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High prevalence of prothrombotic abnormalities in multifocal osteonecrosis: description of a series and review of the literature.

Gómez-Puerta JA, Peris P, Reverter JC, Espinosa G, Martinez-Ferrer A, Monegal A, Monteagudo J, Tàssies D, Guañabens N - Medicine (Baltimore) (2013)

Bottom Line: Sixty-one percent of patients had a history of corticosteroid treatment.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.

View Article: PubMed Central - PubMed

Affiliation: From the Department of Rheumatology (JAG-P, PP, AM-F, AM, NG), CIBERehd; and Hemotherapy and Haemostasis Service (JCR, JM, DT), Hospital Clínic, Barcelona; Department of Autoimmune Diseases (GE), Hospital Clínic, University of Barcelona, Barcelona, Spain; and Division of Rheumatology, Immunology and Allergy (JAG-P), Brigham and Women's Hospital, Boston, Massachusetts, United States.

ABSTRACT
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.

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Related in: MedlinePlus

Bilateral knee X-rays showing extensive bilateral bone infarcts in the distal femur and proximal tibia. (“D” indicates the right side.)
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Figure 1: Bilateral knee X-rays showing extensive bilateral bone infarcts in the distal femur and proximal tibia. (“D” indicates the right side.)

Mentions: Hips were the joint most commonly affected (82%; bilateral in 72.4% of cases), followed by the knees (58%; bilateral in 48%; Figure 1), shoulders (37%; bilateral in 24%), and ankles (13%; bilateral in 3.4%; Figure 2). Other common locations affected were the talus (17%; bilateral in 7%; Figure 3), and the calcaneus (10%). Vertebral involvement was uncommon, observed in only 1 case.


High prevalence of prothrombotic abnormalities in multifocal osteonecrosis: description of a series and review of the literature.

Gómez-Puerta JA, Peris P, Reverter JC, Espinosa G, Martinez-Ferrer A, Monegal A, Monteagudo J, Tàssies D, Guañabens N - Medicine (Baltimore) (2013)

Bilateral knee X-rays showing extensive bilateral bone infarcts in the distal femur and proximal tibia. (“D” indicates the right side.)
© Copyright Policy
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC4553995&req=5

Figure 1: Bilateral knee X-rays showing extensive bilateral bone infarcts in the distal femur and proximal tibia. (“D” indicates the right side.)
Mentions: Hips were the joint most commonly affected (82%; bilateral in 72.4% of cases), followed by the knees (58%; bilateral in 48%; Figure 1), shoulders (37%; bilateral in 24%), and ankles (13%; bilateral in 3.4%; Figure 2). Other common locations affected were the talus (17%; bilateral in 7%; Figure 3), and the calcaneus (10%). Vertebral involvement was uncommon, observed in only 1 case.

Bottom Line: Sixty-one percent of patients had a history of corticosteroid treatment.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.

View Article: PubMed Central - PubMed

Affiliation: From the Department of Rheumatology (JAG-P, PP, AM-F, AM, NG), CIBERehd; and Hemotherapy and Haemostasis Service (JCR, JM, DT), Hospital Clínic, Barcelona; Department of Autoimmune Diseases (GE), Hospital Clínic, University of Barcelona, Barcelona, Spain; and Division of Rheumatology, Immunology and Allergy (JAG-P), Brigham and Women's Hospital, Boston, Massachusetts, United States.

ABSTRACT
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.

Show MeSH
Related in: MedlinePlus