Intramuscular Immunoglobulins as a Therapeutic Modality for Neural Inflammation in patients with ASD and PANS: A Combined Case Report
Main Article Content
Abstract
This report explores the increasing link between autoimmune disorders and neurodiverse conditions, particularly Autism Spectrum Disorder (ASD) and Pediatric Acute-onset Neuropsychiatric Syndrome (PANS). A small study included aims to evaluate the effectiveness of intramuscular immunoglobulins (IMIG) in reducing neuroinflammation in these conditions. Seven children with Autism Spectrum Disorder and five with Pediatric Acute-onset Neuropsychiatric Syndrome were treated with monthly intramuscular immunoglobulins injections. Parents rated improvements in concentration, communication, and behaviour for Autism Spectrum Disorder, and reduction of tics for Pediatric Acute-onset Neuropsychiatric Syndrome. Results showed notable improvements, particularly in Pediatric Acute-onset Neuropsychiatric Syndrome patients. Intramuscular immunoglobulins offer a promising, cost-effective alternative to intravenous immunoglobulins (IVIG) in managing autoimmune neuroinflammation, warranting further investigation.
Article Details
How to Cite
PR, Fourie; JC, Armstrong.
Intramuscular Immunoglobulins as a Therapeutic Modality for Neural Inflammation in patients with ASD and PANS: A Combined Case Report.
Medical Research Archives, [S.l.], v. 12, n. 10, oct. 2024.
ISSN 2375-1924.
Available at: <https://esmed.org/MRA/mra/article/view/5984>. Date accessed: 15 nov. 2024.
doi: https://doi.org/10.18103/mra.v12i10.5984.
Section
Case Reports
The Medical Research Archives grants authors the right to publish and reproduce the unrevised contribution in whole or in part at any time and in any form for any scholarly non-commercial purpose with the condition that all publications of the contribution include a full citation to the journal as published by the Medical Research Archives.
References
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22. Connery K, Tippett M, Delhey LM, et al. Intravenous immunoglobulin for the treatment of autoimmune encephalopathy in children with autism. Transl Psychiatry. 2018;8(1):148. Published 2018 Aug 10. doi:10.1038/s41398-018-0214-7
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25. Hardin H, Shao W, Bernstein JA. An updated review of paediatric autoimmune neuropsychiatric disorders associated with Streptococcus/paediatric acute-onset neuropsychiatric syndrome, also known as idiopathic autoimmune encephalitis: What the allergist should know. Ann Allergy Asthma Immunol. 2023;131(5):567-575. doi:10.1016/j.anai.2023.08.022
26. Rossignol DA, Frye RE. A Systematic Review and Meta-Analysis of Immunoglobulin G Abnormalities and the Therapeutic Use of Intravenous Immunoglobulins (IVIG) in Autism Spectrum Disorder. J Pers Med. 2021;11(6):488. Published 2021 May 30. doi:10.3390/jpm11060488
27. Guglielmi G. Found: the dial in the brain that controls the immune system. Nature. Published online May 1, 2024. doi:10.1038/d41586-024-01259-2
28. Tan J, Smith CH, Goldman RD. Paediatric autoimmune neuropsychiatric disorders associated with streptococcal infections. Can Fam Physician. 2012;58(9):957-959.
29. Frye RE. Mitochondrial Dysfunction in Autism Spectrum Disorder: Unique Abnormalities and Targeted Treatments. Semin Pediatr Neurol. 2020;35:100829. doi:10.1016/j.spen.2020.100829
30. Boris M, Goldblatt A, Edelson SM. Improvements in children with autism treated with intravenous gamma globulin. J. Nutr. Environ. Med. 2005;15:169–176. doi: 10.1080/13590840600681827
31. Griffiths KK, Levy RJ. Evidence of Mitochondrial Dysfunction in Autism: Biochemical Links, Genetic-Based Associations, and Non-Energy-Related Mechanisms. Oxid Med Cell Longev. 2017;2017:4314025. doi:10.1155/2017/4314025
32. Frankovich J, Swedo S, Murphy T, et al. Clinical Management of Pediatric Acute-Onset Neuropsychiatric Syndrome: Part II-Use of Immunomodulatory Therapies. J Child Adolesc Psychopharmacol. 2017;27(7):574-593. doi:10.1089/cap.2016.0148
33. Personal experience.
34. Pfeiffer HCV, Wickstrom R, Skov L, et al. Clinical guidance for diagnosis and management of suspected Pediatric Acute-onset Neuropsychiatric Syndrome in the Nordic countries. Acta Paediatr. 2021;110(12):3153-3160. doi:10.1111/apa.15875
35. Melamed IR, Heffron M, Testori A, Lipe K. A pilot study of high-dose intravenous immunoglobulin 5% for autism: Impact on autism spectrum and markers of neuroinflammation. Autism Res. 2018;11(3):421-433. doi:10.1002/aur.1906
2. D'Auria E, Minutoli M, Colombo A, et al. Allergy and autoimmunity in children: non-mutually exclusive diseases. A narrative review. Front Pediatr. 2023;11:1239365. Published 2023 Nov 2. doi:10.3389/fped.2023.1239365
3. Okada H, Kuhn C, Feillet H, Bach JF. The 'hygiene hypothesis' for autoimmune and allergic diseases: an update. Clin Exp Immunol. 2010;160(1):1-9. doi:10.1111/j.1365-2249.2010.04139.x
4. Zhou D, Zhang H, Bai Z, et al. Exposure to soil, house dust and decaying plants increases gut microbial diversity and decreases serum immunoglobulin E levels in BALB/c mice. Environ Microbiol. 2016;18(5):1326-1337. doi:10.1111/1462-2920.12895
5. Taniya MA, Chung HJ, Al Mamun A, et al. Role of Gut Microbiome in Autism Spectrum Disorder and Its Therapeutic Regulation. Front Cell Infect Microbiol. 2022;12:915701. Published 2022 Jul 22. doi:10.3389/fcimb.2022.915701
6. Dietert RR, Coleman ME, North DW, Stephenson MM. Nourishing the Human Holobiont to Reduce the Risk of Non-Communicable Diseases: A Cow’s Milk Evidence Map Example. Applied Microbiology. 2022; 2(1):25-52. https://doi.org/10.3390/applmicrobiol2010003
7. Arenella M, Fanelli G, Kiemeney LA, McAlonan G, Murphy DG, Bralten J. Genetic relationship between the immune system and autism. Brain Behav Immun Health. 2023;34:100698. Published 2023 Nov 3. doi:10.1016/j.bbih.2023.100698
8. Iinuma T, Okamoto Y, Morimoto Y, et al. Pathogenicity of memory Th2 cells is linked to stage of allergic rhinitis. Allergy. 2018;73(2):479-489. doi:10.1111/all.13295
9. Lampiasi N, Bonaventura R, Deidda I, Zito F, Russo R. Inflammation and the Potential Implication of Macrophage-Microglia Polarisation in Human ASD: An Overview. Int J Mol Sci. 2023;24(3):2703. Published 2023 Jan 31. doi:10.3390/ijms24032703
10. Kolios A, Immune balance and loss of immune tolerance, Lecture presented at: Inflammatory Brain Disorders Conference on 23 June 2024, San Diego, USA.
11. Rice CE, Rosanoff M, Dawson G, et al. Evaluating Changes in the Prevalence of the Autism Spectrum Disorders (ASDs). Public Health Rev. 2012;34(2):1-22. doi:10.1007/BF03391685
12. Pallanti S, Di Ponzio M. PANDAS/PANS in the COVID-19 Age: Autoimmunity and Epstein-Barr Virus Reactivation as Trigger Agents?. Children (Basel). 2023;10(4):648. Published 2023 Mar 30. doi:10.3390/children10040648
13 Toscano CVA, Barros L, Lima AB, Nunes T, Carvalho HM, Gaspar JM. Neuroinflammation in autism spectrum disorders: Exercise as a "pharmacological" tool. Neurosci Biobehav Rev. 2021;129:63-74. doi:10.1016/j.neubiorev.2021.07.023
14 Chiarotti F, Venerosi A. Epidemiology of Autism Spectrum Disorders: A Review of Worldwide Prevalence Estimates Since 2014. Brain Sci. 2020;10(5):274. Published 2020 May 1. doi:10.3390/brainsci10050274
15. Stone R. The inflamed brain. Science. 2024;384(6697):728-733. doi:10.1126/science.adq4311
16. D'Auria E, Minutoli M, Colombo A, et al. Allergy and autoimmunity in children: non-mutually exclusive diseases. A narrative review. Front Pediatr. 2023;11:1239365. Published 2023 Nov 2. doi:10.3389/fped.2023.1239365
17. Wang L, Wang B, Wu C, Wang J, Sun M. Autism Spectrum Disorder: Neurodevelopmental Risk Factors, Biological Mechanism, and Precision Therapy. Int J Mol Sci. 2023;24(3):1819. Published 2023 Jan 17. doi:10.3390/ijms24031819
18 Usui N, Kobayashi H, Shimada S. Neuroinflammation and Oxidative Stress in the Pathogenesis of Autism Spectrum Disorder. Int J Mol Sci. 2023;24(6):5487. Published 2023 Mar 13. doi:10.3390/ijms24065487
19. Mateos-Aparicio P, Rodríguez-Moreno A. The Impact of Studying Brain Plasticity. Front Cell Neurosci. 2019;13:66. Published 2019 Feb 27. doi:10.3389/fncel.2019.00066
20. Chen Z, Wang X, Zhang S, Han F. Neuroplasticity of children in autism spectrum disorder. Front Psychiatry. 2024;15:1362288. Published 2024 Apr 25. doi:10.3389/fpsyt.2024.1362288
21. de Souza DLS, Costa HMGES, Idalina Neta F, et al. Brain Neuroplasticity after Treatment with Antiseizure: A Review. Clin Psychopharmacol Neurosci. 2023;21(4):665-675. doi:10.9758/cpn.23.1058
22. Connery K, Tippett M, Delhey LM, et al. Intravenous immunoglobulin for the treatment of autoimmune encephalopathy in children with autism. Transl Psychiatry. 2018;8(1):148. Published 2018 Aug 10. doi:10.1038/s41398-018-0214-7
23. Citrigno L, Muglia M, Qualtieri A, et al. The Mitochondrial Dysfunction Hypothesis in Autism Spectrum Disorders: Current Status and Future Perspectives. Int J Mol Sci. 2020;21(16):5785. Published 2020 Aug 12. doi:10.3390/ijms21165785
24. Björkstén B, Sepp E, Julge K, Voor T, Mikelsaar M. Allergy development and the intestinal microflora during the first year of life. J Allergy Clin Immunol. 2001;108(4):516-520. doi:10.1067/mai.2001.118130
25. Hardin H, Shao W, Bernstein JA. An updated review of paediatric autoimmune neuropsychiatric disorders associated with Streptococcus/paediatric acute-onset neuropsychiatric syndrome, also known as idiopathic autoimmune encephalitis: What the allergist should know. Ann Allergy Asthma Immunol. 2023;131(5):567-575. doi:10.1016/j.anai.2023.08.022
26. Rossignol DA, Frye RE. A Systematic Review and Meta-Analysis of Immunoglobulin G Abnormalities and the Therapeutic Use of Intravenous Immunoglobulins (IVIG) in Autism Spectrum Disorder. J Pers Med. 2021;11(6):488. Published 2021 May 30. doi:10.3390/jpm11060488
27. Guglielmi G. Found: the dial in the brain that controls the immune system. Nature. Published online May 1, 2024. doi:10.1038/d41586-024-01259-2
28. Tan J, Smith CH, Goldman RD. Paediatric autoimmune neuropsychiatric disorders associated with streptococcal infections. Can Fam Physician. 2012;58(9):957-959.
29. Frye RE. Mitochondrial Dysfunction in Autism Spectrum Disorder: Unique Abnormalities and Targeted Treatments. Semin Pediatr Neurol. 2020;35:100829. doi:10.1016/j.spen.2020.100829
30. Boris M, Goldblatt A, Edelson SM. Improvements in children with autism treated with intravenous gamma globulin. J. Nutr. Environ. Med. 2005;15:169–176. doi: 10.1080/13590840600681827
31. Griffiths KK, Levy RJ. Evidence of Mitochondrial Dysfunction in Autism: Biochemical Links, Genetic-Based Associations, and Non-Energy-Related Mechanisms. Oxid Med Cell Longev. 2017;2017:4314025. doi:10.1155/2017/4314025
32. Frankovich J, Swedo S, Murphy T, et al. Clinical Management of Pediatric Acute-Onset Neuropsychiatric Syndrome: Part II-Use of Immunomodulatory Therapies. J Child Adolesc Psychopharmacol. 2017;27(7):574-593. doi:10.1089/cap.2016.0148
33. Personal experience.
34. Pfeiffer HCV, Wickstrom R, Skov L, et al. Clinical guidance for diagnosis and management of suspected Pediatric Acute-onset Neuropsychiatric Syndrome in the Nordic countries. Acta Paediatr. 2021;110(12):3153-3160. doi:10.1111/apa.15875
35. Melamed IR, Heffron M, Testori A, Lipe K. A pilot study of high-dose intravenous immunoglobulin 5% for autism: Impact on autism spectrum and markers of neuroinflammation. Autism Res. 2018;11(3):421-433. doi:10.1002/aur.1906