Brain & Development最新文献

{"title":"Polysomnographic and clinical features of childhood non-REM parasomnias: A sleep center experience","authors":"Aslı Çıplaklıgil ,&nbsp;Duygu Kurt Gök ,&nbsp;Hüseyin Per ,&nbsp;Sevda İsmailoğulları","doi":"10.1016/j.braindev.2025.104465","DOIUrl":"10.1016/j.braindev.2025.104465","url":null,"abstract":"<div><h3>Introduction</h3><div>Non-REM parasomnias are common in childhood, yet studies investigating their clinical and polysomnographic features remain limited. This study aimed to assess the clinical and sleep characteristics of children diagnosed with non-rapid eye movement (non-REM) parasomnias and to describe the semiological features of episodes confirmed by video-polysomnography (vPSG).</div></div><div><h3>Method</h3><div>We retrospectively evaluated 88 children (2–18 years) referred with abnormal nocturnal motor behaviors who underwent overnight vPSG between 2016 and 2023. Demographic and clinical data, comorbidities, and electroencephalography (EEG) findings were recorded. In cases with observed parasomnia episodes on vPSG, motor patterns, EEG, electrocardiography (ECG) characteristics, and Frontal Lobe Epilepsy-Parasomnia (FLEP) scale scores were analyzed. Parasomnia episodes were classified based on motor complexity: simple, rising, and complex arousal movements.</div></div><div><h3>Results</h3><div>Sixty-nine (78.4 %) patients referred for abnormal nocturnal behaviors were diagnosed with non-REM parasomnias by International Classification of Sleep Disorders-3 (ICSD-3), 20 (29 %) had confirmed episodes during vPSG (15 confusional arousal, 4 sleep terror, 1 somnambulism). Most episodes (91.1 %) occurred during N3 sleep. Simple arousal was the most common pattern (88.2 %). Episodes with complex motor features were longer and associated with autonomic signs such as tachycardia. Patients with comorbid epilepsy had a lower vPSG-confirmation rate (5.2 %) and higher FLEP scores. Electroencephalogram patterns included hypersynchronous delta and mixed-frequency theta-alpha activity.</div></div><div><h3>Conclusion</h3><div>This study characterized the semiological features of non-REM parasomnia episodes in pediatric patients. Although vPSG findings contributed to the objective description of motor patterns, the current data are not sufficient to conclude that vPSG is definitively useful for differential diagnosis. However, vPSG may aid in clarifying symptom profiles and excluding other comorbid conditions.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 6","pages":"Article 104465"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145314267","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Comment on “Survival motor neuron protein is the optimal biomarker for evaluating the risdiplam treatment”","authors":"Gül Yücel","doi":"10.1016/j.braindev.2025.104486","DOIUrl":"10.1016/j.braindev.2025.104486","url":null,"abstract":"","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 6","pages":"Article 104486"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145520346","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Reply to: “When neutral isn't negative: missteps in survey data analysis”","authors":"Tetsuya Okazaki ,&nbsp;Chisako Aoki ,&nbsp;Saki Shinzato ,&nbsp;Kaori Adachi ,&nbsp;Eiji Nanba","doi":"10.1016/j.braindev.2025.104481","DOIUrl":"10.1016/j.braindev.2025.104481","url":null,"abstract":"","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 6","pages":"Article 104481"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145417924","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Effects of therapeutic instrumental music performance on upper limb motor function of children with cerebral palsy: A systematic review","authors":"Zhuolin Wu ,&nbsp;Meijiao Wu","doi":"10.1016/j.braindev.2025.104468","DOIUrl":"10.1016/j.braindev.2025.104468","url":null,"abstract":"<div><div>Therapeutic instrumental music performance (TIMP) can be used to improve limb motor function in children with cerebral palsy (CwCP). The purpose of this systematic review is to further analyze the potential therapeutic effects of TIMP on upper limb motor function in CwCP. We used the terms \"“music,\"” \"“therapeutic instrumental music performance,\"” \"“musical instruments,\"” \"“cerebral palsy,\"” \"“children,\"” \"“upper limb,\"” and \"“upper extremity\"” as our search keywords, and conducted a search of articles published in English and Chinese databases: PubMed, Cochrane, Web of Science, Wiley Online Library, CINAHL, and CNKI, Wanfang Data as of March 2025. Initially, 409 articles were retrieved. After screening titles and abstracts and assessing eligibility, eight studies were included in this review. All studies assessed hand fine motor function, and five also evaluated upper limb gross motor function. Three studies used only keyboard instruments, two exclusively percussion instruments, and three combined keyboard, percussion, and plucked instruments. The results demonstrated that TIMP effectively improves upper limb motor function in CwCP.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 6","pages":"Article 104468"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145294472","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Response to response to the letter to the editor “Decoding the pathophysiological role of Fukutin in Fukuyama congenital muscular dystrophy”","authors":"Christian Messina","doi":"10.1016/j.braindev.2025.104473","DOIUrl":"10.1016/j.braindev.2025.104473","url":null,"abstract":"","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 6","pages":"Article 104473"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145314302","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Refractory myoclonic epilepsy and progressive movement disorder arising from recurrent DHDDS variants in Japanese patients: a case series","authors":"Yu Kobayashi ,&nbsp;Satoru Sakuma ,&nbsp;Emiko Morimoto ,&nbsp;Hitomi Fujii ,&nbsp;Kei Yamada ,&nbsp;Moemi Hojo ,&nbsp;Masaki Miura ,&nbsp;Jun Tohyama ,&nbsp;Fuyuki Miya ,&nbsp;Mitsuhiro Kato ,&nbsp;Hirotomo Saitsu ,&nbsp;Naomichi Matsumoto","doi":"10.1016/j.braindev.2025.104478","DOIUrl":"10.1016/j.braindev.2025.104478","url":null,"abstract":"<div><h3>Objective</h3><div>Heterozygous <em>DHDDS</em> variants have been associated with intellectual disability and seizures, with or without movement disorders. We aimed to expand the known phenotypic spectrum of <em>DHDDS</em>-related disorders by elucidating the clinical characteristics of Japanese patients with recurrent <em>DHDDS</em> variants.</div></div><div><h3>Methods</h3><div>Patients with pathogenic <em>DHDDS</em> variants were recruited from individuals diagnosed with developmental and epileptic encephalopathy or progressive myoclonus epilepsies who were treated at the Department of Child Neurology, National Hospital Organization Nishiniigata Chuo Hospital, or the Department of Pediatrics, Osaka Metropolitan University Graduate School of Medicine. <em>DHDDS</em> pathogenic variants were identified using exome sequencing. Medical records and neurophysiological data were retrospectively reviewed.</div></div><div><h3>Results</h3><div>Three <em>de novo</em> recurrent pathogenic <em>DHDDS</em> variants were identified in four patients: c.632G&gt;A, p.(Arg211Gln) in two; c.614G&gt;A, p.(Arg205Gln) in one; and c.110G&gt;A, p.(Arg37His) in one. Three patients with p.(Arg211Gln) or p.(Arg37His) presented with severe intellectual disability and intractable generalized epilepsy, characterized by myoclonic, myoclonic–atonic, and atonic seizures. Eyelid myoclonia was the initial manifestation in infancy. One patient with p.(Arg205Gln) had a relatively mild course, with focal epilepsy and less severe intellectual disability. Myoclonus and tremor were present from infancy in two patients with p.(Arg211Gln) or p.(Arg37His), and from childhood in the patient with p.(Arg205Gln). One patient with p.(Arg211Gln) exhibited polymicrogyria in the right frontal lobe. Among the three patients with generalized epilepsy, fenfluramine effectively reduced myoclonic seizures in one, while total corpus callosotomy markedly reduced myoclonic–atonic or atonic seizures with falls in another.</div></div><div><h3>Conclusion</h3><div><em>DHDDS</em>-related disorders should be considered in patients with intellectual disability, epilepsy with early-onset eyelid myoclonia and myoclonic or myoclonic–atonic seizures, and movement disorders, including myoclonus and tremor, with slow progression. We identified polymicrogyria as a novel finding in <em>DHDDS</em> patients. For drug-resistant epilepsy in these patients, corpus callosotomy and fenfluramine may represent promising treatment options, with this report providing specific insights into their efficacy.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 6","pages":"Article 104478"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145326434","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Clinical profiles of tuberous sclerosis complex: A regionally based survey","authors":"Misae Yamada ,&nbsp;Jun Natsume ,&nbsp;Yuki Maki ,&nbsp;Soichiro Ishimaru ,&nbsp;Shingo Numoto ,&nbsp;Satoru Kobayashi ,&nbsp;Ayako Hattori ,&nbsp;Yoshihisa Matsukawa ,&nbsp;Keiko Wakahara ,&nbsp;Naoko Ishihara ,&nbsp;Hitomi Sasaki ,&nbsp;Yuji Ito ,&nbsp;Hiroyuki Yamamoto ,&nbsp;Tomohiko Nakata ,&nbsp;Hiroyuki Kidokoro ,&nbsp;Tetsushi Yoshikawa ,&nbsp;Shinji Saitoh ,&nbsp;Akihisa Okumura ,&nbsp;Yoshiyuki Takahashi","doi":"10.1016/j.braindev.2025.104479","DOIUrl":"10.1016/j.braindev.2025.104479","url":null,"abstract":"<div><h3>Background</h3><div>This study aimed to clarify the clinical profiles of patients with tuberous sclerosis complex (TSC) in the general population by a regionally based survey of medical facilities in a region with 7.5 million residents, and investigate differences in clinical profiles according to medical facility size and type.</div></div><div><h3>Methods</h3><div>A survey was sent to 146 hospitals and clinics regarding clinical profiles of patients with TSC who lived in Aichi Prefecture, Japan, between 2013 and 2018. Medical facilities were classified as large hospitals (≥750 beds or a children's hospital), small hospitals (&lt;750 beds), institutions for handicapped children, and private clinics.</div></div><div><h3>Results</h3><div>Information was obtained of 232 patients (median age, 25 years; range, 1–81 years). Estimated prevalence of TSC was 3.1 per 100,000. Cortical tubers were present in 88 %, epilepsy in 81 %, autism spectrum disorder in 44 %, and subependymal giant cell astrocytoma in 17 %. Hypomelanotic macules, facial angiofibroma, renal angiomyolipoma, and cardiac rhabdomyoma were present in &gt;50 % of patients. Rates of epilepsy with frequent seizures and autism spectrum disorder were both higher in patients in institutions for handicapped children. In more than half of patients in institutions for handicapped children information on cranial MRI findings was not obtained.</div></div><div><h3>Conclusions</h3><div>Our regionally based study confirmed the clinical profiles previously reported in specialized hospitals for TSC and found that clinical characteristics differed among the types and sizes of medical facilities. Multicenter information sharing and collaboration between general hospitals and institutions for handicapped children are important for the comprehensive care of patients with TSC.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 6","pages":"Article 104479"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145364704","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"The effect of robotic assisted gait training on physical activity, motor function, and quality of life in children with spastic cerebral palsy: Exploratory pilot study","authors":"Yeon-Gyu Jeong ,&nbsp;Won-Cheol Kim ,&nbsp;Yeon-Jae Jeong ,&nbsp;Ha-Neul Jang ,&nbsp;Joo-Young Lee ,&nbsp;Jae-Soon Chung ,&nbsp;Kyu-Hoon Lee","doi":"10.1016/j.braindev.2025.104482","DOIUrl":"10.1016/j.braindev.2025.104482","url":null,"abstract":"<div><h3>Background</h3><div>Children with spastic cerebral palsy (CP) experience motor impairments and reduced physical activity, negatively impacting health and quality of life. Objective: The study aimed to assess the effects of wearable exoskeleton robot-assisted gait training on physical activity, motor function, and quality of life in children with CP. Methods: Ten children with spastic CP (mean age 9.20 ± 2.57 years; gross motor function classification system levels I–IV) received twelve 30-min sessions of robot-assisted gait training over six weeks at a university hospital rehabilitation center. Physical activity was measured using a tri-axial accelerometer to assess energy expenditure, metabolic equivalents (METs), intensity levels, vector magnitude counts per minute (VM CPM), and step counts. Motor function was evaluated using the Gross Motor Function Measure (GMFM), Timed Up and Go Test (TUG), and 6-Minute Walk Test (6MWT). Quality of life was assessed with the Cerebral Palsy Quality of Life questionnaire (CP-QOL). Repeated measures MANOVA and Cohen's d were used for statistical analysis. Results: Significant improvements were observed in METs (<em>p</em> = 0.02, d = 0.38), light (<em>p</em> = 0.03, d = 0.51) and moderate physical activity time (<em>p</em> = 0.01, d = 0.42), and VM CPM (<em>p</em> = 0.02, d = 0.55), along with reduced sedentary time (p = 0.02, d = −0.49). Functional outcomes improved in GMFM (<em>p</em>&lt;0.01, d = 0.22), TUG (<em>p</em> = 0.03, d = −0.33), and 6MWT (p = 0.02, d = 0.52). No significant changes were found in CP-QOL scores. Conclusion: Wearable robot-assisted gait training appears to enhance physical activity and mobility in children with spastic CP and may be considered a promising therapeutic intervention.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 6","pages":"Article 104482"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145467259","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Diagnostic odyssey of opsoclonus-myoclonus syndrome and barriers to early detection","authors":"Nobuaki Tsuiki ,&nbsp;Itaru Hayakawa ,&nbsp;Yuichi Abe","doi":"10.1016/j.braindev.2025.104484","DOIUrl":"10.1016/j.braindev.2025.104484","url":null,"abstract":"<div><h3>Objectives</h3><div>Opsoclonus-myoclonus syndrome (OMS) is a rare immune-mediated disorder of central nervous systems characterized by chaotic eye movements (opsoclonus), myoclonus, ataxia, and behavioral disturbances. OMS remains diagnostically challenging despite its recognizable clinical features.</div></div><div><h3>Methods</h3><div>We conducted a single-center cohort study of patients with OMS at a tertiary care children's medical center (2002–2024). Using the diagnostic odyssey plot methodology, we mapped individual diagnostic pathways, compared early (≤28 days) versus delayed (&gt;28 days) diagnosis groups, and analyzed symptoms as well as patterns of diagnostic errors.</div></div><div><h3>Results</h3><div>Twenty cases were ascertained. Ten patients were diagnosed early (median 16.5 days), while the remaining experienced a delayed diagnosis (equal to or more than 28 days, median 124 days). Provisional diagnoses of acute cerebellar ataxia, acute disseminated encephalomyelitis, and acute cerebellitis were common. Analysis of symptoms revealed that nystagmus appeared at the same time in both the early- and delayed-diagnosed groups, but opsoclonus was recognized later in the delayed diagnosed group (11.5 days versus 96 days, <em>p</em> &lt; 0.01). The presence or absence of neuroblastoma did not contribute to the diagnostic delay.</div></div><div><h3>Conclusions</h3><div>The unique diagnostic journey of OMS is presented. Reconsidering the diagnosis in cases of prolonged or relapsing ataxia and recognizing opsoclonus early on are vital for the early detection of OMS.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 6","pages":"Article 104484"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145520433","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Neurological manifestations and clinical outcomes in pediatric Alexander disease: single-center cohort and identification of novel GFAP variants","authors":"Renu Suthar ,&nbsp;Yashu Sharma ,&nbsp;Arushi Gahlot Saini ,&nbsp;Pawan Kumar ,&nbsp;Sadhna Lal ,&nbsp;Prateek Bhatia ,&nbsp;Vikas Bhatia ,&nbsp;Sameer Vyas ,&nbsp;Priyanka Srivastava ,&nbsp;Balamurugan Nagarajan ,&nbsp;Savita Attri ,&nbsp;Jitendra Sahu ,&nbsp;Naveen Sankhyan","doi":"10.1016/j.braindev.2025.104488","DOIUrl":"10.1016/j.braindev.2025.104488","url":null,"abstract":"<div><h3>Background</h3><div>We aimed to analyze the prevalence, clinico-radiological and genetic features, and outcomes in children with Alexander disease (AD) with special emphasis on atypical presentations.</div></div><div><h3>Methods</h3><div>This cross-sectional study evaluated children with AD seen over the past 10 years. Neuroimaging was evaluated by a trained neuroradiologist. <em>In-silico</em> tools were used to predict the pathogenicity of the novel variants detected by whole-exome sequencing.</div></div><div><h3>Results</h3><div><em>N</em> = 12 children (males 58.3 %) with genetically-confirmed AD were evaluated. Disease subtypes were infantile (<em>n</em> = 7, 58.3 %), juvenile (<em>n</em> = 3, 25 %), and neonatal (<em>n</em> = 2, 16.7 %) AD. The clinic-based prevalence of AD was 0.34 cases per 1000 pediatric neurology patients per year, or an average of 1.2 cases per year. Clinical features were developmental delay (<em>n</em> = 9, 75 %), macrocephaly (n = 9, 75 %), spasticity (<em>n</em> = 6, 50 %), and epilepsy (<em>n</em> = 8, 66.7 %); two children with juvenile-onset disease had atypical visual and bulbar manifestations. Pathogenic variations were most common in exons 1 (<em>n</em> = 5, 42 %), and exon 4 (<em>n</em> = 4, 33.3 %). These were missense type (<em>n</em> = 11, 91.6 %) or deletion (n = 1, 8.3 %). Three novel variants were detected: c.251T&gt;G (p.Ile84Ser) in exon 1, c.810_818 deletion (p.Asn271_Glu273del) in exon 5, and c.292G&gt;C (p.Ala98Pro) in exon 1 in the <em>GFAP</em> gene (NM_002055). Majority of children developed spastic paresis (<em>n</em> = 9, 83 %) and mortality rate was 33.3 % (<em>n</em> = 4/12).</div></div><div><h3>Conclusion</h3><div>AD is a rare leukodystrophy with high mortality and progressive spastic paraparesis. Neonatal and juvenile types may present atypically and delay correct diagnosis. Deletions may account for a minor proportion of pathogenic variants. Our study expands the clinico-radiological spectrum of AD in children.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 6","pages":"Article 104488"},"PeriodicalIF":1.3,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145617802","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}