Mastoid recurrence after radiotherapy for nasopharyngeal carcinoma
Mastoid recurrence after radiotherapy for nasopharyngeal carcinoma
Mastoid recurrence after radiotherapy for nasopharyngeal carcinoma
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CASE REPORT<br />
Russell B. Smith, MD, Section Editor<br />
MASTOID RECURRENCE AFTER RADIOTHERAPY FOR<br />
NASOPHARYNGEAL CARCINOMA: TWO CASE STUDIES<br />
Ximei Zhang, MD, Jingwei Luo, MD, Li Gao, MD, Guozhen Xu, MD<br />
Department of Radiation Oncology, Cancer Hospital, Chinese Academy of Medical Sciences (CAMS) and Peking<br />
Union Medical College (PUMC), Beijing, People’s Republic of China. E-mail: nqluo@yahoo.com.cn<br />
Accepted 14 January 2010<br />
Published online 31 March 2010 in Wiley Online Library (wileyonlinelibrary.com). DOI: 10.1002/hed.21391<br />
Abstract: Background. The mastoid is a rare site of <strong>nasopharyngeal</strong><br />
<strong>carcinoma</strong> <strong>recurrence</strong> <strong>after</strong> <strong>radiotherapy</strong>, and no<br />
relevant reports are currently in the literature.<br />
Methods. Two case reports are presented describing<br />
patients with a history of <strong>nasopharyngeal</strong> <strong>carcinoma</strong> who<br />
received primary radical <strong>radiotherapy</strong>. The first was treated by<br />
conventional <strong>radiotherapy</strong>, using 2-dimensional techniques,<br />
while the second was delivered by intensity-modulated radiation<br />
therapy (IMRT).<br />
Results. The patients presented with progressive mastoid<br />
<strong>recurrence</strong> <strong>after</strong> <strong>radiotherapy</strong> at 12 and 16 months, respectively.<br />
Clinical presentation of mastoid <strong>recurrence</strong> was similar<br />
to mastoiditis. Meanwhile, distant metastasis occurred in both<br />
cases. To date, 1 has died of distant metastasis, and the other<br />
is alive with disease stabilization following chemotherapy<br />
treatment.<br />
Conclusion. This is the first report of mastoid <strong>recurrence</strong> <strong>after</strong><br />
<strong>radiotherapy</strong> <strong>for</strong> <strong>nasopharyngeal</strong> <strong>carcinoma</strong>, which should<br />
alert clinicians to probe into the pathogenesis and pay attention<br />
to the relationship between mastoid <strong>recurrence</strong> and distant<br />
metastasis. VC 2010 Wiley Periodicals, Inc. Head Neck 33:<br />
1535–1538, 2011<br />
Keywords: <strong>nasopharyngeal</strong> <strong>carcinoma</strong>; <strong>recurrence</strong>; mastoid;<br />
<strong>radiotherapy</strong>; prognosis<br />
Nasopharyngeal <strong>carcinoma</strong> (NPC) is a common tumor<br />
among Chinese people. Radiotherapy is the main<br />
treatment administered <strong>for</strong> almost all NPC cases. Despite<br />
the fact that radical <strong>radiotherapy</strong> achieves a 5-<br />
year overall survival rate of more than 70%, 1,2 local<br />
failure remains the major problem <strong>for</strong> patients with<br />
advanced stages treated by conventional <strong>radiotherapy</strong>.<br />
Local <strong>recurrence</strong>s <strong>after</strong> conventional <strong>radiotherapy</strong><br />
<strong>for</strong> NPC are commonly located in the tissues<br />
within the irradiation field, such as ethmoid sinus<br />
and clivus; it is believed that local <strong>recurrence</strong>s might<br />
be related to low dose delivery there and inappropriate<br />
radiation field. 1 Thus far, no mastoid <strong>recurrence</strong>s<br />
have been detailed in the clinical literature.<br />
Correspondence to: Jingwei Luo, MD<br />
VC<br />
2010 Wiley Periodicals, Inc.<br />
CASE REPORT<br />
Case 1. A 45-year-old man was admitted to<br />
our hospital with complaints of decreased hearing<br />
in the left ear <strong>for</strong> 4 months and epistaxis <strong>for</strong><br />
half a month. Nasopharyngeal biopsy rendered<br />
the diagnosis of poorly differentiated squamous<br />
cell <strong>carcinoma</strong>. An MRI scan demonstrated a<br />
well-circumscribed mass in the left lateral wall<br />
extending into the ipsilateral parapharyngeal<br />
space and an enlargement of the bilateral cervical<br />
nodes (Figure 1). Further clinical workup,<br />
including chest radiograph, isotope bone scan,<br />
and abdominal ultrasonography scan, showed<br />
no evidence of distant metastasis and the disease<br />
was staged as T 2 N 2 M 0 , clinical stage III<br />
according to 2002 Union Internationale Contre<br />
le Cancer classification. The patient then underwent<br />
treatment with conventional radiation<br />
therapy using 2-dimensional techniques accompanied<br />
with a total dose of 70 Gy at 2-Gy daily<br />
fractions. The eustachian tubes were included in<br />
the radiation field and part of the temporal<br />
bones were out of the field, as shown in Figure 2.<br />
Images obtained at the end of <strong>radiotherapy</strong><br />
showed slightly enhanced mucosa in the nasopharynx;<br />
however, endoscopy revealed no residual<br />
tumor. The patient was then followed up<br />
regularly at the outpatient department without<br />
residual tumor evidence. About 1 year <strong>after</strong><br />
initial treatment, the patient presented to the<br />
same hospital with localized redness, swelling,<br />
and pain occurring in the mastoid area. An<br />
MRI showed an obvious mass in the left mastoid<br />
(Figure 3). A biopsy from the external ear<br />
was per<strong>for</strong>med and used to diagnose <strong>recurrence</strong>.<br />
Subsequently, the region of <strong>recurrence</strong><br />
was treated by intensity-modulated radiation<br />
therapy (IMRT) with a dose of 69.96 Gy in 33<br />
fractions. The tumor shrank slightly at the end<br />
NPC <strong>Mastoid</strong> Recurrence <strong>after</strong> RT HEAD & NECK—DOI 10.1002/hed October 2011 1535
FIGURE 1. MR image be<strong>for</strong>e treatment of patient 1 showed a<br />
well-circumscribed mass in the left lateral wall extending into<br />
the ipsilateral parapharyngeal space.<br />
FIGURE 2. The portal image of the radiation field of patient 1.<br />
The eustachian tubes were included in the field and part of the<br />
temporal bones were beyond the field.<br />
FIGURE 3. MR image showed mastoid <strong>recurrence</strong> in patient 1<br />
with an enhanced mass apparent in the left mastoid.<br />
of <strong>radiotherapy</strong>; however, multiple bone metastases<br />
occurred. After 4 cycles of chemotherapy<br />
consisting of vinorelbine (40 mg, days 1 and 8)<br />
and cisplatin (50 mg, days 2, 3, and 4), the disease<br />
remained progression-free at the last follow-up<br />
3 years <strong>after</strong> diagnosis.<br />
Case 2. A 43-year-old woman with a 7-month<br />
history of epistaxis and ear obstruction was<br />
diagnosed with undifferentiated NPC by <strong>nasopharyngeal</strong><br />
mass biopsy. The enhanced mass<br />
involved the right lateral wall extending to the<br />
parapharyngeal space, clivus, petrous apex, and<br />
right cervical nodes which were revealed on an<br />
MRI scan (Figure 4). After a detailed clinical<br />
workup, including chest radiograph, isotope<br />
bone scan, and abdominal ultrasonography scan,<br />
the disease was staged as T 3 N 1 M 0 , clinical stage<br />
III according to 2002 Union Internationale<br />
Contre le Cancer classification. Radiation therapy<br />
was per<strong>for</strong>med by IMRT using 9 coplanar<br />
beams (6-MV photon) 40 apart with a dose of<br />
7200 cGy divided into 33 fractions and delivered<br />
in 7 weeks. Figure 5 illustrates the tumor targets,<br />
dose distribution plan, and the portion of<br />
the temporal bones covered by 5000 cGy isodose<br />
curve. An enhanced T1-weighted MRI scan of<br />
the nasopharynx obtained at the end of the<br />
treatment revealed a high signal area, 1.5 cm <br />
1.5 cm, which was suspected to represent a<br />
region of persistent disease; however, endoscopic<br />
biopsy from the persistent area was unable to<br />
detect any residual tumor. The high-intensity<br />
1536 NPC <strong>Mastoid</strong> Recurrence <strong>after</strong> RT HEAD & NECK—DOI 10.1002/hed October 2011
FIGURE 4. MR image be<strong>for</strong>e treatment of patient 2 showed a<br />
well-circumscribed mass in the right lateral wall extending into<br />
the ipsilateral parapharyngeal space.<br />
feature was most likely radiation edema or<br />
infection of the <strong>nasopharyngeal</strong> mucosa. There<strong>for</strong>e,<br />
routine follow-up was administered and<br />
the disease remained <strong>recurrence</strong> free. Sixteen<br />
months later, the patient was admitted to the<br />
same hospital with complaints of right hearing<br />
loss and hemicrania. On physical examination,<br />
the patient was noted to have some signs of<br />
inflammation including localized redness, swelling,<br />
and increased skin temperature in the<br />
mastoid area which measured approximately<br />
3cm 2 cm. The patient received antiinflammatory<br />
treatment <strong>for</strong> half a month, which had<br />
no effect on suppressing the inflammation. An<br />
MRI scan revealed a relatively defined mass<br />
involving the right petrous bone and mastoid; a<br />
biopsy from the external ear proved <strong>recurrence</strong><br />
(Figure 6). At the same time, the B-ultrasonic<br />
examination showed multiple metastases in the<br />
liver and retroperitoneal lymph nodes. The<br />
patient was placed on palliative treatment with<br />
traditional Chinese medicine and died 3 months<br />
later.<br />
DISCUSSION<br />
The treatment failures <strong>for</strong> <strong>nasopharyngeal</strong> <strong>carcinoma</strong><br />
by <strong>radiotherapy</strong> are often attributed to<br />
local persistence or <strong>recurrence</strong>, regional lymph<br />
node metastasis, and distant metastasis. 1 The<br />
FIGURE 5. CT slice at the level of the mastoid <strong>recurrence</strong>. The<br />
gross target volume (GTV) is highlighted in red, and the planning<br />
target volume (PTV) in sky blue. Part of the mastoid is covered<br />
by 5000 cGy isodose curve. [Color figure can be viewed in<br />
the online issue, which is available at wileyonlinelibrary. com.]<br />
FIGURE 6. MR image showed mastoid <strong>recurrence</strong> in patient 2<br />
with a defined mass involving the right mastoid.<br />
NPC <strong>Mastoid</strong> Recurrence <strong>after</strong> RT HEAD & NECK—DOI 10.1002/hed October 2011 1537
local <strong>recurrence</strong> is usually identified in the<br />
nasopharynx cavity or adjacent organs such as<br />
sphenoid sinus, cavernous sinus, ethmoid sinus,<br />
and clivus; however, mastoid <strong>recurrence</strong> is<br />
extremely rare. To date, no related report citing<br />
such a find could be located in the clinical literature.<br />
It is speculated that eustachian tube<br />
involvement may play an important role during<br />
the process of mastoid <strong>recurrence</strong>. Anatomically,<br />
the eustachian tube links the lateral wall of the<br />
nasopharynx to the middle ear, which further<br />
connects mastoid cells through the mastoid<br />
antrum. Consequently, cancer cells may be able<br />
to spread to the tympanum along the mucosal<br />
lining then into the mastoid antrum. The 2<br />
patients we report here each presented with<br />
<strong>nasopharyngeal</strong> mass involving ipsilateral eustachian<br />
tube, there<strong>for</strong>e, it is logical to extrapolate<br />
that both of the mastoid <strong>recurrence</strong>s<br />
occurred ipsilaterally as described by the abovementioned<br />
anatomical hypothesis. The reason<br />
remains unknown why eustachian tube involvement<br />
with NPC is common, yet mastoid <strong>recurrence</strong><br />
is relatively rare.<br />
Both of our patients initially showed clinical<br />
features similar to mastoiditis when first diagnosed<br />
with mastoid <strong>recurrence</strong>, which suggests<br />
that the differential diagnosis <strong>for</strong> mastoid swelling<br />
<strong>after</strong> <strong>radiotherapy</strong> to treat the NPC should<br />
include both mastoiditis and tumor <strong>recurrence</strong>.<br />
Radiation-induced temporal bone tumors should<br />
also be considered depending on the timing from<br />
radiation therapy. In our 2 cases, the possibility<br />
of a radiation-induced temporal bone tumor having<br />
arisen from overlapping radiation fields was<br />
disproved according to Cahan’s criteria 3–5 ; the<br />
short interval period of <strong>recurrence</strong> (12 and<br />
16 months, respectively) and the same histology<br />
between the mass in the mastoid and the primary<br />
tumor indicated it was a recurrent tumor.<br />
To determine if inflammation was caused by<br />
infection related to posttherapy eustachian tube<br />
dysfunction, we administered antiinflammatory<br />
treatment and observed no therapeutic benefit.<br />
In addition, biopsy was critical to make the diagnosis<br />
clear. It is worth noting that both of our<br />
patients presented with distant metastasis<br />
almost at the same time of mastoid <strong>recurrence</strong>,<br />
there<strong>for</strong>e, it remains imperative that clinicians<br />
be vigilant when patients previously irradiated<br />
<strong>for</strong> <strong>nasopharyngeal</strong> malignancies present with<br />
mastoid complaints. It remains unclear whether<br />
patients with mastoid <strong>recurrence</strong> have an especially<br />
high risk of distant metastasis or poorer<br />
prognosis. Further studies are needed because<br />
of the rarity of such cases.<br />
CONCLUSIONS<br />
This is the first report on mastoid <strong>recurrence</strong> <strong>after</strong><br />
<strong>radiotherapy</strong> <strong>for</strong> <strong>nasopharyngeal</strong> <strong>carcinoma</strong><br />
and the relationship between mastoid <strong>recurrence</strong><br />
and distant metastasis. We also suggest<br />
that a definitive diagnosis of mastoiditis cannot<br />
be made unless imaging and biopsy rule out the<br />
possibility of a malignancy. Although the pathogenic<br />
mechanisms underlying NPC <strong>recurrence</strong><br />
in mastoid tissues remains unknown, the clinical<br />
reporting of these cases will encourage clinicians<br />
to consider this rare <strong>recurrence</strong> and pay<br />
attention to the relationship between mastoid<br />
<strong>recurrence</strong> and distant metastasis.<br />
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1538 NPC <strong>Mastoid</strong> Recurrence <strong>after</strong> RT HEAD & NECK—DOI 10.1002/hed October 2011