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Four and a half LIM protein 1C (FHL1C): a binding partner for voltage-gated potassium channel K(v1.5)
Ivana Poparic1, Wolfgang Schreibmayer, Benedikt Schoser
1Institute of Human Genetics, Institute of Molecular Biology and Biochemistry, Medical University of Graz, Graz, Austria.
Insights
Functional Four-and-a-half LIM domain protein 1 isoform A (FHL1A) is absent in X-linked myopathy with postural muscle atrophy (XMPMA) patient myoblasts. FHL1C interacts with K(v1.5) channels, impacting myoblast proliferation and potentially contributing to XMPMA pathogenesis.
Area of Science:
- Molecular biology
- Cell biology
- Genetics
Background:
- Four-and-a-half LIM domain protein 1 isoform A (FHL1A) is crucial for skeletal and cardiac muscle function.
- Mutations in the FHL1 gene cause hereditary myopathies, such as X-linked myopathy with postural muscle atrophy (XMPMA).
Purpose of the Study:
- To investigate the role of FHL1A and FHL1C in XMPMA patient myoblasts.
- To explore the interaction between FHL1C and the K(v1.5) potassium channel in muscle cells.
Main Methods:
- Studied myoblasts from XMPMA patients and controls.
- Performed pull-down assays, confocal microscopy, and two-electrode voltage clamp experiments.
- Analyzed protein expression, cell proliferation, and ion channel activity.
Main Results:
- XMPMA myoblasts lack functional FHL1A but show unchanged or increased FHL1C expression.
- XMPMA myoblasts exhibit reduced proliferation and an accumulation in the G(0)/G(1) phase.
- FHL1C directly interacts with K(v1.5) channels, colocalizes in atrial cells, and reduces K(+) currents upon coexpression.
- Low K(v1.5) expression was detected in XMPMA myoblasts.
Conclusions:
- FHL1C has a biological relevance and interacts with K(v1.5) channels.
- The FHL1C-K(v1.5) interaction may influence myoblast proliferation and contribute to XMPMA pathophysiology.
Abstract:
Four-and-a-half LIM domain protein 1 isoform A (FHL1A) is predominantly expressed in skeletal and cardiac muscle. Mutations in the FHL1 gene are causative for several types of hereditary myopathies including X-linked myopathy with postural muscle atrophy (XMPMA). We here studied myoblasts from XMPMA patients. We found that functional FHL1A protein is completely absent in patient myoblasts. In parallel, expression of FHL1C is either unaffected or increased. Furthermore, a decreased proliferation rate of XMPMA myoblasts compared to controls was observed but an increased number of XMPMA myoblasts was found in the G(0)/G(1) phase. Furthermore, low expression of K(v1.5), a voltage-gated potassium channel known to alter myoblast proliferation during the G(1) phase and to control repolarization of action potential, was detected. In order to substantiate a possible relation between K(v1.5) and FHL1C, a pull-down assay was performed. A physical and direct interaction of both proteins was observed in vitro. In addition, confocal microscopy revealed substantial colocalization of FHL1C and K(v1.5) within atrial cells, supporting a possible interaction between both proteins in vivo. Two-electrode voltage clamp experiments demonstrated that coexpression of K(v1.5) with FHL1C in Xenopus laevis oocytes markedly reduced K(+) currents when compared to oocytes expressing K(v1.5) only. We here present the first evidence on a biological relevance of FHL1C.
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