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EDITOR D. LUKE GLANCY, MD
VOLUME 170 NUMBER 1 • JANUARY | FEBRUARY 2018
ESTABLISHED 1844
ASSOCIATE EDITOR L.W. JOHNSON, MD
OF THE LOUISIANA STATE MEDICAL SOCIETY JOURNAL
BOARD OF TRUSTEES CHAIR, GEOFFREY W. GARRETT, MD VICE CHAIR, K. BARTON FARRIS, MD SECRETARY/TREASURER, RICHARD PADDOCK, MD ANTHONY P. BLALOCK, MD D. LUKE GLANCY, MD LESTER W. JOHNSON, MD FRED A. LOPEZ, MD
EDITORIAL BOARD MURTUZA J. ALI, MD RONALD AMEDEE, MD SAMUEL ANDREWS, II, MD BOB BATSON, MD EDWIN BECKMAN, MD GERALD S. BERENSON, MD
FEATURED ARTICLES
C. LYNN BESCH, MD JOHN BOLTON, MD BRIAN BOULMAY, MD MICHELLE BOURQUE, JD JAMES N. BRAWNER, III, MD BRETT CASCIO, MD QUYEN CHU, MD WILLIAM PATRICK COLEMAN III, MD RICHARD COULON, MD LOUIS CUCINOTTA, MD VINCENT A. CULOTTA, JR., MD JOSEPH DALOVISIO, MD NINA DHURANDHAR, MD JAMES DIAZ, MD, MPH & TM, D r . PH JOHN ENGLAND, MD JULIO FIGUEROA, MD ELIZABETH FONTHAM, MPH, D r . PH EDWARD FOULKS, MD BEN GUIDER, MD
ASSESSING PSYCHOLOGICAL RESILIENCE AMONG PRE-SURGERY PLIF PATIENTS IN LOUISIANA: PSYCHOMETRIC EVALUATION OF THE BRIEF RESILIENT COPING SCALE Scott Wilks, PhD, Stephen Guillory, PA-C, Jennifer Geiger, MSW, Kevin Goodson, MD, Tatiana Begault, BS, Zibei Chen, MSW, Jorge Isaza, MD COMBINED SURGICAL ONCOLOGY AND NEUROSURGERY APPROACH FOR RESECTION OF SACRAL CHORDOMA Racheal Wolfson, MD, Richard Menger, MD, MPA, Ouyen Dinh Chu, MD, MBA, Anthony Sin, MD INTRACRANIAL SEPTUM PELLUCIDUM DYSEMBRYOPLASTIC NEUROEPITHELIAL TUMOR: CASE PRESENTATION AND REVIEW OF PEDIATRIC SEPTUM PELLUCIDUM TUMORS
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HENRY G. HANLEY, MD ELIAS B. HANNA, MD LYNN H. HARRISON, JR., MD ROBERT HEWITT, MD
Rimal Dossani, MD, Devi Patra, MD, Nimer Adeeb, MD, Elizabeth Wild, MD, Abhilasha Ghildyal, MD, Marjorie Fowler, MD, Christina Notarianni, MD
MICHAEL HILL, MD LARRY HOLLIER, MD JOHN HUNT, MD BERNARD JAFFE, MD NEERAJ JAIN, MD
NEWBORN SCREENING FOR CONGENITAL ADRENAL HYPERPLASIA: REVIEW OF UNDETECTED CASES IN LOUISIANA Dania Felipe, MD, Joseph Ortenberg, MD, Ricardo Gomez, MD, Aaron Martin, MD, MPH, Robin Ortenburg, MD, Michael Marble, MD
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TRENTON L. JAMES, II, MD STEPHEN KANTROW, MD KEVIN KRANE, MD MAUREEN LICHTVELD, MD, MPH FRED A. LOPEZ, MD F. BROBSON LUTZ, JR., MD DAVID MARTIN, MD JORGE A. MARTINEZ, MD, JD ELIZABETH MCBURNEY, MD ELLEN MCLEAN, MD REINHOLD MUNKER, MD DAVID MUSHATT, MD STEVE NELSON, MD NORA OATES, MD DONALD PALMISANO, MD, JD, FACS PATRICK W. PEAVY, MD PAUL PERKOWSKI, MD PETERMAN RIDGE PROSSER, MD ROBERTO QUINTAL, MD RAOULT RATARD, MD, MS, MPH & TM ROBERT RICHARDS, MD
STRONGYLOIDIASIS: A TICKING TIME BOMB IN VIETNAMWAR VETERANS James Diaz, MD
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DEPARTMENTAL ARTICLES
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CLINICAL CASE OF THE MONTH NEW ONSET HYPERTENSION AND DIABETES IN A 24 YEAR-OLD MAN Elizabeth Smith, MD, Catherine Pisano, Robert Richards, MD, Taniya DeSilva, MD, Fred Lopez, MD ECG CASE OF THE MONTH CYANOTIC CONGENITAL HEART DISEASE AND DIARRHEA IN A 42-YEAR-OLD MAN D. Luke Glancy, MD, Chun Tan, MD, and Frederick Helmcke, MD
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DONALD RICHARDSON, MD WILLIAM C. ROBERTS, MD DONNA RYAN, MD JERRY ST. PIERRE, MD CHARLES SANDERS, MD
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OLIVER SARTOR, MD CHARLES SCHER, MD ROGER SMITH, MD RICHARD SPECTOR, MD LEE STEVENS, MD JACK P. STRONG, MD PRAMILLA N. SUBRAMANIAM, MD KEITH VAN METER, MD DIANA VEILLON, MD HECTOR VENTURA, MD
RADIOLOGY CASE OF THE MONTH HYPOVOLEMIC SHOCK COMPLEX Logan Bisset, MD, Justine Kemp, BS, Jeremy Nguyen, MD
CHRIS WINTERS, MD GAZI B. ZIBARI, MD
JOURNAL OF THE LOUISIANA STATE MEDICAL SOCIETY
Assessing Psychological Resilience among Pre-Surgery PLIF Patients in Louisiana: Psychometric Evaluationof theBrief Resilient CopingScale
Scott Wilks, PhD, Stephen Guillory, PA-C, Jennifer Geiger, MSW, Kevin Goodson, MD, Tatiana Begault, BS, Zibei Chen, MSW, Jorge Isaza, MD
Thepurposeof this studywas toexaminepsychometricproperties of theBrief ResilienceCopingScale (BRCS), ameasure of psychological resilience, to determine its applicability among pre-spine surgery patients. The sample comprised 85 adults in south Louisiana scheduled for posterior lumbar interbody fusion (PLIF). We utilized a prospective survey design with independent self-report data one week pre-surgery. Empirical measures included the BRCS, Oswestry Index (back functioning), and three coping strategy scales; the latter measures for descriptive and validity purposes. The typical participant was a 65-year-old married, Caucasian male; no histories of diabetes, heart disease, smoking; moderate back functioning yet a high degree of resilience. BRCS factor analysis revealed its items loading on a single factor, presumably resilience. BRCS reliability was strong. Validity findings were somewhat mixed. The BRCS appears to be an applicable measure of resilience for pre-spine surgery patients, though further research is warranted to endorse BRCS validity.
METHODS
INTRODUCTION
Psychological resilience is a personal characteristic of adaptation and adjustment when facing hardship, trauma, or substantial exposure to stress. Research has shown resilience to be valuable not only for psychological health (e.g., mood, emotions, cognition), but also for immediate and long term physical health outcomes, including AIDS mortality, cardiovascular disease and stroke. 1 The physical health outcome in the current study potentially applicable to resilience chronic back pain, specifically low back pain (LBP). Literature distinguishes resilience as a psychosocial predictor of functionality germane to LBP, as well as healthcare utilization and frequency of such related to chronic LBP. 2 Regarding surgical treatment for LBP, the number of spine fusions in the U.S. have trended dramatically upward over the past two decades; lumbar fusions as the majority at 52%. 3 Given the aforementioned identification of resilience as a predictor of healthcare utilization for LBP and the increasing numbers of lumbar fusions for treatment of LBP, we recognized the relevance of understanding psychological resilience for LBP patients and, equally important, understanding standardized measures to assess said resilience. The purpose of the current study was to conduct an empirical psychometric assessment of a standardized measure of psychological resilience – the Brief Resilient Coping Scale (BRCS). 4 This allowed us to assess descriptively the level of psychological resilience of persons seeking LBP treatment while simultaneously testing the psychometric properties of the BRCS. The sample consisted of LBP patients in Louisiana scheduled for posterior lumbar interbody fusion (PLIF).
Design and Sampling
The current study utilized a prospective survey design with self- reported data. Pre-surgery was the selected data interval based on a premise of resilience theory: For resilience to be observed, demonstrable adversity must be present. 5 Logic and research dictate that patients who undergo surgery typically have higher physical and psychological health adversity levels at pre-surgery compared to post-surgery. 6 Accordingly, we deemed pre- surgery as the optimal period to assess patients’ resilience and to evaluate the resilience measure. One week prior to scheduled posterior lumbar interbody fusion (PLIF), adult participants completed questionnaires, independently self-reported in surgeons’ offices, related to demographics, psychological coping, and a physical back health measure (see measures). Participants were under the care of one of two orthopedic surgeons fellowship trained in spine surgery; these surgeons practiced at one orthopedic clinic in south Louisiana. Numerical codes were assigned to each participant to ensure anonymity. Relevant institutional review boards, academic and clinic, approved the study, thereby protecting HIPAA compliance. Eighty-five patients (N = 85) voluntarily completed all measures and comprised the total sample.
Measures
Demographics and health history . The following demographics were identified in literature as potentially influential to spine/spine surgery health, and thus examined: age, ethnicity, marital status, and insurance/payment
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type. 7-9 Also, the following health risk factors were observed as they were commonly noted by the surgeons prior to surgery: histories of diabetes, heart disease, and smoking. Psychological resilience. The measure under psychometric focus was the 4-item Brief Resilient Coping Scale (BRCS). 4 Content related to creativity to address adversity; locus of control regarding reaction to adversity; personal growth post-adversity; and replacement of loss. The measure contained a 4-point Likert response format ranging from strongly disagree to strongly agree in belief of aforementioned items. Possible global scores in this study ranged from 4-16, with higher scores indicating greater psychological resilience. Psychometric properties were reported as adequate-to-strong in the original evaluation among a sample of rheumatoid arthritis patients. 4 Coping . For validity testing with the BRCS, three specific coping strategy measures were included in the study. These coping scales came from the Proactive Coping Inventory (PCI). 10 Each coping strategy scale contains a 4-point Likert response format ranging from 1 – not at all true to 4 – completely true ; higher global scores represent greater likelihood to utilize the specific coping strategy during adversity. All coping scales have been previously reported as having, at minimum, satisfactory psychometric properties. 10 A brief description of each coping strategy scale is shown below: 10 • proaction . The 14-item Proactive Coping Scale assesses likelihood to cope via goal setting with thoughts and behaviors geared toward self-regulated attainment. Possible global scores range from 14-56. • reflection . The 11-item Reflective Coping Scale assesses likelihood to cope via contemplation of possible alternatives, imaginingeffectiveness, andcreatingplansof actionaccordingly. Possible global scores range from 11-44. • avoidance . The 3-item Avoidance Coping Scale assesses one’s likelihood to cope via delaying or eluding action. Possible global scores range from 3-12. Back functioning . A physical health measure relating to back functioningwas included, again for validity testingwith theBRCS. The Oswestry Disability Index (ODI) 11 based on its popularity and repeatedly proven, robust psychometrics, is considered the gold standard of low back pain and functionality assessment. The ODI evaluates ability to manage everyday activities in 10 general areas (e.g., sitting, standing, walking, sleeping) in light of back pain and related leg pain. Item scores from each area were summed to obtain global scores, potentially ranging from 0-50. Lower ODI scores reflect greater level of back functioning. Analytic Plan. Descriptive statistics were examined on all measures. Principal components analysis identified the underlying BRCS factor structure. Factor analysis was initially rotated to a varimax solution with no limitation on number of factors. Identification of a factor was based on the customary, minimum eigenvalue of 1.0. Minimum factor loading threshold for scale item retention was set at 0.40 with the minimum
Sample Characterisitics
Variable Age Gender Female Male Ethnicity
Valid % Mean (SD, range) n
65.0 (10.25, 36-89)
36.4 64.5
31 54
Caucasian/White African American/Black American Indian
84.7 14.1 1.2
72 12 1
Marital Status Married Divorced Widowed
76.5 15.3
65 13
3.5 2.4 2.4
3 2 2
Common Health Risks Never married Cohabitating Payment/Insurance Type Private Medicare Worker’s Comp. Attorney Other
60.0 20.0 16.5
51 17 14
2.4 1.2
2 1
Back functioning (ODI) ODI: Oswestry Disability Index Diabetes history (no) Heart disease history (no) Smoking history (no)
78.8. 92.9 70.6
67 79 60
26.0 (6.32, 10-43)
Table 1: ODI: Oswestry Disability Index
loading value identified as follows: 0.32 as poor ; 0.45 as fair ; 0.55 as good ; 0.63 as very good ; or 0.71 as excellent . 12 Reliability on the BRCS was observed via Cronbach’s alpha and Guttman coefficients. For Guttman, the split-half coefficients (lambda-4 [λ-4]) and the highest of the lower bound lambdas (if not λ-4) were noted as the reliability estimate. Inter-item correlations also were observed for internal consistency. Validity of the BRCS was observed via Pearson’s r correlations, and directions of such, with each of its theoretically linked measures. Previous literature has shown psychological resilience to be positively (i.e., directly) linked with goal formulation and corresponding plans of action. 13 Accordingly, the BRCS was expected to demonstrate convergent validity as follows: (a) significantly and positively correlate with the Proactive Coping Scale and the Reflective Coping Scale; and (b) significantly but negatively (i.e., inversely) correlate with the Avoidance Coping
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becauseof the lone factor, the solution rotationwas unnecessary. This factor, which was identified assumptively as resilience , held an eigenvalue of 2.8 and explained almost 70% of the variance in BRCS responses. Each of the items loaded strongly with this factor. Table 2 details these findings. BRCS reliability. Reliability analysis on the BRCS revealed a Cronbach’s alpha reliability coefficient of 0.854. Guttman’s coefficient was similar at 0.884. This coefficient, λ-4, was the highest of the lower bound lambdas and thus noted as Guttman’s reliability estimate. Inter-item correlations among all pairings of the four BRCS items were moderate-to-strong, 16 as shown in Table 3. BRCS validity. Convergent validity was examined via zero- order correlations and their specific directions between the BRCS and measures of theoretically linked constructs of coping and back pain/functioning. The BRCS significantly, positively correlated with proactive coping and reflective coping; the BRCS negatively correlated, though not significantly, with avoidance coping and back functioning. These associations showed appropriate directions, per previously mentioned literature. 13,14 Table 4 displays these correlation coefficients relevant to BRCS convergent validity.
Scale. Previous literature has shown resilience to be negatively linked with physical pain. 14 Thus, the BRCS was expected to indicate further convergent validity via a negative correlation with the ODI. Significance ( p ) was observed at the standard 0.05 threshold. Missing Data . Missing values (cases x variables) were less than 1% for the original dataset. Since data absence per variable was less than 20%, single imputation was computed in SPSS. 15 Following calculation of root mean square error, we observed nominal differences in variance between original and imputed datasets. Accordingly, the analyses were conducted using the imputed dataset.
RESULTS
Descriptive Statistics
Sample characteristics . The mean age of sample participants was almost 65 years. The majority of the sample self-identified as male (64%), Caucasian/White (85%) and married (77%). The most common method of payment for scheduled PLIF was private pay (60%). In terms of common health risks to surgery, the majority of patients reported no histories of diabetes (79%), heart disease (93%), or smoking (71%). Table 1 details these sample characteristics. Standardized measures . The aggregate global scores on each of the standardized measures are as follows, beginning with the measure under psychometric focus: • BRCS (resilience): M = 12.6 (SD = 2.74, range: 4-16); • Proactive Coping Scale: M = 44.2 (SD = 6.33, range: 26-55); • Reflective Coping Scale: M = 33.9 (SD = 6.37, range: 11-44); • Avoidance Coping Scale: M = 6.8 (SD = 2.24, range: 3-12); and • ODI (back functioning): M = 26.0 (SD = 6.32, range: 10-43).
BRCS Reliability: Inter-Item Correlations
1
2
Item a. Creativity in addressing adversity b. Locus of control in reacting to adversity c. Personal growth post-adversity d. Replacement of loss
4
3
0.472* a 0.653* b 0.494* a --
0.658* b 0.692* b --
0.596* b --
--
* p < .01 Strength of association (Evans, 1996) a=moderate; b=strong
BRCS Factor Analysis Findings
Table 3: BRCS: Brief Resilient Coping Scale
Factor Loading
BRCS Item a. Creativity in addressing adversity b. Locus of control in reacting to adversity c. Personal growth post-adversity d. Replacement of loss Eigenvalue Variance explained
2.788 0.816* 0.887* 0.858* 0.776* 69.712%
DISCUSSION
Patients in this study reported a high level of psychological resilience, especially when considering their LBP prior to surgery. Regarding scale analysis, factor and reliability observations evidenced support for an internally consistent, single-factor scale for measuring resilience in pre-operative PLIF patients. As anticipated, the BRCS was shown to measure a single factor— resilience—with this factor accounting for a large proportion of the variance in BRCS scores. Relatively strong reliability coefficients across all analyses provided further support for the construct validity of the BRCS. In contrast, convergent validity findings were more mixed. As expected, the BRCS positively correlated with two adaptive coping strategies, proactive and reflective coping. Unexpectedly,
Table 2: BRCS: Brief Resilience Coping Scale * Loading on the factor valued as excellent (Tabachnick & Fidell, 2007) BRCS: Brief Resilience Coping Scale
Psychometric Findings
BRCS factor structure . Principal components analysis extracted a single underlying factor with an eigenvalue greater than 1.0;
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REFERENCES
BRCS Validity: Zero-Order Correlations with Theoretically Linked Measures
1. Tugade MM, Frederickson BL, Barrett LF. Psychological resilience and positive emotional granularity: Examining the benefits of positive emotions on coping and health. J Pers . 2004;72(6):1161-1190. doi:10.1111/ j.1467-6494.2004.00294.x 2. Seerya MD, Leob RJ, Holmand EA, Silvere RC. Lifetime exposure to adversity predicts functional impairment and healthcare utilization among individuals with chronic back pain. Pain . 2010;150(3):507–515. doi:10.1016/j.pain.2010.06.007 3. Andersson G, Watkins-Castillo SI. The burden of musculoskeletal diseases in the United States. The United States Bone and Joint Initiative Website. http://www.boneandjointburden.org/2014-report/iie1/spinal-fusion Accessed October, 5, 2016. 4. Sinclair VG, Wallston KA. The development and psychometric evaluation of the Brief Resilience Coping Scale. Assessment . 2004;11(1):94-101. doi: 10.1177/1073191103258144 5. MastenAS, Best KM, GarmezyN. Resilience anddevelopment: Contributions from the study of children who overcome adversity. Development and Psychopathology . 1990;2(4):425-444. doi: 10.1017/s0954579400005812 6. Wallace LM. Psychological preparation as a method of reducing the stress of surgery. J of Human Stress. 2010;10(2):62-77. doi: 10.1080/0097840X.1984.9934961 7. Schoenfeld AJ, Tipirneni R, Nelson JH, Carpenter JE, Iwashyna TJ. The influence of race and ethnicity on complications and mortality after orthopedic surgery: A systematic review of the literature. Medical Care. 2014;52(9):842-851. doi:10.1097/MLR.0000000000000177 8. Reisbord LS, Greenland S. Factors associated with self-reported back pain prevalence: A population-based study. Journal of Chronic Diseases. 1985;38(8):691-702. doi: 10.1016/0021-9681(85)90023-2 9. Dazenbrock HH, Wolinsky J, Sciubba DM, Witham TF, Gokaslan ZL, Bydon, A. The impact of insurance status on outcomes after surgery for spinal metastases. Cancer. 2012;118(19):4833-4841. doi: 10.1002/cncr.27388 10. Greenglass E, Schwarzer R, Jakubiec SD, Fiksenbaum L, Taubert S. The Proactive Coping Inventory (PCI): A multidimensional research instrument. Paper presented at: The 20th International Conference of the Stress and Anxiety Research Society (July, 1999); Krakow, Poland. http://estherg.info. yorku.ca/files/2014/09/pci.pdf Accessed October 10, 2016. 11. Fairbank JC, Pynsent PB. The Oswestry Disability Index. Spine. 2000;25(22):2940-2952. doi: 10.1097/00007632-200011150-00017 12. Tabachnick BG, Fidell LS. Using Multivariate Statistics . 5th ed. New York, NY: Pearson Education Inc; 2007. 13. Corcoran J, Nichols-Casebolt A. Risk and resilience ecological framework for assessment and goal formulation. Child and Adolesc Social Work J. 2004;21(3):211-235. doi: 10.1023/B:CASW.0000028453.79719.65 14. Ong AD, Zautra AJ, Reid MC. Psychological resilience predicts decreases in pain catastrophizing through positive emotions. Psychology and Aging. 2010;25(3):516-523. doi: 10.1037/a0019384. 15. Yuan Y. Multiple Imputation for Missing Data: Concepts and New Developments . Rockville, MD: SAS Institute; 2000. 16. Evans JD. Straightforward Statistics for the Behavioral Sciences . Pacific Grove, CA: Brooks/Cole;1996. 17. Beasley M, Thompson T, Davidson J. Resilience in response to life stress: The effects of coping style and cognitive hardiness. Personality & Individual Differences . 2003;34(1):77-95. doi: 10.1016/S0191-8869(02)00027-2 18. Chan C, Peng P, Failed Back Surgery Syndrome. Pain Medicine. 2011;12(4):577-606. doi:10.1111/j.1526-4637.2011.01089.x ScottWilks, PhD, is a FellowwithTheGerontological Society of America; Hartford Foundation Faculty Scholar in Geriatric Social Work; and associate professor with Louisiana State University (LSU) School of Social Work. Steve Guillory is a certified physician assistant with Spine Specialists of Louisiana, Baton Rouge, LA. Jen Geiger is a doctoral candidate with LSU School of Social Work. Kevin Goodson, MD, is an orthopedic surgeon with University of Arkansas for Medical Sciences –Orthopedic Surgery, Little Rock, AR. Tatiana Begault is an MSW student with LSU School of Social Work. Zibei Chen is a doctoral candidate with LSU School of SocialWork. Jorge Isaza, MD , is an orthopedic surgeon, fellowship trained in spine surgery, with Spine Specialists of Louisiana, Baton Rouge, LA.
Measure BRCS
PCS
RCS
ACS
ODI
0.580*
0.764*
-0.094
-0.203
however, scores on the BRCS did not significantly correlate with the maladaptive coping strategy (avoidance coping) or with back functioning. These results run contrary to conventional wisdom and previous research indicating significant negative links between resilience and avoidance coping 17 as well as resilience and chronic pain. 14 One possible explanation for the non-significant correlation between resilience and avoidance coping is in the differing conceptions of avoidance coping. The three avoidance coping items of the PCI used in the current study all focus on cognitive distraction (e.g., When I have a problem I like to sleep on it .), which has been shown in a previous study 14 to be associated with fewer symptoms of physical distress in women but not in men. At the same time, the authors also found that emotional avoidance (e.g., rumination) and social diversion (as opposed to social support) were associated with lower levels of resilience. Thus, the coping strategy of distraction may contribute in a limited way to resilience for certain subpopulations. Regarding the non-significant correlation between resilience and lower back functioning, the results may point to the limitations of adaptive coping and resilience in mitigating lower back disability. Every participant in the present study had experienced pain and disability severe and chronic enough to require PLIF. Thus, it may be the case that positive coping and resilience are more effective for individuals with acute pain and higher functioning. While much research has focused on treatment of post-surgical pain and disability, including Failed Back Surgery Syndrome, 18 fewer studies have explored the relationships among resilience, coping, LBP, and disability. Therefore, these results point to the importance of expanding the current study to include a more diverse set of patients in terms of pain and disability. In conclusion, we iterate the primary purpose of this study as a psychometric evaluation of the BRCS, a short resilience scale, for use with pre-surgical PLIF patients. While the scale showed strong construct validity and reliability of the scale, convergent validity findings were less conclusive. Further research is warranted to examine the theoretical underpinnings linking resilience with higher back functioning and less avoidance coping for those with chronic, severe back pain. * p < .01 Note: A positive coefficient represents a direct relationship with BRCS; negative coefficient represents an inverse relationship with BRCS. Table 4 : ACS: Avoidance Coping Scale; BRCS: Brief Resilient Coping Scale; ODI: Owestry Disability Index; PCS: Proactive Coping Scale; RCS: Reflective Coping Scale ACS: Avoidance Coping Scale BRCS: Brief Resilient Coping Scale ODI: Owestry Disability Index PCS: Proactive Coping Scale RCS: Reflective Coping Scale
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Combined Surgical Oncology and Neurosurgery Approach for Resection of Sacral Chordoma
Racheal Wolfson, MD, Richard Menger, MD, MPA, Ouyen Dinh Chu, MD, MBA, Anthony Sin, MD
Sacral chordomas are rare neoplasms of the sacral spine arising from remnants of the notochord. They are poorly responsive to chemotherapy and radiation therapy. The recommended treatment of sacral chordomas is en bloc resection with wide margins. We present a case of a 46-year-oldman with a sacral chordoma who underwent surgical resection of the mass. A combined anterior-posterior approachwas used, with a surgical oncologist freeing the pelvic cavity through an abdominal laparotomy followed by a neurosurgeon resecting the mass and fusing the lumbar spine to the pelvis posteriorly. The patient underwent a laparotomy the second day for exploration of the pelvis and final closure. The procedure was well tolerated the the patient retained ambulation postoperatively. Complete resection of sacral chordomas with wide margins offers the best prognosis for patients. Sacral chordomas should be referred to a tertiary care center with experience treating these lesions. Our center recommends a combined surgery-neurosurgery approach for safe resection of sacral chordomas.
INTRODUCTION
Chordomas are rare but malignant tumors arising from vestigial remnants of the embryological notochord. They account for 1.4% of all primary malignant bone tumors and 17% of primary bone tumors of the spine. 1 The incidence of chordomas is <0.1 per 100,000 people per year 2,3 and increases with age. 4 Chordomas are most commonly found within the sacrococcygeal area (40- 50%), followed by the skull base (35-40%) and the vertebral bodies (15-20%). 5 Patients often present with vague symptoms mimicking lumbosacral stenosis and advanced disease due to the slow growth of these tumors deep within the pelvis. 6 Because chordomas are poorly responsive to chemotherapy, the treatment modality of choice is radical resection. Local recurrence remains common (43-85%) even with complete resection with negative margins. 5 Sacrectomy with wide resection margins also carries a high risk of postoperative morbidity due to extensive nerve root and musculoskeletal resection leading to ambulatory, sexual, bowel, and urinary symptoms. 2 Despite this, wide resection margins provides the best long-term prognosis in terms of progression-free survival and thus the gold standard for surgery is en bloc resection of the chordoma. In sacral chordomas this can be achieved with a combined anterior-posterior approach for lesions extending above S3, whereas a posterior approach alone is usually sufficient for tumors below this level. 6 We present a patient who underwent resection of a sacral chordoma with a combined anterior-posterior approach with an interdisciplinary team of surgeons from surgical oncology and neurosurgery.
Figure 1 : Post-gadolinium T1-weighted MRI showing a contrast-enhancing sacral mass with infiltration into the spinal canal and pelvic cavity.
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5). The posterior incisionwas closed using bilateral advancement flaps to decrease tension on the abdominal wound. The patient remained intubated and was admitted to the ICU overnight. The following day the patient returned to the operating room with the surgical oncology team for planned exploratory laparotomy of the abdominal cavity in order to inspect the resection bed, achieve hemostasis, and final close the abdomen. The patient was extubated on post-operative day two and discharged to home on post-operative day 13. Postoperatively the patient was able to ambulate with a rolling walker but experienced persistent urinary incontinence and erectile dysfunction with perineal numbness. At one year follow-up, the patient continued to have urinary incontinence and radicular leg pain. His ambulation had improved and he was able to walk without assistive device with only a small limp. His surgical scar was well-healed (Figure 6).
CASE PRESENTATION
We present the case of a 46-year-old African American man who was referred to the surgical oncology clinic for findings of a large, rapidly expanding sacral mass on magnetic resonance imaging (MRI) of the lumbar spine obtained for lumbar radiculopathy and urinary retention (Figure 1). A biopsy of the lesion by interventional radiology was positive for chordoma. The patient elected for surgery and underwent resection of the mass with a combined surgical oncology and neurosurgery approach carried out over two days. The surgical oncologist started the resection with an abdominal laparotomy and dissection of the pelvis to free the pelvic viscera, iliac vessels, and ureters off the lateral pelvic walls and sacrum. Once these structures were mobilized, the abdomen was closed and the patient flipped into the prone position. The neurosurgeon then opened a midline lumbosacral incision and placed pedicle screws along L3-L5. The dissection exposed the sacral mass as well as the sacroiliac joints. The thecal sac and nerve roots were tied off at the level of S1 with sacrifice of the right S1 nerve root. An osteotome was then used to cut through the S1 vertebral body and the SI joints and the tumor was removed en bloc (Figures 2 and 3). Bilateral iliac screws were placed, three in each iliac crest. Vertical rods were run from L3- L5 and extended to attach to the last pair of iliac screws. Two horizontal rods were used to connect the top two pairs of iliac screws with a fibula strut wired between the rods (Figures 4 and
DISCUSSION
While sacral chordomas are rare, they present a unique challenge to the treating physician. Their insidious presentation requires careful attention to the patient’s symptoms, with the diagnosis often being made based on imaging workup for lumbar radiculopathy, incontinence, or erectile dysfunction. Often the symptoms are non-specific and the diagnosis is made after the tumor has grown to a large size, rendering the resection of such tumors more difficult. In contrast to osteosarcomas or
Figure 2 : Posterior view of the pelvis after removal of sacral chordoma and ligation of thecal sac and nerve roots. The abdominal packing placed during the anterior part of the procedure is visible.
Figure 3 : Gross pathology specimen resected en bloc.
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chondrosarcomas, chordomas locally invade the intervertebral disc space as they spread, which can be visualized on computed tomography (CT) or MRI. Calcifications are present in 30-70% of chordomas. They are iso- or hypo-intense compared to muscle on T1 and hyperintense on T2-weighted MRI images. Definitive diagnosis is made by core needle biopsy. 5 Once a sacral chordoma is diagnosed, the patient should be referred to a tertiary care center for evaluation by experienced surgeons. The main prognostic factor across several studies is the extent of resection. 1-3,7,8 Local recurrence remains common but is affected by the resection margins. Margin classifications are based on the classifications by Enneking in 1980. A wide margin involves resection of the surrounding healthy tissue without entering the reactive zone surrounding the tumor, whereas a marginal margin extends through the reactive zone, and an intralesional margin enters the tumor. 9 With a wide margin the rate of local recurrence is 5-17%, compared to 71-81% for intralesional or marginal resection margins. Another potential complication is tumor seeding, which can occur due to intraoperative violation of the tumor capsule and can potentially increase the rate of local recurrence. 8 Seeding can also occur from tumor biopsy, therefore every effort should be made to resect the biopsy tract during tumor resection. The choice of surgical approach will be largely dictated by the experience of the surgeon(s) involved. Some centers advocate the use of the anterior-posterior combined approach, while others have foundnodifference in outcomes using the posterior- only approach regardless of tumor level. 3 A combined approach Figure 4 : Posterior lumbo-iliac fixation. The lumbar pedicle screws were connected to the most inferior pair of iliac screws with vertical rods. The superior two pairs of iliac screws were connected with horizontal rods with a fibular strut wired between the rods for additional pelvic stabilization.
allows for careful dissection of the pelvic neurovascular structures, especially when the tumor extends high into the pelvis. 8 The morbidity associated with extensive pelvic resection is generally quite high due to sacrifice of involved nerve roots and extensive muscular dissection. In our patient, using the combined approach allowed for a well-controlled resection with preserved ambulation using a walker, but at the expense of urinary continence. Classical chemotherapy has not been proven to be effective in these tumors; however there are some anecdotal reports of slowed tumoral progression with cisplatin, anthracycline, and alkylating agents. Current studies are aimed at identifying and targeting specific receptors expressed by the chordoma, with promising results from tyrosine kinase inhibitors such as imatinib and sumitinib. Cetuximab, gefitinib, and erlotinib target the epidermal growth factor pathway expressed by these tumors. Use of radiotherapy is controversial, and the radio- sensitivity of surrounding structures such as bladder, small bowel, and sacral plexus limits the extent of radiation therapy. 5 Recent studies have shown a statistically significant increase in overall survival and progression-free survival for patients who undergo total resection followed by proton beam therapy compared to patients with incomplete resection with or without proton therapy, indicating that surgical resection remains the most important variable. 1 Figure 5 : Postoperative x-ray demonstrating instrumentation from L3 to iliac crests. Vertical rods run along L3 to L5 and attach to the bottom-most pair of iliac screws. Horizontal rods connect the remaining two pairs of iliac screws with a fibular strut placed horizontally and wired to the two rods.
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REFERENCES
1. George B, Bresson D, Herman P, Froelich S. Chordomas: A Review. Neurosurg Clin N Am . 2015;26(3):437-452. 2. Kayani B, Sewell MD, Hanna SA, et al. Prognostic factors in the operative management of dedifferentiated sacral chordomas. Neurosurgery. 2014;75(3):269-275; discussion 275. 3. Ruosi C, Colella G, Di Donato SL, Granata F, Di Salvatore MG, Fazioli F. Surgical treatment of sacral chordoma: survival and prognostic factors. Eur Spine J . 2015;24 Suppl 7:912-917. 4. McMaster ML, Goldstein AM, Bromley CM, Ishibe N, Parry DM. Chordoma: incidence and survival patterns in the United States, 1973-1995. Cancer Causes Control . 2001;12(1):1-11. 5. Garofalo F, di Summa PG, Christoforidis D, et al. Multidisciplinary approach of lumbo-sacral chordoma: From oncological treatment to reconstructive surgery. J Surg Oncol. 2015;112(5):544-554. 6. Kayani B, Hanna SA, Sewell MD, Saifuddin A, Molloy S, Briggs TW. A review of the surgical management of sacral chordoma. Eur J Surg Oncol. 2014;40(11):1412-1420. 7. Angelini A, Pala E, Calabro T, Maraldi M, Ruggieri P. Prognostic factors in surgical resection of sacral chordoma. J Surg Oncol. 2015;112(4):344-351. 8. Ruggieri P, Angelini A, Ussia G, Montalti M, Mercuri M. Surgical margins and local control in resection of sacral chordomas. Clin Orthop Relat Res. 2010;468(11):2939-2947. 9. Enneking WF, Spanier SS, Goodman MA. A system for the surgical staging of musculoskeletal sarcoma. Clin Orthop Relat Res . 1980(153):106-120. Racheal Wolfson, MD, is a resident at LSUHSC-Shreveport, Department of Neurosurgery. Richard Menger, MD, MPA, is chief resident at LSUHSC- Shreveport, Department of Neurosurgery. Ouyen Dinh Chu, MD, MBA , is a Professor of Surgery and Chief of Division of Surgical Oncology at LSUHSC- Shreveport, Department of Surgical Oncology. Anthony Sin, MD is an Associate Professor of Neurosurgery and Director of Complex Spinal Disorders at LSUHSC- Shreveport, Department of Neurosurgery.
Figure 6: Surgical scar after healing of posterior incision.
CONCLUSION
The gold standard for treatment of sacral chordoma remains en bloc resection with wide margins. These tumors are often diagnosed after they have grown to considerable size in the pelvis and should be referred to a specialized multidisciplinary center with experience. We advocate use of a team composed of surgical oncology and neurosurgery in order to carefully resect the tumor from the pelvic contents and spinal canal and reconstruct the lumbosacral support.
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Intracranial Septum Pellucidum Dysembryoplastic Neuroepithelial Tumor: Case Presentation and Review of Pediatric Septum PellucidumTumors
Rimal Dossani, MD, Devi Patra, MD, Nimer Adeeb, MD, Elizabeth Wild, MD, Abhilasha Ghildy- al, MD, Marjorie Fowler, MD, Christina Notarianni, MD
Dysembryoplastic neuroepithelial tumors (DNETs) of the septum pellucidum are a rare location for DNETs, which are most commonly located in the temporal cortex. In this case report, we present the case of a 5-year-oldboywhopresentedwith seizures. Magnetic resonance imaging (MRI) revealed a hyperintensemass onT2-weighted imaging arising from the right septumpellucidum. The patient was placed in right lateral decubitus position andunderwent right interhemispheric craniotomy for resectionofmass. Gross total resectionwas achieved and pathology was consistent with DNET. Patient has been free of seizures since the time of operation and MRI scans have been free of recurrence. We review the molecular and histopathological features of DNETs. Most importantly, for the interest of the general medical community, we present a differential diagnosis of pediatric septum pellucidum tumors based on MRI and histopathological findings.
INTRODUCTION
Dysembryoplastic neuroepithelial tumors (DNET) are benign glioneural tumorspresentingwithepilepsy inchildrenandyoung adults. DNETs aremost commonly located in the temporal lobeof the supratentorial cortex, but case reports and small case series have documented DNETs arising from the septum pellucidum and the foramen of Monro. On magnetic resonance imaging (MRI), DNETs appear as hyperintense on T2-weighted and fluid- attenuated inversion recovery (FLAIR) sequences, and do not exhibit contrast enhancement. The best treatment for DNETs is surgical resection. Adjuvant chemotherapy and radiation are not necessary. Pathology demonstrates disorganized microcystic architecture with both glial and neuronal elements without cytological atypia. In this report, we present a septum pellucidumDNET in a 5-year-old male child treated with surgical resection. For the interest of the larger medical community, we present a differential diagnosis of pediatric septum pellucidum tumors based on MRI and histopathological findings.
CASE REPORT
The patient is a 5-year-old boy who presented with worsening headaches for six months. Three months prior to presentation, he developed seizures well controlled on oxcarbazepine. His past medical history was notable for developmental delay because he was unable to walk until the age of three. His neurological exam was within normal limits. T2-weighted and fluid-attenuated inversion recovery MRI sequences showed a hyperintense mass arising from the right septum pellucidum (Figure 1). T1-weighted MRI showed isointense mass that did not enhance with contrast. The mass appeared to extend down to the foramen of Monro without hydrocephalus.
Figure 1: Preoperative magnetic resonance images showing mass arising fromseptumpellucidumandentering the right lateral ventricle. T1-weighted with contrast axial (A) and coronal (B) images and fluid attenuated inversion recovery (FLAIR) axial (C) and coronal (D) cuts demonstrate the tumor in the right lateral ventricle. side down. A small callosotomy via interhemispheric craniotomy was performed to enter the right lateral ventricle. The tumor appeared to be soft and gelatinous and was removed using bipolar cautery and suction. Finally, the septum pellucidum
The patient was taken to the operating room for resection of mass. He was placed in lateral decubitus position with the right
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Figure 2: Postoperative T1-weighted with contrast sagittal (A) , axial (B) and coronal (C) images showing gross total resection of mass from right lateral ventricle.
Figure 3: Tumor composed of oligodendroglia-like cells in a microcystic background (A) (hematoxylin-eosin, original magnifications ×400). At higher magnification (B) the characteristic oligodendroglia-like cells with uniform round nuclei in a fibrillary background (hematoxylin-eosin, original magnifications ×2000). The tumor showed characteristic positivity for immunohistochemical stains, (C) Glial fibrillary acidic protein, (D) Synaptophysin and (E) Neurofilament (original magnifications x2000).
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was cauterized to remove the remainder of the tumor. A small septostomy was performed to promote flow of cerebrospinal fluid between the right and left ventricles. The patient was admitted to the pediatric intensive care unit postoperatively. PostopearativeMR showed gross total resection (Figure 2). The patient was discharged to home on postoperative day four without any new neurological deficit.
• Lipoma • Hamartoma • Cavernous hemangioma • Vascular malformation with hemorrhage • Cysticercosis • Colloid cyst • Central Neurocytoma • Subependymoma • Subependymal giant cell astrocytoma • Low grade astrocytoma • Pilocytic astrocytoma • Oliglioglioma • Dysembroplastic neuroepithelial tumor
Non-Neoplastic
PATHOLOGY
Neoplastic
The specimen was received in two parts. The first part consisted of gelatinous soft tan tissue that measured 0.5x0.3x0.1 cm. The second part consisted of fragments of pink tan tissue that measured in aggregate 0.8x0.5x0.2 cm. Microscopically both parts of the neoplasms consisted of disorganized microcystic architecture with hypocellular neuronal growth without cytological atypia, findings consistent with a low-grade primary neuronal lesion. Subsequent immunohistological staining with glial fibrillary acidic protein (GFAP) and neurofilament highlighted the glial component of the neoplasm. Immunohistochemical staining was also positive for synaptophysin , a synaptic vesicle glycoprotein present in all neurons. In summary, the pathological diagnosis was consistent with dysembryoplastic neuroepithelial tumor (DNET) with presence of both glial and neuronal components and positive immunohistochemical staining for GFAP, neurofilament and synaptophysin (Figure 3).
Table 1: Differential diagnosis of lesions in the septum pellucidum
distinguishing features make preoperative diagnosis extremely difficult. A few differential points may help in diagnosis (Table 2). On MRI, DNETs appear as a gyriform enlargement of involved cortex. 17 They are hypointense onT1-weighted and hyperintense on T2-weighted images, and these tumors do not enhance with contrast. Some tumors may show iso/hypointensity on FLAIR with characteristic hyperintense rim. Among other MRI sequences, apparent diffusion co-efficient (ADC) sequences have been reported to be of clear differentiating value because of high cellularity. In the tumor series by Yamasaki et al, DNETs had much higher ADC values than other WHO grade 1 and grade 2 tumors, including ganglioneural tumors like central neurocytomas. 18 DNETs localized in the septum pellucidummay mimic colloid cysts; however, DNETs may be differentiated from colloid cysts by their clear origin above the foramen of Monro. Still, final diagnosis solely depends upon histopathological analysis. Histologically, DNETs resemble other ganglioneural tumors because of the presence of oligodendroglial like cells (OLC). A multinodular architecture is characteristic especially in cortical locations. Small, round OLC cells along with floating neurons in a gelatinous matrix form a microcystic or alveolar pattern. 11 On immunohistochemistry staining, the OLCs are positive for S-100 and Oligo-2 protein. They also express other neuronal markers like synaptophysin, Neu N, NSE and MAP2. 11 DNETs in theseptumpellucidumdonot differmuchhistologically from cortical DNETS; however, occasional irregularity from the classical pattern has been observed. Firstly, the characteristic multinodular pattern is less frequently observed in DNETs of septum pellucidum. 6,23 Secondly, we observed a less cellular growth in our specimen. Similar to Baisden et al, 6 we also noted single OLCs with a uniform round nuclei floating in a fibrillary background. In 50% of patients in Baisden et al series, synaptophysin positivity was noted on immunohistochemical staining. A possible genetic association has been suggested by Saito et al who found a familial occurrence of DNETs in the septum pellucidum. 24 Recently, Gessi et al studied molecular
DISCUSSION
The septum pellucidum is a midline structure with uncertain functional importance that separates the two halves of the lateral ventricles. Though its role in the limbic system and its connection with the hippocampus and hypothalamus has been demonstrated in few studies, 1 surgeons consider it as a benign membranous structure that may be divided if need arises. The occurrence of de-novo tumors in the septum pellucidum is rare. Tumors of the septum arise either from direct tumor extension from the corpus callosum or via subependymal spread from adjacent periventricular structures. Table 1 lists a differential diagnosis of tumors of the septum pellucidum. DNETs of the septum pellucidum are extremely rare and only sixteen cases have been reported to date. 6-10 Generally, DNETs are located in the supratentorial cortex andpresent with epilepsy in children and young adults. 11 In children with primary CNS tumors, the frequency of DNET is estimated to be 0.6-0.8 %. 12,13 Amongst extracortical locations, DNET has been reported to occur in periventricular white matter, foramen of Monro and the caudate nucleus. DNET origin from midline septum pellucidum location, thought to arise from subpial and subependymal germinal layers, 22 is distinctly rare as a primary location. DNETs of the septum pellucidum may simulate multitudes of lesions that can range from simple benign colloid cysts to high-grade atypical gliomas. Lack of clinical or radiological
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Clinical Presentation
Radiological Features
Pathological Features
Prognosis
• Intraventricular • Midline • MRI: T1 iso, T2 hyper
• Necrosis and cyst formation common • Diffuse synaptophysin reactivity • GFAP+ in ~ 50% of cases
• Higher recurrence rate • Requires adjuvant radiotherapy or radiosurgery
• Young patients • Headache • Features of raised ICP • Rarely, cognitive dysfunction, seizures, etc
Central Neurocytoma
• MRI- Isointense on T1, iso to hyperintense on T2 • Variable contract enhancement • Calcification ± • Heterogenous • MRI- Iso on T1 and Iso to hyper on T2 • Contrast enhancement + • Calcifications + • Ill-defined outline • Paramedian or assymetric • MRI: T1 iso to hypo, T2- Hyper • Contrast enhancement + • May show edema and mass effect • Contrast enhancement + • Heterogenous solid cystic • Calcifications +
• Glial tumor without neuronal component • GFAP+
• Recurrence occurs without total excision • Requires post of radiotherapy
• Any age • Headache • Features of raised ICP
Low grade astrocytoma
• Memory dysfunction (with involvement of fornix and corpus callosum)
• Perinuclear satellosis • Involves white matter • Larger and varied nuclei
• Recurrence occurs without total excision • Requires post of radiotherapy
• Middle aged • Raised ICP due to hydrocephalus
Oligodendroglioma
• Large polygonal cells resembling astrocytes or ganglion cells • Perivascular pseudorosette • No neurons • Glial tumor cells in clusters in an abundant fibrillary matrix • No mature neurons • Intratumoral rosental fibres • Eosinophillic granular bodies • Glial cells without mature neurons • Glial cells major cell type • Perivascular lymphocytic infiltration • May show atypia • GFAP+ • Neoplastic ganglion and glial cells in reticulin network
• Good prognosis with rare recurrence • Adjucant therapy usually not required with total excision
• In children and yong adult • Male preponderence • Seizure is the most common presentation
Ganglioglioma
• Mixed solid cystic • Typical lesion is
• Complete resection carries good prognosis • Radiotherapy needed for incomplete resections
• Young children • Headache • Raised ICP due to hydrocephalus
Pilocytic astrocytoma
• Wall of lateral ventricle near foramen of Monro • MRI- T1 hypo and T2 hyper • Homogenous contrast enhancement characterized by a cystic lesion with mural nodule that enhances with contract • Midline location • Heterogenous with cystic areas • Lobulated apprance • T1 hypo and T2 hyper • Minimal contrast enhancement in septum pellucidum lesions
• Young age • Associated with tuberous sclerosis • Present with raised ICP due to HCP • Middle aged and elderly males • More common in 4 th ventricle • Present with raised ICP due to HCP
• Total excision leads to cure • Recurrence rare • Radiotherapy not needed • Total excision leads to cure • Recurrence rare • Radiotherapy not needed
Subependymal Giant cell astrocytoma
Subependymoma
• Young age • Seizures, headache • Raised ICP due to HCP
• Midline location • Homogenous
• Small round oligodendroglia like cells with floating neurons • Microcystic or alvelolar pattern • May express synaptophysic but not GFAP
• Excellent prognosis • Rare recurrence even if in subtotal excision • Radiotherapy not needed
Dysembroplastic neuroepithelial tumor
• T1 hypo, T2 hyperintense • Does not enhance with contrast • Higher ADC value
Table 2: Differentiating features of various tumors of septum pellucidum
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