of a splicing enhancer sequence within an exon using AS-oligo and removes the exon from the mRNA, thereby converting an out-of-frame deletion (as occurs in DMD) to an in-frame deletion and inducing the expression of functional dystrophin protein. In 2016, eteplirsen, which induces exon 51 skipping, received accelerated approval in the United States. AS-oligo that induce the skipping of exons 45 and 53 are also currently being applied in clinical practice. AS-oligo that induce skipping of other exons are expected to be developed in the future, as well as modified nucleic acids that act more potently. Notably, however, the evaluation of the efficacy of these therapies in clinical practice after accelerated approval remains insufficient. In addition, many issues, such as the effectiveness of early treatment and the combination of these therapies with other novel therapeutic agents, need to be considered. It is therefore important to establish a system to follow-up the long-term efficacy and safety of treatment in the future. The establishment of an early diagnostic system may also need to be considered. The present review outlines the development and future challenges of exon-skipping therapy for DMD and the expansion of splice-switching therapy (a therapy that uses AS-oligo to control splicing), including exon-skipping therapy, to other diseases.
BACKGROUND: Ventilated very-low-birth-weight infants (VLBWIs) frequently receive hydrocortisone (HDC). The HDC dose increases in more serious cases, but the association between the actual HDC dose and neurodevelopmental...BACKGROUND: Ventilated very-low-birth-weight infants (VLBWIs) frequently receive hydrocortisone (HDC). The HDC dose increases in more serious cases, but the association between the actual HDC dose and neurodevelopmental outcomes is unclear. METHOD: A total of 274 VLBWIs were divided into four groups according to the total HDC dose: zero (0 mg/kg), low (1-25 mg/kg), moderate (25-75 mg/kg), high (>75 mg/kg). The developmental quotient (DQ) in each group were compared. Using simple and multiple linear regression analyses, we evaluated the presence of a significant relationship between total DQ and total HDC dose in 274 and 153 VLBWIs receiving HDC therapy. In addition, the association between HDC therapy and the total DQ was investigated in 274 VLBWIs. RESULTS: The total DQ in the zero group was significantly higher than that in the moderate and high groups but equivalent to that in the low group. Despite a significant association between total DQ and total HDC dose in the single linear regression analysis, no significant association was found between total DQ and total HDC dose in 274 VLBWIs (p = 0.07) and 153 VLBWIs receiving HDC (p = 0.18) using a multiple linear regression analysis, after adjusting for the days of artificial ventilation, gestational age, and the onset of late-stage circulatory collapse. No significant association was observed between total DQ and HDC therapy (p = 0.58). CONCLUSION: HDC therapy during NICU admission may not have a significant effect on total DQ at three years old in VLBWIs.
BACKGROUND: Febrile seizures are a common cause of emergency pediatric transport. Although the coronavirus disease (COVID-19) pandemic disrupted healthcare systems, including emergency services, its impact on transport t...BACKGROUND: Febrile seizures are a common cause of emergency pediatric transport. Although the coronavirus disease (COVID-19) pandemic disrupted healthcare systems, including emergency services, its impact on transport times for pediatric febrile seizures remains unclear. This study aimed to evaluate how the pandemic affected transport times. METHODS: We retrospectively reviewed emergency department visits at our hospital between 2018 and 2022. Patients with febrile seizures in 2019 (before the COVID-19 pandemic) and in 2022 (during the pandemic) were included. Transport times and clinical data were compared between those years. Multivariate regression analysis was used to identify factors associated with longer transport times. RESULTS: There were 329 and 282 patients in 2019 and 2022, respectively. The median transport time increased significantly from 33 min in 2019 to 39 min in 2022 (p < 0.001). The proportion of cases exceeding 46 min increased from 10 % to 28 %, whereas those exceeding 60 min increased from 1.2 % to 7.4 %. A longer transport time was associated with seizure duration, onset year (2022), and transport from distant areas. Onset in 2022 was identified as an independent factor. Stratified analysis showed significantly longer transport times in adjacent and remote areas during the pandemic no significant difference in transport times near the hospital. CONCLUSION: Transport times for pediatric febrile seizures increased significantly during the COVID-19 pandemic, with more delays of over 60 min, especially in remote areas. These findings highlight the need to strengthen regional emergency transportation systems to reduce time to care, particularly for patients living farther from hospitals.
PURPOSE: Epileptic spasms are the predominant seizure type in infantile epileptic spasms syndrome (IESS). The pathophysiology of IESS, including blood-brain barrier (BBB) function involvement, remains unclear. To address...PURPOSE: Epileptic spasms are the predominant seizure type in infantile epileptic spasms syndrome (IESS). The pathophysiology of IESS, including blood-brain barrier (BBB) function involvement, remains unclear. To address this issue, we evaluated the serum matrix metallopeptidase-9 (MMP-9) and tissue inhibitor of metalloproteinase-1 (TIMP-1) levels in patients with IESS before and after initiating vigabatrin therapy. METHODS: IESS was defined as epileptic spasms occurring within 2 years after birth. We prospectively assessed serum MMP-9 and TIMP-1 levels before and after initiating vigabatrin therapy in patients with IESS who attended Saitama Children's Medical Center between February 2019 and December 2024 (n = 12; 5 boys) and compared them with those in age-matched controls (n = 14; 8 boys). RESULTS: The median ages at epileptic spasm onset and vigabatrin therapy initiation were 3.5 (1-11) and 8 (3-13) months, respectively. Serum MMP-9 levels were higher in patients with IESS than in the controls (p < 0.001). Serum MMP-9 and MMP-9/TIMP-1 ratios decreased significantly after vigabatrin therapy (MMP-9: 308 [160-664] ng/mL vs. 220 [112-367] ng/mL, p < 0.01; MMP-9/TIMP-1 ratio: 1.48 [0.61-8.14] vs. 1.11 [0.31-1.92], p < 0.05). MMP-9 levels decreased in 9 of 11 patients whose epileptic spasms had disappeared by the time of the last measurement. CONCLUSION: Decreased MMP-9 levels after the initiation of vigabatrin therapy suggested an improvement in BBB dysfunction. Our findings shed light on the role of the BBB in IESS and the role of vigabatrin in the recovery of this function.
BACKGROUND: Both risperidone and aripiprazole are effective in reducing irritability severity in children with autism spectrum disorder (ASD). However, head-to-head comparison trials between these two drugs are scarce in...BACKGROUND: Both risperidone and aripiprazole are effective in reducing irritability severity in children with autism spectrum disorder (ASD). However, head-to-head comparison trials between these two drugs are scarce in the literature and have shown conflicting results. METHODS: This trial compared the efficacy and safety of risperidone and aripiprazole in children and adolescents with ASD, aged 6-18 years. After a two-week placebo trial, placebo responders were excluded. The remaining participants were randomized into two groups. The outcomes were the change in the irritability subscale of the Aberrant Behavior Checklist (ABC-I), Childhood Autism Rating Scale (CARS2), Conners' Parent Rating Scale-Revised (CPRS-R), Children's Sleep Habits Questionnaire (CSHQ), Sensory Profile-2 (SP-2), cognition and the nature and frequency of treatment-emergent adverse events. RESULTS: Seventy-two patients (36 in each group) were recruited. Changes in the ABC-I score (-13.6 ± 4.3 vs. -12.2 ± 3.9, p = 0.15), ABC total score (-27.5 ± 15.9 vs. -26.8 ± 15.7, p = 0.85), CARS score (-2.9 ± 0.7 vs. -2.7 ± 0.8, p = 0.26), CPRS-R Global Index T-score (-10.63 ± 8.54 vs. -9.61 ± 8.92, p = 0.62), number of patients with significant sensory processing abnormalities (18/36 vs. 18/36, p = 1.0), CSHQ score (-4.6 ± 3.8 vs. -3.9 ± 3.1, p = 0.39), and full-scale IQ (1.9 ± 1.6 vs. 1.8 ± 1.5, p = 0.75) were comparable between groups. In multivariate regression analysis, CPRS-R Global Index T-score (p = 0.02) and full-scale intelligence quotient (p = 0.03) were independent predictors of changes in the ABC-I score. The frequency of adverse events was similar in both groups. Serum prolactin levels decreased in the aripiprazole group at 12 weeks but increased in the risperidone group. CONCLUSIONS: Risperidone and aripiprazole demonstrate comparable efficacy and safety in managing irritability in children and adolescents with ASD. Trial Registry no: Clinical Trial Registry of India (CTRI/2021/12/038721).
BACKGROUND: Self-limited epilepsy with centrotemporal spikes (SeLECTS) is a well-known self-limited focal epilepsy in children. While centrotemporal discharges remit with age, the timing and biomarkers predicting EEG nor...BACKGROUND: Self-limited epilepsy with centrotemporal spikes (SeLECTS) is a well-known self-limited focal epilepsy in children. While centrotemporal discharges remit with age, the timing and biomarkers predicting EEG normalization and polytherapy needs are not well established. PURPOSE: This study aimed to identify the timing of EEG remission and electroclinical biomarkers influencing remission and polytherapy needs in SeLECTS. METHODS: We retrospectively reviewed 153 of 401 patients (<18 years) with SeLECTS whose sleep EEGs normalized (2010-2025, Chonnam National University Hospital). RESULTS: The median age at sleep EEG normalization was 11.4 years (IQR, 10.1-13.2), with a median interval of 45.9 months (IQR, 27.0-66.8). Seizure-onset age correlated positively with EEG normalization age (R = 0.457) and negatively with the interval to normalization (R = -0.508; P < 0.001). EEG normalized younger in patients with unilateral (10.9 vs. 12.0 years, P = 0.002) or right-dominant discharges (11.2 vs. 13.0 years, P = 0.023). The EEG normalization interval increased with a longer gap between the first and second seizures (R = 0.279; P = 0.001). It was shorter in unilateral discharges (28.1 vs. 53.9 months; P < 0.001), and longer in the polytherapy group (57.3 vs. 43.5 months; P = 0.006). Polytherapy was more frequent in early childhood (50.5 % vs. 21.0 %; P = 0.005), and in patients with Todd's paralysis (71.4 % vs. 26.0 %; P = 0.019), daytime seizures (64.7 % vs. 23.5 %; P < 0.001), or attention deficit/hyperactivity disorder (ADHD; 66.7 % vs. 25.7 %; P = 0.015). CONCLUSION: EEG remission is associated with seizure-onset age and dipole findings, whereas the need for polytherapy is influenced more by atypical clinical than electrical biomarkers.
OBJECTIVE: Whether intraventricular hemorrhage (IVH) in preterm babies affects the brainstem auditory pathway is undetermined. This study investigated the functional status of the pathway in preterm babies with IVH to as...OBJECTIVE: Whether intraventricular hemorrhage (IVH) in preterm babies affects the brainstem auditory pathway is undetermined. This study investigated the functional status of the pathway in preterm babies with IVH to assess the effect of IVH on the pathway. METHODS: Brainstem auditory evoked response (BAER) was studied at term equivalent age in 83 preterm babies with or without IVH and 35 normal term controls, and between different groups of babies to detect any abnormality in preterm IVH babies. RESULTS: Compared to the normal term group, the preterm IVH group manifested an elevated BAER threshold (p < 0.01). The IVH group also manifested moderate prolongation in BAER wave latencies and significant reduction in waves III and V amplitudes (p < 0.05, and 0.05). Compared to the age-matched non-IVH group, the IVH group manifested a moderately higher BAER threshold and relatively shorter wave V latency. However, the I-V interval in the IVH group was significantly shorter than in the non-IVH group (p < 0.05). The amplitudes of BAER waves in the IVH group were smaller than in the non-IVH group, and the differences were significant for wave III amplitude (p < 0.05). CONCLUSION: This first specific BAER study in IVH revealed that preterm babies with IVH are associated with moderately elevated hearing threshold and shortened brainstem auditory conduction. Brainstem auditory function in preterm babies is affected by IVH.
INTRODUCTION: TANGO2 deficiency disorder (TDD) is a rare autosomal recessive disease that causes recurrent metabolic crises characterized by encephalopathy, rhabdomyolysis, fatal arrhythmia, and hypothyroidism, among oth...INTRODUCTION: TANGO2 deficiency disorder (TDD) is a rare autosomal recessive disease that causes recurrent metabolic crises characterized by encephalopathy, rhabdomyolysis, fatal arrhythmia, and hypothyroidism, among others. However, the clinical course of muscle magnetic resonance imaging (MRI) and thyroid function in TDD have not been well described. CASE PRESENTATION: The patient was a 3-year-old boy with no relevant medical history. After showing episodic ataxia, he developed metabolic crises twice and was eventually diagnosed with TDD with a novel variant. During the clinical course, MRI showed migratory signal changes in the lower limb muscles, primarily affecting the gastrocnemius, soleus, biceps femoris, and vastus medialis during the two episodes of rhabdomyolysis. Furthermore, the patient experienced a thyroid function abnormality with a primary hypothyroidism pattern that resolved spontaneously. CONCLUSION: The case findings provide novel clinical insights into muscle MRI findings and thyroid function abnormalities in TDD and can help in early diagnosis and management of TDD.
Kajiwara K, Setoyama D, Higashi K
… +15 more, Nomiyama T, Ichimiya Y, Kumamoto D, Akamine S, Sonoda Y, Chong PF, Takemoto R, Matsuoka W, Mizuguchi S, Kaku N, Kato TA, Akahoshi T, Kunisaki Y, Sakai Y, Ohga S
BACKGROUNDS: Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is the most frequent form of acute encephalopathy in early childhood in Japan. Magnetic resonance imaging provides useful hallmar...BACKGROUNDS: Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is the most frequent form of acute encephalopathy in early childhood in Japan. Magnetic resonance imaging provides useful hallmarks of diagnosing AESD. However, metabolomic profiles for AESD remain elusive. This study investigates whether measurement of amino acids in the cerebrospinal fluid (CSF) is useful for the diagnosis of AESD before onset. METHODS: In the first study, CSF samples were collected from patients (11 AESD and 17 controls) admitted to Kyushu University Hospital during 2011-2016. Amino acids in the CSF were analyzed using mass spectrometry. Cytometric bead arrays were used to measure cytokine and chemokine levels in the CSF. The second study was performed by recruiting patients (8 AESD patients and 10 controls) admitted during 2011-2024. CSF samples were stored at -20 °C for 1 month to 12 years. RESULTS: In the first study, glutamate levels in the CSF from AESD patients were higher than in controls and correlated with methionine, threonine, and tyrosine levels. A correlation map of cytokines and amino acids revealed that glutamate formed a cluster with IL-1β, IL-10, and IL-12 p70. In the second study, no difference in glutamate levels was observed between AESD and control groups. CONCLUSIONS: CSF glutamate potentially serves as a useful marker for diagnosing AESD. The long-term storage of CSF samples was likely to cause a decay of glutamate in the CSF. Prospective studies using fresh CSF samples are necessary to validate the results in this study.
PURPOSE: To retrospectively analyze the clinical characteristics and relapse factors of children with relapsed anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis in south China. METHODS: Clinical data of children di...PURPOSE: To retrospectively analyze the clinical characteristics and relapse factors of children with relapsed anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis in south China. METHODS: Clinical data of children diagnosed with relapsed anti-NMDAR encephalitis in Guangzhou Women and Children's Medical Center from October 2014 to June 2022 were collected. Patients with monophasic disease course in the same follow-up period were randomly selected as controls. Statistical analysis was performed using SPSS IBM 28.0 and figures were graphed using GraphPad Prism 7.01. RESULTS: A total of 18 children diagnosed with relapsed anti-NMDAR encephalitis (male: female 5:13). The initial onset age was (9.8 ± 3.1) year-old. Relapse interval was 7.0 [interquartile range (IQR) 6.5-15.0] months. Compared with relapses, neurological symptoms were more extensive and mRS scores were higher at the first episode (P = 0.000 in both). Female gender was an independent risk factor for relapse [odds ratio (OR) =0.055, 95 % confidence interval (CI): 0.003-0.907, P = 0.043]. Compared to the patients with monophasic disease course, the relapsed ones were prone to leave neurological sequelae (P = 0.011) at the last follow-up. CONCLUSIONS: Relapses often occur within 1 year after the first episode in children with anti-NMDAR encephalitis. Relapses were milder than first episodes. Female sex is an independent risk factor for relapses. The relapsed individuals were prone to leave neurological sequelae at the last follow-up.
BACKGROUND: Small for gestational age (SGA) and developmental coordination disorder (DCD) are receiving increasing attention in pediatric development. Understanding the risk of DCD, particularly in relation to SGA, would...BACKGROUND: Small for gestational age (SGA) and developmental coordination disorder (DCD) are receiving increasing attention in pediatric development. Understanding the risk of DCD, particularly in relation to SGA, would support children's health and development. However, the relationship between SGA and DCD remains unveiled beyond single-cohort studies. OBJECTIVES: This study aimed to integrate findings on DCD from different cohorts within the nationwide prospective Japanese Birth Cohort Consortium (JBiCC). STUDY DESIGN AND SUBJECTS: DCD was assessed in children aged 4 to 7 years from three birth cohorts participating in the JBiCC: the Hokkaido Study on Environment and Children's Health (Hokkaido Study), the Hamamatsu Birth Cohort for Mothers and Children (HBC Study), and the Tohoku Medical Megabank Project Birth and Three-Generation Cohort Study (TMM BirThree Cohort Study). OUTCOME MEASURES: DCD was assessed using either the Developmental Coordination Disorder Questionnaire Japanese Version (DCDQ-J) or the Ages and Stages Questionnaires Third Edition (ASQ-3). Logistic regression was used to assess the association between SGA and DCD in each cohort. Second, meta-analysis of the association between SGA and DCD defined by DCDQ-J, and individual patient data (IPD) meta-analysis of the association between SGA and DCDQ-J scores were conducted with two cohorts. RESULTS AND CONCLUSIONS: The analysis included 14,233 children in three cohorts. The individual cohort analyses did not explore statistical significance, except for the TMM BirThree Cohort Study. Meta-synthesis of the Hokkaido and HBC studies showed a β-coefficient of -2.63, 95 % CI [-5.22, -0.03]. IPD analysis of linear regression showed a β-coefficient of -2.76, 95 % CI [-5.38, -0.15]. Our results suggest that SGA may be a potential risk factor for DCD.
OBJECTIVES: Febrile seizure (FS) is a worrisome event for parents. This study aimed to investigate how stress and perceived stigma are alleviated after experience FS in their children. METHODS: Parents who visited hospit...OBJECTIVES: Febrile seizure (FS) is a worrisome event for parents. This study aimed to investigate how stress and perceived stigma are alleviated after experience FS in their children. METHODS: Parents who visited hospitals because of FS exhibited by their children were invited to participate this study. One hundred and sixty-one parents answered the questionnaire (1st Q); among them, 52 parents answered the same questionnaire (2nd Q) after 3 months. The questionnaire consisted of the Impact of Event Scale-Revised (IES-R) and the Parent Stigma Scale (PSS). The scores between the 1st Q and 2nd Q were compared with 52 participants who answered both questionnaires. RESULTS: The median value of the IES-R total score was 11, and 20 participants showed >24 in the 1st Q (n = 161). IES-R subscales of Intrusion and Hyperarousal symptoms and one item of PSS, "people who know your children have a FS treat your children differently", decreased significantly (p<0.010, p = 0.013 and p = 0.038, respectively) from the 1st Q (n = 52) to 2nd Q (n = 52). Parents with children having a past history of FS showed significantly higher PSS scores than parents with children having first FS episode (p = 0.029) in the 1st Q (n = 161). Parents who had some knowledge about FS could deal significantly more appropriately with FS attacks compared with those who had no prior knowledge (p<0.001) in both the 1st Q (n = 161) and 2nd Q (n = 52). CONCLUSIONS: Stress and stigma in parents were reduced to some extent 3 months after the FS attacks in their children. Recurrent FS can cause a transient perception of parental stigma. Appropriate education regarding correct management before experiencing the first episode of FS may be important.