Cerebrospinal fluid oligoclonal bands in childhood opsoclonus-myoclonus

Michael R. Pranzatelli; Patricia R. Slev; Elizabeth D. Tate; Anna L. Travelstead; Jerry A. Colliver; Suja Anne Joseph

doi:10.1016/j.pediatrneurol.2011.02.012

Cerebrospinal fluid oligoclonal bands in childhood opsoclonus-myoclonus

Michael R. Pranzatelli^*, Patricia R. Slev, Elizabeth D. Tate, Anna L. Travelstead, Jerry A. Colliver, Suja Anne Joseph

^*Corresponding author for this work

Pediatrics

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

24 Scopus citations

Abstract

Oligoclonal bands in cerebrospinal fluid reflect local B-cell responses associated with various neuroinflammatory disorders. In opsoclonus-myoclonus syndrome, cerebrospinal fluid B-cell expansion was demonstrated, but no studies of oligoclonal bands are available. In a prospective case-control study of 132 children (103 with opsoclonus-myoclonus, 29 neurologic control subjects), cerebrospinal fluid oligoclonal bands, measured by isoelectric focusing with immunofixation, were observed in 35% with opsoclonus-myoclonus and none of the control subjects, with the highest frequency in severe cases (56%). In oligoclonal band-positive patients, the mean band number was 5 ± 3 S.D. (range, 2-10) and the total severity score was significantly higher than in band-negative patients, whereas the frequency of CD19⁺ B cells, opsoclonus-myoclonus duration, neuroblastoma detection, and relapse history did not differ. The cerebrospinal fluid immunoglobulin G synthesis rate, immunoglobulin index, and Q albumin were normal. In 17 untreated children receiving adrenocorticotropic hormone, intravenous immunoglobulins, and rituximab, the number of oligoclonal band-positive decreased by 75%, and the mean band count fell by 80%. Oligoclonal band detection adds useful information to neuroimmunologic "staging" in opsoclonus-myoclonus. However, flow cytometry provides a more sensitive measure of B-cell infiltration. Cerebrospinal fluid oligoclonal bands warrant monitoring in long-term follow-up studies of disease-modifying drugs for opsoclonus-myoclonus.

Original language	English (US)
Pages (from-to)	27-33
Number of pages	7
Journal	Pediatric neurology
Volume	45
Issue number	1
DOIs	https://doi.org/10.1016/j.pediatrneurol.2011.02.012
State	Published - Jul 2011

Funding

M.R.P. holds research grants from Genentech, Inc. (South San Francisco, CA), Biogen IDEC (San Diego, CA), Questcor Pharamaceuticals, Inc. (Union City, CA), the Thrasher Research Fund (Salt Lake City, UT), the Chicago Institute of Neurosurgery and Neuroresearch (Chicago, IL), the Spastic Research Foundation, the Illinois-Eastern Iowa Division of Kiwanis International (DeKalb, IL), and Ronald McDonald House Charities of Central Illinois (Springfield, IL). The authors thank Janalee Renshaw (Technical Supervisor, Electrophoresis/Manual Endocrinology, ARUP Laboratories), Gregory Phillips, BS, MT (ASCP) (Technical Supervisor, Protein Immunology, ARUP Laboratories), Edward Ashwood, MD (ARUP Laboratories), Steven J. Verhulst, PhD (Southern Illinois University Statistics and Research Consulting), former research staff member Kristal J. Adams, BA, and Jennifer A. Swan, BS, Tammy A. Boyd (Office Support Specialist I), Miracle Flights for Kids, and each participating child and family. The authors also thank the collaborating physicians Alma Bicknese, MD (Department of Pediatrics, University of Illinois at Chicago, Chicago, IL), Abigail E. Collins, MD, and Tonia M. Sabo, MD (Department of Neurology, Children’s Hospital, Denver, CO), Glen A. Fenton, MD (Department of Neurology, University of New Mexico, Albuquerque, NM), William J. Logan, MD (Department of Pediatrics, Hospital for Sick Children, Toronto, Ontario, Canada), Subhash H. Shah, MD (Neurology Center of Wichita, Wichita, KS), and Marcos J. Valdez, MD, PA (McAllen, TX).

ASJC Scopus subject areas

Clinical Neurology
Neurology
Developmental Neuroscience
Pediatrics, Perinatology, and Child Health

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

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10.1016/j.pediatrneurol.2011.02.012

Cite this

@article{9c2972ed2b49431b91b54d393391eaf1,

title = "Cerebrospinal fluid oligoclonal bands in childhood opsoclonus-myoclonus",

abstract = "Oligoclonal bands in cerebrospinal fluid reflect local B-cell responses associated with various neuroinflammatory disorders. In opsoclonus-myoclonus syndrome, cerebrospinal fluid B-cell expansion was demonstrated, but no studies of oligoclonal bands are available. In a prospective case-control study of 132 children (103 with opsoclonus-myoclonus, 29 neurologic control subjects), cerebrospinal fluid oligoclonal bands, measured by isoelectric focusing with immunofixation, were observed in 35\% with opsoclonus-myoclonus and none of the control subjects, with the highest frequency in severe cases (56\%). In oligoclonal band-positive patients, the mean band number was 5 ± 3 S.D. (range, 2-10) and the total severity score was significantly higher than in band-negative patients, whereas the frequency of CD19+ B cells, opsoclonus-myoclonus duration, neuroblastoma detection, and relapse history did not differ. The cerebrospinal fluid immunoglobulin G synthesis rate, immunoglobulin index, and Q albumin were normal. In 17 untreated children receiving adrenocorticotropic hormone, intravenous immunoglobulins, and rituximab, the number of oligoclonal band-positive decreased by 75\%, and the mean band count fell by 80\%. Oligoclonal band detection adds useful information to neuroimmunologic {"}staging{"} in opsoclonus-myoclonus. However, flow cytometry provides a more sensitive measure of B-cell infiltration. Cerebrospinal fluid oligoclonal bands warrant monitoring in long-term follow-up studies of disease-modifying drugs for opsoclonus-myoclonus.",

author = "Pranzatelli, \{Michael R.\} and Slev, \{Patricia R.\} and Tate, \{Elizabeth D.\} and Travelstead, \{Anna L.\} and Colliver, \{Jerry A.\} and Joseph, \{Suja Anne\}",

note = "Funding Information: M.R.P. holds research grants from Genentech, Inc. (South San Francisco, CA), Biogen IDEC (San Diego, CA), Questcor Pharamaceuticals, Inc. (Union City, CA), the Thrasher Research Fund (Salt Lake City, UT), the Chicago Institute of Neurosurgery and Neuroresearch (Chicago, IL), the Spastic Research Foundation, the Illinois-Eastern Iowa Division of Kiwanis International (DeKalb, IL), and Ronald McDonald House Charities of Central Illinois (Springfield, IL). The authors thank Janalee Renshaw (Technical Supervisor, Electrophoresis/Manual Endocrinology, ARUP Laboratories), Gregory Phillips, BS, MT (ASCP) (Technical Supervisor, Protein Immunology, ARUP Laboratories), Edward Ashwood, MD (ARUP Laboratories), Steven J. Verhulst, PhD (Southern Illinois University Statistics and Research Consulting), former research staff member Kristal J. Adams, BA, and Jennifer A. Swan, BS, Tammy A. Boyd (Office Support Specialist I), Miracle Flights for Kids, and each participating child and family. The authors also thank the collaborating physicians Alma Bicknese, MD (Department of Pediatrics, University of Illinois at Chicago, Chicago, IL), Abigail E. Collins, MD, and Tonia M. Sabo, MD (Department of Neurology, Children{\textquoteright}s Hospital, Denver, CO), Glen A. Fenton, MD (Department of Neurology, University of New Mexico, Albuquerque, NM), William J. Logan, MD (Department of Pediatrics, Hospital for Sick Children, Toronto, Ontario, Canada), Subhash H. Shah, MD (Neurology Center of Wichita, Wichita, KS), and Marcos J. Valdez, MD, PA (McAllen, TX). ",

year = "2011",

month = jul,

doi = "10.1016/j.pediatrneurol.2011.02.012",

language = "English (US)",

volume = "45",

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AU - Slev, Patricia R.

AU - Tate, Elizabeth D.

AU - Travelstead, Anna L.

AU - Colliver, Jerry A.

AU - Joseph, Suja Anne

N1 - Funding Information: M.R.P. holds research grants from Genentech, Inc. (South San Francisco, CA), Biogen IDEC (San Diego, CA), Questcor Pharamaceuticals, Inc. (Union City, CA), the Thrasher Research Fund (Salt Lake City, UT), the Chicago Institute of Neurosurgery and Neuroresearch (Chicago, IL), the Spastic Research Foundation, the Illinois-Eastern Iowa Division of Kiwanis International (DeKalb, IL), and Ronald McDonald House Charities of Central Illinois (Springfield, IL). The authors thank Janalee Renshaw (Technical Supervisor, Electrophoresis/Manual Endocrinology, ARUP Laboratories), Gregory Phillips, BS, MT (ASCP) (Technical Supervisor, Protein Immunology, ARUP Laboratories), Edward Ashwood, MD (ARUP Laboratories), Steven J. Verhulst, PhD (Southern Illinois University Statistics and Research Consulting), former research staff member Kristal J. Adams, BA, and Jennifer A. Swan, BS, Tammy A. Boyd (Office Support Specialist I), Miracle Flights for Kids, and each participating child and family. The authors also thank the collaborating physicians Alma Bicknese, MD (Department of Pediatrics, University of Illinois at Chicago, Chicago, IL), Abigail E. Collins, MD, and Tonia M. Sabo, MD (Department of Neurology, Children’s Hospital, Denver, CO), Glen A. Fenton, MD (Department of Neurology, University of New Mexico, Albuquerque, NM), William J. Logan, MD (Department of Pediatrics, Hospital for Sick Children, Toronto, Ontario, Canada), Subhash H. Shah, MD (Neurology Center of Wichita, Wichita, KS), and Marcos J. Valdez, MD, PA (McAllen, TX).

PY - 2011/7

Y1 - 2011/7

N2 - Oligoclonal bands in cerebrospinal fluid reflect local B-cell responses associated with various neuroinflammatory disorders. In opsoclonus-myoclonus syndrome, cerebrospinal fluid B-cell expansion was demonstrated, but no studies of oligoclonal bands are available. In a prospective case-control study of 132 children (103 with opsoclonus-myoclonus, 29 neurologic control subjects), cerebrospinal fluid oligoclonal bands, measured by isoelectric focusing with immunofixation, were observed in 35% with opsoclonus-myoclonus and none of the control subjects, with the highest frequency in severe cases (56%). In oligoclonal band-positive patients, the mean band number was 5 ± 3 S.D. (range, 2-10) and the total severity score was significantly higher than in band-negative patients, whereas the frequency of CD19+ B cells, opsoclonus-myoclonus duration, neuroblastoma detection, and relapse history did not differ. The cerebrospinal fluid immunoglobulin G synthesis rate, immunoglobulin index, and Q albumin were normal. In 17 untreated children receiving adrenocorticotropic hormone, intravenous immunoglobulins, and rituximab, the number of oligoclonal band-positive decreased by 75%, and the mean band count fell by 80%. Oligoclonal band detection adds useful information to neuroimmunologic "staging" in opsoclonus-myoclonus. However, flow cytometry provides a more sensitive measure of B-cell infiltration. Cerebrospinal fluid oligoclonal bands warrant monitoring in long-term follow-up studies of disease-modifying drugs for opsoclonus-myoclonus.

AB - Oligoclonal bands in cerebrospinal fluid reflect local B-cell responses associated with various neuroinflammatory disorders. In opsoclonus-myoclonus syndrome, cerebrospinal fluid B-cell expansion was demonstrated, but no studies of oligoclonal bands are available. In a prospective case-control study of 132 children (103 with opsoclonus-myoclonus, 29 neurologic control subjects), cerebrospinal fluid oligoclonal bands, measured by isoelectric focusing with immunofixation, were observed in 35% with opsoclonus-myoclonus and none of the control subjects, with the highest frequency in severe cases (56%). In oligoclonal band-positive patients, the mean band number was 5 ± 3 S.D. (range, 2-10) and the total severity score was significantly higher than in band-negative patients, whereas the frequency of CD19+ B cells, opsoclonus-myoclonus duration, neuroblastoma detection, and relapse history did not differ. The cerebrospinal fluid immunoglobulin G synthesis rate, immunoglobulin index, and Q albumin were normal. In 17 untreated children receiving adrenocorticotropic hormone, intravenous immunoglobulins, and rituximab, the number of oligoclonal band-positive decreased by 75%, and the mean band count fell by 80%. Oligoclonal band detection adds useful information to neuroimmunologic "staging" in opsoclonus-myoclonus. However, flow cytometry provides a more sensitive measure of B-cell infiltration. Cerebrospinal fluid oligoclonal bands warrant monitoring in long-term follow-up studies of disease-modifying drugs for opsoclonus-myoclonus.

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Cerebrospinal fluid oligoclonal bands in childhood opsoclonus-myoclonus

Abstract

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ASJC Scopus subject areas

UN SDGs

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