{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,9,11]],"date-time":"2025-09-11T17:26:46Z","timestamp":1757611606736,"version":"3.44.0"},"reference-count":21,"publisher":"BMJ","issue":"1","funder":[{"name":"CureJM Foundation Program of Excellence in Care and Research"}],"content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["RMD Open"],"accepted":{"date-parts":[[2017,6,9]]},"published-print":{"date-parts":[[2017,7]]},"abstract":"<jats:p>The study aimed to document the utility of the absolute number of natural killer cells as a biomarker in paediatric orbital myositis (OM). Extracted data from four children with OM included demographics, laboratory values, imaging and treatment response. Stored sera (\u221280\u00b0C) were tested for IgG4 levels in three cases and antibody to Coxsackie B in two cases. Their first symptom was at 14.4\u00b11.2 years (mean\u00b1SD). At diagnosis three had creatine phosphokinase (CPK) of 97.3\u00b144.2, aldolase of 8.5\u00b12.8 (n=2), alanine aminotransferase (ALT) of 13\u00b12.8 (n=2) and aspartate aminotransferase (AST) of 21.3\u00b12.9. IG4 level was 87.7\u00b166 (normal=8\u201389\u2009mg\/dL); two sera (patients 1and4) were positive (&gt;1:8 dilution) for anti-Coxsackievirus antigen B5. The CD3-CD16+CD56+ natural killer absolute count was 96.7\u00b128.7 (lower limit of normal=138), increasing to 163\u00b157.2 with disease resolution in three patients. The fourth patient was followed elsewhere. CT showed involvement of bilateral superior oblique, lateral rectus or the left medial rectus muscles. Treatment included intravenous methylprednisolone, methotrexate (n=2) and other immunosuppressants. Paediatric OM disease activity was associated with initially low absolute CD3-CD16+CD56+ natural killer cell counts, which normalised with improvement. We speculate (1) infection, such as Coxsackie B virus, may be associated with paediatric OM; and (2) the absolute count of circulating CD3-CD16+CD56+ natural killer lymphocytes may serve as a biomarker to guide medical therapy.<\/jats:p>","DOI":"10.1136\/rmdopen-2016-000385","type":"journal-article","created":{"date-parts":[[2017,7,11]],"date-time":"2017-07-11T20:40:11Z","timestamp":1499805611000},"page":"e000385","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":8,"title":["Decreased CD3-CD16+CD56+ natural killer cell counts in children with orbital myositis: a clue to disease activity"],"prefix":"10.1136","volume":"3","author":[{"given":"Melissa R","family":"Briones","sequence":"first","affiliation":[{"name":"Division of Allergy, Immunology, and Rheumatology, Loyola University Medical Center, Maywood, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gabrielle A","family":"Morgan","sequence":"additional","affiliation":[{"name":"Pediatrics, Stanly Manne Children's Research Institute, Children's Hospital of Chicago; Ann and Robert H. Lurie Children's Hospital, Chicago, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maria C","family":"Amoruso","sequence":"additional","affiliation":[{"name":"Pediatrics, Stanly Manne Children's Research Institute, Children's Hospital of Chicago; Ann and Robert H. Lurie Children's Hospital, Chicago, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bahram","family":"Rahmani","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Northwestern University Feinberg School of Medicine, Chicago, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maura E","family":"Ryan","sequence":"additional","affiliation":[{"name":"Department of Medical Imaging, Ann & Robert H Lurie Children\u2019s Hospital of Chicago, Northwestern University Feinberg School of Medicine, Chicago, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lauren M.","family":"Pachman","sequence":"additional","affiliation":[{"name":"Division of Pediatric Rheumatology, Ann and Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine, Chicago, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2017,7,11]]},"reference":[{"key":"2025090313073477000_3.1.e000385.1","doi-asserted-by":"publisher","DOI":"10.1007\/s11882-012-0319-7"},{"key":"2025090313073477000_3.1.e000385.2","doi-asserted-by":"publisher","DOI":"10.1007\/s11882-009-0045-y"},{"key":"2025090313073477000_3.1.e000385.3","doi-asserted-by":"publisher","DOI":"10.1080\/01676830500505608"},{"key":"2025090313073477000_3.1.e000385.4","doi-asserted-by":"publisher","DOI":"10.1016\/j.rdc.2013.06.001"},{"key":"2025090313073477000_3.1.e000385.5","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajo.2010.05.012"},{"key":"2025090313073477000_3.1.e000385.6","doi-asserted-by":"publisher","DOI":"10.1016\/j.survophthal.2011.07.004"},{"key":"2025090313073477000_3.1.e000385.7","doi-asserted-by":"publisher","DOI":"10.1136\/bjo.2010.191866"},{"key":"2025090313073477000_3.1.e000385.8","doi-asserted-by":"crossref","first-page":"173","DOI":"10.1179\/1465328111Y.0000000004","article-title":"Recurrent bilateral orbital myositis: case report and review of the literature","volume":"31","author":"Abd-Rashid","year":"2011","journal-title":"Ann Trop Paediatr"},{"key":"2025090313073477000_3.1.e000385.9","doi-asserted-by":"crossref","first-page":"677","DOI":"10.1001\/jamapediatrics.2013.4525","article-title":"Adolescent with right orbital swelling and proptosis. Idiopathic orbital inflammatory syndrome","volume":"168","author":"Chang","year":"2014","journal-title":"JAMA Pediatr"},{"key":"2025090313073477000_3.1.e000385.10","doi-asserted-by":"crossref","unstructured":"Gil P , Gil J , Paiva C , et al . Medical and Surgical Treatment in Pediatric Orbital Myositis Associated with Coxsackie Virus. Case Rep Ophthalmol Med 2015;:1\u20134 Art ID 917275.doi:10.1155\/2015\/917275","DOI":"10.1155\/2015\/917275"},{"key":"2025090313073477000_3.1.e000385.11","doi-asserted-by":"crossref","first-page":"e86","DOI":"10.1097\/IOP.0b013e318275b649","article-title":"Atypical idiopathic orbital inflammation in a young girl","volume":"29","author":"Spindle","year":"2013","journal-title":"Ophthal Plast Reconstr Surg"},{"key":"2025090313073477000_3.1.e000385.12","doi-asserted-by":"crossref","first-page":"585","DOI":"10.1111\/j.1445-5994.1987.tb01264.x","article-title":"Orbital myositis: a study of six cases","volume":"17","author":"Hankey","year":"1987","journal-title":"Aust N Z J Med"},{"key":"2025090313073477000_3.1.e000385.13","doi-asserted-by":"crossref","first-page":"270","DOI":"10.1097\/IOP.0000000000000494","article-title":"Pediatric Idiopathic Orbital Inflammation: Clinical Features of 30 Cases","volume":"32","author":"Spindle","year":"2016","journal-title":"Ophthal Plast Reconstr Surg"},{"key":"2025090313073477000_3.1.e000385.14","first-page":"1374","article-title":"Treatment of orbital myositis with adalimumab (Humira)","volume":"32","author":"Adams","year":"2005","journal-title":"J Rheumatol"},{"key":"2025090313073477000_3.1.e000385.15","doi-asserted-by":"publisher","DOI":"10.1001\/archopht.1978.03910060164013"},{"key":"2025090313073477000_3.1.e000385.16","doi-asserted-by":"crossref","first-page":"664","DOI":"10.1016\/j.survophthal.2016.03.002","article-title":"Orbital inflammation: Biopsy first","volume":"61","author":"Mombaerts","year":"2016","journal-title":"Surv Ophthalmol"},{"key":"2025090313073477000_3.1.e000385.17","first-page":"S225","article-title":"Absolute number of circulating CD3-CD56+\/CD16+ natural killer (NK) cells, a potential biomarker of disease activity in Juvenile Dermatomyositis (JDM)","volume":"58","author":"Pachman","year":"2008","journal-title":"Arthritis Rheum"},{"key":"2025090313073477000_3.1.e000385.18","first-page":"640","article-title":"Natural and antibody-dependent cellular cytotoxicity in children with systemic lupus erythematosus and juvenile dermatomyositis","volume":"10","author":"Miller","year":"1983","journal-title":"J Rheumatol"},{"key":"2025090313073477000_3.1.e000385.19","doi-asserted-by":"publisher","DOI":"10.3899\/jrheum.121031"},{"key":"2025090313073477000_3.1.e000385.20","doi-asserted-by":"crossref","first-page":"470","DOI":"10.5114\/ceji.2015.56971","article-title":"The role of natural killer cells in pathogenesis of autoimmune diseases","volume":"40","author":"Popko","year":"2015","journal-title":"Cent Eur J Immunol"},{"key":"2025090313073477000_3.1.e000385.21","doi-asserted-by":"publisher","DOI":"10.1002\/art.1780291109"}],"container-title":["RMD Open"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/rmdopen-2016-000385","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,9,3]],"date-time":"2025-09-03T20:07:43Z","timestamp":1756930063000},"score":1,"resource":{"primary":{"URL":"https:\/\/rmdopen.bmj.com\/lookup\/doi\/10.1136\/rmdopen-2016-000385"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2017,7]]},"references-count":21,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2017,7,11]]},"published-print":{"date-parts":[[2017,7]]}},"alternative-id":["10.1136\/rmdopen-2016-000385"],"URL":"https:\/\/doi.org\/10.1136\/rmdopen-2016-000385","relation":{},"ISSN":["2056-5933"],"issn-type":[{"type":"electronic","value":"2056-5933"}],"subject":[],"published":{"date-parts":[[2017,7]]}}}