Milestones in Goodpasture Research

from EdREN, the website of the Renal Unit of the Royal Infirmary of Edinburgh

This is far from a comprehensive survey - but does show how Goodpasture's disease has progressively revealed information that has proved important in understanding and in treating other, more common kidney disorders.

Year

Observations

First authors
1919 First case report Goodpasture
1964 Associated with anti-GBM antibodies Scheer and Grossman
1975 Effective treatment reported in several patients Lockwood
1977 Injury to kidney enhanced by infection Rees
1978 HLA (tissue type) association noted Rees
1980 Antigen absent from Alport GBM
1988 type IV collagen a3 chain is the target of autoantibodies Saus
1990-94 cDNA cloning of type IV collagen chains Morrison, Turner, et al
1996-8 Peptides presented to T lymphocytes identified Phelps
2004 Peptides attacked by patients identified Cairns et al
2007 Mechanisms of avoiding tolerance Phelps, Turner

Antibodies cause kidney damage. In the 1960s, landmark investigations showed that at least part of the kidney damage in Goodpasture's disease was caused by the anti-GBM antibodies that define the disease. These findings had been predicted from much earlier experiments in animals, but this was the first proof that the same could happen in man.


Effective treatments are developed. Observations in individual patients were followed by the developments in the 1970s described previously. These have subsequently been used (generally even more successfully) for the treatment of other types of crescentic nephritis, particularly SLE and vasculitis
.

The antibodies bind to a single component of certain basement membranes. In the 1980s it was shown that the immune response was to a highly specific component of basement membranes, found in lung and glomerulus (GBM) and a few other locations, but not in all basement membranes.


The Goodpasture antigen is missing in Alport syndrome. Patients with this inherited disease develop kidney failure as young adults. Their GBMs degenerate and glomeruli are destroyed. Goodpasture antigen is usually completely absent from the GBM.


Some people are genetically at risk of Goodpasture's disease. The tissue type of an individual can predispose to or protect from Goodpasture's disease. This is seen in all autoimmune diseases, but it is stronger in Goodpasture's disease than most others. Other genetic factors, so far unknown, will also be involved, but can only be a part of the story.


Lung haemorrhage only occurs if the lungs are damaged. Antibodies cannot reach the basement membrane of the lung until there is damage to lung blood vessels that makes them leaky. This is not true in the kidney. Nor is it true in vasculitis, where the attack may be directed towards the cells lining blood vessels, rather than the basement membrane.


Infection can increase the damage caused by immune attack. During recovery from Goodpasture's disease, infection can worsen kidney and lung damage. It is now known that this is a general principle of immune damage, not limited to Goodpasture's disease. Treatments to control infection and inflammation are as important as controlling the disease.


Alport syndrome is more complicated than first thought. The gene for the Goodpasture antigen is not involved in most patients with Alport syndrome. Six basement membrane collagen genes have been identified. Three of these proteins are found in GBM, and abnormalities of any of these three can cause Alport syndrome.


The Goodpasture antigen is identified. First identified as a collagen in the 1980s, then as a specific, new type of basement membrane collagen, then 'cloned' by recombinant DNA techniques, and now made as a recombinant molecule for use in research into the cause of Goodpasture's disease and Alport syndrome.

 

Peptides that the immune system may see from the Goodpasture antigen were first identified in the late 1990s.

 

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