Purpose of StudyATL is a rare and aggressive peripheral T-cell neoplasm characterized by clonal human T-cell lymphotropic virus type-1 (HTLV-1) proviral DNA integration with host T lymphocytes.These patients commonly present with lymphadenopathy, skin rash, fever, fatigue or altered mental status. The prevalence of CNS disease varies from 3 to 50% and is always in the presence of systemic disease. Isolated cranial neuropathy as a presenting symptom has not been described in literature.Methods UsedRetrospective chart review and review of literature.Summary of Results49 year old Caribbean male presented with 2 month history of left sided headache, 5 weeks of right sided jaw numbness and pain which progressed to contralateral side. He was now unable to smile and had food falling from the side of his mouth. He denied fever, fatigue, night sweats, rash, weakness or abnormal lumps. He had normal mental status and good motor strength. Facial exam reveal bilateral upper and lower facial paralysis, left lateral rectus palsy and horizontal gaze diplopia. Rest of the physical exam was unremarkable. Labs revealed WBC of 6 k/mm3 with normal differential, HB 16.5 gm/dl and platelets 238 k/mm3. Complete metabolic profile and peripheral smear was normal. MRI Brain showed irregular, fusiform enhancement of left trigeminal nerve, bilateral facial and abducent nerves. CSF flow cytometry showed clonal CD4+ CD25+ T cell population. HTLV-1 serology was reactive. Left infraorbital nerve biopsy confirmed involvement with ATL. CT Chest/abdomen/pelvis did not reveal enlarged lymphadenopathy. He was started on treatment with EPOCH and twice weekly intrathecal methotrexate for 4 months with clearance of CNS fluid. His jaw pain and vision improved but facial nerve paralysis persisted. He developed local relapse four months after treatment and was treated with high dose methotrexate for 5 cycles. Ultimately his performance status deteriorated and he succumbed to the disease progression.ConclusionsThis case illustrates the unique presentation of this disease and gives an insight on one treatment approach. This patient achieved remission with our approach of aggressive chemotherapy with intrathecal methotrexate although the duration of remission was short lived.
Purpose of StudyThrombocytopenia during chemotherapy is not always due to myelosuppression. We report an unusual case of isolated acute thrombocytopenia after oxaliplatin and irinotecan administration. We reviewed 11 reported cases to better understand the nature of the presentation and variability in response to treatment.Case ReportPatient is a 63 year old female with metastatic colon cancer treated with palliative chemotherapy with FOLFOX. Follwing her 14th cycle she had an episode of acute drop in platelet count to 8,000/microliter. Peripheral smear revealed no evidence of thrombotic microangiopathy. She was managed with supportive platelet transfusions with slow recovery of platelet count. Subsequently she was treated with second line chemotherapy with FOLFIRI. Following the first cycle of Irinotecan, she again had a catastrophic drop in platelets from 136,000/microliter to 6,000/microliter within 10 hours. Due to this recurrent episode, a drug mediated thrombocytopenia was suspected and work up was initiated. She was initially treated with dexamethasone without a significant response. Platelet count normalized after 7 days with supportive platelet transfusions.Methods UsedBlood was tested for drug dependent platelet antibodies by Flow Cytometry at the Platelet and Neutrophil Immunology Laboratory at the Blood Center of Wisconsin.Summary of ResultsThe patient's serum showed evidence of drug dependent platelet antibodies to both oxaliplatin and irinotecan.ConclusionsDrug mediated immune thrombocytopenia is not uncommon. Time to severe acute thrombocytopenia and platelet recovery time varied post exposure of the drug. It is unclear whether steroid or IVIG administration had any effect on the platelet recovery time. Recovery from thrombocytopenia was observed in all 11 cases after the discontinuation of the insulting agent. Confirmation of the presence of drug dependent platelet antibodies against the chemotherapeutic agent by flow cytometry essential for diagnosis. This would be the first reported case of acute thrombocytopenia to two different chemotherapeutic agents in the same patient. Whether the reaction is two different mechanisms or if there is a cross reactivity between Oxaliplatin and Irinotecan has yet to be investigated.
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