Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study

Sébastien Peytrignet; Christopher P Denton; Mark Lunt; Roger Hesselstrand; Luc Mouthon; Alan Silman; Xiaoyan Pan; Edith Brown; László Czirják; Jörg H W Distler; Oliver Distler; Kim Fligelstone; William J Gregory; Rachel Ochiel; Madelon Vonk; Codrina Ancuta; Voon H Ong; Dominique Farge; Marie Hudson; Marco Matucci-Cerinic; Alexandra Balbir-Gurman; Øyvind Midtvedt; Alison C Jordan; Wendy Stevens; Pia Moinzadeh; Frances C Hall; Christian Agard; Marina E Anderson; Elisabeth Diot; Rajan Madhok; Mohammed Akil; Maya H Buch; Lorinda Chung; Nemanja Damjanov; Harsha Gunawardena; Peter Lanyon; Yasmeen Ahmad; Kuntal Chakravarty; Søren Jacobsen; Alexander J MacGregor; Neil McHugh; Ulf Müller-Ladner; Gabriela Riemekasten; Michael Becker; Janet Roddy; Patricia E Carreira; Anne Laure Fauchais; Eric Hachulla; Jennifer Hamilton; Murat Inanç; John S McLaren; Jacob M van Laar; Sanjay Pathare; Susanna Proudman; Anna Rudin; Joanne Sahhar; Brigitte Coppere; Christine Serratrice; Tom Sheeran; Douglas J Veale; Claire Grange; Georges-Selim Trad; Ariane L Herrick

doi:10.1093/rheumatology/kex410

Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study

Sébastien Peytrignet, Christopher P Denton, Mark Lunt, Roger Hesselstrand, Luc Mouthon, Alan Silman, Xiaoyan Pan, Edith Brown, László Czirják, Jörg H W Distler, Oliver Distler, Kim Fligelstone, William J Gregory, Rachel Ochiel, Madelon Vonk, Codrina Ancuta, Voon H Ong, Dominique Farge, Marie Hudson, Marco Matucci-CerinicAlexandra Balbir-Gurman, Øyvind Midtvedt, Alison C Jordan, Wendy Stevens, Pia Moinzadeh, Frances C Hall, Christian Agard, Marina E Anderson, Elisabeth Diot, Rajan Madhok, Mohammed Akil, Maya H Buch, Lorinda Chung, Nemanja Damjanov, Harsha Gunawardena, Peter Lanyon, Yasmeen Ahmad, Kuntal Chakravarty, Søren Jacobsen, Alexander J MacGregor, Neil McHugh, Ulf Müller-Ladner, Gabriela Riemekasten, Michael Becker, Janet Roddy, Patricia E Carreira, Anne Laure Fauchais, Eric Hachulla, Jennifer Hamilton, Murat Inanç, John S McLaren, Jacob M van Laar, Sanjay Pathare, Susanna Proudman, Anna Rudin, Joanne Sahhar, Brigitte Coppere, Christine Serratrice, Tom Sheeran, Douglas J Veale, Claire Grange, Georges-Selim Trad, Ariane L Herrick

Department of Clinical Medicine

23 Citations (Scopus)

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Abstract

Objectives. Our aim was to describe the burden of early dcSSc in terms of disability, fatigue and pain in the European Scleroderma Observational Study cohort, and to explore associated clinical features. Methods. Patients completed questionnaires at study entry, 12 and 24 months, including the HAQ disability index (HAQ-DI), the Cochin Hand Function Scale (CHFS), the Functional Assessment of Chronic Illness Therapy-fatigue and the Short Form 36 (SF36). Associates examined included the modified Rodnan skin score (mRSS), current digital ulcers and internal organ involvement. Correlations between 12-month changes were also examined. Results. The 326 patients recruited (median disease duration 11.9 months) displayed high levels of disability [mean (S.D.) HAQ-DI 1.1 (0.83)], with 'grip' and 'activity' being most affected. Of the 18 activities assessed in the CHFS, those involving fine finger movements were most affected. High HAQ-DI and CHFS scores were both associated with high mRSS (r = 0.34, P < 0.0001 and r = 0.35, P < 0.0001, respectively). HAQ-DI was higher in patients with digital ulcers (P = 0.004), pulmonary fibrosis (P = 0.005), cardiac (P = 0.005) and muscle involvement (P = 0.002). As anticipated, HAQ-DI, CHFS, the Functional Assessment of Chronic Illness Therapy and SF36 scores were all highly correlated, in particular the HAQ-DI with the CHFS (ρ = 0.84, P < 0.0001). Worsening HAQ-DI over 12 months was strongly associated with increasing mRSS (ρ = 0.40, P < 0.0001), decreasing hand function (ρ = 0.57, P < 0.0001) and increasing fatigue (ρ =-0.53, P < 0.0001). Conclusion. The European Scleroderma Observational Study highlights the burden of disability in early dcSSc, with high levels of disability and fatigue, associating with the degree of skin thickening (mRSS). Impaired hand function is a major contributor to overall disability.

Original language	English
Journal	Rheumatology
Volume	57
Issue number	2
Pages (from-to)	370-381
ISSN	1462-0324
DOIs	https://doi.org/10.1093/rheumatology/kex410
Publication status	Published - 1 Feb 2018

Keywords

Adult
Cost of Illness
Disability Evaluation
Europe
Fatigue/etiology
Female
Fingers
Hand Strength
Health Surveys
Humans
Male
Pain/etiology
Prospective Studies
Scleroderma, Diffuse/complications
Severity of Illness Index
Skin Ulcer/etiology

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Peytrignet, S., Denton, C. P., Lunt, M., Hesselstrand, R., Mouthon, L., Silman, A., Pan, X., Brown, E., Czirják, L., Distler, J. H. W., Distler, O., Fligelstone, K., Gregory, W. J., Ochiel, R., Vonk, M., Ancuta, C., Ong, V. H., Farge, D., Hudson, M., ... Herrick, A. L. (2018). Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study. Rheumatology, 57(2), 370-381. https://doi.org/10.1093/rheumatology/kex410

Peytrignet, S, Denton, CP, Lunt, M, Hesselstrand, R, Mouthon, L, Silman, A, Pan, X, Brown, E, Czirják, L, Distler, JHW, Distler, O, Fligelstone, K, Gregory, WJ, Ochiel, R, Vonk, M, Ancuta, C, Ong, VH, Farge, D, Hudson, M, Matucci-Cerinic, M, Balbir-Gurman, A, Midtvedt, Ø, Jordan, AC, Stevens, W, Moinzadeh, P, Hall, FC, Agard, C, Anderson, ME, Diot, E, Madhok, R, Akil, M, Buch, MH, Chung, L, Damjanov, N, Gunawardena, H, Lanyon, P, Ahmad, Y, Chakravarty, K, Jacobsen, S, MacGregor, AJ, McHugh, N, Müller-Ladner, U, Riemekasten, G, Becker, M, Roddy, J, Carreira, PE, Fauchais, AL, Hachulla, E, Hamilton, J, Inanç, M, McLaren, JS, van Laar, JM, Pathare, S, Proudman, S, Rudin, A, Sahhar, J, Coppere, B, Serratrice, C, Sheeran, T, Veale, DJ, Grange, C, Trad, G-S & Herrick, AL 2018, 'Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study', Rheumatology, vol. 57, no. 2, pp. 370-381. https://doi.org/10.1093/rheumatology/kex410

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title = "Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study",

abstract = "Objectives. Our aim was to describe the burden of early dcSSc in terms of disability, fatigue and pain in the European Scleroderma Observational Study cohort, and to explore associated clinical features. Methods. Patients completed questionnaires at study entry, 12 and 24 months, including the HAQ disability index (HAQ-DI), the Cochin Hand Function Scale (CHFS), the Functional Assessment of Chronic Illness Therapy-fatigue and the Short Form 36 (SF36). Associates examined included the modified Rodnan skin score (mRSS), current digital ulcers and internal organ involvement. Correlations between 12-month changes were also examined. Results. The 326 patients recruited (median disease duration 11.9 months) displayed high levels of disability [mean (S.D.) HAQ-DI 1.1 (0.83)], with 'grip' and 'activity' being most affected. Of the 18 activities assessed in the CHFS, those involving fine finger movements were most affected. High HAQ-DI and CHFS scores were both associated with high mRSS (r = 0.34, P < 0.0001 and r = 0.35, P < 0.0001, respectively). HAQ-DI was higher in patients with digital ulcers (P = 0.004), pulmonary fibrosis (P = 0.005), cardiac (P = 0.005) and muscle involvement (P = 0.002). As anticipated, HAQ-DI, CHFS, the Functional Assessment of Chronic Illness Therapy and SF36 scores were all highly correlated, in particular the HAQ-DI with the CHFS (ρ = 0.84, P < 0.0001). Worsening HAQ-DI over 12 months was strongly associated with increasing mRSS (ρ = 0.40, P < 0.0001), decreasing hand function (ρ = 0.57, P < 0.0001) and increasing fatigue (ρ =-0.53, P < 0.0001). Conclusion. The European Scleroderma Observational Study highlights the burden of disability in early dcSSc, with high levels of disability and fatigue, associating with the degree of skin thickening (mRSS). Impaired hand function is a major contributor to overall disability.",

keywords = "Adult, Cost of Illness, Disability Evaluation, Europe, Fatigue/etiology, Female, Fingers, Hand Strength, Health Surveys, Humans, Male, Pain/etiology, Prospective Studies, Scleroderma, Diffuse/complications, Severity of Illness Index, Skin Ulcer/etiology",

author = "S{\'e}bastien Peytrignet and Denton, {Christopher P} and Mark Lunt and Roger Hesselstrand and Luc Mouthon and Alan Silman and Xiaoyan Pan and Edith Brown and L{\'a}szl{\'o} Czirj{\'a}k and Distler, {J{\"o}rg H W} and Oliver Distler and Kim Fligelstone and Gregory, {William J} and Rachel Ochiel and Madelon Vonk and Codrina Ancuta and Ong, {Voon H} and Dominique Farge and Marie Hudson and Marco Matucci-Cerinic and Alexandra Balbir-Gurman and {\O}yvind Midtvedt and Jordan, {Alison C} and Wendy Stevens and Pia Moinzadeh and Hall, {Frances C} and Christian Agard and Anderson, {Marina E} and Elisabeth Diot and Rajan Madhok and Mohammed Akil and Buch, {Maya H} and Lorinda Chung and Nemanja Damjanov and Harsha Gunawardena and Peter Lanyon and Yasmeen Ahmad and Kuntal Chakravarty and S{\o}ren Jacobsen and MacGregor, {Alexander J} and Neil McHugh and Ulf M{\"u}ller-Ladner and Gabriela Riemekasten and Michael Becker and Janet Roddy and Carreira, {Patricia E} and Fauchais, {Anne Laure} and Eric Hachulla and Jennifer Hamilton and Murat Inan{\c c} and McLaren, {John S} and {van Laar}, {Jacob M} and Sanjay Pathare and Susanna Proudman and Anna Rudin and Joanne Sahhar and Brigitte Coppere and Christine Serratrice and Tom Sheeran and Veale, {Douglas J} and Claire Grange and Georges-Selim Trad and Herrick, {Ariane L}",

note = "{\textcopyright} The Author 2017. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For Permissions, please email: [email protected]",

year = "2018",

month = feb,

day = "1",

doi = "10.1093/rheumatology/kex410",

language = "English",

volume = "57",

pages = "370--381",

journal = "Rheumatology",

issn = "1462-0324",

publisher = "Oxford University Press",

number = "2",

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TY - JOUR

T1 - Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis

T2 - the European Scleroderma Observational Study

AU - Peytrignet, Sébastien

AU - Denton, Christopher P

AU - Lunt, Mark

AU - Hesselstrand, Roger

AU - Mouthon, Luc

AU - Silman, Alan

AU - Pan, Xiaoyan

AU - Brown, Edith

AU - Czirják, László

AU - Distler, Jörg H W

AU - Distler, Oliver

AU - Fligelstone, Kim

AU - Gregory, William J

AU - Ochiel, Rachel

AU - Vonk, Madelon

AU - Ancuta, Codrina

AU - Ong, Voon H

AU - Farge, Dominique

AU - Hudson, Marie

AU - Matucci-Cerinic, Marco

AU - Balbir-Gurman, Alexandra

AU - Midtvedt, Øyvind

AU - Jordan, Alison C

AU - Stevens, Wendy

AU - Moinzadeh, Pia

AU - Hall, Frances C

AU - Agard, Christian

AU - Anderson, Marina E

AU - Diot, Elisabeth

AU - Madhok, Rajan

AU - Akil, Mohammed

AU - Buch, Maya H

AU - Chung, Lorinda

AU - Damjanov, Nemanja

AU - Gunawardena, Harsha

AU - Lanyon, Peter

AU - Ahmad, Yasmeen

AU - Chakravarty, Kuntal

AU - Jacobsen, Søren

AU - MacGregor, Alexander J

AU - McHugh, Neil

AU - Müller-Ladner, Ulf

AU - Riemekasten, Gabriela

AU - Becker, Michael

AU - Roddy, Janet

AU - Carreira, Patricia E

AU - Fauchais, Anne Laure

AU - Hachulla, Eric

AU - Hamilton, Jennifer

AU - Inanç, Murat

AU - McLaren, John S

AU - van Laar, Jacob M

AU - Pathare, Sanjay

AU - Proudman, Susanna

AU - Rudin, Anna

AU - Sahhar, Joanne

AU - Coppere, Brigitte

AU - Serratrice, Christine

AU - Sheeran, Tom

AU - Veale, Douglas J

AU - Grange, Claire

AU - Trad, Georges-Selim

AU - Herrick, Ariane L

PY - 2018/2/1

Y1 - 2018/2/1

N2 - Objectives. Our aim was to describe the burden of early dcSSc in terms of disability, fatigue and pain in the European Scleroderma Observational Study cohort, and to explore associated clinical features. Methods. Patients completed questionnaires at study entry, 12 and 24 months, including the HAQ disability index (HAQ-DI), the Cochin Hand Function Scale (CHFS), the Functional Assessment of Chronic Illness Therapy-fatigue and the Short Form 36 (SF36). Associates examined included the modified Rodnan skin score (mRSS), current digital ulcers and internal organ involvement. Correlations between 12-month changes were also examined. Results. The 326 patients recruited (median disease duration 11.9 months) displayed high levels of disability [mean (S.D.) HAQ-DI 1.1 (0.83)], with 'grip' and 'activity' being most affected. Of the 18 activities assessed in the CHFS, those involving fine finger movements were most affected. High HAQ-DI and CHFS scores were both associated with high mRSS (r = 0.34, P < 0.0001 and r = 0.35, P < 0.0001, respectively). HAQ-DI was higher in patients with digital ulcers (P = 0.004), pulmonary fibrosis (P = 0.005), cardiac (P = 0.005) and muscle involvement (P = 0.002). As anticipated, HAQ-DI, CHFS, the Functional Assessment of Chronic Illness Therapy and SF36 scores were all highly correlated, in particular the HAQ-DI with the CHFS (ρ = 0.84, P < 0.0001). Worsening HAQ-DI over 12 months was strongly associated with increasing mRSS (ρ = 0.40, P < 0.0001), decreasing hand function (ρ = 0.57, P < 0.0001) and increasing fatigue (ρ =-0.53, P < 0.0001). Conclusion. The European Scleroderma Observational Study highlights the burden of disability in early dcSSc, with high levels of disability and fatigue, associating with the degree of skin thickening (mRSS). Impaired hand function is a major contributor to overall disability.

AB - Objectives. Our aim was to describe the burden of early dcSSc in terms of disability, fatigue and pain in the European Scleroderma Observational Study cohort, and to explore associated clinical features. Methods. Patients completed questionnaires at study entry, 12 and 24 months, including the HAQ disability index (HAQ-DI), the Cochin Hand Function Scale (CHFS), the Functional Assessment of Chronic Illness Therapy-fatigue and the Short Form 36 (SF36). Associates examined included the modified Rodnan skin score (mRSS), current digital ulcers and internal organ involvement. Correlations between 12-month changes were also examined. Results. The 326 patients recruited (median disease duration 11.9 months) displayed high levels of disability [mean (S.D.) HAQ-DI 1.1 (0.83)], with 'grip' and 'activity' being most affected. Of the 18 activities assessed in the CHFS, those involving fine finger movements were most affected. High HAQ-DI and CHFS scores were both associated with high mRSS (r = 0.34, P < 0.0001 and r = 0.35, P < 0.0001, respectively). HAQ-DI was higher in patients with digital ulcers (P = 0.004), pulmonary fibrosis (P = 0.005), cardiac (P = 0.005) and muscle involvement (P = 0.002). As anticipated, HAQ-DI, CHFS, the Functional Assessment of Chronic Illness Therapy and SF36 scores were all highly correlated, in particular the HAQ-DI with the CHFS (ρ = 0.84, P < 0.0001). Worsening HAQ-DI over 12 months was strongly associated with increasing mRSS (ρ = 0.40, P < 0.0001), decreasing hand function (ρ = 0.57, P < 0.0001) and increasing fatigue (ρ =-0.53, P < 0.0001). Conclusion. The European Scleroderma Observational Study highlights the burden of disability in early dcSSc, with high levels of disability and fatigue, associating with the degree of skin thickening (mRSS). Impaired hand function is a major contributor to overall disability.

KW - Adult

KW - Cost of Illness

KW - Disability Evaluation

KW - Europe

KW - Fatigue/etiology

KW - Female

KW - Fingers

KW - Hand Strength

KW - Health Surveys

KW - Humans

KW - Male

KW - Pain/etiology

KW - Prospective Studies

KW - Scleroderma, Diffuse/complications

KW - Severity of Illness Index

KW - Skin Ulcer/etiology

U2 - 10.1093/rheumatology/kex410

DO - 10.1093/rheumatology/kex410

M3 - Journal article

C2 - 29207002

SN - 1462-0324

VL - 57

SP - 370

EP - 381

JO - Rheumatology

JF - Rheumatology

IS - 2

ER -

Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study

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