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

被引:29
作者
Hale, Andrew T. [1 ,2 ]
Adelson, P. David [3 ]
Albert, Gregory W. [4 ]
Aldana, Philipp R. [5 ]
Alden, Tord D. [6 ]
Anderson, Richard C. E. [7 ]
Bauer, David F. [8 ]
Bonfield, Christopher M. [2 ,9 ]
Brockmeyer, Douglas L. [10 ]
Chern, Joshua J. [11 ]
Couture, Daniel E. [12 ]
Daniels, David J. [13 ]
Durham, Susan R. [14 ]
Ellenbogen, Richard G. [15 ]
Eskandari, Ramin [16 ]
George, Timothy M. [17 ]
Grant, Gerald A. [18 ]
Graupman, Patrick C. [19 ]
Greene, Stephanie [20 ]
Greenfield, Jeffrey P. [21 ]
Gross, Naina L. [22 ]
Guillaume, Daniel J. [23 ]
Heuer, Gregory G. [24 ]
Iantosca, Mark [25 ]
Iskandar, Bermans J. [26 ]
Jackson, Eric M. [27 ]
Johnston, James M. [28 ]
Keating, Robert F. [29 ]
Leonard, Jeffrey R. [30 ]
Maher, Cormac O. [31 ]
Mangano, Francesco T. [32 ]
McComb, J. Gordon [33 ]
Meehan, Thanda [34 ]
Menezes, Arnold H. [35 ]
O'Neill, Brent [36 ]
Olavarria, Greg [37 ]
Park, Tae Sung [34 ]
Ragheb, John [38 ]
Selden, Nathan R. [39 ,40 ]
Shah, Manish N. [41 ]
Smyth, Matthew D. [34 ]
Stone, Scellig S. D. [42 ]
Strahle, Jennifer M. [34 ]
Wait, Scott D. [43 ]
Wellons, John C. [2 ,9 ]
Whitehead, William E. [44 ]
Shannon, Chevis N. [2 ,9 ]
Limbrick, David D., Jr. [34 ]
机构
[1] Vanderbilt Univ, Sch Med, Med Scientist Training Program, Nashville, TN 37212 USA
[2] Vanderbilt Univ, Monroe Carell Jr Childrens Hosp, Surg Outcomes Ctr Kids, 221 Kirkland Hall, Nashville, TN 37235 USA
[3] Phoenix Childrens Hosp, Barrow Neurol Inst, Div Pediat Neurosurg, Phoenix, AZ USA
[4] Arkansas Childrens Hosp, Div Neurosurg, 800 Marshall St, Little Rock, AR 72202 USA
[5] Univ Florida, Coll Med, Div Pediat Neurosurg, Jacksonville, FL USA
[6] Ann & Robert H Lurie Childrens Hosp Chicago, Div Pediat Neurosurg, Chicago, IL 60611 USA
[7] Columbia Presbyterian, Childrens Hosp New York, Dept Neurol Surg, Div Pediat Neurosurg, New York, NY USA
[8] Dartmouth Hitchcock Med Ctr, Dept Neurosurg, Lebanon, NH 03766 USA
[9] Vanderbilt Univ, Monroe Carell Jr Childrens Hosp, Div Pediat Neurosurg, 221 Kirkland Hall, Nashville, TN 37235 USA
[10] Primary Childrens Med Ctr, Div Pediat Neurosurg, Salt Lake City, UT USA
[11] Childrens Healthcare Atlanta Univ, Div Pediat Neurosurg, Atlanta, GA USA
[12] Wake Forest Univ, Bowman Gray Sch Med, Dept Neurol Surg, Winston Salem, NC USA
[13] Mayo Clin, Dept Neurosurg, Rochester, MN USA
[14] Univ Vermont, Dept Neurosurg, Burlington, VT USA
[15] Seattle Childrens Hosp, Div Pediat Neurosurg, Seattle, WA USA
[16] Med Univ South Carolina, Dept Neurosurg, Charleston, SC 29425 USA
[17] Dell Childrens Med Ctr, Div Pediat Neurosurg, Austin, TX USA
[18] Lucile Packard Childrens Hosp, Div Pediat Neurosurg, Palo Alto, CA USA
[19] Gillette Childrens Hosp, Div Pediat Neurosurg, St Paul, MN USA
[20] Univ Pittsburgh, Childrens Hosp Pittsburgh, Med Ctr, Div Pediat Neurosurg, Pittsburgh, PA 15213 USA
[21] NewYork Presbyterian Hosp, Weill Cornell Med Coll, Dept Neurol Surg, New York, NY USA
[22] Univ Oklahoma, Hlth Sci Ctr, Dept Neurosurg, Oklahoma City, OK USA
[23] Univ Minnesota, Sch Med, Dept Neurosurg, Minneapolis, MN 55455 USA
[24] Univ Penn, Childrens Hosp Penn, Perelman Sch Med, Div Pediat Neurosurg, Philadelphia, PA 19104 USA
[25] Penn State Milton S Hershey Med Ctr, Dept Neurosurg, Hershey, PA USA
[26] Univ Wisconsin, Dept Neurol Surg, Madison, WI USA
[27] Johns Hopkins Univ, Sch Med, Dept Neurosurg, Baltimore, MD 21205 USA
[28] Univ Alabama Birmingham, Div Pediat Neurosurg, Birmingham, AL USA
[29] Childrens Natl Med Ctr, Dept Neurosurg, Washington, DC 20010 USA
[30] Nationwide Childrens Hosp, Div Pediat Neurosurg, Columbus, OH USA
[31] Univ Michigan, Dept Neurosurg, Ann Arbor, MI 48109 USA
[32] Cincinnati Childrens Med Ctr, Div Pediat Neurosurg, Cincinnati, OH USA
[33] Childrens Hosp Los Angeles, Div Pediat Neurosurg, Los Angeles, CA 90027 USA
[34] Washington Univ, Sch Med, Dept Neurol Surg, St Louis, MO USA
[35] Univ Iowa Hosp & Clin, Dept Neurosurg, Iowa City, IA 52242 USA
[36] Childrens Hosp Colorado, Dept Neurosurg, Aurora, CO USA
[37] Arnold Palmer Hosp Children, Div Pediat Neurosurg, Orlando, FL USA
[38] Univ Miami, Miller Sch Med, Dept Neurol Surg, Miami, FL 33136 USA
[39] Oregon Hlth & Sci Univ, Dept Neurol Surg, Portland, OR 97201 USA
[40] Oregon Hlth & Sci Univ, Doernbecher Childrens Hosp, Portland, OR 97201 USA
[41] McGovern Med Sch, Div Pediat Neurosurg, Houston, TX USA
[42] Boston Childrens Hosp, Div Pediat Neurosurg, Boston, MA USA
[43] Carolina Neurosurg & Spine Associates, Charlotte, NC USA
[44] Texas Childrens Hosp, Div Pediat Neurosurg, Houston, TX 77030 USA
基金
美国国家卫生研究院;
关键词
Chiari malformation type I; syrinx; Park-Reeves Syringomyelia Research Consortium; POSTERIOR-FOSSA DECOMPRESSION; DURA-SPLITTING-DECOMPRESSION; SURGICAL-TREATMENT; SUBOCCIPITAL DECOMPRESSION; DURAPLASTY; CHILDREN; OUTCOMES;
D O I
10.3171/2020.1.PEDS19493
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
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 0.160.91], 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.
引用
收藏
页码:629 / 639
页数:11
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