Intravenous immunoglobulin for the treatment of Kawasaki disease in children

Oates-Whitehead, Richmal; Baumer, J H; Haines, Linda; Love, Samantha; Maconochie, Ian; Gupta, Amit; Roman, Kevin S.; Dua, Jaspal; Flynn, Ichiko

doi:10.1002/14651858.cd004000

Cited by 179 publications

(151 citation statements)

References 49 publications

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“…(Pillebout et al, 2002). Correlation with acute myocardial infarction or multivessel disease has been rarely reported (AbdelHadi et al, 1981;Lutz et al, 2009;Osman & McCreery, 2000). (Table 1) An interesting finding of the present case, is the discrepancy between absence of serious risk factors for coronary artery disease in a young patient and severity of infarction (three-vessel disease), which was also reported in two similar cases, with thrombotic thrombocytopenic purpura.…”

supporting

confidence: 70%

Coronary Artery Disease and Systemic Vasculitis: Case Report and Review

Eleftheriadis¹,

Eleftheriadis²

2012

Coronary Artery Diseases

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supporting

confidence: 70%

Coronary Artery Disease and Systemic Vasculitis: Case Report and Review

Eleftheriadis¹,

Eleftheriadis²

2012

Coronary Artery Diseases

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“…27 In pediatric narcolepsy, patients often present close to the onset of the disease, which affords the opportunity to intervene rapidly with immunomodulatory therapies. Among the potential immunomodulatory therapies available, and despite its lack of specificity, IVIg is an attractive option for children with narcolepsy because of established efficacy and good tolerability in a number of other pediatric autoimmune diseases, including Guillain-Barré syndrome, 28 Kawasaki disease, 29 and Stevens-Johnson syndrome. 30 The first published reports on the use of IVIg in patients with narcolepsy have been uncontrolled case studies involving very small numbers of patients.…”

mentioning

confidence: 99%

Intravenous Immunoglobulin Therapy in Pediatric Narcolepsy: A Nonrandomized, Open-Label, Controlled, Longitudinal Observational Study

Lecendreux

Berthier

Corny

et al. 2017

Journal of Clinical Sleep Medicine

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Study Objectives: Previous case reports of intravenous immunoglobulins (IVIg) in pediatric narcolepsy have shown contradictory results. Methods: This was a nonrandomized, open-label, controlled, longitudinal observational study of IVIg use in pediatric narcolepsy with retrospective data collection from medical files obtained from a single pediatric national reference center for the treatment of narcolepsy in France. Of 56 consecutively referred patients with narcolepsy, 24 received IVIg (3 infusions administered at 1-mo intervals) in addition to standard care (psychostimulants and/or anticataplectic agents), and 32 continued on standard care alone (controls). Results: For two patients in each group, medical files were unavailable. Of the 22 IVIg patients, all had cerebrospinal fluid (CSF) hypocretin ≤ 110 pg/mL and were HLA-DQB1*06:02 positive. Of the 30 control patients, 29 were HLA-DQB1*06:02 positive and of those with available CSF measurements, all 12 had hypocretin ≤ 110 pg/mL. Compared with control patients, IVIg patients had shorter disease duration, shorter latency to sleep onset, and more had received H1N1 vaccination. Mean (standard deviation) follow-up length was 2.4 (1.1) y in the IVIg group and 3.9 (1.7) y in controls. In multivariate-adjusted linear mixed-effects analyses of change from baseline in Ullanlinna Narcolepsy Scale (UNS) scores, high baseline UNS, but not IVIg treatment, was associated with a reduction in narcolepsy symptoms. On time-to-event analysis, among patients with high baseline UNS scores, control patients achieved a UNS score < 14 (indicating remission) less rapidly than IVIg patients (adjusted hazard ratio 0.18; 95% confidence interval: 95% confidence interval: 0.03, 0.95; p = 0.043). Shorter or longer disease duration did not influence treatment response in any analysis. Conclusions: Overall, narcolepsy symptoms were not significantly reduced by IVIg. However, in patients with high baseline symptoms, a subset of IVIgtreated patients achieved remission more rapidly than control patients. I NTRO DUCTI O NNarcolepsy is a debilitating and currently an incurable neurological condition with a characteristic constellation of symptoms, including excessive daytime sleepiness (EDS), cataplexy, sleep paralysis, hypnagogic hallucinations, and disturbed nocturnal sleep.Until recently, and at the time patients in this study were treated, the disorder was categorized into narcolepsy with cataplexy and narcolepsy without cataplexy according to the revised second edition of the International Classification of Sleep Disorders (ICSD).1 ICSD-2-revised has now been replaced by ICSD-3, which classifies narcolepsy into type 1 and type 2 narcolepsy. 2The majority of patients with type 1 narcolepsy have hypocretin deficiency (cerebrospinal fluid [CSF] hypocretin-1 level ≤ 110 pg/ mL), which is considered to result from an autoimmune-mediated loss of hypocretin-secreting neurons in those who are genetically predisposed (most commonly those with human leucocyte antigen [HLA] DQB1*06:02 haplotype). The...

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“…Administration in divided doses in five days has been shown to be more efficient in preventing coronary artery aneurism. Acetylssalicylic acid is used in addition to IVIG for treatment of this disease (28,30,33).…”

Section: B) Kawasaki Diseasementioning

confidence: 99%