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. 2022 Mar 28;17:137. doi: 10.1186/s13023-022-02267-z

Table 2.

The clinical phenotype comparison between different clinical subgroup

Connatal group (n = 28) Transitional group (n = 56) Classic group (n = 27) p value
Disease onset
Nystagmus, n (%) 28 (100) 56(100) 26(96.3) 0.208
Onset age (range) 0.85 m (0, 12) 1 (0, 24) 2 m (0, 16)b 0.016*
Hypotonia, n (%) 18 (64.3)a 50 (89.3) 25 (92.6)b 0.005*
Onset age (range) 6.5 m (2. 18) 8 m (3, 26) 7 m (2, 18) 0.381
Disease progression
Nystagmus decrease 13 (46.4) 32 (57.1) 19 (73.1) 0.136
Decrease age (range) 12 m (4, 96) 26 m (8, 79) 24 m (7, 260) 0.291
Stridor 15 (53.6)a 15 (26.8) 5 (18.5)b 0.011*
Motor milestone
Head control, n (%) 12 (42.9) 48 (85.7) 27 (100) < 0.001*
Acquire age (range) 11 m (4, 71) 10 (3, 60)c 5 (3, 18)b < 0.001*
Sit, n (%) 4 (14.3) 14 (25)c 26 (88.9)b < 0.001*
Acquire age (range) 47 (7, 96) 33 (8, 86)c 12 (6, 84) 0.027*
Stand, n (%) 1 (3.6) 4 (7.1) 5 (18.5) 0.121
Acquire age (range) 13 (13, 13) 41 (26, 48) 36 (16, 84) 0.273
Walk, n (%) 1 (3.6) 2 (3.6) 2 (7.4) 0.705
Acquire age (range) 15 (15, 15) 38.5 (29, 48) 108 (96, 120) 0.165
Cognitive milestone
Recognize stranger, n (%) 21 (75.0) 51 (91.1) 25 (92.6) 0.072
Acquire age (range) 8 (3, 48) 8 (4, 60) 8 (4, 36) 0.955
Speak, n (%) 11 (39.3)a 44 (78.6) 25 (92.6)b < 0.001*
Acquire age(range) 36 (12, 72)a 19 (6, 86) 12 (6, 30)b 0.002*
Other neurological finding
Joint contracture, n (%) 8 (28.6) 10 (17.9) 5 (18.5) 0.494
Spasticity tetraparesis, n (%) 0 4 (7.1) 1 (3.7) 0.322
Pyramidal signs, n (%) 3 (10.7) 3 (5.4) 6 (22.2) 0.068
Brainstem dysfunction, n (%) (Swallowing difficulty, respiration dysfunction) 15 (53.6)a 8 (14.3) 0 (0)b < 0.001*
Ataxia, n (%) 2 (7.1) 3 (5.4) 2 (7.4) 0.917
Seizure, n (%) 4 (14.3) 4 (7.1) 0 0.123

*Significant difference between three groups

aSignificant between connatal and transitional group was significant

bSignificant between connatal and classis group was significant, cSignificant between transitional and classic group was significant