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Factors associated with syrinx size in pediatric patients treated for Chiari malformation type I and syringomyelia: a study from the Park-Reeves Syringomyelia Research Consortium.
J Neurosurg Pediatr. 2020 Mar 06 [Online ahead of print]JN

Abstract

OBJECTIVE

Factors associated with syrinx size in pediatric patients undergoing posterior fossa decompression (PFD) or PFD with duraplasty (PFDD) for Chiari malformation type I (CM-I) with syringomyelia (SM; CM-I+SM) are not well established.

METHODS

Using the Park-Reeves Syringomyelia Research Consortium registry, the authors analyzed variables associated with syrinx radiological outcomes in patients (< 20 years old at the time of surgery) with CM-I+SM undergoing PFD or PFDD. Syrinx resolution was defined as an anteroposterior (AP) diameter of ≤ 2 mm or ≤ 3 mm or a reduction in AP diameter of ≥ 50%. Syrinx regression or progression was defined using 1) change in syrinx AP diameter (≥ 1 mm), or 2) change in syrinx length (craniocaudal, ≥ 1 vertebral level). Syrinx stability was defined as a < 1-mm change in syrinx AP diameter and no change in syrinx length.

RESULTS

The authors identified 380 patients with CM-I+SM who underwent PFD or PFDD. Cox proportional hazards modeling revealed younger age at surgery and PFDD as being independently associated with syrinx resolution, defined as a ≤ 2-mm or ≤ 3-mm AP diameter or ≥ 50% reduction in AP diameter. Radiological syrinx resolution was associated with improvement in headache (p < 0.005) and neck pain (p < 0.011) after PFD or PFDD. Next, PFDD (p = 0.005), scoliosis (p = 0.007), and syrinx location across multiple spinal segments (p = 0.001) were associated with syrinx diameter regression, whereas increased preoperative frontal-occipital horn ratio (FOHR; p = 0.007) and syrinx location spanning multiple spinal segments (p = 0.04) were associated with syrinx length regression. Scoliosis (HR 0.38 [95% CI 0.16-0.91], p = 0.03) and smaller syrinx diameter (5.82 ± 3.38 vs 7.86 ± 3.05 mm; HR 0.60 [95% CI 0.34-1.03], p = 0.002) were associated with syrinx diameter stability, whereas shorter preoperative syrinx length (5.75 ± 4.01 vs 9.65 ± 4.31 levels; HR 0.21 [95% CI 0.12-0.38], p = 0.0001) and smaller pB-C2 distance (6.86 ± 1.27 vs 7.18 ± 1.38 mm; HR 1.44 [95% CI 1.02-2.05], p = 0.04) were associated with syrinx length stability. Finally, younger age at surgery (8.19 ± 5.02 vs 10.29 ± 4.25 years; HR 1.89 [95% CI 1.31-3.04], p = 0.01) was associated with syrinx diameter progression, whereas increased postoperative syrinx diameter (6.73 ± 3.64 vs 3.97 ± 3.07 mm; HR 3.10 [95% CI 1.67-5.76], p = 0.003), was associated with syrinx length progression. PFD versus PFDD was not associated with syrinx progression or reoperation rate.

CONCLUSIONS

These data suggest that PFDD and age are independently associated with radiological syrinx improvement, although forthcoming results from the PFDD versus PFD randomized controlled trial (NCT02669836, clinicaltrials.gov) will best answer this question.

Authors+Show Affiliations

1Vanderbilt University School of Medicine, Medical Scientist Training Program, Nashville, Tennessee. 2Surgical Outcomes Center for Kids, Monroe Carell Jr. Children's Hospital of Vanderbilt University, Nashville, Tennessee.3Division of Pediatric Neurosurgery, Barrow Neurological Institute at Phoenix Children's Hospital, Phoenix, Arizona.4Division of Neurosurgery, Arkansas Children's Hospital, Little Rock, Arkansas.5Division of Pediatric Neurosurgery, University of Florida College of Medicine, Jacksonville, Florida.6Division of Pediatric Neurosurgery, Ann and Robert H. Lurie Children's Hospital of Chicago, Illinois.7Division of Pediatric Neurosurgery, Department of Neurological Surgery, Children's Hospital of New York, Columbia-Presbyterian, New York, New York.8Department of Neurosurgery, Dartmouth-Hitchcock Medical Center, Lebanon, New Hampshire.2Surgical Outcomes Center for Kids, Monroe Carell Jr. Children's Hospital of Vanderbilt University, Nashville, Tennessee. 9Division of Pediatric Neurosurgery, Monroe Carell Jr. Children's Hospital of Vanderbilt University, Nashville, Tennessee.10Division of Pediatric Neurosurgery, Primary Children's Hospital, Salt Lake City, Utah.11Division of Pediatric Neurosurgery, Children's Healthcare of Atlanta University, Atlanta, Georgia.12Department of Neurological Surgery, Wake Forest University School of Medicine, Winston-Salem, North Carolina.13Department of Neurosurgery, Mayo Clinic, Rochester, Minnesota.14Department of Neurosurgery, University of Vermont, Burlington, Vermont.15Division of Pediatric Neurosurgery, Seattle Children's Hospital, Seattle, Washington.16Department of Neurosurgery, Medical University of South Carolina, Charleston, South Carolina.17Division of Pediatric Neurosurgery, Dell Children's Medical Center, Austin, Texas.18Division of Pediatric Neurosurgery, Lucile Packard Children's Hospital, Palo Alto, California.19Division of Pediatric Neurosurgery, Gillette Children's Hospital, St. Paul, Minnesota.20Division of Pediatric Neurosurgery, Children's Hospital of Pittsburgh, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.21Department of Neurological Surgery, Weill Cornell Medical College, NewYork-Presbyterian Hospital, New York, New York.22Department of Neurosurgery, University of Oklahoma Health Sciences Center, Oklahoma City, Oklahoma.23Department of Neurosurgery, University of Minnesota Medical School, Minneapolis, Minnesota.24Division of Pediatric Neurosurgery, Children's Hospital of Pennsylvania, Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania.25Department of Neurosurgery, Penn State Milton S. Hershey Medical Center, Hershey, Pennsylvania.26Department of Neurological Surgery, University of Wisconsin at Madison, Wisconsin.27Department of Neurosurgery, Johns Hopkins University School of Medicine, Baltimore, Maryland.28Division of Pediatric Neurosurgery, University of Alabama at Birmingham, Alabama.29Department of Neurosurgery, Children's National Medical Center, Washington, DC.30Division of Pediatric Neurosurgery, Nationwide Children's Hospital, Columbus, Ohio.31Department of Neurosurgery, University of Michigan, Ann Arbor, Michigan.32Division of Pediatric Neurosurgery, Cincinnati Children's Medical Center, Cincinnati, Ohio.33Division of Pediatric Neurosurgery, Children's Hospital of Los Angeles, California.34Department of Neurological Surgery, Washington University in St. Louis School of Medicine, St. Louis, Missouri.35Department of Neurosurgery, University of Iowa Hospitals and Clinics, Iowa City, Iowa.36Department of Neurosurgery, Children's Hospital Colorado, Aurora, Colorado.37Division of Pediatric Neurosurgery, Arnold Palmer Hospital for Children, Orlando, Florida.34Department of Neurological Surgery, Washington University in St. Louis School of Medicine, St. Louis, Missouri.38Department of Neurological Surgery, University of Miami Miller School of Medicine, Miami, Florida.39Department of Neurological Surgery and Doernbecher Children's Hospital, Oregon Health & Science University, Portland, Oregon.40Division of Pediatric Neurosurgery, McGovern Medical School, Houston, Texas.34Department of Neurological Surgery, Washington University in St. Louis School of Medicine, St. Louis, Missouri.41Division of Pediatric Neurosurgery, Boston Children's Hospital, Boston, Massachusetts.34Department of Neurological Surgery, Washington University in St. Louis School of Medicine, St. Louis, Missouri.42Carolina Neurosurgery & Spine Associates, Charlotte, North Carolina; and.2Surgical Outcomes Center for Kids, Monroe Carell Jr. Children's Hospital of Vanderbilt University, Nashville, Tennessee. 9Division of Pediatric Neurosurgery, Monroe Carell Jr. Children's Hospital of Vanderbilt University, Nashville, Tennessee.43Division of Pediatric Neurosurgery, Texas Children's Hospital, Houston, Texas.2Surgical Outcomes Center for Kids, Monroe Carell Jr. Children's Hospital of Vanderbilt University, Nashville, Tennessee. 9Division of Pediatric Neurosurgery, Monroe Carell Jr. Children's Hospital of Vanderbilt University, Nashville, Tennessee.34Department of Neurological Surgery, Washington University in St. Louis School of Medicine, St. Louis, Missouri.No affiliation info available

Pub Type(s)

Journal Article

Language

eng

PubMed ID

32114543

Citation

Hale, Andrew T., et al. "Factors Associated With Syrinx Size in Pediatric Patients Treated for Chiari Malformation Type I and Syringomyelia: a Study From the Park-Reeves Syringomyelia Research Consortium." Journal of Neurosurgery. Pediatrics, 2020, pp. 1-11.
Hale AT, Adelson PD, Albert GW, et al. Factors associated with syrinx size in pediatric patients treated for Chiari malformation type I and syringomyelia: a study from the Park-Reeves Syringomyelia Research Consortium. J Neurosurg Pediatr. 2020.
Hale, A. T., Adelson, P. D., Albert, G. W., Aldana, P. R., Alden, T. D., Anderson, R. C. E., Bauer, D. F., Bonfield, C. M., Brockmeyer, D. L., Chern, J. J., Couture, D. E., Daniels, D. J., Durham, S. R., Ellenbogen, R. G., Eskandari, R., George, T. M., Grant, G. A., Graupman, P. C., Greene, S., ... Limbrick, D. D. (2020). Factors associated with syrinx size in pediatric patients treated for Chiari malformation type I and syringomyelia: a study from the Park-Reeves Syringomyelia Research Consortium. Journal of Neurosurgery. Pediatrics, 1-11. https://doi.org/10.3171/2020.1.PEDS19493
Hale AT, et al. Factors Associated With Syrinx Size in Pediatric Patients Treated for Chiari Malformation Type I and Syringomyelia: a Study From the Park-Reeves Syringomyelia Research Consortium. J Neurosurg Pediatr. 2020 Mar 6;1-11. PubMed PMID: 32114543.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Factors associated with syrinx size in pediatric patients treated for Chiari malformation type I and syringomyelia: a study from the Park-Reeves Syringomyelia Research Consortium. AU - Hale,Andrew T, AU - Adelson,P David, AU - Albert,Gregory W, AU - Aldana,Philipp R, AU - Alden,Tord D, AU - Anderson,Richard C E, AU - Bauer,David F, AU - Bonfield,Christopher M, AU - Brockmeyer,Douglas L, AU - Chern,Joshua J, AU - Couture,Daniel E, AU - Daniels,David J, AU - Durham,Susan R, AU - Ellenbogen,Richard G, AU - Eskandari,Ramin, AU - George,Timothy M, AU - Grant,Gerald A, AU - Graupman,Patrick C, AU - Greene,Stephanie, AU - Greenfield,Jeffrey P, AU - Gross,Naina L, AU - Guillaume,Daniel J, AU - Heuer,Gregory G, AU - Iantosca,Mark, AU - Iskandar,Bermans J, AU - Jackson,Eric M, AU - Johnston,James M, AU - Keating,Robert F, AU - Leonard,Jeffrey R, AU - Maher,Cormac O, AU - Mangano,Francesco T, AU - McComb,J Gordon, AU - Meehan,Thanda, AU - Menezes,Arnold H, AU - O'Neill,Brent, AU - Olavarria,Greg, AU - Park,Tae Sung, AU - Ragheb,John, AU - Selden,Nathan R, AU - Shah,Manish N, AU - Smyth,Matthew D, AU - Stone,Scellig S D, AU - Strahle,Jennifer M, AU - Wait,Scott D, AU - Wellons,John C, AU - Whitehead,William E, AU - Shannon,Chevis N, AU - Limbrick,David D, AU - ,, Y1 - 2020/03/06/ PY - 2019/08/23/received PY - 2020/01/07/accepted PY - 2020/3/2/entrez PY - 2020/3/3/pubmed PY - 2020/3/3/medline KW - AP = anteroposterior KW - CM-I = Chiari malformation type I KW - CM-I+SM = CM-I with SM KW - CPH = Cox proportional hazards KW - CXA = clivus canal angle KW - Chiari malformation type I KW - FOHR = frontal-occipital horn ratio KW - PFD = posterior fossa decompression KW - PFDD = PFD with duraplasty KW - PRSRC = Park-Reeves Syringomyelia Research Consortium KW - Park-Reeves Syringomyelia Research Consortium KW - SM = syringomyelia KW - pB-C2 distance = distance from the ventral dura perpendicular to the line between the basion and C2 KW - syrinx SP - 1 EP - 11 JF - Journal of neurosurgery. Pediatrics JO - J Neurosurg Pediatr N2 - OBJECTIVE: Factors associated with syrinx size in pediatric patients undergoing posterior fossa decompression (PFD) or PFD with duraplasty (PFDD) for Chiari malformation type I (CM-I) with syringomyelia (SM; CM-I+SM) are not well established. METHODS: Using the Park-Reeves Syringomyelia Research Consortium registry, the authors analyzed variables associated with syrinx radiological outcomes in patients (< 20 years old at the time of surgery) with CM-I+SM undergoing PFD or PFDD. Syrinx resolution was defined as an anteroposterior (AP) diameter of ≤ 2 mm or ≤ 3 mm or a reduction in AP diameter of ≥ 50%. Syrinx regression or progression was defined using 1) change in syrinx AP diameter (≥ 1 mm), or 2) change in syrinx length (craniocaudal, ≥ 1 vertebral level). Syrinx stability was defined as a < 1-mm change in syrinx AP diameter and no change in syrinx length. RESULTS: The authors identified 380 patients with CM-I+SM who underwent PFD or PFDD. Cox proportional hazards modeling revealed younger age at surgery and PFDD as being independently associated with syrinx resolution, defined as a ≤ 2-mm or ≤ 3-mm AP diameter or ≥ 50% reduction in AP diameter. Radiological syrinx resolution was associated with improvement in headache (p < 0.005) and neck pain (p < 0.011) after PFD or PFDD. Next, PFDD (p = 0.005), scoliosis (p = 0.007), and syrinx location across multiple spinal segments (p = 0.001) were associated with syrinx diameter regression, whereas increased preoperative frontal-occipital horn ratio (FOHR; p = 0.007) and syrinx location spanning multiple spinal segments (p = 0.04) were associated with syrinx length regression. Scoliosis (HR 0.38 [95% CI 0.16-0.91], p = 0.03) and smaller syrinx diameter (5.82 ± 3.38 vs 7.86 ± 3.05 mm; HR 0.60 [95% CI 0.34-1.03], p = 0.002) were associated with syrinx diameter stability, whereas shorter preoperative syrinx length (5.75 ± 4.01 vs 9.65 ± 4.31 levels; HR 0.21 [95% CI 0.12-0.38], p = 0.0001) and smaller pB-C2 distance (6.86 ± 1.27 vs 7.18 ± 1.38 mm; HR 1.44 [95% CI 1.02-2.05], p = 0.04) were associated with syrinx length stability. Finally, younger age at surgery (8.19 ± 5.02 vs 10.29 ± 4.25 years; HR 1.89 [95% CI 1.31-3.04], p = 0.01) was associated with syrinx diameter progression, whereas increased postoperative syrinx diameter (6.73 ± 3.64 vs 3.97 ± 3.07 mm; HR 3.10 [95% CI 1.67-5.76], p = 0.003), was associated with syrinx length progression. PFD versus PFDD was not associated with syrinx progression or reoperation rate. CONCLUSIONS: These data suggest that PFDD and age are independently associated with radiological syrinx improvement, although forthcoming results from the PFDD versus PFD randomized controlled trial (NCT02669836, clinicaltrials.gov) will best answer this question. SN - 1933-0715 UR - https://www.unboundmedicine.com/medline/citation/32114543/Factors_associated_with_syrinx_size_in_pediatric_patients_treated_for_Chiari_malformation_type_I_and_syringomyelia:_a_study_from_the_Park_Reeves_Syringomyelia_Research_Consortium_ DB - PRIME DP - Unbound Medicine ER -
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