Early diagnosis and successful treatment of paraneoplastic melanocytic proliferation

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

Research output: Contribution to journalArticle

13 Citations (Scopus)

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 languageEnglish (US)
Pages (from-to)943-948
Number of pages6
JournalBritish Journal of Ophthalmology
Volume99
Issue number7
DOIs
StatePublished - Jul 1 2015

Fingerprint

Ocular Paraneoplastic Syndromes
Plasmapheresis
Melanocytes
Early Diagnosis
Peptide Elongation Factors
Serum
Therapeutics
Eye Diseases
Rare Diseases
Disease Progression
Radiation
Drug Therapy

ASJC Scopus subject areas

  • Ophthalmology
  • Sensory Systems
  • Cellular and Molecular Neuroscience

Cite this

Early diagnosis and successful treatment of paraneoplastic melanocytic proliferation. / Jansen, Joyce C G; Van Calster, Joachim; Pulido, Jose S; Miles, Sarah L.; Vile, Richard Geoffrey; Van Bergen, Tine; Cassiman, Catherine; Spielberg, Leigh H.; Leys, Anita M.

In: British Journal of Ophthalmology, Vol. 99, No. 7, 01.07.2015, p. 943-948.

Research output: Contribution to journalArticle

Jansen, JCG, Van Calster, J, Pulido, JS, Miles, SL, Vile, RG, Van Bergen, T, Cassiman, C, Spielberg, LH & Leys, AM 2015, 'Early diagnosis and successful treatment of paraneoplastic melanocytic proliferation', British Journal of Ophthalmology, vol. 99, no. 7, pp. 943-948. https://doi.org/10.1136/bjophthalmol-2014-305893
Jansen, Joyce C G ; Van Calster, Joachim ; Pulido, Jose S ; Miles, Sarah L. ; Vile, Richard Geoffrey ; Van Bergen, Tine ; Cassiman, Catherine ; Spielberg, Leigh H. ; Leys, Anita M. / Early diagnosis and successful treatment of paraneoplastic melanocytic proliferation. In: British Journal of Ophthalmology. 2015 ; Vol. 99, No. 7. pp. 943-948.
@article{2edcfa3cae9c4d35a2457072042da0e5,
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.",
author = "Jansen, {Joyce C G} and {Van Calster}, Joachim and Pulido, {Jose S} and Miles, {Sarah L.} and Vile, {Richard Geoffrey} and {Van Bergen}, Tine and Catherine Cassiman and Spielberg, {Leigh H.} and Leys, {Anita M.}",
year = "2015",
month = "7",
day = "1",
doi = "10.1136/bjophthalmol-2014-305893",
language = "English (US)",
volume = "99",
pages = "943--948",
journal = "British Journal of Ophthalmology",
issn = "0007-1161",
publisher = "BMJ Publishing Group",
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 Geoffrey

AU - Van Bergen, Tine

AU - Cassiman, Catherine

AU - Spielberg, Leigh H.

AU - Leys, Anita M.

PY - 2015/7/1

Y1 - 2015/7/1

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.

UR - http://www.scopus.com/inward/record.url?scp=84937694708&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=84937694708&partnerID=8YFLogxK

U2 - 10.1136/bjophthalmol-2014-305893

DO - 10.1136/bjophthalmol-2014-305893

M3 - Article

C2 - 25908835

AN - SCOPUS:84937694708

VL - 99

SP - 943

EP - 948

JO - British Journal of Ophthalmology

JF - British Journal of Ophthalmology

SN - 0007-1161

IS - 7

ER -