[ { "key": "B27BRDE5", "version": 19, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/B27BRDE5", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/B27BRDE5", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/M95Q3Q6Z", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "numChildren": 0 }, "data": { "key": "B27BRDE5", "version": 19, "parentItem": "M95Q3Q6Z", "itemType": "attachment", "linkMode": "imported_file", "title": "xyrem study 2005.pdf", "accessDate": "", "url": "", "note": "", "contentType": "application/pdf", "charset": "", "filename": "xyrem study 2005.pdf", "md5": "6fbf30465ead446637304f7f56c8001e", "mtime": 1418938325000, "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:11:37Z", "dateModified": "2013-11-20T23:11:37Z" } }, { "key": "WN5MDQS2", "version": 17, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/WN5MDQS2", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/WN5MDQS2", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/M95Q3Q6Z", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } } }, "data": { "key": "WN5MDQS2", "version": 17, "parentItem": "M95Q3Q6Z", "itemType": "attachment", "linkMode": "linked_url", "title": "PubMed entry", "accessDate": "2013-11-20T23:11:25Z", "url": "http://www.ncbi.nlm.nih.gov/pubmed/16099718", "note": "", "contentType": "text/html", "charset": "", "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:11:25Z", "dateModified": "2013-11-20T23:11:25Z" } }, { "key": "M95Q3Q6Z", "version": 17, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/M95Q3Q6Z", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/M95Q3Q6Z", "type": "text/html" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "creatorSummary": "Xyrem International Study Group", "parsedDate": "2005-09", "numChildren": 2 }, "data": { "key": "M95Q3Q6Z", "version": 17, "itemType": "journalArticle", "title": "Further evidence supporting the use of sodium oxybate for the treatment of cataplexy: a double-blind, placebo-controlled study in 228 patients", "creators": [ { "creatorType": "author", "name": "Xyrem International Study Group" } ], "abstractNote": "BACKGROUND AND PURPOSE: To measure the effect of the nocturnal administration of sodium oxybate on cataplexy in patients with narcolepsy.\nPATIENTS AND METHODS: This trial was conducted with 228 adult narcolepsy/cataplexy patients in 42 sleep clinics. Patients using anticataplectic medications were weaned from these medications, then randomized to receive 4.5, 6 or 9 g sodium oxybate nightly or placebo for 8 weeks. Patients receiving 6 and 9 g doses were titrated to their final dose in weekly 1.5 g increments. Placebo patients underwent a randomized mock dose-titration schedule. The effect of sodium oxybate on weekly cataplexy attacks was measured using patient daily diaries.\nRESULTS: Compared to placebo, nightly doses of 4.5, 6 and 9 g sodium oxybate for 8 weeks resulted in statistically significant median decreases in weekly cataplexy attacks of 57.0, 65.0 and 84.7%, respectively. The decrease in cataplexy at the 4.5 g dose represents a novel finding. The weekly increase in sodium oxybate dose was associated with fewer adverse events than previously reported in double-blind sodium oxybate trials using fixed doses. Some adverse events reported demonstrated a clear dose-response relationship.\nCONCLUSIONS: In the largest study of its kind, sodium oxybate was highly effective for the treatment of cataplexy. The improvements in cataplexy are dependent on the dosage of sodium oxybate as well on the duration of treatment. Weekly dose titration appears to be well-tolerated.", "publicationTitle": "Sleep medicine", "publisher": "", "place": "", "date": "Sep 2005", "volume": "6", "issue": "5", "section": "", "partNumber": "", "partTitle": "", "pages": "415-421", "series": "", "seriesTitle": "", "seriesText": "", "journalAbbreviation": "Sleep Med.", "DOI": "10.1016/j.sleep.2005.03.010", "citationKey": "", "url": "", "accessDate": "", "PMID": "", "PMCID": "", "ISSN": "1389-9457", "archive": "", "archiveLocation": "", "shortTitle": "Further evidence supporting the use of sodium oxybate for the treatment of cataplexy", "language": "eng", "libraryCatalog": "NCBI PubMed", "callNumber": "", "rights": "", "extra": "PMID: 16099718", "tags": [ { "tag": "Adolescent", "type": 1 }, { "tag": "Adult", "type": 1 }, { "tag": "Aged", "type": 1 }, { "tag": "Cataplexy", "type": 1 }, { "tag": "Central Nervous System Agents", "type": 1 }, { "tag": "Dose-Response Relationship, Drug", "type": 1 }, { "tag": "Double-Blind Method", "type": 1 }, { "tag": "Drug Administration Schedule", "type": 1 }, { "tag": "Female", "type": 1 }, { "tag": "Humans", "type": 1 }, { "tag": "Male", "type": 1 }, { "tag": "Middle Aged", "type": 1 }, { "tag": "Sodium Oxybate", "type": 1 }, { "tag": "Treatment Outcome", "type": 1 } ], "collections": [], "relations": {}, "dateAdded": "2013-11-20T23:11:25Z", "dateModified": "2013-11-20T23:11:25Z" } }, { "key": "K9FG4I4Z", "version": 15, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/K9FG4I4Z", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/K9FG4I4Z", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/WRZWUVIS", "type": "application/json" } }, 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"9BAPZB6W", "version": 13, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/9BAPZB6W", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/9BAPZB6W", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/5NA2C7RU", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } } }, "data": { "key": "9BAPZB6W", "version": 13, "parentItem": "5NA2C7RU", "itemType": "attachment", "linkMode": "linked_url", "title": "PubMed entry", "accessDate": "2013-11-20T23:08:54Z", "url": "http://www.ncbi.nlm.nih.gov/pubmed/16542521", "note": "", "contentType": "text/html", "charset": "", "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:08:54Z", "dateModified": "2013-11-20T23:08:54Z" } }, { "key": "5NA2C7RU", "version": 13, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/5NA2C7RU", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/5NA2C7RU", "type": "text/html" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "creatorSummary": "Stores", "parsedDate": "2006-04", "numChildren": 2 }, "data": { "key": "5NA2C7RU", "version": 13, "itemType": "journalArticle", "title": "The protean manifestations of childhood narcolepsy and their misinterpretation", "creators": [ { "creatorType": "author", "firstName": "Gregory", "lastName": "Stores" } ], "abstractNote": "Narcolepsy often begins in childhood but is infrequently recognized, partly because of its many manifestations that can be confused with other conditions. The clinical presentations of excessive sleepiness, cataplexy, hallucinatory phenomena, and sleep paralysis (not always occurring together) are very varied. The picture may be further complicated by the occurrence of automatic behaviour, memory and visual problems, and associated sleep disorders, as well as the psychological and social consequences of having narcolepsy. Not surprisingly, therefore, misinterpretation of the symptoms as primarily psychological, or otherwise physical, may well occur leading to inappropriate management including initial referral to psychiatric or educational rather than neurological or sleep disorder services. The wide-ranging and special features of childhood narcolepsy need to be appreciated by health care and other professionals as well as by parents.", "publicationTitle": "Developmental medicine and child neurology", "publisher": "", "place": "", "date": "Apr 2006", "volume": "48", "issue": "4", "section": "", "partNumber": "", "partTitle": "", "pages": "307-310", "series": "", "seriesTitle": "", "seriesText": "", "journalAbbreviation": "Dev Med Child Neurol", "DOI": "10.1017/S0012162206000661", "citationKey": "", "url": "", "accessDate": "", "PMID": "", "PMCID": "", "ISSN": "0012-1622", "archive": "", "archiveLocation": "", "shortTitle": "", "language": "eng", "libraryCatalog": "NCBI PubMed", "callNumber": "", "rights": "", "extra": "PMID: 16542521", "tags": [ { "tag": "Child", "type": 1 }, { "tag": "Diagnosis, Differential", "type": 1 }, { "tag": "Disorders of Excessive Somnolence", "type": 1 }, { "tag": "Humans", "type": 1 }, { "tag": "Mood Disorders", "type": 1 }, { "tag": "Narcolepsy", "type": 1 }, { "tag": "Sleep Paralysis", "type": 1 } ], "collections": [], "relations": {}, "dateAdded": "2013-11-20T23:08:54Z", "dateModified": "2013-11-20T23:08:54Z" } }, { "key": "7N4J86Z3", "version": 12, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/7N4J86Z3", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/7N4J86Z3", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/D8N9S95Q", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "numChildren": 0 }, "data": { "key": "7N4J86Z3", "version": 12, "parentItem": "D8N9S95Q", "itemType": "attachment", "linkMode": "imported_file", "title": "oxybate in paed narcolepsy.pdf", "accessDate": "", "url": "", "note": "", "contentType": "application/pdf", "charset": "", "filename": "oxybate in paed narcolepsy.pdf", "md5": "b8a5f376c794ba7ac1eb822dbe80b6e6", "mtime": 1384988900000, "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:08:20Z", "dateModified": "2013-11-20T23:08:20Z" } }, { "key": "D8N9S95Q", "version": 11, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/D8N9S95Q", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/D8N9S95Q", "type": "text/html" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "creatorSummary": "Murali and Kotagal", "parsedDate": "2006-08", "numChildren": 2 }, "data": { "key": "D8N9S95Q", "version": 11, "itemType": "journalArticle", "title": "Off-label treatment of severe childhood narcolepsy-cataplexy with sodium oxybate", "creators": [ { "creatorType": "author", "firstName": "Hema", "lastName": "Murali" }, { "creatorType": "author", "firstName": "Suresh", "lastName": "Kotagal" } ], "abstractNote": "STUDY OBJECTIVES: To evaluate the efficacy and side-effect profile of off-label sodium oxybate (gamma hydroxy butyrate) therapy in severe childhood narcolepsy-cataplexy.\nDESIGN: Retrospective; chart review.\nSETTING: A multidisciplinary tertiary sleep center.\nPATIENTS: A group of eight children with severe narcolepsy-cataplexy diagnosed on the basis of clinical history, nocturnal polysomnography and the multiple sleep latency test were studied. A modified Epworth Sleepiness Scale and an arbitrary cataplexy severity scale (1 = minimal weakness, 2 = voluntarily preventable falls, 3 = falls to the ground) were utilized.\nINTERVENTIONS: Sodium oxybate therapy; concurrent medications were maintained.\nMEASUREMENTS AND RESULTS: Before sodium oxybate therapy, all subjects had suboptimally controlled sleepiness and cataplexy. Following treatment with sodium oxybate, 7/8 subjects (88%) improved. Cataplexy frequency decreased from a median of 38.5 to 4.5/ week (p = 0.0078). Cataplexy severity decreased from 2.75 to 1.75 (p = 0.06). The Epworth Sleepiness Scores improved from a median of 19 to 12.5 (p = 0.02). Suicidal ideation, dissociative episodes, tremor and constipation occurred in one subject each and terminal insomnia in two. Three of the 8 (38%) discontinued therapy. Two stopped the drug owing to side effects and one due to problems with postal delivery of the medication.\nCONCLUSIONS: This is the first report on sodium oxybate therapy in childhood narcolepsy-cataplexy. Our finding of improvement in cataplexy and sleepiness suggests that this medication is effective in treating severe childhood narcolepsy-cataplexy.", "publicationTitle": "Sleep", "publisher": "", "place": "", "date": "Aug 2006", "volume": "29", "issue": "8", "section": "", "partNumber": "", "partTitle": "", "pages": "1025-1029", "series": "", "seriesTitle": "", "seriesText": "", "journalAbbreviation": "Sleep", "DOI": "", "citationKey": "", "url": "", "accessDate": "", "PMID": "", "PMCID": "", "ISSN": "0161-8105", "archive": "", "archiveLocation": "", "shortTitle": "", "language": "eng", "libraryCatalog": "NCBI PubMed", "callNumber": "", "rights": "", "extra": "PMID: 16944670", "tags": [ { "tag": "Adolescent", "type": 1 }, { "tag": "Body Mass Index", "type": 1 }, { "tag": "Child", "type": 1 }, { "tag": "Dose-Response Relationship, Drug", "type": 1 }, { "tag": "Drug Administration Schedule", "type": 1 }, { "tag": "Female", "type": 1 }, { "tag": "Follow-Up Studies", "type": 1 }, { "tag": "Humans", "type": 1 }, { "tag": "Male", "type": 1 }, { "tag": "Narcolepsy", "type": 1 }, { "tag": "Polysomnography", "type": 1 }, { "tag": "Quality of Life", "type": 1 }, { "tag": "Retrospective Studies", "type": 1 }, { "tag": "Sleep, REM", "type": 1 }, { "tag": "Sodium Oxybate", "type": 1 }, { "tag": "Treatment Outcome", "type": 1 }, { "tag": "Wakefulness", "type": 1 } ], "collections": [], "relations": {}, "dateAdded": "2013-11-20T23:08:06Z", "dateModified": "2013-11-20T23:08:06Z" } }, { "key": "X27N39BX", "version": 11, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/X27N39BX", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/X27N39BX", "type": "text/html" }, "up": { 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"BAG4KVIT", "version": 9, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/BAG4KVIT", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/BAG4KVIT", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/TTWGG7BA", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } } }, "data": { "key": "BAG4KVIT", "version": 9, "parentItem": "TTWGG7BA", "itemType": "attachment", "linkMode": "linked_url", "title": "PubMed entry", "accessDate": "2013-11-20T23:07:15Z", "url": "http://www.ncbi.nlm.nih.gov/pubmed/18410846", "note": "", "contentType": "text/html", "charset": "", "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:07:15Z", "dateModified": "2013-11-20T23:07:15Z" } }, { "key": "TTWGG7BA", "version": 9, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/TTWGG7BA", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/TTWGG7BA", "type": "text/html" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "creatorSummary": "Vendrame et al.", "parsedDate": "2008-05", "numChildren": 2 }, "data": { "key": "TTWGG7BA", "version": 9, "itemType": "journalArticle", "title": "Narcolepsy in children: a single-center clinical experience", "creators": [ { "creatorType": "author", "firstName": "Martina", "lastName": "Vendrame" }, { "creatorType": "author", "firstName": "Navasuma", "lastName": "Havaligi" }, { "creatorType": "author", "firstName": "Chandra", "lastName": "Matadeen-Ali" }, { "creatorType": "author", "firstName": "Ruth", "lastName": "Adams" }, { "creatorType": "author", "firstName": "Sanjeev V", "lastName": "Kothare" } ], "abstractNote": "Although the initial manifestations of narcolepsy in children may differ from those with adult onset, hypersomnia remains the most common presenting sign. This study aimed to (1) describe the clinical and polysomnographic features, and treatment outcomes, of a group of children with narcolepsy, and (2) describe other sleep disorders to be considered in the differential diagnosis of hypersomnia and which may coexist with narcolepsy. A retrospective review of 125 children referred in 1 year for hypersomnia revealed 20 patients (16%) with narcolepsy. Of these, only 15% exhibited cataplexy, 10% experienced hallucinations, and none manifested sleep paralysis. Eighty-five percent of children with narcolepsy had sleep-disordered breathing on polysomnography. Body mass indices of these children were higher than for healthy, age-matched controls subjects. Other polysomnography findings included periodic limb movements of sleep (25%) and parasomnias (5%). The multiple sleep latency test revealed a mean sleep latency of 6.14 minutes, with sleep-onset rapid eye movement periods (median, 2/5 naps). Treatment with modafinil and sodium oxybate provided optimal control of daytime sleepiness. Physicians should routinely screen for hypersomnia in children by obtaining a detailed history and, in appropriate situations, ordering polysomnographic testing to rule out narcolepsy and other causes of hypersomnia.", "publicationTitle": "Pediatric neurology", "publisher": "", "place": "", "date": "May 2008", "volume": "38", "issue": "5", "section": "", "partNumber": "", "partTitle": "", "pages": "314-320", "series": "", "seriesTitle": "", "seriesText": "", "journalAbbreviation": "Pediatr. Neurol.", "DOI": "10.1016/j.pediatrneurol.2007.12.010", "citationKey": "", "url": "", "accessDate": "", "PMID": "", "PMCID": "", "ISSN": "0887-8994", "archive": "", "archiveLocation": "", "shortTitle": "Narcolepsy in children", "language": "eng", "libraryCatalog": "NCBI PubMed", "callNumber": "", "rights": "", "extra": "PMID: 18410846", "tags": [ { "tag": "Adolescent", "type": 1 }, { "tag": "Adult", "type": 1 }, { "tag": "Child", "type": 1 }, { "tag": "Child, Preschool", "type": 1 }, { "tag": "Female", "type": 1 }, { "tag": "Humans", "type": 1 }, { "tag": "Male", "type": 1 }, { "tag": "Narcolepsy", "type": 1 }, { "tag": "Polysomnography", "type": 1 }, { "tag": "Retrospective Studies", "type": 1 } ], "collections": [], "relations": {}, "dateAdded": "2013-11-20T23:07:15Z", "dateModified": "2013-11-20T23:07:15Z" } }, { "key": "H2VN9M3V", "version": 8, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/H2VN9M3V", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/H2VN9M3V", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/5AWHAX6P", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "numChildren": 0 }, "data": { "key": "H2VN9M3V", "version": 8, "parentItem": "5AWHAX6P", "itemType": "attachment", "linkMode": "imported_file", "title": "Clinical and Therapeutic Aspects of Childhood Narcolepsy-Cataplexy A Retrospective Study of 51 Children.pdf", "accessDate": "", "url": "", "note": "", "contentType": "application/pdf", "charset": "", "filename": "Clinical and Therapeutic Aspects of Childhood Narcolepsy-Cataplexy A Retrospective Study of 51 Children.pdf", "md5": "25d8aaff857d0f12eda074b9630b1486", "mtime": 1384988802000, "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:06:42Z", "dateModified": "2013-11-20T23:06:42Z" } }, { "key": "KKAQECPE", "version": 7, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/KKAQECPE", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/KKAQECPE", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/5AWHAX6P", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } } }, "data": { "key": "KKAQECPE", "version": 7, "parentItem": "5AWHAX6P", "itemType": "attachment", "linkMode": "linked_url", "title": "PubMed entry", "accessDate": "2013-11-20T23:06:35Z", "url": "http://www.ncbi.nlm.nih.gov/pubmed/21102987", "note": "", "contentType": "text/html", "charset": "", "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:06:35Z", "dateModified": "2013-11-20T23:06:35Z" } }, { "key": "5AWHAX6P", "version": 7, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/5AWHAX6P", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/5AWHAX6P", "type": "text/html" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "creatorSummary": "Aran et al.", "parsedDate": "2010-11", "numChildren": 2 }, "data": { "key": "5AWHAX6P", "version": 7, "itemType": "journalArticle", "title": "Clinical and therapeutic aspects of childhood narcolepsy-cataplexy: a retrospective study of 51 children", "creators": [ { "creatorType": "author", "firstName": "Adi", "lastName": "Aran" }, { "creatorType": "author", "firstName": "Mali", "lastName": "Einen" }, { "creatorType": "author", "firstName": "Ling", "lastName": "Lin" }, { "creatorType": "author", "firstName": "Guiseppe", "lastName": "Plazzi" }, { "creatorType": "author", "firstName": "Seiji", "lastName": "Nishino" }, { "creatorType": "author", "firstName": "Emmanuel", "lastName": "Mignot" } ], "abstractNote": "STUDY OBJECTIVE: to report on symptoms and therapies used in childhood narcolepsy-cataplexy.\nDESIGN, PATIENTS, AND SETTING: retrospective series of 51 children who completed the Stanford Sleep Inventory. HLA-DQB1*0602 typing (all tested, and 100% positive), polysomnography or Multiple Sleep Latency Test (76%), and cerebrospinal fluid hypocretin-1 measurements (26%, all with low levels) were also conducted. Prospective data on medication response was collected in 78% using a specially designed questionnaire.\nMEASUREMENTS AND RESULTS: patients were separated into children with onset of narcolepsy prior to (53%), around (29%), and after (18%) puberty. None of the children had secondary narcolepsy. Clinical features were similar across puberty groups, except for sleep paralysis, which increased in frequency with age. Common features included excessive weight gain (84% ≥ 4 kg within 6 months of onset of narcolepsy) and earlier puberty (when compared with family members), notably in subjects who gained the most weight. Streptococcus-positive throat infections were reported in 20% of cases within 6 months of onset of narcolepsy. Polysomnographic features were similar across groups, but 3 prepubertal children did not meet Multiple Sleep Latency Test diagnostic criteria. Regarding treatment, the most used and continued medications were modafinil (84% continued), sodium oxybate (79%), and venlafaxine (68%). Drugs such as methylphenidate, tricyclic antidepressants, or selective serotonin reuptake inhibitors were often tried but rarely continued. Modafinil was reported to be effective for treating sleepiness, venlafaxine for cataplexy, and sodium oxybate for all symptoms, across all puberty groups. At the conclusion of the study, half of children with prepubertal onset of narcolepsy were treated \"off label\" with sodium oxybate alone or with the addition of one other compound. In older children, however, most patients needed more than 2 drugs.\nCONCLUSION: this study reports on the clinical features of childhood narcolepsy and documents the safe use of treatments commonly used in adults in young children.", "publicationTitle": "Sleep", "publisher": "", "place": "", "date": "Nov 2010", "volume": "33", "issue": "11", "section": "", "partNumber": "", "partTitle": "", "pages": "1457-1464", "series": "", "seriesTitle": "", "seriesText": "", "journalAbbreviation": "Sleep", "DOI": "", "citationKey": "", "url": "", "accessDate": "", "PMID": "", "PMCID": "", "ISSN": "0161-8105", "archive": "", "archiveLocation": "", "shortTitle": "Clinical and therapeutic aspects of childhood narcolepsy-cataplexy", "language": "eng", "libraryCatalog": "NCBI PubMed", "callNumber": "", "rights": "", "extra": "PMID: 21102987", "tags": [ { "tag": "Adjuvants, Anesthesia", "type": 1 }, { "tag": "Adolescent", "type": 1 }, { "tag": "Age Distribution", "type": 1 }, { "tag": "Age of Onset", "type": 1 }, { "tag": "Antidepressive Agents, Second-Generation", "type": 1 }, { "tag": "Benzhydryl Compounds", "type": 1 }, { "tag": "Central Nervous System Stimulants", "type": 1 }, { "tag": "Child", "type": 1 }, { "tag": "Cyclohexanols", "type": 1 }, { "tag": "Drosophila Proteins", "type": 1 }, { "tag": "Drug Therapy, Combination", "type": 1 }, { "tag": "Female", "type": 1 }, { "tag": "Follow-Up Studies", "type": 1 }, { "tag": "Humans", "type": 1 }, { "tag": "Male", "type": 1 }, { "tag": "Narcolepsy", "type": 1 }, { "tag": "Nerve Tissue Proteins", "type": 1 }, { "tag": "Nuclear Proteins", "type": 1 }, { "tag": "Overweight", "type": 1 }, { "tag": "Polysomnography", "type": 1 }, { "tag": "Puberty", "type": 1 }, { "tag": "Questionnaires", "type": 1 }, { "tag": "Retrospective Studies", "type": 1 }, { "tag": "Sodium Oxybate", "type": 1 }, { "tag": "Streptococcal Infections", "type": 1 }, { "tag": "Transcription Factors", "type": 1 }, { "tag": "United States", "type": 1 }, { "tag": "Weight Gain", "type": 1 } ], "collections": [], "relations": {}, "dateAdded": "2013-11-20T23:06:35Z", "dateModified": "2013-11-20T23:06:35Z" } }, { "key": "3QFN5PI2", "version": 6, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/3QFN5PI2", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/3QFN5PI2", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/Q33PKVXC", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "numChildren": 0 }, "data": { "key": "3QFN5PI2", "version": 6, "parentItem": "Q33PKVXC", "itemType": "attachment", "linkMode": "imported_file", "title": "childhhod narcolepsy1998.pdf", "accessDate": "", "url": "", "note": "", "contentType": "application/pdf", "charset": "", "filename": "childhhod narcolepsy1998.pdf", "md5": "a07a89358c58c384a3a8368b3ffb89c2", "mtime": 1384988727000, "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:05:27Z", "dateModified": "2013-11-20T23:05:27Z" } }, { "key": "Q33PKVXC", "version": 5, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/Q33PKVXC", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/Q33PKVXC", "type": "text/html" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "creatorSummary": "Wise", "parsedDate": "1998-02", "numChildren": 2 }, "data": { "key": "Q33PKVXC", "version": 5, "itemType": "journalArticle", "title": "Childhood narcolepsy", "creators": [ { "creatorType": "author", "firstName": "M S", "lastName": "Wise" } ], "abstractNote": "Narcolepsy may present during childhood and is probably underrecognized and underdiagnosed in this population. The core symptoms of narcolepsy in children are similar to those in adults, but the expression may be different because of maturational factors. This report focuses on the presenting features that are unique to childhood narcolepsy and the appropriate diagnostic evaluation for suspected narcolepsy in children. Psychosocial and academic problems are almost universal in children with narcolepsy, and management strategies should address these areas. Although currently available stimulant medications may be helpful to some extent, the cornerstone of management is education, emotional and academic support, and careful follow-up over time. The overall goal for managing childhood narcolepsy is to assist the child and family in achieving optimal quality of life.", "publicationTitle": "Neurology", "publisher": "", "place": "", "date": "Feb 1998", "volume": "50", "issue": "2 Suppl 1", "section": "", "partNumber": "", "partTitle": "", "pages": "S37-42", "series": "", "seriesTitle": "", "seriesText": "", "journalAbbreviation": "Neurology", "DOI": "", "citationKey": "", "url": "", "accessDate": "", "PMID": "", "PMCID": "", "ISSN": "0028-3878", "archive": "", "archiveLocation": "", "shortTitle": "", "language": "eng", "libraryCatalog": "NCBI PubMed", "callNumber": "", "rights": "", "extra": "PMID: 9484422", "tags": [ { "tag": "Adolescent", "type": 1 }, { "tag": "Adult", "type": 1 }, { "tag": "Child", "type": 1 }, { "tag": "Child, Preschool", "type": 1 }, { "tag": "Humans", "type": 1 }, { "tag": "Narcolepsy", "type": 1 }, { "tag": "Quality of Life", "type": 1 } ], "collections": [], "relations": {}, "dateAdded": "2013-11-20T23:05:15Z", "dateModified": "2013-11-20T23:05:15Z" } }, { "key": "GWQQT2U2", "version": 5, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/GWQQT2U2", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/GWQQT2U2", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/Q33PKVXC", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } } }, "data": { "key": "GWQQT2U2", "version": 5, "parentItem": "Q33PKVXC", "itemType": "attachment", "linkMode": "linked_url", "title": "PubMed entry", "accessDate": "2013-11-20T23:05:15Z", "url": "http://www.ncbi.nlm.nih.gov/pubmed/9484422", "note": "", "contentType": "text/html", "charset": "", "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:05:15Z", "dateModified": "2013-11-20T23:05:15Z" } }, { "key": "G95P5SEH", "version": 4, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/G95P5SEH", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/G95P5SEH", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/GR7KU6EE", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "numChildren": 0 }, "data": { "key": "G95P5SEH", "version": 4, "parentItem": "GR7KU6EE", "itemType": "attachment", "linkMode": "imported_file", "title": "sodium oxybate in tx of childhood narcolepsy cataplexy retrospective study sleep medicine 2012.pdf", "accessDate": "", "url": "", "note": "", "contentType": "application/pdf", "charset": "", "filename": "sodium oxybate in tx of childhood narcolepsy cataplexy retrospective study sleep medicine 2012.pdf", "md5": "0949b04b2edd45c00e64d4f7ed0dfc6b", "mtime": 1384988594000, "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:03:14Z", "dateModified": "2013-11-20T23:03:14Z" } }, { "key": "8JNADKTX", "version": 2, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/8JNADKTX", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/8JNADKTX", "type": "text/html" }, "up": { "href": "https://api.zotero.org/groups/233156/items/GR7KU6EE", "type": "application/json" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } } }, "data": { "key": "8JNADKTX", "version": 2, "parentItem": "GR7KU6EE", "itemType": "attachment", "linkMode": "linked_url", "title": "PubMed entry", "accessDate": "2013-11-20T23:02:02Z", "url": "http://www.ncbi.nlm.nih.gov/pubmed/22445591", "note": "", "contentType": "text/html", "charset": "", "tags": [], "relations": {}, "dateAdded": "2013-11-20T23:02:02Z", "dateModified": "2013-11-20T23:02:02Z" } }, { "key": "GR7KU6EE", "version": 2, "library": { "type": "group", "id": 233156, "name": "Narcolepsy & Cataplexy in Children", "links": { "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children", "type": "text/html" } } }, "links": { "self": { "href": "https://api.zotero.org/groups/233156/items/GR7KU6EE", "type": "application/json" }, "alternate": { "href": "https://www.zotero.org/groups/narcolepsy__cataplexy_in_children/items/GR7KU6EE", "type": "text/html" } }, "meta": { "createdByUser": { "id": 1503175, "username": "mattjt", "name": "", "links": { "alternate": { "href": "https://www.zotero.org/mattjt", "type": "text/html" } } }, "creatorSummary": "Mansukhani and Kotagal", "parsedDate": "2012-06", "numChildren": 2 }, "data": { "key": "GR7KU6EE", "version": 2, "itemType": "journalArticle", "title": "Sodium oxybate in the treatment of childhood narcolepsy-cataplexy: a retrospective study", "creators": [ { "creatorType": "author", "firstName": "Meghna P", "lastName": "Mansukhani" }, { "creatorType": "author", "firstName": "Suresh", "lastName": "Kotagal" } ], "abstractNote": "OBJECTIVE: To evaluate the efficacy and side effect profile of sodium oxybate in the treatment for narcolepsy-cataplexy in the pediatric age group.\nMETHODS: A retrospective study was conducted on 15 children and adolescents with narcolepsy-cataplexy who had been treated with sodium oxybate. The mean age at diagnosis of narcolepsy was 11 years (range 3-17 years). Subjects were followed for 3-90 months (mean 33) after starting sodium oxybate. During this period of time they were also maintained on other medications for sleepiness (n=14) and cataplexy (n=6). The charts were reviewed for documentation of improvement in sleepiness or cataplexy, side effects, and functioning in daily life.\nRESULTS: Subsequent to the addition of sodium oxybate, sleepiness improved in 13/15 patients. In patients who had Epworth Sleepiness Scale (ESS) assessments, the score fell from a baseline median of 18 to 12 (n=10, p=0.01). The number of cataplexy episodes estimated by parents decreased from a median of 38/week pre-treatment to <1/week post treatment (n=14, p<0.001). Cataplexy severity, measured on an arbitrary scale, fell from a median of 3 (severe) to 1 (mild) in all 15 subjects (p<0.001). Two of the 15 patients (13%) discontinued sodium oxybate, one for insurance reasons and the other due to constipation and dissociative feelings. A third patient stopped the medication temporarily due to body aches and dizziness, but then resumed treatment without recurrence of symptoms. Side effects in four others included tremor, blurring of vision, nocturnal awakenings, and increased nightmares. Overall, side effects occurred in 6/15 (40%) individuals. Improvement in social/academic spheres was noted in 11/15 (73%) subjects after starting sodium oxybate. The median BMI before and after treatment remained unchanged at 23 (n=14, p=0.99). Median values of height and weight before and after treatment also did not change significantly. The mean dose of sodium oxybate was 5 ± 2 g. Dose escalation owing to development of tolerance was not encountered.\nCONCLUSIONS: Sodium oxybate is effective in alleviating sleepiness and cataplexy in childhood onset narcolepsy-cataplexy. The therapeutic response was sustained over time, and without development of tolerance. Forty percent of the subjects experienced adverse effects.", "publicationTitle": "Sleep medicine", "publisher": "", "place": "", "date": "Jun 2012", "volume": "13", "issue": "6", "section": "", "partNumber": "", "partTitle": "", "pages": "606-610", "series": "", "seriesTitle": "", "seriesText": "", "journalAbbreviation": "Sleep Med.", "DOI": "10.1016/j.sleep.2011.10.032", "citationKey": "", "url": "", "accessDate": "", "PMID": "", "PMCID": "", "ISSN": "1878-5506", "archive": "", "archiveLocation": "", "shortTitle": "Sodium oxybate in the treatment of childhood narcolepsy-cataplexy", "language": "eng", "libraryCatalog": "NCBI PubMed", "callNumber": "", "rights": "", "extra": "PMID: 22445591", "tags": [ { "tag": "Adjuvants, Anesthesia", "type": 1 }, { "tag": "Adolescent", "type": 1 }, { "tag": "Child", "type": 1 }, { "tag": "Child, Preschool", "type": 1 }, { "tag": "Drug Tolerance", "type": 1 }, { "tag": "Female", "type": 1 }, { "tag": "Humans", "type": 1 }, { "tag": "Male", "type": 1 }, { "tag": "Narcolepsy", "type": 1 }, { "tag": "Off-Label Use", "type": 1 }, { "tag": "Retrospective Studies", "type": 1 }, { "tag": "Sleep Stages", "type": 1 }, { "tag": "Sleep, REM", "type": 1 }, { "tag": "Sodium Oxybate", "type": 1 }, { "tag": "Treatment Outcome", "type": 1 }, { "tag": "Wakefulness", "type": 1 } ], "collections": [], "relations": {}, "dateAdded": "2013-11-20T23:02:02Z", "dateModified": "2013-11-20T23:02:02Z" } } ]