The first clinical case of carpal tunnel syndrome (CTS) in Cuban HIV-infected patient was described, and the scientific literature indexed in: PUBMED/MEDLINE, LILACS and BIREME were revised. The case presented was a male with HIV infection without preceding opportunistic illnesses, CD4+ T cell count over 200 cells/mm3 and clinical symptoms of pain, tingling and numbness in the right hand and wrist for three months. The electrophysiological study was compatible with CTS. The pharmacological treatment did not modify the symptoms and the patient received specific surgical treatment with absolute resolution of symptoms. CTS is a compressive neuropathy that can occur in HIV-positive individuals with as similar frequency as in the general population. The association between HIV infection and CTS is scarcely described in the medical scientific literature and probably does not represent a different phenomenon from what happens in the HIV-negative population. Nevertheless, its clinical recognition among other neurological and muscle-skeletal manifestations in HIV-infected patients is important.