Osteonecrosis of both shoulders and hips is a rare presentation of primary antiphospholipid syndrome. A female patient aged 23 years old with no systemic diseases has her only complaint which was pain and limitations in both hips followed by both shoulders. Careful detailed history and clinical examination is essential for reaching optimum diagnosis thus good management. MRI for hip and shoulder joints is essential for the diagnosis of osteonecrosis. Exclusion of all causes of secondary osteonecrosis by history, clinical examination, and laboratory studies should be done before diagnosing the rare causes of osteonecrosis or the primary type. Pregnancy is an exacerbation factor for primary osteonecrosis. We recommend that the female patient with primary osteonecrosis should receive prophylactic antithrombotic during pregnancy. In conclusion, exclusion of all secondary causes of osteonecrosis is mandatory to reach an accurate diagnosis.
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