TY - JOUR
T1 - Early diagnosis and successful treatment of paraneoplastic melanocytic proliferation
AU - Jansen, Joyce C G
AU - Van Calster, Joachim
AU - Pulido, Jose S
AU - Miles, Sarah L
AU - Vile, Richard G
AU - Van Bergen, Tine
AU - Cassiman, Catherine
AU - Spielberg, Leigh H
AU - Leys, Anita M
N1 - Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://group.bmj.com/group/rights-licensing/permissions.
PY - 2015/7
Y1 - 2015/7
N2 - BACKGROUND: Paraneoplastic melanocytic proliferation (bilateral diffuse uveal melanocytic proliferation, BDUMP) is a rare but devastating disease that causes progressive visual loss in patients who usually have an occult malignancy. Visual loss occurs as a result of paraneoplastic changes in the uveal tissue.METHODS: In a masked fashion, the serum of two patients with BDUMP was evaluated for the presence of cultured melanocyte elongation and proliferation (CMEP) factor using cultured human melanocytes. We evaluated the efficacy of plasmapheresis as a treatment modality early in the disease in conjunction with radiation and chemotherapy.RESULTS: The serum of the first case patient was investigated after plasmapheresis and did not demonstrate proliferation of cultured human melanocytes. The serum of the second case was evaluated prior to treatment with plasmapheresis and did induce this proliferation. These findings are in accordance with the diminution of CMEP factor after plasmapheresis. Treatment with plasmapheresis managed to stabilise the ocular disease progression in both patients.CONCLUSIONS: In the past, visual loss due to paraneoplastic melanocytic proliferation was considered progressive and irreversible. We treated two patients successfully with plasmapheresis and demonstrated a relation between CMEP factor in the serum of these patients and proliferation of cultured melanocytes.
AB - BACKGROUND: Paraneoplastic melanocytic proliferation (bilateral diffuse uveal melanocytic proliferation, BDUMP) is a rare but devastating disease that causes progressive visual loss in patients who usually have an occult malignancy. Visual loss occurs as a result of paraneoplastic changes in the uveal tissue.METHODS: In a masked fashion, the serum of two patients with BDUMP was evaluated for the presence of cultured melanocyte elongation and proliferation (CMEP) factor using cultured human melanocytes. We evaluated the efficacy of plasmapheresis as a treatment modality early in the disease in conjunction with radiation and chemotherapy.RESULTS: The serum of the first case patient was investigated after plasmapheresis and did not demonstrate proliferation of cultured human melanocytes. The serum of the second case was evaluated prior to treatment with plasmapheresis and did induce this proliferation. These findings are in accordance with the diminution of CMEP factor after plasmapheresis. Treatment with plasmapheresis managed to stabilise the ocular disease progression in both patients.CONCLUSIONS: In the past, visual loss due to paraneoplastic melanocytic proliferation was considered progressive and irreversible. We treated two patients successfully with plasmapheresis and demonstrated a relation between CMEP factor in the serum of these patients and proliferation of cultured melanocytes.
KW - Adenocarcinoma/secondary
KW - Aged
KW - Angiogenesis Inhibitors/therapeutic use
KW - Antibodies, Monoclonal, Humanized/therapeutic use
KW - Antineoplastic Agents/therapeutic use
KW - Bevacizumab
KW - Cell Proliferation/drug effects
KW - Combined Modality Therapy
KW - Early Diagnosis
KW - Fluorescein Angiography
KW - Humans
KW - Intravitreal Injections
KW - Lung Neoplasms/pathology
KW - Lymphatic Metastasis
KW - Male
KW - Melanocytes/pathology
KW - Paraneoplastic Syndromes, Ocular/diagnosis
KW - Plasmapheresis
KW - Subretinal Fluid
KW - Tomography, Optical Coherence
KW - Vascular Endothelial Growth Factor A/antagonists & inhibitors
KW - Visual Acuity/physiology
KW - Visual Field Tests
KW - Visual Fields
U2 - 10.1136/bjophthalmol-2014-305893
DO - 10.1136/bjophthalmol-2014-305893
M3 - Article
C2 - 25908835
SN - 0007-1161
VL - 99
SP - 943
EP - 948
JO - British Journal of Ophthalmology
JF - British Journal of Ophthalmology
IS - 7
ER -