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Paediatric Acute-onset Neuropsychiatric Syndrome (PANS) and intravenous immunoglobulin (IVIG) : comprehensive open-label trial in ten children

Hajjari, Parisa ; Oldmark, Malin Huldt ; Fernell, Elisabeth ; Jakobsson, Klara ; Vinsa, Ingrid ; Thorsson, Max LU ; Monemi, Mehran ; Stenlund, Lotta ; Fasth, Anders and Furuhjelm, Catrin , et al. (2022) In BMC Psychiatry 22. p.1-13
Abstract

BACKGROUND: Treatment with intravenous immunoglobulin (IVIG) in children with Paediatric Acute-onset Neuropsychiatric Syndrome (PANS) has for many years been used on clinical indications, but the research evidence for its efficacy is insufficient.

METHODS: Open-label prospective in-depth trial including ten children (median age 10.3 years) with PANS, who received IVIG treatment 2 g/kg monthly for three months. Primary outcomes were changes in symptom severity and impairment from baseline to first and second follow-up visits one month after first and one month after third treatment, using three investigator-rated scales: Paediatric Acute Neuropsychiatric Symptom (PANS) scale, Clinical Global Impression - Severity and Improvement... (More)

BACKGROUND: Treatment with intravenous immunoglobulin (IVIG) in children with Paediatric Acute-onset Neuropsychiatric Syndrome (PANS) has for many years been used on clinical indications, but the research evidence for its efficacy is insufficient.

METHODS: Open-label prospective in-depth trial including ten children (median age 10.3 years) with PANS, who received IVIG treatment 2 g/kg monthly for three months. Primary outcomes were changes in symptom severity and impairment from baseline to first and second follow-up visits one month after first and one month after third treatment, using three investigator-rated scales: Paediatric Acute Neuropsychiatric Symptom (PANS) scale, Clinical Global Impression - Severity and Improvement (CGI-S and CGI-I) scales. Secondary outcomes reported here were changes in Children's Yale-Brown Obsessive Compulsive Scale (CY-BOCS) scores, and side effects.

RESULTS: All ten children received three treatments at one-month intervals according to study plan. From baseline to second follow-up marked reductions were seen in mean total PANS scale scores (p = .005), and CGI-S scores (p = .004). CGI-I ratings showed much to very much global improvement (mean CGI-I 1.8). Nine children had clinical response defined as > 30% reduction in PANS Scale scores. Improvements were also noted for CY-BOCS scores (p = .005), and in school attendance. Three children suffered moderate to severe temporary side effects after the first treatment, and the remaining seven had mild to moderate side effects. Side effects were much less severe after second and third treatments.

CONCLUSIONS: Considerable and pervasive improvements in symptoms and clinical impairments were seen in these ten children after three monthly IVIG treatments. Moderate to severe transient side effects occurred in three cases.

TRIAL REGISTRATION: EudraCT no. 2019-004758-27, Clinicaltrials.gov no. NCT04609761, 05/10/2020.

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publishing date
type
Contribution to journal
publication status
published
keywords
Autoimmune Diseases/diagnosis, Child, Humans, Immunoglobulins, Intravenous/therapeutic use, Obsessive-Compulsive Disorder/diagnosis, Prospective Studies
in
BMC Psychiatry
volume
22
article number
535
pages
1 - 13
publisher
BioMed Central (BMC)
external identifiers
  • scopus:85135580793
  • pmid:35933358
ISSN
1471-244X
DOI
10.1186/s12888-022-04181-x
language
English
LU publication?
no
additional info
© 2022. The Author(s).
id
9b8a7c21-e3ad-417d-8cdb-2b654826eeda
date added to LUP
2024-11-21 22:15:26
date last changed
2025-01-31 10:55:36
@article{9b8a7c21-e3ad-417d-8cdb-2b654826eeda,
  abstract     = {{<p>BACKGROUND: Treatment with intravenous immunoglobulin (IVIG) in children with Paediatric Acute-onset Neuropsychiatric Syndrome (PANS) has for many years been used on clinical indications, but the research evidence for its efficacy is insufficient.</p><p>METHODS: Open-label prospective in-depth trial including ten children (median age 10.3 years) with PANS, who received IVIG treatment 2 g/kg monthly for three months. Primary outcomes were changes in symptom severity and impairment from baseline to first and second follow-up visits one month after first and one month after third treatment, using three investigator-rated scales: Paediatric Acute Neuropsychiatric Symptom (PANS) scale, Clinical Global Impression - Severity and Improvement (CGI-S and CGI-I) scales. Secondary outcomes reported here were changes in Children's Yale-Brown Obsessive Compulsive Scale (CY-BOCS) scores, and side effects.</p><p>RESULTS: All ten children received three treatments at one-month intervals according to study plan. From baseline to second follow-up marked reductions were seen in mean total PANS scale scores (p = .005), and CGI-S scores (p = .004). CGI-I ratings showed much to very much global improvement (mean CGI-I 1.8). Nine children had clinical response defined as &gt; 30% reduction in PANS Scale scores. Improvements were also noted for CY-BOCS scores (p = .005), and in school attendance. Three children suffered moderate to severe temporary side effects after the first treatment, and the remaining seven had mild to moderate side effects. Side effects were much less severe after second and third treatments.</p><p>CONCLUSIONS: Considerable and pervasive improvements in symptoms and clinical impairments were seen in these ten children after three monthly IVIG treatments. Moderate to severe transient side effects occurred in three cases.</p><p>TRIAL REGISTRATION: EudraCT no. 2019-004758-27, Clinicaltrials.gov no. NCT04609761, 05/10/2020.</p>}},
  author       = {{Hajjari, Parisa and Oldmark, Malin Huldt and Fernell, Elisabeth and Jakobsson, Klara and Vinsa, Ingrid and Thorsson, Max and Monemi, Mehran and Stenlund, Lotta and Fasth, Anders and Furuhjelm, Catrin and Johnels, Jakob Åsberg and Gillberg, Christopher and Johnson, Mats}},
  issn         = {{1471-244X}},
  keywords     = {{Autoimmune Diseases/diagnosis; Child; Humans; Immunoglobulins, Intravenous/therapeutic use; Obsessive-Compulsive Disorder/diagnosis; Prospective Studies}},
  language     = {{eng}},
  month        = {{08}},
  pages        = {{1--13}},
  publisher    = {{BioMed Central (BMC)}},
  series       = {{BMC Psychiatry}},
  title        = {{Paediatric Acute-onset Neuropsychiatric Syndrome (PANS) and intravenous immunoglobulin (IVIG) : comprehensive open-label trial in ten children}},
  url          = {{http://dx.doi.org/10.1186/s12888-022-04181-x}},
  doi          = {{10.1186/s12888-022-04181-x}},
  volume       = {{22}},
  year         = {{2022}},
}