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Re: Gentamicin-SARA

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Sara,

That is also Eilish's mutation so I would love to hear any information

you get about it.

(Australia)

Re: Gentamicin

I discussed this with Zach's doctor since Zach has a stop mutation

(K710X). His theory was that gentamicin would be altered in such a

way that it would no long be an " antibiotic " , but it would still be

able to correct the CFTR malfunction in stop mutations. How this

might be accomplished is anyone's guess.

He also theorized that if one mutation could be corrected then the

person might only have the CFTR malfunction of a carrier.

In France they are testing ways to turn on the sodium chloride

channel in the G551D mutation. That is Zach's other mutation so I

am very interested in that too.

Sara

> > I read that in testing Gentamicin in nasal applications it can

result

> > in read-through of CTFR protein for people with Grade I

mutations

> > (ex. G542, W1282X, R553X, 1717-1G->A)

> >

> > http://content.nejm.org/cgi/content/short/349/15/1433

> >

> > I think this drug is usually used for urinary infections. Has

anyone

> > ever heard of it being used for CF?

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This is a pubmed article I found. It is very difficult to

understand but I think the last sentence means that the new drug

they were testing restored the CFTR function for G551D. Others

might be able to interpret this better. - Sara

Comparative pharmacology of the activity of wild-type and G551D

mutated CFTR chloride channel: effect of the benzimidazolone

derivative NS004.

Derand R, Bulteau-Pignoux L, Becq F.

Laboratoire des Biomembranes et Signalisation Cellulaire, UMR 6558

CNRS, Universite de Poitiers, 86022 Poitiers, France.

The pharmacological activation of the cystic fibrosis transmembrane

conductance regulator (CFTR) chloride channel mutated at glycine 551

(G551D-CFTR) was studied in the presence of the benzimidazolone

derivative NS004 and compared to that of wild-type (wt) CFTR. Using

iodide ((125)I) efflux and whole-cell patch-clamp techniques we

found dose-dependent stimulation of phosphorylated wt-CFTR channels

by NS004 with an EC(50) approximately 11 microM. With non-

phosphorylated CFTR, the effect of NS004 was apparent only at

concentration >100 microM. In G551D-CFTR-expressing CHO cells,

neither forskolin (from 0.1 to 10 microM) nor NS004 (from 0.1 to 200

microM) added separately were able to stimulate channel activity.

However, in the presence of 10 microM forskolin, NS004 stimulated

G551D-CFTR activity in a dose-dependent manner with an EC(50)

approximately 1.5 microM. We also determined the half-maximal

effective concentration of forskolin ( EC(50) approximately 3.2

microM) required to stimulate G551D channel activity in presence of

1.5 micro M NS004. No inhibitory effect was observed at high

concentration of NS004 with both wt- and G551D-CFTR. Whole-cell

recordings of CFTR chloride currents from cells expressing wild-type

or G551D-CFTR in the presence of NS004 were linear, time- and

voltage-independent. The inhibitory profile of G551D-CFTR channel

activity was similar to that of wild type, i.e., inhibition by

glibenclamide (100 microM) and DPC (250 microM) but not by DIDS (200

microM) nor calixarene (100 nM). These results show that NS004

activates wt-CFTR channel and restores G551D-CFTR channel activity,

the potency of which depends on both the concentration of NS004 and

the phosphorylation status of CFTR.

> Sara,

>

> That is also Eilish's mutation so I would love to hear any

information

> you get about it.

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This is one of 's as well.

jan

> This is a pubmed article I found. It is very difficult to

> understand but I think the last sentence means that the new drug

> they were testing restored the CFTR function for G551D. Others

> might be able to interpret this better. - Sara

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