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Robert E. Elliott and Jeffrey H. Wisoff

Object

Prior work by the authors' group and reports of other authors suggest worse functional outcomes and decreased survival in children with larger craniopharyngiomas. The purpose of this study was to assess the oncological, endocrinological, and functional outcomes in children who underwent radical resection of giant craniopharyngiomas (defined as 5 cm or greater in largest diameter).

Methods

Between 1986 and 2006, 26 children under the age of 18 (14 boys, 12 girls; mean age 10.5 years) underwent radical resection of giant craniopharyngiomas performed by the senior author. Data were retrospectively collected to assess the outcome of surgical treatment.

Results

Twenty (77%) of 26 patients underwent gross-total resection (GTR) confirmed by intraoperative inspection and postoperative imaging. All primary tumors (17 of 17) and 3 (33%) of 9 recurrent tumors were treated with GTR. There was no operative mortality, and 18 of 26 patients (69%) were alive at a mean follow-up of 8.9 years (median 9.3 years). Disease control was achieved in 21 (84%) of the 25 patients followed up for more than 6 months and was more successful in patients who underwent GTR (95%) than in those who underwent STR (50%, p = 0.03). New-onset diabetes insipidus (DI) occurred in 63.2% of patients (73% of patients had DI postoperatively). New or worsened deficits in visual acuity and visual fields occurred in 16% and 28%, respectively, of the 25 patients for whom postoperative visual data were available. Five patients (19%) experienced significant, permanent neurological deficits, and 5 (19%) had mild to moderate deficits. New or worsened hypothalamic disturbance occurred in 35% and 22% of patients, respectively, but obesity developed in only 15%.

Conclusions

In this retrospective series, radical resection of giant craniopharyngiomas in children was found to lead to excellent rates of disease control with acceptable or good functional outcomes but slightly higher rates of neurological complications compared with rates in patients with smaller tumors. Radical resection is less successful in recurrent tumors that reach very large sizes, especially previously irradiated tumors, with resultant diminished survival.

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Robert E. Elliott and Jeffrey H. Wisoff

Object

Given the potential morbidity of cranial irradiation in young children, the risk-benefit analysis of limited surgery plus irradiation versus radical resection may favor the latter strategy. The purpose of this study was to assess the oncological, endocrinological, and functional outcomes of patients 5 years of age and younger who underwent radical resection of craniopharyngiomas.

Methods

Between 1991 and 2008, 19 children age ≤ 5 years were diagnosed with a craniopharyngioma and underwent radical resection by the senior author (J.H.W.). Data were retrospectively collected on these 19 patients (11 males, 8 females; mean age 3.2 years) to assess the efficacy and impact of surgical treatment.

Results

Eighteen (95%) of 19 patients underwent gross-total resection (GTR) confirmed by intraoperative inspection and postoperative imaging. There was no operative death and 18 of (95%) 19 patients were alive at a mean follow-up of 9.4 years (median 8.3 years). Six patients (31%) had a total of 7 tumor recurrences treated by repeat GTR in 5 patients and Gamma knife surgery in 1 patient. No patient required conventional, fractionated radiation therapy. Disease control was achieved surgically in 17 (89.5%) patients and with surgery and Gamma knife surgery in 1 patient, yielding an overall rate of disease control of 95% without the use of conventional radiotherapy. New-onset diabetes insipidus occurred in 50% of patients. Vision worsened in 1 patient, and there was no long-term neurological morbidity.

Conclusions

In this retrospective series, children aged ≤ 5 years with craniopharyngiomas can have excellent outcomes with minimal morbidity after radical resection by an experienced surgeon. Disease control in this population can be successfully achieved with GTR alone in the majority of cases, avoiding the detrimental effects of radiotherapy in this vulnerable population.

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Robert E. Elliott and Jeffrey H. Wisoff

Object

Fusiform dilation of the supraclinoid internal carotid artery (FDCA) is a reported occurrence following surgery for suprasellar tumors, in particular craniopharyngiomas. We report our experience of the incidence and natural history of FDCA following aggressive surgical resection of craniopharyngiomas in children.

Methods

Between 1986 and 2006, 86 patients under the age of 21 underwent radical resection of craniopharyngiomas at our institution. Ten cases with < 1 year of follow-up imaging (6), perioperative death (3), or nonsuprasellar tumors (1) were excluded. Data were retrospectively collected on the remaining 76 patients (43 male, 33 female; mean age 9.5 years; mean tumor size 3.3 cm) to determine the risk factors for and the rate and clinical significance of FDCA.

Results

Fifty patients had primary tumors and 26 patients received treatment before referral to our center. Sixty-six children (87%) had gross-total resection. At a mean follow-up time of 9.9 years, FDCA had developed in 7 patients (9.2%), all of whom had primary tumors and gross-total resection. The mean time to onset of FDCA was 6.8 months (range 3–11 months) with stabilization occurring at mean of 17.7 months (range 9–29.5 months). The mean size of the aneurysms was 9.1 mm (range 7.1–12 mm). After arrest, no lesions showed continued growth on serial imaging or produced symptoms or required treatment. There were no significant differences in age, sex, tumor size, pre- or retrochiasmatic location, extent of resection, or surgical approach (p > 0.05) between patients with and without FDCA.

Conclusions

Fusiform dilation of the supraclinoid internal carotid artery occurred in almost 10% of children following radical resection of craniopharyngiomas. In agreement with other reports, the authors concluded that FDCA probably occurs as a result of surgical manipulation of the supraclinoid carotid artery and should be managed conservatively because very few patients exhibit continued symptoms or experience growth or rupture of the lesion.

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Editorial

Less is less

Ossama Al-Mefty

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Robert E. Elliott, Stephen A. Sands, Russell G. Strom, and Jeffrey H. Wisoff

Object

Controversy persists concerning the optimal treatment of craniopharyngiomas in children, and no standard outcome metric exists for comparison across treatment modalities, nor is there one that adequately reflects the multisystem dysfunction that may arise.

Methods

The authors retrospectively analyzed the records of 86 consecutive children who underwent a uniform treatment paradigm of attempted radical resection performed by a single surgeon. Excluding 3 perioperative deaths and 3 patients with inadequate follow-up, 80 children (34 girls and 46 boys; mean age 9.56 years; mean follow-up 9.6 years) composed the study group (53 primary and 27 previously treated/recurrent tumors). Building on existing classification schemes proposed by De Vile for hypothalamic dysfunction and Wen for overall functional outcome, the authors devised a more nuanced classification system (Craniopharyngioma Clinical Status Scale [CCSS]) that assesses outcome across 5 axes, including neurological examination, visual status, pituitary function, hypothalamic dysfunction, and educational/occupational status at last follow-up (there is a 4-tiered grading scale in each domain, with increasing values reflecting greater dysfunction).

Results

There was a significant increase in pituitary dysfunction following treatment—consistent with the high rates of diabetes insipidus and hypopituitarism common to the surgical management of craniopharyngiomas—and less dramatic deterioration in hypothalamic function or cognitive domains. Significant improvement in vision was also demonstrated, with no significant overall change in neurological status. Preoperative CCSS scores predicted postoperative outcome better than clinical characteristics like patient age, sex, tumor size, and the location or presence of hydrocephalus.

Conclusions

Preoperative CCSS scores predicted outcome with higher accuracy than clinical or imaging characteristics. In lieu of randomized trials, the CCSS may provide a useful outcome assessment tool for comparison across treatment paradigms and surgical approaches. Long-term follow-up is critical to the analysis of outcomes of craniopharyngioma treatment, given the often-delayed sequelae of all therapies and the high recurrence rates of these tumors.

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Yaron A. Moshel, Robert E. Elliott, David J. Monoky, and Jeffrey H. Wisoff

Object

The choice of surgical approach during resection of a thalamic juvenile pilocytic astrocytoma (JPA) is dictated by the location of the displaced normal thalamus and posterior limb of the internal capsule (PLIC). Diffusion tensor (DT) imaging and white matter tractography can identify the location of the PLIC in relation to the tumor and may be useful in planning the operative trajectory.

Methods

Diffusion tensor imaging was used to localize the PLIC on preoperative MR imaging in 6 children undergoing resection of thalamic JPAs. After review of the standard T2-weighted MR imaging sequences, the anticipated position of the PLIC was determined. This result was compared with the location of the PLIC determined by a blinded radiologist with the use of DT imaging. The utility of DT imaging in determining the surgical approach to a thalamic JPA, degree of resection, and neurological outcomes were all evaluated.

Results

Diffusion tensor imaging confirmed the expected location of the PLIC as approximated on conventional T2-weighted images in all 6 cases. In 1 patient in particular, unexpected medial deviation of the PLIC was identified, and this proved useful in tailoring the approach to a more lateral trajectory. Gross-total resection of all cystic and solid tumor components was confirmed on postoperative imaging in all cases. All patients experienced mild to moderate worsening of neurological status immediately following resection, but 4 of 6 patients were back to their preoperative baseline at 6-month follow-up.

Conclusions

Diffusion tensor imaging and white matter tractography successfully identified the white matter fibers emanating from the precentral gyrus within the PLIC in children with thalamic JPAs prior to surgery. Diffusion tensor imaging served as a valuable tool for stereotactic planning of operative approaches to thalamic JPAs. Localizing the position of the PLIC helped minimize potential neurological morbidity and facilitated gross-total resection.

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Robert E. Elliott, Omar Tanweer, Akwasi Boah, Michael L. Smith, and Anthony Frempong-Boadu

Object

Some centers report a lower incidence of vertebral artery (VA) injury with C-2 pars screws compared with pedicle screws without sacrificing construct stability, despite biomechanical studies suggesting greater load failures with C-2 pedicle screws. The authors reviewed published series describing C-2 pars and pedicle screw implantation and atlantoaxial fusions and compared the incidence of VA injury, screw malposition, and successful atlantoaxial fusion with each screw type.

Methods

Online databases were searched for English-language articles between 1994 and April of 2011 describing the clinical and radiographic outcomes following posterior atlantoaxial fusion with C-1 lateral mass and either C-2 pars interarticularis or pedicle screws. Thirty-three studies describing 2975 C-2 pedicle screws and 11 studies describing 405 C-2 pars screws met inclusion criteria for the safety analysis. Seven studies describing 113 patients treated with C-2 pars screws and 20 studies describing 918 patients treated with C-2 pedicle screws met inclusion criteria for fusion analysis. Standard and formal meta-analysis techniques were used to compare outcomes.

Results

All studies provided Class III evidence. Ten instances of VA injury occurred with C-2 pedicle screws (0.3%) and no VA injury occurred with pars screws. The point estimate of VA injury for C-2 pedicle screws was 1.09% (95% CI 0.73%–1.63%) and was similar to that of C-2 pars screws (1.48%, 95% CI 0.62%–3.52%). Similarly, there was no statistically significant difference in the rate of clinically significant screw malpositions (1.14% [95% CI 0.77%–1.69%) vs 1.69% [95% CI 0.73%–3.84%]). Radiographically identified screw malposition occurred in a higher proportion of C-2 pedicle screws compared with C-2 pars screws (6.0% [95% CI 3.7%–9.6%] vs 4.0% [95% CI 2.0%–7.6%], p < 0.0001). Pseudarthrosis occurred in a greater proportion of patients treated with C-2 pars screws (5 [4.4%] of 113) compared with those treated with C-2 pedicle screws (2 [0.22%] of 900). Point estimates with 95% confidence intervals show a slightly higher rate of successful atlantoaxial fusion in the pedicle screw cohort (97.8% [CI 96.0%–98.8%] vs 93.5% [CI 86.6%–97.0%]; p < 0.0001). Q-testing ruled out heterogeneity between the study groups.

Conclusions

With a thorough knowledge of axis anatomy, surgeons can place both C-2 pars and C-2 pedicle screws accurately with a small risk of VA injury or clinically significant malposition. There may be subtle trade-off of safety for rigidity when using axial pedicle instead of pars screws, and the decision to use either screw type must be made only after careful review of the preoperative CT imaging and must take into account the surgeon's expertise and the particular demands of the clinical scenario in any given case.

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Robert E. Elliott, Yaron A. Moshel, and Jeffrey H. Wisoff

Object

The purpose of this study was to assess the impact of minimal residual calcification without enhancing tumor on the rate of recurrence after gross-total resection (GTR) of craniopharyngioma in children.

Methods

Data were retrospectively collected in 86 patients younger than 21 years of age in whom 103 craniopharyngioma resections were performed by the senior author between 1986 and 2008. Forty-nine patients (27 boys and 22 girls, with a mean age of 8.6 years) fulfilled the criteria for inclusion in this study by having tumor calcification on the preoperative CT scan, undergoing GTR, and having complete postoperative CT and MR imaging and clinical follow-up.

Results

Thirteen patients (27%) had residual calcification (≤ 2 mm in 12 patients; 3.5 mm in 1 patient) on their postoperative CT scan. At a mean follow-up of 9.4 years (median 10 years), 2 (15%) of 13 patients with and 10 (28%) of 36 patients without residual calcification experienced tumor recurrence. There were no significant differences between these groups in terms of the duration of follow-up, time to recurrence, rate of recurrence, or recurrence-free survival.

Conclusions

The absence or presence of minimal residual calcification does not have an impact on the risk of recurrence after GTR in pediatric craniopharyngiomas. The authors recommend withholding irradiation or other adjuvant therapy in the setting of minimal residual calcification without enhancing tumor. Close follow-up with frequent serial imaging in all patients after GTR is imperative to identify and treat early recurrence.

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Robert E. Elliott, Yaron A. Moshel, and Jeffrey H. Wisoff

Local recurrence following radical resection is one of the most common complications of pediatric craniopharyngioma. Only 28 cases of ectopic recurrence of craniopharyngioma have been reported in the literature, and only 13 cases occurred in patients originally treated as children. In this consecutive series of 86 children who underwent radical resection of primary and recurrent craniopharyngiomas, 4 patients (4.7%) experienced ectopic tumor recurrence, accounting for 27% of all recurrences after gross-total resection. The authors report on the successful surgical treatment of these 4 patients and the impact of ectopic craniopharyngioma recurrence on survival.

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Richard W. Byrne