( ssembly takes place. Finally in 1...)Prestigious Wilson Award Goes To Yale Cell Biologist Joel Rosenbaum,Health

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Finally, in 1992, using new high-resolution microscopes in the laboratory of Paul Forscher, who had recently joined the department, Rosenbaum’s graduate student, Keith Kozminski, was first able to observe these elevators. He saw particles moving up and down the length of the flagella, between the membrane and the microtubule-based core of the flagella. They named the process intraflagellar transport (IFT).

These initial observations led to biochemical and genetic studies showing that molecular motors controlled movement of the IFT particles. When the motors were stopped, flagella would not form, and already-formed flagella became shorter. They also showed that the IFT particles served as transports, carrying prefabricated parts of the flagellar core to the tip for assembly, and recycling turnover products from the tip back to the cell body. Over the next several years, IFT particles were isolated, their polypeptides identified, and many of the genes for IFT cloned.

When the IFT genes were compared with the various available “on line” gene libraries, they were surprised to find that a sequence matching one Chlamydomonas IFT gene, called “IFT88” was in the mouse genome and was the gene which was defective in the then-current mouse model of polycystic kidney disease (PKD).

The relationship between an IFT particle gene that would block flagella assembly in Chlamydomonas when mutated and produce PKD in the mouse model seemed remote. Few had paid attention to the fact that the cells forming the kidney tubules each had a single non-motile “primary” cilium pointing toward the center of the tubule.

When Rosenbaum and his colleagues Gregory Pazour and George Witman at University of Massachusetts Medical School examined kidney tubules of the PKD mutant mouse with the scanning electron microscope, they found that the cilia were either short or missing. They also provided conclusive evidence for the im
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