Development and validation of a IHS4 dichotomous outcome for assessing anti-inflammatory treatment efficacy for the treatment of hidradenitis suppurativa

Lead Investigator: Thrasyvoulos Tzellos, Norland University Hospital
Title of Proposal Research: Development and validation of a IHS4 dichotomous outcome for assessing anti-inflammatory treatment efficacy for the treatment of hidradenitis suppurativa
Vivli Data Request: 5902
Funding Source: None
Potential Conflicts of Interest:
Abbvie: Consultant, clinical investigator, speaker
UCB: Clinical investigator
Sanofi: Consultant, clinical investigator
All members of the research group declare clearly their conflict of interest and the analysis proposed has no effort comparing effect of treatments but most importantly it will try to provide a scoring system – outcome that can be beneficial for all, patients, MDs and drug industry in general. In all cases, for all conflict of interests, we have signed a confidential agreement, which we always respect. The Conflicts of Interest (COI) are managed in a manner that they will not bias our interpretation of the data, the data is kept confidential and only used for the purpose of this study.

Summary of the Proposed Research:

For a chronic systemic inflammatory skin disease like Hidradenitis Suppurativa it is essential to have validated and easy to use outcomes that can be used both in clinical trial setting and daily clinical practice. Our effort will be to develop and validate a new dichotomous outome that can serve exactly this purpose. We are going to utilize a Post-hoc analysis of individual patient data from PIONEER studies that evaluated the efficacy of adalimumab versus placebo. A recent study
(Frew JW, et al. J Am Acad Dermatol 2020; 82: 1150-1157) concluded that when the International Hidradenitis Suppurativa Severity Scoring System (IHS4) was used, the placebo response rates were measured to be lower, as opposed to the hidradenitis suppurativa clinical response (HiSCR) dichotomous score, which was included in the study protocol. The latter study also assessed the impact of draining tunnels upon therapeutic response.
We believe that integrating draining tunnel status (using the IHS4) was the core fact that reduced placebo response rates in both PIONEERs regardless of whether binary or continuous outcomes were used. IHS4 use as a continuous outcome variable resulted in placebo response rates consistent with those in studies of Psoriasis and Atopic Dermatitis (4.5%-12%). We will use the Pioneer I as a training dataset and then Pioneer II as a validation dataset. Should this not provide adequate verification, we will create random datasets from either or both Pioneer I and II studies, which will be used for training and validation. Correlations between the new binary outcome and VAS pain, DLQI (Dermatology Life Quality Index), Sartorius, HiSCR, the physician and patient derived outcomes will be examined. Minimally clinical important differences corresponding to outcome change, will also be explored. We will use SPSS and Stata for the statistical analyses.

Statistical Analysis Plan:

We will use the Pioneer I as a training dataset and then Pioneer II as a validation dataset. Should this not provide adequate verification, we will create random datasets from either or both Pioneer I and II studies, which will be used for training and validation.

Correlations between the new binary outcome and VAS pain, DLQI, Sartorius, HiSCR, the physician and patient derived outcomes will be examined. Minimally clinical important differences corresponding to outcome change, will also be explored.

We will use SPSS and Stata for the statistical analyses.

We will use the Pioneer I as a training dataset and then Pioneer II as a validation dataset. We will employ discriminant analyses to identify the optimal cut-off threshold for the continuous IHS4 score, to provide a dichotomous variable. Diagnostic accuracy (Sensitivity-Se and Specificity-Sp) will be used to determine the cut-off. Alternative cut-offs for enhanced Se and Sp will also be determined.

We shall then use the above cut-offs to determine the classification of the patients from the Pioneer II validation dataset. Thus we shall have a number of dichotomous (testing) variables which would then be compared against VAS pain, DLQI, Sartorius, HiSCR, the physician and patient derived outcomes. Minimally clinical important differences of the latter scores corresponding to IHS4 derived dichotomous outcome (under testing) change will also be measured.

In the case that the IHS4 binary variables (created using the cut-offs from Pioneer I) will not provide sufficient fit to the data of the Pioneer II (allowing for some differences between the two studies populations?) we will create random datasets from both Pioneer I and II studies, which will be used for training and validation (in an attempt to homogenize the populations, in the –not likely- case they prove different).

To examine the association of the new under test IHSD4 binary outcomes and the various established scores we shall use correlation matrices; subsequent quantification of the plausible association will follow using linear or logistic regression analysis (depending on the variable correlated) in an attempt to model the association and plot the curve. Then, minimally clinical important differences corresponding to outcome changes will be determined. We will use SPSS (accessed via Vivli) for the analyses, using a license from A.U.Th.

Requested Studies:

A Phase 3 Multicenter Study of the Safety and Efficacy of Adalimumab in Subjects With Moderate to Severe Hidradenitis Suppurativa – PIONEER I (PIONEER I)
Sponsor: Abbvie
Study ID: NCT01468207
Sponsor ID: M11-313

A Phase 3 Multicenter Study of the Safety and Efficacy of Adalimumab in Subjects With Moderate to Severe Hidradenitis Suppurativa – PIONEER II (PIONEER II)
Sponsor: Abbvie
Study ID: NCT01468233
Sponsor ID: M11-810

Public Disclosures:

  1. Thrasivoulos Tzellos Et al. Development and validation of IHS4-55, an IHS4 dichotomous outcome to assess treatment effect for hidradenitis suppurativa. J Eur Acad Dermatol Venereol. 2022 Oct 2. doi: 10.1111/jdv.18632. Online ahead of print.
  2. K. R. van Straalen, T. Tzellos, A. Alavi, F. Benhadou, C. Cuenca-Barrales, M. Daxhelet, M. Daoud, O. Efthymiou, E. J. Giamarellos-Bourboulis, P. Guillem, W. Gulliver, G. B. Jemec, A. Katoulis, A. Koenig, E. Lazaridou, M. Lowes, A. V. Marzano, L. Matusiak, A. Molina-Leyva, C. Moltrasio, A. Pinter, C. Potenza, E. P. Prens, J. Romaní, D. M. Saunte, C. J. Sayed, N. Skroza, D. Stergianou, J. C. Szepietowski, A. Trigoni, E. Vilarrasa, H. H. van der Zee, A. Kyrgidis, C. C. Zouboulis. (2022), 11th EHSF Conference 2022 Abstracts. Exp Dermatol, 31: 40-125. S-01-12 – Development and validation of an IHS4 dichotomous outcome to assess treatment effect. (p.47).Doi: 10.11111/exd.14557