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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 />

REFERENCES<br />

1. Lee AWM, Perez CA, Law SCK, Chua DTT, Wei WI,<br />

Chong V. Nasopharynx. In: Perez CA, Brady LW, Halperin<br />

EC, editors. Principles and Practice of Radiation Oncology.<br />

Philadelphia: Lippincott Williams & Wilkins; 2008.<br />

p 1583–1586.<br />

2. Yi JL, Gao L, Huang XD, Li SY, Luo JW, Cai WM. Nasopharyngeal<br />

<strong>carcinoma</strong> treated by radical <strong>radiotherapy</strong><br />

alone: ten-year experience of a single institution. Int J<br />

Radiat Oncol Biol Phys 2006;65:161–168.<br />

3. Cahan WG, Woodard HQ, Higinbotham NL. Sarcoma arising<br />

in irradiated bone; report of 11 cases. Cancer 1948;<br />

1:3–29.<br />

4. Lustig LR, Jackler RK, Lanser MJ. Radiation-induced<br />

tumors of the temporal bone. Am J Otol 1997;18:230–<br />

235.<br />

5. Goh YH, Chong VF, Low WK. Temporal bone tumours in<br />

patients irradiated <strong>for</strong> <strong>nasopharyngeal</strong> neoplasm. J Laryngol<br />

Otol 1999;113:222–228.<br />

1538 NPC <strong>Mastoid</strong> Recurrence <strong>after</strong> RT HEAD & NECK—DOI 10.1002/hed October 2011

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