Early diagnosis and successful treatment of paraneoplastic melanocytic proliferation

Joyce C G Jansen; Joachim Van Calster; Jose S Pulido; Sarah L Miles; Richard G Vile; Tine Van Bergen; Catherine Cassiman; Leigh H Spielberg; Anita M Leys

doi:10.1136/bjophthalmol-2014-305893

Early diagnosis and successful treatment of paraneoplastic melanocytic proliferation

Joyce C G Jansen, Joachim Van Calster, Jose S Pulido, Sarah L Miles, Richard G Vile, Tine Van Bergen, Catherine Cassiman, Leigh H Spielberg, Anita M Leys

Research output: Contribution to journal › Article › Research › peer-review

Abstract

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.

Original language	English
Pages (from-to)	943-8
Number of pages	6
Journal	British Journal of Ophthalmology
Volume	99
Issue number	7
DOIs	https://doi.org/10.1136/bjophthalmol-2014-305893
Publication status	Published - Jul 2015

Keywords

Adenocarcinoma/secondary
Aged
Angiogenesis Inhibitors/therapeutic use
Antibodies, Monoclonal, Humanized/therapeutic use
Antineoplastic Agents/therapeutic use
Bevacizumab
Cell Proliferation/drug effects
Combined Modality Therapy
Early Diagnosis
Fluorescein Angiography
Humans
Intravitreal Injections
Lung Neoplasms/pathology
Lymphatic Metastasis
Male
Melanocytes/pathology
Paraneoplastic Syndromes, Ocular/diagnosis
Plasmapheresis
Subretinal Fluid
Tomography, Optical Coherence
Vascular Endothelial Growth Factor A/antagonists & inhibitors
Visual Acuity/physiology
Visual Field Tests
Visual Fields

Access to Document

10.1136/bjophthalmol-2014-305893

Cite this

@article{63be0a91b2b3440aa417f446f42b73c1,

title = "Early diagnosis and successful treatment of paraneoplastic melanocytic proliferation",

abstract = "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.",

keywords = "Adenocarcinoma/secondary, Aged, Angiogenesis Inhibitors/therapeutic use, Antibodies, Monoclonal, Humanized/therapeutic use, Antineoplastic Agents/therapeutic use, Bevacizumab, Cell Proliferation/drug effects, Combined Modality Therapy, Early Diagnosis, Fluorescein Angiography, Humans, Intravitreal Injections, Lung Neoplasms/pathology, Lymphatic Metastasis, Male, Melanocytes/pathology, Paraneoplastic Syndromes, Ocular/diagnosis, Plasmapheresis, Subretinal Fluid, Tomography, Optical Coherence, Vascular Endothelial Growth Factor A/antagonists & inhibitors, Visual Acuity/physiology, Visual Field Tests, Visual Fields",

author = "Jansen, {Joyce C G} and {Van Calster}, Joachim and Pulido, {Jose S} and Miles, {Sarah L} and Vile, {Richard G} and {Van Bergen}, Tine and Catherine Cassiman and Spielberg, {Leigh H} and Leys, {Anita M}",

note = "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.",

year = "2015",

month = jul,

doi = "10.1136/bjophthalmol-2014-305893",

language = "English",

volume = "99",

pages = "943--8",

number = "7",

}

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 -

Early diagnosis and successful treatment of paraneoplastic melanocytic proliferation

Abstract

Keywords

Access to Document

Fingerprint

Cite this