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Vol. 12, Issue 7, 2075-2085, July 2001
*Section on Human Biochemical Genetics, Heritable Disorders Branch,
National Institute of Child Health and Human Development, National
Institutes of Health, Bethesda, Maryland 20892; and
Patients with Hermansky-Pudlak syndrome type 2 (HPS-2) have
mutations in the Department of Dermatology, University of Cincinnati,
Cincinnati, Ohio 45267-0592
3A subunit of adaptor complex-3 (AP-3) and
functional deficiency of this complex. AP-3 serves as a coat protein in
the formation of new vesicles, including, apparently, the platelet's dense body and the melanocyte's melanosome. We used HPS-2 melanocytes in culture to determine the role of AP-3 in the trafficking of the
melanogenic proteins tyrosinase and tyrosinase-related protein-1 (TRP-1). TRP-1 displayed a typical melanosomal pattern in both normal
and HPS-2 melanocytes. In contrast, tyrosinase exhibited a melanosomal
(i.e., perinuclear and dendritic) pattern in normal cells but only a
perinuclear pattern in the HPS-2 melanocytes. In addition, tyrosinase
exhibited a normal pattern of expression in HPS-2 melanocytes
transfected with a cDNA encoding the 3A subunit of the AP-3 complex.
This suggests a role for AP-3 in the normal trafficking of tyrosinase
to premelanosomes, consistent with the presence of a dileucine
recognition signal in the C-terminal portion of the tyrosinase
molecule. In the AP-3-deficient cells, tyrosinase was also present in
structures resembling late endosomes or multivesicular bodies; these
vesicles contained exvaginations devoid of tyrosinase. This suggests
that, under normal circumstances, AP-3 may act on multivesicular bodies
to form tyrosinase-containing vesicles destined to fuse with
premelanosomes. Finally, our studies demonstrate that tyrosinase and
TRP-1 use different mechanisms to reach their premelanosomal destination.
Corresponding author. E-mail address:
boissyre{at}ucmail.uc.edu.
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|
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|
|
|
|
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|
|
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|
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