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Bejerot, S., Hesselmark, E., Mobarrez, F., Wallén, H., Hietala, M. A., Nybom, R. & Wetterberg, L. (2019). Neuromyelitis optica spectrum disorder with increased aquaporin-4 microparticles prior to autoantibodies in cerebrospinal fluid: a case report. Journal of Medical Case Reports, 13(1), Article ID 27.
Open this publication in new window or tab >>Neuromyelitis optica spectrum disorder with increased aquaporin-4 microparticles prior to autoantibodies in cerebrospinal fluid: a case report
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2019 (English)In: Journal of Medical Case Reports, ISSN 1752-1947, E-ISSN 1752-1947, Vol. 13, no 1, article id 27Article in journal (Refereed) Published
Abstract [en]

BACKGROUND: Neuromyelitis optica spectrum disorders are severe autoimmune inflammatory diseases of the central nervous system associated with the presence of immunoglobulin G antibodies against the water channel protein aquaporin-4. During exacerbation, specific aquaporin-4 immunoglobulin G may be produced intrathecally. We measured extracellular aquaporin-4 microparticles in the cerebrospinal fluid of a patient who later developed the typical symptoms and signs of a neuromyelitis optica spectrum disorder.

CASE PRESENTATION: A 17-year-old South American girl developed acute severe motor and vocal tics and difficulties in walking, peripheral numbness, muscle pain, and bilateral headache. At age 22, she had a multitude of motor and psychiatric symptoms. Over the years, she fulfilled the diagnostic criteria for anorexia nervosa, depression, sleep disorder, obsessive-compulsive disorder, generalized anxiety disorder, panic disorder, agoraphobia, social anxiety disorder, development coordination disorder, attention-deficit/hyperactivity disorder, hypomania, pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections, conversion disorder, psychosis, and schizotypal personality syndrome. At age 24, she was found to have elevated titers of aquaporin-4 antibodies in serum, suggestive of probable neuromyelitis optica. She subsequently developed visual impairment, and swollen optic nerves were verified by magnetic resonance imaging. She was thus treated with a chimeric monoclonal antibody targeted against the pan-B-cell marker CD20 (rituximab), and almost all symptoms, including the psychiatric symptoms, rapidly decreased. We found a significant increase of extracellular microparticles of aquaporin-4 in cerebrospinal fluid sampled from our patient when she was 22 years old, 2 years before the full clinical development of neuromyelitis optica.

CONCLUSIONS: Microparticles of aquaporin-4 represent subcellular arrangements that may influence the pathogenesis of neuromyelitis optica spectrum disorders and may serve as biomarkers for the underlying cellular disturbances. The increase of aquaporin-4 microparticles in cerebrospinal fluid may be used for early diagnostic purposes; for prevention; and for evaluation of effective treatment, long-term follow-up studies, and elucidating the pathophysiology in neuromyelitis optica spectrum disorders. Further studies of aquaporin-4 microparticles in cerebrospinal fluid of patients with neuromyelitis optica and similar neuropsychiatric disorders are thus called for.

Place, publisher, year, edition, pages
BioMed Central, 2019
Keywords
Antibodies, Aquaporin-4, Case report, Conversion disorder, La belle indifférence, Microparticles, Neuromyelitis optica spectrum disorder, Obsessive-compulsive disorder, Pediatric autoimmune neuropsychiatric disorders
National Category
Rheumatology and Autoimmunity
Identifiers
urn:nbn:se:oru:diva-72040 (URN)10.1186/s13256-018-1929-z (DOI)30696485 (PubMedID)2-s2.0-85060700660 (Scopus ID)
Available from: 2019-02-12 Created: 2019-02-12 Last updated: 2019-02-12Bibliographically approved
Bejerot, S. & Hesselmark, E. (2019). The Cunningham Panel is an unreliable biological measure [Letter to the editor]. Translational Psychiatry, 9(1), Article ID 49.
Open this publication in new window or tab >>The Cunningham Panel is an unreliable biological measure
2019 (English)In: Translational Psychiatry, ISSN 2158-3188, E-ISSN 2158-3188, Vol. 9, no 1, article id 49Article in journal, Letter (Refereed) Published
Place, publisher, year, edition, pages
Nature Publishing Group, 2019
National Category
Otorhinolaryngology Pediatrics
Identifiers
urn:nbn:se:oru:diva-72037 (URN)10.1038/s41398-019-0413-x (DOI)30705260 (PubMedID)2-s2.0-85060934571 (Scopus ID)
Available from: 2019-02-12 Created: 2019-02-12 Last updated: 2019-02-12Bibliographically approved
Bilevicute-Ljunger, I., Maroti, D. & Bejerot, S. (2018). Patients with chronic fatigue syndrome do not score higher on the autism-spectrum quotient than healthy controls: comparison with autism spectrum disorder. Scandinavian Journal of Psychology, 59(4), 428-432
Open this publication in new window or tab >>Patients with chronic fatigue syndrome do not score higher on the autism-spectrum quotient than healthy controls: comparison with autism spectrum disorder
2018 (English)In: Scandinavian Journal of Psychology, ISSN 0036-5564, E-ISSN 1467-9450, Vol. 59, no 4, p. 428-432Article in journal (Refereed) Published
Abstract [en]

Background: Clinically, there is an overlap of several symptoms of chronic fatigue syndrome (CFS) and autism spectrum disorder (ASD), including fatigue; brain “fog”; cognitive impairments; increased sensitivity to sound, light, and odour; increased pain and tenderness; and impaired emotional contact.

Methods: Adults with CFS (n = 59) or ASD (n = 50) and healthy controls (HC; n = 53) were assessed with the Autism-Spectrum Quotient (AQ) in a cross-sectional study. Non-parametric analysis was used to compare AQ scores among the groups. Univariate analysis of variance (ANCOVA) was used to identify if age, sex, or diagnostic group influenced the differences in scores.

Results: Patients with ASD scored significantly higher on the AQ than the CFS group and the HC group. No differences in AQ scores were found between the CFS and HC groups. AQ results were influenced by the diagnostic group but not by age or sex, according to ANCOVA.    

Conclusions: Despite clinical observations of symptom overlap between ASD and CFS, adult patients with CFS report few autistic traits in the self-report instrument, the AQ. The choice of instrument to assess autistic traits may influence the results.

Place, publisher, year, edition, pages
John Wiley & Sons, 2018
Keywords
autism spectrum disorder, chronic fatigue syndrome, Autism-Spectrum Quotient
National Category
Psychology (excluding Applied Psychology)
Identifiers
urn:nbn:se:oru:diva-65829 (URN)10.1111/sjop.12451 (DOI)000437295200009 ()29738079 (PubMedID)2-s2.0-85049527625 (Scopus ID)
Available from: 2018-03-15 Created: 2018-03-15 Last updated: 2018-07-25Bibliographically approved
Sigra, S., Hesselmark, E. & Bejerot, S. (2018). Treatment of PANDAS and PANS: a systematic review. Neuroscience and Biobehavioral Reviews, 86, 51-56
Open this publication in new window or tab >>Treatment of PANDAS and PANS: a systematic review
2018 (English)In: Neuroscience and Biobehavioral Reviews, ISSN 0149-7634, E-ISSN 1873-7528, Vol. 86, p. 51-56Article, review/survey (Refereed) Published
Abstract [en]

Pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections (PANDAS) are a subtype of acute-onset obsessive-compulsive disorder (OCD) thought to be caused by an autoimmune response to group A streptococcal infection. Based on this proposed pathophysiology, alternative treatments for acute-onset OCD have been introduced, including antibiotics and immunomodulatory interventions. However, the literature on treatment of PANDAS is diverse, and clinical consensus regarding optimal treatment strategy is lacking. We conducted a systematic review of articles in PubMed, Cochrane Library, and Scopus that addressed treatment for PANDAS and related disorders. Twelve research studies involving the following treatments met inclusion criteria: penicillin, azithromycin, intravenous immunoglobulin, plasma exchange, tonsillectomy, cognitive behavior therapy, NSAID and corticosteroids. In addition, 65 case reports in which patients received immunomodulatory treatments, antibiotics, and/or psychotropics were identified. We determined that rigorously conducted research regarding treatments for PANDAS is scarce, and published studies have a high risk of bias. Further research is needed in which promising treatment strategies for PANDAS and other variants of OCD with proposed autoimmune etiology are rigorously investigated.

Place, publisher, year, edition, pages
Elsevier, 2018
Keywords
Pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections; PANDAS; Pediatric acute-onset neuropsychiatric syndrome; PANS; PITAND; Childhood acute neuropsychiatric symptoms; CANS; Obsessive-compulsive disorder; OCD; Obsessive-compulsive symptoms; Treatment; Therapy; Systematic review
National Category
Psychiatry Neurology
Identifiers
urn:nbn:se:oru:diva-64031 (URN)10.1016/j.neubiorev.2018.01.001 (DOI)000426224600005 ()29309797 (PubMedID)2-s2.0-85040358056 (Scopus ID)
Funder
Swedish Research Council, 523-2011-3646
Note

Funding Agency:

Stockholm County Council (PPG)  20130671  20150150

Available from: 2018-01-11 Created: 2018-01-11 Last updated: 2018-09-05Bibliographically approved
Hesselmark, E. & Bejerot, S. (2017). Biomarkers for diagnosis of Pediatric Acute Neuropsychiatric Syndrome (PANS): Sensitivity and specificity of the Cunningham Panel. Journal of Neuroimmunology, 312, 31-37
Open this publication in new window or tab >>Biomarkers for diagnosis of Pediatric Acute Neuropsychiatric Syndrome (PANS): Sensitivity and specificity of the Cunningham Panel
2017 (English)In: Journal of Neuroimmunology, ISSN 0165-5728, E-ISSN 1872-8421, Vol. 312, p. 31-37Article in journal (Refereed) Published
Abstract [en]

OBJECTIVE: Pediatric Acute Neuropsychiatric Syndrome (PANS) and pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections (PANDAS) are conditions marked by sudden onset of obsessive-compulsive disorder (OCD), tics, or avoidant/restrictive food intake in combination with multiple psychiatric symptoms. A diagnosis of PANS or PANDAS may be supported by the Cunningham Panel, a commercially available set of immunologic assays currently in clinical use. However, the relationship between Cunningham Panel results and patient symptoms remains unclear. This study was done to assess the diagnostic accuracy of the Cunningham Panel in patients with suspected PANS or PANDAS.

METHOD: All Swedish patients who had taken the Cunningham Panel prior to June 2014 (n=154) were invited and 53 patients participated in the study. Based on comprehensive psychiatric assessment (the reference standard of diagnosis), subjects were classified as PANS, PANDAS, or neither. Prior Cunningham Panel test results were collected from patient records, and new blood samples were similarly analyzed within the scope of this study. In addition, results were compared to healthy controls (n=21) and a test-retest reliability analysis was performed.

RESULTS: Sensitivities of individual biomarkers in the Cunningham Panel ranged from 15 to 60%, and specificities from 28 to 92%. Positive predictive values ranged from 17 to 40%, and negative predictive values from 44 to 74%. A majority of the healthy controls had pathological Cunningham Panel results and test-retest reliability proved insufficient.

CONCLUSION: Clinical use of the Cunningham Panel in diagnosing PANS or PANDAS is not supported by this study.

Place, publisher, year, edition, pages
Amsterdam, Netherlands: Elsevier, 2017
Keywords
PANDAS; PANS; Obsessive-compulsive disorder; Sensitivity and specificity; Biomarkers; Antibodies; Calcium/calmodulin kinase II
National Category
Neurology Immunology in the medical area
Identifiers
urn:nbn:se:oru:diva-61456 (URN)10.1016/j.jneuroim.2017.09.002 (DOI)000413390500007 ()28919236 (PubMedID)2-s2.0-85029516525 (Scopus ID)
Funder
Swedish Research Council, 523-2011-3646
Note

Funding Agency:

Stockholm County Council  20130671  20150150

Available from: 2017-10-13 Created: 2017-10-13 Last updated: 2018-08-06Bibliographically approved
Manouilenko, I., Humble, M. B., Georgieva, J. & Bejerot, S. (2017). Brainstem Auditory Evoked Potentials for diagnosing Autism Spectrum Disorder, ADHD and Schizophrenia Spectrum Disorders in adults: A blinded study. Psychiatry Research, 257, 21-26
Open this publication in new window or tab >>Brainstem Auditory Evoked Potentials for diagnosing Autism Spectrum Disorder, ADHD and Schizophrenia Spectrum Disorders in adults: A blinded study
2017 (English)In: Psychiatry Research, ISSN 0165-1781, E-ISSN 1872-7123, Vol. 257, p. 21-26Article in journal (Refereed) Published
Abstract [en]

The aim of the present study was to examine the clinical utility of complex auditory brainstem response (c-ABR) and investigate if c-ABR is helpful in the diagnostic procedure. Thirty-one adult psychiatric patients, thoroughly diagnosed with autism spectrum disorder (ASD) (n=16), ADHD (n=8), or schizophrenia spectrum disorder (SSD) (n=7) and 15 healthy controls (HC), were blindly assessed with SensoDetect BERA. This c-ABR correctly identified psychiatric diagnoses in 4 patients (13%) and provided partially correct diagnoses in 11 more patients. Of the 15 HC, 6 were misclassified as psychiatric patients. The Cohen´s kappa coefficient (κ) was substantial for HC (κ=0.67), fair for SSD (κ=0.37), slight for ADHD (κ=0.09) and without agreement in ASD (κ=-0.03). In conclusion, we found the c-ABR method unhelpful and unreliable as a tool in clinical diagnostics.

Place, publisher, year, edition, pages
Clare, Ireland: Elsevier, 2017
Keywords
Auditory brainstem response, double-blind method, mental disorder
National Category
Psychiatry Neurology
Identifiers
urn:nbn:se:oru:diva-61468 (URN)10.1016/j.psychres.2017.06.085 (DOI)000413385300004 ()28710948 (PubMedID)2-s2.0-85022338791 (Scopus ID)
Funder
Swedish Research Council, K2012-62X -22130-04-6
Note

Funding Agencies:

Praktikertjanst AB, Stockholm, Sweden  

Stockholm County Council  20110307 

Karolinska Institutet  20110307 

Region Örebro County, Sweden 

Available from: 2017-10-13 Created: 2017-10-13 Last updated: 2018-08-06Bibliographically approved
Hesselmark, E. & Bejerot, S. (2017). Corrigendum to Biomarkers for diagnosis of Pediatric Acute Neuropsychiatric Syndrome (PANS): Sensitivity and specificity of the Cunningham Panel [J. Neuroimmunol. 312. (2017) 31-37]. Journal of Neuroimmunology, 313, 116-117
Open this publication in new window or tab >>Corrigendum to Biomarkers for diagnosis of Pediatric Acute Neuropsychiatric Syndrome (PANS): Sensitivity and specificity of the Cunningham Panel [J. Neuroimmunol. 312. (2017) 31-37]
2017 (English)In: Journal of Neuroimmunology, ISSN 0165-5728, E-ISSN 1872-8421, Vol. 313, p. 116-117Article in journal, Editorial material (Refereed) Published
Place, publisher, year, edition, pages
Amsterdam, Netherlands: Elsevier, 2017
National Category
Neurology Immunology in the medical area
Identifiers
urn:nbn:se:oru:diva-62950 (URN)10.1016/j.jneuroim.2017.11.001 (DOI)000423006700017 ()29153602 (PubMedID)2-s2.0-85033399582 (Scopus ID)
Available from: 2017-12-07 Created: 2017-12-07 Last updated: 2018-08-13Bibliographically approved
Bejerot, S., Edman, G., Frisén, L. & Humble, M. B. (2017). Evidence-Based Brief Obsessive-Compulsive Scale [Letter to the editor]. Journal of Central Nervous System Disease, 9, Article ID UNSP 1179573517702867.
Open this publication in new window or tab >>Evidence-Based Brief Obsessive-Compulsive Scale
2017 (English)In: Journal of Central Nervous System Disease, ISSN 1179-5735, E-ISSN 1179-5735, Vol. 9, article id UNSP 1179573517702867Article in journal, Letter (Other academic) Published
Place, publisher, year, edition, pages
Sage Publications, 2017
National Category
Neurology
Identifiers
urn:nbn:se:oru:diva-58957 (URN)10.1177/1179573517702867 (DOI)000404684800001 ()28579868 (PubMedID)
Available from: 2017-08-18 Created: 2017-08-18 Last updated: 2017-10-05Bibliographically approved
Glans, M., Bejerot, S. & Humble, M. B. (2017). Generalised joint hypermobility and neurodevelopmental traits in a non-clinical adult population. BJPsych Open, 3(5), 236-242
Open this publication in new window or tab >>Generalised joint hypermobility and neurodevelopmental traits in a non-clinical adult population
2017 (English)In: BJPsych Open, E-ISSN 2056-4724, Vol. 3, no 5, p. 236-242Article in journal (Refereed) Published
Abstract [en]

BACKGROUND: Generalised joint hypermobility (GJH) is reportedly overrepresented among clinical cases of attention deficit/hyperactivity disorder (ADHD), autism spectrum disorder (ASD) and developmental coordination disorder (DCD). It is unknown if these associations are dimensional and, therefore, also relevant among non-clinical populations.

AIMS: To investigate if GJH correlates with sub-syndromal neurodevelopmental symptoms in a normal population.

METHOD: Hakim-Grahame's 5-part questionnaire (5PQ) on GJH, neuropsychiatric screening scales measuring ADHD and ASD traits, and a DCD-related question concerning clumsiness were distributed to a non-clinical, adult, Swedish population (n=1039).

RESULTS: In total, 887 individuals met our entry criteria. We found no associations between GJH and sub-syndromal symptoms of ADHD, ASD or DCD.

CONCLUSIONS: Although GJH is overrepresented in clinical cases with neurodevelopmental disorders, such an association seems absent in a normal population. Thus, if GJH serves as a biomarker cutting across diagnostic boundaries, this association is presumably limited to clinical populations.

Place, publisher, year, edition, pages
Royal College of Psychiatrists, 2017
National Category
Psychiatry
Identifiers
urn:nbn:se:oru:diva-62454 (URN)10.1192/bjpo.bp.116.004325 (DOI)000418507300005 ()28959454 (PubMedID)
Funder
Swedish Research Council, K2012-62X-22130-04-6The Karolinska Institutet's Research FoundationStockholm County Council
Available from: 2017-12-04 Created: 2017-12-04 Last updated: 2018-08-13Bibliographically approved
Bejerot, S. & Hesselmark, E. (2017). Pediatric autoimmune neuropsychiatric syndrome (PANS), developmental regression and autism. European psychiatry, 41(Suppl.), S123-S123
Open this publication in new window or tab >>Pediatric autoimmune neuropsychiatric syndrome (PANS), developmental regression and autism
2017 (English)In: European psychiatry, ISSN 0924-9338, E-ISSN 1778-3585, Vol. 41, no Suppl., p. S123-S123Article in journal, Meeting abstract (Other academic) Published
Abstract [en]

Introduction: Pediatric autoimmune neuropsychiatric syndrome (PANS) is a term used to describe a clinical picture which includes sudden onset of psychiatric symptoms and a possible autoimmune genesis. The sudden decline in neuropsychiatric functioning as well as the multiple combinations of symptoms may lead to a clinical phenotype similar to that in infantile autism (IA) with regressive features. We are conducting a study with the aim to evaluate a diagnostic test for PANS currently marketed by Moleculera Labs. All patients in Sweden who had taken the test (n = 154) were invited to the study.

Objectives: The aim of the study is to characterize a subgroup of patients with IA within the PANS diagnosis study.

Methods: Participants (n = 53) were examined for psychiatric and somatic symptoms and evaluated for PANS caseness by an experienced psychiatrist. Because the criteria for entering the study was having taken the diagnostic test for PANS, the participants in the study comprise a group with mixed symptoms.

Results: Twelve participants had IA. Eleven of these reported a developmental regression with loss of abilities. Two of the IA patients also fulfill criteria for PANS. Eight of the IA patients had been treated with antibiotics for psychiatric symptoms and 4 reported a positive effect of this treatment. Nine of the patients had elevated test results suggesting possible PANS according to Moleculera Labs.

Conclusions: Very early onset on PANS may be phenotypically similar to IA with regressive features. Further analysis of the immunological attributes of patients with autism with regressive features is warranted.

Place, publisher, year, edition, pages
Elsevier, 2017
National Category
Psychiatry
Research subject
Psychiatry
Identifiers
urn:nbn:se:oru:diva-58969 (URN)10.1016/j.eurpsy.2017.01.1921 (DOI)000404952200364 ()
Available from: 2017-08-04 Created: 2017-08-04 Last updated: 2018-09-04Bibliographically approved
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ORCID iD: ORCID iD iconorcid.org/0000-0002-3587-6075

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