Table 3

Multivariate Cox regression analysis for event-time endpoints

Endpoint, predictor, and cutoffN (%)%HR95% CIP
Orthopedic endpoints      
 Thoracolumbar kyphosis (surgical intervention) 67 (34)     
 Cervical instability (surgical intervention) 9 (5)     
 Cord compression (surgical intervention) 21 (11)     
  IDUA level      
   <Reference*  22   
   ≥Reference*  0.34 0.14-0.82 .02 
  Age at HCT      
   <16 months    
   ≥16 months  16 2.84 1.02-1.41 .04 
 Hip dysplasia (surgical intervention) 71 (36)     
  IDUA level      
   <Reference*  52   
   ≥Reference*  31 0.53 0.32-0.86 .01 
 Genu valgum (surgical intervention) 75 (38)     
  IDUA level      
   <Reference*  54   
   ≥Reference*  33 0.50 0.31-0.81 .005 
 Carpal tunnel syndrome (surgical intervention) 89 (45)     
  Age at HCT      
   <16 months  33   
   ≥16 months  56 1.72 1.11-2.68 .02 
  IDUA level      
   <Reference*  61   
   ≥Reference*  39 0.58 0.37-0.92 .02 
 Trigger fingers (surgical intervention) 33 (18)     
Cardiac endpoints      
 Cardiomyopathy (treatment) 33 (18)     
Respiratory endpoint      
 Overnight hypoxia (respiratory support) 9 (4)     
  IDUA level      
   <Reference*  12   
   ≥Reference*  0.15 0.03-0.78 .02 
Ophthalmologic endpoints      
 Corneal clouding (surgical intervention) 20 (10)     
 Glaucoma (surgical intervention) 11 (6)     
 Cataract (surgical intervention) 6 (3)     
  TBI      
   No    
   Yes  22 40.13 4.66-345.72 .001 
Audiologic endpoint      
 Hearing loss (hearing aids) 59 (32)     
  IDUA level      
   <Reference*  51   
   ≥Reference*  26 0.42 0.24-0.73 .002 
Endocrinologic endpoints      
 Growth retardation (GH treatment) 26 (13)     
  TBI      
   No  11   
   Yes  32 4.82 1.84-12.65 .001 
  Follow-up center   1.20 1.02-1.41 .03 
 Hypothyroidism (treatment) 12 (7)     
  TBI      
   No    
   Yes  18 6.12 1.42-26.33 .02 
  Follow-up center   1.37 1.03-1.81 .03 
Endpoint, predictor, and cutoffN (%)%HR95% CIP
Orthopedic endpoints      
 Thoracolumbar kyphosis (surgical intervention) 67 (34)     
 Cervical instability (surgical intervention) 9 (5)     
 Cord compression (surgical intervention) 21 (11)     
  IDUA level      
   <Reference*  22   
   ≥Reference*  0.34 0.14-0.82 .02 
  Age at HCT      
   <16 months    
   ≥16 months  16 2.84 1.02-1.41 .04 
 Hip dysplasia (surgical intervention) 71 (36)     
  IDUA level      
   <Reference*  52   
   ≥Reference*  31 0.53 0.32-0.86 .01 
 Genu valgum (surgical intervention) 75 (38)     
  IDUA level      
   <Reference*  54   
   ≥Reference*  33 0.50 0.31-0.81 .005 
 Carpal tunnel syndrome (surgical intervention) 89 (45)     
  Age at HCT      
   <16 months  33   
   ≥16 months  56 1.72 1.11-2.68 .02 
  IDUA level      
   <Reference*  61   
   ≥Reference*  39 0.58 0.37-0.92 .02 
 Trigger fingers (surgical intervention) 33 (18)     
Cardiac endpoints      
 Cardiomyopathy (treatment) 33 (18)     
Respiratory endpoint      
 Overnight hypoxia (respiratory support) 9 (4)     
  IDUA level      
   <Reference*  12   
   ≥Reference*  0.15 0.03-0.78 .02 
Ophthalmologic endpoints      
 Corneal clouding (surgical intervention) 20 (10)     
 Glaucoma (surgical intervention) 11 (6)     
 Cataract (surgical intervention) 6 (3)     
  TBI      
   No    
   Yes  22 40.13 4.66-345.72 .001 
Audiologic endpoint      
 Hearing loss (hearing aids) 59 (32)     
  IDUA level      
   <Reference*  51   
   ≥Reference*  26 0.42 0.24-0.73 .002 
Endocrinologic endpoints      
 Growth retardation (GH treatment) 26 (13)     
  TBI      
   No  11   
   Yes  32 4.82 1.84-12.65 .001 
  Follow-up center   1.20 1.02-1.41 .03 
 Hypothyroidism (treatment) 12 (7)     
  TBI      
   No    
   Yes  18 6.12 1.42-26.33 .02 
  Follow-up center   1.37 1.03-1.81 .03 

Only statistically significant results are shown.

*Lower limit of local reference.

Median age at HCT.

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