Measuring Disease Activity During Maintenance Therapy in Chronic Inflammatory Demyelinating Polyradiculoneuropathy (CIDP)
Grace Swart1, Nidhi Garg1, Con Yiannikas1, Ostoja (Steve) Vucic2, John Pollard1, Antonia Carroll3, Matthew Silsby4, Matthew Kiernan5, Judith Spies6, Susanna Park1
1University of Sydney, 2Concord Hospital, 3St Vincent's Hospital, 4Westmead Hospital, 5Neuroscience Research Australia, 6Royal Prince Alfred Hospital
Objective:

To investigate clinically meaningful outcome measures associated with minimum symptom expression (MSE) in a cohort of treated CIDP patients. 

Background:

Clinically derived endpoints for CIDP reflecting patient function are necessary to optimally assess disease activity and treatment efficacy.

Design/Methods:

We retrospectively analyzed a consecutive Australian cohort fulfilling 2021-EAN/PNS CIDP criteria1 on therapy between April 2015 and January 2017. Comprehensive assessments of clinical impairment, disability and patient-reported outcomes were performed pre- and post-treatment for patients on cyclic therapy. MSE was systematically defined as an I-RODS score ≥44/48, with no meaningful functional limitation (all activities scored as possible).

Results:

Forty-two patients were identified (mean age 57 years; 64% male; median disease duration 8.5 years[range 0.33-45]). Phenotypes were typical (48%), multifocal/focal (24%), distal (12%), sensory/sensory-predominant (12%), and motor-predominant (4%). Treatments included intravenous immunoglobulin (IVIG) (90%), plasma exchange (5%), prednisone (14%), mycophenolate (12%) and azathioprine (2%). Average IVIG regimen was 0.7g/kg, 4 weekly. 21% (8/38) at baseline and 27% (9/33) post-treatment (mean interval 12.4 days) fulfilled MSE criteria. MSE status was not significantly different by age, disease duration, muscle strength (MRC-SS), dominant grip strength (p=0.089), sensory function (mISS), fatigue (mFSS) or pain. Outcomes significantly distinguishing MSE were 10MWT (7.19 vs 9.65 sec, median faster walk 2.37 sec, 95%CI 1.25-3.51, p<0.001) and INCAT (2.5 vs 3, median decrease 1-point, 95%CI 0-2, p=0.026). Logistic regression showed MSE associated with faster 10MWT (OR7.7, 95%CI 1.6–37.0, p=0.011; and lower INCAT (OR 2.3, 95%CI 1.06-5, p=0.036). 10MWT showed excellent discrimination of MSE (AUC 0.93, 95% CI 0.82–1.00, p<0.001) with optimal cut-off ≤6.5 s (sensitivity 0.90, specificity 0.80, p<0.001). Discrimination was fair for INCAT (AUC 0.758, 95%CI 0.603–0.914; p = .001); cut-off ≤2 yielding 63% sensitivity and 73% specificity.

Conclusions:

MSE was achieved in 21% of CIDP patients on maintenance treatment, best distinguished by measures of gait (10MWT) and disability (INCAT).

10.1212/WNL.0000000000217482
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