Late neuropsychological and behavioural outcome of children surgically treated for craniopharyngioma

Riva, Daria; Pantaleoni, Chiara; Devoti, Monica; Saletti, Veronica; Nichelli, Francesca; Giorgi, C.

doi:10.1007/s003810050207

Cited by 88 publications

(57 citation statements)

References 15 publications

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“…All but 2 of 50 surviving patients enjoy a normal social life. Riva et al 88 reported no instances of cognitive or memory deficits in 12 children who underwent radical resection but noted increased emotional lability and im pulse control, possibly related to the subfrontal approach or hypothalamic injury. In a series by Hoffman et al, 46 26 of 27 children who underwent aggressive resection had IQ scores at or above average levels.…”

Section: Functional Outcomementioning

confidence: 99%

“…Side effects of radiotherapy include dysfunction of the hy pothalamicpituitary axis and visual apparatus, benign and malignant radiationinduced CNS tumors, cognitive and at tentional deficits, and cerebrovasculopathy/moyamoya dis ease. [5][6][7]26,28,29,31,36,37,41,51,52,60,63,64,72,75,84,87,88,90,96,97,102,105,106 These effects are more pronounced in younger children 64,75 and are correlated with increasing target volume and total dos age. 28,36,72,86,87 These late effects must be recognized when comparing risk-benefit profiles of radical versus limited resection and radiotherapy.…”

Section: Side Effects Of Radiation Therapy and Impact On Reoperationmentioning

confidence: 99%

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Efficacy and safety of radical resection of primary and recurrent craniopharyngiomas in 86 children

Elliott¹,

Hsieh²,

Hochm

et al. 2010

PED

163

148

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Object Optimal treatment of primary and recurrent craniopharyngiomas remains controversial. Radical resection and limited resection plus radiation therapy yield similar rates of disease control and overall survival. The data are much less clear for recurrent tumors. The authors report their experience with radical resection of both primary and recurrent craniopharyngiomas in children and compare the outcomes between the 2 groups. Methods A retrospective analysis was performed in 86 children younger than 21 years of age who underwent a total of 103 operations for craniopharyngioma between 1986 and 2008; these were performed by the senior author. The goal was resection with curative intent in all patients. Two patients were lost to follow-up and were excluded from analysis. The mean age at the time of surgery was 9.6 years, and the mean follow-up was 9.0 years. Results All 57 children with primary tumors underwent gross-total resection (GTR). A GTR was achieved in significantly fewer children with recurrent tumors (18 [62%] of 29). There were 3 perioperative deaths (3%). Tumor recurred after GTR in 14 (20%) of 71 patients. Overall survival and progression-free survival were significantly better in patients with primary tumors at time of presentation to the authors' institution. There were no significant differences in the neurological, endocrinological, visual, or functional outcomes between patients with primary and those with recurrent tumors. Factors negatively affecting overall survival and progression-free survival include subtotal resection (recurrent tumors only), tumor size ≥ 5 cm, or presence of hydrocephalus or a ventriculoperitoneal shunt. Prior radiation therapy and increasing tumor size were both risk factors for incomplete resection at reoperation. Conclusions In the hands of surgeons with experience with craniopharyngiomas, the authors believe that radical resection at presentation offers the best chance of disease control and potential cure with acceptable morbidity. While GTR does not preclude recurrence and is more difficult to achieve in recurrent tumors, especially large and previously irradiated tumors, radical resection is still possible in patients with recurrent craniopharyngiomas with morbidity similar to that of primary tumors.

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Section: Functional Outcomementioning

confidence: 99%

Section: Side Effects Of Radiation Therapy and Impact On Reoperationmentioning

confidence: 99%

Efficacy and safety of radical resection of primary and recurrent craniopharyngiomas in 86 children

Elliott¹,

Hsieh²,

Hochm

et al. 2010

PED

163

148

View full text Add to dashboard Cite

show abstract

“…4,15,20,29,31,41,44,52,64,73 Given the improved rates of survival with treatment advances over the past few decades, the focus has turned toward functional outcome and QOL metrics. 6,7,41,46,48,50,55,58,61 Neuropsychological testing of small series of patients has revealed cognitive and intellectual sequelae in as many as 60% of children treated for craniopharyngioma.…”

Section: Functional Outcome In Reported Surgical Seriesmentioning

confidence: 99%

“…6,7,41,46,50,55,58,61 Although investigators in a handful of studies have attempted to create grading scales to assess functional outcomes and QOL in patients with craniopharyngiomas, each scale has its own limitations. Wen and colleagues 67 developed a 4-tiered outcome…”

mentioning

confidence: 99%

Craniopharyngioma Clinical Status Scale: a standardized metric of preoperative function and posttreatment outcome

Elliott¹,

Sands

Strom³

et al. 2010

FOC

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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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“…Side effects of irradiation include dysfunction of the hypo thalamicpituitary axis, visual deterioration, radiationin duced CNS tumors, age-dependent cognitive deficits, pro gressive decline of IQ, and cerebrovasculopathy/moyamoya disease. [2][3][4]11,12,14,18,19,[24][25][26]28,36,37,43,45,46,54,56,63,[65][66][67][68][71][72][73]76,78,79,83 Critical risk factors for predicting increased morbidity from radiotherapy are younger age at irradiation, volume of the irradiated field, and total dosage. 18,24,46,54,65,66 Specifically, Neglia et al 56 noted these effects to be more pronounced in patients under 5 years of age.…”

Section: Discussionmentioning

confidence: 99%

Successful surgical treatment of craniopharyngioma in very young children

Elliott¹,

Wisoff

2009

PED

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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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Late neuropsychological and behavioural outcome of children surgically treated for craniopharyngioma

Cited by 88 publications

References 15 publications

Efficacy and safety of radical resection of primary and recurrent craniopharyngiomas in 86 children

Efficacy and safety of radical resection of primary and recurrent craniopharyngiomas in 86 children

Craniopharyngioma Clinical Status Scale: a standardized metric of preoperative function and posttreatment outcome

Successful surgical treatment of craniopharyngioma in very young children

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