Abstract
Objective: The goal of this study was to investigate treatment histories and outcomes in a large community sample of youth with Pediatric Acute-onset Neuropsychiatric Syndrome (PANS), and, where appropriate, to examine the impact of immune deficiency on treatment outcomes.Methods: A comprehensive internet-based survey was completed by parents or guardians of youth who had received physician diagnoses of PANS, or by young adults (age 18+) who had themselves been diagnosed by a physician (N = 698). Data regarding the treatment histories of these patients, including the variety of medical and psychological treatments employed and the caregiver- or self-reported response to each, are presented.Results: The PANS patients in this study had commonly been treated with antibiotic (N = 675), anti-inflammatory (N = 437), and/or psychotropic therapy (N = 378). Response to antibiotic treatment was best when treatment was relatively aggressive, with broad-spectrum antibiotics and courses of >30 days generally producing the best results (i.e., up to 52% of patients achieving a “very effective” response). For immune-deficient patients (caregiver-reported laboratory studies below normal limits; N = 108), use of broad-spectrum antibiotics appeared to be particularly desirable. Anti-inflammatory therapies, including over-the-counter medications such as ibuprofen, were at least “somewhat effective” for most patients. Intravenous immunoglobulin (IVIG) had been used to treat PANS in 193 (28%) of the patients and was at least “somewhat effective” for 89%, although for 18% of these, the effect was not sustained. The highest rate of sustained response to IVIG treatment was seen in immune-deficient patients who received doses of at least 0.8 g/kg IVIG on a regular basis. Psychotropic medications, most commonly SSRIs (38% reported a trial), were commonly employed, but were often ineffective (e.g., 44% found SSRIs “somewhat” to “very effective”). Many patients (N = 473) had received some form of psychotherapy with some benefit, with cognitive behavioral therapy found to be at least somewhat effective in a majority of those treated with this modality.Conclusion: 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. The results of this study should be considered in light of the limitations inherent in a self-selected and administered online survey.
Highlights
Among the Pediatric Acute-onset Neuropsychiatric Syndrome (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
The diagnostic criteria for Pediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcus (PANDAS) formulated by Swedo et al in the late 1990’s (Swedo et al 1998) describe an acute-onset presentation of neuropsychiatric symptoms that is temporally related to a group A streptococcal (GAS) infection
The potential for certain antimicrobials to possess neurochemical or immunemodulating properties is a consideration (Obregon et al 2012) The need for sufficiently aggressive antibiotic treatment from the first PANS presentation is important given that, as reported previously (Calaprice et al 2017), resolution of the inciting infection with the initial course of antibiotic treatment is achieved for only 59% of patients, and recurrence of PANS appears to be significantly more probable for patients for whom the infection associated with the initial PANS episode does not resolve completely with antibiotic treatment
Summary
The diagnostic criteria for Pediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcus (PANDAS) formulated by Swedo et al in the late 1990’s (Swedo et al 1998) describe an acute-onset presentation of neuropsychiatric symptoms that is temporally related to a group A streptococcal (GAS) infection. A broader variation of this disorder is PANS (Pediatric Acute-onset Neuropsychiatric Syndrome), which diagnostically requires acute-onset OCD or food refusal, most often but not necessarily in association with GAS or nonGAS infections, plus ancillary symptoms such as those described above for PANDAS (Swedo et al 2012). Similar to Sydenham’s chorea, on which the original PANDAS model was based (Swedo 1994), PANS is presumed to have an autoimmune pathophysiology, and most PANS patients exhibit additional immune abnormalities, including comorbid autoimmune or inflammatory conditions and/or below normal or low-normal levels of immunoglobulins, in addition to having neuroactive autoantibodies (Frankovich et al 2015; Nadeau et al 2015)
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