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Mar 30, 2017 - Robotic radiosurgery of head and neck paragangliomas: a single institution experience. ˙Ilker Tosun1. Banu Atalar2. Bilgehan Sahin2. Görkem ...
Received: 5 December 2016

Revised: 29 March 2017

Accepted: 30 March 2017

DOI: 10.1111/ajco.12695

ORIGINAL ARTICLE

Robotic radiosurgery of head and neck paragangliomas: a single institution experience ˙Ilker Tosun1

Banu Atalar2

Gökhan Aydin2

Bülent Yapici2

1 Radiation Oncology Division, Acıbadem

2 Bilgehan Sahin ¸

Görkem Güngör2

Enis Özyar2

Abstract

˘ Turkey Bodrum Hospital, Mugla, 2 Department of Radiation Oncology, Acibadem

R Aim: CyberKnife is a robotic stereotactic radiotherapy system. The aim of this study is to evalR uate the effectiveness and the safety of CyberKnife on treating head and neck paragangliomas

University, Istanbul, Turkey Correspondence ˙Ilker Tosun, Ortakent Mah Gölba¸sı Sok No:11 ˘ Turkey. 48420, Bodrum – Mugla, Email: ilker.tosun@acibadem.com.tr

and to report our results.

The study has not been previously presented.

body paragangliomas and three cases had tympanic paragangliomas. One patient had bilateral

Methods: Between March 2009 and June 2014, 12 patients with head and neck paragangliomas have been referred to our clinic: in three cases had jugular paragangliomas, five cases had carotid neck paragangliomas (right neck; carotid body paraganglioma, left neck; jugular pargangliomas). R up to a total All of them received fractionated stereotactic radiotherapy with CyberKnife

median dose of 24 Gy (reference isodose 67–90%). The median tumor volume was 35.5 cc (range, 5.3—113.8 cc). The median follow up was 30 months (range, 0–66 months). Local tumor control was assessed according to RECIST criteria on follow-up imaging studies. Results: There were no acute or late toxicity related with stereotactic radiotherapy after treatment. No local tumor progression was observed on magnetic resonance imaging and none of our patients showed progressive clinical status. Seven tumors shrinked in size (54%). Five tumors (46%) had stable size during follow up. Local control rate was 100%. Conclusion: Stereotactic radiotherapy is a good alternative to surgery for the treatment of head and neck paragangliomas coming up with a clear benefit of acute and late side effects. R CyberKnife seems to be a safe and efficient system treating head and neck paragangliomas.

KEYWORDS

CyberKnife radiosurgery, glomus jugulare tumor, paraganglioma, radiosurgery, radiotherapy

1

INTRODUCTION

older than abdominal PGLs at the time of diagnosis (43 years vs. 36 years of age).5 The male-to-female ratio of PGLs are usually

Paragangliomas (PGLs) are rare neuroendocrine tumors, which are

equal.6

paraganglia.1

Most of PGLs have benign nature and have manageable size (the

Parasympathetic ganglia-derived PGLs are mostly located at the neck

average volume was 17.1 cm3 for HNPGLs and 94.1 cm3 for other

and skull base region.2 Head and neck PGLs (HNPGLs), formerly

tumors).2 A wait and scan approach is often advised as the majority

known as glomus tumors mostly occurs in the bifurcation of carotid

of HNGPLs have slow growing character.7 Although HNPGLs are gen-

arteries (carotid body PGL), tympanic area (tympanic PGL), the dome

erally benign tumors, tumor bulk may result neurovascular structure

of internal jugular vein (jugular PGL) and along the vagal nerve (vagal

damage by cranial nerve deficits and cause serious morbidity. The main

PGL).3

treatment options for HNPGLs are surgical resection and radiation

derived from either parasympathetic or sympathetic

The specific incidence of PGLs are unknown as they are usually reported together with other pheochromocytomas. The annual incidence of pheochromocytoma/PGLs has been estimated to be year.4

therapy (RT). Although tumors can be surgically resected it may lead to neurovascular injury approximately in 60% of patients.8 RT is an alternative treatment strategy for HNPGL patients.

PGLs are mostly discovered in

HNPGLs can be treated by both conventionally fractionated RT

the fourth and fifth decades of life.3 HNPGL patients tend to be

(CFRT) and stereotactic radiotherapy (SRT) approaches. Local

0.8 per 100.000 person /

Asia-Pac J Clin Oncol. 2017;1–5.

wileyonlinelibrary.com/journal/ajco

c 2017 John Wiley & Sons Australia, Ltd 

1

2

CyberKnife

CyberKnife

CyberKnife

CyberKnife

CyberKnife

CyberKnife

and have a better dose distribution which spares healthy struc-

CyberKnife

of HNPGLs.16 Compared to CFRT, SRT provides better precision

CyberKnife

been widely used as a promising treatment modality for treatment

CyberKnife

effects for critical structures.12 Within the past 15 years, SRT has

CyberKnife

However, administering CFRT to wide fields may cause serious side

CyberKnife

Treatment machine

control rate with CFRT is reported to be between 79% and 100%.11

CyberKnife

TOSUN ET AL .

no follow-up

66 months

49 months

52 months

57 months

no follow-up

28 months

36 months

33 months

found to be safe and efficacious for the treatment of HNPGLs.13–15

22 months

USA) and linac-based stereotactic radiotherapy systems have been

2 months

Follow-up

R (CK; Accuray Incorporated, Sunnyvale, CA, Sweden), CyberKnife

17 months

R (GK; Elekta AB, Stockholm, tures around the target. Gamma Knife

7 Gy

7 Gy

7 Gy

10 Gy

8 Gy

8 Gy

5 Gy

5 Gy

We retrospectively reviewed 12 HNPGL patients treated with CK in

5 Gy

(SRS).16

8 Gy

to be 97% and 95% respectively after stereotactic radiosurgery

8 Gy

Dose/fraction

patients, tumor control and clinical control rates were reported

8 Gy

In a meta-analysis, which included a total of 335 glomus jugulare

21 Gy Yes

21 Gy

21 Gy Yes

Yes

24 Gy

30 Gy No

No

25 Gy

24 Gy No

No

25 Gy

24 Gy

24 Gy

25 Gy Yes

between March 2009 and June 2014. From our patient database,

Yes

Total of 12 patients with HNPGLs were treated with SRT at our clinic

No

METHODS AND MATERIALS

Yes

Total dose Prior surgery

2

Yes

as describing our results.

24 Gy

our clinic. This report evaluates effectiveness and safety of CK as well

C1

D2

D2

D2

C2

C2, C2

C3

staging (Table 2).

D2

eters are summarized in Table 1. Fisch classification is used for tumor

D2

and tumor response. Patient characteristics and radiotherapy param-

C

rence status, radiotherapy parameters, acute and late complications

C

Fisch classification

der, tumor size and location, pretreatment symptoms and signs, recur-

D2

we identified patient and tumor characteristics including age, gen-

created by defining a 2 to 5 mm margin to GTV (median 3 mm). The

Glomus caroticum 49 12

Female

Glomus jugulare/nasopharyngeal componant

Glomus timpanicum Female

Male

40

61

10

11

Left carotis bulbus, glomus caroticum

Glomus caroticum Female

Right: glomus caroticum Left: glomus jugulare Male

Glomus timpanicum

Glomus timpanicum Female

Male

Glomus caroticum

Tumor localization

Glomus jugulare

Glomus jugulare Female

Male

Female

41

29

8

9

able. GTV was defined according to the radiological findings. PTV was

27

were fused with contrast-enhanced T1- and T2-weighted MRI if avail-

44

each patient underwent a thin (1 mm) slice CT scan and those images

4

sion: 3.5.4) was used for treatment planning. For target delineation,

5

R (verand received fractionated SRT with curative intent. MultiPlan

65

All patients were treated with CK G4 system using 6-MV photons

34

diagnosis.

2

Five patients had no prior treatment therewith no histopathological

3

tary PGLs. Seven patients had prior surgery and recurrent tumor.

Gender

tation and family history, this patient was regarded as a heredi-

Male

succinate dehydrogenase D gene. Because bilateral disease presen-

Age

had PGLs as well and genetic studies have revealed a mutation in

43

left neck; jugular PGL). It was learned that his father and sister

1

One patient had bilateral neck PGLs (right neck; carotid body PGL,

Patient number

patients), vertigo (one patient) and hypoglossal paralysis (one patient).

TA B L E 1

ing loss (three patients), neck mass (two patients), hoarseness (two

Patient characteristics and radiotherapy modality

other symptoms presented were pulsatile mass (three patients), hear-

Male

toms were pulsatile tinnitus (five patients) and pain (five patients),

36

Type C (50%) or D (50%) disease. The most common presenting symp-

57

three patients had tympanic PGLs. All patients presented either with

6

patients had jugular PGLs, five patients had carotid body PGLs and

7

this report. The median age was 42 years (range, 27–65 years). Three

Glomus caroticum

Six male (50%) and six female (50%) patients were analyzed in

3

TOSUN ET AL .

TA B L E 2

Fisch classification of glomus tumors

3

RESULTS

A

Limited to middle ear cleft

B

Limited to the tympanomastoid area

3.1

C

Involving the infralabyrinthine compartment and petrous apex of the temporal bone

All patients tolerated radiotherapy well. No acute or late toxicity

D1

Intracranial extension 2 cm in greatest dimension

Clinical response

related with SBRT was seen after treatment. None of the patients developed new cranial nerve deficits. All of our patients’ clinical status was stable and one patient had symptomatic relief.

3.2

Tumor control

median number of fractions was 3 (range, 3–5) and the median fraction

MRI was performed for all patients after treatment. No local tumor

per dose was 7 Gy (range, 5–10 Gy). Total dose was ranged between 21

progression was observed. Seven of 13 tumors (54%) had partial

and 30 Gy (median 24 Gy) and prescription isodose lines were selected

response and five tumors (46%) were considered as stable disease dur-

between 67% and 90% (median 75%; Figure 1). The median overall

ing follow-up. Decreases in tumor volume did not correlate with symp-

treatment time was 3 days (range, 2–5 days). The median maximum

tomatic improvement. Overall local control rate of 100% was obtained.

tumor diameter was 56 mm (range, 26–92 mm) and the median tumor volume was 35.5 cc (range, 5.3–113.8 cc). In case with bilateral disease

3.3

Toxicity

both lesions at the neck were treated with fractionated SRT using CK system one month apart. The median follow up was 30 months (range, 0–66 months). Follow up time was calculated from the last day of SRT procedure. Two

Following SBRT none of our patients experienced acute or late toxicities including carotid blowout syndrome, vertigo, hearing loss, otorrhoe, pulsatile tinnitus and pulsatile mass.

patients have no follow up. The responses to treatment were evaluated by serial physical and imaging examinations performed after completion of SRT by an experienced radiologist and radiation oncologist.

4

DISCUSSION

Stabilization or absence of symptoms were considered as clinical control. Local tumor control was assessed according to RECIST criteria17

HNPGLs treatment strategy is based upon retrospective data; the

on follow-up imaging studies.

optimal management relies on clinical and radiological assessment.18

F I G U R E 1 Definitive RT planning with CK. A total dose of 24 Gy (8 Gy/fraction, prescribed isodose: 82%) to a Fisch C class left glomus jugulare tumor (tumor size; 26 × 22 × 22 mm, volume; 5340 cc) [Colour figure can be viewed at wileyonlinelibrary.com]

4

TOSUN ET AL .

Complication risks have to be minimized while deciding on curative

alternative to surgery particularly where operation would be highly

approach. Three main roads up to management exist; observation,

dangerous for vascular and/or neural structures around. External RT

resection and primary RT.

history of HNPGLs starts with conventional fractionation. Daily doses

Although these tumors reflect a broad spectrum of clinical behav-

of 1.8 to 2 Gy RT, five days/week, delivered between 45 and 50.4

ior, fortunately most of them are in benign behavior.2 In a retrospec-

Gy to the PTV. Lower doses than 40 Gy showed higher risk for local

tive study7 including 48 HNPGL patients, patients were followed with-

recurrence.10 Hinerman et al.23 presented their 35 years results of

out any treatment. The median tumor growth rate was 1 mm annu-

HNPGL treatment with external RT. A total of 104 patients with 121

ally and the median tumor doubling time was reported as four years.

PGLs received RT. A total of 104 tumors had no prior treatment. A

In another study, 43 patients were followed with serial imaging (CT

total of 89 PGLs received CFRT, 11 PGLs received intensity modulated

or MRI) and 70% of patients were asymptomatic. Forty-two percent

radiotherapy (IMRT), 15 PGLs received fractionated SRT and 6 PGLs

of tumors were stable during five years follow-up time. Twenty per-

underwent SRS. The overall crude local control rate for all 121 lesions

cent of tumors’ size was reduced. Thirty-eight percent of tumors grew

was 95%, with an ultimate local control rate of 96%. They reported

but the mean growth rate was 0.2 cm annually.19 From this point of

20 mild or moderate complications in 104 patients (19%) following

view, initial observation might be the choice of decision for small and

RT. There were no severe complications. They concluded that exter-

asymptomatic nonsecreting HNPGLs while close monitoring is war-

nal RT provides high tumor control therewith minimal complications.

ranted. Seven of our patients had prior surgical intervention and all of

Despite the scarcity of literature evidence, RT may not be as effective

our patients received curative SBRT. All treatment options including

as surgery for catecholamine secreting HNPGLs. RT seems to be impo-

observation were discussed with patients as well.

tent in reducing catecholamine secretion.26 Surgery must be taken into

Historically, surgical removal of HNPGLs was the first treatment choice.20

consideration for these specific subgroup.

The major complication associated with surgery is cra-

After SRS was introduced for HNPGLs, CFRT usage shifted towards

nial nerve damage.16,18,20,21 Less common complications are vascular

SRS in many clinics. SRS enables clinician to spare healthy structures

problems (stroke and bleeding).18,21 In a systemic review of retrospec-

while prescribing ablative radiation doses for the tumor.

tive series which includes 1084 juguler PGL patients, local control was

Theoretically, SRS has lower complication rates than CFRT depend-

85% and 965 new cranial nerve deficits were reported after surgery.21

ing on smaller radiation fields and it provides shorter treatment

During vagal PGL resection, vagal nerve deficit is unavoidable and

times.24 Guss et al.16 published a meta-analysis including 19 studies

nerve-preserving try-outs usually bring out postoperative failure of

with a total of 335 glomus jugulare patients who are treated with SRS.

complete resection as well as nerve palsies.20,21 Green et al.22 reported

They reported 97% tumor control (unchanged or reduced tumor vol-

high rate of cranial nerve deficits after surgery up to 46% of patients,

ume) and 95% clinical control (unchanged or improved clinical sta-

in addition, they found out a 15% decrease in quality of life. As a result

tus). In another meta-analysis,25 46 publications describing outcomes

of experience, surgical removal of HNPGLs potentially cause morbidity

for 869 jugular PGL patients, SBRT success was shown compared to

and mortality especially for tumors surrounded by critical structures.

surgery. Tumor control rates for gross total resection alone (GTR; 351

Moreover, some surgeons use preoperative arterial embolization for

patients) was 86%, subtotal resection alone (STR; 82 patients) was

HNPGLs. This procedure adds some additional risks for severe mor-

69%, STR plus SBRT (97 patients) was 71%, and SBRT alone (339

bidity such as blindness, cranial nerve deficits, stroke and death. For-

patients) was 95%. Furthermore, SBRT alone group was more challeng-

tunately, the risk is reported to be in low range (0–13%).14,20 Six of our

ing in terms of treatment success. Fisch class D tumor rates were the

patients had prior surgery and two of them had arterial embolization

highest in SBRT alone group (57%); GTR (19%), STR (29%) and STR plus

before surgery. One patient had right vision loss, left hemiplesia, right

SBRT (9%). Cranial nerve (CN) neuropathy rates for patients follow-

facial paralysis, hypoglossus paralysis after embolization. Despite small

ing GTR was worse than SRS (CN IX [38% vs 9.7%], X (26% vs 9.7%),

patient size, our results support the risk related with embolization.

and XI (40% vs 12%), although the rates of CN XII deficits were compa-

In this study, we retrospectively evaluated 10 HNPGL patients

rable (18% vs 8.7%). Current study is well matched with similar stud-

treated with fractionated SRT in our clinic. After a median 30 months

ies published in literature. All of our patients had stable or improved

follow-up, our findings indicated either stable disease or partial

clinical status. Lim et al. also revealed parallel results with us.15 How-

response. Local tumor control was 100%. Our follow-up time is not

ever, treatment success with SBRT doesn’t seem to be correlated with

covering long time range but our primary goal here is evaluating effi-

tumor shrinkage.18 This may create radiological assessment problems.

cacy and safety of CK for HNPGL tumors. Although this retrospec-

We achieved tumor regression in seven patients (1 patient had >50%)

tive analysis involves limited patient size, our results support the of

and remaining five patients had stable tumor. We believe that radiolog-

CK for HNPGL treatment. There is lack of data in literature regard-

ical and clinical status must be considered together while evaluating

ing HNPGLs, which are primarily treated with CK.15 To date, published

HNPGLs during follow-up.

meta-analyses showed that GK experience is

overwhelming.16,25

Because of lack of previously published CK data, we chose fractionated SBRT, perhaps much safer than single fraction SBRT, and none of our

5

CONCLUSION

patients came up with post-SBRT complications. Local control rates of RT ranged between 79% and 100% in

HNPGLs are usually non catecholamine-secreting benign tumors. Sur-

the literature9 and it seems to be a reasonable approach as an

gical removal provides good local control rates but comes up with

5

TOSUN ET AL .

increased risk of complications. RT is a good alternative for surgery. FCRT and SBRT local control rates are comparable and also they are higher than surgery with far less complication risk. Besides this, SBRT must be planned and administered meticulously since an inaccuracy may cause marginal miss and tumor recurrence. In our clinic, we have treated HNPGL patients with CK (fractionated SBRT) and achieved 100% local control. No patient had post-SBRT complications and none of them presented with disease progression during follow-up. Our results are well matched with published literature data. CK seems to be a safe and efficient system treating HNPGLs. Current study is valuable supporting literature data; however, our results must be confirmed by studies with larger patient size and longer follow-up. CONFLICT OF INTEREST No conflict of interest was declared by the authors. FINANCIAL DISCLOSURE The authors declared that this study has received no financial support.

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How to cite this article: Tosun ˙I, Atalar B, Sahin ¸ B, et al. Robotic radiosurgery of head and neck paragangliomas: a single institution experience. Asia-Pac J Clin Oncol. 2017;00:1-5. https://doi.org/10.1111/ajco.12695