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       #Post#: 548--------------------------------------------------
       Article about the KIR4.1 debate
   DIR By: Matt2
       Date: November 10, 2014, 3:34 pm
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       The Lancet Neurology, Volume 13, Issue 8, Pages 753 - 755,
       August 2014 <Previous Article|Next Article>
       doi:10.1016/S1474-4422(14)70149-8Cite or Link Using DOI
       This article can be found in the following collections:
       Neurology (Multiple sclerosis)
       Published Online: 07 July 2014
       Copyright © 2014 Elsevier Ltd All rights reserved.
       KIR4.1: another misleading expectation in multiple sclerosis?
       Massimo Filippi a bEmail Address, Maria A Rocca a b, Hans
       Lassmann c
       The identification of a biomarker for the diagnosis, prognosis,
       and monitoring of patients with multiple sclerosis is needed.
       Several biomarkers have been proposed (eg, immunological and
       clinical markers) but have had disappointing results.
       Pathological changes in the immunological response in patients
       with multiple sclerosis affect not only T cells, but also B
       cells, and increase immunoglobulin synthesis in the CSF,
       oligoclonal bands, and complement deposition; therefore, CNS
       antigens have been regarded as potential autoimmune targets.
       Such research has been fuelled by the discovery of a pathogenic
       antibody against the aquaporin-4 channel in neuromyelitis
       optica, which has revolutionised the diagnosis and treatment of
       this disorder.1
       A serum autoantibody specific for multiple sclerosis would
       profoundly affect the diagnostic work-up of people suspected of
       having this disease, the monitoring of disease evolution, and
       ultimately treatment. In 2012, KIR4.1 (also known as KCNJ10) was
       proposed as the target of IgG antibodies in a proportion of
       patients with multiple sclerosis.2 Using ELISA, serum antibodies
       against KIR4.1 were detected in 47% of 397 patients with
       multiple sclerosis, 1% of 329 patients with other neurological
       diseases, and none of 59 healthy donors.2 Only two of 19
       patients who were serum IgG positive had detectable
       concentrations of KIR4.1 antibodies in the CSF, which is not
       surprising considering the high expression of the target antigen
       on the surface of CSF cells, which might lead to absorption of
       antibodies from the extracellular space and CSF.3 Using the same
       method, KIR4.1 serum antibodies have been reported in roughly
       57% of paediatric patients with acquired demyelinating diseases
       and more than 50% of paediatric patients with multiple sclerosis
       or clinically isolated syndromes,4 independently of the presence
       of antibodies against myelin oligodendrocyte glycoproteins.
       According to these data, KIR4.1 would be an excellent candidate
       for a pathogenic antibody in multiple sclerosis, since
       demyelination associated with complement activation has been
       reported in about half of patients in pathological-histological
       studies of early multiple sclerosis,5 and a similar incidence of
       reactivity to oligodendrocytes by serum IgG from patients with
       multiple sclerosis has been reported.6 The prevalence of
       anti-KIR4.1 antibodies in patients with multiple sclerosis has
       been questioned in a few studies,7, 8 including a study by
       Brickshawana and colleagues,8 the results of which are reported
       in The Lancet Neurology.
       KIR4.1 is an inward rectifying potassium channel expressed on
       oligodendrocytes and the endfeet of astrocytes (including
       retinal Müller glia and Hensen's and Claudius' cells in the
       cochlea). It contributes to the maintenance of electrochemical
       gradient by removing potassium from the extracellular space and
       to the maintenance of potassium and water balance by acting in
       concert with aquaporin-4 to maintain osmotic homoeostasis.
       Similar to aquaporin-4, KIR4.1 is not uniquely expressed in CNS
       cells—it is present also in the retina, kidney, and parietal
       cells of gastric mucosal epithelium.9 Antibodies against
       parietal cells have been detected in the sera of patients with
       multiple sclerosis, particularly in those with gastrointestinal
       disturbances.10 Therefore, the occurrence of antibodies against
       KIR4.1 might merely be an epiphenomenon rather than a sign of
       causality.
       A study by Nerrant and colleagues,7 using ELISA, reported that
       only 7·5% of 268 patients with multiple sclerosis had
       anti-KIR4.1 antibodies, and that this proportion did not
       significantly differ to those of healthy controls or patients
       with other neurological diseases; immunofluorescence analysis
       did not detect any specific staining. Brickshawana and
       colleagues8 detect reactivity to KIR4.1 in sera of three (<1%)
       of 268 people with multiple sclerosis and two (<1%) of 208
       controls.
       Click to toggle image size
       Click to toggle image size
       Full-size image (91K) Animated Healthcare Ltd/Science Photo
       Library
       The discrepancies between the results of Srivastava and
       colleagues' study2 and those reported by others (Nerrant and
       colleagues7 and Brickshawana and colleagues8) are difficult to
       explain, since very similar techniques were used for the
       detection of KIR4.1 antibodies. However, in a subsequent study
       from the Munich group (Schirmer and colleagues11), tetramers or
       high-order polymers of KIR4.1 were used for antibody detection
       in the ELISA assay. This approach might be important, since such
       human channel antibodies might be directed against
       conformational epitopes, which might be detected with higher
       affinity in clustered proteins within the cell membrane, but
       binding of the antibodies might still be blocked by a specific
       competing peptide as shown by Srivastava and colleagues.2 Thus,
       whether subtle differences in the detection assays between the
       studies2, 7, 8, 11 might explain the divergent results awaits
       further clarification.
       When KIR4.1 antibodies, which are directed against epitopes
       expressed on the surface of oligodendrocytes or astrocytes, are
       present in patients with multiple sclerosis, the cells
       recognised by these antibodies are thought to be destroyed in a
       complement-dependent manner in early stages of the development
       of multiple sclerosis lesions. Furthermore, a selective loss of
       KIR4.1 from the remaining glia is expected. Studies by the
       Munich group2, 11 suggest that both complement activation and
       selective loss of KIR4.1 occur in active multiple sclerosis
       lesions; Brickshawana and colleagues8 report no KIR4.1 loss from
       glia in early active demyelinating lesions, but rather an
       apparent increase in astrocytic and oligodendrocytic KIR4.1
       immunoreactivity in early active and remyelinated plaques and in
       periplaque white matter. The interpretation of these findings is
       challenging, since KIR4.1 might occur as homotetramers in
       oligodendrocytes and as heterotetramers with KIR5.1 in
       astrocytes.11 Furthermore, KIR4.1 loss will also occur when
       oligodendrocytes are lost in the demyelinating process or
       astrocyte processes are damaged by mechanisms independent of
       antibody-mediated and complement-mediated immune reactions.12
       Finally, technical issues of immunocytochemistry (eg, use of
       frozen vs paraffin sections) and exact staging of lesions might
       partly differ between the studies.
       Do these findings, from independent research groups, mean that
       research into KIR4.1 in multiple sclerosis should end? We hope
       not. The reasons for the discrepancies between the
       investigations might be at least partly technical and call for
       additional work. Pathological studies using different analytical
       approaches are also warranted to deepen the understanding of
       this potentially revolutionary aspect of multiple sclerosis
       research. Many unanswered questions related to KIR4.1 function
       still remain. The coexpression of KIR4.1 and aquaporin-4
       channels in endfeet of astrocytes and their synergistic effect
       in maintaining osmotic homoeostasis is intriguing, especially
       when considering that most retinal pathological changes
       characterised by Müller cell damage are accompanied by changes
       of the amount or spatial distribution of both channels. Finally,
       the potential relation between anti-KIR4.1 antibodies and a more
       general dysfunction of immune-mediated mechanisms in patients
       with multiple sclerosis deserves further investigation.
       MF serves on the scientific advisory board for Teva
       Pharmaceutical Industries; has received compensation for
       consulting services or speaking activities from Bayer Schering
       Pharma, Biogen Idec, Merck Serono, and Teva Pharmaceutical
       Industries; and receives research support from Bayer Schering
       Pharma, Biogen Idec, Merck Serono, Teva Pharmaceutical
       Industries, Italian Ministry of Health, Fondazione Italiana
       Sclerosi Multipla, Cure PSP, and the Jacques and Gloria
       Gossweiler Foundation (Switzerland). MAR has received speakers'
       honoraria from Novartis, Biogen Idec, and Serono Symposia
       International Foundation and receives research support from the
       Italian Ministry of Health and Fondazione Italiana Sclerosi
       Multipla. HL has received honoraria for lectures from Teva,
       Novartis, and Biogen and served as a consultant for Amgen and
       Baxter.
       References
       1 Lennon VA, Wingerchuk DM, Kryzer TJ, et al. A serum
       autoantibody marker of neuromyelitis optica: distinction from
       multiple sclerosis. Lancet 2004; 364: 2106-2112. Summary | Full
       Text | PDF(1049KB) | CrossRef | PubMed
       2 Srivastava R, Aslam M, Kalluri SR, et al. Potassium channel
       KIR4.1 as an immune target in multiple sclerosis. N Engl J Med
       2012; 367: 115-123. CrossRef | PubMed
       3 Bradl M, Misu T, Takahashi T, et al. Neuromyelitis optica:
       pathogenicity of patient immunoglobulin in vivo. Ann Neurol
       2009; 66: 630-643. CrossRef | PubMed
       4 Kraus V, Srivastava R, Kalluri SR, et al. Potassium channel
       KIR4.1-specific antibodies in children with acquired
       demyelinating CNS disease. Neurology 2014; 82: 470-473. CrossRef
       | PubMed
       5 Lucchinetti C, Bruck W, Parisi J, Scheithauer B, Rodriguez M,
       Lassmann H. Heterogeneity of multiple sclerosis lesions:
       implications for the pathogenesis of demyelination. Ann Neurol
       2000; 47: 707-717. CrossRef | PubMed
       6 Lily O, Palace J, Vincent A. Serum autoantibodies to cell
       surface determinants in multiple sclerosis: a flow cytometric
       study. Brain 2004; 127: 269-279. CrossRef | PubMed
       7 Nerrant E, Salsac C, Charif M, et al. Lack of confirmation of
       anti-inward rectifying potassium channel 4.1 antibodies as
       reliable markers of multiple sclerosis. Mult Scler
       201410.1177/1352458514531086. published online April 22. PubMed
       8 Brickshawana A, Hinson SR, Romero MF, et al. Investigation of
       the KIR4.1 potassium channel as a putative antigen in patients
       with multiple sclerosis: a comparative study. Lancet Neurol
       2014. published online July 7.
  HTML http://dx.doi.org/10.1016/S1474-4422(14)70141-3.
       9 Jin X, Yu L, Wu Y, et al. S-Glutathionylation underscores the
       modulation of the heteromeric Kir4.1-Kir5.1 channel in oxidative
       stress. J Physiol 2012; 590: 5335-5348. CrossRef | PubMed
       10 Banati M, Csecsei P, Koszegi E, et al. Antibody response
       against gastrointestinal antigens in demyelinating diseases of
       the central nervous system. Eur J Neurol 2013; 20: 1492-1495.
       PubMed
       11 Schirmer L, Srivastava R, Kalluri SR, et al. Differential
       loss of KIR4.1 immunoreactivity in multiple sclerosis lesions.
       Ann Neurol 201410.1002/ana.24168. published online April 29.
       PubMed
       12 Sharma R, Fischer MT, Bauer J, et al. Inflammation induced by
       innate immunity in the central nervous system leads to primary
       astrocyte dysfunction followed by demyelination. Acta
       Neuropathol 2010; 120: 223-236. CrossRef | PubMed
       a Neuroimaging Research Unit, Institute of Experimental
       Neurology, Division of Neuroscience, Scientific Institute and
       University Ospedale San Raffaele, 20132 Milan, Italy
       b Department of Neurology, Scientific Institute and University
       Ospedale San Raffaele, 20132 Milan, Italy
       c Center for Brain Research, Medical University of Vienna,
       Austria
       #Post#: 550--------------------------------------------------
       Re: Article about the KIR4.1 debate
   DIR By: agate
       Date: November 10, 2014, 3:39 pm
       ---------------------------------------------------------
       Matt, thanks so much for posting this!
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