Developing consensus for the management of pediatric cervical spine disorders and stabilization: a modified Delphi study

*Yosef M. DastagirzadaDepartment of Neurological Surgery, New York University, Hassenfeld Children’s Hospital, New York, New York;

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Nikita G. AlexiadesUniversity of Arizona College of Medicine–Phoenix, Arizona;

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David B. KurlandDepartment of Neurological Surgery, New York University, Hassenfeld Children’s Hospital, New York, New York;

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Sebastián N. AndersonNorthern Highlands Regional High School, Allendale, New Jersey;

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Douglas L. BrockmeyerDepartment of Pediatric Neurosurgery, Primary Children’s Medical Center, University of Utah, Salt Lake City, Utah;

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David B. BumpassDepartment of Orthopedic Surgery, University of Arkansas, Little Rock, Arkansas;

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Sandip ChatterjeeDepartment of Neurological Surgery, Park Clinic, Kolkata, India;

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Mari L. GrovesDepartment of Neurosurgery, Johns Hopkins University School of Medicine, Baltimore, Maryland;

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Todd C. HankinsonDepartment of Pediatric Neurosurgery, Children’s Hospital Colorado, University of Colorado Anschutz Medical Campus, Aurora, Colorado;

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David HarterDepartment of Neurological Surgery, New York University, Hassenfeld Children’s Hospital, New York, New York;

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Daniel HedequistDepartment of Neurosurgery, Boston Children’s Hospital, Harvard Medical School, Boston, Massachusetts;

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Andrew JeaDepartment of Neurological Surgery, University of Oklahoma, Oklahoma City, Oklahoma;

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Jeffrey R. LeonardDepartment of Neurosurgery, Nationwide Children’s Hospital, The Ohio State University College of Medicine, Columbus, Ohio;

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Jonathan E. MartinDivision of Pediatric Neurosurgery, Connecticut Children’s, Hartford, Connecticut;

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Matthew E. OetgenDivision of Orthopedic Surgery and Sports Medicine, Children’s National Hospital, Washington, DC;

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Joshua PahysDepartment of Pediatric Orthopedic Surgery, Shriners Hospital for Children, Philadelphia, Pennsylvania;

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Curtis RozzelleDepartment of Neurosurgery, Division of Pediatric Neurosurgery, University of Alabama, Birmingham, Alabama;

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Jennifer M. StrahleDepartment of Neurological Surgery, Washington University School of Medicine, St. Louis, Missouri;

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Dominic ThompsonDepartment of Neurosurgery, Great Ormond Street Hospital for Children, London, United Kingdom; and

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Burt YaszayDepartment of Orthopedics, University of Washington, Seattle Children’s Hospital, Seattle, Washington

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Richard C. E. AndersonDepartment of Neurological Surgery, New York University, Hassenfeld Children’s Hospital, New York, New York;

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OBJECTIVE

Cervical spine disorders in children are relatively uncommon; therefore, paradigms for surgical and nonsurgical clinical management are not well established. The purpose of this study was to bring together an international, multidisciplinary group of pediatric cervical spine experts to build consensus via a modified Delphi approach regarding the clinical management of children with cervical spine disorders and those undergoing cervical spine stabilization surgery.

METHODS

A modified Delphi method was used to identify consensus statements for the management of children with cervical spine disorders requiring stabilization. A survey of current practices, supplemented by a literature review, was electronically distributed to 17 neurosurgeons and orthopedic surgeons experienced with the clinical management of pediatric cervical spine disorders. Subsequently, 52 summary statements were formulated and distributed to the group. Statements that reached near consensus or that were of particular interest were then discussed during an in-person meeting to attain further consensus. Consensus was defined as ≥ 80% agreement on a 4-point Likert scale (strongly agree, agree, disagree, strongly disagree).

RESULTS

Forty-five consensus-driven statements were identified, with all participants willing to incorporate them into their practice. For children with cervical spine disorders and/or stabilization, consensus statements were divided into the following categories: A) preoperative planning (12 statements); B) radiographic thresholds of instability (4); C) intraoperative/perioperative management (15); D) postoperative care (11); and E) nonoperative management (3). Several important statements reaching consensus included the following recommendations: 1) to obtain pre-positioning baseline signals with intraoperative neuromonitoring; 2) to use rigid instrumentation when technically feasible; 3) to provide postoperative external immobilization for 6–12 weeks with a rigid cervical collar rather than halo vest immobilization; and 4) to continue clinical postoperative follow-up at least until anatomical cervical spine maturity was reached. In addition, preoperative radiographic thresholds for instability that reached consensus included the following: 1) translational motion ≥ 5 mm at C1–2 (excluding patients with Down syndrome) or ≥ 4 mm in the subaxial spine; 2) dynamic angulation in the subaxial spine ≥ 10°; and 3) abnormal motion and T2 signal change on MRI seen at the same level.

CONCLUSIONS

In this study, the authors have demonstrated that a multidisciplinary, international group of pediatric cervical spine experts was able to reach consensus on 45 statements regarding the management of pediatric cervical spine disorders and stabilization. Further study is required to determine if implementation of these practices can lead to reduced complications and improved outcomes for children.

ABBREVIATIONS

MEP = motor evoked potential; SSEP = somatosensory evoked potential.

OBJECTIVE

Cervical spine disorders in children are relatively uncommon; therefore, paradigms for surgical and nonsurgical clinical management are not well established. The purpose of this study was to bring together an international, multidisciplinary group of pediatric cervical spine experts to build consensus via a modified Delphi approach regarding the clinical management of children with cervical spine disorders and those undergoing cervical spine stabilization surgery.

METHODS

A modified Delphi method was used to identify consensus statements for the management of children with cervical spine disorders requiring stabilization. A survey of current practices, supplemented by a literature review, was electronically distributed to 17 neurosurgeons and orthopedic surgeons experienced with the clinical management of pediatric cervical spine disorders. Subsequently, 52 summary statements were formulated and distributed to the group. Statements that reached near consensus or that were of particular interest were then discussed during an in-person meeting to attain further consensus. Consensus was defined as ≥ 80% agreement on a 4-point Likert scale (strongly agree, agree, disagree, strongly disagree).

RESULTS

Forty-five consensus-driven statements were identified, with all participants willing to incorporate them into their practice. For children with cervical spine disorders and/or stabilization, consensus statements were divided into the following categories: A) preoperative planning (12 statements); B) radiographic thresholds of instability (4); C) intraoperative/perioperative management (15); D) postoperative care (11); and E) nonoperative management (3). Several important statements reaching consensus included the following recommendations: 1) to obtain pre-positioning baseline signals with intraoperative neuromonitoring; 2) to use rigid instrumentation when technically feasible; 3) to provide postoperative external immobilization for 6–12 weeks with a rigid cervical collar rather than halo vest immobilization; and 4) to continue clinical postoperative follow-up at least until anatomical cervical spine maturity was reached. In addition, preoperative radiographic thresholds for instability that reached consensus included the following: 1) translational motion ≥ 5 mm at C1–2 (excluding patients with Down syndrome) or ≥ 4 mm in the subaxial spine; 2) dynamic angulation in the subaxial spine ≥ 10°; and 3) abnormal motion and T2 signal change on MRI seen at the same level.

CONCLUSIONS

In this study, the authors have demonstrated that a multidisciplinary, international group of pediatric cervical spine experts was able to reach consensus on 45 statements regarding the management of pediatric cervical spine disorders and stabilization. Further study is required to determine if implementation of these practices can lead to reduced complications and improved outcomes for children.

In Brief

A group of international, multidisciplinary pediatric cervical spine experts aimed to provide a clinical framework for clinicians caring for children with cervical spine disorders. Using a modified Delphi approach, they developed consensus statements addressing various aspects of clinical management and decision-making (e.g., preoperative planning, radiographic thresholds of instability, perioperative management, and nonoperative management) for this unique patient population. These statements reflect current practices and can serve as a foundation for further studies in the field.

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.14 Throughout the past 2 decades, rigid fixation techniques have overtaken semirigid techniques for stabilization of craniocervical and subaxial cervical instability, with improved outcomes.57 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.811

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.1317

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.

TABLE 1.

Demographics of participants in survey

CharacteristicNo. (%)
No. of participants17
 Neurosurgery trained13 (76.5%)
 Orthopedic trained4 (23.5%)
Geographic distribution
 US15
 United Kingdom1
 India1
Clinical experience
 >20 yrs5 (29%)
 16–20 yrs4 (24%)
 11–15 yrs5 (29%)
 6–10 yrs3 (18%)
TABLE 2.

Participant case volume by procedure type

Stabilization ProcedureNo. (%)
Oc–C1
 ≤5/yr8 (47%)
 6–10/yr5 (29%)
 11–15/yr3 (18%)
 16–20/yr1 (6%)
C1–2
 ≤5/yr10 (59%)
 6–10/yr5 (29%)
 11–15/yr2 (12%)
Subaxial
 ≤5/yr8 (47%)
 6–10/yr6 (35%)
 11–15/yr1 (6%)
 16–20/yr2 (12%)
Anterior
 ≤5/yr12 (70%)
 6–10/yr3 (18%)
 11–15/yr2 (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.

TABLE 3.

Statements that reached consensus regarding the management of children with cervical spine disorders and stabilization*

StatementResponse (%)
Total ConsensusStrongly AgreeAgree
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.885929
 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). 897712
 3. In children with cervical spine instability, a CT scan of the cervical spine as part of the preoperative workup is recommended.897712
 4. In children with cervical spine instability, MRI of the cervical spine as part of the preoperative workup is recommended.1008812
 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.1007129
 6. In symptomatic children with traumatic instability, a rigid cervical collar should be recommended until stabilization is performed.1007723
 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. 1006535
 8. In symptomatic children with vertical instability (e.g., basilar invagination), a rigid cervical collar should be recommended until stabilization is performed.824735
 9. In asymptomatic children with traumatic instability, a rigid cervical collar should be recommended until stabilization is performed.895336
 10. For hospitalized children with acute traumatic instability, activity restrictions (e.g., bedrest or out of bed with assistance) should be recommended.945341
 11. For children with non-traumatic instability, activity restrictions including avoidance of athletic activities should be recommended.944153
 12. Aspirin should be discontinued prior to pediatric cervical stabilization procedures, unless medically contraindicated.1005545
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.952471
 14. The pediatric subaxial cervical spine should be considered unstable if there is translational motion ≥4 mm.831271
 15. The pediatric subaxial cervical spine should be considered unstable if there is dynamic angulation ≥10 degrees.831271
 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.1004753
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.944747
 18. In children undergoing stabilization procedures requiring reduction of the cervical spine, an arterial line is recommended.943559
 19. In children undergoing stabilization procedures requiring reduction of the cervical spine, intraoperative neuromonitoring including MEPs and SSEPs are recommended.1005347
 20. In children undergoing stabilization procedures requiring reduction of the cervical spine, prepositioning baseline signals are recommended when there is concern for significant instability.1006535
 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.1008812
 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.1007723
 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.892465
 24. In children undergoing posterior stabilization procedures with rigid fixation, some method of image guidance (e.g., fluoroscopy or navigation) is recommended.897118
 25. In children undergoing anterior stabilization procedures with rigid fixation, image guidance with fluoroscopy is recommended.945935
 26. In children undergoing cervical spine stabilization procedures, the choice of autograft or allograft should be made based on the clinical situation.1005347
 27. In children undergoing posterior C1–2 cervical stabilization procedures, a postoperative drain is not routinely recommended.1002971
 28. In children undergoing posterior occipital–cervical stabilization procedures, a postoperative drain is not routinely recommended.882959
 29. In children undergoing posterior subaxial cervical stabilization procedures, a postoperative drain is not routinely recommended.942965
 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.942965
 31. Intrawound antibiotic powder is recommended following pediatric posterior stabilization procedures.805030
D. Postoperative care
 32. Intravenous antibiotic coverage for 24 hours following pediatric cervical stabilization procedures is recommended.823547
 33. In children undergoing cervical stabilization procedures, mobilization with physical therapy as early as postoperative day 1 is recommended if possible.1005941
 34. Halo vest immobilization is not routinely recommended for children undergoing cervical stabilization procedures with rigid instrumentation.884741
 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).825329
 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). 824141
 37. In children undergoing cervical stabilization procedures, upright static radiographs of the cervical spine prior to discharge are recommended.883553
 38. In children undergoing cervical stabilization procedures, postoperative external immobilization with a rigid cervical collar is recommended for a duration of 6–12 weeks.824141
 39. Upright static or dynamic radiographs are recommended prior to discontinuing external immobilization after pediatric cervical stabilization procedures.1004753
 40. Solid bridging bone seen on a CT scan or plain radiographs indicates successful arthrodesis following pediatric cervical stabilization procedures.1006535
 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).943559
 42. In children undergoing cervical stabilization procedures, nonsteroidal antiinflammatory drugs (NSAIDs) can be used for short-term pain control (up to 2 weeks).895633
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.952471
 44. In children with cervical spine disorders that do not have current instability, routine MRI scan is recommended only if a clinical change occurs.952471
 45. In children with cervical spine disorders that do not have current instability, clinical follow-up is recommended at least until skeletal maturity.1002278

Statements requiring discussion and modification are indicated in italics.

TABLE 4.

Statements that did not reach consensus regarding the management of children with cervical spine disorders and stabilization (round 1)

StatementResponse (%)
Total ConsensusStrongly AgreeAgreeDisagreeStrongly Disagree
1. In asymptomatic children with vertical instability (e.g., basilar invagination), preoperative immobilization in a rigid cervical collar is not routinely necessary.591841356
2. It is not necessary to routinely discontinue NSAIDs prior to pediatric cervical stabilization procedures.771859240
3. Aspirin should be discontinued prior to pediatric cervical stabilization procedures.7724531212
4. In children <3 years of age undergoing posterior cervical stabilization with structural autograft, rib is the preferred donor site.7035351812
5. Intrawound vancomycin powder is recommended following pediatric posterior stabilization procedures.771859240
6. Children undergoing posterior occipital–cervical stabilization procedures should routinely be admitted to the pediatric intensive care unit postoperatively.703535246
7. In children undergoing cervical stabilization procedures, NSAIDs can be used at any time postoperatively.704129290
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.771859186
9. In children with cervical spine disorders that do not have current instability, clinical follow-up is recommended at least annually.65659296
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.711853246
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.771265240
TABLE 5.

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.8,1821 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.2527

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.2,9,2931 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.3234 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.3840 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.4547 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.

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Supplementary Materials

  • Collapse
  • Expand

Figure from Candela-Cantó et al. (pp 61–70).

  • 1

    Smith MD, Phillips WA, Hensinger RN. Complications of fusion to the upper cervical spine. Spine (Phila Pa 1976). 1991;16(7):702-705.

  • 2

    Lowry DW, Pollack IF, Clyde B, Albright AL, Adelson PD. Upper cervical spine fusion in the pediatric population. J Neurosurg. 1997;87(5):671676.

  • 3

    Smith MD, Phillips WA, Hensinger RN. Fusion of the upper cervical spine in children and adolescents. An analysis of 17 patients. Spine (Phila Pa 1976). 1991;16(7):695-701.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4

    Hwang SW, Gressot LV, Rangel-Castilla L, et al. Outcomes of instrumented fusion in the pediatric cervical spine. J Neurosurg Spine. 2012;17(5):397409.

  • 5

    Haque A, Price AV, Sklar FH, Swift DM, Weprin BE, Sacco DJ. Screw fixation of the upper cervical spine in the pediatric population. Clinical article. J Neurosurg Pediatr. 2009;3(6):529533.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    Hedequist D. Modern instrumentation of the pediatric occiput and upper cervical spine: review article. HSS J. 2015;11(1):914.

  • 7

    Hedequist DJ. Modern posterior screw techniques in the pediatric cervical spine. World J Orthop. 2014;5(2):9499.

  • 8

    Brockmeyer DL, York JE, Apfelbaum RI. Anatomical suitability of C1-2 transarticular screw placement in pediatric patients. J Neurosurg. 2000;92(1 suppl):711.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9

    Anderson RC, Ragel BT, Mocco J, Bohman LE, Brockmeyer DL. Selection of a rigid internal fixation construct for stabilization at the craniovertebral junction in pediatric patients. J Neurosurg. 2007;107(1 suppl):3642.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Menezes AH. Craniocervical fusions in children. J Neurosurg Pediatr. 2012;9(6):573585.

  • 11

    Ahmed R, Traynelis VC, Menezes AH. Fusions at the craniovertebral junction. Childs Nerv Syst. 2008;24(10):12091224.

  • 12

    Dalkey N, Helmer O. An experimental application of the Delphi method to the use of experts. Manage Sci. 1963;9(3):458467.

  • 13

    Alexiades NG, Shao B, Braga BP, et al. Development of best practices in the utilization and implementation of pediatric cervical spine traction: a modified Delphi study. J Neurosurg Pediatr. 2021;27(6):649660.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Pezold ML, Pusic AL, Cohen WA, et al. Defining a research agenda for patient-reported outcomes in surgery: using a Delphi survey of stakeholders. JAMA Surg. 2016;151(10):930936.

    • Crossref
    • Search Google Scholar
    • Export Citation
  • 15

    Alexiades NG, Ahn ES, Blount JP, et al. Development of best practices to minimize wound complications after complex tethered spinal cord surgery: a modified Delphi study. J Neurosurg Pediatr. 2018;22(6):701709.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 16

    Roye BD, Campbell ML, Matsumoto H, et al. Establishing consensus on the best practice guidelines for use of halo gravity traction for pediatric spinal deformity. J Pediatr Orthop. 2020;40(1):e42e48.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 17

    Vitale MG, Riedel MD, Glotzbecker MP, et al. Building consensus: development of a Best Practice Guideline (BPG) for surgical site infection (SSI) prevention in high-risk pediatric spine surgery. J Pediatr Orthop. 2013;33(5):471478.

    • Crossref
    • Search Google Scholar
    • Export Citation
  • 18

    Ould-Slimane M, Le Pape S, Leroux J, et al. CT analysis of C2 pedicles morphology and considerations of useful parameters for screwing. Surg Radiol Anat. 2014;36(6):537542.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 19

    Resnick DK, Lapsiwala S, Trost GR. Anatomic suitability of the C1-C2 complex for pedicle screw fixation. Spine (Phila Pa 1976). 2002;27(14):14941498.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 20

    Barker R, Fareedi S, Thompson D, Saunders D. The use of CT angiography in the preoperative planning of cervical spine surgery in children. Childs Nerv Syst. 2009;25(8):955959.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 21

    Moon BJ, Choi KH, Shin DA, et al. Anatomical variations of vertebral artery and C2 isthmus in atlanto-axial fusion: consecutive surgical 100 cases. J Clin Neurosci. 2018;53:147152.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 22

    Yamazaki M, Okawa A, Furuya T, et al. Anomalous vertebral arteries in the extra- and intraosseous regions of the craniovertebral junction visualized by 3-dimensional computed tomographic angiography: analysis of 100 consecutive surgical cases and review of the literature. Spine (Phila Pa 1976). 2012;37(22):E1389-E1397.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 23

    Pang D, Nemzek WR, Zovickian J. Atlanto-occipital dislocation—part 2: the clinical use of (occipital) condyle-C1 interval, comparison with other diagnostic methods, and the manifestation, management, and outcome of atlanto-occipital dislocation in children. Neurosurgery. 2007;61(5):9951015.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 24

    White AA III, Johnson RM, Panjabi MM, Southwick WO. Biomechanical analysis of clinical stability in the cervical spine. Clin Orthop Relat Res. 1975;(109):85-96.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 25

    Wellborn CC, Sturm PF, Hatch RS, Bomze SR, Jablonski K. Intraobserver reproducibility and interobserver reliability of cervical spine measurements. J Pediatr Orthop. 2000;20(1):6670.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 26

    Douglas TS, Sanders V, Machers S, Pitcher R, van As AB. Digital radiographic measurement of the atlantodental interval in children. J Pediatr Orthop. 2007;27(1):2326.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 27

    Karol LA, Sheffield EG, Crawford K, Moody MK, Browne RH. Reproducibility in the measurement of atlanto-occipital instability in children with Down syndrome. Spine (Phila Pa 1976). 1996;21(21):2463-2468.

    • Crossref
    • PubMed
    • Search Google Scholar
    • Export Citation
  • 28

    Martinez-Del-Campo E, Turner JD, Rangel-Castilla L, Soriano-Baron H, Kalb S, Theodore N. Pediatric occipitocervical fixation: radiographic criteria, surgical technique, and clinical outcomes based on experience of a single surgeon. J Neurosurg Pediatr. 2016;18(4):452462.

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