NATURE | VOL 406 | 17 AUGUST 2000 | www.nature.com 739
letters to nature
.................................................................
Progression of autoimmune diabetes
driven by avidity maturation
of a T-cell population
Abdelaziz Amrani*, Joan Verdaguer*, Pau Serra*, Sabrina Tafuro²,
Rusung Tan³ & Pere Santamaria*
* Department of Microbiology and Infectious Diseases, Faculty of Medicine,
University of Calgary, Health Sciences Centre, 3330 Hospital Drive NW, Calgary,
Alberta T2N 4N1, Canada
² MRC Human Immunology Unit, Institute of Molecular Medicine,
John Radcliffe Hospital, Oxford OX3 9DU, UK
³ Department of Pathology and Laboratory Medicine, University of British
Columbia and BC's Children's Hospital, 4480 Oak Street, Vancouver, V6H 3V4,
British Columbia, Canada
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For unknown reasons, autoimmune diseases such as type 1
diabetes develop after prolonged periods of in¯ammation of
mononuclear cells in target tissues
1
. Here we show that progres-
sion of pancreatic islet in¯ammation to overt diabetes in non-
obese diabetic (NOD) mice is driven by the `avidity maturation' of
a prevailing, pancreatic beta-cell-speci®c T-lymphocyte popula-
tion carrying the CD8 antigen. This T-lymphocyte population
recognizes two related peptides (NRP and NRP-A7)
2
in the context
of H-2K
d
class I molecules of the major histocompatibility com-
plex (MHC). As pre-diabetic NOD mice age, their islet-associated
CD8
+
T lymphocytes contain increasing numbers of NRP-A7-
reactive cells, and these cells bind NRP-A7/H-2K
d
tetramers with
increased speci®city, increased avidity and longer half-lives.
Repeated treatment of pre-diabetic NOD mice with soluble
NRP-A7 peptide blunts the avidity maturation of the NRP-A7-
reactive CD8
+
T-cell population by selectively deleting those
clonotypes expressing T-cell receptors with the highest af®nity
and lowest dissociation rates for peptide±MHC binding. This
inhibits the local production of T cells that are cytotoxic to beta
cells, and halts the progression from severe insulitis to diabetes.
We conclude that avidity maturation of pathogenic T-cell popula-
tions may be the key event in the progression of benign in¯am-
mation to overt disease in autoimmunity.
Nonobese diabetic mice develop in¯ammation of pancreatic
islets (insulitis) at 3 weeks of age, but do not begin to develop
diabetes until 10 weeks later
1
. Although CD8
+
cytotoxic T lympho-
cytes (CTLs) are well established as killers of insulin-producing
pancreatic beta cells in autoimmune diabetes
3±9
, the mechanisms
that drive the progression of insulitis to overt diabetes remain a
mystery. It has been shown that most of the islet-associated CD8
+
CTLs in NOD mice are cytotoxic to beta cells in the context of the
MHC class I molecule H-2K
d
, and that many of these CTLs express
highly homologous T-cell receptor (TCR) a-subunit chains (Va17
and Ja42 elements joined by the N-region sequence M-R-D/E)
7,9,10
.
By screening combinatorial peptide libraries, we have identi®ed two
peptide ligands for this group of CTLs: NRP (amino-acid sequence
KYNKANWFL), and NRP-A7 (KYNKANAFL), an alanine mutant
analogue of NRP with superior agonistic properties
2
. As many of the
CTL clones derived from islets of diabetic NOD mice recognize
these peptides
2
, and as transgenic NOD mice expressing an NRP/
NRP-A7-reactive TCR (8.3-TCRab-transgenic NOD mice) become
diabetic shortly after developing insulitis
8
, we thought that progres-
sion of insulitis to diabetes might be driven by the accumulation of
NRP/NRP-A7-reactive CTLs in islets.
To follow the accumulation of antigen-speci®c CD8
+
T cells in
pancreatic islets of pre-diabetic NOD mice, we generated H-2K
d
tetramers containing the peptides NRP, NRP-A7, TUM
(KYQAVTTTL, a negative control
11
) or INS (LYLVCGERG, an
insulin-derived peptide recognized by islet-associated T cells from
young NOD mice
12
). The NRP and NRP-A7 tetramers stained
virtually all the splenic CD8
+
T cells from 8.3-TCRab-transgenic
NOD mice, but less than 1% of the splenic CD8
+
T cells from NOD
mice. The INS tetramer stained the INS-reactive CTL clone G9C8
(ref. 12), but neither this tetramer nor the negative control tetramer
(TUM) stained the splenic CD8
+
T cells from NOD or 8.3-TCRab-
transgenic NOD mice (data not shown).
As tetramer-reactive cells represent a very small fraction of all the
T cells contained in freshly isolated islets (see Fig. 1 legend), we
assayed for tetramer-reactive clonotypes within the cell population
that has undergone antigen-driven activation and thus is likely to
contain autoreactive speci®cities. Islets from non-diabetic 5-, 9-, 15-
and 20-week-old NOD mice were cultured in the presence of
recombinant interleukin-2 (rIL-2) for 6±7 days, to selectively
expand IL-2 receptor-positive T cells, which constitute about 10%
of all the islet-associated CD8
+
T cells
7
. There were age-dependent
increases in the absolute number of T cells recovered from NOD
a TUM
INS
NRP
NRP-A7
% Tetramer
+
CD8
+
T cells
Tetramer
+
CD8
+
T cells (× 10
5
)
5 9 15 20
0
5
10
15
20
25
1
1
c
0
0.2
0.4
0.6
5 9 15 20
b
Tetramer-PE
TUM NRP NRP-A7
CD8-FITC
8.3-TCR-
transgenic
NOD
NOD
0.7 89 95
0.8 23 25
0.6 6 25
2
Age (weeks)
Figure 1 TUM-, INS-, NRP- and NRP-A7-reactive CD8
+
T cells derived from pancreatic
islets. a, Percentage of tetramer-reactive T cells. Data (mean 6 s.e.m.) are from mice
aged 5 weeks (n= 3), 9 weeks (n= 13), 15 weeks (n= 7) and 20 weeks (n= 11).
P values: INS and NRP versus TUM at 5 weeks, P , 0.04; NRP and NRP-A7 versus TUM or
INS at 9 weeks, P , 0.0006; NRP or NRP-A7 versus TUM or INS at 15 weeks, P , 0.013
and P , 0.003; NRP and NRP-A7 versus TUM at 20 weeks, P , 0.0001. `1', NRP-A7
versus NRP at 15 and 20 weeks, P , 0.05 and P , 0.008, respectively. Percentage of
tetramer-reactive CD8
+
cells in freshly isolated islets are 0.13 6 0.38% (n = 6; 5 weeks),
0.95 6 0.67% (n = 4; 9 weeks) and 1.6 6 0.5% (n = 2; 15 weeks) (NRP-A7); and
0.71 6 0.4 (n = 4; 5 weeks) and 0.31 6 0.4 (n = 4; 9 weeks) (INS). b, Absolute number
(mean 6 s.e.m.) of tetramer-reactive T cells from the same samples as in a. P values:
NRP-A7 at 15 and 20 weeks versus 9 and 5 weeks, P , 0.04; NRP and NRP-A7 versus
TUM at 9 weeks, P , 0.001; NRP or NRP-A7 versus INS at 9 weeks, P , 0.035; NRP or
NRP-A7 versus TUM or INS at 15 weeks, P , 0.037; NRP or NRP-A7 versus TUM at
20 weeks, P , 0.0001. `2', NRP-A7 versus NRP at 20 weeks, P , 0.05.
c, Representative tetramer staining patterns. Over 90% of the CD8-negative cells shown
in the plots are CD4
+
.
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