Introduction: Ankylosing spondylitis (AS) and autoimmune thrombocytopenia are two distinct diseases, and they can occur rarely. Objective: To determine the epidemiological, clinical, therapeutic profiles and impact of this rare association in the literature Observation: This was a 57-year-old teaching patient received on September 07, 2020. The examination of the musculoskeletal system revealed lumbar pain. The onset was 26 years ago and was marked by the progressive onset of pain located in the dorsal spine, lumbar and manubrio-sternal region, then a progressive extension to both wrists and the left knee with a VAS of 6/10. Associated signs were stiffness and synovitis of the left knee. The biology showed a non-specific biological inflammatory syndrome with accelerated blood pressure at 70 minutes in the first hour and CRP increased to 15 mg/l, normocytic normochromic anemia (hemoglobin 7.6 g/dl), thrombocytopenia at 51,000/mm3 (N : leukopenia at 3,100/mm3 and neutropenia at 1115/mm3 (pancytopenia). The transaminases ALAT was 60 IU/L and Gamma GT was increased to 265 IU/L, the immunological tests were negative with the presence of the HLA B27 positive antigen, and the fixed anti-platelet antibodies showed the presence of fixed autoantibodies directed against the glycoprotein complex IIb and IIIa and the absence of circulating antibodies directed against the glycoprotein complexes IaIIb, IIbIIIa and IbIX. The lumbar CT scan shows syndesmophytes and osteophytes (Fig 2) and the CT scan of the pelvis shows Forestier stage III sacroiliitis (Fig 1). Conclusion: The association of ankylosing spondylitis and autoimmune thrombocytopenia is rare but possible, and should be considered whenever a known spondyloarthritis patient presents with a platelet collapse. Treatment with methotrexate and anti-inflammatory drugs may be effective in some cases.