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Cavernous malformations of the brainstem in infants

Report of two cases and review of the literature

Bruno P. Braga, Leodante B. Costa Jr., Sergio Lemos, and Marcelo D. Vilela

✓ Cavernous malformations of the brainstem (CMB) occur less commonly in children than in adults. Their appearance is even rarer in infants, with only five cases reported in the literature. The authors report two additional cases in which giant CMBs were diagnosed in two infants, one when the patient was 1 month old and the other when the patient was 15 months old. A median suboccipital approach in one patient and a pterional–orbitozygomatic approach in the other were used to obtain complete resection of the malformations. Excellent outcomes were achieved in both children.

A review of the literature is also presented. It seems that CMBs in infants usually follow a progressive course of growth and associated neurological deterioration. Patients with symptomatic lesions abutting the pial surface should undergo surgical treatment with the goal of cure. An increase may be expected in the number of CMBs diagnosed in children as a result of regular screening of relatives with the familial form of the disease. Nevertheless, due to the small confines of the brainstem, incidental or asymptomatic CMB should still be extraordinary. In the case of such a rare occurrence, conservative treatment should be advocated.

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Hannah E. Goldstein, Justin A. Neira, Matei Banu, Philipp R. Aldana, Bruno P. Braga, Douglas L. Brockmeyer, Michael L. DiLuna, Daniel H. Fulkerson, Todd C. Hankinson, Andrew H. Jea, Sean M. Lew, David D. Limbrick, Jonathan Martin, Joshua M. Pahys, Luis F. Rodriguez, Curtis J. Rozzelle, Gerald F. Tuite, Nicholas M. Wetjen, and Richard C. E. Anderson

OBJECTIVE

The long-term effects of surgical fusion on the growing subaxial cervical spine are largely unknown. Recent cross-sectional studies have demonstrated that there is continued growth of the cervical spine through the teenage years. The purpose of this multicenter study was to determine the effects of rigid instrumentation and fusion on the growing subaxial cervical spine by investigating vertical growth, cervical alignment, cervical curvature, and adjacent-segment instability over time.

METHODS

A total of 15 centers participated in this multi-institutional retrospective study. Cases involving children less than 16 years of age who underwent rigid instrumentation and fusion of the subaxial cervical spine (C-2 and T-1 inclusive) with at least 1 year of clinical and radiographic follow-up were investigated. Charts were reviewed for clinical data. Postoperative and most recent radiographs, CT, and MR images were used to measure vertical growth and assess alignment and stability.

RESULTS

Eighty-one patients were included in the study, with a mean follow-up of 33 months. Ninety-five percent of patients had complete clinical resolution or significant improvement in symptoms. Postoperative cervical kyphosis was seen in only 4 patients (5%), and none developed a swan-neck deformity, unintended adjacent-level fusion, or instability. Of patients with at least 2 years of follow-up, 62% demonstrated growth across the fusion construct. On average, vertical growth was 79% (4-level constructs), 83% (3-level constructs), or 100% (2-level constructs) of expected growth. When comparing the group with continued vertical growth to the one without growth, there were no statistically significant differences in terms of age, sex, underlying etiology, surgical approach, or number of levels fused.

CONCLUSIONS

Continued vertical growth of the subaxial spine occurs in nearly two-thirds of children after rigid instrumentation and fusion of the subaxial spine. Failure of continued vertical growth is not associated with the patient’s age, sex, underlying etiology, number of levels fused, or surgical approach. Further studies are needed to understand this dichotomy and determine the long-term biomechanical effects of surgery on the growing pediatric cervical spine.

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Nikita G. Alexiades, Belinda Shao, Bruno P. Braga, Christopher M. Bonfield, Douglas L. Brockmeyer, Samuel R. Browd, Michael DiLuna, Mari L. Groves, Todd C. Hankinson, Andrew Jea, Jeffrey R. Leonard, Sean M. Lew, David D. Limbrick Jr., Francesco T. Mangano, Jonathan Martin, Joshua Pahys, Alexander Powers, Mark R. Proctor, Luis Rodriguez, Curtis Rozzelle, Phillip B. Storm, and Richard C. E. Anderson

OBJECTIVE

Cervical traction in pediatric patients is an uncommon but invaluable technique in the management of cervical trauma and deformity. Despite its utility, little empirical evidence exists to guide its implementation, with most practitioners employing custom or modified adult protocols. Expert-based best practices may improve the care of children undergoing cervical traction. In this study, the authors aimed to build consensus and establish best practices for the use of pediatric cervical traction in order to enhance its utilization, safety, and efficacy.

METHODS

A modified Delphi method was employed to try to identify areas of consensus regarding the utilization and implementation of pediatric cervical spine traction. A literature review of pediatric cervical traction was distributed electronically along with a survey of current practices to a group of 20 board-certified pediatric neurosurgeons and orthopedic surgeons with expertise in the pediatric cervical spine. Sixty statements were then formulated and distributed to the group. The results of the second survey were discussed during an in-person meeting leading to further consensus. Consensus was defined as ≥ 80% agreement on a 4-point Likert scale (strongly agree, agree, disagree, strongly disagree).

RESULTS

After the initial round, consensus was achieved with 40 statements regarding the following topics: goals, indications, and contraindications of traction (12), pretraction imaging (6), practical application and initiation of various traction techniques (8), protocols in trauma and deformity patients (8), and management of traction-related complications (6). Following the second round, an additional 9 statements reached consensus related to goals/indications/contraindications of traction (4), related to initiation of traction (4), and related to complication management (1). All participants were willing to incorporate the consensus statements into their practice.

CONCLUSIONS

In an attempt to improve and standardize the use of cervical traction in pediatric patients, the authors have identified 49 best-practice recommendations, which were generated by reaching consensus among a multidisciplinary group of pediatric spine experts using a modified Delphi technique. Further study is required to determine if implementation of these practices can lead to reduced complications and improved outcomes for children.