High Prevalence of Prothrombotic Abnormalities in Multifocal Osteonecrosis Description of a Series and Review of the Literature

被引:24
作者
Gomez-Puerta, Jose A. [1 ,4 ]
Peris, Pilar [1 ]
Carles Reverter, Joan [2 ]
Espinosa, Gerard [3 ]
Martinez-Ferrer, Angeles [1 ]
Monegal, Ana [1 ]
Monteagudo, Juan [2 ]
Tassies, Dolors [2 ]
Guanabens, Nuria [1 ]
机构
[1] CIBERehd, Dept Rheumatol, Madrid, Spain
[2] Hosp Clin Barcelona, Hemotherapy & Haemostasis Serv, Barcelona, Spain
[3] Univ Barcelona, Hosp Clin, Dept Autoimmune Dis, Barcelona, Spain
[4] Brigham & Womens Hosp, Div Rheumatol Immunol & Allergy, Boston, MA 02115 USA
关键词
PRIMARY ANTIPHOSPHOLIPID SYNDROME; FACTOR-VIII LEVELS; FACTOR-V-LEIDEN; VON-WILLEBRAND-FACTOR; FEMORAL-HEAD; AVASCULAR NECROSIS; VENOUS THROMBOEMBOLISM; NATURAL-HISTORY; GENE-MUTATIONS; PLASMA-LEVELS;
D O I
10.1097/MD.0000000000000007
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Multifocal or multiple osteonecrosis (ON), defined by the involvement of 3 or more anatomic sites, is unusual, being observed in only 3%-10% of patients diagnosed with ON. We report the clinical characteristics of a cohort of 29 patients with multifocal ON from a single center and evaluate the prevalence of associated prothrombotic abnormalities in 26 of these patients. We conducted a retrospective study of all patients diagnosed with multifocal ON evaluated in our institution during the last 20 years. We recorded clinical manifestations and underlying diagnoses. A wide thrombophilic profile was performed, including antithrombin, protein C, protein S, lupus anticoagulant, anticardiolipin antibodies, activated protein C resistance, factor V Leiden, mutation G-20210-A of the prothrombin gene, and factor VIII. Coagulation test results were compared with those in a healthy control group and a group of patients with history of lower-extremity deep venous thrombosis. The mean age of the patients was 49.2 +/- 15 years (range, 28-81 yr). The mean number of ON localizations per patient was 5.2 +/- 2.3 (range, 3-11). Hips were the most commonly affected joint (82%), followed by knees (58%), shoulders (37%), and ankles (13%). Most patients had an underlying disease process, and 12 of 25 (48%) patients had coagulation test abnormalities. The most common alterations were high factor VIII levels and antiphospholipid antibody (aPL) positivity in 24% and 20% of cases, respectively. These abnormalities were more prevalent in patients with multifocal ON compared with patients in the control groups. Sixty-one percent of patients had a history of corticosteroid treatment. Patients with coagulation abnormalities had a higher number of ON localizations per patient (6.5 +/- 2.7 vs. 3.88 +/- 0.8; p = 0.002) and a higher prevalence of atypical ON localizations (25% vs. 0%; p = 0.05). In conclusion, in the present cohort of patients with multifocal ON, 48% of the patients had at least 1 prothrombotic factor, especially high levels of factor VIII and aPL. These findings have major implications for the diagnosis and treatment of multifocal ON and clearly indicate the need to perform a thrombophilic profile in these patients.
引用
收藏
页码:295 / 304
页数:10
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