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Case Report

Laser Interstitial Thermotherapy for Treatment of Symptomatic Peritumoral Edema After


Radiosurgery for Meningioma
Christopher S. Hong1, Jason M. Beckta2, Adam J. Kundishora1, Aladine A. Elsamadicy1, Veronica L. Chiang1,2

Key words - BACKGROUND: Symptomatic peritumoral edema (PTE) is a known compli-


- Laser interstitial thermotherapy cation after radiosurgical treatment of meningiomas. Although the edema in
- Meningioma
- Neurosurgery
most patients can be successfully managed conservatively with corticosteroid
- Peritumoral edema therapy or bevacizumab, some medically refractory cases may require surgical
- Radiation necrosis resection of the underlying lesion when feasible. Laser interstitial thermother-
Abbreviations and Acronyms
apy (LITT) continues to gain traction as an effective therapeutic modality for the
FLAIR: Fluid-attenuated inversion recovery treatment of radiation necrosis where its biggest impact is through the control of
LITT: Laser interstitial thermotherapy peritumoral edema.
MRI: Magnetic resonance imaging
PTE: Peritumoral edema - CASE DESCRIPTION: A 56-year-old woman with neurofibromatosis 2 pre-
WHO: World Health Organization sented with a symptomatic, regrowing left frontotemporal lesion that had pre-
From the Departments of 1Neurosurgery and 2Therapeutic
viously been radiated, then resected with confirmed recurrence of grade I
Radiology, Yale University School of Medicine, New Haven, meningioma, and subsequently radiated again for lesion recurrence. Given her
Connecticut, USA history of 2 prior same-side craniotomies, including a complication of wound
To whom correspondence should be addressed: infection, she was not a candidate for further open surgical resection. Having
Veronica L. Chiang, M.D.
[E-mail: veronica.chiang@yale.edu]
failed conservative management, she underwent LITT with intraoperative biopsy
Citation: World Neurosurg. (2020) 136:295-300.
demonstrating viable grade I meningioma. Postoperatively, she demonstrated
https://doi.org/10.1016/j.wneu.2020.01.143 radiographic marked, serial reduction of PTE and experienced resolution of her
Journal homepage: www.journals.elsevier.com/world- symptoms.
neurosurgery
- CONCLUSIONS: This case demonstrates that LITT may be a viable alternative
Available online: www.sciencedirect.com
1878-8750/$ - see front matter ª 2020 Elsevier Inc. All
treatment for patients with meningioma with symptomatic PTE who have failed
rights reserved. medical therapy and require surgical intervention.

INTRODUCTION
The development of symptomatic peritu- procedure for both the treatment of path- knife radiosurgery 7 years prior to tumor
moral edema (PTE) causing focal neuro- ologic diagnoses and delivery of ablative in the same location (18 Gy to the 50%
logic deficits, seizures, and intracranial therapy. The most robust indication for isodose line) and 3 adjacent tumors, fol-
hypertension is a known complication LITT is the treatment of symptomatic lowed 1 year later by a large fronto-
after radiosurgical treatment of meningi- radiation necrosis after previous radio- temporal craniotomy for resection of the
omas.1-3 Its pathophysiology has been surgery for brain metastases.8-11 In this regrowing and symptomatic dominant left
attributed to both factors related to radi- report, we present a patient, who having frontotemporal lesion and 4 other adjacent
ation type and dosing and characteristics exhausted medical, surgical, and radio- tumors. Pathology confirmed viable World
intrinsic to the treated meningioma.4-7 therapy options, underwent successful Health Organization (WHO) grade I
First-line therapy for symptomatic PTE LITT for treatment of symptomatic PTE psammomatous meningioma with areas
after meningioma radiation remains high- related to her tumor. As such, LITT may be a of focal necrosis in all resected specimens.
dose corticosteroids, but alternative treat- viable alternative treatment of PTE after Surgery was complicated by a wound
ments have included bevacizumab or sur- radiosurgical treatment of meningioma. infection resulting in the removal of the
gical resection in those with accessible original bone flap and subsequent
lesions. However, in cases of persistent replacement with a cranioplasty. Five years
PTE refractory to corticosteroids and CASE DESCRIPTION after craniotomy, given progressive tumor
otherwise not amenable to bevacizumab or The index patient is a 56-year-old women regrowth in the previously surgically
surgical resection, further treatment op- with known neurofibromatosis type 2. As resected region associated with develop-
tions remain limited. part of her extensive neurosurgical history, ment of recurrent, albeit mild, right-sided
Magnetic resonance imaging (MRI)e she had previously undergone multiple hemiparesis, she underwent repeat
guided laser interstitial thermotherapy resections of both cranial and spinal me- gamma knife radiosurgery. A total of 15 Gy
(LITT) has become an increasingly ningiomas. In particular, in the region of to the 50% isodose line was delivered to
popular minimally invasive neurosurgical interest, she had undergone initial gamma the dominant left frontotemporal lesion

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CASE REPORT
CHRISTOPHER S. HONG ET AL. LITT FOR EDEMA IN IRRADIATED MENINGIOMAS

left frontotemporal extra-axial lesion


measuring 2.2  2.3  3.3 cm (16.7 cm3)
with significant increase in the amount of
surrounding T2 fluid-attenuated inversion
recovery (FLAIR) changes extending into
the adjacent temporal lobe (Figure 2AeD).
Her symptoms improved significantly with
steroids, but her clinical course was
complicated by development of shingles,
significant weight gain, and severe
gastrointestinal reflux. Bevacizumab was
felt to be contraindicated given her recent
fracture. Repeat surgical resection was
considered, but because of her history of
multiple same-sided craniotomies and
prior infection, LITT was proposed with the
possibility of craniotomy salvage if needed.
The patient tolerated the LITT proced-
ure without any significant events. An
alternative trajectory to her lesion avoiding
her prior craniotomies was chosen allow-
ing the laser fiber also to be introduced
along the long axis of the lesion
(Figure 3AeC). LITT was performed using
the full-fire 3.3-mm diameter laser fiber
and the NeuroBlate system (Monteris
Medical, Minneapolis, Minnesota, USA) in
accordance with our previously published
institutional experience.8,12 Complete
ablation was achieved (Figure 3D and E).
Biopsy was also performed prior to
thermocoagulation and again
demonstrating viable WHO grade I
psammomatous meningioma with a
Ki-67 index <5%.
An MRI obtained 2 weeks after treatment
demonstrated similar but slightly
decreased T2 FLAIR signal and thinner rim
enhancement compared with preoperative
imaging. Clinically, she was without motor
deficits, and her speech had returned to
baseline, in the setting of a slow steroid
taper, which was eventually successfully
weaned off by the time of her 6-week sur-
veillance MRI. At 6 weeks there was further
reduction in perilesional edema associated
with a notable decrease in the size of the
contrast-enhancing lesion (Figure 2EeH).
Scans at 3-month follow-up revealed a
further decrease in size of the ablated lesion,
Figure 1. Gamma knife radiosurgery plan. Representative T1-weighted magnetic resonance imaging and imaging at 1-year post-LITT showed res-
after gadolinium administration showing (A) coronal, (B) sagittal, and (C) axial slices of 15 Gy to the olution of T2 FLAIR edema and minimal
50% isodose line (inner yellow line), 8 Gy to the 25% isodose line (inner green line), and 4 Gy to the residual enhancement (Figure 4). At the last
12.5% isodose line (outer green line).
follow-up, 3 years after LITT, the ablated
lesion remained as a small area of nodular
(Figure 1). Six months after this second right-sided hemiparesis resulting in a enhancement with no perilesional edema, and
gamma knife treatment, she presented fall and fracture of her right wrist. the patient remained neurologically asymp-
with new onset aphasia and worsening MRI demonstrated a persistent, dominant tomatic from this lesion (Figure 4EeH).

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CASE REPORT
CHRISTOPHER S. HONG ET AL. LITT FOR EDEMA IN IRRADIATED MENINGIOMAS

Figure 2. Preoperative and 2-week follow-up magnetic resonance imaging fluid-attenuated inversion recovery (FLAIR). Postoperative MRI 2 weeks
(MRI) after laser interstitial thermotherapy (LITT). Preoperative MRI of the after LITT is shown on (E) axial and (F) coronal T1-weighted MRI after
ablated lesion is shown on (A) axial and (B) coronal T1-weighted MRI after gadolinium administration and (G) axial and (H) coronal T2-weighted FLAIR.
gadolinium administration and (C) axial and (D) coronal T2-weighted

DISCUSSION symptomatic. Although high-dose corti- predominantly described its use in


Meningiomas remain the most common costeroids remain first-line therapy, in intra-axial pathologies for primary and
primary brain tumor and differ from most refractory cases, few alternative medical secondary brain tumors and radiation ne-
intra-axial pathologies by nature of their therapies exist. Other systemic therapies crosis.12,27,28 Recently, its use was
extra-axial location and typically benign with mixed results but potential efficacy in affirmed in a multicenter prospective
pathology. Although first-line treatment meningiomas are somatostatin analogues study for the treatment of radiosurgical
remains surgical resection, which fortu- in somatostatin receptor-positive tu- failure or complication in brain
nately is curative in most cases of gross or mors,17-19 interferon alpha,20,21 and metastases.29 Furthermore, LITT has
near-total resection, adjuvant radiation bevacizumab.22,23 Surgical resection is been shown to compare favorably with
may be necessary for higher-grade tumors possible in patients with accessible craniotomy for the same patient
(WHO grade II or III) or in patients with lesions, but when these therapies fail population.9
syndromic conditions such as neurofibro- and surgery is not an option, few Development of symptomatic PTE is a
matosis 2, multiple endocrine neoplasia 1, alternative treatments exist. known complication after radiosurgery to
Cowden syndrome, and Werner syn- LITT for meningiomas has also been meningiomas, with rates ranging widely
drome, who are at high risk of multifocal described in a handful of prior case series, from 5% to 46%2,3; however, larger series
tumor recurrence.13-16 Radiosurgery is typically involving patients with multiple have reported lower values closer to 5%e
often first-line treatment of these tumors prior craniotomies for whom further sur- 10%.30,31 However, it is often difficult to
given patient reluctance and intrinsic risks gery was deemed high risk or because of determine whether the main cause of
of multiple craniotomies. Radiosurgery, patient preference.24-26 As this report PTE is related to tumor progression or
especially when repeated to the same demonstrates, however, LITT may be a postradiation changes, with a recent
location, carries a risk of development of viable surgical alternative for the treatment study demonstrating that previously
delayed PTE that can become significantly of symptomatic PTE. Prior studies have radiated meningiomas with predominant

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CASE REPORT
CHRISTOPHER S. HONG ET AL. LITT FOR EDEMA IN IRRADIATED MENINGIOMAS

Figure 3. Laser interstitial thermotherapy (LITT) trajectories and demonstrate trajectory of the catheter to administer LITT within
thermal damage threshold lines. Representative intraoperative the center of the lesion. Representative axial slices of thermal
(A) coronal, (B) sagittal, and (C) axial slices of T1-weighted damage threshold lines are shown for (D) protein denaturation in
magnetic resonance imaging after gadolinium administration yellow and (E) cell death in blue.

radiation necrosis on biopsy responded that there was significant and prompt edema. Further studies that compare
most readily to low-dose bevacizumab reduction of this edema after ablative biopsy of the center versus the periphery
compared with samples with mostly therapy. Based on these observations, we of these lesions may confirm this
viable/recurrent tumor.22 In the case of our hypothesize that the centers of previously possibility.
index patient, intraoperative biopsy radiated meningiomas may harbor viable
demonstrated viable tumor and the tumor, but these lesions may develop
etiology of the perilesional edema symptomatic secondary radiation necrosis CONCLUSIONS
remains uncertain. The edema however pathology at the periphery of the lesion As LITT continues to become increasingly
responded to LITT in a time frame interface with normal brain tissue which commonplace in the neurosurgical arma-
similar to radiation necrosis pathology in may then be the cause of the perilesional mentarium, the applications for this

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CASE REPORT
CHRISTOPHER S. HONG ET AL. LITT FOR EDEMA IN IRRADIATED MENINGIOMAS

Figure 4. Longer-term follow-up surveillance magnetic resonance imaging fluid-attenuated inversion recovery (FLAIR). Postoperative MRI at 3 years
(MRI) after laser interstitial thermotherapy (LITT). Postoperative MRI at 1 after LITT is shown on (E) axial and (F) coronal T1-weighted MRI after
year after LITT is shown on (A) axial and (B) coronal T1-weighted MRI after gadolinium administration and (G) axial and (H) coronal T2-weighted FLAIR.
gadolinium administration and (C) axial and (D) coronal T2-weighted

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changes, but not viable tumor tissue, may Management of intracranial metastatic disease rights reserved.

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