craniosynostosis is a craniofacial abnormality occurring in approximately 1 in 2500 births, and is defined by the premature fusion of at least one cranial suture.1 It is estimated that 8%–30% of craniosynostosis cases occur as part of congenital syndromes—most commonly Apert, Crouzon, and Pfeiffer syndromes.2–5 These children often have other developmental abnormalities including dental abnormalities, syndactyly, hearing loss, structural cardiac abnormalities, and elevated intracranial pressure.6–8 On the other hand, nonsyndromic craniosynostosis cases have historically been considered isolated events in otherwise healthy children and are resolved by surgical correction of the synostotic suture. However, there is a growing body of literature describing persistent developmental and psychiatric comorbidities after craniosynostosis repair in both syndromic and nonsyndromic patients spanning beyond infancy and into childhood.9–12 Thus, a diagnosis of craniosynostosis can have long-term functional consequences in both syndromic and nonsyndromic cases, suggesting that children with syndromic as well as nonsyndromic craniosynostosis may benefit from developmental, behavioral, and mental health supports. Unfortunately, data on utilization of these services are lacking. We hypothesize that children with any form of craniosynostosis use healthcare services at higher rates than controls. Using a nationwide database, we analyzed use of mental health care services, rehabilitation therapies, and other medical specialties in children who underwent surgery for repair of craniosynostosis and compared them with healthy controls and children with plagiocephaly.
Methods
Data Source
All data were derived from the Optum Clinformatics Data Mart database (Optum, Inc.). This nationwide administrative claims database provides de-identified, longitudinal data for 77 million enrollees of a major insurance provider from 2003 to 2020. In this database, diagnoses are identified using the International Classification of Diseases, Ninth Revision (ICD-9) and the International Classification of Diseases, Tenth Revision (ICD-10), and procedures are identified using Current Procedural Terminology (CPT) codes.
Cohort Definition
The craniosynostosis cohort was defined with a combination of ICD-9, ICD-10, and CPT codes. To meet criteria for the cohort, children must have had the ICD-9 code of 756.0 or the ICD-10 code of Q75.0 and any of the following CPT codes: 61550, 61552, 61556, 61557, 61558, 61559, 21172, 21175, 21179, 21180, 21230, 21299, 64999 (Supplementary Table 1). Children must have had a surgical procedure for craniosynostosis before the age of 3 years and must have had at least 60 days of insurance coverage after surgical correction. This cohort was further subdivided into syndromic or nonsyndromic craniosynostosis. The syndromic subgroup was determined by the presence of a multisuture CPT code (61552 or 61558) or the presence of ICD codes for Crouzon, Apert, Pfeiffer, Muenke, Saethre-Chotzen, and Carpenter syndromes. Children without the presence of a multisuture CPT code or a syndromic ICD code were in the nonsyndromic cohort (Fig. 1). The plagiocephaly group was defined by the presence of either ICD-9 code 754.0 or ICD-10 code Q67.3 without any of the craniosynostosis ICD or CPT codes. Children with positional plagiocephaly were included as a comparison group in this study because they have an abnormal head shape without a synostotic suture and do not require surgical correction.13
Craniosynostosis cohort definition diagram. *ICD codes include Crouzon, Apert, Pfeiffer, Carpenter, Muenke, and Saethre-Chotzen syndromes.
Last, the healthy control cohort was determined based on at least one well-child visit to a pediatrician or family medicine physician before the age of 3 years without any of the craniosynostosis or plagiocephaly ICD or CPT codes. Both plagiocephaly and healthy control cohorts were required to have at least 60 days of insurance coverage after the diagnosis of plagiocephaly or the first pediatrician or family medicine physician visit, respectively.
Study Design and Statistical Analysis
The primary outcome of interest was utilization (claims) of mental health care, rehabilitation therapies, and other medical services that support development in children with craniosynostosis after surgical correction (Supplementary Table 2). Mental health care included any service provided by a psychiatrist, psychologist, licensed clinical social worker, or other mental health care practitioner. Rehabilitation therapies included occupational therapy (OT), physical therapy (PT), and speech–language pathology (SLP). Last, other medical services of interest included visits to developmental pediatricians, optometrists, ophthalmologists, and audiologists. Utilization was defined as at least one visit to the provider after surgery and before the age of 6 years.
Continuous variables and categorical variables were compared using Wilcoxon rank-sum tests, ANOVA tests, Fisher’s exact tests, and chi-square tests for independence. For all tests, p values < 0.05 were considered statistically significant. All statistical analyses were done using Stata version 16 (StataCorp LLC);14 graphs were constructed using GraphPad Prism version 9.15 This study was approved by the institutional review board.
Results
A total of 4166 children were included in our study. Of these, 1340 met our criteria for the craniosynostosis cohort (1140 nonsyndromic, 200 syndromic), 1249 for the plagiocephaly cohort, and 1577 were healthy controls. Characteristics of the cohorts can be found in Table 1.
Characteristics in patient and control cohorts
| Syndromic Craniosynostosis | Nonsyndromic Craniosynostosis | Plagiocephaly | Healthy Controls | |
| No. of pts | 200 | 1140 | 1249 | 1577 |
| % male | 62% | 66% | 60% | 60% |
| Age in yrs at index surgery (mean) | 0.60 | 0.63 | NA | NA |
| Race & ethnicity | ||||
| White | 104 (52%) | 622 (55%) | 893 (71%) | 752 (48%) |
| Black | 6 (3%) | 56 (5%) | 53 (4%) | 66 (4%) |
| Hispanic | 17 (9%) | 79 (7%) | 134 (11%) | 109 (7%) |
| Asian | 7 (4%) | 36 (3%) | 124 (10%) | 76 (5%) |
| Unknown | 66 (33%) | 347 (30%) | 45 (4%) | 574 (36%) |
| Household income | ||||
| >$40K | 6 (3%) | 57 (5%) | 61 (5%) | 78 (5%) |
| $40K–$49K | 3 (2%) | 34 (3%) | 45 (4%) | 42 (3%) |
| $50K–$59K | 6 (3%) | 24 (2%) | 54 (4%) | 49 (3%) |
| $60K–$74K | 9 (5%) | 64 (6%) | 84 (7%) | 67 (4%) |
| $75K–$99K | 14 (7%) | 102 (9%) | 125 (10%) | 130 (8%) |
| ≥$100K | 72 (36%) | 369 (32%) | 656 (53%) | 502 (32%) |
| Unknown | 90 (45%) | 490 (43%) | 224 (18%) | 709 (45%) |
| Yrs w/ insurance coverage (mean) | 3.6 | 3.2 | 3.4 | 3.4 |
NA = not applicable; pts = patients.
Of the 1340 children with craniosynostosis, 108 (8.1%) used mental health care services at least once before 6 years of age (Table 2). Eleven percent of children with syndromic craniosynostosis and 7.5% of children with nonsyndromic craniosynostosis used mental health care services (p = 0.1198). Collectively, children with craniosynostosis used mental health care services at a significantly higher rate than both the plagiocephaly (5.0%, p = 0.0020) and healthy control (2.9%, p < 0.0001) cohorts (Table 3).
Number of patients using mental health care, rehabilitation therapies, and other medical services at least once before the age of 6 years
| Service | All Craniosynostosis | Syndromic | Nonsyndromic | Plagiocephaly | Healthy Controls |
|---|---|---|---|---|---|
| No. of pts | 1340 | 200 | 1140 | 1249 | 1577 |
| Mental health care | 108 (8.1%) | 22 (11.0%) | 86 (7.5%) | 63 (5.0%) | 45 (2.9%) |
| Rehabilitation therapies | 150 (11.2%) | 32 (16.0%) | 118 (10.4%) | 176 (14.1%) | 61 (3.9%) |
| OT | 32 (2.4%) | 4 (2.0%) | 28 (2.5%) | 29 (2.3%) | 13 (0.8%) |
| PT | 111 (8.3%) | 27 (13.5%) | 84 (7.4%) | 138 (11.0%) | 40 (2.5%) |
| SLP | 52 (3.9%) | 12 (6.0%) | 40 (3.5%) | 37 (3.0%) | 16 (1.0%) |
| Other medical services | 497 (37.0%) | 76 (38.0%) | 421 (36.9%) | 261 (20.9%) | 238 (15.1%) |
| Developmental pediatrics | 22 (1.6%) | 3 (1.5%) | 19 (1.7%) | 25 (2.0%) | 7 (0.4%) |
| Optometry | 50 (3.7%) | 10 (5.0%) | 40 (3.5%) | 15 (1.2%) | 18 (1.1%) |
| Ophthalmology | 441 (32.9%) | 70 (35.0%) | 371 (32.5%) | 188 (15.1%) | 116 (7.4%) |
| Audiology | 63 (4.7%) | 11 (5.5%) | 52 (4.6%) | 70 (5.6%) | 97 (6.2%) |
| Total | 578 (43.1%) | 87 (43.5%) | 491 (43.1%) | 401 (32.1%) | 297 (18.9%) |
Statistical comparison of utilization patterns between children with syndromic versus nonsyndromic craniosynostosis, all craniosynostosis versus plagiocephaly, and all craniosynostosis versus healthy controls
| Service | Syndromic vs Nonsyndromic | Craniosynostosis vs Plagiocephaly | Craniosynostosis vs Healthy Controls |
|---|---|---|---|
| Mental health care | 0.1198 | 0.0020* | >0.0001* |
| Rehabilitation therapies | 0.0278* | 0.0283* | >0.0001* |
| OT | >0.9999 | >0.9999 | 0.0008* |
| PT | 0.0076* | 0.0194* | >0.0001* |
| SLP | 0.1097 | 0.235 | >0.0001* |
| Other medical services | 0.8119 | >0.0001* | >0.0001* |
| Developmental pediatrics | >0.9999 | 0.5566 | 0.0012* |
| Optometry | 0.3109 | >0.0001* | >0.0001* |
| Ophthalmology | 0.5143 | >0.0001* | >0.0001* |
| Audiology | 0.5859 | 0.3273 | 0.1025 |
| Total | 0.9384 | >0.0001* | >0.0001* |
Indicates statistically significant association at p < 0.05 level.
Rehabilitation therapies (PT, OT, and SLP) were most commonly used by children with syndromic craniosynostosis and children with plagiocephaly, with 16.0% and 14.1% of the cohorts using a service at least once, respectively. In contrast, 10.4% of children with nonsyndromic craniosynostosis and 3.9% of healthy controls used at least one rehabilitation therapy. Among rehabilitation therapies, PT was more frequently used by patients with plagiocephaly and syndromic craniosynostosis (11.0% and 13.5%, respectively). There was no difference in OT and SLP utilization rates among patients with craniosynostosis (p > 0.9999 and p = 0.1097, respectively), and there was no difference between patients with craniosynostosis and plagiocephaly (p > 0.9999 and p = 0.2350, respectively). However, patients with craniosynostosis did use all rehabilitation therapies at a higher rate than healthy controls (11.2% vs 3.9%, respectively; p < 0.0001).
Thirty-seven percent of children with craniosynostosis used other medical subspecialty services, with 38.0% and 36.9% of syndromic and nonsyndromic patients using these services, respectively (p = 0.8119). The utilization rate of other healthcare services was significantly greater in the craniosynostosis cohort than the rates observed in the plagiocephaly (20.9%, p < 0.0001) and healthy control (15.1%, p < 0.0001) groups. Across all 3 cohorts, ophthalmology was the most commonly used service. In the craniosynostosis cohort, nearly one-third (32.9%) of patients visited an ophthalmologist at least once, as compared with 15.1% (p < 0.0001) of children with plagiocephaly and 7.4% (p < 0.0001) of healthy controls. Developmental pediatrics was only used by 1.6% of children with craniosynostosis (1.5% of syndromic patients and 1.7% of nonsyndromic patients, p > 0.9999), which was not significantly different than in children with plagiocephaly (2.0%, p = 0.5566), but was noted to be significantly greater than healthy controls (0.4%, p = 0.0012).
There was no statistically significant difference in utilization of services when comparing utilization rate in children with craniosynostosis by race/ethnicity (Table 4). Mental health care services were used by 9.5% of non-White craniosynostosis patients—including Hispanic, Black, and Asian children—as compared to 8.1% among White children (p = 0.5661). White children used rehabilitation services at a higher rate than their non-White counterparts, with 3.3% of White children using OT, 8.7% using PT, and 4.8% using SLP versus 2.0% of non-White children using OT, 8.5% using PT, and 3.5% using SLP (p = 0.4844, p > 0.9999, and p = 0.5651, respectively). Last, ophthalmology, the most commonly used service, was used by 34.6% of White children versus 34.8% of non-White children (p > 0.9999).
Utilization of healthcare services in children with craniosynostosis before the age of 6 years by race/ethnicity
| Service | White, n = 726 | Non-White (Hispanic, Black, Asian), n = 201 | p Value |
|---|---|---|---|
| Mental health care | 8.1% | 9.5% | 0.5661 |
| OT | 3.3% | 2.0% | 0.4844 |
| PT | 8.7% | 8.5% | >0.9999 |
| SLP | 4.8% | 3.5% | 0.5651 |
| Developmental pediatrics | 1.5% | 3.0% | 0.2288 |
| Optometry | 5.5% | 3.0% | 0.1974 |
| Ophthalmology | 34.6% | 34.8% | >0.9999 |
| Audiology | 5.2% | 5.5% | 0.8597 |
Excludes 413 patients with craniosynostosis who had unknown race/ethnicity.
Similarly, there was no statistically significant difference in utilization of services in children with craniosynostosis by annual household income (Table 5). Children whose families had an annual household income < $40,000 utilized SLP (7.9%), developmental pediatrics (3.2%), ophthalmology (39.7%), and audiology (7.9%) at higher rates than children whose annual household income was $40,000–$99,999 and ≥ $100,000. However, they used mental health services (6.3%), OT (1.6%), PT (6.3%), and optometry (4.8%) at lower rates than their counterparts with higher socioeconomic status (i.e., $40,000–$99,999 and ≥ $100,000 annual household income). Children whose families had annual household incomes exceeding $100,000 used PT more frequently than their counterparts (10.2%), although the difference was not statistically significant (p = 0.6166).
Utilization of healthcare services in children with craniosynostosis before the age of 6 years by annual household income brackets
| Service | >$40K, n = 63 | $40K–$99K, n = 256 | ≥$100K, n = 441 | p Value |
|---|---|---|---|---|
| Mental health care | 6.3% | 10.5% | 10.0% | 0.6018 |
| OT | 1.6% | 2.3% | 3.6% | 0.4978 |
| PT | 6.3% | 9.4% | 10.2% | 0.6166 |
| SLP | 7.9% | 3.5% | 5.2% | 0.3002 |
| Developmental pediatrics | 3.2% | 2.3% | 2.0% | 0.8419 |
| Optometry | 4.8% | 5.1% | 5.4% | 0.9619 |
| Ophthalmology | 39.7% | 32.4% | 37.9% | 0.2945 |
| Audiology | 7.9% | 6.3% | 4.8% | 0.4840 |
Excludes 580 patients with craniosynostosis who had unknown household income.
Among children with craniosynostosis, utilization was not uniform by age. Whereas utilization of ophthalmology services did not differ by age (p = 0.1003), mental health care was most commonly used among older children (p = 0.0107) (Fig. 2). SLP was most commonly used among children with craniosynostosis at the ages of 3 and 4 years (p = 0.0005), and developmental pediatrics was most commonly used at the age of 5 years (p = 0.0030).
Distribution of services utilization by age (in years) in patients with craniosynostosis.
Discussion
In this study, we demonstrate that children with craniosynostosis use healthcare services at higher rates than children with plagiocephaly and healthy controls. Notably, we demonstrate that rates of mental health services, OT, SLP, and other medical services do not differ between patients with syndromic and nonsyndromic craniosynostosis. Mental health care utilization was significantly higher in children with craniosynostosis compared to children with plagiocephaly, which exceeded utilization by healthy controls. PT utilization was significantly higher in the syndromic craniosynostosis and plagiocephaly cohorts compared to the nonsyndromic craniosynostosis and healthy controls, whereas OT and SLP utilization was higher in both craniosynostosis subgroups and plagiocephaly groups compared to healthy controls. Ophthalmology and optometry utilization was significantly higher in the craniosynostosis groups compared with plagiocephaly and healthy controls, whereas developmental pediatrics referrals were higher in craniosynostosis and plagiocephaly groups compared with the healthy control cohort. There was no difference in audiology utilization between craniosynostosis, plagiocephaly, and healthy control groups. Last, among children with craniosynostosis, we found no statistically significant difference in utilization by race or household income.
Mental Health Services
Children with craniosynostosis have been found to have higher rates of psychiatric comorbidities. In a large nationwide Swedish population-based study, children treated for nonsyndromic craniosynostosis showed a statistically significant increase in adjusted hazard ratios for any psychiatric disorder (1.7) and other psychiatric disorders (1.3), which include bipolar disorder, depression, eating disorders, anxiety disorders, substance use disorder, personality disorders, suicide, and suicide attempt.16 Deficits in mental health and self-esteem were also noted among children with syndromic craniosynostosis, as assessed by the Child Health Questionnaire–Parent Form 50.17 In our study, we found that mental health care utilization is nearly 4 times greater in children with syndromic craniosynostosis than in healthy controls and nearly 3 times greater among children with nonsyndromic craniosynostosis as compared to healthy controls, consistent with the observations by both Tillman et al. and Bannink et al.16,17 Increased utilization may also reflect increased evaluation by psychiatrists and psychologists for signs of inattention or hyperactivity, which have been noted in both syndromic and nonsyndromic cases of craniosynostosis,9,16,18,19 as well as the emotional and self-esteem difficulties experienced by these patient cohorts.17
Rehabilitation Therapies
Rehabilitation therapies were most commonly used by patients with syndromic craniosynostosis and plagiocephaly, which was driven by PT utilization. This finding is consistent with management principles of plagiocephaly. In the last decade, results from randomized controlled trials have demonstrated that adding PT is superior to parental counseling on head positioning alone, and that mild disfigurement can be treated with alternate head positioning and PT.13,20–22 With regard to patients with syndromic craniosynostosis, prior studies have demonstrated decreased physical functioning scores (including limitations in movement, coordination, grasping, or playing due to health problems), which may explain the need for physical rehabilitation through PT or OT visits.17 Although the majority of OT claims are for functional therapy, it is possible that OT services are also being billed for postsurgical orthotic helmet fittings and adjustments, as is typically used after endoscopic suturectomies.23,24 Interestingly, rates of SLP utilization did not differ between children with craniosynostosis and those with plagiocephaly. This can be explained by some evidence showing that patients with plagiocephaly exhibit language deficiencies, including limitations in articulation, phonology, and lexicon, similar to their single-suture craniosynostosis counterparts.25
Other Medical Subspecialities
Among other medical services, ophthalmology was the most commonly used service across all 4 cohorts, with utilization ranging from 7.4% in the healthy control group to 35.0% in the syndromic craniosynostosis group (total 32.9% across all patients with craniosynostosis). The fact that nearly one-third of patients with craniosynostosis are using ophthalmological care in the first 6 years of life is consistent with the concern for papilledema secondary to increased intracranial pressure that can occur as part of craniosynostosis.26 Moreover, the craniofacial abnormalities seen in craniosynostosis, including exorbitism and hypertelorism, have been associated with other visual complications such as strabismus, amblyopia, and high refractive errors, which would explain significant utilization beyond optic neuropathy screenings.27,28 Importantly, several studies have demonstrated that patients with nonsyndromic craniosynostosis who require surgical correction are also susceptible to developing strabismus, amblyopia, and high refractive errors compared to their syndromic counterparts, which is consistent with our findings demonstrating similar ophthalmology utilization of syndromic and nonsyndromic patients.29,30
We found increased utilization of developmental pediatrics in craniosynostosis and plagiocephaly cohorts compared to controls. While there is some evidence that developmental attainment improves after corrective surgery for sagittal synostosis,10 some evidence suggests that cognitive and motor disabilities may persist even after surgical correction. In one study, children with sagittal synostosis were shown to have worse performance than healthy controls on language, learning, and memory tasks in neuropsychological testing, especially in tasks requiring consolidation of learning.11 These findings were consistent among nonsyndromic patients broadly, in whom increased rates of abnormalities in speech, behavioral development, and school function have been noted.12
Utilization by Demographics
Prior studies in craniosynostosis have identified an association between non-White race and older age at time of surgical consultation and older age at surgery.31–35 Importantly, though, at least one study has found that once patients had a surgical evaluation, time to surgery did not differ by race, suggesting that delay in referral to a specialist is a key driver of disparities in craniosynostosis.31 Thus, the lack of differences in utilization by race and household income are not surprising, given that our patient sample has already received initial access to specialist care and in many cases may continue to be followed by them. Importantly, all patients in our sample are privately insured, which may mask differences in access to care by socioeconomic factors that have been noted in craniosynostosis and other neurosurgical conditions.34,36–38
Limitations
Although the Optum Clinformatics database offers a large population to study, we are limited by the granularity of the data. With respect to the description of cases of craniosynostosis, we are limited by ICD and CPT codes and are unable to segment our data based on type of suture involvement or repair modality (open vs endoscopic). Additional granularity may help elucidate different utilization patterns, given that there is some evidence suggesting different long-term developmental outcomes (e.g., IQ scores) by suture type.39 Specifically, in our study we defined the syndromic cohort by using the CPT codes for multisuture repairs; however, we understand that nonsyndromic cases may involve multiple sutures. Thus, although our sample may be sensitive, we are limited in specificity. Despite this limitation, our approach resulted in 15% of our craniosynostosis sample categorized as syndromic, which is consistent with estimates of prevalence of syndromic craniosynostosis reported in the literature.4,5
Moreover, CPT code 64999 is a general code for "other procedures in the nervous system" that is not specific to craniosynostosis. Although we have sought to increase the specificity by requiring craniosynostosis-specific ICD codes, technically these may be used for a range of craniofacial and nervous system procedures. Another limitation of this study is that it only includes utilization of healthcare services offered and billed through patients’ primary medical insurance. It excludes any services that may be provided through other venues, including care provided through educational institutions (e.g., SLP through the school district, therapy through an in-school counselor), or through other forms of supplemental insurance (e.g., vision-specific insurance for optometry and ophthalmology visits). Last, as previously stated, this database includes only commercially insured patients; thus, patients covered by the Children’s Health Insurance Plan or other Medicaid services are excluded from this study.
Conclusions
In this study, we demonstrate that more than 40% of children with craniosynostosis repair will use mental health care, rehabilitation therapies, and other healthcare services supporting development. We also demonstrate that utilization of these services does not differ between patients with nonsyndromic and syndromic craniosynostosis. Although the multidisciplinary care approach is well established in syndromic patients, our findings suggest that this care approach can be applied to patients with nonsyndromic cases too, given that this study demonstrates that the body of literature identifying developmental deficits in nonsyndromic cases does indeed manifest itself as increased healthcare utilization. Thus, although craniosynostosis may be considered an isolated surgical event, parents, caregivers, and providers—including but not limited to neurosurgeons, plastic surgeons, and pediatricians—need to be aware of these needs and ensure that care or referrals are being offered to patients longitudinally. Moreover, these findings can serve to guide families on long-term utilization after surgery and may facilitate subsequent research aimed at increasing efficiency and quality of care. Future work should explore risk factors driving utilization, including suture involvement, repair type, and comorbidities.
Disclosures
The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.
Author Contributions
Conception and design: Prolo, Ruiz Colón, Grant. Acquisition of data: Ruiz Colón, Jin. Analysis and interpretation of data: Ruiz Colón, Jin. Drafting the article: Ruiz Colón. Critically revising the article: Prolo, Jin, Grant. Reviewed submitted version of manuscript: all authors. Approved the final version of the manuscript on behalf of all authors: Prolo. Statistical analysis: Ruiz Colón. Study supervision: Prolo.
Supplemental Information
Previous Presentations
A portion of the results described in Tables 2 and 3 and Fig. 2 were presented at the CNS Annual Meeting in Austin, TX, October 19, 2021. The contents of this manuscript have not been previously published.
Online-Only Content
Supplemental material is available with the online version of the article.
Supplementary Tables 1 and 2. https://thejns.org/doi/suppl/10.3171/2022.2.PEDS2253.
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