Utility of the EULAR Sjögren syndrome disease activity index in Japanese children: a retrospective multicenter cohort study.

Adolescent Child Cohort Studies Europe Female Glucocorticoids / therapeutic use Humans Immunosuppressive Agents / therapeutic use Japan Male Outcome Assessment, Health Care Prednisolone / therapeutic use Retrospective Studies Severity of Illness Index Sjogren's Syndrome / drug therapy Societies, Medical
Children ESSDAI Primary Sjögren’s syndrome Treatment

Journal

Pediatric rheumatology online journal
ISSN: 1546-0096
Titre abrégé: Pediatr Rheumatol Online J
Pays: England
ID NLM: 101248897

Informations de publication

Date de publication:
17 Sep 2020
Historique:
received: 14 02 2020
accepted: 03 08 2020
entrez: 18 9 2020
pubmed: 19 9 2020
medline: 3 8 2021
Statut: epublish

Résumé

The European League Against Rheumatism (EULAR) Sjögren Syndrome Disease Activity Index (ESSDAI) has been utilized to assess Sjögren syndrome-related systemic involvement in adult patients. To date, however, the ESSDAI has not been validated in children with primary Sjögren's syndrome. This study evaluated the applicability of the ESSDAI to Japanese children with primary Sjögren's syndrome. The medical records of children who had been diagnosed with Sjogren syndrome at age ≤ 16 years between June 2011 and October 2016 were collected, and their ESSDAIs at initial presentation were calculated. Clinical symptoms and treatment regimens were surveyed by questionnaire, and patients were divided into groups based on ESSDAI and glucocorticoid dosages. The associations of ESSDAI scores with treatment regimens were analyzed statistically. The study subjects included 31 children (3 boys, 28 girls) with primary Sjögren's syndrome. Their median age at disease onset was 10 years (interquartile range [IQR], 8-13 years), and their median initial ESSDAI was 7.0 (IQR; 5.0-15.0). ESSDAI-determined disease activity was high in nine patients (29.0%), moderate in 15 (48.4%), and low in seven (22.6%). During the first year after their initial visit, 14 patients (45.2%) were treated with prednisolone (PSL) and six (19.4%) with immunosuppressants. Dose of PSL was significantly associated with ESSDAI score. Median ESSDAI score was significantly higher in patients treated with high/medium- than with no/low-dose PSL (16.5 [IQR 10.5-18.0] vs 5.0 [IQR 3.0-8.5]). Eight (66.7%) of 12 patients administered medium/high-dose PSL and one (5.3%) of 19 administered no/low-dose PSL had high disease activity on ESSDAI. Disease activity assessed by ESSDAI tended to be consistent with disease activity assessed by pediatric rheumatologists in determining treatment regimens. ESSDAI is useful for assessing disease activity in Japanese children with primary Sjögren's syndrome.

Sections du résumé

BACKGROUND BACKGROUND
The European League Against Rheumatism (EULAR) Sjögren Syndrome Disease Activity Index (ESSDAI) has been utilized to assess Sjögren syndrome-related systemic involvement in adult patients. To date, however, the ESSDAI has not been validated in children with primary Sjögren's syndrome. This study evaluated the applicability of the ESSDAI to Japanese children with primary Sjögren's syndrome.
METHODS METHODS
The medical records of children who had been diagnosed with Sjogren syndrome at age ≤ 16 years between June 2011 and October 2016 were collected, and their ESSDAIs at initial presentation were calculated. Clinical symptoms and treatment regimens were surveyed by questionnaire, and patients were divided into groups based on ESSDAI and glucocorticoid dosages. The associations of ESSDAI scores with treatment regimens were analyzed statistically.
RESULTS RESULTS
The study subjects included 31 children (3 boys, 28 girls) with primary Sjögren's syndrome. Their median age at disease onset was 10 years (interquartile range [IQR], 8-13 years), and their median initial ESSDAI was 7.0 (IQR; 5.0-15.0). ESSDAI-determined disease activity was high in nine patients (29.0%), moderate in 15 (48.4%), and low in seven (22.6%). During the first year after their initial visit, 14 patients (45.2%) were treated with prednisolone (PSL) and six (19.4%) with immunosuppressants. Dose of PSL was significantly associated with ESSDAI score. Median ESSDAI score was significantly higher in patients treated with high/medium- than with no/low-dose PSL (16.5 [IQR 10.5-18.0] vs 5.0 [IQR 3.0-8.5]). Eight (66.7%) of 12 patients administered medium/high-dose PSL and one (5.3%) of 19 administered no/low-dose PSL had high disease activity on ESSDAI.
CONCLUSION CONCLUSIONS
Disease activity assessed by ESSDAI tended to be consistent with disease activity assessed by pediatric rheumatologists in determining treatment regimens. ESSDAI is useful for assessing disease activity in Japanese children with primary Sjögren's syndrome.

Identifiants

DOI: 10.1186/s12969-020-00458-1 PMID: 32943063 PMC: PMC7499954
pubmed: 32943063
doi: 10.1186/s12969-020-00458-1
pii: 10.1186/s12969-020-00458-1
pmc: PMC7499954
doi:

Substances chimiques

Glucocorticoids 0
Immunosuppressive Agents 0
Prednisolone 9PHQ9Y1OLM

Types de publication

Journal Article Multicenter Study

Langues

eng

Sous-ensembles de citation

IM

Pagination

73

Subventions

Organisme : Ministry of Health, Labour and Welfare
ID : 201510096A

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Auteurs

Naomi Iwata (N)

Department of Infection and Immunology, Aichi Children's Health and Medical Center, Obu, Japan. naomi_iwata@mx.achmc.pref.aichi.jp.
The Japan Pediatric Sjögren's Syndrome Study Group, Tokyo, Japan. naomi_iwata@mx.achmc.pref.aichi.jp.
ORCID: 0000-0001-6149-1358

Minako Tomiita (M)

The Japan Pediatric Sjögren's Syndrome Study Group, Tokyo, Japan.
Department of Pediatrics, Shimoshizu National Hospital, Yotsukaido, Japan.

Ichiro Kobayashi (I)

The Japan Pediatric Sjögren's Syndrome Study Group, Tokyo, Japan.
Center for Pediatric Allergy and Rheumatology, KKR Sapporo Medical Center, Sapporo, Japan.

Yusaburo Inoue (Y)

The Japan Pediatric Sjögren's Syndrome Study Group, Tokyo, Japan.
Department of Allergy and Rheumatology, Chiba Children's Hospital, Chiba, Japan.

Yukiko Nonaka (Y)

The Japan Pediatric Sjögren's Syndrome Study Group, Tokyo, Japan.
Department of Pediatrics, Kagoshima University Hospital, Kagoshima, Japan.

Nami Okamoto (N)

The Japan Pediatric Sjögren's Syndrome Study Group, Tokyo, Japan.
Department of Pediatrics, Osaka Medical College, Osaka, Japan.

Hiroaki Umebayashi (H)

The Japan Pediatric Sjögren's Syndrome Study Group, Tokyo, Japan.
Department of General Pediatrics, Miyagi Children's Hospital, Sendai, Japan.

Ryoki Hara (R)

The Japan Pediatric Sjögren's Syndrome Study Group, Tokyo, Japan.
Department of Pediatrics, Yokohama City University School of Medicine, Yokohama, Japan.

Yasuhiko Ito (Y)

The Japan Pediatric Sjögren's Syndrome Study Group, Tokyo, Japan.
Department of Pediatrics, Nippon Medical School, Tokyo, Japan.

Yasunori Sato (Y)

Department of Preventive Medicine and Public Health, Keio University School of Medicine, Tokyo, Japan.

Masaaki Mori (M)

The Japan Pediatric Sjögren's Syndrome Study Group, Tokyo, Japan.
Department of Lifetime Clinical Immunology, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, Tokyo, Japan.

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