Research

Intravenous immunoglobulin for the treatment of autoimmune encephalopathy in children with autism

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
Transl Psychiatry-2018

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.

Family Impacts Reported By Parents Raising Children With Pediatric Acute-onset Neuropsychiatric Syndrome (PANS)

Marcey Mettica, B.S., M.S
A Dissertation Submitted in Partial Fulfillment of the Requirements for The Degree of Doctor of Family Studies in The Graduate School of The Texas Woman’s University
PDF of Dissertation

This study explored variables that were thought to possiblyinfluence family impact and explored relationships between demographic factors, illness factors, and symptom severity with impact on the family

Gut Microbiota Profiling and Gut-Brain Crosstalk in Children Affected by Pediatric Acute-Onset Neuropsychiatric Syndrome and Pediatric Autoimmune Neuropsychiatric Disorders Associated With Streptococcal Infections

Quagliariello A, Del Chierico F, Russo A, Reddel S, Conte G, Lopetuso LR, Ianiro G, Dallapiccola B, Cardona F, Gasbarrini A, Putignani L.
Front Microbiology-2018
PDF

Excerpt: The results obtained from this study suggest that streptococcal infections can alter gut microbial communities leading to a pro-inflammatory state in the gut by selecting for specific bacterial strains that are normally associated with gut inflammation and activation of the immune response. This condition is likely maintained in patients, even after the infection itself has resolved.

Principles and approaches to the treatment of immune-mediated movement disorders

Mohammad SS, Dale RC
Eur J Paediatric Neurol-2018

Excerpt: Immune mediated movement disorders include movement disorders in the context of autoimmune encephalitis such as anti-NMDAR encephalitis, post-infectious autoimmune movement disorders such as Sydenham chorea, paraneoplastic autoimmune movement disorders such as opsoclonus myoclonus ataxia syndrome, and infection triggered conditions such as paediatric acute neuropsychiatric syndrome. This review focuses on the approach to treatment of immune mediated movement disorders, which requires an understanding of the immunopathogenesis, whether the disease is destructive or ‘altering’, and the natural history of disease. 

Paediatric Autoimmune Neuropsychiatric Disorder Associated with Group A Beta-Haemolytic Streptococcal Infection: An Indication for Tonsillectomy? A Review of the Literature

Rajgor AD, Hakim NA, Ali S, Darr A.
Int J Otolaryngology-2018
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PANDAS is a significantly debilitating condition, with detrimental effects on a child’s psychophysical development. This review provides an insight into the current literature and the potential for surgery to treat PANDAS. However due to the current limitations of the literature drawing definitive conclusions is difficult. Thus, the next required steps would involve conducting a prospective collaborative study.

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 
Neuropsychiatry-2018
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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.

 

A Double-Blind Randomized Placebo-Controlled Pilot Study of Azithromycin in Youth with Acute-Onset Obsessive-Compulsive Disorder

Murphy, Brennan, Johnco, Parker-Athill, Miladinovic, Storch, Lewin 
J Child Adolesc Psychopharmacology-2017

This double blind pilot study suggests that azithromycin may be helpful in treating youth meeting the PANS diagnosis, especially those with elevated levels of both OCD and tic symptoms. Azithromycin was well tolerated, but the potential for cardiac risks suggests that additional monitoring may be needed to ensure safety.

Treatment of Pediatric Acute-Onset Neuropsychiatric Disorder in a Large Survey Population

Denise Calaprice, Janice Tona, and Tanya K. Murphy
Journal of Child and Adolescent Psychopharmacology-2017

Among the PANS patients represented in this study, relatively aggressive treatment courses targeted at eradicating infection and modulating the inflammatory response appeared to provide the best caregiver-reported therapeutic results, and to be generally well tolerated. Given its relative efficacy and tolerability, treatment targeting the inflammatory response may represent an underutilized approach in this population.

Inflammation in the Neurocircuitry of Obsessive-Compulsive Disorder

Sophia Attwells, HBS; Elaine Setiawan, PhD; Alan A. Wilson, PhD, Pablo M. Rusjan, PhD; Romina Mizrahi, MD, PhD, FRCP(C); Laura Miler, HBS; Cynthia Xu, MD; Margaret Anne Richter, MD, FRCP(C); Alan Kahn, MD, FRCP(C); Stephen J. Kish, PhD; Sylvain Houle, MD, PhD, FRCP(C); Lakshmi Ravindran, MD, FRCP(C); Jeffrey H. Meyer, MD, PhD, FRCP(C)
JAMA Psychiatry 2017

The regional distribution of elevated TSPO VT argues that the autoimmune/neuroinflammatory theories of OCD should extend beyond the basal ganglia to include the cortico-striato-thalamo-cortical circuit. Immunomodulatory therapies should be investigated in adult OCD, rather than solely childhood OCD, particularly in cases with prominent distress when preventing compulsions.

Mental Health News Article
Brain Behavior Research Foundation Article

Association of Streptococcal Throat Infection With Mental Disorders Testing Key Aspects of the PANDAS Hypothesis in a Nationwide Study

Sonja Orlovska, Claus Høstrup Vestergaard, Bodil Hammer Bech, PhD
JAMA Psychiatry-2017

This large-scale study investigating key aspects of the PANDAS hypothesis found that individuals with a streptococcal throat infection had elevated risks of mental disorders, particularly OCD and tic disorders. However, nonstreptococcal throat infection was also associated with increased risks, although less than streptococcal infections for OCD and any mental disorder, which could also support important elements of the diagnostic concept of pediatric acute-onset neuropsychiatric syndrome.