The authors report the case of a 39-year-old woman with leukemic meningitis. A right frontal Ommaya reservoir was placed for intrathecal chemotherapy. During and immediately following the first injection of chemotherapy, the patient developed an episode of nausea, emesis, frontal headache and diarrhea. These same symptoms were later elicited during a second and third administration of chemotherapy. Post-placement head computed tomography showed the tip of the catheter projecting approximately 1.5 cm inferior to the floor of the left frontal ventricle. After a revision of the Ommaya catheter due to suboptimal positioning, subsequent intrathecal chemotherapy administration was tolerated without any of the adverse symptoms previously encountered. The case documents an unusual complication arising from catheter migration in the setting of intrathecal chemotherapy and also demonstrates the value in troubleshooting Ommaya reservoir complications rather than prematurely abandoning its use in favor of lumbar puncture.Copyright © 2020, Spandidos Publications.