Insights on chronic-relapsing opsoclonus-myoclonus from a pilot study of mycophenolate mofetil.

Abstract:

:Opsoclonus-myoclonus syndrome is characterized by abnormal lymphocyte trafficking into brain. The authors hypothesized that mycophenolate mofetil, a lymphocyte proliferation inhibitor, might be therapeutic. The cerebrospinal fluid and blood immunophenotypes of 15 children with predominantly chronic-relapsing opsoclonus-myoclonus syndrome were compared before and after treatment by flow cytometry. Mycophenolate mofetil reduced the cerebrospinal fluid expansion of HLA-DR+ activated T cells (-40%); the frequency of other T-cell or natural killer cell subsets remained unchanged, but cerebrospinal fluid B cells increased significantly. Adrenocorticotropic hormone dose was lowered by 64% over an average of 1.5 years, yet 73% eventually relapsed despite therapeutic drug levels. Prior treatment with rituximab prevented relapse-associated increase in cerebrospinal fluid B cells, without hindering mycophenolate mofetil-induced reduction in T-cell activation. These data demonstrate resistant immunologic problems in chronic-relapsing opsoclonus-myoclonus syndrome. Mycophenolate mofetil did not prevent relapse. The novel effect of mycophenolate mofetil on chronically activated T cells may contribute to its efficacy in T-cell mediated neurological disorders.

journal_name

J Child Neurol

authors

Pranzatelli MR,Tate ED,Travelstead AL,Baumgardner CA,Gowda NV,Halthore SN,Kerstan P,Kossak BD,Mitchell WG,Taub JW

doi

10.1177/0883073808324217

subject

Has Abstract

pub_date

2009-03-01 00:00:00

pages

316-22

issue

3

eissn

0883-0738

issn

1708-8283

pii

24/3/316

journal_volume

24

pub_type

杂志文章
  • Manic-depressive illness in children: treatment with lithium carbonate.

    abstract::A behavior questionnaire was used retrospectively in 21 manic-depressive children to quantitate manic-depressive behaviors before and after treatment with lithium carbonate. The study children were matched with 21 control children for age, race, sex, and socioeconomic status. The study children had significantly more ...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/088307388600100409

    authors: Younes RP,DeLong GR,Neiman G,Rosner B

    更新日期:1986-10-01 00:00:00

  • Steroid-responsive chronic cerebellitis with positive glutamate receptor delta 2 antibody.

    abstract::We report the clinical course of a 4-year-old girl with chronic cerebellitis (onset 2 days after diphtheria-pertussis-tetanus vaccination at 1 year and 7 months old) associated with anti-glutamate receptor delta 2 antibody, who improved dramatically with steroid therapy (methylprednisolone pulse therapy plus oral pred...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073807307973

    authors: Kubota M,Takahashi Y

    更新日期:2008-02-01 00:00:00

  • Automated tandem mass spectrometry for mass newborn screening for disorders in fatty acid, organic acid, and amino acid metabolism.

    abstract::Development of acylcarnitine and amino acid profiling using tandem mass spectrometry, and its application for use with dried blood specimens collected on filter-paper cards, has introduced an innovative new technology for detecting inborn errors of fatty acid, organic acid, and amino acid metabolism. From November 1, ...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073899014001021

    authors: Naylor EW,Chace DH

    更新日期:1999-11-01 00:00:00

  • Corpus Callosotomy for Intractable Epilepsy Revisited: The Children's Hospital of Michigan Series.

    abstract::Corpus callosotomy is a palliative procedure performed to reduce the severity of drug-resistant epilepsy. The authors assessed its efficacy on different seizure types in 20 subjects (age range 5-19 years); 8 with active vagus nerve stimulator. Fifteen had complete callosotomy, 3 had anterior 2/3, and 2 had anterior 2/...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073817697847

    authors: Luat AF,Asano E,Kumar A,Chugani HT,Sood S

    更新日期:2017-06-01 00:00:00

  • Children who can't smell the coffee: isolated congenital anosmia.

    abstract::Two children with isolated congenital anosmia, a rare syndrome of deficient restricted neuronal migration, are presented with early diagnosis confirmed by standardized smell testing and detailed neuroimaging studies. Recognition of this disorder and its spectrum of presentations provides important insights into the mo...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/088307389801300404

    authors: Assouline S,Shevell MI,Zatorre RJ,Jones-Gotman M,Schloss MD,Oudjhane K

    更新日期:1998-04-01 00:00:00

  • Unilateral schizencephaly and contralateral polymicrogyria associated with umbilical cord mass.

    abstract::We report a 6-month-old boy with diffuse hypertonia and developmental delay who had unilateral separated-lip schizencephaly and contralateral polymicrogyria. The contralateral polymicrogyria was associated with an incomplete clefting in that hemisphere. An umbilical cord hamartoma is presumed to have caused hypoperfus...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/08830738030180031401

    authors: Hahn JS,Lewis AJ

    更新日期:2003-03-01 00:00:00

  • Association of adenosine deaminase polymorphism with mild mental retardation.

    abstract::The etiology of mild mental retardation remains undefined in about 60% of cases. Even though the causes of mild mental retardation are likely to be heterogeneous, the evidence for genetic involvement is increasing, along with the development of specific diagnostic techniques. To improve our understanding of the geneti...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/08830738060210091201

    authors: Saccucci P,Arpino C,Rizzo R,Gagliano A,Volzone A,Lalli C,Galasso C,Curatolo P

    更新日期:2006-09-01 00:00:00

  • Autonomic dysfunction in childhood Guillain-Barré syndrome.

    abstract::This investigation correlated incidence and degree of autonomic dysfunction with the degree of motor impairment in children hospitalized with Guillain-Barré syndrome. Motor weakness varies, as does the effect on autonomic function including heart rate, vasomotor stability, sweating, continence, and blood pressure. Aft...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073811420872

    authors: Dimario FJ Jr,Edwards C

    更新日期:2012-05-01 00:00:00

  • Primary antiphospholipid syndrome presenting with a branch retinal artery occlusion in a 15-year-old boy.

    abstract::Acute vascular events are rare in the pediatric population, but there is an association with the presence of antiphospholipid antibodies. When there is no other underlying medical disorder, this is referred to as primary antiphospholipid syndrome. We present a case of a 15-year-old boy who developed an acute superior ...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/088307380201700517

    authors: Saxonhouse MA,Bhatti MT,Driebe WT Jr,Freeman BE,Maria BL,Carney PR

    更新日期:2002-05-01 00:00:00

  • Wilms' tumor in a case with Möbius' syndrome associated with arthrogryposis and mega cisterna magna.

    abstract::Möbius' syndrome is a rare congenital anomaly characterized by paralysis of the 7th and other cranial nerves and musculoskeletal abnormalities. We report a patient with Möbius' syndrome associated with arthrogryposis and mega cisterna magna in addition to the classic components of this syndrome. The case is interestin...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/08830738040190010710

    authors: Yaris N,Aynaci FM,Kalyoncu M,Odemiş E,Okten A

    更新日期:2004-01-01 00:00:00

  • Neurobiology of specific language impairment.

    abstract::This review summarizes what is known about the neurobiology of specific language impairment. Despite its name, specific language impairment is frequently not specific. It is common to find associated impairments in motor skills, cognitive function, attention, and reading in children who meet criteria for specific lang...

    journal_title:Journal of child neurology

    pub_type: 杂志文章,评审

    doi:10.1177/08830738040190070101

    authors: Webster RI,Shevell MI

    更新日期:2004-07-01 00:00:00

  • Cognitive profile of Rett syndrome.

    abstract::We report preliminary studies in 18 girls with Rett syndrome (15 typical, three atypical cases) who were studied using a number of neuropsychologic measures. Results indicate a relative preservation of gross motor and daily living skills at the developmental level of the age of onset of the condition. Other adaptive f...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073888003001s05

    authors: Fontanesi J,Haas RH

    更新日期:1988-01-01 00:00:00

  • Attentional disturbance after pediatric closed head injury.

    abstract::The influence of severity of closed head injury and age on attentional functioning was prospectively investigated in 36 children (age range, 7 to 16 years) 6 months after injury. Children were placed into mild, moderate, and severe injury groups using established neurologic criteria. Each child received the Wechsler I...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/088307389300800410

    authors: Kaufmann PM,Fletcher JM,Levin HS,Miner ME,Ewing-Cobbs L

    更新日期:1993-10-01 00:00:00

  • Sydenham's chorea: a clinical follow-up of 65 patients.

    abstract::Sydenham's chorea, the neurological manifestation of rheumatic fever, is the most common acquired chorea of childhood. In this retrospective study, the authors aim to present the clinical and laboratory findings of 65 Sydenham's chorea patients, followed up in a clinic over less than 7 years. The mean age at the onset...

    journal_title:Journal of child neurology

    pub_type: 临床试验,杂志文章

    doi:10.1177/0883073807302614

    authors: Demiroren K,Yavuz H,Cam L,Oran B,Karaaslan S,Demiroren S

    更新日期:2007-05-01 00:00:00

  • Spongy degeneration of the central nervous system (Van Bogaert-Bertrand disease): report of a case in a Saudi family.

    abstract::Spongy degeneration of the central nervous system in infancy (Van Bogaert-Bertrand disease) is a disorder usually thought to occur in Ashkenazi Jewish infants. We report a case occurring in an Arab infant of consanguineous parents. ...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/088307388600100111

    authors: Mahdi AH,Elidirssy AT,Wright EA

    更新日期:1986-01-01 00:00:00

  • Head Impact Exposure During a Weekend Youth Soccer Tournament.

    abstract::Concussion is a known risk in youth soccer, but little is known about subconcussive head impacts. The authors provided a prospective cohort study measuring frequency and magnitude of subconcussive head impacts using accelerometry in a middle school-age soccer tournament, and association between head impacts and change...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073816634857

    authors: Chrisman SP,Mac Donald CL,Friedman S,Andre J,Rowhani-Rahbar A,Drescher S,Stein E,Holm M,Evans N,Poliakov AV,Ching RP,Schwien CC,Vavilala MS,Rivara FP

    更新日期:2016-07-01 00:00:00

  • Electroencephalogram discharges in atypical cognitive development.

    abstract::To investigate the significance of electroencephalogram (EEG) discharges and their treatment, we retrospectively reviewed the charts of 22 children with atypical cognitive development that did not respond to standard educational therapy and demonstrated discharges on EEG. Most children demonstrated no obvious symptoms...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073809344743

    authors: Frye RE,Butler I,Strickland D,Castillo E,Papanicolaou A

    更新日期:2010-05-01 00:00:00

  • Atypical teratoid/rhabdoid tumor of the spine in a 4-year-old girl.

    abstract::Primary spinal atypical teratoid/rhabdoid tumor is extremely rare. The authors present a case of atypical teratoid/rhabdoid tumor occurring in a 4-year-old girl. Magnetic resonance imaging The authors showed an intramedullary mass extending from the bulbomedullary junction to T1 with leptomeningeal dissemination. The ...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073808319319

    authors: Tinsa F,Jallouli M,Douira W,Boubaker A,Kchir N,Hassine DB,Boussetta K,Bousnina S

    更新日期:2008-12-01 00:00:00

  • Head circumference, brain weight, and tumor burden.

    abstract::The failure to diagnose an ependymoma at an appropriately early age led to an incomplete excision and a tumor burden too great for radiotherapy to control. The development of normative curves for brain weight correlated with head circumference allowed for the estimates of the extremes of possible growth rates of the t...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/088307388600100311

    authors: Alvord EC Jr

    更新日期:1986-07-01 00:00:00

  • What is attention-deficit hyperactivity disorder (ADHD)?

    abstract::Attention-deficit hyperactivity disorder (ADHD) is described as the most common neurobehavioral condition of childhood. We raise the concern that ADHD is not a disease per se but rather a group of symptoms representing a final common behavioral pathway for a gamut of emotional, psychological, and/or learning problems....

    journal_title:Journal of child neurology

    pub_type: 杂志文章,评审

    doi:10.1177/08830738050200121301

    authors: Furman L

    更新日期:2005-12-01 00:00:00

  • Applying the International Classification of Functioning-Children and Youth Version to Pediatric Neuro-oncology.

    abstract::Given the increased survival rates in patients with pediatric central nervous system tumors, late effects such as treatment- and/or illness-related neurologic sequelae as well as neuropsychological deficits and social difficulties have moved into focus in follow-up care. In order to provide personalized treatment reco...

    journal_title:Journal of child neurology

    pub_type: 杂志文章,评审

    doi:10.1177/0883073816669647

    authors: Pletschko T,Felnhofer A,Schwarzinger A,Weiler L,Slavc I,Leiss U

    更新日期:2017-01-01 00:00:00

  • Gingival enlargement in children treated with antiepileptics.

    abstract::This study was conducted to determine the occurrence, severity, and risk factors of gingival enlargement in children treated with valproate and other nonvalproate antiepileptic drugs. A cross-sectional study was carried out in which data obtained from 68 epileptic children under treatment were compared with those from...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/08830738040190120901

    authors: Tan H,Gürbüz T,Dağsuyu IM

    更新日期:2004-12-01 00:00:00

  • Schilder's disease: case study with serial neuroimaging.

    abstract::Schilder's myelinoclastic diffuse sclerosis is a rare sporadic demyelinating disease that usually affects children between 5 and 14 years old. The disease often mimics intracranial neoplasm or abscess. We report a 9-year-old girl with Schilder's disease who presented with left hemiparesis. Cranial computed tomography ...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/08830738030180011301

    authors: Kurul S,Cakmakçi H,Dirik E,Kovanlikaya A

    更新日期:2003-01-01 00:00:00

  • Ornithine transcarbamylase deficiency presenting with acute reversible cortical blindness.

    abstract::Acute focal neurologic deficits are a rare but known presentation of ornithine transcarbamylase deficiency, particularly in females. We describe here a 6-year-old girl with newly diagnosed ornithine transcarbamylase deficiency who presents with an episode of acute cortical blindness lasting for 72 hours in the absence...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073814535490

    authors: Prasun P,Altinok D,Misra VK

    更新日期:2015-05-01 00:00:00

  • Developmental and behavior problems predict parenting stress in young children with global delay.

    abstract::To identify parent-reported symptoms that predict parenting stress in preschoolers with global developmental delay, 201 parents/guardians of 142 boys and 59 girls with global delay, mean age 39.1 months (range, 18 to 63 months) were studied retrospectively. Parents completed the following: (a) a semistructured intervi...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073811418230

    authors: Tervo RC

    更新日期:2012-03-01 00:00:00

  • Nutritional vitamin D deficiency presenting as hemichorea.

    abstract::The authors describe a pediatric patient with repaired hypoplastic left heart syndrome developing protein-losing enteropathy, hypocalcemia, vitamin D deficiency, and hemichorea. After correction of nutritional vitamin D deficiency with calcium and vitamin D supplementation, the chorea resolved. Hypoalbuminemia also im...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073807299956

    authors: Fernandez R,Ashraf A,Dure LS

    更新日期:2007-01-01 00:00:00

  • Research in Rett syndrome: past, present, and future.

    abstract::Past, present, and future research activities in Rett syndrome are described. Studies to this point have failed to define a biologic or chemical marker. It is hoped that heightened activity in this unique syndrome will provide guidance for the diagnosis of this disorder, and with it, better understanding. ...

    journal_title:Journal of child neurology

    pub_type: 杂志文章,评审

    doi:10.1177/0883073888003001s14

    authors: Percy AK

    更新日期:1988-01-01 00:00:00

  • The multiple causes of multiple sclerosis: the importance of age of infections in childhood.

    abstract::The geographic distribution of multiple sclerosis (MS) may relate to the age of initial exposure and degree of sensitization to common viruses or bacteria which have proteins with epitopes (antigenic determinants) which are homologous with potentially encephalitogenic peptides in central myelin proteins, such as basic...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/088307388700200418

    authors: Alvord EC Jr,Jahnke U,Fischer EH,Kies MW,Driscoll BF,Compston DA

    更新日期:1987-10-01 00:00:00

  • Learning disabilities spectrum and sexual dimorphic abilities in girls with congenital adrenal hyperplasia.

    abstract::Congenital adrenal hyperplasia could provide a promising model for the study of the effects of hormones on cognition. The aim of this study was to assess sexual dimorphic abilities related to oral language, spatial abilities, and verbal fluency and to determine the existence of learning disabilities in 11 congenital g...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073808315618

    authors: Inozemtseva O,Matute E,Juárez J

    更新日期:2008-08-01 00:00:00

  • Macrodactyly and fibrous hamartoma in a child with tuberous sclerosis complex.

    abstract::A 10-year-old boy with tuberous sclerosis complex and macrodactyly of the right index and middle fingers is described. He also had a fibrous hamartoma at the front of the right wrist. The association of fibrous hamartoma and macrodactyly has not been previously described in tuberous sclerosis complex. ...

    journal_title:Journal of child neurology

    pub_type: 杂志文章

    doi:10.1177/0883073810375616

    authors: Sharma S,Sankhyan N,Gulati S,Kumar A,Srinivas M,Shukla B,Mathur SR

    更新日期:2011-01-01 00:00:00