Pediatric cervical spine disorders and deformities are rare, with nuances specific to each disorder, age group, and the anatomical level affected. Clinical management of these problems is often challenging because clinicians must consider variable and smaller bony anatomy, practicality and compliance with external bracing, technical feasibility of instrumentation, the child’s growth potential, graft material use, long-term arthrodesis rates, bone strength related to congenital conditions, and other related issues. Early empirical evidence regarding surgical outcomes primarily consisted of single-institution series, with surgical techniques focusing on posterior wiring with onlay bone graft and postoperative external orthosis, which often led to notable morbidity, nonunion, and higher construct failure rates.1–4 Throughout the past 2 decades, rigid fixation techniques have overtaken semirigid techniques for stabilization of craniocervical and subaxial cervical instability, with improved outcomes.5–7 However, large, multicenter, or multidisciplinary studies investigating the surgical outcomes, clinical and radiographic criteria for instability, or the nonoperative management of these patients have been very limited.8–11
Originally developed by the RAND Corporation during the Cold War, the Delphi method was first used to predict which American industrial targets might be bombed by the Soviet Union and to determine the quantity of bombs required to damage US munitions.12 Its purpose is to build consensus among groups of experts about topics with a paucity of evidence-based data.12 The process of the Delphi method begins with an initial series of iterative anonymous questionnaires, with experts providing background information and responses to statements using a 4-point Likert scale to generate consensus statements. With this method the skewing effects of direct confrontation, preconceived notions, resistance to new ideas, and defensiveness are minimized, and constructive criticism is optimized. The process culminates in an in-person meeting in which statements that achieved near consensus in prior rounds of questioning can then be discussed, debated, and modified to achieve consensus. Over the last decade, this method has been modified and used to develop consensus statements aimed at minimizing wound complications following complex tethered cord surgery, to minimize surgical site infections after pediatric deformity surgery, to optimize responses to intraoperative neuromonitoring changes during spinal surgery, to manage children requiring cervical spine traction after spinal cord injury, and for other clinical topics.13–17
Stabilization of the pediatric cervical spine is an uncommon procedure with limited consensus regarding the diagnosis of instability, nonoperative management, indications for operative intervention, and determination of successful fusion. This is largely due to the relative rarity of pediatric cervical instability, limiting the rigorous study of patient care in this population. The goal of this study was to bring together an international, multidisciplinary group of pediatric cervical spine experts to build consensus via a modified Delphi approach and to identify areas of equipoise regarding the clinical management of children with cervical spine disorders and those undergoing cervical spine stabilization surgery.
Methods
Focus and Design of Modified Delphi Study
The focus of this study was to query expert opinion regarding practices surrounding the clinical management of children with cervical spine disorders including those with instability requiring stabilization. A systematic literature review was conducted using PubMed/MEDLINE, Google Scholar, Cochrane Reviews, and EMBASE databases to examine the existing evidence supporting the following topics: preoperative planning and radiographic evaluation for patients with cervical spine disorders, intraoperative management, postoperative management with follow-up care, and nonoperative management. An electronic survey was then generated and distributed to participants to assess general practice patterns. Primary survey responses were collected and analyzed, and potential consensus statements were formulated based on initial responses. A second survey that included these statements was then distributed to participants. Results of the second survey were collected and statements were modified and revised. A hybrid meeting (both in-person and virtual) was then convened to foster discussion and provide an opportunity to revise statements that reached near consensus (70%–79% agreement) or were of particular interest to the group. A final document including consensus statements was then generated.
Participants
Thirteen pediatric neurosurgeons and 4 pediatric orthopedic surgeons from major children’s hospitals in the US, United Kingdom, and India participated. Participants had significant experience managing children with cervical spine disorders (defined as fellowship training in pediatric neurosurgery, pediatric orthopedics, and/or spine; more than 5 years in clinical practice; history of relevant publications; and a national/international reputation) and were selected based on prior collaboration, clinical experience, and volume; prior research into pediatric cervical pathologies; and membership in academic pediatric neurosurgical, orthopedic, and spine organizations.
Initial Survey
A 98-item online survey (SurveyMonkey) was distributed to participating surgeons in November 2021. The survey was divided into 5 sections, with initial questions obtaining background information on the participants, followed by sections detailing preoperative clinical and radiographic evaluation and planning, intraoperative management, postoperative management, and nonoperative management. Using a 3-point Likert scale (very willing, somewhat willing, not willing), participants were also asked how willing they would be to change their practices based on the results of the current study.
Delphi Round 1
Using results of the initial survey, 52 potential consensus statements were generated (Supplementary Table). Open-ended questions from the initial survey were adapted and modified into statement form and a 4-point Likert scale (strongly agree, agree, disagree, strongly disagree) was used. Whereas the language and structure of statements were altered, the clinical management topics were unchanged. The survey was electronically distributed in November 2021, and anonymous responses were collected. Responses achieving ≥ 80% total agreement (strongly agree plus agree responses) or total disagreement (disagree plus strongly disagree responses) were considered to have reached consensus. Responses achieving near consensus (70%–79%) were the primary focus of discussion at the in-person meeting (round 2).
Delphi Round 2
An in-person meeting was held in December 2021, with both in-person and virtual participation due to the SARS-CoV-2 pandemic. An anonymous audience response system (Poll Everywhere) was used in which participants entered their responses to statements using their personal smartphone or computer anonymously. Responses were tallied and projected in real time to all participants. Statements that were near consensus were discussed in detail, and modifications to the statements were made to try to achieve consensus. Finalized statements achieving ≥ 80% total consensus were then incorporated into a consensus statement of proposed best practices. At the conclusion of the discussion, participants were asked whether they would 1) agree to the publication of these consensus statements; 2) implement the consensus statements into their practice; and 3) be willing to participate in multicenter prospective studies to investigate the efficacy of the implementation of the statements. All initial participants (including those unable to attend the in-person meeting) reviewed and approved the final consensus statements and manuscript prior to publication.
Results
Participant Characteristics
Of the 17 individuals invited to participate in the study, 100% completed the initial survey of current practices. Table 1 describes the participants’ demographics and clinical experience and Table 2 displays the case volumes by procedure type. The most common pathologies requiring stabilization were syndromic/genetic, followed by trauma, purely ligamentous instability, basilar invagination, neoplastic, and infectious pathologies, respectively.
Demographics of participants in survey
| Characteristic | No. (%) |
|---|---|
| No. of participants | 17 |
| Neurosurgery trained | 13 (76.5%) |
| Orthopedic trained | 4 (23.5%) |
| Geographic distribution | |
| US | 15 |
| United Kingdom | 1 |
| India | 1 |
| Clinical experience | |
| >20 yrs | 5 (29%) |
| 16–20 yrs | 4 (24%) |
| 11–15 yrs | 5 (29%) |
| 6–10 yrs | 3 (18%) |
Participant case volume by procedure type
| Stabilization Procedure | No. (%) |
|---|---|
| Oc–C1 | |
| ≤5/yr | 8 (47%) |
| 6–10/yr | 5 (29%) |
| 11–15/yr | 3 (18%) |
| 16–20/yr | 1 (6%) |
| C1–2 | |
| ≤5/yr | 10 (59%) |
| 6–10/yr | 5 (29%) |
| 11–15/yr | 2 (12%) |
| Subaxial | |
| ≤5/yr | 8 (47%) |
| 6–10/yr | 6 (35%) |
| 11–15/yr | 1 (6%) |
| 16–20/yr | 2 (12%) |
| Anterior | |
| ≤5/yr | 12 (70%) |
| 6–10/yr | 3 (18%) |
| 11–15/yr | 2 (12%) |
Delphi Method
The initial round of 52 proposed consensus statements (Supplementary Table) resulted in ≥ 80% total agreement for 41 statements, of which 14 reached 100% consensus (Table 3). Eleven statements failed to reach consensus; however, 9 reached near consensus in agreement (70%–79% strongly agreed or agreed) (Table 4). Of the 9 statements, 7 were discussed at the in-person meeting and 4 reached consensus after modifications (Table 5, italicized statements in Table 3), yielding a total of 45 statements reaching final consensus.
Statements that reached consensus regarding the management of children with cervical spine disorders and stabilization*
| Statement | Response (%) | ||
|---|---|---|---|
| Total Consensus | Strongly Agree | Agree | |
| A. Preoperative planning for pediatric patients requiring posterior cervical instrumentation | |||
| 1. In children with cervical spine instability, static radiographs of the cervical spine as part of the preoperative workup are recommended. | 88 | 59 | 29 |
| 2. In children with cervical spine instability, dynamic flexion/extension radiographs of the cervical spine as part of the preoperative workup are recommended unless contraindicated (e.g., acute traumatic instability). | 89 | 77 | 12 |
| 3. In children with cervical spine instability, a CT scan of the cervical spine as part of the preoperative workup is recommended. | 89 | 77 | 12 |
| 4. In children with cervical spine instability, MRI of the cervical spine as part of the preoperative workup is recommended. | 100 | 88 | 12 |
| 5. In children with cervical spine instability, CT angiogram of the cervical spine should be considered in select cases of severe deformity or congenital anomalies. | 100 | 71 | 29 |
| 6. In symptomatic children with traumatic instability, a rigid cervical collar should be recommended until stabilization is performed. | 100 | 77 | 23 |
| 7. In symptomatic children with translational instability (e.g., excessive motion on dynamic radiographs), a rigid cervical collar should be recommended until stabilization is performed. | 100 | 65 | 35 |
| 8. In symptomatic children with vertical instability (e.g., basilar invagination), a rigid cervical collar should be recommended until stabilization is performed. | 82 | 47 | 35 |
| 9. In asymptomatic children with traumatic instability, a rigid cervical collar should be recommended until stabilization is performed. | 89 | 53 | 36 |
| 10. For hospitalized children with acute traumatic instability, activity restrictions (e.g., bedrest or out of bed with assistance) should be recommended. | 94 | 53 | 41 |
| 11. For children with non-traumatic instability, activity restrictions including avoidance of athletic activities should be recommended. | 94 | 41 | 53 |
| 12. Aspirin should be discontinued prior to pediatric cervical stabilization procedures, unless medically contraindicated. | 100 | 55 | 45 |
| B. Radiographic thresholds of instability | |||
| 13. Excluding children with Down syndrome, the pediatric cervical spine should be considered unstable at C1-C2 if there is translational motion ≥5 mm. | 95 | 24 | 71 |
| 14. The pediatric subaxial cervical spine should be considered unstable if there is translational motion ≥4 mm. | 83 | 12 | 71 |
| 15. The pediatric subaxial cervical spine should be considered unstable if there is dynamic angulation ≥10 degrees. | 83 | 12 | 71 |
| 16. The spine should be considered unstable in children if both abnormal motion and T2 signal change on MRI are seen at the same level. | 100 | 47 | 53 |
| C. Intraoperative and perioperative management | |||
| 17. In children ≥2 years old undergoing cervical spine stabilization procedures, a Mayfield head clamp or other rigid fixation of the head is preferred. | 94 | 47 | 47 |
| 18. In children undergoing stabilization procedures requiring reduction of the cervical spine, an arterial line is recommended. | 94 | 35 | 59 |
| 19. In children undergoing stabilization procedures requiring reduction of the cervical spine, intraoperative neuromonitoring including MEPs and SSEPs are recommended. | 100 | 53 | 47 |
| 20. In children undergoing stabilization procedures requiring reduction of the cervical spine, prepositioning baseline signals are recommended when there is concern for significant instability. | 100 | 65 | 35 |
| 21. In children undergoing posterior stabilization that are old enough to accommodate commercially available cervical spine screws (e.g., ≥2 years old at the craniovertebral junction and 3–4 years old in the subaxial spine), rigid instrumentation is recommended. | 100 | 88 | 12 |
| 22. In children undergoing anterior stabilization that are old enough to accommodate commercially available instrumentation (e.g., ≥4–5 years old), rigid instrumentation is recommended. | 100 | 77 | 23 |
| 23. In children undergoing anterior stabilization that are too young to accommodate commercially available instrumentation (e.g., <4–5 years old), rigid instrumentation using nonspinal instrumentation systems (e.g., hand or oral–maxillofacial systems) is recommended. | 89 | 24 | 65 |
| 24. In children undergoing posterior stabilization procedures with rigid fixation, some method of image guidance (e.g., fluoroscopy or navigation) is recommended. | 89 | 71 | 18 |
| 25. In children undergoing anterior stabilization procedures with rigid fixation, image guidance with fluoroscopy is recommended. | 94 | 59 | 35 |
| 26. In children undergoing cervical spine stabilization procedures, the choice of autograft or allograft should be made based on the clinical situation. | 100 | 53 | 47 |
| 27. In children undergoing posterior C1–2 cervical stabilization procedures, a postoperative drain is not routinely recommended. | 100 | 29 | 71 |
| 28. In children undergoing posterior occipital–cervical stabilization procedures, a postoperative drain is not routinely recommended. | 88 | 29 | 59 |
| 29. In children undergoing posterior subaxial cervical stabilization procedures, a postoperative drain is not routinely recommended. | 94 | 29 | 65 |
| 30. In children undergoing cervical stabilization procedures where a postoperative drain is left in place, prophylactic antibiotics do not need to be routinely continued until discontinuation of the drain. | 94 | 29 | 65 |
| 31. Intrawound antibiotic powder is recommended following pediatric posterior stabilization procedures. | 80 | 50 | 30 |
| D. Postoperative care | |||
| 32. Intravenous antibiotic coverage for 24 hours following pediatric cervical stabilization procedures is recommended. | 82 | 35 | 47 |
| 33. In children undergoing cervical stabilization procedures, mobilization with physical therapy as early as postoperative day 1 is recommended if possible. | 100 | 59 | 41 |
| 34. Halo vest immobilization is not routinely recommended for children undergoing cervical stabilization procedures with rigid instrumentation. | 88 | 47 | 41 |
| 35. In children undergoing posterior cervical stabilization procedures, postoperative external immobilization with a rigid cervical collar is recommended (e.g., all the time or when upright and out of bed). | 82 | 53 | 29 |
| 36. In children undergoing anterior cervical stabilization procedures, postoperative external immobilization with a rigid cervical collar is recommended (e.g., all the time or when upright and out of bed). | 82 | 41 | 41 |
| 37. In children undergoing cervical stabilization procedures, upright static radiographs of the cervical spine prior to discharge are recommended. | 88 | 35 | 53 |
| 38. In children undergoing cervical stabilization procedures, postoperative external immobilization with a rigid cervical collar is recommended for a duration of 6–12 weeks. | 82 | 41 | 41 |
| 39. Upright static or dynamic radiographs are recommended prior to discontinuing external immobilization after pediatric cervical stabilization procedures. | 100 | 47 | 53 |
| 40. Solid bridging bone seen on a CT scan or plain radiographs indicates successful arthrodesis following pediatric cervical stabilization procedures. | 100 | 65 | 35 |
| 41. In children who have undergone cervical spine stabilization procedures, clinical follow-up should continue at least until anatomic cervical spine maturity (around 14–15 years old in girls, 16–17 years old in boys). | 94 | 35 | 59 |
| 42. In children undergoing cervical stabilization procedures, nonsteroidal antiinflammatory drugs (NSAIDs) can be used for short-term pain control (up to 2 weeks). | 89 | 56 | 33 |
| E. Nonoperative management | |||
| 43. In children with cervical spine disorders that do not have current instability, routine CT scan is recommended only if a clinical change occurs. | 95 | 24 | 71 |
| 44. In children with cervical spine disorders that do not have current instability, routine MRI scan is recommended only if a clinical change occurs. | 95 | 24 | 71 |
| 45. In children with cervical spine disorders that do not have current instability, clinical follow-up is recommended at least until skeletal maturity. | 100 | 22 | 78 |
Statements requiring discussion and modification are indicated in italics.
Statements that did not reach consensus regarding the management of children with cervical spine disorders and stabilization (round 1)
| Statement | Response (%) | ||||
|---|---|---|---|---|---|
| Total Consensus | Strongly Agree | Agree | Disagree | Strongly Disagree | |
| 1. In asymptomatic children with vertical instability (e.g., basilar invagination), preoperative immobilization in a rigid cervical collar is not routinely necessary. | 59 | 18 | 41 | 35 | 6 |
| 2. It is not necessary to routinely discontinue NSAIDs prior to pediatric cervical stabilization procedures. | 77 | 18 | 59 | 24 | 0 |
| 3. Aspirin should be discontinued prior to pediatric cervical stabilization procedures. | 77 | 24 | 53 | 12 | 12 |
| 4. In children <3 years of age undergoing posterior cervical stabilization with structural autograft, rib is the preferred donor site. | 70 | 35 | 35 | 18 | 12 |
| 5. Intrawound vancomycin powder is recommended following pediatric posterior stabilization procedures. | 77 | 18 | 59 | 24 | 0 |
| 6. Children undergoing posterior occipital–cervical stabilization procedures should routinely be admitted to the pediatric intensive care unit postoperatively. | 70 | 35 | 35 | 24 | 6 |
| 7. In children undergoing cervical stabilization procedures, NSAIDs can be used at any time postoperatively. | 70 | 41 | 29 | 29 | 0 |
| 8. Intact hardware without solid bridging bone seen on plain or dynamic radiographs after 2 years in an asymptomatic patient indicates successful arthrodesis following pediatric cervical stabilization procedures. | 77 | 18 | 59 | 18 | 6 |
| 9. In children with cervical spine disorders that do not have current instability, clinical follow-up is recommended at least annually. | 65 | 6 | 59 | 29 | 6 |
| 10. In children with cervical spine disorders that do not have current instability, routine static radiographs performed at least every 2 years or in the setting of a clinical change are recommended. | 71 | 18 | 53 | 24 | 6 |
| 11. In children with cervical spine disorders that do not have current instability, routine dynamic radiographs performed at least every 2 years or in the setting of a clinical change are recommended. | 77 | 12 | 65 | 24 | 0 |
Revisions made to selected statements at the hybrid meeting to achieve consensus
| Initial Statement | Final Statement (% agree or strongly agree) |
|---|---|
| 1. Aspirin should be discontinued prior to pediatric cervical stabilization procedures. | Aspirin should be discontinued prior to pediatric cervical stabilization procedures, unless medically contraindicated. (100%) |
| 2. In children <3 years of age undergoing posterior cervical stabilization with structural autograft, rib is the preferred donor site. | Unable to reach consensus |
| 3. Intrawound vancomycin powder is recommended following pediatric posterior stabilization procedures. | Intrawound antibiotic powder is recommended following pediatric posterior stabilization procedures. (80%) |
| 4. Children undergoing posterior occipital–cervical stabilization procedures should routinely be admitted to the pediatric intensive care unit postoperatively. | Unable to reach consensus |
| 5. In children undergoing cervical stabilization procedures, NSAIDs can be used at any time postoperatively. | In children undergoing cervical stabilization procedures, NSAIDs can be used for short-term pain control for up to 2 weeks. (100%) |
| 6. Intact hardware without solid bridging bone seen on plain or dynamic radiographs after 2 years in an asymptomatic patient indicates successful arthrodesis following pediatric cervical stabilization procedures. | Unable to reach consensus |
| 7. In children with cervical spine disorders that do not have current instability, clinical follow-up is recommended at least annually. | In children with cervical spine disorders that do not have current instability, clinical follow-up is recommended at least until skeletal maturity. (100%) |
Preoperative Planning for Patients Requiring Cervical Stabilization
Regarding preoperative planning for pediatric patients requiring posterior cervical instrumentation, total consensus was reached for 12 of 14 (86%) statements. A 100% consensus was obtained for 5 statements that recommended the following: 1) obtaining a preoperative MRI; 2) considering a preoperative CT angiogram in select cases; 3) using a rigid cervical collar in symptomatic children with traumatic instability; 4) using a rigid cervical collar in symptomatic children with translational instability; and 5) discontinuing aspirin prior to surgery unless medically contraindicated. Between 82% and 94% agreement was reached for the remaining 7 consensus statements. Consensus could not be reached for 2 of 14 (14%) statements.
Radiographic Thresholds of Instability
Total consensus was reached for 4 of 4 (100%) statements regarding radiographic thresholds of instability. A 100% consensus was obtained for 1 statement that the spine should be considered unstable if both abnormal motion and T2 signal change on MRI are seen at the same level. Between 83% and 94% agreement was reached for the remaining 3 consensus statements.
Intraoperative and Perioperative Management
Regarding intraoperative and perioperative management, total consensus was reached for 15 of 17 (88%) statements. A 100% consensus was obtained for 6 statements that recommended 1) intraoperative somatosensory evoked potential (SSEP) and motor evoked potential (MEP) neuromonitoring; 2) including pre-positioning baselines; 3) rigid instrumentation when anatomically feasible for posterior fixation; 4) rigid instrumentation when anatomically feasible for anterior fixation; 5) clinical discretion when determining autograft versus allograft; and 6) that the use of a postoperative drain is not routinely necessary after posterior C1–2 stabilization. Between 80% and 94% agreement was reached for the remaining 9 consensus statements. Consensus could not be reached for 2 of 17 (12%) statements.
Postoperative Care
Total consensus was reached for 11 of 12 (92%) statements regarding postoperative care. A 100% consensus was obtained for 3 statements recommending the following: 1) mobilization with physical therapy starting on postoperative day 1 if possible; 2) upright radiographs prior to discontinuing external immobilization after surgery; and 3) that solid bridging bone on radiographs or CT scans indicates successful arthrodesis. Between 82% and 94% agreement was reached for the remaining 8 consensus statements. Consensus could not be reached for 1 of 12 (8%) statements.
Nonoperative Management
Regarding nonoperative management, total consensus was reached for 3 of 5 (60%) statements. A 100% consensus was obtained for 1 statement that recommended clinical follow-up at least until skeletal maturity in children with cervical spine disorders. A 94% agreement was reached for the remaining 2 consensus statements. Consensus could not be reached for 2 of 5 (40%) statements.
Discussion
In this paper, we demonstrate that using a modified Delphi approach, a multidisciplinary, international group of pediatric cervical spine experts was able to reach consensus on 45 statements regarding the preoperative, intraoperative, postoperative, and nonoperative management of pediatric cervical spine disorders and stabilization (Tables 3and 4). Our hope is that these consensus statements can provide a foundation of clinical guidance for the management of this challenging patient population and encourage future research into the topic.
Consensus Surrounding Preoperative Planning for Patients Requiring Cervical Stabilization
Imaging is critical for both the diagnosis and clinical management of children with cervical spine disorders. However, imaging in children is often more complicated than in adults due to the concerns of radiation exposure, need for sedation, and compliance with dynamic studies. In this study, consensus was achieved on the need for preoperative static cervical radiographs (88.2% agreement) as well as dynamic radiographs unless clinically contraindicated (88.2%). All participants agreed that preoperative MRI of the cervical spine is recommended, and 88.2% recommended a CT scan as well. For select cases of severe deformity or congenital anomalies, 100% recommend considering a CT angiogram of the cervical spine for operative planning. This is consistent with prior literature; several authors have recommended CT with multiplanar reconstructions to plan screw trajectories, and others have advocated for CT angiography for select cases to identify anomalous vertebral arteries or abnormal trajectories.8,18–21 Using CT angiography, for instance, Yamazaki et al. demonstrated an increased incidence of aberrant vertebral arteries in patients with atlantoaxial subluxation and congenital skeletal anomalies.22
Questions often arise regarding the need for activity restrictions and preoperative cervical immobilization in children, especially in those with long-standing congenital anomalies. For symptomatic children with preoperative translational (excessive motion on dynamic radiographs) and vertical (basilar invagination) instability, 100% and 82.4% of participants, respectively, recommended a rigid cervical collar until stabilized. The group also recommended the use of rigid cervical immobilization for both symptomatic (100% consensus) and asymptomatic (88.2% consensus) patients with traumatic instability. Regarding activity restrictions, there was consensus in recommending activity restrictions including avoidance of athletic activities for children with traumatic instability (94.1%) and for hospitalized children with acute traumatic instability (94.1%).
Consensus Regarding Radiographic Thresholds of Instability
Few comprehensive studies investigating radiographic thresholds of instability of the pediatric cervical spine have been reported. One excellent example is the establishment of the measurement at the condyle–C1 interval for the diagnosis of atlantooccipital dislocation.23 Most reported radiographic thresholds for instability in children, however, have been based on single-surgeon expert opinion8,10 or extrapolation from adult studies.24 For the first time to our knowledge, in this study an international, multidisciplinary group of experts agreed that children’s cervical spine should be considered unstable if there is 1) translational motion (measured by the atlantodens interval) ≥ 5 mm at C1–2 (excluding patients with Down syndrome); 2) translational motion > 4 mm or dynamic angulation > 10° in the subaxial spine; or 3) abnormal motion and T2 signal change on MRI seen at the same level.
Because of the high level of agreement reached regarding instability at C1–2 and the subaxial spine, there was an attempt to reach consensus for a radiographic threshold of instability at the occipitocervical junction (Oc–C1). Our initial survey resulted in various measurements for this threshold ranging from 3 to 5 mm, and we therefore attempted to discuss the point further. Following extensive discussion, consensus was not attainable primarily due to concerns regarding the difficulty of reliably measuring parameters on plain radiographs, which has previously been expressed in the literature.25–27
Consensus Surrounding Intraoperative and Perioperative Management
The operative management of pediatric cervical spine stabilization has transformed from semirigid fixation techniques using sublaminar wires and interlaminar bone to rigid techniques with screw-rod constructs and allograft or autograft.2,9,28 Biomechanical studies and outcome data outlining high fusion rates coupled with low complication rates have been the impetus behind this shift. Fusion rates at the craniovertebral junction in children have been reported to be between 85% and 100% when using rigid fixation techniques, in comparison to the lower fusion rates associated with traditional nonrigid fixation techniques and halo immobilization.2,9,29–31 For these reasons, it is not surprising that in our study, 100% of participants recommended the use of rigid instrumentation when possible. In addition, 88.2% recommended the use of nonspinal instrumentation systems (e.g., hand or oral–maxillofacial systems) in patients too young to accommodate commercially available anterior cervical instrumentation.
The potential role of intraoperative navigation in pediatric cervical spine surgery was also investigated. The numerous advantages of intraoperative navigation, including improved accuracy and avoidance of critical neurovascular structures, have been well documented in the spine surgery literature.32–34 Navigation systems have specifically demonstrated improved safety and accuracy for cervical pedicle screw fixation in light of altered anatomy.35,36 Although much less commonly reported in pediatric cervical spine surgery, a feasibility study in children younger than 10 years of age undergoing atlantoaxial screw placement with navigation found no immediate complications, and all screws were deemed properly placed.37 Although consensus for the use of intraoperative navigation was not reached in this study due to concerns with accuracy and availability, 88.2% of our participants recommended some type of image guidance (fluoroscopy or navigation) for posterior stabilization procedures, and 94.1% recommended fluoroscopy for anterior procedures.
Prior Delphi studies have established checklists to help surgeons respond to changes in intraoperative neuromonitoring during pediatric and adult spinal deformity surgery.38–40 These studies, however, did not consider pediatric cervical spine stabilization surgery. In this study, 100% of surgeons agreed that intraoperative neuromonitoring including MEPs and SSEPs is recommended, including pre-positioning baseline signals when there is concern for significant instability.
We also aimed to determine if there was consensus regarding the need for postoperative drains after cervical spine stabilization procedures. Among our cohort of pediatric spine surgeons, there was agreement that postoperative drains are not routinely necessary after C1–2, occipital–cervical, and subaxial stabilization procedures, and if used, continuation of prophylactic antibiotics until discontinuation of a drain is not necessary.
Consensus Surrounding Postoperative and Nonoperative Management
One goal of this study was to elucidate what practices are commonly used for postoperative immobilization after pediatric cervical spine surgery, given that there is no consensus reported in the pediatric cervical spine literature.41 Although there was agreement that halo vest immobilization is not routinely recommended for children undergoing cervical stabilization procedures with rigid instrumentation, the use of a rigid cervical collar (at all times or when the patient is upright and out of bed) postoperatively after both anterior and posterior procedures was recommended. Furthermore, 82.4% of participants recommended that postoperative external immobilization with a rigid cervical collar be continued for a duration of 6–12 weeks. These results differ from those of studies in adults, in which a survey of 83 international Cervical Spine Research Society members demonstrated that there was an equal likelihood of treatment without a brace (33%), a soft collar (32%), or a rigid collar (34%) after cervical fusion procedures for degenerative cervical spine disease.42 The reason for this discrepancy between adult and pediatric experience is unknown, but may be due to the increased frequency of limited screw fixation in young children.
Additional goals of the study were to determine if consensus could be achieved regarding the type and frequency of postoperative imaging and the radiographic determinants of successful fusion. Overall, 88.2% of surgeons recommended that upright static radiographs be obtained prior to discharge from the hospital, and 100% recommended upright static or dynamic radiographs prior to discontinuing external mobilization. Clinical equipoise remains regarding radiographic criteria for successful fusion after pediatric cervical spine surgery. Although classic imaging modalities for fusion assessment include static or dynamic radiographs and CT, more rarely used modalities in children, such as MRI, bone scans, ultrasound, and radiostereometric analysis, have also been investigated.43 Not surprisingly, 100% of participants agreed that solid bridging bone on CT or plain radiographs indicates successful arthrodesis after pediatric cervical spine stabilization procedures. Importantly, although near consensus at 77%, the group was not able to reach final consensus for the following statement even after extensive discussion at the in-person meeting: "Intact hardware without solid bridging bone seen on plain or dynamic radiographs after two years in an asymptomatic patient indicates successful arthrodesis following pediatric cervical stabilization procedures." This is a departure from the pediatric spinal deformity literature, in which stable plain upright radiographs in an asymptomatic patient 2 years after thoracolumbar spinal surgery are generally considered to indicate successful fusion.44 Overall, the group believed that after pediatric cervical spine surgery, this situation may indicate clinical success but not radiographic evidence of fusion. This highlights an important topic for future clinical study.
All surgeons in this study advocated for early mobilization (as early as postoperative day 1) with physical therapy following cervical stabilization procedures. This is concordant with recent literature emphasizing enhanced recovery after surgery in the adult population.45–47 Others have advocated for early mobilization after pediatric orthopedic spine surgery as well as improving multimodal pain management, given its association with mobilization.48,49
Participants also recommended that clinical follow-up should continue at least until anatomical cervical spine maturity is reached (approximately 14–16 years old in girls and 16–17 years old in boys)50 in postoperative patients and at least until skeletal maturity in children with cervical spine disorders without current instability.
Limitations and Future Studies
Consensus-driven studies such as this have limitations inherent to their design. The need for such a study highlights the lack of quality empirical evidence available and leads to a reliance on expert opinion. Although this study was able to generate consensus statements regarding the management of pediatric cervical spine disorders and stabilization, it cannot provide firm evidence-based guidelines for the management of these rare cases. Furthermore, the group of experts in this study all practice at large academic children’s hospitals; thus, the generalizability of these statements to community settings is not clear. Nevertheless, the goal of this study was to provide a framework for clinicians to build upon and guide future multicenter studies to standardize the clinical management of children with cervical spine disorders and stabilization.
Conclusions
In an attempt to improve and standardize the management of children with cervical spine disorders and stabilization, this paper reports 45 consensus statements generated by an international, multidisciplinary group of pediatric cervical spine experts using a modified Delphi technique. These statements aim to provide a clinical framework for clinicians caring for children with cervical spine disorders and to survey current practices in search of common techniques/management strategies. Further study is required to determine if implementation of these practices can lead to improved outcomes for children.
Disclosures
Dr. Bumpass: consultant for Medtronic and member of their speaker’s bureau; ownership in NuShores Biosciences. Dr. Pahys: consultant for DePuy Synthes, NuVasive, Zimmer Biomet. Dr. Yaszay: consultant for DePuy Synthes, NuVasive, Stryker, Medtronic; royalties from Stryker, NuVasive, OrthoPediatrics, Globus.
Author Contributions
Conception and design: Dastagirzada, Alexiades, Brockmeyer, Anderson. Acquisition of data: Dastagirzada, Alexiades, Kurland, Brockmeyer, Bumpass, Chatterjee, Groves, Hankinson, Harter, Hedequist, Jea, Leonard, Martin, Oetgen, Rozzelle, Strahle, Thompson, Yaszay, Anderson. Analysis and interpretation of data: Dastagirzada, Alexiades, Anderson, Pahys. Drafting the article: Dastagirzada, Alexiades, Kurland, Anderson, Anderson. Critically revising the article: Alexiades, Kurland, Brockmeyer, Bumpass, Chatterjee, Groves, Hankinson, Harter, Hedequist, Jea, Leonard, Martin, Oetgen, Pahys, Rozzelle, Strahle, Thompson, Yaszay, Anderson. Reviewed submitted version of manuscript: Alexiades, Brockmeyer, Bumpass, Chatterjee, Groves, Hankinson, Harter, Hedequist, Jea, Leonard, Martin, Oetgen, Pahys, Rozzelle, Strahle, Thompson, Yaszay, Anderson. Administrative/technical/material support: Dastagirzada, Anderson. Study supervision: Alexiades, Brockmeyer, Anderson.
Supplemental Information
Online-Only Content
Supplemental material is available with the online version of the article.
Supplementary Table. https://thejns.org/doi/suppl/10.3171/2022.9.PEDS22319.
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