PMC:1420314 / 4542-10244 JSONTXT

{"target":"https://pubannotation.org/docs/sourcedb/PMC/sourceid/1420314","sourcedb":"PMC","sourceid":"1420314","source_url":"http://www.ncbi.nlm.nih.gov/pmc/1420314","text":"Results\n\nGeneration of ADAM11-deficient mice\nMice carrying a targeted mutation in their Adam11 gene were generated by homologous recombination (Fig. 1A). Correct targeting events were confirmed by Southern blot analysis (Fig. 1B). As a result of this procedure, since a termination codon was introduced in exon 5 in the pro-protein domain, only the truncated form of the ADAM11 protein would be synthesised from this targeted allele. Because pro-protein domains are always removed in mature functional ADAM-proteins by furin-like proteases and are thus thought to be non-functional, we concluded that this truncated form of ADAM11 protein would have no function [26]. Absence of mature ADAM11 protein in homozygous mutants was confirmed by Western blot analysis using a specific antibody that recognises the ADAM11 protein (Fig. 1C). Analysis by Western blot showed that heterozygous mutants produced approximately half of the wild-type levels of ADAM11 protein.\n\nADAM11-deficient mice are viable and appear normal\nMating of mice heterozygous for the ADAM11 mutation yielded a near-Mendelian distribution of genotypes in the offspring and the ratio of females and males was the same in all groups (data not shown). For the present experiments, ADAM11-deficient male mice (backcrossed generations into C57BL/6NCrj, n = 14) were used in all behavioural analyses and anatomical-histological studies. Homozygous ADAM11-deficient mice were viable and appeared normal, indicating that the Adam11 gene is not essential to development and survival. The gain in body weight of (-/-) mice was comparable to that of (+/+) and (+/-) mice at least until 24 weeks of age (data not shown). There was no significant difference in locomotor activity among (+/+), (+/-) and (-/-) genotypes (data not shown). No apparent anatomical-histological change was observed in the spinal cord, heart, lung, liver, kidney, spleen, testis or ovary (data not shown). The brain of (-/-) mice, including the hippocampus (Fig. 1D) and cerebellum (Fig. 1E) appeared normal after HE staining when (+/+), (+/-) and (-/-) mice were compared. Since Adam11 is predominantly expressed in the hippocampus and cerebellum, we applied hippocampus- or cerebellum-dependent behavioural analyses.\n\nSpatial learning in ADAM11-deficient mice\nTo study the functions of ADAM11 in the hippocampus, we examined the spatial learning ability of ADAM11-deficient mice using the Morris Water Maze task [27]. First, mice were trained in a hidden platform task, in which mice searched for a submerged platform to escape from the water. Adam11 (-/-) mice were found to take a significantly longer time to locate the hidden platform than (+/+) and (+/-) mice (Fig. 2A). Nonetheless, the ability of (+/+), (+/-) and (-/-) mice to find the hidden platform improved over test sessions (Fig. 2A). Thus, (+/+), (+/-) and (-/-) mice were able to learn the location of the hidden platform during the course of the trials, although the capacity of (-/-) mice to find the platform was lower than that of (+/+) and (+/-) mice. We further performed a probe test, in which the platform was removed from the pool after completion of the hidden platform task, and the trained mice were allowed to swim freely for 60 sec. The time spent in the target quadrant of (-/-) is significantly lower than that of (+/+) mice (Fig. 2B). Adam11 (+/-) mice appeared to spend a shorter time in the target quadrant, but there was no significant difference between (+/+) and (+/-) mice (Fig. 2B). These results indicate a spatial learning impairment in (-/-) mice. The swimming distance of (+/+), (+/-) and (-/-) mice, however, was similar during the probe trial (Fig. 2C). In the visible platform task, the performance of mice with (+/+), (+/-) and (-/-) genotypes improved during the course of the test sessions, as indicated by a progressive decrease in the latency for locating the platform (Fig. 2D), suggesting that (-/-) mice learn this visible platform task just as well as (+/+) and (+/-) mice. These results also indicated that (-/-) mice have normal visible and motor function and the motivation needed in the water maze task.\n\nMotor function of ADAM11-deficient mice\nTo study the functions of ADAM11 in the cerebellum, we examined the motor function of ADAM11-deficient mice using a rotating rod task [28]. In the rotating rod test, the retention time of (+/-) and (-/-) mice on the rod was not significantly different from that of (+/+) mice at 0 rpm (stationary) [+/-: F (1, 80) = 1.67, p = 0.21; -/-: F (1, 88) = 0.002, p = 0.96] (Fig. 3A). In (+/-) mice, the tendency of decreased retention times was seen at 5, 10 and 15 rpm, but there was no significant difference between (+/+) and (+/-) mice [5 rpm: F (1, 20) = 1.27, p = 0.27; 10 rpm: F (1, 20) = 1.07, p = 0.31; 15 rpm: F (1, 20) = 2.716, p = 0.11] (Figs. 3B, 3C and 3D). However, (-/-) mice fell more quickly from the rod than (+/+) mice at 5, 10 and 15 rpm [5 rpm: F (1, 27.3) = 17.51, p = 0.0002; 10 rpm: F (1, 22) = 11.05, p = 0.003; 15 rpm: F (1, 22) = 19.22, p = 0.0002] (Figs. 3B, 3C and 3D). The retention time was not significantly improved in (-/-) mice across trials of training at 5, 10 and 15 rpm [5 rpm: F (3, 15.3) = 2.05, p = 0.15; 10 rpm: F (7, 11) = 2.61, p = 0.07; 15 rpm: F (19, 26.6) = 1.57, p = 0.14] (Figs. 3B, 3C and 3D). In a traction test, (-/-) mice showed a high grip strength similar to that of (+/+) and (+/-) mice (Fig. 4). The hanging score and time of (-/-) mice were also similar to those of (+/+) and (+/-) mice (Figs. 5A and 5B). A footprint test did not reveal any abnormalities in walking patterns, including stride length and step width, as a function of genotypes (Figs. 6A and 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