- Department of Neurosurgery, The Rose Ella Burkhardt Brain Tumor and Neuro-Oncology Center, Neurological and Taussig Cancer Institutes, Cleveland Clinic, Cleveland, Ohio, United States
- Department of Neurosurgery, Mayo Clinic, Jacksonville, Florida
Gene H. Barnett
Department of Neurosurgery, The Rose Ella Burkhardt Brain Tumor and Neuro-Oncology Center, Neurological and Taussig Cancer Institutes, Cleveland Clinic, Cleveland, Ohio, United States
DOI:10.4103/2152-7806.121612Copyright: © 2012 Rahmathulla G. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
How to cite this article: Rahmathulla G, Barnett GH. Minimally invasive management of adult craniopharyngiomas: An analysis of our series and review of literature. Surg Neurol Int 20-Nov-2013;4:
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Background:Craniopharyngiomas (CPs) are slow growing tumors with an incidence of between 1.2% and 4.6%, having a bimodal age distribution typically peaking in childhood and in adults between 45 and 60 years. Recurrences occur even after documented gross total resections necessitating a combination of therapeutic strategies. Obtaining a cure of this tumor in adults without producing major side effects continues to remain elusive.
Methods:We describe our results in 11 patients with CP treated in a minimally invasive fashion using a combination of techniques like burr hole aspiration, Ommaya reservoir placement, ventriculo-peritoneal (VP) shunting and focal radiation (Gamma Knife stereotactic radiosurgery/Intensity modulated radiotherapy [GKRS/IMRT]).
Results:Visual function remained intact in all patients; endocrine status remained stable with two patients developing new postoperative diabetes insipidus. There was no periprocedural morbidity or mortality, with hospital stays for any in-patient procedure being 48 hours or less.
Conclusions:Minimally invasive techniques such as cyst aspiration, insertion of a catheter with Ommaya reservoir, when combined with stereotactic radiosurgery/IMRT is an effective and safe option for management and long-term control of adult CPs. We believe the Ommaya catheter by itself could act as a stent, creating a tract allowing gradual drainage of cyst fluid and stabilization without necessitating any further interventions in selected cases.
Keywords: Craniopharyngioma, focal radiotherapy, minimally invasive, radiosurgery
Craniopharyngiomas (CPs) are benign, slow growing tumors whose ideal management remains controversial and challenging. They arise from remnants of the craniopharyngeal duct and/or Rathkes cleft. These tumors have an incidence of between 1.2% and 4.6%[
Traditional management of these tumors is microsurgical total resection. However, their propensity to recur had necessitated a combination of strategies such as local chemotherapy (e.g., bleomycin)[
The aim of this study was to present our outcomes treating adult CP patients using a combination of minimally invasive strategies including cyst aspiration (with or without Ommaya reservoir system insertion), ventriculo-peritoneal (VP) shunting and either upfront or adjuvant conformal radiotherapy and a review of current literature.
An institutional review board (IRB) approved retrospective chart review of all patients undergoing treatment for CPs between 1995 and 2010. Fifty-two patients with a diagnosis of CP underwent management at our institute between 1995 and 2010. We included all adult patients (>21 years) with an imaging diagnosis of CP with a predominantly cystic component, had undergone either a biopsy with cyst drainage, insertion of a catheter or Ommaya reservoir system, VP shunt or radiation therapy (RT) as their procedures, without any prior surgical intervention categorizing all these procedures together as minimally invasive. Out of the 52 patients, 11 patients were treated using the minimally invasive strategy. The median age for all CP patients at diagnosis was 49 years (range 24-80) with an equal number of male and female patients. In the minimally invasive treated group there were five male and six female patients with a median age of 58 years (range 24-80 years). Seven patients had histologically proven CPs with stereotactic biopsy (4 adamantinomatous, 2 WHO grade I, and 1 papillary) and of the remaining four patients, two underwent a stereotactic biopsy aspiration with characteristic machine oil fluid but no tissue sample and two patients in the cohort had undergone VP shunting elsewhere. For these 4/11 patients’ clinical findings, laboratory workup, and neuroimaging was the basis for further management. In most cases after biopsy-cyst aspiration, patients were followed with magnetic resonance imaging (MRI) studies initially at 1 month, then every 3 months for a year and then every 6 months for a year, and subsequently at yearly intervals to evaluate tumor size and to determine the feasibility of early intervention with focal RT (Gamma knife radiosurgery or Intensity modulated radiotherapy [IMRT]). Detailed neuroendocrinological assessment was done prior to and following treatment. A brief description of three representative cases is presented.
HS, a 46-year-old male presented with decreased vision in his left eye along with impotence and diminished energy levels of about 3 months duration. His endocrine evaluation revealed pan-hypopituitarism. He was diagnosed with a suprasellar mass, mixed solid-cystic [Figure
Figure 1 (a and b)
HS, a 46-year-old male presented with symptoms of visual and endocrine dysfunction. T1-MR postcontrast images on presentation with sagittal (a) and coronal (b) sequences revealing a mixed tumor with sellar-suprasellar involvement, contrast enhancement at the periphery and mass effect over the optic chiasm
MO, a 24-year-old male presented at an outside hospital with headaches of a few months in duration with a loss of libido and decreased energy levels. His endocrine workup revealed hypopituitarism and imaging showed a 2.5 cm sellar-suprasellar solid cystic mass for which he underwent a cyst aspiration and biopsy. He subsequently had tumor progression and was treated with GKRS. Long-term follow-up at 9 years continues with the patient remaining stable and requiring no further interventions.
MC, a 69-year-old female who following a head injury, was diagnosed with an incidental suprasellar cystic-solid mass. She remained asymptomatic for 4 years, subsequently developing diminished vision along with weight gain with an increase in lesion size. She underwent biopsy-aspiration of the cyst followed by Gamma Knife radiosurgery a month later. Subsequently 4 months later she developed a cyst recurrence on imaging and underwent Ommaya reservoir insertion, having no associated clinical changes. On follow-up, her MRI revealed an increase in the size of the cyst. Although further aspiration of the cystic component via the Ommaya reservoir was advised, she declined any intervention. Repeat imaging revealed a spontaneous decrease in the size of the cystic component that remained stable over time and required no further intervention.
All the patients were evaluated clinically for their neurological, endocrine status, and MRI imaging at our institution. Initial follow-up imaging was obtained at 1 month, then every 3 months for a year, subsequently at 6 months intervals for a year, and then at yearly intervals. Earlier imaging was done in cases of symptomatic worsening or if progression was observed on their MRIs. In patients with stable disease yearly clinical and neuroimaging follow-up was performed.
There were no procedural or perioperative associated morbidity or mortality. The mean follow-up period in these patients was 66 months (range 2-144 months) and the median was 60 months. There was always a residual component on posttreatment MRI scans because the interventions did not include gross surgical resection. Two patients who were 77 and 78 years at the time of initial diagnosis and treatment were lost to follow-up after a period of 11 and 9 years, respectively. On the last clinic visit, they had no progression of their disease and were 88 and 87 years old. The remaining patients continue to be observed with regular clinic visits and with no mortalities in this group.
Of the 11 patients, 8 underwent stereotactic biopsy aspiration followed by clinical and neuroimaging follow-up, 4 patients had insertion of an Ommaya reservoir and aspiration of the cystic component either as an initial or subsequent procedure. Duration of in-hospital stay for these procedures ranged from 24 to 48 hours. Four (4/11) patients had GKRS, and three (3/11) had IMRT following tumor cyst decompression. The choice of RT was determined by the dose constraints and proximity of the tumor to the optic nerves. One patient (1/11) in the cohort was treated at an outside facility with upfront GKRS (case no. 7). This patient subsequently developed visual deterioration associated with tumor progression and was treated with an IMRT boost [
Serial neuroendocrine evaluations revealed anterior pituitary hormone dysfunction in 8/11 of the patients. Two patients had improvement of their preexisting partial DI after intervention. Two patients (7 and 10) underwent VP shunts for obstructive hydrocephalus as the only invasive procedure prior to initiating RT. There was no formal neuropsychological evaluation prior to or after radiation treatments, making it difficult to objectively assess cognitive decline or improvement.
Response of the tumor to minimally invasive interventions
The tumors treated, were mixed tumors having both a solid as well as a predominantly cystic component. In our series, we defined progression as a symptomatic worsening with corresponding changes in neuroimaging after the last intervention, stable disease in cases where there was no symptomatic worsening but there could be fluctuation in the size of the cyst on MRI, and a complete response when there was neither symptomatic nor neuroimaging changes in the tumor. According to our criteria, there was one patient (1/11) in whom symptomatic progression occurred, along with radiological worsening of the solid component about 6 years following his treatment, at which time the tumor was amenable to surgical intervention (case 1). There were five (5/11) patients with stable disease, in whom two (2/11) had a fluctuation in cyst size on follow-up imaging. These two patients continue to remain clinically stable from their baseline initial evaluations, and at present have reduced cystic components without the need for further intervention. One (1/11) patient has progressed on imaging without symptomatic clinical worsening and continues to be observed. Two (2/11) patients have stable tumors on imaging following biopsy-cyst aspiration without radiation (case no. 6 and 8) and continue to be observed. Five (5/11) patients have been defined as having a complete response according to our criteria (case nos. 4, 7, 9, 10, 11), as they have no clinical or MRI changes after receiving focal RT (GKRS/IMRT or 32P). A fluctuation in the cyst dimensions is a common occurrence, subsequent to radiotherapy and although patients may not be symptomatic, they requiring diligent follow-up. Hence according to our criteria, only one patient (case no. 1) had a true progression, whereas the others are either stable or improved.
Eight patients (73%) had one or more endocrine deficits on presentation, hypogonadism being the most common at presentation five (5/11, 45%). There were two (2/11, 18%) who presented with hypothyroidism and (3/11, 27.2%) had pan-hypopituitarism. Diabetes insipidus was seen at presentation in one (1/11, 9%) patient (case 5).
In the course of their treatments, two new patients had developed pan-hypopituitarism and required hormone supplementation (preoperatively 3/11 (27.2%); postoperatively 5/11 (45%)) following intervention. The patients with hypothyroidism required hormone replacement during their follow-up. Transient diabetes insipidus was seen in one patient following a biopsy (case no. 8) and was permanent in one (case no. 5). The endocrine evaluation and management was performed by the neuroendocrinologist in the pre- and postoperative periods.
Eight patients (73%) had a visual field defect at presentation. Visual deficits included blurring of vision, diplopia, and field cuts and all affected patients were symptomatic. Six (6/8) of these patients had improvement in vision at last follow-up irrespective of the type of intervention, one had no change and one patient had a worsening of vision. Although one patient (case 7) did undergo GKRS and subsequently IMRT, her vision remained stable on follow-up.
Discharge time and associated medical morbidity
In-hospital stay was required for performing a biopsy, insertion of an Ommaya reservoir or placement of a VP shunt. Patients who underwent any of these procedures were discharged within 48 hours, with no associated complications related to hospital stay. Endocrine issues were taken care of by the neuroendocrinologist on an out-patient follow up basis. For adjuvant radiation procedures no hospital admission was required.
CPs are histologically benign tumors involving the sellar-suprasellar region, but often invading or adherent to adjacent critical structures. The intimate association between the tumor and the optic apparatus, hypothalamus, and pituitary gland makes treating them challenging. In reports published prior to the 1980s, recurrence rates for PCs varied from 30% to 50%.[
Of the various therapeutic strategies available, microsurgical resection is still considered to be the mainstay of management for adult patients with PCs.[
Although our cohort of patients is small, we observed good long-term control rates as seen by the duration of follow-up [
Moussa et al. treated 52 patients with cystic CPs using an Ommaya reservoir catheter to aspirate the cyst contents.[
Patient, a 55-year-old male presented with visual blurring and diplopia. MR imaging, T1WI axial and coronal preoperative (a and b) postcontrast shows a suprasellar cystic lesions measuring 2.8 × 2.5 × 3.3 cm. He underwent an Ommaya insertion with aspiration of the cystic component and was discharged a day later. His vision improved and he had no DI, neurological, or endocrine worsening. Follow-up MR imaging at 6 weeks, T1WI postcontrast axial and coronal (c and d) reveal the catheter in the cavity. At 1 year follow-up without any intervention his MRI T1WI axial and coronal (e and f) show a smaller cyst. The catheter may act as a stent gradually allowing cyst fluid to egress.
Algorithm outlining our decision pathway in the minimally invasive management of the patients included in this study with craniopharyngiomas. Our preferred choice for this subset is an initial aspiration and biopsy of the wall, although in the elderly, if the lesion is not amenable to any intervention, radiosurgery or radiation therapy may be a first line option There is no ‘one size fits all’ paradigm, as each patient presents with their own clinical and imaging findings upon which further treatment decisions should be based.
In literature, control rates appear to be better for solid or cystic tumors compared with mixed, large, and multi-cystic tumors, factors that predict poorer outcomes.[
With variable results from surgery, data from the Royal Marsden hospital[
Evolution in conformal RT techniques such as intensity modulated radiation therapy (IMRT), stereotactic fractionated radiotherapy, proton beam therapy, and SRS has been used more frequently for treating these tumors.[
Our patients receiving IMRT were treated with a standard dose of 54 Gy and the GKRS patients received marginal doses varying from 9 to 15 Gy with various series report marginal doses ranging from 3 to 25 Gy for long-term tumor control. Alternative dose plans prescribing lower margin doses with a high central dose at the same time avoid adverse radiation effects to adjacent organs at risk with good tumor control.[
Alternative treatment options include intracavitary radiation and chemotherapy,[
Although we tried to obtain histology in all our cases prior to treatment, this was not possible in four (4/11) patients. In two (2/4) of these patients, we attempted to biopsy the wall, however, doing a stereotactic biopsy of the cyst wall is technically challenging because of capsule shift after puncturing the wall, the associated adjacent vessels and the deep seated location of the tumor mass. Of the remaining two (2/11) patients, one patient had undergone VP shunting elsewhere and another had undergone shunt insertion and GKRS following which they had presented to us for further management. Even though other lesions have to be considered in the differential diagnosis of sellar–suprasellar neuroimaging,[
The overall 10-year survival rates for CPs presently range from 85% to 90%[
CP management requires an individualized, multi-disciplinary approach. Radical surgery is not justified in all cases, especially with adjunct treatment modalities now readily available. The good long-term control rates, low endocrine, visual and cognitive morbidity and minimal to no mortality, and short hospital stays make minimally invasive strategies a viable option for the long-term control of these tumors in the elderly and other well-selected patients.
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