Validation of a score tool for measurement of histological severity in juvenile dermatomyositis and association with clinical severity of disease

Hemlata Varsani; Susan C. Charman; Charles K. Li; Suely K.N. Marie; Anthony A. Amato; Brenda Banwell; Kevin E. Bove; Andrea M. Corse; Alison M. Emslie-Smith; Thomas S. Jacques; Ingrid E. Lundberg; Carlo Minetti; Inger Nennesmo; Elisabeth J. Rushing; Adriana M.E. Sallum; Caroline Sewry; Clarissa A. Pilkington; Janice L. Holton; Lucy R. Wedderburn; Liza McCann; Ian Roberts; Eileen Baildam; Louise Hanna; Olivia Lloyd; Phil Riley; Ann McGovern; Clive Ryder; Janis Scott; Sue Wyatt; Gillian Jackson; Tania Amin; Mark Wood; Vanessa Van Rooyen; Joyce Davidson; Janet Gardner-Medwin; Neil Martin; Sue Ferguson; Liz Waxman; Mark Friswell; Helen Foster; Alison Swift; Sharmila Jandial; Vicky Stevenson; Debbie Wade; Ethan Sen; Eve Smith; Lisa Qiao; Helen Venning; Rangaraj Satyapal; Elizabeth Stretton; Mary Jordan; Kate Armon; Joe Ellis-Gage; Holly Roper; Clarissa Pilkington; N. Hasson; Sue Maillard; Elizabeth Halkon; Virginia Brown; Audrey Juggins; Sally Smith; Sian Lunt; Elli Enayat; Miss Laura Beard; Miss Laura Kassoumeri; Miss Katie Arnold; Kevin Murray; John Ioannou; Linda Suffield

doi:10.1136/annrheumdis-2013-203396

Validation of a score tool for measurement of histological severity in juvenile dermatomyositis and association with clinical severity of disease

Hemlata Varsani, Susan C. Charman, Charles K. Li, Suely K.N. Marie, Anthony A. Amato, Brenda Banwell, Kevin E. Bove, Andrea M. Corse, Alison M. Emslie-Smith, Thomas S. Jacques, Ingrid E. Lundberg, Carlo Minetti, Inger Nennesmo, Elisabeth J. Rushing, Adriana M.E. Sallum, Caroline Sewry, Clarissa A. Pilkington, Janice L. Holton, Lucy R. Wedderburn, Liza McCannIan Roberts, Eileen Baildam, Louise Hanna, Olivia Lloyd, Phil Riley, Ann McGovern, Clive Ryder, Janis Scott, Sue Wyatt, Gillian Jackson, Tania Amin, Mark Wood, Vanessa Van Rooyen, Joyce Davidson, Janet Gardner-Medwin, Neil Martin, Sue Ferguson, Liz Waxman, Mark Friswell, Helen Foster, Alison Swift, Sharmila Jandial, Vicky Stevenson, Debbie Wade, Ethan Sen, Eve Smith, Lisa Qiao, Helen Venning, Rangaraj Satyapal, Elizabeth Stretton, Mary Jordan, Kate Armon, Joe Ellis-Gage, Holly Roper, Clarissa Pilkington, N. Hasson, Sue Maillard, Elizabeth Halkon, Virginia Brown, Audrey Juggins, Sally Smith, Sian Lunt, Elli Enayat, Miss Laura Beard, Miss Laura Kassoumeri, Miss Katie Arnold, Kevin Murray, John Ioannou, Linda Suffield

School of Medicine

Research output: Contribution to journal › Article › peer-review

31 Scopus citations

Abstract

Objectives: To study muscle biopsy tissue from patients with juvenile dermatomyositis (JDM) in order to test the reliability of a score tool designed to quantify the severity of histological abnormalities when applied to biceps humeri in addition to quadriceps femoris. Additionally, to evaluate whether elements of the tool correlate with clinical measures of disease severity.

Methods: 55 patients with JDM with muscle biopsy tissue and clinical data available were included. Biopsy samples (33 quadriceps, 22 biceps) were prepared and stained using standardised protocols. A Latin square design was used by the International Juvenile Dermatomyositis Biopsy Consensus Group to score cases using our previously published score tool. Reliability was assessed by intraclass correlation coefficient (ICC) and scorer agreement (α) by assessing variation in scorers' ratings. Scores from the most reliable tool items correlated with clinical measures of disease activity at the time of biopsy. Results Inter- and intraobserver agreement was good or high for many tool items, including overall assessment of severity using a Visual Analogue Scale. The tool functioned equally well on biceps and quadriceps samples. A modified tool using the most reliable score items showed good correlation with measures of disease activity.

Conclusions: The JDM biopsy score tool has high interand intraobserver agreement and can be used on both biceps and quadriceps muscle tissue. Importantly, the modified tool correlates well with clinical measures of disease activity. We propose that standardised assessment of muscle biopsy tissue should be considered in diagnostic investigation and clinical trials in JDM.

Original language	English (US)
Pages (from-to)	204-210
Number of pages	7
Journal	Annals of the rheumatic diseases
Volume	74
Issue number	1
DOIs	https://doi.org/10.1136/annrheumdis-2013-203396
State	Published - Jan 1 2015

ASJC Scopus subject areas

Rheumatology
Immunology and Allergy
Immunology
General Biochemistry, Genetics and Molecular Biology

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10.1136/annrheumdis-2013-203396

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Varsani, H., Charman, S. C., Li, C. K., Marie, S. K. N., Amato, A. A., Banwell, B., Bove, K. E., Corse, A. M., Emslie-Smith, A. M., Jacques, T. S., Lundberg, I. E., Minetti, C., Nennesmo, I., Rushing, E. J., Sallum, A. M. E., Sewry, C., Pilkington, C. A., Holton, J. L., Wedderburn, L. R., ... Suffield, L. (2015). Validation of a score tool for measurement of histological severity in juvenile dermatomyositis and association with clinical severity of disease. Annals of the rheumatic diseases, 74(1), 204-210. https://doi.org/10.1136/annrheumdis-2013-203396

Varsani, H, Charman, SC, Li, CK, Marie, SKN, Amato, AA, Banwell, B, Bove, KE, Corse, AM, Emslie-Smith, AM, Jacques, TS, Lundberg, IE, Minetti, C, Nennesmo, I, Rushing, EJ, Sallum, AME, Sewry, C, Pilkington, CA, Holton, JL, Wedderburn, LR, McCann, L, Roberts, I, Baildam, E, Hanna, L, Lloyd, O, Riley, P, McGovern, A, Ryder, C, Scott, J, Wyatt, S, Jackson, G, Amin, T, Wood, M, Van Rooyen, V, Davidson, J, Gardner-Medwin, J, Martin, N, Ferguson, S, Waxman, L, Friswell, M, Foster, H, Swift, A, Jandial, S, Stevenson, V, Wade, D, Sen, E, Smith, E, Qiao, L, Venning, H, Satyapal, R, Stretton, E, Jordan, M, Armon, K, Ellis-Gage, J, Roper, H, Pilkington, C, Hasson, N, Maillard, S, Halkon, E, Brown, V, Juggins, A, Smith, S, Lunt, S, Enayat, E, Beard, ML, Kassoumeri, ML, Arnold, MK, Murray, K, Ioannou, J & Suffield, L 2015, 'Validation of a score tool for measurement of histological severity in juvenile dermatomyositis and association with clinical severity of disease', Annals of the rheumatic diseases, vol. 74, no. 1, pp. 204-210. https://doi.org/10.1136/annrheumdis-2013-203396

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title = "Validation of a score tool for measurement of histological severity in juvenile dermatomyositis and association with clinical severity of disease",

abstract = "Objectives: To study muscle biopsy tissue from patients with juvenile dermatomyositis (JDM) in order to test the reliability of a score tool designed to quantify the severity of histological abnormalities when applied to biceps humeri in addition to quadriceps femoris. Additionally, to evaluate whether elements of the tool correlate with clinical measures of disease severity.Methods: 55 patients with JDM with muscle biopsy tissue and clinical data available were included. Biopsy samples (33 quadriceps, 22 biceps) were prepared and stained using standardised protocols. A Latin square design was used by the International Juvenile Dermatomyositis Biopsy Consensus Group to score cases using our previously published score tool. Reliability was assessed by intraclass correlation coefficient (ICC) and scorer agreement (α) by assessing variation in scorers' ratings. Scores from the most reliable tool items correlated with clinical measures of disease activity at the time of biopsy. Results Inter- and intraobserver agreement was good or high for many tool items, including overall assessment of severity using a Visual Analogue Scale. The tool functioned equally well on biceps and quadriceps samples. A modified tool using the most reliable score items showed good correlation with measures of disease activity.Conclusions: The JDM biopsy score tool has high interand intraobserver agreement and can be used on both biceps and quadriceps muscle tissue. Importantly, the modified tool correlates well with clinical measures of disease activity. We propose that standardised assessment of muscle biopsy tissue should be considered in diagnostic investigation and clinical trials in JDM.",

author = "Hemlata Varsani and Charman, {Susan C.} and Li, {Charles K.} and Marie, {Suely K.N.} and Amato, {Anthony A.} and Brenda Banwell and Bove, {Kevin E.} and Corse, {Andrea M.} and Emslie-Smith, {Alison M.} and Jacques, {Thomas S.} and Lundberg, {Ingrid E.} and Carlo Minetti and Inger Nennesmo and Rushing, {Elisabeth J.} and Sallum, {Adriana M.E.} and Caroline Sewry and Pilkington, {Clarissa A.} and Holton, {Janice L.} and Wedderburn, {Lucy R.} and Liza McCann and Ian Roberts and Eileen Baildam and Louise Hanna and Olivia Lloyd and Phil Riley and Ann McGovern and Clive Ryder and Janis Scott and Sue Wyatt and Gillian Jackson and Tania Amin and Mark Wood and {Van Rooyen}, Vanessa and Joyce Davidson and Janet Gardner-Medwin and Neil Martin and Sue Ferguson and Liz Waxman and Mark Friswell and Helen Foster and Alison Swift and Sharmila Jandial and Vicky Stevenson and Debbie Wade and Ethan Sen and Eve Smith and Lisa Qiao and Helen Venning and Rangaraj Satyapal and Elizabeth Stretton and Mary Jordan and Kate Armon and Joe Ellis-Gage and Holly Roper and Clarissa Pilkington and N. Hasson and Sue Maillard and Elizabeth Halkon and Virginia Brown and Audrey Juggins and Sally Smith and Sian Lunt and Elli Enayat and Beard, {Miss Laura} and Kassoumeri, {Miss Laura} and Arnold, {Miss Katie} and Kevin Murray and John Ioannou and Linda Suffield",

year = "2015",

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doi = "10.1136/annrheumdis-2013-203396",

language = "English (US)",

volume = "74",

pages = "204--210",

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T1 - Validation of a score tool for measurement of histological severity in juvenile dermatomyositis and association with clinical severity of disease

AU - Varsani, Hemlata

AU - Charman, Susan C.

AU - Li, Charles K.

AU - Marie, Suely K.N.

AU - Amato, Anthony A.

AU - Banwell, Brenda

AU - Bove, Kevin E.

AU - Corse, Andrea M.

AU - Emslie-Smith, Alison M.

AU - Jacques, Thomas S.

AU - Lundberg, Ingrid E.

AU - Minetti, Carlo

AU - Nennesmo, Inger

AU - Rushing, Elisabeth J.

AU - Sallum, Adriana M.E.

AU - Sewry, Caroline

AU - Pilkington, Clarissa A.

AU - Holton, Janice L.

AU - Wedderburn, Lucy R.

AU - McCann, Liza

AU - Roberts, Ian

AU - Baildam, Eileen

AU - Hanna, Louise

AU - Lloyd, Olivia

AU - Riley, Phil

AU - McGovern, Ann

AU - Ryder, Clive

AU - Scott, Janis

AU - Wyatt, Sue

AU - Jackson, Gillian

AU - Amin, Tania

AU - Wood, Mark

AU - Van Rooyen, Vanessa

AU - Davidson, Joyce

AU - Gardner-Medwin, Janet

AU - Martin, Neil

AU - Ferguson, Sue

AU - Waxman, Liz

AU - Friswell, Mark

AU - Foster, Helen

AU - Swift, Alison

AU - Jandial, Sharmila

AU - Stevenson, Vicky

AU - Wade, Debbie

AU - Sen, Ethan

AU - Smith, Eve

AU - Qiao, Lisa

AU - Venning, Helen

AU - Satyapal, Rangaraj

AU - Stretton, Elizabeth

AU - Jordan, Mary

AU - Armon, Kate

AU - Ellis-Gage, Joe

AU - Roper, Holly

AU - Pilkington, Clarissa

AU - Hasson, N.

AU - Maillard, Sue

AU - Halkon, Elizabeth

AU - Brown, Virginia

AU - Juggins, Audrey

AU - Smith, Sally

AU - Lunt, Sian

AU - Enayat, Elli

AU - Beard, Miss Laura

AU - Kassoumeri, Miss Laura

AU - Arnold, Miss Katie

AU - Murray, Kevin

AU - Ioannou, John

AU - Suffield, Linda

PY - 2015/1/1

Y1 - 2015/1/1

N2 - Objectives: To study muscle biopsy tissue from patients with juvenile dermatomyositis (JDM) in order to test the reliability of a score tool designed to quantify the severity of histological abnormalities when applied to biceps humeri in addition to quadriceps femoris. Additionally, to evaluate whether elements of the tool correlate with clinical measures of disease severity.Methods: 55 patients with JDM with muscle biopsy tissue and clinical data available were included. Biopsy samples (33 quadriceps, 22 biceps) were prepared and stained using standardised protocols. A Latin square design was used by the International Juvenile Dermatomyositis Biopsy Consensus Group to score cases using our previously published score tool. Reliability was assessed by intraclass correlation coefficient (ICC) and scorer agreement (α) by assessing variation in scorers' ratings. Scores from the most reliable tool items correlated with clinical measures of disease activity at the time of biopsy. Results Inter- and intraobserver agreement was good or high for many tool items, including overall assessment of severity using a Visual Analogue Scale. The tool functioned equally well on biceps and quadriceps samples. A modified tool using the most reliable score items showed good correlation with measures of disease activity.Conclusions: The JDM biopsy score tool has high interand intraobserver agreement and can be used on both biceps and quadriceps muscle tissue. Importantly, the modified tool correlates well with clinical measures of disease activity. We propose that standardised assessment of muscle biopsy tissue should be considered in diagnostic investigation and clinical trials in JDM.

AB - Objectives: To study muscle biopsy tissue from patients with juvenile dermatomyositis (JDM) in order to test the reliability of a score tool designed to quantify the severity of histological abnormalities when applied to biceps humeri in addition to quadriceps femoris. Additionally, to evaluate whether elements of the tool correlate with clinical measures of disease severity.Methods: 55 patients with JDM with muscle biopsy tissue and clinical data available were included. Biopsy samples (33 quadriceps, 22 biceps) were prepared and stained using standardised protocols. A Latin square design was used by the International Juvenile Dermatomyositis Biopsy Consensus Group to score cases using our previously published score tool. Reliability was assessed by intraclass correlation coefficient (ICC) and scorer agreement (α) by assessing variation in scorers' ratings. Scores from the most reliable tool items correlated with clinical measures of disease activity at the time of biopsy. Results Inter- and intraobserver agreement was good or high for many tool items, including overall assessment of severity using a Visual Analogue Scale. The tool functioned equally well on biceps and quadriceps samples. A modified tool using the most reliable score items showed good correlation with measures of disease activity.Conclusions: The JDM biopsy score tool has high interand intraobserver agreement and can be used on both biceps and quadriceps muscle tissue. Importantly, the modified tool correlates well with clinical measures of disease activity. We propose that standardised assessment of muscle biopsy tissue should be considered in diagnostic investigation and clinical trials in JDM.

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Validation of a score tool for measurement of histological severity in juvenile dermatomyositis and association with clinical severity of disease

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