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Benefits of IVIG in Pediatric Acute-Onset Neuropsychiatric Syndrome (2411)
Isaac Melamed, Roger Kobayashi, Maeve O’Connor, Ai Lan Kobayashi, Andrew Schechterman, Melinda Heffron, Sharon Canterberry, Holly Miranda, Nazia Rashid Neurology Apr 14 2020, 94
“Conclusions: In PANS, which may be associated with an underlying immune dysregulation, IVIG [Octagam 5%] successfully ameliorated psychological symptoms and dysfunction, with sustained benefits for at least 8 weeks, and up to 46 weeks, following the final infusion. In addition, baseline immune and autoimmune profiles demonstrated significant elevations in a majority of subjects, which requires further evaluation, characterization, and study to clarify the potential immune dysfunction by which PANS manifests and progresses.”
Kathleen Connery, Marie Tippett, Leanna M. Delhey, Shannon Rose, John C. Slattery, Stephen G. Kahler, Juergen Hahn, Uwe Kruger, Madeleine W. Cunningham, Craig Shimasaki, Richard E. Frye
In an open-labeled IVIG study in children with comorbid ASD and PANS/ PANDAS, anti-tubulin and anti-D2R (as measured by the Cunningham panel) were associated with responsiveness to IVIG treatment, suggesting that these autoantibodies could be biomarkers to select for positive IVIG treatment outcomes. Research continues to explore serum biomarkers and genetic risk factors that can provide a diagnostic tool and/or complement diagnosis in children with PANS/PANDAS.
Pediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcal Infection (PANDAS): Clinical Manifestations, IVIG Treatment Outcomes, Results from a Cohort of Italian Patients
Piero Pavone, Raffaele Falsaperia, Francesco Nicita, Andreana Zecchini, Chiari Battaglia, Alberto Spalice, Lucia Iozzi, Enrico Parano, Giovanna Vitaliti, Alberto Verrotti, Vincenzo Belcastro, Sung Yoon Cho, Dong-Kyu Jin, Salvatore Savasta
In this cohort, 29 out of 34 of children treated with IVIG showed a reduction or disappearance of the symptomatology, while in 5 cases (15%), despite 3 IVIG courses, there was only temporary improvement of clinical symptoms, with their reappearance within 1 to 6 months. According to the results of literature and ours no definitive conclusions may be drawn from IVIG treatment for PANDAS. Limitations of the present study include the absence of casecontrols and long-term follow up. However, in our opinion, treatment with IVIG has been shown to be effective in PANDAS children with a serious-severe type and to be well tolerated. As it has been reported by Frankovic et al. most of the PANS Research Consortium (PRC) members prefer use of IVIG in treatment of patients with this disorder in moderate to severe forms.
Randomized, Controlled Trial of Intravenous Immunoglobulin for Pediatric Autoimmune Neuropsychiatric Disorders Associated With Streptococcal Infections
Kyle A. Williams, MD, PhD, Susan E. Swedo, MD, Cristan A. Farmer, PhD, Heidi Grantz, LCSW, Paul J. Grant, MD, Precilla D’Souza, CRNP, Rebecca Hommer, MD, Liliya Katsovich, MA, Robert A. King, MD, James F. Leckman, MD, Phd
Journal of the American Academy of Child & Adolescent Psychiatry–2016
IVIG was safe and well tolerated. Between-group differences were smaller than anticipated, and the double-blind comparison failed to demonstrate superiority of IVIG over placebo. The observed open-label improvements indicate that future trials would benefit from larger sample sizes designed in part to aid in the identification of biomarkers predictive of a positive response to immunotherapy. Future investigations focused on the natural history of PANDAS are also warranted.
PANDAS: Baseline Immunoglobulin Levels Predict Achievement of Remission at One Year Following IVIG Therapy
David S. Younger, Phyllis A. Mast, and Denis A. Bouboulis
Journal of Neurology and Neurosurgery-2016
Children with PANDAS who had baseline low IgA, IgG or IgG subclass levels were more likely than others to achieve 100 % improvement after IVIg therapy at 12 months follow-up.
Use of Intravenous Immunoglobulin in the Treatment of Twelve Youths with Pediatric Autoimmune Neuropsychiatric Disorders Associated with Streptococcal infections
This case series demonstrates the benefits of IVIG therapy for youths with PANDAS/PANS, including those who had been symptomatic for several years prior to treatment. Although the generalizability of this retrospective report is limited, the selected cases represent the breadth of symptom presentations in PANDAS/PANS and provide additional evidence that IVIG may be useful in the management of children with moderate-severe symptoms.