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Table 2 Statistical analysis of GAG percentage variation from pre-ERT values

From: Clinical efficacy of Enzyme Replacement Therapy in paediatric Hunter patients, an independent study of 3.5 years

Age group

Time point from start of ERT

n

Median (minimum-maximum)

p-value

A

1 yr ± 4 m

12

37.6 (7.8-76.0)

0.0005*

2 yrs ± 4 m

9

29.4 (3.9-92.1)

0.0039*

3 yrs ± 4 m

7

24.5 (13.7-81.8)

0.0156*

Last

13

25.4 (5.7-79.2)

0.0002*

B

1 yr ± 4 m

6

33.9 (17.1-88.1)

0.0313*

2 yrs ± 4 m

4

19.5 (17.4-36.0)

0.1250

3 yrs ± 4 m

4

19.2 (18.0-23.1)

0.1250

Last

7

18.2 (14.6-55.8)

0.0156*

C

1 yr ± 4 m

5

26.4 (8.9-38.8)

0.0625

2 yrs ± 4 m

4

16.5 (9.3-29.6)

0.1250

3 yrs ± 4 m

5

26.4 (5.7-35.1)

0.0625

Last

5

13.3 (5.1-28.2)

0.0625

A + B

1 yr ± 4 m

18

35.6 (7.8-88.1)

< .0001*

2 yrs ± 4 m

13

20.2 (3.9-92.1)

0.0002*

3 yrs ± 4 m

11

21.0 (13.7-81.8)

0.0010*

Last

20

24.0 (5.7-79.2)

< .0001*

A + B + C

1 yr ± 4 m

23

31.8 (7.8-88.1)

< .0001*

2 yrs ± 4 m

17

20.0 (3.9-92.1)

< .0001*

3 yrs ± 4 m

16

22.0 (5.7-81.8)

< .0001*

Last

25

18.2 (5.1-79.2)

< .0001*

  1. Urinary GAG values are expressed at each time-point, from start of ERT as percentage with respect to the pre-ERT baseline level, taken as 100%. The number of available samples considered in the calculation (n), the median value and minimum and maximum values, together with the p-value of the statistical test applied (Wilcoxon signed rank test), are reported. Statistically significant values (p < 0.05) are marked with an asterisk.