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Gitelman syndrome, calcium pyrophosphate dihydrate deposition disease and crowned dens syndrome. A new association?

2009; Oxford University Press; Volume: 49; Issue: 3 Linguagem: Inglês

10.1093/rheumatology/kep324

1462-0332

Marwin Gutiérrez, F Silveri, Chiara Bertolazzi, Fausto Salaffi, Gilberta Giacchetti, Rita Girolimetti, Emilio Filippucci, W. Grassi,

Caveolin-1 and cellular processes

oral prednisolone.Thereafter tacrolimus was introduced to provide ongoing control of her eye disease.The cornea comprises five layers: epithelium, basement membrane zone, stroma, Descemet's membrane and endothelium.Within the stromal layer is a dynamic process of repair and regeneration.Modified fibroblasts constantly remodel the cornea, and their activities are regulated by the presence of MMPs, produced by stromal fibroblasts that degrade the extracellular matrix, and their tissue inhibitors, e.g.tissue inhibitor of metalloproteinases-1, along with other non-specific protease inhibitors [3].PUK is hypothesized to occur when this balance is upset [2].Another study [3] suggests that PUK is due to a cellmediated process, as MMP-1, in particular, is strongly associated with a mononuclear cell infiltrate as described in ulcerating corneas.It is possible that PUK is triggered by the release of MMPs from fibroblasts in response to pro-inflammatory cytokines, e.g.IL-1 produced by activated macrophages.Alternatively, MMPs could be produced by the macrophages themselves [3].There is also evidence for HLA-DR expressing cells, along with helper and cytotoxic T cells, in the cell infiltrate [3].This finding may explain why PUK is a recurring pathology when the cornea is damaged by any other reason.In this case, there had been a long history of keratoconjunctivitis sicca and recurrent episcleritis.As far as we are aware, this is the first case report of PUK following treatment with rituximab.At the time of her corneal melt, our patient did not have any obvious trigger such as cataract surgery, but her cornea might be vulnerable from her previous pathology.Could B-cell depletion cause disequilibrium within the cellular network?Interestingly, rituximab has actually been found to be a useful treatment for PUK, although these cases were associated with WG rather than RA [4,5]. Rheumatology key message. We highlight a possible link between rituximab and PUK.