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Publikacje naukowe

Zalecenia dotyczące oceny i leczenia

Tran, P, Frankovich J, Van Mater H, Dale R, Or-Geva N, McHugh A, Thienemann M. “Pediatric Inflammatory Brain Disease” Complex Disorders in Pediatric Psychiatry. A Clinician’s Guide 1st Edition Editors: David I Driver & Shari Thomas. December 2017.

Cellucci T, Van Mater H, Graus F, Muscal E, Gallentine W, Klein-Gitelman MS, Benseler SM, Frankovich J, Gorman MP, Van Haren K, Dalmau J, Dale RC. Clinical approach to the diagnosis of autoimmune encephalitis in the pediatric patient. Neurol Neuroimmunol Neuroinflamm. 2020 Jan 17;7(2). pii: e663. doi: 10.1212/NXI.0000000000000663. Print 2020 Mar. PMID:31953309

Chang K*, Frankovich J*, Cooperstock M, Cunningham M, Latimer E, Murphy T, Pasternack M, Thienemann M, Williams K, Walter J, Swedo S. Clinical Evaluation of Youth with Pediatric Acute-Onset Neuropsychiatric Syndrome (PANS): Recommendations from the 2013 PANS Consensus Conference. Journal of Child and Adolescent Psychopharmacology, Vol. 25, No. 1, February 2015: 3-13. PMID: 25325534

Swedo S, Frankovich J, Murphy T. Overview of Treatment of Pediatric Acute-Onset Neuropsychiatric Syndrome. J of Child and Adolescent Psychopharmacology. Vol. 27, No.7, September 2017. doi:10.1089/cap/2017.0042

Thienemann M, Murphy T, Leckman J, Shaw R, Williams K, Kapphahn C, Frankovich J, Geller D, Bernstein G, Chang K, Elia J, Swedo S. Clinical Management of Pediatric Acute-Onset Neuropsychiatric Syndrome: Part I-Psychiatric and Behavioral Interventions. J of Child and Adolescent Psychopharmacology. Vol. 27, No.7, 566-573. September 2017. doi:10.1089/cap.2016.0145

Frankovich J, Swedo S, Murphy T, Dale R C, Agalliu D, Williams K, Daines M, Hornig M, Chugani H, Sanger T, Muscal E, Pasternack M, Cooperstock, et al. Clinical Management of Pediatric Acute-Onset Neuropsychiatric Syndrome: Part II—Use of Immunomodulatory Therapies. Journal of Child and Adolescent Psychopharmacology, Vol. 27, No. 7, 574-593. September 2017. doi: 10.1089/cap.2016.0148

Cooperstock M, Swedo SE, Pasternack M, Murphy T, and for the PANS/PANDAS Consortium. Clinical Management of Pediatric Acute-Onset Neuropsychiatric Syndrome: Part III—Treatment and Prevention of Infections. Journal of Child and Adolescent Psychopharmacology.Sep 2017.594-606.http://doi.org/10.1089/cap.2016.0151

Pfeiffer HCV, Wickstrom R, Skov L, Sørensen CB, Sandvig I, Gjone IH, Ygberg S, de Visscher C, Idring Nordstrom S, Herner LB, Hesselmark E, Hedderly T, Lim M, Debes NM. Clinical guidance for diagnosis and management of suspected Pediatric Acute-onset Neuropsychiatric Syndrome in the Nordic countries. Acta Paediatr. 2021 Dec;110(12):3153-3160. doi: 10.1111/apa.15875. Epub 2021 May 6. PMID: 33848371.

Johnson M, Ehlers S, Fernell E, Hajjari P, Wartenberg C, Wallerstedt SM. Anti-inflammatory, antibacterial and immunomodulatory treatment in children with symptoms corresponding to the research condition PANS (Pediatric Acute-onset Neuropsychiatric Syndrome): A systematic review. PLoS One. 2021 Jul 1;16(7):e0253844. doi: 10.1371/journal.pone.0253844. PMID: 34197525; PMCID: PMC8248649.

Pollak TA, Lennox BR, Müller S, Benros ME, Prüss H, Tebartz van Elst L, Klein H, Steiner J, Frodl T, Bogerts B, Tian L, Groc L, Hasan A, Baune BT, Endres D, Haroon E, Yolken R, Benedetti F, Halaris A, Meyer JH, Stassen H, Leboyer M, Fuchs D, Otto M, Brown DA, Vincent A, Najjar S, Bechter K. Autoimmune psychosis: An international consensus on an approach to the diagnosis and management of psychosis of suspected autoimmune origin. Lancet Psychiatry. 2020 Jan;7(1):93-108. doi: 10.1016/S2215-0366(19)30290-1. Epub 2019 Oct 24. Erratum in: Lancet Psychiatry. 2019 Dec;6(12):e31. PMID: 31669058.

Mahmud FH, Lteif AN, Renaud DL, Reed AM, Brands CK. Steroid-responsive encephalopathy associated with Hashimoto’s thyroiditis in an adolescent with chronic hallucinations and depression: case report and review. Pediatrics. 2003 Sep;112(3 Pt 1):686-90. doi: 10.1542/peds.112.3.686. PMID: 12949305

Aktywacja mikrogleju w PANS, PANDAS, OCD i Zespole Tourette'a

Frick L, Pittenger C. Microglial Dysregulation in OCD, Tourette Syndrome, and PANDAS. J Immunol Res. 2016;2016:8606057. doi:10.1155/2016/8606057

Kumar A, Williams MT, Chugani HT. Evaluation of Basal Ganglia and Thalamic Inflammation in Children With Pediatric Autoimmune Neuropsychiatric Disorders Associated With Streptococcal Infection and Tourette Syndrome: A Positron Emission Tomographic (PET) Study Using 11C-[R]-PK11195. J Child Neurol 30: 749-756, 2015.

Attwells S, Setiawan E, Wilson AA, et al. Inflammation in the Neurocircuitry of Obsessive-Compulsive Disorder. JAMA Psychiatry. 2017;74(8):833-840. doi:10.1001/jamapsychiatry.2017.1567

Alergie i zaburzenia pokarmowe o podłożu immunologicznym w PANS

Rosa J, Hernandez J, Sherr J, Smith B, Brown K, Farhadian B, Mahony T, McGhee S, Lewis D B, Thienemann M, Frankovich J. Allergic Diseases and Immune-Mediated Food Disorders in Pediatric Acute-Onset Neuropsychiatric Syndrome. Pediatric Allergy, Immunology, and Pulmonology, Vol 31, No.3. Sept 2018. doi: 10.1089/ped.2018.0888

Toufexis MD, Hommer R, Gerardi DM, et al. Disordered eating and food restrictions in children with PANDAS/PANS. J Child Adolesc Psychopharmacol. 2015;25(1):48-56. doi:10.1089/cap.2014.0063

Artykuły przeglądowe

Swedo SE, Leckman JF, Rose NR.  From Research Subgroup to Clinical Syndrome: Modifying the PANDAS Criteria to Describe PANS (Pediatric Acute-onset Neuropsychiatric Syndrome). Pediatr Therapeut 2:113. 2012. doi:10.4172/2161-0665.1000113

Swedo SE, Frankovich J, Murphy TM. Overview of Treatment of Pediatric Acute-Onset Neuropsychiatric Syndrome. Journal of Child and Adolescent Psychopharmacology. Sep 2017.562-565.http://doi.org/10.1089/cap.2017.0042

Murphy TK, Gerardi DM, Leckman JF. Pediatric acute-onset neuropsychiatric syndrome. Psychiatr Clin North Am. 2014 Sep;37(3):353-74. doi: 10.1016/j.psc.2014.06.001. PMID: 25150567.

Williams KA, Swedo SE. Post-infectious autoimmune disorders: Sydenham’s chorea, PANDAS and beyond. Brain Res. 2015 Aug 18;1617:144-54. doi: 10.1016/j.brainres.2014.09.071. Epub 2014 Oct 7. PMID: 25301689.

Autoprzeciwciała w PANS/PANDAS

Xu J, Liu RJ, Fahey S, et al. Antibodies From Children With PANDAS Bind Specifically to Striatal Cholinergic Interneurons and Alter Their Activity [published online ahead of print, 2020 Jun 16]. Am J Psychiatry. 2020;appiajp202019070698. doi:10.1176/appi.ajp.2020.19070698

Frick, L. R., Rapanelli, M., Jindachomthong, K., Grant, P., Leckman, J. F., Swedo, S., Williams, K., & Pittenger, C. (2018). Differential binding of antibodies in PANDAS patients to cholinergic interneurons in the striatum. Brain, behavior, and immunity, 69, 304–311. https://doi.org/10.1016/j.bbi.2017.12.004

Chain JL, Alvarez K, Mascaro-Blanco A, et al. Autoantibody Biomarkers for Basal Ganglia Encephalitis in Sydenham Chorea and Pediatric Autoimmune Neuropsychiatric Disorder Associated With Streptococcal Infections. Front Psychiatry. 2020;11:564. Published 2020 Jun 24. doi:10.3389/fpsyt.2020.00564

Kirvan CA, Swedo SE, Heuser JS, Cunningham MW. Mimicry and autoantibody-mediated neuronal cell signaling in Sydenham chorea. Nat Med. 2003 Jul;9(7):914-20. doi: 10.1038/nm892. PMID: 12819778.

Kirvan CA, Cox CJ, Swedo SE, Cunningham MW. Tubulin is a neuronal target of autoantibodies in Sydenham’s chorea. J Immunol 178: 7412-7421, 2007.

Kirvan CA, Swedo SE, Kurahara D, Cunningham MW. Streptococcal mimicry and antibody-mediated cell signaling in the pathogenesis of Sydenham’s chorea. Autoimmunity 39: 21-29, 2006

Kirvan CA, Swedo SE, Snider LA, Cunningham MW. Antibody-mediated neuronal cell signaling in behavior and movement disorders. J Neuroimmunol. 2006 Oct;179(1-2):173-9. doi: 10.1016/j.jneuroim.2006.06.017. Epub 2006 Jul 27. PMID: 16875742.

Cox CJ, Zuccolo AJ, Edwards EV, Mascaro-Blanco A, Alvarez K, Stoner J, Chang K, Cunningham MW. Antineuronal antibodies in a heterogeneous group of youth and young adults with tics and obsessive-compulsive disorder. J Child Adolesc Psychopharmacol 25: 76-85, 2015.

Shimasaki C, Frye RE, Trifiletti R, Cooperstock M, et al. Evaluation of the Cunningham Panel™ in pediatric autoimmune neuropsychiatric disorder associated with streptococcal infection (PANDAS) and pediatric acute-onset neuropsychiatric syndrome (PANS): Changes in antineuronal antibody titers parallel changes in patient symptoms. J Neuroimmunol. 2020 Feb 15;339:577138. doi: 10.1016/j.jneuroim.2019.577138. Epub 2019 Dec 15. PMID: 31884258.

Frye RE, Shimasaki C. Reliability of the Cunningham Panel. Transl Psychiatry. 2019 Apr 8;9(1):129. doi: 10.1038/s41398-019-0462-1. PMID: 30962420; PMCID: PMC6453973.

Badania obserwacyjne, badania przypadków i badania kliniczno-kontrolne

Swedo SE, Leonard HL, Garvey M, Mittleman B, Allen AJ, Perlmutter S, Lougee L, Dow S, Zamkoff J, Dubbert BK. Pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections: clinical description of the first 50 cases. Am J Psychiatry. 1998 Feb;155(2):264-71. doi: 10.1176/ajp.155.2.264. Erratum in: Am J Psychiatry 1998 Apr;155(4):578. PMID: 9464208.

Murphy TK, Patel PD, McGuire JF, et al. Characterization of the pediatric acute-onset neuropsychiatric syndrome phenotype. J Child Adolesc Psychopharmacol. 2015;25(1):14-25. doi:10.1089/cap.2014.0062

Frankovich J, Thienemann M, Pearlstein J, Crable A, Brown K, Chang K. Multidisciplinary clinic dedicated to treating youth with pediatric acute-onset neuropsychiatric syndrome: presenting characteristics of the first 47 consecutive patients. J Child Adolesc Psychopharmacol. 2015 Feb;25(1):38-47. doi: 10.1089/cap.2014.0081. PMID: 25695943; PMCID: PMC4340335.

Frankovich J, Thienemann M, Rana S, Chang K. Five Youth with Pediatric Acute-Onset Neuropsychiatric Syndrome of Different Etiologies. Journal of Child and Adolescent Psychopharmacology, Vol. 25, No. 1, February 2015: 31-37. PMID: 25695942

Gromark C, Harris RA, Wickström R, et al. Establishing a Pediatric Acute-Onset Neuropsychiatric Syndrome Clinic: Baseline Clinical Features of the Pediatric Acute-Onset Neuropsychiatric Syndrome Cohort at Karolinska Institutet. J Child Adolesc Psychopharmacol. 2019;29(8):625-633. doi:10.1089/cap.2018.0127

Gromark C, Hesselmark E, Djupedal IG, et al. A Two-to-Five Year Follow-Up of a Pediatric Acute-Onset Neuropsychiatric Syndrome Cohort [published online ahead of print, 2021 Feb 9]. Child Psychiatry Hum Dev. 2021;1-11. doi:10.1007/s10578-021-01135-4

Mahony T, Sidell D, Gans, H, Cooperstock M, Brown K, Cheung J, Farhadian B, Gustafson M, Thienemann M, Frankovich J. Palatal Petechiae in the Absence of Group A Streptococcus in Pediatric Patients with Acute-onset Neuropsychiatric Deterioration: A Cohort Study. Journal of Child and Adolescent Psychopharmacology, Vol. 27, No. 7, April 2017, online ahead of print. DOI: 10.1089/cap.2016.0153

Gagliano A, Galati C, Ingrassia M, Ciuffo M, Alquino MA, Tanca MG, Carucci S, Zuddas A, Grossi E. Pediatric Acute-Onset Neuropsychiatric Syndrome: A Data Mining Approach to a Very Specific Constellation of Clinical Variables. J Child Adolesc Psychopharmacol. 2020 Oct;30(8):495-511. doi: 10.1089/cap.2019.0165. Epub 2020 May 28. PMID: 32460516.

Swedo SE, Seidlitz J, Kovacevic M, et al. Clinical presentation of pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections in research and community settings. J Child Adolesc Psychopharmacol. 2015;25(1):26-30. doi:10.1089/cap.2014.0073

Lepri G, Rigante D, Bellando Randone S, Meini A, Ferrari A, Tarantino G, Cunningham MW, Falcini F. Clinical-Serological Characterization and Treatment Outcome of a Large Cohort of Italian Children with Pediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcal Infection and Pediatric Acute Neuropsychiatric Syndrome. J Child Adolesc Psychopharmacol. 2019 Oct;29(8):608-614. doi: 10.1089/cap.2018.0151. Epub 2019 May 29. PMID: 31140830.

Murgia F, Gagliano A, Tanca MG, Or-Geva N, Hendren A, Carucci S, Pintor M, Cera F, Cossu F, Sotgiu S, Atzori L, Zuddas A. Metabolomic Characterization of Pediatric Acute-Onset Neuropsychiatric Syndrome (PANS). Front Neurosci. 2021 May 28;15:645267. doi: 10.3389/fnins.2021.645267. PMID: 34121984; PMCID: PMC8194687.

Johnson M, Fernell E, Preda I, et al. Paediatric acute-onset neuropsychiatric syndrome in children and adolescents: an observational cohort study. The Lancet Child & Adolescent Health. 2019;3(3):175-180. doi:10.1016/s2352-4642(18)30404-8

Tucker DM, Leckman JF, Scahill L, Wilf GE, LaCamera R, Cardona L, Cohen P, Heidmann S, Goldstein J, Judge J, Snyder E, Bult A, Peterson BS, King R, Lombroso P: A putative poststreptococcal case of OCD with chronic tic disorder, not otherwise specified. J Am Acad Child Adolesc Psychiatry 35: 1684-1691, 1996. DOI: 10.1097/00004583-199612000-00022

Waldrep DA: Two cases of ADHD following GABHS infection: a PANDAS subgroup? J Am Acad Child Adolesc Psychiatry 41: 1273-1274, 2002.

COVID, POTS i PANS

Vernino S, Stiles LE. Autoimmunity in postural orthostatic tachycardia syndrome: Current understanding. Auton Neurosci. 2018 Dec;215:78-82. doi: 10.1016/j.autneu.2018.04.005. Epub 2018 Apr 26. PMID: 29909990.

Pavone P, Ceccarelli M, Marino S, Caruso D, Falsaperla R, Berretta M, Rullo EV, Nunnari G. SARS-CoV-2 related paediatric acute-onset neuropsychiatric syndrome. Lancet Child Adolesc Health. 2021 Jun;5(6):e19-e21. doi: 10.1016/S2352-4642(21)00135-8. Epub 2021 May 4. PMID: 33961798; PMCID: PMC8096321.

Miglis MG, Prieto T, Shaik R, Muppidi S, Sinn DI, Jaradeh S. A case report of postural tachycardia syndrome after COVID-19. Clin Auton Res. 2020 Oct;30(5):449-451. doi: 10.1007/s10286-020-00727-9. Epub 2020 Sep 3. PMID: 32880754; PMCID: PMC7471493.

Chan A, Gao J, Houston M, Willett T, Farhadian B, Silverman M, Tran P, Jaradeh S, Thienemann M, Frankovich J. Children With PANS May Manifest POTS. Front Neurol. 2022 Apr 26;13:819636. doi: 10.3389/fneur.2022.819636. PMID: 35557616; PMCID: PMC9086964.

Diagnostyka różnicowa

Yu CH, Lee MB, Tseng MM, Liao SC. Obsessive-compulsive symptoms as a manifestation of neuropsychiatric systemic lupus erythematosus. J Formos Med Assoc 107: 68-72, 2008.

Mercadante MT, Diniz JB, Hounie AG, Ferrao Y, Alvarenga P, Brotto S, Miguel EC. Obsessive-compulsive spectrum disorders in rheumatic fever patients. J Neuropsychiatry Clin Neurosci 17: 544-547, 2005.

Mahmud FH, Lteif AN, Renaud DL, Reed AM, Brands CK. Steroid-responsive encephalopathy associated with Hashimoto’s thyroiditis in an adolescent with chronic hallucinations and depression: case report and review. Pediatrics 112: 686-690, 2003.

Muller N, Riedel M, Forderreuther S, Blendinger C, Abele-Horn M. Tourette’s syndrome and mycoplasma pneumoniae infection. Am J Psychiatry 157: 481-482, 2000.

Peatfield RC. Basal ganglia damage and subcortical dementia after possible insidious Coxsackie virus encephalitis. Acta Neurol Scand 76: 340-345, 1987.

Beleza P, Soares-Fernandes J, Jordao MJ, Almeida F. From juvenile parkinsonism to encephalitis lethargica, a new phenotype of post-streptococcal disorders: case report. Eur J Paediatr Neurol 12: 505-507, 2008.

Casanova MF, Crapanzano KA, Mannheim G, Kruesi M. Sydenham’s chorea and schizophrenia: a case report. Schizophr Res 16: 73-76, 1995.

Mink J, Kurlan R. Acute postinfectious movement and psychiatric disorders in children and adolescents. J Child Neurol 26: 214-217, 2011.

Długoterminowe wyniki w PANS/PANDAS

Leon, J., Hommer, R., Grant, P. et al. Longitudinal outcomes of children with pediatric autoimmune neuropsychiatric disorder associated with streptococcal infections (PANDAS). Eur Child Adolesc Psychiatry 27, 637–643 (2018). https://doi.org/10.1007/s00787-017-1077-9

Gromark C, Hesselmark E, Djupedal IG, et al. A Two-to-Five Year Follow-Up of a Pediatric Acute-Onset Neuropsychiatric Syndrome Cohort [published online ahead of print, 2021 Feb 9]. Child Psychiatry Hum Dev. 2021;1-11. doi:10.1007/s10578-021-01135-4

Genetyka

Chan A, Phu T, Farhadian B, Willet T, Thienemann M, Frankovich J. Familial Clustering of Immune-Mediated Diseases in Children with Abrupt-Onset Obsessive Compulsive Disorder. J Child Adolesc Psychopharmacol. 2020 Apr 20. Doi: 10.1089/cap.2019.0167 PMID 32311283

Bey AL, Gorman MP, Gallentine W, Kohlenberg TM, Frankovich J, Jiang YH, Van Haren K. Subacute Neuropsychiatric Syndrome in Girls With SHANK3 Mutations Responds to Immunomodulation. Pediatrics. 2020 Feb;145(2). pii: e20191490. doi: 10.1542/peds.2019-1490.

Lewin AB, Storch EA, Murphy TK. Pediatric autoimmune neuropsychiatric disorders associated with Streptococcus in identical siblings. J Child Adolesc Psychopharmacol 21: 177-182, 2011.

Mataix-Cols D, Frans E, Pérez-Vigil A, et al. A total-population multigenerational family clustering study of autoimmune diseases in obsessive-compulsive disorder and Tourette’s/chronic tic disorders. Mol Psychiatry. 2018;23(7):1652-1658. doi:10.1038/mp.2017.215

HLA PANS

Fernell E, Sundin M, Fasth A, Dinkler L, Galazka M, Gillberg C, Johnson M. Paediatric Acute onset Neuropsychiatric Syndrome: Exploratory study finds no evidence of HLA class II association but high rate of autoimmunity in first-degree relatives. Acta Paediatr. 2021 Feb 10. doi: 10.1111/apa.15805.

Immunomodulacja

Frankovich J, Swedo S, Murphy T, Dale R C, Agalliu D, Williams K, Daines M, Hornig M, Chugani H, Sanger T, Muscal E, Pasternack M, Cooperstock, et al. Clinical Management of Pediatric Acute-Onset Neuropsychiatric Syndrome: Part II—Use of Immunomodulatory Therapies. Journal of Child and Adolescent Psychopharmacology, Vol. 27, No. 7, 574-593. September 2017. doi: 10.1089/cap.2016.0148

Perlmutter SJ, Leitman SF, Garvey MA, Hamburger S, Feldman E, Leonard HL, Swedo SE. Therapeutic plasma exchange and intravenous immunoglobulin for obsessive-compulsive disorder and tic disorders in childhood. Lancet. 1999 Oct 2;354(9185):1153-8. doi: 10.1016/S0140-6736(98)12297-3. PMID: 10513708.

Williams KA, Swedo SE, Farmer CA, et al. Randomized, Controlled Trial of Intravenous Immunoglobulin for Pediatric Autoimmune Neuropsychiatric Disorders Associated With Streptococcal Infections. J Am Acad Child Adolesc Psychiatry. 2016;55(10):860-867.e2. doi:10.1016/j.jaac.2016.06.017

Brown K, Farmer C, Freeman GM, Spartz E, Farhadian B, Thienemann M, Frankovich J. Effect of Early and Prophylactic Nonsteroidal Anti-Inflammatory Drugs (NSAIDS) on Flare Duration in Pediatric Acute-onset Neuropsychiatric Syndrome (PANS): An Observational Study of Patients Followed by an Academic Community-Based Pediatric Acute-onset Neuropsychiatric Syndrome Clinic. Journal of Child and Adolescent Psychopharmacology. Vol. 27, No. 7, July 2017, online ahead of print. PMID: 28696786. DOI: 10.1089/cap.2016.0193

Spartz E, Freeman GM, Brown K, Farhadian B, Thienemann M, Frankovich J. Course of Neuropsychiatric Symptoms after Introduction or Removal of Non-Steroidal Anti-Inflammatory Drugs: A Pediatric Observational Study. Journal of Child and Adolescent Psychopharmacology, Vol. 27, No. 7, July 2017, online ahead of print. PMID: 28696783. DOI: 10.1089/cap.2016.0179

Brown K, Farmer C, Farhadian B, Hernandez J, Thienemann M, Frankovich J. Pediatric Acute-onset Neuropsychiatric Syndrome (PANS) – Response to Oral Corticosteroid Bursts: An Observational Study of Patients in an Academic Community-Based PANS Clinic. Journal of Child and Adolescent Psychopharmacology, Vol. 27, No.7, July 2017, online ahead of print. PMID: 28714753. DOI: 10.1089/cap.2016.0139

Latimer ME, L’Etoile N, Seidlitz J, Swedo SE. Therapeutic plasma apheresis as a treatment for 35 severely ill children and adolescents with pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections. J Child Adolesc Psychopharmacol. 2015;25(1):70-75. doi:10.1089/cap.2014.0080

Pavone P, Falsaperia R, Nicita F, Zecchini A, et al Pediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcal Infection (PANDAS): Clinical Manifestations, IVIG Treatment Outcomes, Results from a Cohort of Italian Patients. Neuropsych 2018; 8 (3)

Pavone P, Falsaperla R, Cacciaguerra G, et al PANS/PANDAS: Clinical Experience in IVIG Treatment and State of the Art in Rehabilitation Approaches. NeuroSci 2020, 1, 75-84; doi:10.3390/neurosci1020007

Kovacevic M, Grant P, Swedo SE. Use of intravenous immunoglobulin in the treatment of twelve youths with pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections. J Child Adolesc Psychopharmacol. 2015;25(1):65-69. doi:10.1089/cap.2014.0067

Infekcje i antybiotyki w PANS/PANDAS

Murphy TK, Brennan EM, Johnco C, Parker-Athill EC, Miladinovic B, Storch EA, Lewin AB. A Double-Blind Randomized Placebo-Controlled Pilot Study of Azithromycin in Youth with Acute-Onset Obsessive-Compulsive Disorder. J Child Adolesc Psychopharmacol. 2017 Sep;27(7):640-651. doi: 10.1089/cap.2016.0190. Epub 2017 Mar 30. PMID: 28358599.

Murphy TK, Snider LA, Mutch PJ, Harden E, Zaytoun A, Edge PJ, Storch EA, Yang MC, Mann G, Goodman WK, Swedo SE. Relationship of movements and behaviors to Group A Streptococcus infections in elementary school children. Biol Psychiatry. 2007 Feb 1;61(3):279-84. doi: 10.1016/j.biopsych.2006.08.031. Epub 2006 Nov 27. PMID: 17126304.

Orlovska S, Vestergaard CH, Bech BH, Nordentoft M, Vestergaard M, Benros ME. Association of Streptococcal Throat Infection With Mental Disorders: Testing Key Aspects of the PANDAS Hypothesis in a Nationwide Study. JAMA Psychiatry. 2017 Jul 1;74(7):740-746. doi: 10.1001/jamapsychiatry.2017.0995. PMID: 28538981; PMCID: PMC5710247.

Chan A, Frankovich J. Infections, Antibiotics, and Mental Health Deteriorations. J Child Adolesc Psychopharmacol. 2019 Jul 29. doi: 10.1089/cap.2019.0100. [Epub ahead of print]

Mahony T, Sidell D, Gans H, Brown K, Farhadian B, Gustafson M, Sherr J, Thienemann M, Frankovich J. Improvement of Psychiatric Symptoms in Youth Following Resolution of Sinusitis. International Journal of Pediatric Otorhinolaryngology, Vol. 92, January 2017: 38-44. DOI: 10.1016/j.ijporl.2016.10.034

Murphy TK, Parker-Athill EC, Lewin AB, Storch EA, Mutch PJ. Cefdinir for recent-onset pediatric neuropsychiatric disorders: a pilot randomized trial. J Child Adolesc Psychopharmacol. 2015 Feb;25(1):57-64. doi: 10.1089/cap.2014.0010. Epub 2014 Oct 9. PMID: 25299463; PMCID: PMC4340343.

Swedo S, Menendez ChM, Cunningham MW. Chapter 26 Pediatric Autoimmune Neuropsychiatric Disorders Associated with Streptococcal Infections (PANDAS). Streptococcus pyogenes: Basic Biology to Clinical Manifestations [Internet]. 2nd edition. 

Interwencje psychiatryczne i behawioralne

Thienemann M, Park M, Chan A, Frankovich J. Patients with abrupt early-onset OCD due to PANS tolerate lower doses of antidepressants and antipsychotics. Journal of Psychiatric Research. 2021 Jan;135:270-278. doi:10.1016/j.jpsychires.2021.01.022 doi:10.1016/j.jpsychires.2021.01.022

Thienemann M, Murphy T, Leckman J, Shaw R, Williams K, Kapphahn C, Frankovich J, Geller D, Bernstein G, Chang K, Elia J, Swedo S. Clinical Management of Pediatric Acute-Onset Neuropsychiatric Syndrome: Part I-Psychiatric and Behavioral Interventions. J of Child and Adolescent Psychopharmacology. Vol. 27, No.7, 566-573. September 2017. doi:10.1089/cap.2016.0145

Nadeau JM, Jordan C, Selles RR, et al. A pilot trial of cognitive-behavioral therapy augmentation of antibiotic treatment in youth with pediatric acute-onset neuropsychiatric syndrome-related obsessive-compulsive disorder. J Child Adolesc Psychopharmacol. 2015;25(4):337-343. doi:10.1089/cap.2014.0149

Storch EA, Murphy TK, Geffken GR, Mann G, Adkins J, Merlo LJ, Duke D, Munson M, Swaine Z, Goodman WK. Cognitive-behavioral therapy for PANDAS-related obsessive-compulsive disorder: findings from a preliminary waitlist controlled open trial. J Am Acad Child Adolesc Psychiatry. 2006 Oct;45(10):1171-8. doi: 10.1097/01.chi.0000231973.43966.a0. PMID: 17003662.

Kwas folinowy

Wells, L.; O’Hara, N.; Frye, R.E.; Hullavard, N.; Smith, E. Folate Receptor Alpha Autoantibodies in the Pediatric Acute-Onset Neuropsychiatric Syndrome (PANS) and Pediatric Autoimmune Neuropsychiatric Disorders Associated with Streptococcal Infections (PANDAS) Population J. Pers. Med. 2024, 14, 166.

Frye RE, McCarty PJ, Werner BA, Scheck AC, Collins HL, Adelman SJ, Rossignol DA, Quadros EV. Binding Folate Receptor Alpha Autoantibody Is a Biomarker for Leucovorin Treatment Response in Autism Spectrum Disorder J Pers Med. 1 Jan 2024.

Frye RE, Lane A, Worner A, Werner BA, McCarty PJ, Scheck AC, Collins HL, Adelman SJ, Quadros EV, Rossignol DA. The Soluble Folate Receptor in Autism Spectrum Disorder: Relation to Autism Severity and Leucovorin Treatment. J Pers Med. 2022 Dec 8;12(12):2033. doi: 10.3390/jpm12122033. PMID: 36556254; PMCID: PMC9786140.

Rossignol DA, Frye RE. Cerebral Folate Deficiency, Folate Receptor Alpha Autoantibodies and Leucovorin (Folinic Acid) Treatment in Autism Spectrum Disorders: A Systematic Review and Meta-Analysis J Pers Med. 2021 Nov 3;11(11):1141. doi: 10.3390/jpm11111141. Erratum in: J Pers Med. 2022 Apr 29;12(5): PMID: 34834493; PMCID: PMC8622150.

Frye RE, Rossignol DA, Scahill L, McDougle CJ, Huberman H, Quadros EV. Treatment of Folate Metabolism Abnormalities in Autism Spectrum Disorder Semin Pediatr Neurol. 2020 Oct;35:100835. doi: 10.1016/j.spen.2020.100835. Epub 2020 Jun 25. PMID: 32892962; PMCID: PMC7477301.

Frye RE, Slattery J, Delhey L, Furgerson B, Strickland T, Tippett M, Sailey A, Wynne R, Rose S, Melnyk S, Jill James S, Sequeira JM, Quadros EV. Folinic acid improves verbal communication in children with autism and language impairment: a randomized double-blind placebo-controlled trial. Mol Psychiatry. 2018 Feb;23(2):247-256. doi: 10.1038/mp.2016.168. Epub 2016 Oct 18. PMID: 27752075; PMCID: PMC5794882.

Frye RE, Slattery JC, Quadros EV. Folate metabolism abnormalities in autism: potential biomarkers Biomark Med. 2017 Aug;11(8):687-699. doi: 10.2217/bmm-2017-0109. Epub 2017 Aug 3. PMID: 28770615.

Frye RE, Wynne R, Rose S, Slattery J, Delhey L, Tippett M, Kahler SG, Bennuri SC, Melnyk S, Sequeira JM, Quadros EV. Thyroid dysfunction in children with autism spectrum disorder is associated with folate receptor α autoimmune disorder. J Neuroendocrinol. 2017; 29(3).

Frye RE, Delhey L, Slattery J, Tippett M, Wynne R, Rose S, Kahler SG, Bennuri SC, Melnyk S, Sequeira JM, Quadros E. Front Blocking and Binding Folate Receptor Alpha Autoantibodies Identify Novel Autism Spectrum Disorder Subgroups Neurosci. 2016; 10:80.

Frye RE, Melnyk S, Fuchs G, Reid T, Jernigan S, Pavliv O, Hubanks A, Gaylor DW, Walters L, James SJ. Effectiveness of methylcobalamin and folinic Acid treatment on adaptive behavior in children with autistic disorder is related to glutathione redox status. Autism Res Treat. 2013;2013:609705. doi: 10.1155/2013/609705. Epub 2013 Oct 12. PMID: 24224089; PMCID: PMC3810468.

Frye RE, Sequeira JM, Quadros EV, James SJ, Rossignol DA. Cerebral folate receptor autoantibodies in autism spectrum disorder. Mol Psychiatry. 2013; 18(3):369-81.

Bobrowski-Khoury N, Ramaekers VT, Sequeira JM, Quadros EV. Folate Receptor Alpha Autoantibodies in Autism Spectrum Disorders: Diagnosis, Treatment and Prevention. J Pers Med. 2021 Jul 24;11(8):710. doi: 10.3390/jpm11080710.

Ramaekers VT,Quadros EV, Sequeira JM. Role of folate receptor autoantibodies in infantile autism. Mol Psychiatry. 2013 Mar;18(3):270-1. doi: 10.1038/mp.2012.22. Epub 2012 Apr 10.

Ramaekers VTh, Segers K,Sequeira JM, Koenig M,Van Maldergem L, Bours V, Kornak U, Quadros EV. Genetic assessment and folate receptor autoantibodies in infantile-onset cerebral folate deficiency (CFD) syndrome. Mol Genet Metab. 2018 May;124(1):87-93. doi: 10.1016/j.ymgme.2018.03.001. Epub 2018 Mar 3.

Wells L, O’Hara N, Frye RE, Hullavard N, Smith E. Folate Receptor Alpha Autoantibodies in the Pediatric Acute-Onset Neuropsychiatric Syndrome (PANS) and Pediatric Autoimmune Neuropsychiatric Disorders Associated with Streptococcal Infections (PANDAS) Population. J Pers Med. 2024 Jan 31;14(2):166. doi: 10.3390/jpm14020166.

Ramaekers VTh, Quadros EV. Cerebral Folate Deficiency Syndrome: Early Diagnosis, Intervention and Treatment Strategies. Nutrients. 2022 Jul 28;14(15):3096. doi: 10.3390/nu14153096.

Ramaekers VT, Thöny B, Sequeira JM, Ansseau M, Philippe P, Boemer F, Bours V, Quadros EV. Folinic acid treatment for schizophrenia associated with folate receptor autoantibodies. Mol Genet Metab. 2014 Dec;113(4):307-14. doi: 10.1016/j.ymgme.2014.10.002. Epub 2014 Oct 12.

Hong-Fang Yuan, Kai Zhao, Yu Zang, Chun-Yan Liu, Zhi-Yong Hu, Jia-Jing Wei, Ting Zhou, Ying Li, Hui-Ping Zhang. Effect of folate deficiency on promoter methylation and gene expression of Esr1, Cav1, and Elavl1, and its influence on spermatogenesis. Oncotarget. 2017 Apr 11;8(15):24130-24141. doi: 10.18632/oncotarget.15731.

Altmäe S, Stavreus-Evers A, Ruiz JR, Laanpere M, Syvänen T, Yngve A, Salumets A, Nilsson TK. Variations in folate pathway genes are associated with unexplained female infertility. Fertil Steril. 2010 Jun;94(1):130-7. doi: 10.1016/j.fertnstert.2009.02.025. Epub 2009 Mar 26.

Ramaekers VT, Blau N, Sequeira JM, Nassogne M-C, Quadros EV. Folate receptor autoimmunity and cerebral folate deficiency in low-functioning autism with neurological deficits. Neuropediatrics. 2007 Dec;38(6):276-81. doi: 10.1055/s-2008-1065354.

Renard E, Leheup B, Guéant-Rodriguez R-M, Oussalah A, Quadros EV, Guéant J-L. Folinic acid improves the score of Autism in the EFFET placebo-controlled randomized trial. Biochimie. 2020 Jun:173:57-61. doi: 10.1016/j.biochi.2020.04.019. Epub 2020 May 7.

Gordon N. Cerebral folate deficiency. Developmental Medicine & Child Neurology: Volume 51, Issue 3 https://doi.org/10.1111/j.1469-8749.2008.03185.x

Ramaekers VT, Rothenberg SP, Sequeira JM, Opladen T, Blau N, Quadros EV, Selhub J. Autoantibodies to Folate Receptors in the Cerebral Folate Deficiency Syndrome. N Engl J Med 2005;352:1985-1991 DOI: 10.1056/NEJMoa043160 VOL. 352 NO. 19

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Niski poziom ferrytyny i anemia w PANS

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Obciążenie opiekuna

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