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Cryptorchidism is Associated with Lower Extremity Contracture Location and Severity in Patients with Cerebral Palsy: a PHIS Cohort Study
Eric M. Bortnick, MD, Tanya Logvinenko, PhD, HH Scott Wang, MD, David J. Fogelman, MD, Benjamin J. Shore, MD MPH, Caleb P. Nelson, MD MPH, Michael P. Kurtz, MD MPH.
Boston Childrens Hospital, Boston, MA, USA.

BACKGROUND: Cryptorchidism affects 24-50% of boys with Cerebral Palsy (CP) and the cause remains unknown. Proposed etiologies range from hormonal to muscular. We propose cremasteric contracture as one cause, as the cremaster is likely subject to the same processes that drive other skeletal muscular contractures in CP. We hypothesized that among boys with CP, those with more severe lower limb spasticity, as defined by need for intervention, and those with more proximal muscular contractures (i.e. closer to the groin), are more likely to have undergone orchidopexy for cryptorchidism.
METHODS: Using the Pediatric Health Information System (PHIS) database (2016-2020), we examined male patients with CP and stratified these patients into those with and without lower limb spasticity, as defined by relevant ICD 10 and CPT codes, and compared these groups for occurrence of orchidopexy for cryptorchidism. Demographic characteristics were summarized and compared between patients who underwent and those without orchidopexy using Chi-squared and Mann-Whitney U tests for categorical and continuous variables, respectively. Logistic regression was used to investigate association between orchidopexy and types of contractions and interventions.
RESULTS: 44,561 male patients with CP were identified. 1.6% of patients underwent orchidopexy at a median age of 7.7 years (IQR: 4.5-11.3). 16.0% of patients had lower extremity contractures, 1.1% had only upper extremity contractures, 82.8% had no documented extremity contractures. Presence of lower extremity contracture was significantly associated with a higher rate of orchidopexy for cryptorchidism compared to absence of any extremity contracture (OR=1.33 [1.10-1.59], p=0.0026); importantly, upper extremity contracture was not associated with orchidopexy risk (OR=0.507 [0.16-1.19], p=0.1774). Of the 7,134 patients with lower extremity contractures, need for intervention for extremity spasticity was significantly associated with higher rate of orchidopexy (injection procedures: (OR=2.47 [1.17-6.39], p=0.0337); limb surgical procedure: (OR=2.60 [1.22-6.76], p=0.0259)). Among patients with lower extremity contractures, proximity of contracture to the groin was significantly associated with an increased risk of orchidopexy (proximal contracture vs distal contracture OR=2.52 [1.42-4.96], p=0.0034).
CONCLUSIONS: In the largest series to date on CP and UDT, we show a relationship between lower extremity contracture and orchidopexy in the CP population, as well as association between the location and severity of contracture and the probability of orchidopexy. These findings support a hypothesis that cremaster muscle contracture is an important factor in presence of UDT in CP. Providers should continue to examine for cryptorchidism in boys with CP as they age, particularly those with more severe lower extremity contractures. Future studies may provide insight into whether including treatment of the cremaster in concert with treatment of lower extremity muscle spasticity could prevent future testicular ascent.
Table 1: Demographics

nNo UDTUDTp-value
Overall (%)4456143844 (98.39)717 (1.61)
Age (median years [IQR])
At last follow up10.91 [6.07, 16.05]10.95 [6.08, 16.11]8.87 [5.69, 12.34]<0.001
At time of orchidopexy7.7 [4.5-11.3]
Ethnicity (%)0.046
Hispanic or Latino8612 (19.3)8448 (19.3)164 (22.9)
Not Hispanic or Latino32934 (73.9)32424 (74.0)510 (71.1)
NA3015 (6.8)2972 (6.8)43 (6.0)
White (%)27441 (61.6)26974 (61.5)467 (65.1)
Non-White (%)17210 (38.4)16870 (38.5)250 (34.9)
Census Region (%)0.047
Midwest11572 (26.0)11361 (25.9)211 (29.4)
Northeast5300 (11.9)5234 (11.9)66 (9.2)
South17864 (40.1)17583 (40.1)281 (39.2)
West9825 (22.0)9666 (22.0)159 (22.2)
Insurance (%)0.194
Private15553 (34.9)15320 (34.9)233 (32.5)
Public27217 (61.1)26761 (61.0)456 (63.6)
Other1221 (2.7)1206 (2.8)15 (2.1)
NA570 (1.3)557 (1.3)13 (1.8)
Prematurity (%)0.001
Yes2013 (4.5)1961 (4.5)52 (7.3)
No42548 (95.5)41883 (95.5)665 (92.7)
Extremity Contracture Location (%)0.003
Lower7134 (16.0)6989 (15.9)145 (20.2)
Upper Only509 (1.1)505 (1.2)4 (0.6)
None36918 (82.8)36350 (82.9)568 (79.2)

Table 2: Logistic Regression for Risk of Orchidopexy for UDT
Whole Cohort (n=44561)OR95% CIp-value
Types of Contractures0.0035
Upper Only Contraction vs None0.51(0.16, 1.19)0.1774
Lower Contraction vs None1.33(1.10, 1.59)0.0026
Lower Contraction vs Upper Only2.62(1.10, 8.54)0.0585
Lower Extremity Contracture (n=7134)
Need for Intervention0.0361
Injection Procedure vs None2.47(1.17, 6.39)0.0337
Limb Surgery vs None2.60(1.22, 6.76)0.0259
Limb Surgery vs Injection Procedure1.05(0.75, 1.47)0.7660
Contracture Location
Proximal Contracture vs Distal Contracture2.52(1.42, 4.96)0.0034

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