{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,29]],"date-time":"2026-10-29T11:54:04Z","timestamp":1793274844151,"version":"4.5.0"},"reference-count":56,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2005,12,9]],"date-time":"2005-12-09T00:00:00Z","timestamp":1134086400000},"content-version":"vor","delay-in-days":5701,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Arthritis &amp; Rheumatism"],"published-print":{"date-parts":[[1990,5]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Three generations of a nonconsanguineous family with premature onset of primary (idiopathic) osteoarthritis (OA) were studied for clues to the etiopathogenesis of their disorder. Articular symptoms began in their second and third decades of life and involved multiple joints, both typical and atypical for primary OA. Radiographs of the majority of involved peripheral joints showed abnormalities typical of primary OA. Evidence of chondrodysplasia was found in the spines. Pathologic examination of femoral heads obtained at total hip arthroplasty from 3 affected family members showed moderate to severe OA. Articular cartilage proteoglycans from these specimens were evaluated for aggregatability with hyaluronic acid, levels of chondroitin sulfate and keratan sulfate, and core protein structure. The results from each patient's specimen differed from the results of the other specimens. We conclude that this family's disorder, primary OA associated with a mild chondrodysplasia, was a late\u2010onset overlap form of an epiphyseal dysplasia, that a defect common to hyaline articular and physeal cartilage was primary, and that a single structural proteoglycan abnormality was not likely to be the underlying cause.<\/jats:p>","DOI":"10.1002\/art.1780330510","type":"journal-article","created":{"date-parts":[[2007,2,14]],"date-time":"2007-02-14T23:27:55Z","timestamp":1171495675000},"page":"674-684","source":"Crossref","is-referenced-by-count":24,"title":["Early\u2010onset primary osteoarthritis and mild chondrodysplasia"],"prefix":"10.1002","volume":"33","author":[{"given":"Paul L.","family":"Katzenstein","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charles J.","family":"Malemud","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mini N.","family":"Pathria","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John R.","family":"Carter","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert P.","family":"Sheon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roland W.","family":"Moskowitz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2005,12,9]]},"reference":[{"key":"e_1_2_1_2_2","volume-title":"Osteoarthritis, Diagnosis and Management","author":"Meachim G","year":"1984"},{"key":"e_1_2_1_3_2","volume-title":"Osteoarthritis, Diagnosis and Management","author":"Moskowitz RW","year":"1984"},{"key":"e_1_2_1_4_2","volume-title":"Osteoarthritis, Diagnosis and Management","author":"Schumacher HR","year":"1984"},{"key":"e_1_2_1_5_2","volume-title":"Heritable Disorders of Connective Tissue","author":"McKusick VA","year":"1972"},{"key":"e_1_2_1_6_2","doi-asserted-by":"crossref","first-page":"40","DOI":"10.1302\/0301-620X.46B1.40","article-title":"Persistent joint laxity and congenital dislocation of the hip.","volume":"46","author":"Carter C","year":"1964","journal-title":"J Bone Joint Surg"},{"key":"e_1_2_1_7_2","doi-asserted-by":"crossref","first-page":"79","DOI":"10.1097\/00003086-198305000-00013","article-title":"Acetabular dysplasia in the adult.","volume":"175","author":"Cooperman DR","year":"1983","journal-title":"Clin Orthop"},{"key":"e_1_2_1_8_2","doi-asserted-by":"publisher","DOI":"10.3109\/03008207809152628"},{"key":"e_1_2_1_9_2","doi-asserted-by":"crossref","first-page":"6151","DOI":"10.1016\/S0021-9258(19)41171-X","article-title":"Isolation and characterization of proteoglycans from the Swarm rat chondrosarcoma.","volume":"250","author":"Oegema TR","year":"1975","journal-title":"J Biol Chem"},{"key":"e_1_2_1_10_2","doi-asserted-by":"publisher","DOI":"10.1016\/0003-2697(62)90095-7"},{"key":"e_1_2_1_11_2","doi-asserted-by":"publisher","DOI":"10.1042\/bj2060329"},{"key":"e_1_2_1_12_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0021-9673(01)80009-1"},{"key":"e_1_2_1_13_2","first-page":"875","article-title":"Effects of polymethylmethacrylate on rabbit articular chondrocytes in monolayer culture.","volume":"189","author":"Boyd AD","year":"1984","journal-title":"Clin Orthop"},{"key":"e_1_2_1_14_2","first-page":"4234","article-title":"Aggregation of cartilage proteoglycans.","volume":"240","author":"Hascall VC","year":"1974","journal-title":"J Biol Chem"},{"key":"e_1_2_1_15_2","doi-asserted-by":"publisher","DOI":"10.3109\/03008208309004860"},{"key":"e_1_2_1_16_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0174-173X(86)80030-9"},{"key":"e_1_2_1_17_2","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.84.3.832"},{"key":"e_1_2_1_18_2","first-page":"7","article-title":"Changes in human osteoarthritic hip cartilage maintained in explant culture: implications for understanding repair in osteoarthritis.","volume":"77","author":"Malemud CJ","year":"1989","journal-title":"Scand J Rheumatol [Suppl]"},{"key":"e_1_2_1_19_2","doi-asserted-by":"crossref","first-page":"1536","DOI":"10.1016\/S0021-9258(18)93575-1","article-title":"Enzymatic methods for the determination of small quantities of isomeric chondroitin sulfates.","volume":"243","author":"Saito H","year":"1968","journal-title":"J Biol Chem"},{"key":"e_1_2_1_20_2","first-page":"25","article-title":"Keratansulphate\u2010containing proteoglycans in human osteochondrophytic spurs of the femoral head.","volume":"16","author":"Malemud CJ","year":"1988","journal-title":"Biochem Int"},{"key":"e_1_2_1_21_2","doi-asserted-by":"publisher","DOI":"10.1038\/227680a0"},{"key":"e_1_2_1_22_2","doi-asserted-by":"publisher","DOI":"10.1016\/0003-2697(80)90470-4"},{"key":"e_1_2_1_23_2","doi-asserted-by":"publisher","DOI":"10.1016\/0047-6374(88)90112-1"},{"key":"e_1_2_1_24_2","doi-asserted-by":"publisher","DOI":"10.1016\/0003-2697(83)90177-X"},{"key":"e_1_2_1_25_2","doi-asserted-by":"crossref","DOI":"10.1016\/B978-0-12-750650-0.50005-7","volume-title":"Monoclonal antibodies as probes for elucidating proteoglycan structure and function","author":"Caterson B","year":"1987"},{"key":"e_1_2_1_26_2","doi-asserted-by":"crossref","first-page":"239","DOI":"10.1016\/S0307-742X(21)00541-5","article-title":"Articular degeneration as a sequela of osteochondrodysplasias.","volume":"11","author":"Stanescu Y","year":"1985","journal-title":"Clin Rheum Dis"},{"key":"e_1_2_1_27_2","volume-title":"Atlas of Skeletal Dysplasias","author":"Wynne\u2010Davies R","year":"1985"},{"key":"e_1_2_1_28_2","volume-title":"Fairbank's Atlas of General Affections of the Skeleton","author":"Wynne\u2010Davies R","year":"1976"},{"key":"e_1_2_1_29_2","volume-title":"The chondrodysplasias, Principles and Practice of Medical Genetics","author":"Rimoin DL","year":"1983"},{"key":"e_1_2_1_30_2","doi-asserted-by":"publisher","DOI":"10.1136\/ard.43.2.275"},{"key":"e_1_2_1_31_2","first-page":"1917!","article-title":"Premature arthritis as manifestation of epiphyseal dysplasia.","volume":"66","author":"Levy RN","year":"1966","journal-title":"NY State J Med"},{"key":"e_1_2_1_32_2","first-page":"46","article-title":"The epiphyseal dysplasias.","volume":"114","author":"Spranger J","year":"1976","journal-title":"Clin Orthop"},{"key":"e_1_2_1_33_2","doi-asserted-by":"publisher","DOI":"10.1002\/bjs.18003915815"},{"key":"e_1_2_1_34_2","doi-asserted-by":"crossref","first-page":"1587","DOI":"10.2106\/00004623-197052080-00006","article-title":"A family study of spondyloepiphyseal dysplasia.","volume":"52","author":"Diamond LS","year":"1970","journal-title":"J Bone Joint Surg"},{"key":"e_1_2_1_35_2","doi-asserted-by":"crossref","first-page":"228","DOI":"10.1302\/0301-620X.37B2.228","article-title":"Dysplasia epiphysialis multiplex.","volume":"37","author":"Maudsley RH","year":"1955","journal-title":"J Bone Joint Surg"},{"key":"e_1_2_1_36_2","first-page":"449","article-title":"Familial precocious polyarticular osteoarthritis of chondrodysplastic type.","volume":"65","author":"Brown AR","year":"1966","journal-title":"NZ Med J"},{"key":"e_1_2_1_37_2","doi-asserted-by":"publisher","DOI":"10.1002\/art.1780130106"},{"key":"e_1_2_1_38_2","first-page":"1061","article-title":"Pathologic features of the femoral heads in a patient aged 14 1\/2 years with spondyloepiphyseal dysplasia with osteoarthritis.","volume":"14","author":"Stanescu R","year":"1987","journal-title":"J Rheumatol"},{"key":"e_1_2_1_39_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0046-8177(85)80059-9"},{"key":"e_1_2_1_40_2","first-page":"137","article-title":"Chondroosseous pathology in the chondrodystrophies.","volume":"114","author":"Rimoin DL","year":"1976","journal-title":"Clin Orthop"},{"key":"e_1_2_1_41_2","doi-asserted-by":"publisher","DOI":"10.2106\/00004623-198466060-00002"},{"key":"e_1_2_1_42_2","doi-asserted-by":"publisher","DOI":"10.1159\/000157111"},{"key":"e_1_2_1_43_2","doi-asserted-by":"publisher","DOI":"10.1007\/BF00710070"},{"key":"e_1_2_1_44_2","doi-asserted-by":"publisher","DOI":"10.1126\/science.2543071"},{"key":"e_1_2_1_45_2","doi-asserted-by":"publisher","DOI":"10.1007\/BF00441206"},{"key":"e_1_2_1_46_2","doi-asserted-by":"publisher","DOI":"10.1016\/8756-3282(88)90118-4"},{"key":"e_1_2_1_47_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.1982.tb09434.x"},{"key":"e_1_2_1_48_2","doi-asserted-by":"crossref","first-page":"181","DOI":"10.1042\/bj2300181","article-title":"The core proteins of large and small interstitial proteoglycans from various connective tissues form distinct groups.","volume":"230","author":"Heing\u00e5rd D","year":"1985","journal-title":"Biochem J"},{"key":"e_1_2_1_49_2","doi-asserted-by":"publisher","DOI":"10.1002\/jor.1100010310"},{"key":"e_1_2_1_50_2","first-page":"23","article-title":"Quantitation of component proteoglycans during long\u2010term organ explant culture of human osteoarthritic hip cartilage.","volume":"113","author":"Shuckett R","year":"1989","journal-title":"J Lab Clin Med"},{"key":"e_1_2_1_51_2","doi-asserted-by":"publisher","DOI":"10.1042\/bj2310129"},{"key":"e_1_2_1_52_2","doi-asserted-by":"publisher","DOI":"10.2106\/00004623-198466010-00013"},{"key":"e_1_2_1_53_2","first-page":"321","article-title":"Newly synthesized and endogenous proteoglycans in human osteoarthritic knee cartilage.","volume":"14","author":"Martel\u2010Pelletier J","year":"1987","journal-title":"J Rheumatol"},{"key":"e_1_2_1_54_2","doi-asserted-by":"publisher","DOI":"10.1172\/JCI106660"},{"key":"e_1_2_1_55_2","doi-asserted-by":"publisher","DOI":"10.1136\/ard.47.10.801"},{"key":"e_1_2_1_56_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM198408093110606"},{"key":"e_1_2_1_57_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199002223220807"}],"container-title":["Arthritis &amp; Rheumatism"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fart.1780330510","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/art.1780330510","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,11,23]],"date-time":"2023-11-23T22:20:41Z","timestamp":1700778041000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/art.1780330510"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[1990,5]]},"references-count":56,"journal-issue":{"issue":"5","published-print":{"date-parts":[[1990,5]]}},"alternative-id":["10.1002\/art.1780330510"],"URL":"https:\/\/doi.org\/10.1002\/art.1780330510","archive":["Portico"],"relation":{},"ISSN":["0004-3591","1529-0131"],"issn-type":[{"value":"0004-3591","type":"print"},{"value":"1529-0131","type":"electronic"}],"subject":[],"published":{"date-parts":[[1990,5]]}}}