Patient-reported Outcomes for Children With Unilateral Congenital Below Elbow Deficiency
Azarin Shoghi1, Anita Bagley1,2, Lisa V Wagner3
1University of California Davis School of Medicine.
Insights
Children with unilateral congenital below elbow deficiency (UCBED) show mild upper extremity impairment but normal function in other areas. Parents of younger children report greater functional limitations than older children with UCBED.
Area of Science:
- Pediatric Orthopedics
- Rehabilitation Medicine
- Outcome Measurement
Background:
- Patient Reported Outcomes Measurement Information System (PROMIS) is a validated functional assessment tool.
- PROMIS has not been previously validated in children with unilateral congenital below elbow deficiency (UCBED).
Purpose of the Study:
- To validate the PROMIS tool for assessing functional impairment in children with UCBED.
- To compare functional outcomes of children with UCBED to the general population.
- To hypothesize that children with UCBED experience mild upper extremity impairment but normal mobility, pain interference, and peer relations.
Main Methods:
- Retrospective chart review of 55 children (aged 5-17) with UCBED.
- PROMIS questionnaires administered during clinic visits (April 2017 - March 2020).
- Comparison of UCBED patient PROMIS scores with a general reference population; Mann Whitney and ANOVA tests used for subgroup analysis.
Main Results:
- Children with UCBED demonstrated similar PROMIS scores in mobility, peer relations, and pain interference compared to the reference population.
- Mild impairment was observed in upper extremity function for children with UCBED (44.3±10.7).
- Parents of younger children (5-7 years) reported significantly greater upper extremity functional impairment than older children (8-17 years).
Conclusions:
- The PROMIS tool effectively identifies functional status in children with UCBED.
- Children with UCBED generally report functional outcomes similar to the general population, with the exception of upper extremity function.
- Younger children with UCBED, as reported by parents, experience more significant upper extremity functional limitations.
Background:
The Patient Reported Outcomes Measurement Information System (PROMIS ® ) is a validated tool used to evaluate different domains of function in patients with chronic health conditions. This tool has not been validated in children with unilateral congenital below elbow deficiency (UCBED). The purpose of this study was to determine whether PROMIS discerns functional impairment for children with UCBED and whether children with UCBED differ from the general population with respect to PROMIS outcomes. We hypothesized that children with UCBED report mild impairment in upper extremity function but normal mobility, pain interference and peer relations.
Methods:
A retrospective chart review of children aged 5 to 17 years with a diagnosis of UCBED who completed a PROMIS questionnaire at their clinic visit at the [blinded locations] was conducted between April 1, 2017 and March 31, 2020. The mean PROMIS scores of UCBED patients were compared with that of the general reference population. Mann Whitney and ANOVA tests were used to explore the differences across the PROMIS upper extremity function domain by arm length and prosthesis use.
Results:
Fifty-five children (28 boys) with a mean age of 11±3.6 years met the inclusion criteria. Children with UCBED had similar PROMIS scores as the reference population in mobility (51.9±6.2), peer relations (53.5±9.4), and pain interference (40.1±7.2), with mild impairment in the upper extremity function (44.3±10.7). Compared with the 8 to 17-year-old cohort, the parent-proxy (5 to 7-year-old group) reported significantly more upper extremity function impairment (31.3±5.9) vs (48.0±8.8) ( P =0.000). The two age groups did not differ with respect to mobility, pain interference and peer relations.
Conclusions:
Our study confirms previous findings that children with UCBED report upper extremity function, peer relationships, pain interference, and mobility, similar to the reference population. In addition, parents of younger children with UCBED report more upper extremity functional impairment than is self-reported by older children with UCBED.
Level Of Evidence:
Prognostic Level III (comparison with reference population).
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