Publications by authors named "Yushiro Yamashita"

109 Publications

Validation of actigraphy in hospitalised newborn infants using video polysomnography.

J Sleep Res 2021 Jul 15:e13437. Epub 2021 Jul 15.

Department of Paediatrics and Child Health, Kurume University School of Medicine, Kurume, Japan.

Actigraphy has been established as a reliable sleep assessment tool in adults; however, its utility in newborns remains unknown. Validation of actigraphy in newborns may provide a significant insight into the physiological and pathological acquisition process of mature diurnal sleep patterns and subsequent morbidities in both newborns and their mothers. Thus, the present study aimed to evaluate the accuracy of sleep-wake detection by overnight actigraphy in a cohort of newborns. Simultaneous recording of polysomnography and actigraphy data was performed in 40 newborns admitted to a tertiary neonatal intensive care unit (NICU). A mixed-effects logistic regression model to explain the sleep state identified by polysomnography was employed using the actigraphic activity score as a fixed independent variable and the individual newborn's identity as a random effect. To evaluate the usefulness of the actigraphic activity score as a surrogate marker of sleep, a receiver operating characteristic (ROC) curve analysis was performed using the variables that were used in the mixed-effects logistic regression model, and the area under the curve (AUC) was assessed. The results showed that polysomnography-determined sleep epochs were associated with a smaller activity index on actigraphy (odds ratio per 10 activity indices increase 0.81, 95% confidence interval [CI] 0.79-0.84). The AUC for the ROC curve was 0.87 (95% CI 0.87-0.88, range 0.54-0.99). An activity score of 124 showed the maximum overall accuracy (90.2%, 95% CI 87.7-92.1). Our present study suggests that sleep-wake states of NICU-hospitalised newborns can be precisely determined using actigraphy on the ankle.
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http://dx.doi.org/10.1111/jsr.13437DOI Listing
July 2021

Melatonin Treatment and Adequate Sleep Hygiene Interventions in Children with Autism Spectrum Disorder: A Randomized Controlled Trial.

J Autism Dev Disord 2021 Jun 28. Epub 2021 Jun 28.

Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, Japan.

Robust clinical evidence has not been available for melatonin, a drug commonly administered for treating sleep problems of children with autism spectrum disorder (ASD). In a phase 3 randomized, placebo-controlled clinical trial, we administered 1-mg melatonin (n = 65), 4-mg melatonin (n = 65), or placebo (n = 66) to196 children with ASD once daily before bedtime under adequate sleep hygiene interventions. The primary outcome was sleep onset latency (SOL) assessed with the electronic sleep diary. SOL shortened significantly in the 1- and 4-mg melatonin groups compared to the placebo group (- 22.0, - 28.0, and - 5.0 min, respectively; p < 0.0001 each). This therapeutic regimen of melatonin is a reasonable clinical approach to cope with ASD-emergent difficulties in children with ASD.
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http://dx.doi.org/10.1007/s10803-021-05139-wDOI Listing
June 2021

Gene Mutation Presenting at Age 2 Years With Autoimmune Retinopathy and Steroid-Responsive Acute Liver Failure: A Case Report and Literature Review.

Front Immunol 2021 28;12:687280. Epub 2021 May 28.

Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Japan.

Autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED) is a rare monogenic autosomal recessive disorder caused by mutation in the autoimmune regulator (AIRE) gene. Patients usually are diagnosed at ages between 5 and 15 years when they show 3 or more manifestations, most typically mucocutaneous candidiasis, Addison's disease, and hypoparathyroidism. APECED-associated hepatitis (APAH) develops in only 10% to 40% of patients, with severity varying from subclinical chronic active hepatitis to potentially fatal acute liver failure (ALF). Ocular abnormalities are fairly common, most often keratopathy but sometimes retinopathy. Here we report a 2-year-old Japanese girl with an gene mutation who developed APAH with ALF, preceded by autoimmune retinopathy associated with anti-recoverin antibody before major symptoms suggested a diagnosis of APECED. Intravenous pulse methylprednisolone therapy followed by a corticosteroid combined with azathioprine treatment resolved ALF and achieved control of APAH. To our knowledge, our patient is the youngest reported to have ALF resulting from an gene mutation. Pulse methylprednisolone induction therapy followed by treatment with corticosteroid plus azathioprine may well be effective in other children with APAH and gene mutations.
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http://dx.doi.org/10.3389/fimmu.2021.687280DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8194255PMC
May 2021

Serum Zinc and Selenium in Children with Inflammatory Bowel Disease: A Multicenter Study in Japan.

Dig Dis Sci 2021 Jun 8. Epub 2021 Jun 8.

Department of Pediatrics and Child Health, Kurume University School of Medicine, 67 Asahi-machi, Kurume, 8300011, Japan.

Background: Reports of zinc and selenium deficiencies accompanying inflammatory bowel disease (IBD) mostly have originated from Western countries and concerned adult patients. Whether Japanese children with IBD have similar deficiencies remained unclear.

Aim: We aimed to elucidate differences in serum zinc and selenium concentrations in Japanese children between types of IBD.

Methods: Children under 17 years old undergoing care at 12 Japanese pediatric centers were retrospectively enrolled between November 2016 and February 2018 to 3 groups representing Crohn's disease (CD), ulcerative colitis (UC), and normal controls (NC) with irritable bowel syndrome or no illnesses. Serum zinc and selenium were measured by atomic absorption spectrophotometry. Zinc and selenium deficiencies were defined by serum concentrations < 70 μg/dL and < 9.5 μg/dL, respectively.

Results: Subjects included 98 patients with CD (median age, 13 years), 118 with UC (11 years), and 43 NC (11 years). Serum zinc and selenium were significantly lower in CD (median, 64 and 12.6 μg/dL respectively) than in UC (69 and 14.6; P < 0.05 and P < 0.001) or NC (77 and 15.7; P < 0.01 and P < 0.001). Zinc deficiency was significantly more prevalent in CD (60.2%) than in NC (37.2%; P < 0.05), but not than in UC (51.7%; P = 0.22). Selenium deficiency was significantly more prevalent in CD (15.3%) than in UC (5.9%; P < 0.05) or NC (0%; P < 0.01).

Conclusions: In Japanese children under 17 years old, serum zinc and selenium were significantly lower in CD than in UC or NC. Zinc and selenium should be monitored, and supplemented when deficient, in children with IBD, especially CD.
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http://dx.doi.org/10.1007/s10620-021-07078-zDOI Listing
June 2021

Revisions of clinical protocols using the Plan Do Check Act cycle improved outcomes of extremely preterm infants at 2 years.

Acta Paediatr 2021 Jul 25;110(7):2100-2109. Epub 2021 Mar 25.

Department of Paediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, Japan.

Aim: Clinical quality improvement is often cumbersome due to established protocols. We aimed to investigate whether outcomes of preterm infants improve with protocol revisions using iteration cycles.

Methods: Preterm infants born <28 weeks gestation between January 2006 and December 2015 were retrospectively analysed. Protocols were revised using Plan Do Check Act cycle. Death and serious adverse events at term were reviewed in six-monthly quality improvement meetings. Adverse outcome of death or motor/sensory impairments at two years was compared before and after two major protocol changes, which were implemented in January 2008 and January 2012.

Results: Based on the appraisal for period 2006-2007, strategies for surfactant, narcotics, parenteral nutrition, respiratory gas humidity and prophylactic indomethacin and antibiotics were changed for period 2008-2011. For period 2012-2015, stabilisation of infants was accelerated via very early catheterisation. Of 162 infants (84 males, 25.5 ± 1.5 weeks gestation) within the whole cohort, 63 developed adverse outcomes, which were fewer for periods 2008-2011 (p = 0.013) and 2012-2015 (p = 0.035) compared with period 2006-2007 (adjusted for gestational age, Apgar scores and sex).

Conclusion: Careful bottom-up revisions of protocols using iteration cycles, accounting for local settings, successfully improved the outcomes of preterm infants.
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http://dx.doi.org/10.1111/apa.15840DOI Listing
July 2021

A Japanese prospective multicenter study of urinary oxysterols in biliary atresia.

Sci Rep 2021 Mar 2;11(1):4986. Epub 2021 Mar 2.

Department of Pediatrics and Child Health, Kurume University School of Medicine, 67 Asahi-machi, Kurume, 8300011, Japan.

Diagnosis of biliary atresia (BA) can involve uncertainties. In the present prospective multicenter study, we considered whether urinary oxysterols represent a useful marker for diagnosis of BA in Japanese children. Subjects under 6 months old at 7 pediatric centers in Japan were prospectively enrolled, including patients with cholestasis and healthy controls (HC) without liver disease. Patients with cholestasis constituted 2 groups representing BA patients and others with cholestasis from other causes (non-BA). We quantitatively analyzed 7 oxysterols including 4β-, 20(S)-, 22(S)-, 22(R)-, 24(S)-, 25-, and 27-hydroxycholesterol by liquid chromatography/electrospray ionization-tandem mass spectrometry. Enrolled subjects included 14 with BA (median age 68 days; range 26-170) and 10 non-BA cholestatic controls (59; 14-162), as well as 10 HC (57; 25-120). Total urinary oxysterols were significantly greater in BA (median, 153.0 μmol/mol creatinine; range 24.1-486.7; P < 0.001) and non-BA (36.2; 5.8-411.3; P < 0.05) than in HC (2.7; 0.8-7.6). In patients with BA, urinary 27-hydroxycholesterol (3.61; 0.42-11.09; P < 0.01) was significantly greater than in non-BA (0.71; 0-5.62). In receiver operating characteristic (ROC) curve analysis for distinguishing BA from non-BA, the area under the ROC curve for urinary 27-hydroxycholesterol was 0.83. In conclusion, this first report of urinary oxysterol analysis in patients with BA indicated that 27-hydroxycholesterol may be a useful marker for distinguishing BA from other causes of neonatal cholestasis.
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http://dx.doi.org/10.1038/s41598-021-84445-wDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7925559PMC
March 2021

Complications in patients with neurological impairment after gastrostomy.

Pediatr Int 2021 Feb 9. Epub 2021 Feb 9.

Department of Pediatric Surgery, Kurume University School of Medicine, Fukuoka, Japan.

Background: Neurological impairment (NI) is responsible for most conditions that require a permanent gastrostomy tube. The present study assessed the occurrence of short- and long-term complications after video-assisted gastrostomy (VAG) in patients with NI.

Methods: The incidence rates of short- (<6 months) and long-term (over two years) complications of VAG were analyzed in a retrospective study. The differences between the incidence rates of the complications of VAG according to the age at surgery (≥16 vs. ≤15 years old) were also evaluated. The short- and long-term complications observed were granulation tissue formation, infection requiring antibiotic treatment, skin problems, perigastrostomy leakage, vomiting, accidental tube dislodgement, dumping syndrome, ileus, and peritonitis.

Results: Eighty-two patients were evaluated for short- and long-term complications. The long-term complication rate was significantly lower than the short-term complication rate (p=0.0026). Onodera's prognostic nutritional index before VAG in patients with long-term complications was significantly lower than in patients without such complications (p=0.046). The incidence rates of long-term granulation tissue formation, infection, and vomiting were significantly lower than those of similar short-term complications. Long-term skin problems were associated with short-term skin problems (odds ratio: 18.95; 95% confidence interval: 4.53-92.98; p<0.001). The number of patients ≥16 years old with short- and long-term skin problems was significantly higher than in patients ≤15 years old (p=0.0014 and p=0.0073, respectively) CONCLUSIONS: The incidence rate of granulation tissue formation and infection after VAG were lower in the long term than in the short term. However, patients ≥16 years old presented with persistent complications.
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http://dx.doi.org/10.1111/ped.14645DOI Listing
February 2021

Using the drug repositioning approach to develop a novel therapy, tipepidine hibenzate sustained-release tablet (TS-141), for children and adolescents with attention-deficit/hyperactivity disorder.

BMC Psychiatry 2020 11 10;20(1):530. Epub 2020 Nov 10.

Aiiku Counselling Office, Aiiku Research Institute, Imperial Gift Foundation Boshi-Aiiku-Kai, Tokyo, Japan.

Background: Asverin® (tipepidine hibenzate) has been used as an antitussive for > 50 years in Japan. Studies revealed that tipepidine modulates monoamine levels, by inhibiting G-protein-activated inwardly rectifying potassium (GIRK) channels, expecting the potential therapeutic effects of tipepidine for attention-deficit/hyperactivity disorder (ADHD) in recent years. In this study, TS-141, a sustained-release tablet of tipepidine, was developed for the treatment of ADHD through a drug repositioning approach.

Methods: The sustained-release profile of TS-141 in healthy adults was investigated, and tipepidine exposure in the plasma after the TS-141 administration was compared to that of Asverin in the phase I study. Phase II study was conducted to examine the effects of TS-141 30 (once a day), 60 (once a day), 120 mg (60 mg twice a day), or placebo, that is within the exposure in the maximum dosage of Asverin, in children and adolescents with ADHD, and was designed as an 8-week treatment, randomized, parallel group, double-blind, placebo-controlled trial recruiting 6-17-year-old children and adolescents diagnosed with ADHD. A total of 216 patients were randomized according to the CYP2D6 phenotype. The primary end-point was ADHD Rating Scale IV-J changes. Furthermore, effects of CYP2D6 phenotype on the efficacy in the subgroup analysis were investigated.

Results: TS-141 had the sustained-release profile, and the CYP2D6 phenotype had effects on the plasma exposure of tipepidine. ADHD RS-IV-J scores in all TS-141 dosages decreased from their baseline scores; however, no significant difference was observed in ADHD RS-IV-J score changes between the placebo and TS-141-administered groups. In patients with intermediate metabolizer CYP2D6, ADHD RS-IV-J score changes in the 120 mg group tended to be larger than that in the placebo group.

Conclusions: ADHD RS-IV-J changes on TS-141 may depend on the interaction between the TS-141 dose and CYP2D6 phenotype, suggesting that further clinical trials should be conducted with careful consideration of polymorphism. Drug repositioning approach of TS-141 was attempted at the same dose as that of antitussive; however, dose setting according to the indication was necessary.

Trial Registration: Phase I study: JapicCTI-205235 (Registered 25 March 2020), Phase II study: JapicCTI-163244 (Registered 9 May 2016), https://www.clinicaltrials.jp/cti-user/trial/Show.jsp.
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http://dx.doi.org/10.1186/s12888-020-02932-2DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7653993PMC
November 2020

Diagnostic accuracy of serum proteinase 3 antineutrophil cytoplasmic antibodies in children with ulcerative colitis.

J Gastroenterol Hepatol 2021 Jun 29;36(6):1538-1544. Epub 2020 Oct 29.

Division of Gastroenterology, Department of Medicine, Kurume University School of Medicine, Kurume, Japan.

Background And Aim: Serologic markers such as myeloperoxidase (MPO) antineutrophil cytoplasmic antibodies (ANCA) (MPO-ANCA) have been used to screen patients for ulcerative colitis (UC). However, MPO-ANCA shows limited accuracy in Asians. Proteinase 3 ANCA (PR3-ANCA) has performed better at UC diagnosis in Japanese adults than MPO-ANCA. The present study aimed to evaluate usefulness of PR3-ANCA for diagnosis of UC in Japanese pediatric practice.

Methods: Patients under 17 years old undergoing assessment at 12 Japanese pediatric centers between November 2016 and February 2018 were prospectively enrolled and divided into groups with UC, Crohn's disease (CD), intestinal disease control (IC), and healthy control (HC). Serum PR3-ANCA and MPO-ANCA were analyzed using chemiluminescence enzyme immunoassay kits.

Results: Sera from 367 patients (148 with UC at a median age of 12 years; 120 with CD, 13 years; 56 with IC, 10.5 years; and 43 with HC, 10 years) were examined. Median PR3-ANCA values in UC (1.6 U/mL) were greater than in CD (0.2; P < 0.001), IC (0.15; P < 0.001), and HC (0.1; P < 0.001). In receiver operating characteristic curve analyses, the area under the curve for PR3-ANCA was 0.79, significantly greater than for MPO-ANCA (0.58; P < 0.001). Using a cut-off value of 0.8 U/mL determined from the receiver operating characteristic analyses, PR3-ANCA showed significantly greater sensitivity (64.9%) than MPO-ANCA (cut-off, 0.2 U/mL; sensitivity, 19.6%; P < 0.001) and good specificity (83.6%).

Conclusions: In Japanese children and adolescents, PR3-ANCA performed better as a serologic marker for diagnosis of UC than MPO-ANCA. To our knowledge, this is the first report of such a comparison.
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http://dx.doi.org/10.1111/jgh.15296DOI Listing
June 2021

Diagnostic accuracy of serum proteinase 3 antineutrophil cytoplasmic antibodies in children with ulcerative colitis.

J Gastroenterol Hepatol 2021 Jun 29;36(6):1538-1544. Epub 2020 Oct 29.

Division of Gastroenterology, Department of Medicine, Kurume University School of Medicine, Kurume, Japan.

Background And Aim: Serologic markers such as myeloperoxidase (MPO) antineutrophil cytoplasmic antibodies (ANCA) (MPO-ANCA) have been used to screen patients for ulcerative colitis (UC). However, MPO-ANCA shows limited accuracy in Asians. Proteinase 3 ANCA (PR3-ANCA) has performed better at UC diagnosis in Japanese adults than MPO-ANCA. The present study aimed to evaluate usefulness of PR3-ANCA for diagnosis of UC in Japanese pediatric practice.

Methods: Patients under 17 years old undergoing assessment at 12 Japanese pediatric centers between November 2016 and February 2018 were prospectively enrolled and divided into groups with UC, Crohn's disease (CD), intestinal disease control (IC), and healthy control (HC). Serum PR3-ANCA and MPO-ANCA were analyzed using chemiluminescence enzyme immunoassay kits.

Results: Sera from 367 patients (148 with UC at a median age of 12 years; 120 with CD, 13 years; 56 with IC, 10.5 years; and 43 with HC, 10 years) were examined. Median PR3-ANCA values in UC (1.6 U/mL) were greater than in CD (0.2; P < 0.001), IC (0.15; P < 0.001), and HC (0.1; P < 0.001). In receiver operating characteristic curve analyses, the area under the curve for PR3-ANCA was 0.79, significantly greater than for MPO-ANCA (0.58; P < 0.001). Using a cut-off value of 0.8 U/mL determined from the receiver operating characteristic analyses, PR3-ANCA showed significantly greater sensitivity (64.9%) than MPO-ANCA (cut-off, 0.2 U/mL; sensitivity, 19.6%; P < 0.001) and good specificity (83.6%).

Conclusions: In Japanese children and adolescents, PR3-ANCA performed better as a serologic marker for diagnosis of UC than MPO-ANCA. To our knowledge, this is the first report of such a comparison.
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http://dx.doi.org/10.1111/jgh.15296DOI Listing
June 2021

Long-term melatonin treatment for the sleep problems and aberrant behaviors of children with neurodevelopmental disorders.

BMC Psychiatry 2020 09 10;20(1):445. Epub 2020 Sep 10.

Department of Pediatrics and Child Health, Kurume University School of Medicine, 67 Asahi-machi, Kurume, Fukuoka, 830-0011, Japan.

Background: Clinical evidence is required about the long-term efficacy and safety of melatonin treatment for sleep problems in children with neurodevelopmental disorders (NDDs) who underwent adequate sleep hygiene interventions.

Methods: We conducted a 26-week, multicenter, collaborative, uncontrolled, open-label, phase III clinical trial of melatonin granules in children 6 to 15 years of age who had NDDs and sleep problems. The study consisted of the 2-week screening phase, the 26-week medication phases I and II, and the 2-week follow-up phase. Children received 1, 2, or 4 mg melatonin granules orally in the medication phases. Variables of sleep status including sleep onset latency (SOL), aberrant behaviors listed on the Aberrant Behavior Check List-Japanese version (ABC-J), and safety were examined. The primary endpoint was SOL in the medication phase I.

Results: Between June 2016 and July 2018, 99 children (80 males and 19 females, 10.4 years in mean age) were enrolled at 17 medical institutions in Japan-74, 60, 22, 9, 6, and 1 of whom had autism spectrum disorder, attention-deficit/hyperactivity disorder, intellectual disabilities, motor disorders, specific learning disorder, and communication disorders, respectively, at baseline. Fifteen children received the maximal dose of 4 mg among the prespecified dose levels. SOL recorded with the electronic sleep diary shortened significantly (mean ± standard deviation [SD], - 36.7 ± 46.1 min; 95% confidence interval [CI], - 45.9 to - 27.5; P <  0.0001) in the medication phase I from baseline, and the SOL-shortening effect of melatonin persisted in the medication phase II and the follow-up phase. Temper upon wakening and sleepiness after awakening improved significantly (P <  0.0001 each) in the medication phase I from baseline and persisted in the follow-up phase. The following subscales of the ABC-J improved significantly: stereotypic behavior (P = 0.0322) in the medication phase I; and irritability, hyperactivity, and inappropriate speech (P <  0.0001) in the medication phase II. Treatment-emergent adverse events did not occur subsequent to week 16 after medication onset, and NDDs did not deteriorate in the follow-up phase.

Conclusions: Long-term melatonin treatment in combination with adequate sleep hygiene interventions may afford clinical benefits to children with NDDs and potentially elevates their well-being.

Trial Registration: ClinicalTrils.gov , NCT02757066 . Registered April 27, 2016.
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http://dx.doi.org/10.1186/s12888-020-02847-yDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7488027PMC
September 2020

Meaningful word acquisition is associated with walking ability over 10 years in Rett syndrome.

Brain Dev 2020 Nov 17;42(10):705-712. Epub 2020 Jul 17.

Department of Mental Retardation and Birth Defect Research, National Center of Neurology and Psychiatry, 4-1-1 Ogawahigashi-machi, Kodaira, Tokyo 187-8502, Japan.

Purpose: To investigate walking ability in Japanese patients with Rett syndrome (RTT).

Methods: Walking ability was assessed in 100 female Japanese patients with RTT using univariate and multivariate analysis in all age groups, and in patients over 10 years of age. We analyzed walking ability and confounding factors including prenatal-perinatal histories, developmental milestones, somatic and head growth, anthropometric data, body mass index, age of loss of purposeful hand use, age at onset of stereotypic hand movement, history of autistic behavior, age at regression, presence or absence of seizures, and the results of MECP2 genetic examination from the Japanese Rett syndrome database.

Results: Univariate analysis revealed that acquisition of walking in all age groups was significantly correlated with the acquisition of meaningful words, microcephaly, and crawling (P < 0.0001, P = 0.005, P < 0.0001, respectively). Univariate analysis revealed that walking ability over 10 years of age was significantly correlated with acquisition of meaningful words, microcephaly, and body mass index (P < 0,0001, P = 0.005, P = 0.0018, respectively). MECP2 mutations R306C, R133C, and R294X were significantly associated with different acquisition of crawling (P = 0.004) and walking (P = 0.01). Multivariate analysis revealed that only acquisition of meaningful words was significantly correlated with walking ability over 10 years of age. This trend excluded the genetic effects of R306C, R133C, and R294X.

Conclusions: Meaningful word acquisition was robustly associated with walking ability over 10 years. Prognosis of walking ability may be predicted by the acquisition of meaningful words. This information is potentially useful for early intervention and the planning of comprehensive treatment for young children with RTT.
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http://dx.doi.org/10.1016/j.braindev.2020.06.012DOI Listing
November 2020

Executive dysfunction in medication-naïve children with ADHD: A multi-modal fNIRS and EEG study.

Brain Dev 2020 Sep 10;42(8):555-563. Epub 2020 Jun 10.

Department of Developmental Disorders, National Institute of Mental Health, National Center of Neurology and Psychiatry, 4-1-1 Ogawahigashi-cho, Kodaira, Tokyo 187-8553, Japan.

Objective: Children with attention deficit hyperactivity disorder (ADHD) exhibit deficits in executive function. Since there are no clear biomarkers for the disorder, this study aimed to investigate the neurophysiological biomarkers for deficits in executive function in children with ADHD using functional near-infrared spectroscopy (fNIRS) and electroencephalography.

Methods: Twenty patients diagnosed with ADHD and 19 typically developing children (TDC; 8-11 years old) were included. Event related potentials (ERPs) were recorded using an electroencephalogram (EEG) and oxygenated hemoglobin concentrations (Oxy-Hb) were recorded using fNIRS during a colored Go/NoGo task, simultaneously. Latencies and amplitudes of NoGo-N2 and NoGo/Go-P3 tasks were measured using EEG.

Results: Children with ADHD showed significantly decreased Oxy-Hb in the right frontal cortex as well as longer NoGo-P3 latencies and a decreased NoGo/Go-P3 amplitude. There was a significant positive correlation between the Oxy-Hb and NoGo/Go-P3 amplitude.

Conclusions: These results suggest that children with ADHD experience executive dysfunction. Hemodynamic and electrophysiological findings during the Go/NoGo task might be useful as a biomarker of executive function.

Significance: These findings have key implications for understanding the pathophysiology of deficits in executive function in ADHD.
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http://dx.doi.org/10.1016/j.braindev.2020.05.007DOI Listing
September 2020

Association between children's sleep patterns and problematic behaviors at age 5.

Pediatr Int 2020 Oct;62(10):1189-1196

Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Japan.

Background: Night-shift lifestyles affect children as well as adults, and are associated with sleep and behavioral problems among children. This study aimed to investigate associations among sleep patterns, individual/environmental factors, and problematic behaviors in children at age 5 years.

Methods: Data for sleep patterns, individual / environmental factors, and problematic behaviors for 8,689 5-year-old children were collected from health-checkup records. Problematic behaviors investigated were anxious behavior (being afraid, difficulty being separated from the mother), developmental behavior (violence, restlessness, rebellious behavior, restrictive diet, stereotypic play), personal habits (thumb-sucking, nail-biting, tic, masturbation), and excretory problems. The relationships between sleep patterns (bedtime, sleep duration) and the presence of these behaviors were analyzed. Individual / environmental factors that affected problematic behaviors were statistically identified using a tree-form model.

Results: Late bedtime and short sleep duration showed significant adverse effects on children's problematic behaviors - odds ratio (OR): 1.07, 95% confidence interval (CI): 1.03-1.11 and OR: 0.92, 95% CI: 0.87-0.97, respectively. Long television watching time, abnormality at birth, and lack of father's support also showed significant adverse effects on problematic behaviors (OR: 2.34, 95% CI: 1.87-2.94), and significantly affected late bedtime and short sleep duration.

Conclusions: There were significant associations among sleep patterns, individual / environmental factors, and problematic behaviors in 5-year-old children. Improving children's sleep patterns, reducing the duration of television watching, and improving support from fathers may reduce problematic behaviors.
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http://dx.doi.org/10.1111/ped.14267DOI Listing
October 2020

Urinary and serum oxysterols in children: developmental pattern and potential biomarker for pediatric liver disease.

Sci Rep 2020 04 21;10(1):6752. Epub 2020 Apr 21.

Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, Japan.

Few reports describe oxysterols in healthy children or in children with liver disease. We aimed to determine whether developmental changes in urinary and serum oxysterols occur during childhood, and to assess whether oxysterols might be biomarkers for pediatric liver disease. Healthy children enrolled as subjects (36 and 35 for urine and serum analysis, respectively) included neonates, infants, preschoolers, and school-age children, studied along with 14 healthy adults and 8 children with liver disease. We quantitated 7 oxysterols including 4β-, 20(S)-, 22(S)-, 22(R)-, 24(S)-, 25-, and 27-hydroxycholesterol using liquid chromatography/electrospray ionization-tandem mass spectrometry. Urinary total oxysterols were significantly greater in neonates than in infants (P < 0.05), preschoolers (P < 0.001), school-age children (P < 0.001), or adults (P < 0.001), declining with age. Serum total oxysterols in neonates were significantly lower than in infants (P < 0.05), preschoolers (P < 0.001), school-age children (P < 0.05), or adults (P < 0.01). Compared with healthy children, total oxysterols and 24(S)-hydroxycholesterol in liver disease were significantly increased in both urine (P < 0.001 and P < 0.001, respectively) and serum (P < 0.001 and P < 0.05, respectively). Oxysterols in liver disease, particularly 24(S)-hydroxycholesterol, were greater in urine than serum. Oxysterols change developmentally and might serve as a biomarker for pediatric liver disease. To our knowledge, this is the first such report.
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http://dx.doi.org/10.1038/s41598-020-63758-2DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7174363PMC
April 2020

Association between problematic behaviors and individual/environmental factors in difficult children.

Brain Dev 2020 Jun 7;42(6):431-437. Epub 2020 Apr 7.

Department of Pediatrics and Child Health, Kurume University School of Medicine, Japan.

Background: Difficult children are ones whose behavior deviates from the norm, which manifests as restlessness, violence, and difficulty in separating from the mother. Such problematic behaviors usually exhaust their parents during child rearing. This study aimed to identify individual and environmental factors that influence children's problematic behavior, which could be helpful in supporting parents' child rearing.

Methods: Records of children's problematic behaviors and their individual or environmental information were collected from 8691 children at their 5-year-old health checks. Problematic behaviors were divided into three categories; anxious behaviors, developmental behaviors, and personal habits. Individual factors included sex, parental age, birth order, birth weight, and birth abnormalities. The environmental factors were mother's smoking during pregnancy or currently, partner's cooperation in child rearing, having someone to consult about child rearing, and television viewing time. Using logistic regression, we identified the association between such behaviors and aggravating factors.

Results: Problematic behavior was identified in 2.2%, 11.5%, and 16.1% of cases, respectively, with regard to anxious behaviors, developmental behaviors, and personal habits. The individual factors (including birth order and birth abnormality), and the environmental factors (including mothers currently smoking, lack of someone to consult about child rearing, and long television-watching time) were associated with the odd ratio of increased risk for some problematic behaviors.

Conclusion: Behaviors in difficult children are not influenced by individual factors but by several environmental factors. To reduce the parental child rearing burden, health providers should be aware of these aggravating factors.
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http://dx.doi.org/10.1016/j.braindev.2020.03.002DOI Listing
June 2020

Antibodies to Crohn's disease peptide 353 as a diagnostic marker for pediatric Crohn's disease: a prospective multicenter study in Japan.

J Gastroenterol 2020 May 24;55(5):515-522. Epub 2020 Jan 24.

Division of Gastroenterology, Department of Medicine, Kurume University School of Medicine, Kurume, Japan.

Background: Various serologic markers such as anti-glycoprotein 2 antibodies and anti-Saccharomyces cerevisiae antibodies have been reported to be diagnostically useful in Crohn's disease. Mitsuyama et al. reported that antibodies to Crohn's disease peptide 353, a newly proposed serologic marker, were more useful in Japanese adults than anti-Saccharomyces. We addressed the same issue in Japanese children and adolescents.

Methods: Prospectively enrolled subjects under 17 years old assessed and treated at 12 pediatric centers in Japan included groups with Crohn's disease, ulcerative colitis, other intestinal diseases, or good health. The 3 serum markers were analyzed by enzyme-linked immunosorbent assays.

Results: Enrolled subjects, numbering 367, included 120 with Crohn's disease, 148 with ulcerative colitis, 56 with other intestinal diseases, and 43 healthy subjects. In Crohn's disease, anti-Crohn's disease peptide 353, anti-glycoprotein 2, and anti-Saccharomyces concentrations (median, 2.25, 3.0, and 8.9 U/mL) were significantly greater than in ulcerative colitis (1.1, 1.9, and 3.4; all P < 0.001), other intestinal diseases (1.1, 1.85, and 2.95; all P < 0.001), and healthy controls (1.1, 1.7, and 2.8; all P < 0.001), respectively. At 95% specificity, sensitivity of anti-Crohn's disease peptide (45.0%) was significantly higher than for anti-glycoprotein 2 (30.8%; P < 0.05) or anti-Saccharomyces (26.7%; P < 0.01).

Conclusions: Anti-Crohn's disease peptide 353 proved more useful for diagnosis of Crohn's disease in Japanese children than the other 2 markers. To our knowledge, this is the first pediatric report to that effect.
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http://dx.doi.org/10.1007/s00535-019-01661-yDOI Listing
May 2020

Long-term study of lisdexamfetamine dimesylate in Japanese children and adolescents with attention-deficit/hyperactivity disorder.

Neuropsychopharmacol Rep 2020 03 8;40(1):52-62. Epub 2019 Dec 8.

Aiiku Counselling Office, Aiiku Research Institute, Imperial Gift Foundation Boshi-Aiiku-Kai, Tokyo, Japan.

Aims: As an extension of a phase 2/3 study evaluating the efficacy and safety of lisdexamfetamine dimesylate (LDX) 30, 50, or 70 mg/d for 4 weeks in Japanese patients aged 6-17 years with attention-deficit/hyperactivity disorder (ADHD), this study evaluated its long-term safety and efficacy.

Methods: This was a multicenter, open-label study of LDX for 53 weeks. Safety was assessed by regular medical examination for treatment-emergent adverse events (TEAEs); regular recording of body weight, vital signs, and laboratory test values; and completion of dependence questionnaires. Efficacy was assessed using Japanese versions of the ADHD-Rating Scale-IV (ADHD-RS-IV) and Conners' 3rd edition Parent Rating Scale (Conners 3); plus Clinical Global Impression-Improvement (CGI-I), Clinical Global Impression-Severity, and Parent Global Assessment (PGA) scales.

Results: Of 132 enrolled patients, 104 completed the trial. Most frequent treatment-related TEAEs were decreased appetite (73.5%), initial insomnia (39.4%), and weight decrease (22.0%). Most TEAEs were mild (82.6% of patients). There were no serious or severe TEAEs or deaths. No treatment-related TEAEs were associated with blood pressure or pulse rate, and no patient had a QTcF interval >500 ms. Statistically significant improvement from baseline to week 53 was observed in the mean ADHD-Rating Scale-IV total score and mean Conners 3 subscale scores. Most patients showed improvement on the CGI-I (78%) and PGA (76.5%) scales.

Conclusions: No significant safety issues were observed with LDX 30, 50, or 70 mg/d administered for 1 year in Japanese children and adolescents with ADHD. LDX was associated with long-term reductions in ADHD symptoms and severity.
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http://dx.doi.org/10.1002/npr2.12091DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7292222PMC
March 2020

Characteristics of socially high-risk pregnant women and children's outcomes.

Pediatr Int 2020 Feb 30;62(2):140-145. Epub 2020 Jan 30.

Department of Pediatrics and Child Health, Kurume University School of Medicine, Fukuoka, Japan.

Background: The number of reports of child abuse and neglect in Japan has increased each year. A causal relationship between socially high-risk pregnant women and child abuse is strongly suggested. This study aims to investigate the characteristics of socially high-risk pregnant women and their children's outcomes, to help prevent child abuse.

Methods: In total, 2,342 births were retrospectively analyzed from medical records. We extracted the frequency, factors, and circumstances of socially high-risk pregnant women, and the presence of social interventions for their children.

Results: There were 538 (23%) socially high-risk pregnant women out of 2,342 cases investigated. Related factors (with duplication) were: economic problems (258 cases, 48%), mental disorders (139 cases, 26%), teenage pregnancies (112 cases, 21%), multiple pregnancies (90 cases, 17%), and pregnancy conflict (73 cases, 14%). Sixty-four (12%) expectant mothers received their first health examination in late pregnancy or were not receiving pregnancy health examinations. An analysis of births showed neonatal intensive care unit hospitalization in 40% of the children born to socially high-risk pregnant women. The hospital Child Abuse Prevention Committee intervened in 71 cases, and child consultation centers intervened in 55 cases. Twenty-two children entered social care facilities and four children died of unknown causes.

Conclusions: Socially high-risk pregnant women had various social and individual problems, and received multidisciplinary interventions for child rearing support. Antenatal assessment and multidisciplinary early intervention for socially high-risk pregnant women are necessary to prevent child abuse.
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http://dx.doi.org/10.1111/ped.14058DOI Listing
February 2020

Canakinumab eliminates resistant familial Mediterranean fever in a Japanese girl.

Pediatr Int 2019 Nov 13;61(11):1173-1174. Epub 2019 Nov 13.

Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, Japan.

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http://dx.doi.org/10.1111/ped.13968DOI Listing
November 2019

Phase II/III Study of Lisdexamfetamine Dimesylate in Japanese Pediatric Patients with Attention-Deficit/Hyperactivity Disorder.

J Child Adolesc Psychopharmacol 2020 02 12;30(1):21-31. Epub 2019 Nov 12.

Aiiku Counseling Office, Aiiku Research Institute, Imperial Gift Foundation Boshi-Aiiku-Kai, Tokyo, Japan.

To further define the efficacy and safety profiles of lisdexamfetamine dimesylate (LDX) in Japanese pediatric patients with attention-deficit/hyperactivity disorder (ADHD). This was a multicenter, randomized, double-blind, placebo-controlled study of LDX 30, 50, or 70 mg/day for 4 weeks in 76 patients 6-17 years of age with ADHD in Japan. The primary efficacy endpoint was the change in the ADHD Rating Scale-IV (ADHD-RS-IV) total score from baseline to 4 weeks. Secondary efficacy endpoints were: Conners' Third Edition (Japanese version) Parent Rating Scale (Conners 3), Clinical Global Impression-Improvement (CGI-I) scale, and Parent Global Assessment (PGA) scale. Change in the ADHD-RS-IV total score from baseline to 4 weeks was significantly greater ( < 0.0001) in all LDX dosage groups versus placebo (30 mg, -16.38; 50 mg, -18.10; 70 mg, -16.47; placebo, -2.78). At all time points, improvements (decreases) in the ADHD-RS-IV total score were significantly greater in all LDX groups versus placebo. At weeks 3 and 4, improvements from baseline in Conners 3 inattention plus hyperactivity/impulsivity subscale scores were significantly greater ( ≤ 0.0082) for all LDX dosages versus placebo. At week 4, the proportion of LDX-treated patients "much improved" or "very much improved" was 61%-71% on the CGI-I scale ( ≤ 0.0019) and 56%-65% on the PGA scale ( ≤ 0.0170). LDX was generally well tolerated. The most frequent treatment-emergent adverse events (AEs) were decreased appetite, headache, and initial insomnia. No severe/serious AEs occurred, and no AEs specific to Japanese patients were evident. The superiority of LDX 30, 50, and 70 mg/day over placebo was confirmed in Japanese pediatric patients with ADHD, and no major safety or tolerability concerns were identified.
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http://dx.doi.org/10.1089/cap.2019.0076DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7041327PMC
February 2020

Molecular epidemiology, antimicrobial susceptibility, and characterization of fluoroquinolone non-susceptible Streptococcus pyogenes in Japan.

J Infect Chemother 2020 Mar 1;26(3):280-284. Epub 2019 Nov 1.

Department of Infection Control and Prevention, Kurume University School of Medicine, Kurume, Japan.

Streptococcus pyogenes (Group A streptococci: GAS) are known to cause a wide variety of human illnesses, some of which can be life-threatening. Usually, penicillin is the first-choice agent for the treatment of GAS infections. For patients with penicillin or beta-lactam antibiotics allergies, macrolide drugs are recommended as an alternative therapy. However, an increased prevalence of macrolide-resistant GAS (MRGAS) has been reported in many countries. Furthermore, fluoroquinolone non-susceptible GAS has been reported. The present study was focused on determining the features of fluoroquinolone non-susceptible strains collected from children with pharyngotonsillitis in the southwestern areas of Japan. To reveal the characteristics of fluoroquinolone non-susceptible GAS, we investigated the MIC, T-serotype, emm typing, and PFGE of 298 GAS strains isolated in the Fukuoka southwest area of Japan between 2011 and 2013. We determined that fluoroquinolone non-susceptibility shows a MIC to tosufloxacin of ≧1 μg/ml. We identified 33 (11.1%) fluoroquinolone non-susceptible GAS strains. In these strains, 6 T-serotypes and 9 emm/MLST patterns were detected. The predominant combinations were emm6/ST382 (14 strains, 42.4%) and emm89/ST101 (5 strains, 15.2%). PFGE classified 10 pulsotypes, and each was quite different. These results showed that fluoroquinolone non-susceptible GAS strains have a variety of origins. The usage of fluoroquinolone drugs could have a negative effect on the antimicrobial drug sensitivity of GAS in Japan. Considering such a situation, continuous monitoring of quinolone non-susceptible GAS is necessary.
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http://dx.doi.org/10.1016/j.jiac.2019.10.004DOI Listing
March 2020

Validation of a childhood eating disorder outcome scale.

Biopsychosoc Med 2019 11;13:21. Epub 2019 Sep 11.

1Department of Pediatrics and Child Health, Kurume University School of Medicine, 67 Asahi-machi, Kurume, Fukuoka, 830-0011 Japan.

We developed and validated a childhood eating disorder outcome scale based on outcomes associated with body mass index standard deviation score (BMI-SDS). This prospective observational study included 131 children with eating disorders (aged 5-15 years). Participants' outcomes scales were completed at the first visit and at 1, 3, 6, and 12 months. The scale evaluated 12 outcomes: body weight change (BW), eating attitude (EA), fear of being fat (FF), body image distortion (BD), menstruation (ME), perceived physical condition (PC), attending school (AS), disease recognition by school (RS), family function (FA), disease recognition by parent (RP), social adaptation (SA), and relationships with friends (RF). Responses to all items were on a four-point Likert scale. Exploratory factor analysis was used to determine the number of factors based on the 12 outcomes. The relation between outcome scale scores and BMI-SDS over the 12-month follow-up period was analyzed. Two types of factors were extracted: disease-specific factors (EA, FF, BD) and biopsychosocial factors (BW, PC, AS, FA, SA, RF). Three items (ME, RS, RP) were excluded because they showed no significant loading effect. There was a significant negative correlation between the outcome scale and BMI-SDS, and changes in outcome scale scores from baseline to 12 months were significantly associated with improvement in BMI-SDS. We developed a childhood eating disorder outcome scale characterized by disease-specific and biopsychosocial factors. Biopsychosocial management combined with a therapeutic approach for disease-specific symptoms may support body weight recovery for children with eating disorders.
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http://dx.doi.org/10.1186/s13030-019-0162-3DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6737718PMC
September 2019

Clinical Features, Molecular Genetics, and Long-Term Outcome in Congenital Chloride Diarrhea: A Nationwide Study in Japan.

J Pediatr 2019 11 30;214:151-157.e6. Epub 2019 Aug 30.

Department of Medical Biochemistry, Kurume University School of Medicine, Kurume, Japan.

Objective: To clarify clinical and genetic features of Japanese children with congenital chloride diarrhea (CCD).

Study Design: This was a multi-institutional, retrospective survey of 616 pediatric centers in Japan with identified patients with CCD between 2014 and 2018. Mutations involving SLC26A3 were detected by Sanger sequencing.

Results: Thirteen patients met all entry criteria including mutations in SLC26A3, and 14 patients satisfied clinical diagnostic criteria. Homozygous or compound heterozygous mutations in SLC26A3, including 6 novel mutations, were identified in 13 of these 14 patients (93%). The most common (detected in 7 of 13) was c.2063-1g>t. Median age at diagnosis was 1 day. Nine of the patients meeting all criteria were diagnosed as neonates (69%). Median follow-up duration was 10 years. When studied, 8 patients had <5 stools daily (62%), and all had fewer than in infancy. Only 1 patient had nephrocalcinosis, and 3 (23%) had mild chronic kidney disease. Neurodevelopment was generally good; only 1 patient required special education. Five patients (38%) received long-term sodium, potassium, and chloride supplementation.

Conclusions: Early fetal ultrasound diagnosis and prompt long-term sodium, potassium, and chloride supplementation were common management features. Genetic analysis of SLC26A3 provided definitive diagnosis of CCD. In contrast with previously reported localities, c.2063-1g>t might be a founder mutation in East Asia.
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http://dx.doi.org/10.1016/j.jpeds.2019.07.039DOI Listing
November 2019

Reliability and validity of the Children's Depression Inventory-Japanese version.

Pediatr Int 2019 Nov;61(11):1159-1167

Department of Psychiatry and Neuroscience, Hiroshima University Graduate School of Biomedical Sciences, Hiroshima, Japan.

Background: Depression has major negative consequences for individuals and society, and psychological assessment tools for early disease detection are needed. The aim of this study was to investigate the reliability and validity of an updated Japanese version of the Children's Depression Inventory (CDI-J) and set a cut-off score for the detection of depression.

Methods: The participants consisted of 465 children and adolescents aged 7-17 years. The control (CON) groups consisted of students recruited from elementary and junior-high school (CONEJ) and children recruited from among hospital staff members (CONRE), while the outpatient clinical (OPC) groups consisted of pediatric psychosomatic outpatients (OPCPD) and adolescent psychiatric outpatients (OPCPS). The CON and OPC CDI-J scores underwent factor analysis using varimax rotation, followed by measurement invariance analysis. The Youth Self-Report (YSR) was administered to assess concurrent validity. The Mini-International Neuropsychiatric Interview was administered to the OPC group to diagnose current depressive symptoms. Receiver operating characteristics (ROC) analysis was conducted to evaluate case-finding performance and to set cut-off points for the detection of depression.

Results: The CDI-J was reliable in terms of internal consistency (Cronbach α = 0.86; mean inter-item correlation, 0.16). Re-test reliability was substantial (mean interval 18 days: γ = 0.59, P < 0.05). The four-factor solution exhibited adequate internal consistency (range, 0.52-0.73) and correspondence (Pearson correlation of 0.65 with the YSR) for both the CON and OPC groups. On ROC analysis the optimal cut-off score was 23/24.

Conclusion: The CDI-J can be used as a reliable and well-validated instrument alongside standard diagnostic procedures.
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http://dx.doi.org/10.1111/ped.13984DOI Listing
November 2019

Laboratory values in Japanese children with newly diagnosed inflammatory bowel disease.

Pediatr Int 2019 Jul;61(7):720-725

Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, Japan.

Background: Laboratory data in children with newly diagnosed inflammatory bowel disease (IBD) have been reported from Europe and North America, but not Asia. The aim of this study was to clarify laboratory data in Japanese children with newly diagnosed IBD, and to compare them with those in Western reports.

Methods: We retrospectively reviewed patients <16 years old, newly diagnosed with ulcerative colitis (UC) or Crohn's disease (CD) at Kurume University Hospital between January 2008 and December 2015.

Results: UC and CD patients numbered 31 and 15, respectively. The percentages of patients with normal values for hemoglobin (Hb), platelet count (Plt), albumin (Alb), C-reactive protein (CRP), and erythrocyte sedimentation rate (ESR) in the UC and CD groups were 45% and 47%; 68% and 53%; 84% and 40%; 81% and 7%; and 35% and 0%, respectively. The frequency of normal results for these five tests were similar to Western findings except for the greater frequency of normal CRP in UC. Alb and ESR differed significantly between UC and CD in both mild and moderate-severe cases. Plt, Alb, CRP, and ESR differed significantly between diseases in late-onset IBD, whereas early onset IBD showed no differences. In UC, ESR correlated positively, while Hb and Alb correlated negatively, with disease activity. In CD, CRP and ESR correlated positively with activity.

Conclusions: The proportion of Japanese children with IBD having normal values at diagnosis was mostly similar to that in Western reports. In early onset cases, UC parameters may be similar to CD. Of the five tests, ESR was particularly indicative of disease activity at diagnosis in both pediatric UC and CD.
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http://dx.doi.org/10.1111/ped.13892DOI Listing
July 2019

Age-related differences in frontal lobe function in children with ADHD.

Brain Dev 2019 Aug 2;41(7):577-586. Epub 2019 Apr 2.

Department of Developmental Disorders, National Institute of Mental Health, Kumamoto University, National Center of Neurology and Psychiatry (NCNP), Kodaira, Chuo-ku, Kumamoto, Japan.

Background: The neural correlates of executive function disorders are thought to be predominantly localized within the prefrontal cortex (PFC). However, no study to date has investigated changes in this system across different age groups in children with attention deficit hyperactivity disorder (ADHD). Thus, this study aimed to explore changes in PFC function in children with ADHD.

Methods: Study participants included typically developing (TD) children (n = 140) and children with ADHD (n = 67) of primary school age. Behavioral executive functions and their neural basis were evaluated between the TD children and children with ADHD and also across different age periods (younger and older children). To examine executive function, inhibitory control was assessed using the reverse Stroop task, and PFC near-infrared spectroscopic measurements were used to investigate the neural mechanisms involved.

Results: Both ADHD symptoms and the ability to inhibit color interference improved with age. Compared to TD children, children with ADHD demonstrated decreased activation of the right and middle PFC across all age groups. Interestingly, the left PFC appeared to play a compensatory role.

Conclusion: Children with ADHD exhibited changes in PFC function that varied with age. Longitudinal studies are required to assess the potential of using PFC function as an early biomarker of ADHD.
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http://dx.doi.org/10.1016/j.braindev.2019.03.006DOI Listing
August 2019

Anti-myelin Oligodendrocyte Glycoprotein Antibody-positive Optic Neuritis in a Girl With Ulcerative Colitis.

J Pediatr Gastroenterol Nutr 2019 10;69(4):e117

Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume.

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http://dx.doi.org/10.1097/MPG.0000000000002281DOI Listing
October 2019

Infantile-onset inflammatory bowel disease in a patient with Hermansky-Pudlak syndrome: a case report.

BMC Gastroenterol 2019 Jan 11;19(1). Epub 2019 Jan 11.

Department of Pediatrics and Child Health, Kurume University School of Medicine, 67 Asahi-machi, Kurume, 830-0011, Japan.

Background: Hermansky-Pudlak syndrome (HPS) is a rare, genetically heterogeneous disorder that manifests oculocutaneous albinism together with bleeding diatheses that reflect a platelet storage pool deficiency. Ten genetic subtypes of this autosomal recessive condition have been described to date. Some patients with Hermansky-Pudlak syndrome type 1, 4, or 6 develop Crohn's-like inflammatory bowel disease at any age including early childhood, but most often in adolescence or young adulthood. Here we report infantile-onset of inflammatory bowel disease in a patient with Hermansky-Pudlak syndrome type 1 who responded to infliximab.

Case Presentation: A Japanese boy, the second child of non-consanguineous healthy parents, was born with chalky white skin, silvery-white hair, and gray eyes, representing oculocutaneous albinism. He developed frequent diarrhea and fever accompanied by weight loss at 6 months, and was diagnosed with Crohn's-like inflammatory bowel disease based on the endoscopic finding of longitudinal ulcerations in the colon and the histopathologic finding of nonspecific chronic inflammation without granulomas at the age of 11 months. Treatment with an elemental diet, salazosulfapyridine, and corticosteroids failed to improve clinical or laboratory abnormalities, and the diarrhea became bloody. At 13 months he began treatment with infliximab, which produced marked improvement followed by clinical remission. Endoscopy at 20 months demonstrated healing of the colonic mucosa. At 22 months he is in sustained clinical remission receiving only infliximab. Because albinism with inflammatory bowel disease suggested Hermansky-Pudlak syndrome, we performed genetic screening using next-generation sequencing in a targeted gene panel analysis for primary immunodeficiency disease and/or inflammatory bowel disease. The patient proved to have a compound heterozygous mutation of the HPS1 gene resulting in Hermansky-Pudlak syndrome type 1.

Conclusions: We consider this report to be the first account of type 1 Hermansky-Pudlak syndrome with infantile-onset of inflammatory bowel disease. Early administration of infliximab was effective. We recommend next-generation sequencing for patients with very early-onset inflammatory bowel disease suspected to be monogenic.
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http://dx.doi.org/10.1186/s12876-019-0929-9DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6329123PMC
January 2019

Resting energy expenditure prediction using bioelectrical impedance analysis in patients with severe motor and intellectual disabilities.

Brain Dev 2019 Apr 28;41(4):352-358. Epub 2018 Nov 28.

Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, Japan.

Introduction: Resting energy expenditure (REE) is expected to be lower in with severe motor and intellectual disabilities (SMID) patients than in healthy subjects because of their relatively low fat-free mass (FFM). Therefore, an REE predictive equation for SMID patients may be required. The aim of this study was to validate existing REE predictive weight-based equations (Harris-Benedict, WHO, Mifflin, Owen, Schofield) and FFM-based REE equations (Mifflin, Owen and Cunningham) and to develop a new SMID patient-specific FFM-based REE equation.

Methods: Twenty-eight (22 males, 6 females) SMID patients over 18 years of age were included. The REE was measured using indirect calorimetry. FFM were measured using bioelectrical impedance analysis. A multiple linear regression analysis was used to develop a new FFM-based REE predictive equation. The accurate predictions compared the measured REE and root mean square error.

Results: The median measured REE was 950 (25th,75th percentile:712.75, 1102.75) kcal/day. The new FFM-based equation was as follows: REE (kcal/day) = 550.62 + 16.62 FFM (kg). The new FFM-based REE resulted in the highest percentage of accurate predictions within 10% of measured REE (42.9%). The root mean square errors were the smallest for the new FFM-based REE and largest for Harris-Benedict (91.00 and 185.22 kcal/day).

Conclusion: For SMID patients, the REE cannot accurately be predicted using the existing weight-based REE equations. Furthermore, the existing FFM-based REE equations are less accurate with regard to the measured REE than the new FFM-based REE equation. The new FFM-based equation is advised for use in SMID patients.
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http://dx.doi.org/10.1016/j.braindev.2018.11.003DOI Listing
April 2019