Carpal tunnel syndrome occurring as a complication of hemophilia is documented in the literature. Most reports, however, indicate that the condition can be relieved by splinting and replacement therapy. Two cases of carpal tunnel syndrome in hemophiliacs are presented. In one patient, replacement therapy was successful in relieving the condition. However, in the other patient, decompression of the carpal tunnel and internal neurolysis of the median nerve were carried out after replacement therapy failed. To our knowledge, this is the first time that intraneural bleeding in the nerve has been documented as a cause of peripheral neuropathy in hemophilia. Surgical release of the carpal canal together with the epineurectomy and internal neurolysis resulted in complete recovery.