De f o r m at i o n a l or positional plagiocephaly is the

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1 J Neurosurg Pediatrics 5: , 5: , 2010 Comparison of perceptions and treatment practices between neurosurgeons and plastic surgeons for infants with deformational plagiocephaly Clinical article Am y Le e, M.D., 1 And r e a E. Va n Pe lt, M.D., 2 Al e x A. Ka n e, M.D., 2 Th o m a s K. Pi l g r a m, Ph.D., 3 Da n i e l P. Go v i e r, 2 Al b e r t S. Wo o, M.D., 2 a n d Ma t t h e w D. Sm y t h, M.D. 1 Departments of 1 Neurosurgery, 2 Plastic and Reconstructive Surgery, and 3 Radiology, Washington University School of Medicine and St. Louis Children s Hospital, Saint Louis, Missouri Object. Deformational plagiocephaly (DP) is the leading cause of head shape abnormalities in infants. Treatment options include conservative measures and cranial molding. Pediatric neurosurgeons and craniofacial plastic surgeons have yet to agree on an ideal therapy, and no definable standards exist for initiating treatment with helmets. Furthermore, there may be differences between specialties in their perceptions of DP severity and need for helmet therapy. Methods. Requests to participate in a web-based questionnaire were sent to diplomates of the American Board of Pediatric Neurological Surgery and US and Canadian members of the Pediatric Joint Section of the American Association of Neurological Surgeons and the Congress of Neurological Surgeons and the American Cleft Palate Craniofacial Association. Questions focused on educational background; practice setting; volume of DP patients; preferences for evaluation, treatment, follow-up; and incentives or deterrents to treat with helmet therapy. Six examples of varying degrees of DP were presented to delineate treatment preferences. Results. Requests were sent to 302 neurosurgeons and 470 plastic surgeons, and responses were received from 71 neurosurgeons (24%) and 64 plastic surgeons (14%). The following responses represented the greatest variations between specialties: 1) 8% of neurosurgeons and 26% of plastic surgeons strongly agreed with the statement that helmet therapy is more beneficial than conservative therapy (p < 0.01); and 2) 25% of neurosurgeons and 58% of plastic surgeons would treat moderate to severe DP with helmets (p < 0.01). Conclusions. Survey responses suggest that neurosurgeons are less likely to prescribe helmet therapy for DP than plastic surgeons. Parents of children with DP are faced with a costly treatment decision that may be influenced more strongly by referral and physician bias than medical evidence. (DOI: / PEDS0983) Ke y Wo r d s plagiocephaly craniosynostosis deformational plagiocephaly De f o r m at i o n a l or positional plagiocephaly is the leading cause of head shape abnormalities in infants. The incidence has increased steadily since 1992, when the American Academy of Pediatrics released its Back to Sleep campaign, recommending that infants sleep on their backs after multiple studies demonstrated an association between sudden infant death syndrome and prone sleep position. 1,2,7 Deformational plagiocephaly describes a skull-shape deformity caused by repetitive, Abbreviation used in this paper: DP = deformational plagiocephaly. prolonged external pressure on the occipital region. This typically results in right- or left-sided posterior flattening, with ipsilateral frontal bulging and ear displacement, depending on the infant s preferred resting position. Currently, there are 2 primary nonsurgical treatment options for infants with DP: conservative therapy with repositioning and avoidance of continued, prolonged pressure on the flattened side; or cranial remolding therapy with an orthotic helmet, taking advantage of the malleability of the infant cranium and the rapid brain growth that occurs during the first year of life. Both pediatric neurosurgeons and craniofacial pe- 368 J Neurosurg: Pediatrics / Volume 5 / April 2010

2 Treatment practices for infants with deformational plagiocephaly diatric plastic surgeons are faced with an increasing population of infants with DP and the controversy surrounding treatment. There is no standard tool for quantitative assessment of treatment progress or efficacy for DP described in the literature. 9 Additionally, no studies compare outcomes from the 2 approaches with sufficient sample size to provide evidence-based guidelines. 6 From a neurodevelopmental standpoint, studies have yet to establish clear relationships between DP and developmental delays or whether remolding therapy has any benefit on developmental outcomes. 10 Parents of children with DP are faced with a costly treatment decision that may be influenced more strongly by referral and physician bias than by medical evidence. Furthermore, there may be differences between specialties with respect to the perception of DP severity and need for helmet therapy. The purpose of this study was to identify perception and treatment patterns among and between neurosurgeons and craniofacial plastic surgeons regarding DP. Methods Study Design An Internet-based questionnaire was created through a commercial web-based product called Survey Monkey ( Institutional review board approval was obtained to use anonymous physician response data. Requests to participate in the web-based questionnaire were sent by means of messages that included an electronic link to the survey document. The messages were sent to diplomates of the American Board of Pediatric Neurological Surgery and US and Canadian members of the Pediatric Joint Section of the American Association of Neurological Surgeons and the Congress of Neurological Surgeons as well as members of the American Cleft Palate Craniofacial Association (ACPA). They were sent to all identified surgeons for whom addresses were available. Participation was completely voluntary and no penalty was incurred for either refusal to participate or withdrawal from the study. Measurement The Internet-based questionnaire included 30 multiple choice questions (Appendix) focused on the following: specialty training (plastic surgery vs neurosurgery); training background within each specialty (residency only vs additional fellowship training); practice setting (urban, rural, academic, private); volume of DP patients seen; and preference for evaluation, treatment (duration and maximum age for candidacy of helmet therapy, type and cost of helmet used), follow-up (monitoring techniques), and incentives or deterrents to treat with helmet therapy (parental influence and financial background). The survey also contained 6 computer-generated images of infants heads as viewed from above. The images showed varying degrees of DP severity, and for each image, the respondents were asked to indicate whether they would treat the child with a helmet. Increasing severity was gauged by increasing amounts of posterior flattening and ipsilateral frontal bulging. The survey was open for a response period of approximately 2 J Neurosurg: Pediatrics / Volume 5 / April 2010 months. Nonresponders were sent a single reminder request to participate. During the response period, several respondents notified us that the survey did not include options to indicate whether they did not treat patients with helmets at all. Since many questions were based on the premise that helmet therapy was used, the survey was amended to account for this discrepancy. Respondents were then notified to clarify whether they simply did not evaluate infants with DP in their practice or if they did evaluate infants, but had a strict no helmet policy. If they did not prescribe helmets and preferred conservative management, the survey did not explore what respondents deemed to be appropriate conservative therapy. Statistical Analysis Categorical responses, such as background and preferences, were compared by specialty with contingency tables. Patterns were tested for statistical significance with chisquare tests. Measurement data (such as patient volume) were compared by calculating means, and differences were tested with Student t-tests. All analyses were performed using JMP 6.0 (SAS Institute). Results Of the 772 practitioners to whom the requests to participate were ed, 135 (71 [24%] of 302 neurosurgeons and 64 [14%] of 470 plastic surgeons) responded to the questionnaire. Practice backgrounds were found to be similar among all respondents. Most had undergone fellowship training after residency and practiced in urban, academic settings (Fig. 1). With respect to the questions about maximum age for initiating helmet therapy, duration of helmet therapy, parental influence, or parents financial background affecting decisions to prescribe helmets, there was no significant difference between the 2 specialties. However, in response to questions about the cost of helmet therapy, there were differences that suggested that neurosurgeons were less likely than plastic surgeons to recommend helmets when cost was taken into consideration (p < 0.05, chi-square) (Table 1). Fig. 1. Bar graph showing the training and practice backgrounds of respondents. A+P = academic and private; NS = neurosurgeon; PS = plastic surgeon. 369

3 A. Lee et al. TABLE 1: Comparison of responses between neurosurgeons and plastic surgeons Highest Frequency Response Survey Item NS PS Difference Btwn Specialties p Value maximum age to initiate therapy (range 6 mos to 2 yrs)* 12 mos 12 mos not significant 0.33 typical duration of helmet therapy (range 2 mos to 6 mos) 4 mos 4 mos not significant 0.60 extent of parents influence on whether to prescribe helmet great moderate not significant 0.26 influenced by family financial background disagree disagree not significant 0.16 influenced by helmet cost in decision to prescribe agree disagree significant <0.05 * Maximum ages were unintentionally presented with the > symbol in the survey. Chi-square. Difference suggests that neurosurgeons are less likely than plastic surgeons to prescribe helmets when cost is taken into consideration. Other responses that showed notable variation between specialties were as follows: 1) the mean percentage of patients with DP who had helmets prescribed by neurosurgeons was 21% (median 10%) versus 35% (median 25%) for patients of plastic surgeons (p < 0.05, t-test; Fig. 2); 2) 8% of neurosurgeons versus 26% of plastic surgeons strongly agreed that helmet therapy was more beneficial than conservative therapy (Fig. 3); and 3) choices based on sample overhead views of infants heads revealed no difference between treatment practices for mild DP, but plastic surgeons were more likely than neurosurgeons to prescribe helmets in moderate to severe examples (p < 0.05, chi-square; Fig. 4). Also, when evaluating practice Fig. 2. Box and whiskers plots showing the 25th and 75th percentiles (box) and range (whiskers) of the percentage of patients that actually receive helmet therapy by specialty. volumes of those surgeons in both groups who saw more than 200 DP patients per year (10 of 71 neurosurgeons, 14 of 64 plastic surgeons), an increase was seen in the number of helmets prescribed when compared with the number prescribed by those who saw fewer than 200 DP patients per year (mean values 25% for < 200 patients, 43% for > 200 patients; p = 0.009, t-test). There was no difference between neurosurgeons and plastic surgeons regarding the type or brand of helmet prescribed. The most frequent choice was the use of custom helmets manufactured by local orthotists (58% of neurosurgeons, 37% of plastic surgeons). The second most commonly used type of helmet was the Starband Cranial Orthosis (Orthomerica) followed by Hanger Cranial Band (Hanger Orthopedic Group, Inc.). Tools to monitor head shape or progress did not differ significantly between groups and are listed in Table 2. Interestingly, the group mean values for the estimates of the percentage of DP patients seen by neurosurgeons or plastic surgeons at the respondent s institutions were strikingly similar, but the proportion allocated to one or the other specialty depended on the respondent group. Neurosurgeons reported that they saw a mean of 66% of the patients with DP and that plastic surgeons saw 33%, whereas plastic surgeons reported they saw a mean of 67% and that neurosurgeons saw 34%. Discussion The incidence of DP has increased dramatically in recent years and has been temporally associated with the Back to Sleep campaign to combat sudden infant death syndrome. 1,2,7 Craniofacial plastic surgeons and pediatric neurosurgeons have consequently been inundated with an increasing number of visits from parents concerned about their infants asymmetrical head shape. The escalating incidence of DP has sparked controversy over the need to treat these infants and which therapy is most efficacious. Most clinicians agree that surgery is almost never warranted. 4 Conservative treatment options include counterpositioning to prevent prolonged pressure on the flattened side, and passive cranial remolding with an orthotic helmet. Decisions to treat are clouded by parental preferences and physician bias, the lack of standardized tools to 370 J Neurosurg: Pediatrics / Volume 5 / April 2010

4 Treatment practices for infants with deformational plagiocephaly Fig. 3. Bar graph showing responses to the statement: Helmet therapy is more beneficial than conservative management for deformational plagiocephaly. measure outcomes, and the high cost of helmet therapy (~$1500 to $3000). At our own institution, we observed empirically that the overlap in referrals to both specialties did not correlate with similar prescribing trends for helmet therapy. We therefore designed an Internet-based survey to determine if this reflected the general North American trend between pediatric neurosurgeons and craniofacial plastic surgeons. Based on memberships to professional organizations, 772 requests for participation were electronically distributed, yielding a 24% response rate from neurosurgeons and a 14% response rate from plastic surgeons. The primary limitation of this study is the relatively low response rate. Response rates are a common, and increasing, 3 problem in Fig. 4. Overhead-view computer-generated images of varying degrees of DP (mild, moderate, severe) and bar graphs showing physicians responses to being asked whether they would treat these infants with a helmet. Avg = average. J Neurosurg: Pediatrics / Volume 5 / April

5 A. Lee et al. TABLE 2: Monitoring methods used by 71 neurosurgeons and 64 plastic surgeons* Method Neurosurgeons Plastic Surgeons Total clinical evaluation 58 (82) 58 (92) 116 (86) parental observation 45 (63) 34 (53) 9 (59) photographs 16 (23) 37 (58) 53 (39) calipers/cephalometrics 19 (27) 21 (33) 40 (30) 3D surface scans 5 (7) 10 (16) 15 (11) CT 4 (6) 2 (3) 6 (4) plain radiographs 0 (0) 0 (0) * Responses to the question, How do you monitor for change in head shape over time? Values represent the number of surgeons (%) who indicated that they used the method in question. surveys, particularly those that rely on self-administered modes, such as web-based surveys. 5 The drawback of a low response rate is the lack of knowledge about the nonrespondents. Although there is no reason to believe that their clinical approaches differ those of respondents, there is no evidence to prove that they are the same. Even among the respondents, we relied on their recollection rather than hospital records, but the focus was more on attitude than behavior. Still, our results clearly suggest that neurosurgeons and plastic surgeons may have different beliefs, as well as practices, involving the use of helmets for treating DP in infants, and a larger study to explore this issue more rigorously is certainly warranted. Survey results from 71 neurosurgeons and 64 plastic surgeons suggest that there are differences in treatment practices between the 2 specialties, with plastic surgeons more likely to prescribe orthotic helmets, especially in moderate to severe cases. Among respondents, the median values for the percentage of patients receiving helmet therapy were 25% among plastic surgeons and 10% among neurosurgeons. More plastic surgeons than neurosurgeons strongly agreed that helmet therapy was more beneficial than conservative therapy (26 vs 8%). We hypothesize that this difference might be related to the fact that DP is considered predominantly a cosmetic problem rather than a physiological one. Parents do not want their children to be perceived as abnormal and worry about psychosocial implications and teasing by peers. 4 Our survey indicated that both neurosurgeons and plastic surgeons were influenced by parents to at least a moderate extent in treatment decisions. Yet because the functional or neurodevelopmental outcomes in DP have not been firmly established, plastic surgeons may be more sensitive to cosmetic concerns of parents and thus more likely to treat with helmets. One recent study by Kordestani et al. 8 demonstrated an association between DP and mental/psychomotor delay. The authors note, however, that the etiology of delays in each case could be explained by the presence of one or more confounding factors such as prematurity, low birth weight, maternal substance abuse, early illness, or failure to thrive. Infants with DP in the absence of such confounding factors did not have an increased incidence of delays. This questions whether children with existing delays are more prone to DP, or if DP places children at higher risk for developmental delays. 10 If DP does indeed cause developmental delays, does correction of head shape in infancy affect developmental outcomes? Evidence-based longitudinal outcomes studies are needed to substantiate a compelling functional benefit to helmet therapy. Referrals to either neurosurgeons or plastic surgeons for evaluation of DP should be made before 1 year of age. At 1 year old, the width of a child s head is approximately 85% of its adult size, whereas, at 5 years old, the head width increases to more than 90% of its adult size. Similarly, with respect to head circumference, approximately 85% of growth is complete at 1 year of age and 95% by age 5. Therefore, earlier referrals and evaluation will be more beneficial if helmet therapy is initiated because the therapy will take advantage of the rapid growth of the skull. When considering referral, it is also important to differentiate DP from true lambdoid synostosis. Lambdoid synostosis is relatively rare and may have a few morphological similarities with nonsynostotic plagiocephaly or DP, but these 2 entities must be distinguished to select appropriate treatment. Children with lambdoid synostosis will characteristically have a trapezoid-shaped head in association with a posteriorly displaced ear, contralateral occipital bossing, and sometimes ridging of the lambdoid suture. In contrast, DP patients will exhibit a parallelogram-shaped head with anterior displacement of the tragus and ipsilateral frontal bossing. This distinction is critical since lambdoid synostosis may often require surgical intervention, while DP warrants simpler, noninvasive options. In this survey, of those respondents who did use helmets, the most frequently noted duration of therapy was 4 months; nearly all surgeons used a combination of methods for monitoring progress over time. In addition to subjective clinical evaluation and parental observation, the most common adjuncts for monitoring head shape over time were caliper measurements/cephalometrics and photographs. Smaller numbers of respondents relied on 3D surface scanning, and even fewer on CT scans. Multiple centers have developed specialized methods to measure infant head shape in DP, but there are no defined standards for initiating therapy or monitoring change. In situations in which parents and practitioners agree that a helmet would be beneficial, the cost of helmet manufacture (often exceeding $1500) unfortunately forces some families to opt out of therapy. Many insurers will not cover the cost, placing parents in a difficult position. Parents can also be burdened with extensive travel costs for regular follow-up appointments if they do not live near the institution where their child is treated. In our survey, neither group was influenced by the financial background of patients families in deciding whether to recommend helmet therapy. However, neurosurgeons were less likely than plastic surgeons to prescribe helmets when cost was taken into consideration. Conclusions Survey responses support the assumption that the treatment of patients with severe DP differs significantly based on the specialty of the physician. Specifically, neu- 372 J Neurosurg: Pediatrics / Volume 5 / April 2010

6 Treatment practices for infants with deformational plagiocephaly rosurgeons are less likely to prescribe helmet therapy for DP than plastic surgeons. Parents of children with DP are faced with a costly treatment decision that may be influenced more strongly by referral and physician bias than medical evidence. Appendix: Survey Questions Are you a neurosurgeon or plastic surgeon? What is your training background? Check all that apply. For Plastics: Plastic surgery residency only Pediatric/Craniofacial surgery fellowship Other, please specify For Neurosurgery: Neurosurgery residency only Pediatric neurosurgery fellowship Other, please specify How many years have you been in practice post-training? [text box answer, integers only] Which of the following best describes your practice setting? Check all that apply. Urban Rural Solo Group Are you in private practice or affiliated with an academic in stitution? Academic Private Private with academic affiliation (For those who answered academic): In your academic institution, estimate the percentage of deformational plagiocephaly (DP) patients seen by neurosurgery vs plastic surgery. Numbers should total 100. How certain are you about your estimation on the above question? Please give a percent certainty. What percentage of your patient population is pediatric? 0 33% 34 66% > 66% Approximately how many new patients with deformational plagiocephaly (DP) are evaluated per year in your practice? Include patients evaluated by physicians, physician assistants (PAs) and nurse practitioners (NPs) > 200 If you simply do not evaluate infants with DP at all, please click in the circle below and exit the survey. Otherwise, please proceed. What percentage of your patients with deformational plagiocephaly actually receives helmet therapy? (answer in %, integer only) To what extent do parents desires influence your decision to prescribe helmets? to a great extent to a moderate extent a little not at all undecided In your opinion, at what maximum age is a patient no longer a candidate for initiating helmet therapy, e.g. no longer likely to achieve maximum benefit from cranial remolding? > 6 mos > 7 mos J Neurosurg: Pediatrics / Volume 5 / April 2010 > 8 mos > 10 mos > 12 mos > 18 mos > 2 years In your experience, what is the typical duration of helmet therapy? 2 months 3 months 4 months 5 months 6 months or longer How many helmets per patient are typically required (e.g. due to growth) through duration of therapy? After helmet therapy is prescribed, who follows patients progress and how often are they evaluated? Check all that apply. You (physician) PA or NP Orthotist Pediatrician Other: please specify [Respondents were asked to chose from the following responses to indicate timing of evaluation: q 2 wks, q month, q 6 wks, q 2 months, not sure] How do you monitor for change in shape over time? Check all that apply. Caliper measurements/cephalometrics Photographs 3D surface imaging Plain radiographs CT scans Clinical experience/physical exam Parental observation Other: please specify What type of helmet do you use? DOC Band (Cranial Technologies, Inc.) Hanger Cranial Band (Hanger Orthopedic Group, Inc.) Starband Cranial Orthosis (Orthomerica) Custom helmet manufactured by local orthotist Other: please specify How much does the helmet cost? < $500 $501 1,000 $1,001 1,500 $1,501 2,000 $2,001 2,500 > $2,500 don t know What percent of families have to pay for helmet therapy be - cause of insurance denial? 0 25% 26 50% 51 75% % don t know Please indicate your agreement/disagreement with the following statements: The cost of helmets influences your decision to prescribe them. Knowledge of a family s financial background influences your likelihood of recommending helmet therapy. Helmet therapy is more beneficial than conservative management for deformational plagiocephaly. [Respondents were provided with check boxes with the following options: strongly disagree, agree, neither agree nor disagree, disagree, strongly disagree] In which of the following ways do you receive financial benefit by treating patients with deformational plagiocephaly? Check all that apply. Scanning fees 373

7 A. Lee et al. Orthotist in office or one who generates income for your practice Helmet construction Management fees for follow-up visits I receive no financial benefit Other, please specify Based on the following 6 images, would you recommend helmet therapy for these individuals? [The images in Fig. 4 were displayed with with checkboxes for yes, no, and maybe.] Disclosure 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 to the study and manuscript preparation include the following. Conception and design: AA Kane, AS Woo, MD Smyth. Acquisition of data: A Lee, AE Van Pelt. Analysis and interpretation of data: A Lee, AE Van Pelt, TK Pilgram, DP Go - vier. Drafting the article: A Lee, AE Van Pelt. Critically revising the article: A Lee, AE Van Pelt, AA Kane, TK Pilgram, AS Woo, MD Smyth. Reviewed final version of article and approved it for submission: AA Kane, AS Woo, MD Smyth. Statistical analysis: TK Pilgram. Administrative/technical/material support: DP Govier. Study supervision: AA Kane, MD Smyth. Other (figure design): DP Govier. References 1. American Academy of Pediatrics AAP Task Force on Infant Positioning and SIDS: Positioning and SIDS. Pediatrics 89 (6 Pt 1): , Argenta LC, David LR, Wilson JA, Bell WO: An increase in infant cranial deformity with supine sleeping position. J Craniofac Surg 7:5 11, Biemer PP, Lyberg LE: Introduction to Survey Quality. Hobo ken, NJ: John Wiley & Sons, Bruner TW, David LR, Gage HD, Argenta LC: Objective outcome analysis of soft shell helmet therapy in the treatment of deformational plagiocephaly. J Craniofac Surg 15: , de Leeuw E, de Heer W: Trends in household survey nonresponse: a longitudinal and international comparison, in Groves RM, Dillman DA, Eltinge JL, et al (eds): Survey Nonresponse. New York: John Wiley & Sons, 2002, pp Graham JM Jr, Gomez M, Halberg A, Earl DL, Kreutzman JT, Cui J, et al: Management of deformational plagiocephaly: repositioning versus orthotic therapy. J Pediatr 146: , Kane AA, Mitchell LE, Craven KP, Marsh JL: Observations on a recent increase in plagiocephaly without synostosis. Pediatrics 97: , Kordestani RK, Patel S, Bard DE, Gurwitch R, Panchal J: Neurodevelopmental delays in children with deformational plagiocephaly. Plast Reconstr Surg 117: , Loveday BP, de Chalain TB: Active counterpositioning or orthotic device to treat positional plagiocephaly? J Craniofac Surg 12: , Steinbok P, Lam D, Singh S, Mortenson PA, Singhal A: Longterm outcome of infants with positional occipital plagiocephaly. Childs Nerv Syst 23: , 2007 Manuscript submitted February 11, Accepted November 9, Address correspondence to: Amy Lee, M.D., Washington University School of Medicine, Department of Neurosurgery, Campus Box 8057, 660 South Euclid Avenue, St. Louis, Missouri leeam@nsurg.wustl.edu. 374 J Neurosurg: Pediatrics / Volume 5 / April 2010