Kelch-like Protein 11 antibodies in seminoma-Associated paraneoplastic encephalitis

Caleigh Mandel-brehm; Divyanshu Dubey; Thomas J. Kryzer; Brian D. O'donovan; Baouyen Tran; Sara E. Vazquez; Hannah A. Sample; Kelsey C. Zorn; Lillian M. Khan; Ian O. Bledsoe; Andrew Mckeo; Samuel J. Pleasure; Vanda A Lennon; Joseph L. Derisi; Michael R. Wilson; Sean J Pittock

doi:10.1056/NEJMoa1816721

Kelch-like Protein 11 antibodies in seminoma-Associated paraneoplastic encephalitis

Caleigh Mandel-brehm, Divyanshu Dubey, Thomas J. Kryzer, Brian D. O'donovan, Baouyen Tran, Sara E. Vazquez, Hannah A. Sample, Kelsey C. Zorn, Lillian M. Khan, Ian O. Bledsoe, Andrew Mckeo, Samuel J. Pleasure, Vanda A Lennon, Joseph L. Derisi, Michael R. Wilson, Sean J Pittock

Research output: Contribution to journal › Article

9 Scopus citations

Abstract

A 37-year-old man with a history of seminoma presented with vertigo, ataxia, and diplopia. An autoantibody specific for kelch-like protein 11 (KLHL11) was identified with the use of programmable phage display. Immunoassays were used to identify KLHL11 IgG in 12 other men with similar neurologic features and testicular disease. Immunostaining of the patient's IgG on mouse brain tissue showed sparse but distinctive points of staining in multiple brain regions, with enrichment in perivascular and perimeningeal tissues. The onset of the neurologic syndrome preceded the diagnosis of seminoma in 9 of the 13 patients. An age-adjusted estimate of the prevalence of autoimmune KLHL11 encephalitis in Olmsted County, Minnesota, was 2.79 cases per 100,000 men.

Original language	English (US)
Pages (from-to)	47-54
Number of pages	8
Journal	New England Journal of Medicine
Volume	381
Issue number	1
DOIs	https://doi.org/10.1056/NEJMoa1816721
State	Published - Jul 4 2019

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Medicine(all)

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Mandel-brehm, C., Dubey, D., Kryzer, T. J., O'donovan, B. D., Tran, B., Vazquez, S. E., Sample, H. A., Zorn, K. C., Khan, L. M., Bledsoe, I. O., Mckeo, A., Pleasure, S. J., Lennon, V. A., Derisi, J. L., Wilson, M. R., & Pittock, S. J. (2019). Kelch-like Protein 11 antibodies in seminoma-Associated paraneoplastic encephalitis. New England Journal of Medicine, 381(1), 47-54. https://doi.org/10.1056/NEJMoa1816721

Kelch-like Protein 11 antibodies in seminoma-Associated paraneoplastic encephalitis. / Mandel-brehm, Caleigh; Dubey, Divyanshu; Kryzer, Thomas J.; O'donovan, Brian D.; Tran, Baouyen; Vazquez, Sara E.; Sample, Hannah A.; Zorn, Kelsey C.; Khan, Lillian M.; Bledsoe, Ian O.; Mckeo, Andrew; Pleasure, Samuel J.; Lennon, Vanda A; Derisi, Joseph L.; Wilson, Michael R.; Pittock, Sean J.

In: New England Journal of Medicine, Vol. 381, No. 1, 04.07.2019, p. 47-54.

Research output: Contribution to journal › Article

Mandel-brehm, C, Dubey, D, Kryzer, TJ, O'donovan, BD, Tran, B, Vazquez, SE, Sample, HA, Zorn, KC, Khan, LM, Bledsoe, IO, Mckeo, A, Pleasure, SJ, Lennon, VA, Derisi, JL, Wilson, MR & Pittock, SJ 2019, 'Kelch-like Protein 11 antibodies in seminoma-Associated paraneoplastic encephalitis', New England Journal of Medicine, vol. 381, no. 1, pp. 47-54. https://doi.org/10.1056/NEJMoa1816721

Mandel-brehm, Caleigh ; Dubey, Divyanshu ; Kryzer, Thomas J. ; O'donovan, Brian D. ; Tran, Baouyen ; Vazquez, Sara E. ; Sample, Hannah A. ; Zorn, Kelsey C. ; Khan, Lillian M. ; Bledsoe, Ian O. ; Mckeo, Andrew ; Pleasure, Samuel J. ; Lennon, Vanda A ; Derisi, Joseph L. ; Wilson, Michael R. ; Pittock, Sean J. / Kelch-like Protein 11 antibodies in seminoma-Associated paraneoplastic encephalitis. In: New England Journal of Medicine. 2019 ; Vol. 381, No. 1. pp. 47-54.

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abstract = "A 37-year-old man with a history of seminoma presented with vertigo, ataxia, and diplopia. An autoantibody specific for kelch-like protein 11 (KLHL11) was identified with the use of programmable phage display. Immunoassays were used to identify KLHL11 IgG in 12 other men with similar neurologic features and testicular disease. Immunostaining of the patient's IgG on mouse brain tissue showed sparse but distinctive points of staining in multiple brain regions, with enrichment in perivascular and perimeningeal tissues. The onset of the neurologic syndrome preceded the diagnosis of seminoma in 9 of the 13 patients. An age-adjusted estimate of the prevalence of autoimmune KLHL11 encephalitis in Olmsted County, Minnesota, was 2.79 cases per 100,000 men.",

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AU - Tran, Baouyen

AU - Vazquez, Sara E.

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AU - Zorn, Kelsey C.

AU - Khan, Lillian M.

AU - Bledsoe, Ian O.

AU - Mckeo, Andrew

AU - Pleasure, Samuel J.

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AB - A 37-year-old man with a history of seminoma presented with vertigo, ataxia, and diplopia. An autoantibody specific for kelch-like protein 11 (KLHL11) was identified with the use of programmable phage display. Immunoassays were used to identify KLHL11 IgG in 12 other men with similar neurologic features and testicular disease. Immunostaining of the patient's IgG on mouse brain tissue showed sparse but distinctive points of staining in multiple brain regions, with enrichment in perivascular and perimeningeal tissues. The onset of the neurologic syndrome preceded the diagnosis of seminoma in 9 of the 13 patients. An age-adjusted estimate of the prevalence of autoimmune KLHL11 encephalitis in Olmsted County, Minnesota, was 2.79 cases per 100,000 men.

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10.1056/NEJMoa1816721