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Socioeconomic and demographic factors in the diagnosis and treatment of Chiari malformation type I and syringomyelia

Syed Hassan A. Akbari, Asad A. Rizvi, Travis S. CreveCoeur, Rowland H. Han, Jacob K. Greenberg, James Torner, Douglas L. Brockmeyer, John C. Wellons III, Jeffrey R. Leonard, Francesco T. Mangano, James M. Johnston, Manish N. Shah, Bermans J. Iskandar, Raheel Ahmed, Gerald F. Tuite, Bruce A. Kaufman, David J. Daniels, Eric M. Jackson, Gerald A. Grant, Alexander K. Powers, Daniel E. Couture, P. David Adelson, Tord D. Alden, Philipp R. Aldana, Richard C. E. Anderson, Nathan R. Selden, Karin Bierbrauer, William Boydston, Joshua J. Chern, William E. Whitehead, Robert C. Dauser, Richard G. Ellenbogen, Jeffrey G. Ojemann, Herbert E. Fuchs, Daniel J. Guillaume, Todd C. Hankinson, Brent R. O’Neill, Mark Iantosca, W. Jerry Oakes, Robert F. Keating, Paul Klimo Jr., Michael S. Muhlbauer, J. Gordon McComb, Arnold H. Menezes, Nickalus R. Khan, Toba N. Niazi, John Ragheb, Chevis N. Shannon, Jodi L. Smith, Laurie L. Ackerman, Andrew H. Jea, Cormac O. Maher, Prithvi Narayan, Gregory W. Albert, Scellig S. D. Stone, Lissa C. Baird, Naina L. Gross, Susan R. Durham, Stephanie Greene, Robert C. McKinstry, Joshua S. Shimony, Jennifer M. Strahle, Matthew D. Smyth, Ralph G. Dacey Jr., Tae Sung Park, and David D. Limbrick Jr.

OBJECTIVE

The goal of this study was to assess the social determinants that influence access and outcomes for pediatric neurosurgical care for patients with Chiari malformation type I (CM-I) and syringomyelia (SM).

METHODS

The authors used retro- and prospective components of the Park-Reeves Syringomyelia Research Consortium database to identify pediatric patients with CM-I and SM who received surgical treatment and had at least 1 year of follow-up data. Race, ethnicity, and insurance status were used as comparators for preoperative, treatment, and postoperative characteristics and outcomes.

RESULTS

A total of 637 patients met inclusion criteria, and race or ethnicity data were available for 603 (94.7%) patients. A total of 463 (76.8%) were non-Hispanic White (NHW) and 140 (23.2%) were non-White. The non-White patients were older at diagnosis (p = 0.002) and were more likely to have an individualized education plan (p < 0.01). More non-White than NHW patients presented with cerebellar and cranial nerve deficits (i.e., gait ataxia [p = 0.028], nystagmus [p = 0.002], dysconjugate gaze [p = 0.03], hearing loss [p = 0.003], gait instability [p = 0.003], tremor [p = 0.021], or dysmetria [p < 0.001]). Non-White patients had higher rates of skull malformation (p = 0.004), platybasia (p = 0.002), and basilar invagination (p = 0.036). Non-White patients were more likely to be treated at low-volume centers than at high-volume centers (38.7% vs 15.2%; p < 0.01). Non-White patients were older at the time of surgery (p = 0.001) and had longer operative times (p < 0.001), higher estimated blood loss (p < 0.001), and a longer hospital stay (p = 0.04). There were no major group differences in terms of treatments performed or complications. The majority of subjects used private insurance (440, 71.5%), whereas 175 (28.5%) were using Medicaid or self-pay. Private insurance was used in 42.2% of non-White patients compared to 79.8% of NHW patients (p < 0.01). There were no major differences in presentation, treatment, or outcome between insurance groups. In multivariate modeling, non-White patients were more likely to present at an older age after controlling for sex and insurance status (p < 0.01). Non-White and male patients had a longer duration of symptoms before reaching diagnosis (p = 0.033 and 0.004, respectively).

CONCLUSIONS

Socioeconomic and demographic factors appear to influence the presentation and management of patients with CM-I and SM. Race is associated with age and timing of diagnosis as well as operating room time, estimated blood loss, and length of hospital stay. This exploration of socioeconomic and demographic barriers to care will be useful in understanding how to improve access to pediatric neurosurgical care for patients with CM-I and SM.

In Journal of Neurosurgery: Pediatrics Volume 29: Issue 3 (Mar 2022)

Free access

Obex position is associated with syringomyelia and use of posterior fossa decompression among patients with Chiari I malformation

Gabe Haller, Brooke Sadler, Timothy Kuensting, Nivan Lakshman, Jacob K. Greenberg, Jennifer M. Strahle, Tae Sung Park, Matthew B. Dobbs, Christina A. Gurnett, and David D. Limbrick Jr.

OBJECTIVE

Chiari I malformation (CM-I) has traditionally been defined by measuring the position of the cerebellar tonsils relative to the foramen magnum. The relationships of tonsillar position to clinical presentation, syringomyelia, scoliosis, and the use of posterior fossa decompression (PFD) surgery have been studied extensively and yielded inconsistent results. Obex position has been proposed as a useful adjunctive descriptor for CM-I and may be associated with clinical disease severity.

METHODS

A retrospective chart review was performed of 442 CM-I patients with MRI who presented for clinical evaluation between 2003 and 2018. Clinical and radiological variables were measured for all patients, including presence/location of headaches, Chiari Severity Index (CSI) grade, tonsil position, obex position, clival canal angle, pB-C2 distance, occipitalization of the atlas, basilar invagination, syringomyelia, syrinx diameter, scoliosis, and use of PFD. Radiological measurements were then used to predict clinical characteristics using regression and survival analyses, with performing PFD, the presence of a syrinx, and scoliosis as outcome variables.

RESULTS

Among the radiological measurements, tonsil position, obex position, and syringomyelia were each independently associated with use of PFD. Together, obex position, tonsil position, and syringomyelia (area under the curve [AUC] 89%) or obex position and tonsil position (AUC 85.4%) were more strongly associated with use of PFD than tonsil position alone (AUC 76%) (Pdiff = 3.4 × 10−6 and 6 × 10−4, respectively) but were only slightly more associated than obex position alone (AUC 82%) (Pdiff = 0.01 and 0.18, respectively). Additionally, obex position was significantly associated with occipital headaches, CSI grade, syringomyelia, and scoliosis, independent of tonsil position. Tonsil position was associated with each of these traits when analyzed alone but did not remain significantly associated with use of PFD when included in multivariate analyses with obex position.

CONCLUSIONS

Compared with tonsil position alone, obex position is more strongly associated with symptomatic CM-I, as measured by presence of a syrinx, scoliosis, or use of PFD surgery. These results support the role of obex position as a useful radiological measurement to inform the evaluation and potentially the management of CM-I.

In Journal of Neurosurgery: Pediatrics Volume 26: Issue 1 (Jul 2020)