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Disease State: Capillary malformations

Background

Capillary malformations (a.k.a. port wine stains; PWS) are red to purple lesions affecting the dermis and are usually present at birth. These congenital lesions have been associated with somatic mosaic mutations in the GNA (Q or 11) gene and PIK3CA gene. Besides physical and functional effects, PWS often lead to a decreased emotional and social overall health-related quality of life (HR-QoL) as most are visibly located in the head and neck region. Multiple therapeutic strategies are available, including cosmetic camouflage, medical tattooing, surgical excision, and laser and light therapies.

Currently, there is a large variety in treatment outcomes and a lack of outcomes relevant to patients. This hampers proper comparison of treatment outcomes. As a result, there is limited evidence about what the best available therapeutic options are.


Project Goal

The Core Outcome Set for CApillary Malformations (COSCAM) project aims to reach international consensus on a core outcome set (COS) for clinical research on capillary malformations, hereby standardizing reporting of outcomes.

To date, we have reached international consensus on the core outcome domain set (CDS), containing the most important outcomes that are recommended to be used in all future clinical research on PWS. A total of 269 stakeholders (patients, caregivers, physicians, researchers) from 45 countries have participated in defining the CDS. The next step will be defining the core outcome measurement set (COMS), containing the most important outcome measurement instruments to measure the core outcomes.


Project Lead

Ginger Beau Langbroek
Ginger Beau Langbroek

Key Project Team Members:

Albert Wolkerstorfer
Albert Wolkerstorfer
Sophie E.R. Horbach
Sophie E.R. Horbach
Phyllis Spuls
Phyllis Spuls
Kristen M Kelly
Kristen M Kelly
Susan J Robertson
Susan J Robertson
M Ingmar van Raath
M Ingmar van Raath
Firas Al-Niaimi
Firas Al-Niaimi
Taro Kono
Taro Kono
Pablo Boixeda
Pablo Boixeda
Hans J Laubach
Hans J Laubach
Ashraf M Badawi
Ashraf M Badawi
Agneta Troilius Rubin
Agneta Troilius Rubin
Merete Haedersdal
Merete Haedersdal
Woraphong Manuskiatti
Woraphong Manuskiatti
Chantal M.A.M. van der Horst
Chantal M.A.M. van der Horst
Dirk T Ubbink
Dirk T Ubbink

C3 Methods Partner:

Phyllis Spuls
Phyllis Spuls

Contact:

Ginger Beau Langbroek (g.b.langbroek@amsterdamumc.nl)

 

Publications: 

Core outcome domain set

  1. van Raath MI, Chohan S, Wolkerstorfer A, van der Horst CMAM, Limpens J, Huang X, Ding B, Storm G, van der Hulst RRWJ, Heger M. Clinical outcome measures and scoring systems used in prospective studies of port wine stains: A systematic review. PLoS One. 2020;15(7):e0235657-e0235657. doi:10.1371/journal.pone.0235657
  2. G.B. Langbroek, A. Wolkerstorfer, S.E.R. Horbach, P.I. Spuls, K.M. Kelly, S.J. Robertson, M.I. van Raath, F. Al-Niaimi, T. Kono, P. Boixeda, H.J. Laubach, A.M. Badawi, A. Troilius Rubin, M. Haedersdal, W. Manuskiatti, C.M.A.M. van der Horst and D.T. Ubbink. Development of a core outcome domain set for clinical research on capillary malformations (the COSCAM project). Journal of the European Academy of Dermatology and Venereology. 2021 Sep;35(9):1888-95.
  3. B. Langbroek, A. Wolkerstorfer, S.E.R. Horbach, P.I. Spuls, K.M. Kelly, S.J. Robertson, M.I. van Raath, F. Al-Niaimi, T. Kono, P. Boixeda, H.J. Laubach, A.M. Badawi, A. Troilius Rubin, M. Haedersdal, W. Manuskiatti, C.M.A.M. van der Horst and D.T. Ubbink. A core outcome domain set for clinical research on capillary malformations (the COSCAM project): an e-Delphi process and consensus meeting. British Journal of Dermatology. Provisionally accepted.

 

Updated on Date