Barriers to Upper Extremity Reconstruction for Patients With Cerebral Palsy

Abstract

Background

Reconstructive surgery for upper extremity manifestations of cerebral palsy (CP) has been demonstrated to be safe and effective, yet many potential candidates are never evaluated for surgery. The purpose of this study was to determine barriers to upper extremity reconstruction for patients with CP in a cohort of upper extremity surgeons and nonsurgeons.

Methods

We sent a questionnaire to 4167 surgeons and nonsurgeon physicians, aggregated responses, and analyzed for differences in perceptions regarding surgical efficacy, patient candidacy for surgery, compliance with rehabilitation, remuneration, complexity of care, and physician comfort providing care.

Results

Surgeons and nonsurgeons did not agree on the literature support of surgical efficacy (73% vs 35% agree or strongly agree, respectively). Both surgeons and nonsurgeons felt that many potential candidates exist, yet there was variability in their confidence in identifying them. Most surgeons (59%) and nonsurgeons (61%) felt comfortable performing surgery and directing the associated rehabilitation, respectively. Neither group reported that patient compliance, access to rehabilitation services, and available financial resources were a major barrier, but surgeons were more likely than nonsurgeons to feel that remuneration for services was inadequate (37% vs 13%). Both groups agreed that surgical treatments are complex and should be performed in the setting of a multidisciplinary team.

Conclusions

Surgeons and nonsurgeons differ in their views regarding upper extremity reconstructive surgery for CP. Barriers to reconstruction may be addressed by performing higher level research, implementing multispecialty educational outreach, developing objective referral criteria, increasing surgical remuneration, improving access to trained upper extremity surgeons, and implementing multidisciplinary CP clinics.

Background

Cerebral palsy (CP) is an irreversible, nonprogressive upper motor neuron disorder that manifests with variable deficits in upper and lower extremity function. ¹ Cerebral palsy occurs in 1 of 500 births, making it one of the most common causes of childhood physical disability. ² In 2017, the inflation-adjusted lifetime economic cost per child born with CP was estimated to be $1.3 million. As such, preventing and treating the manifestations of CP have substantial economic implications.^3-5

In patients with CP, the primary insult to the central nervous system frequently results in secondary manifestations in the limb, such as spasticity, dystonia, and weakness. Over time, these imbalances in muscle tone and control can lead to joint contractures, muscle contractures/fibrosis, and osteopenia. Upper limb postural abnormalities often include internal rotation of the shoulder, flexion of the elbow, forearm pronation, flexed wrist and fingers, intrinsic spasticity, and thumb-in-palm deformity. The associated disability secondary to pain, decreased function, and poor ability to perform self-care result in a decreased quality of life. ⁶ Surgery, therefore, offers the possibility for improving the patient’s quality of life and decreasing the economic burden of the disease.

Surgical treatment for upper extremity manifestations of CP was described well over half a century ago ⁷ and includes arthrodesis, ⁸ tendon lengthening,^9,10 selective neurectomy, ¹¹ and tendon transfers. ¹² The majority of research supports improved function after surgery to treat thumb-in-palm,^12-17 wrist,^8,12,18-20 forearm,^12,15,18 and elbow^21-23 deformities associated with CP. ²⁴ In many patients, however, there is an improvement in upper extremity posture only, without a clear gain in function.^22,25 Functional improvements are greatest in patients with voluntary muscle control^17,26 and less baseline functional impairment. ²⁷ Surgery has also been shown to improve hygiene, ²⁸ enhance appearance,^19,28 and results in a high rate of patient satisfaction.^8,12

Despite these data outlining benefits of surgery, experience shows that a limited number of potential candidates are evaluated for reconstruction. In this study, we sought to identify perceptions of surgical management for patients with upper extremity manifestations of CP to uncover perceived barriers to surgical evaluation. We hypothesized that there would be differences between surgeons’ and nonsurgeons’ beliefs about surgical efficacy, knowledge of surgical candidacy, beliefs regarding financial and other resources available to support surgery, complexity of surgery and recovery, and comfort providing care.

Materials and Methods

We employed a cross-sectional study strategy surveying physicians who treat patients with CP. Institutional review board approval was obtained, and participants provided informed consent. Physicians were divided into surgeon and nonsurgeon cohorts based on self-identification. Nonsurgeons were identified through the American Academy for CP and Developmental Medicine (AACPDM) member directory as members with a Doctor of Medicine (MD) or Doctor of Osteopathic Medicine (DO) degree who categorized their specialty as physical medicine and rehabilitation, neurology, pediatric neurology, pediatrics, neurodevelopmental disabilities, and developmental and behavioral pediatrics. Surgeons were identified through the membership directories of the American Society for Surgery of the Hand (ASSH) or the Pediatric Orthopaedic Society of North America (POSNA); any duplicates were removed.

A survey instrument was developed in consultation with a survey research specialist and national experts on the upper extremity manifestations of CP (Supplemental Table 1). Responses were based on a 5-point Likert scale ranging from strongly disagree to strongly agree. A think-out-loud protocol was performed and recorded with 5 test subjects to confirm survey face validity. The survey also included demographic information with categorical responses, a free-text-response question about the greatest barrier to obtaining surgery, and a free-text-response question about the greatest benefit of surgery.

An email containing a cover letter highlighting the informed consent process was sent to each eligible participant along with a unique, deidentified hyperlink to the survey. A single reminder email was sent 2 weeks later to each participant who did not initially respond or indicate they did not want to participate. Participants were given a total of 1 month to respond to the survey. The survey and data were administered and stored on a secure, anonymous, Web-based Research Electronic Data Capture (REDCap) application.

We used χ² and Fisher exact tests to evaluate the differences between categorical variables and the Student t test to evaluate the difference between the continuous variables. Free-text responses related to the greatest barrier and benefit of reconstructive surgery were reviewed by the study team, categorized in a blinded fashion, and analyzed as a categorical variable. In the minority of respondents who listed multiple categories in the free-text response, the first one listed was prioritized and used for categorization. The categories for the greatest benefit included cosmetic improvement, enhanced extremity function, improved patient hygiene, decreased pain/increased comfort, improved psychological well-being, no improvements, or an improvement that does not fit into any other category. The categories for the greatest barrier included patient access to therapy or medical resources, complexity inherent to the patient disease, not knowing which patients would benefit from a referral or to whom to refer, limited financial resources, technical limitations of surgery, social support system, and access to a trained surgeon.

Results

One hundred forty-five (5%) ASSH, 55 (8%) POSNA, and 63 (18%) AACPDM members completed the questionnaire, and the vast majority of responders were physicians who treated patients with CP (Table 1). Post hoc examination for differences between ASSH and POSNA responding surgeons found similar demographics (Supplemental Table 2), with the notable exception that surgeons responding from ASSH treated fewer patients with CP annually than those responding from POSNA (eg, 9.7% vs 96.4% treated >20 patients/year, respectively; P < .001). Ultimately, there were 200 surgeons and 63 nonsurgeons who participated in the survey. All respondents from POSNA and ASSH self-identified as surgeons, whereas all respondents from AACPDM self-identified as nonsurgeons. The average age (±SD) was not statistically different between surgeons (52.9 ± 11.3) and nonsurgeons (52.0 ± 10.8) (Table 1). Surgeons were more likely to be men (P < .001), and nonsurgeons treated more patients with CP per year (P = .040). Seventy-one percent of ASSH respondents and 48% of POSNA respondents state that they perform upper extremity reconstructive surgery for patients with CP.

Table 1.

Physician Characteristics.

Characteristic	Surgeons, no. (%)	Nonsurgeons, no. (%)	P value
Respondents	200	63
Age (SD)	52.9 (11.3)	52.0 (10.8)	.230
Gender			<.001
Male	154 (77.0)	26 (41.3)
Female	33 (16.5)	31 (49.2)
Other	1 (0.5)	0 (0.0)
Race			.240
White	166 (83.0)	47 (74.6)
Black or African American	1 (0.5)	2 (3.2)
American Indian or Alaska Native	1 (0.5)	0 (0.0)
Asian	10 (5.0)	6 (9.5)
Native Hawaiian or Other Pacific Islander	0 (0.0)	0 (0.0)
Other	13 (6.5)	5 (7.9)
Geographic region			.541
Northeast	36 (18.0)	7 (11.1)
Midwest (North Central)	48 (24.0)	21 (33.3)
South	58 (29.0)	19 (30.2)
West	38 (19.0)	11 (17.5)
Years in practice			.457
0-4	17 (8.5)	5 (7.9)
5-9	25 (12.5)	12 (19.0)
10-14	21 (10.5)	8 (12.7)
15-19	25 (12.5)	11 (17.5)
20-24	37 (18.5)	7 (11.1)
≥25	75 (37.5)	20 (31.7)
Number of patients with CP treated annually			.040
0	37 (18.5)	1 (1.6)
1-5	20 (10.0)	0 (0.0)
6-10	25 (12.5)	1 (1.6)
11-20	14 (7.0)	2 (3.2)
>20	67 (33.5)	59 (93.7)

Note. Categories may not add to 100% for questions where 1 or more participants chose not to respond. CP = cerebral palsy.

Surgeons were more likely than nonsurgeons to feel that the literature supports the efficacy of surgery (73% vs 35% agree or strongly agree, respectively; P < .001). Surgeons were more likely to feeel that the functional benefits were worth the risk (77% vs 38%, P < .001) and would want surgery if they had CP (68% vs 37%, P < .001; Figure 1).

Figure 1.

Physician perspectives on effectiveness. Physician opinions on the effectiveness of upper extremity reconstructive surgery for patients with cerebral palsy.

Fewer surgeons than nonsurgeons felt that patients with CP were generally poor candidates for surgery (4% vs 16%, P = .006; Figure 2). The majority of each group felt they know who were good candidates for surgery (56% vs 61%, P = 0.547), but there was a wide range in confidence regarding their ability to identify candidates.

Figure 2.

Physician perspectives on surgical candidacy. Physician perspectives on candidacy of patients with cerebral palsy for upper extremity reconstruction.

Surgeons were more likely than nonsurgeons to feel that patients with CP had difficulty complying with treatment (21% vs 8%, P = .049; Figure 3) and lack the necessary social support to complete the postoperative regimen (23% vs 16%, P = .043). There was no difference between groups regarding their beliefs about access to clinical resources for postoperative rehabilitation (60% vs 70%, P = .483).

Figure 3.

Physician perspectives on patient compliance and financial resources. Physician appraisal of ability of patients with cerebral palsy to adhere to treatment recommendations, and physician assessment of the financial resources available for treating patients with cerebral palsy.

Few surgeons and nonsurgeons felt that insurance does not cover upper extremity reconstructive surgery (6% vs 3%, P = .983; Figure 3), but more surgeons felt that the costs associated with upper extremity reconstructive surgery were not adequately reimbursed (37% vs 13%, P < .001).

Most surgeons and nonsurgeons felt that surgery was complex (61% vs 68%, P = 0.599; Figure 4), surgery should be performed under the direction of a multidisciplinary team (55% vs 94%, P < .001), and individuals with CP are challenging patients to treat (71% vs 58%, P < .001). Seventy-eight percent of surgeons and 81% of nonsurgeons did not feel that the procedures were lengthy (P = 0.035). Most surgeons (61%) were comfortable performing surgery, and most nonsurgeons were comfortable managing the postoperative rehabilitation after surgery (59%; Figure 4). The greatest perceived barrier was similarly distributed among surgeons and nonsurgeons, but the greatest benefit differed between the 2 groups (P = .049; Table 2).

Figure 4.

Physician perspectives on complexity and physician comfort. Physician opinions on the complexity of upper extremity reconstructive surgery, and physician perceived comfort with treating patients with cerebral palsy.

Table 2.

Greatest Benefits and Barriers for Upper Extremity Reconstructive Surgery.

Category	Surgeons	Nonsurgeons	P value
Greatest benefit (n)	(119)	(47)	.049
Cosmetic	8%	2%
Functional	78%	77%
Hygiene	3%	11%
None	0%	2%
Decreased pain/increased comfort	3%	6%
Other	3%	2%
Psychological	6%	0%
Greatest barrier (n)	(125)	(48)	.154
Patient access to medical resources or therapy	15%	19%
Disease or patient complexity	6%	4%
Knowledge of referring, outcomes, or candidates	41%	29%
Financial resources	6%	0%
Surgery-related factors	9%	13%
Social issues	8%	6%
Access to trained surgeon	14%	29%

Discussion

Specialty-specific differences in opinions regarding the efficacy of hand surgery for a variety of conditions have been previously reported.^29,30 In this study, we found many differences in perspectives regarding surgical management of upper extremity manifestations of CP. Notably, most surgeons felt that the literature supports surgical efficacy, the benefits are worth the risks, and they would want surgery if they had CP, whereas most nonsurgeons did not. The differences in beliefs between surgeons and nonsurgeons regarding the risk/benefit ratio and efficacy of surgery may contribute to the seemingly limited referrals for surgical evaluation. One potential method to increase the rate of appropriate referrals is by increasing the level of evidence in future research beyond single surgeon/institution case reviews and disseminating this information among all relevant providers. Of the previously cited studies evaluating the efficacy of upper extremity surgery for CP, only 1 study ¹² contains prospective, randomized data. Given that most nonsurgeons expressed uncertainty regarding literature support for reconstruction, providing relevant outcomes data may address any knowledge gaps and result in surgical consultation for many patients who may benefit from reconstruction.

Both study groups reported that more patients should undergo reconstruction, and sufficient resources were available for postoperative rehabilitation, although nonsurgeons were more likely to characterize patients with CP as poor surgical candidates. Interestingly, there was wide variation in both groups regarding confidence in identifying patients who would be good candidates for surgery, and knowledge of who would be a good candidate was the barrier most commonly cited in the free-text entry of the survey in both study groups. This disparity could be addressed by developing objective criteria that surgeons and nonsurgeons alike can use to select whom to refer for surgical evaluation.

We discovered disparities between groups regarding perceived financial barriers to reconstruction. Although both groups felt that insurance covers upper extremity reconstructive surgery, more surgeons listed being uninsured as a barrier or the greatest barrier to surgery. The majority in each group were not sure whether reimbursement was adequate, and surgeons were nearly 3 times as likely to feel that reimbursement was inadequate. Given that most surgeons responded that patients with CP are challenging to treat and surgery is complex, it is not surprising that some surgeons may feel inadequately compensated for their efforts. As a result, surgeons may choose to not treat patients with CP, which decreases access to a qualified surgeon. This may explain why 29% of nonsurgeons felt that access to a trained surgeon was the greatest barrier to reconstructive surgery. Based on these findings, one way to increase access to a trained surgeon is to increase financial remuneration for performing these procedures; this would address one potential concern identified by a significant minority of surgeons.

Most surgeons and nonsurgeons feel that patients with CP are challenging to treat, reconstructive surgery is complex, and surgery should only be performed under the direction of a multidisciplinary team, although there were group differences. For example, nearly all (94%) nonsurgeons felt that a multidisciplinary team is a necessary prerequisite to reconstructive surgery compared with only 55% of surgeons. Given the complex milieu in which these patients live and are cared for, our team recommends a multidisciplinary approach to evaluation, medical management, and shared decision-making regarding surgery. ³¹ The multidisciplinary approach can also provide an opportunity to offer both surgical and nonsurgical options, such as botulinum toxin A, in an effort to optimize outcomes.

This study has several limitations, most notably related to survey biases. On the surface, the study appears to have a low response rate. Experience shows that there are relatively few surgeons who currently perform surgeries for upper extremity manifestations of CP. The study finding “access to a trained surgeon” as a barrier corroborates this. Therefore, we believe that there was a far greater response rate among surgeons who treat upper extremity manifestations of CP. Another limitation is that we did not define physician specialty with more specificity than “surgeon” or “nonsurgeon.” This broad classification, for example, did not allow for the assessment of differences in perceptions between medical providers who treat patients with CP.

Nevertheless, our study uncovered several barriers to potentially beneficial surgical reconstruction for patients with upper extremity manifestations of CP. Based on the results of this study, an action plan to eliminate these barriers includes performing higher level research and educating nonsurgeons about the outcomes of upper extremity reconstructive surgery for patients with CP, developing objective referral criteria, practicing in the setting of a multidisciplinary team, and increasing compensation for surgical management in an effort to increase access to qualified surgeons. Overall, our data support the concept that the lack of coordinated cross-specialty relationships is the greatest barrier to utilization of upper extremity reconstructive surgery. ²⁹