10.1007/s00381-007-0446-y

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Child's Nervous System

=A9 Springer-Verlag 2007

Original Paper

Neurological outcome after = resection of=20 intramedullary spinal cord tumors in children

Matthew J. McGirt^{1, 2 =},=20 Kaisorn L. Chaichana¹, = April Atiba¹,=20 Frank Attenello¹, = Graeme F. Woodworth¹ and=20 George I. Jallo¹

(1)	Department of Neurosurgery, Johns = Hopkins=20 School of Medicine, Baltimore, MD, = USA

(2)	3553 Newland Rd, Baltimore, = MD 21218,=20 USA

= =

=	Matthew J. McGirt= Email:=20 mmcgirt1@jhmi.edu

Received:=20 25 June 2007 Published online:=20 31 July 2007

Abstract

Objective With modern surgical = advances,=20 radical resection of pediatric intramedullary spinal cord tumors (IMSCT) = can be=20 achieved with preservation of long-term neurological function. Clinical = and=20 radiographic risk factors predictive of postoperative neurological = outcome may=20 serve as a guide for surgical risk stratification.

Materials = and=20 methods We prospectively reviewed the outcomes of 16=20 consecutive cases of pediatric IMSCT resection at a single institution.=20 Clinical, radiographic, and operative variables were analyzed as = predictors of=20 postoperative neurological function defined by the modified McCormick = score=20 (MMS).

Results Sixteen children = 10 =B1 5 years old=20 presented with median (interquartile range) MMS score of 2 (1=962) with = IMSCTs=20 (eight cervical, eight thoracic) involving 4 =B1 2 levels. = Pathology revealed=20 astrocytoma in 12 cases (three pilocytic, four grade II, three gradeIII, = two=20 GBM), gangliogliomas in two, ependymoma in one, and gliosis in one case. = At=20 median follow-up of 7 months, six (38%) patients experienced = improved=20 neurological function, eight (50%) remained stable, one (6%) experienced = a=20 delayed decrease in neurological function (GBM progression), and one = (6%) died=20 (GBM progression). Five (31%) patients developed persistent dysesthetic=20 symptoms. Four (80%) patients with cystic tumors experienced = neurological=20 improvement compared to only two (18%) patients with noncystic tumors,=20 p < 0.05. Preoperative steroid use (odds ratio, OR = [95% confidence=20 interval, CI] =3D 18.0 [1.24=96260.1], = p =3D 0.03) and cystic tumor (OR=20 [95%CI] =3D 18.0 [1.24=96260.1], = p =3D 0.03) predicted neurological improvement=20 after surgery.

Conclusion Radical resection of = pediatric=20 IMSCTs can be achieved with low incidence of neurological injury. = Sensory=20 syndromes frequently occur after pediatric IMSCT resection and = frequently affect=20 patient=92s quality of life. Tumors with compressive cysts may identify = patients=20 more likely to experience improved neurological function after surgical=20 resection.

Keywords Intramedullary spinal = cord=20 tumor - Outcome - Pediatric - Predictors =

Introduction

Intramedullary spinal cord tumors (IMSCTs) are relatively rare tumors = that=20 account for 4=9610% of central nervous system tumors but account for = approximately=20 35% of pediatric intraspinal tumors [1,=20 2,=20 26].=20 Conventionally, conservative management consisted of biopsy, dural=20 decompression, and radiation therapy. This approach avoided sequelae = associated=20 with iatrogenic injury to adjacent neurological tissue but yielded only = moderate=20 improvement in disease progression and survival. Recent advances in=20 microsurgical techniques, operative equipment, neuroimaging, and = intraoperative=20 neurophysiology have made more aggressive resection of IMSCT feasible = [3,=20 4,=20 9,=20 10,=20 18].=20 In fact, this approach has been associated with increased long-term = survival and=20 improved quality of life [3,=20 4,=20 15,=20 17,=20 25].=20

While these developments now allow radical resection in the majority = of=20 cases, overall morbidity rates remain high [5,=20 23].=20 Many studies have examined prognostic factors associated with functional = outcome=20 after IMSCT resection in adults [5,=20 11,=20 15,=20 23,=20 25].=20 However, studies in the pediatric population are sparse. We set out to = identify=20 preoperative variables that may identify patients most likely to = experience=20 improvement in neurological deficits with surgical resection of IMSCTs.=20

Materials and methods

Sixteen consecutive pediatric patients undergoing surgery for = IMSCTs at a=20 single academic institution were prospectively reviewed. Demographics, = clinical=20 presentation, comorbidities, preoperative magnetic resonance imaging = (MRI)=20 characteristics, intraoperative records, pathological data, and = neurological=20 function at discharge and at clinic follow-up were recorded. Pre- and=20 postoperative neurological function was defined by the modified = McCormick score=20 [21].=20 Preoperative steroid use was defined as oral dexamethasone given several = days=20 before surgery. Radical resection was defined as removal of at least 95% = of the=20 tumor based on comparison of the pre- and immediate postoperative MRI. = The=20 resection was defined as subtotal removal if 80 to 95% of the tumor was=20 resected. Improvement in neurological function was defined as both (1) = an=20 improvement in deficit on physical exam and (2) patients and their = families=20 described a subjective improvement in symptoms. Sensory discomfort = arising from=20 burning, tingling, or hyperesthesia was defined as dysesthetic symptoms = and=20 documented if they affected quality of life according to patients and = their=20 families. A tumor-associated cyst was defined as an eccentrically = located=20 cavitary lesion within the tumor mass (Fig. 1).=20 A tumor-associated syrinx was defined as a fluid-filled cavity extending = centrally within the spinal cord rostral and/or caudal to the tumor=20 (Fig. 1).=20

Fig. 1 Mid-sagittal T1=20 postcontrast MRI demonstrating an intramedullary spinal cord tumor with = both=20 tumor-associated cysts and syringomyelia. Syringomyelia can be seen = immediately=20 caudal to the enhancing tumor. The paracentric tumor associated cysts = can be=20 seen both within and immediately rostral to the enhancing tumor

Surgical technique

All patients underwent osteoplastic laminotomy for access to the = IMSCT. Only=20 medial facet joint exposure was performed, and an effort was made to = preserve=20 the facet joint capsules in all cases. The interspinous ligaments at the = rostral-most and caudal-most levels of the laminoplasty segment were = removed=20 with a Leksell rongeur. Bilateral laminotomies were then made at a width = equal=20 to the spinal canal in a caudal-to-rostral direction spanning the entire = laminoplasty segment utilizing the osteotome drill with a footplate. The = tumor=20 was approached and removed via a midline dural incision spanning the = entire=20 length of the tumor in all cases. Sensory-evoked and motor-evoked = potentials=20 were used. Resection was deemed complete once interface with the white = matter=20 was reached or if a sustained decrease in motor-evoked potentials = greater than=20 50% of the baseline occurred. Tumor-associated syrinx were not exposed = or=20 surgically manipulated in any case because of frequent resolution after = tumor=20 resection alone. After tumor resection and primary dural closure, the = laminae=20 were reapproximated and attached with titanium microplates. Instrumented = or in=20 situ fusion was not utilized in any case.

Statistical analysis

For intergroup comparison, the Student=92s t test was used for = parametric data, and the Mann=96Whitney U test was used for = nonparametric=20 data. Percentages were compared via chi-squared tests or the Fisher = exact test=20 where appropriate. The univariate association of all recorded variables = to=20 postoperative neurological improvement was assessed via logistic = regression=20 analysis (Statview, [24]).=20

Results

Patient population

Sixteen patients underwent resection of an IMSCT. = Mean =B1 standard deviation=20 (SD) age was 10 =B1 5 years, and nine (56%) patients = were male. The mean=20 duration of symptoms was 8 months (range 1=9636) before resection. = The median=20 (interquartile range, IQR) McCormick grade at presentation was 2 = (1=962). Eight=20 (50%) patients presented primarily with motor symptoms while eight (50%) = presented primarily with sensory symptoms. One (6%) and two (12%) = patients=20 received preoperative chemotherapy and radiotherapy before surgery,=20 respectively. Five (31%) patients were started on oral decadron at least = 1 week before surgery. The spinal location of the IMSCT was = primarily=20 cervical in eight (50%) patients and primarily thoracic in eight (50%) = patients,=20 and the location crossed the cervicothoracic junction in four (25%) = patients.=20 Tumor spanned 4 =B1 2 (mean =B1 SD) spinal = levels and was associated with a syrinx=20 in five (31%) cases. A tumor associated cyst was evident on preoperative = MRI in=20 five (31%) cases. Radical resection (>95%) was achieved in ten (72%) = of=20 non-glioblastoma multiforme (GBM) cases. Subtotal resection (80=9695%) = was=20 achieved in four (28%) of the non-GBM cases and in both cases of GBM. = Surgical=20 pathology revealed astrocytoma in 12 cases (three pilocytic, four grade = II,=20 three grade III, two GBM), gangliogliomas in two cases, ependymoma in = one case,=20 and gliosis in one case.

Outcome

At median (IQR) follow-up of 7 months (1=9624), the median (IQR) = modified=20 McCormick grade [21]=20 was 1 (1=962). Six (38%) patients experienced improved neurological = function,=20 eight (50%) remained stable, one (6%) experienced a delayed decrease in=20 neurological function (GBM progression), and one (6%) died (GBM = progression). No=20 patient experienced an acute decline in neurological function = immediately after=20 IMSCT resection. Five (31%) patients developed persistent dysesthetic = symptoms=20 that felt to affect quality of life by last follow-up.

Four (80%) patients with cystic tumors experienced neurological = improvement=20 compared to only two (18%) patients with noncystic tumors, = p < 0.05,=20 Fig. 1.=20 Preoperative steroid use (odds ratio, OR [95% confidence interval, = CI] =3D 18.0=20 [1.24=96260.1], p =3D 0.03) and cystic tumor (OR = [95%CI] =3D 18.0 [1.24=96260.1],=20 p =3D 0.03) were associated with neurological = improvement after surgery.=20 All other clinical and radiographic variables (age, gender, duration = symptoms,=20 tumor size, location or pathology, presence of syrinx, degree of = resection,=20 radiotherapy) were not associated with neurological improvement at last=20 follow-up.

Discussion

In this study, we identified clinical and radiographic variables = predictive=20 of neurological improvement in 16 consecutive pediatric patients = undergoing=20 IMSCT resection. These variables remain relatively uninvestigated and = may serve=20 as a guide for surgical risk stratification. For non-GBM cases, more = than 40% of=20 patients experienced improvement in their neurological deficits by a = median of=20 7 months after surgery, while neurological function remained stable = in all=20 other patients. Preoperative steroid use and the presence of = tumor-associated=20 cysts on MRI each markedly increased the likelihood of neurological = improvement=20 with surgical resection.

In the adult IMSCT literature, several studies have attempted to = determine=20 predictors of neurological outcomes after IMSCT resection. = Intramedullary=20 ependymomas, which primarily occur in adults, are associated with better = neurological outcomes compared to intramedullary astrocytomas [5,=20 13].=20 The use of intraoperative monitoring and postoperative radiation therapy = have=20 also been linked to improved morbidity, while the presence of arachnoid = scarring=20 [14],=20 spinal atrophy [14],=20 and solid tumor components [6]=20 have been associated with increased morbidity. These studies, however, = involve=20 adult patients, and factors associated with neurological outcome in the=20 pediatric population remain largely unstudied.

IMSCTs in pediatric patients are inherently different than in adults. = Histologically, infiltrating astrocytomas are the most common type of = tumor in=20 children, as opposed to adults who more commonly have intramedullary = ependymomas=20 [3,=20 4,=20 15,=20 16,=20 25].=20 Astrocytomas are often eccentrically located and, as a result, cause an=20 asymmetric enlargement of the spinal cord [16].=20 The pediatric spine is also more susceptible to postoperative deformity = because=20 of lax ligamentous structures, horizontal orientation of the facet = complexes,=20 and the dynamic growth of the immature spine [12,=20 22,=20 28].=20 Furthermore, children more often present with insidious, nonspecific = symptoms of=20 diffuse and nonlocalized pain, motor deficits, and, in young infants, = motor=20 regression [3,=20 4,=20 16].=20 Adults, however, typically present with sensory deficits, including = dysesthesia,=20 extremity numbness, and myelopathy [10,=20 15,=20 25].=20 These intrinsic differences between IMSCTs in adults and children = highlight the=20 importance of identifying outcome predictors solely for pediatric IMSCTs = to draw=20 conclusions most applicable to pediatric patients.

The overwhelming majority of patients in this study had improved or=20 maintained neurological function after IMSCT resection at last = follow-up. The=20 two patients who worsened neurologically had GBM and deteriorated = secondary to=20 tumor progression. These results are similar to previous pediatric = studies=20 [3,=20 4].=20 Constantini et al. [4]=20 reported that approximately 75% of the 164 children and young adults had = improved or maintained neurological function at last follow-up. In a = separate=20 study, Epstein et al. documented that 25 of 27 patients under = 3 years of=20 age had improved or maintained neurological function after IMSCT = resection.=20 Therefore, this study and others support the finding that IMSCT can be = safely=20 resected in pediatric patients and often lead to improved and/or = maintained=20 neurological function at last follow-up [3,=20 4].=20

Patients who were administered preoperative dexamethasone several = days before=20 surgery in this study were more likely to improve their neurological = function at=20 last follow-up. Steroids presumably function by decreasing both = tumor-associated=20 inflammation and spinal cord edema, hypothetically making tumors more = amenable=20 to resection and decreasing the chance that surrounding neurological = tissue=20 would be damaged [7,=20 8,=20 27].=20 Steroids have been shown to decrease surgical pain, length of stay, and = improve=20 functional outcome after routine spine surgery [19,=20 20].=20 Furthermore, steroids improve neurologic outcomes after surgery for = metastatic=20 spinal cord compression [7,=20 8,=20 27].=20 The findings in our study also support the use of steroids in the = preoperative=20 period.

Finally, patients who possess IMSCTs with associated macroscopic = cysts were=20 more likely to have improved neurological outcomes after surgical = resection.=20 Prior studies have noted the presence of cysts but do not address their=20 predictive value or effect on neurological outcomes [3,=20 4].=20 Nevertheless, the detection of cysts on preoperative neuroimaging may = indicate a=20 better likelihood of improved outcomes. This may be due to the fact that = cysts=20 typically occur at the tumor=96spinal cord interface allowing safer = tumor margin=20 identification and resection [18].=20 Cystic components of IMSCT may also contribute to compressive spinal = cord injury=20 that may be acutely relieved at the time of decompression and resection. = Decompression of compressed adjacent spinal cord not yet infiltrated = with the=20 tumor may underlie the greater improvement observed in patients with = intratumor=20 cysts.

Conclusion

Prior studies on IMSCTs primarily involve adult patients. Studies = that=20 exclusively focus on pediatric patients are limited, although IMSCTs in=20 pediatric patients are distinctly different than adults. This study = supports the=20 finding that IMSCTs can be safely and radically resected in the = pediatric=20 population, with the slight majority experiencing improvement in = deficits.=20 Tumors with compressive cysts may identify patients more likely to = experience=20 improved neurological function after surgical resection.

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