Name: surgical treatment of an endolymphatic sac tumor
Uploaded: 2023-05-26T13:10:00
Description: Watch this Scientific Journal Video about Surgical Treatment of an Endolymphatic Sac Tumor at JoVE.com

The local ethical committee approved the publication of this clinical case. A written informed consent form was collected from the patient.
1. Clinical and audiological evaluation
<ol>
	<li>Collect personal or familiar history of VHL disease or compatible symptoms from the patient.</li>
	<li>Perform a neurological examination to investigate central causes. Ensure that there is no pathological cause to be underlined and the facial nerve function is normal.</li>
	<li>Assess ex...

<ol>
	<li>Heffner, D. K. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Low-grade+adenocarcinoma+of+probable+endolymphatic+sac+origin+A+clinicopathologic+study+of+20+cases.">Low-grade adenocarcinoma of probable endolymphatic sac origin A clinicopathologic study of 20 cases.</a> Cancer. 64 (11), 2292-2302 (1989).</li><li>Lonser, R. R., Baggenstos, M., Kim, H. J., Butman, J. A., Vortmeyer, A. O. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=The+vestibular+aqueduct:+site+of+origin+of+endolymphatic+sac+tumors.">The vestibular aqueduct: site of origin of endolymphatic sac tumors.</a> J Neurosurg. 108 (4), 751-756 (2008).</li><li>Megerian, C. A., et al. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Endolymphatic+sac+tumors:+histopathologic+confirmation,+clinical+characterization,+and+implication+in+von+Hippel-Lindau+disease.">Endolymphatic sac tumors: histopathologic confirmation, clinical characterization, and implication in von Hippel-Lindau disease.</a> Laryngoscope. 105, 801-808 (1995).</li><li>Zanoletti, E., et al. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Endolymphatic+sac+tumour+in+von+Hippel-Lindau+disease:+management+strategies.">Endolymphatic sac tumour in von Hippel-Lindau disease: management strategies.</a> Acta Otorhinolaryngol Ital. 37 (5), 423-429 (2017).</li><li>Mendenhall, W. M., et al. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Current+Treatment+of+Endolymphatic+Sac+Tumor+of+the+Temporal+Bone.">Current Treatment of Endolymphatic Sac Tumor of the Temporal Bone.</a> Adv Ther. 35 (7), 887-898 (2018).</li><li>Wu, N., et al. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Surgical+management+of+endolymphatic+sac+tumor:+classification,+outcomes+and+strategy.+A+single+institution's+experience.">Surgical management of endolymphatic sac tumor: classification, outcomes and strategy. A single institution's experience.</a> Eur Arch Otorhinolaryngol. , (2022).</li><li>Bausch, B., et al. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Characterization+of+endolymphatic+sac+tumors+and+von+Hippel-Lindau+disease+in+the+International+Endolymphatic+Sac+Tumor+Registry.">Characterization of endolymphatic sac tumors and von Hippel-Lindau disease in the International Endolymphatic Sac Tumor Registry.</a> Head Neck. 38, E673-E679 (2016).</li><li>Tang, J. D., et al. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Systematic+Review+of+Endolymphatic+Sac+Tumor+Treatment+and+Outcomes.">Systematic Review of Endolymphatic Sac Tumor Treatment and Outcomes.</a> Otolaryngol Head Neck Surg. , (2022).</li><li>Balasubramaniam, S., Deshpande, R. B., Misra, B. K. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Gamma+knife+radiosurgery+in+jugular+foramen+endolymphatic+sac+adenocarcinoma.">Gamma knife radiosurgery in jugular foramen endolymphatic sac adenocarcinoma.</a> J Clin Neurosci. 16 (5), 710-711 (2009).</li><li>Poletti, A. M., Dubey, S. P., Colombo, G., Cugini, G., Mazzoni, A. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Treatment+of+endolymphatic+sac+tumour+(Papillary+adenocarcinoma)+of+the+temporal+bone.">Treatment of endolymphatic sac tumour (Papillary adenocarcinoma) of the temporal bone.</a> Rep Pract Oncol Radiother. 21 (4), 391-394 (2016).</li><li>Bambakidis, N. C., Megerian, C. A., Ratcheson, R. A. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Differential+grading+of+endolymphatic+sac+tumor+extension+by+virtue+of+von+Hippel-Lindau+disease+status.">Differential grading of endolymphatic sac tumor extension by virtue of von Hippel-Lindau disease status.</a> Otol Neurotol. 25 (5), 773-781 (2004).</li><li>Schipper, J., et al. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Endolymphatic+sac+tumours:+surgical+management.">Endolymphatic sac tumours: surgical management.</a> J Otolaryngol. 35 (6), 387-394 (2006).</li><li>Li, F., Zhang, Y. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Grading+system+and+surgical+approaches+for+endolymphatic+sac+tumors.">Grading system and surgical approaches for endolymphatic sac tumors.</a> Arch Otorhinolaryngol. 278 (5), 1345-1353 (2021).</li><li>Chittiboina, P., Lonser, R. R. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Von+Hippel-Lindau+disease.">Von Hippel-Lindau disease.</a> Handb Clin Neurol. 132, 139-156 (2015).</li><li>Sykopetrites, V., et al. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=The+endolymphatic+sac+tumor:+challenges+in+the+eradication+of+a+localized+disease.">The endolymphatic sac tumor: challenges in the eradication of a localized disease.</a> Eur Arch Otorhinolaryngol. 278 (7), 2297-2304 (2021).</li><li>Devaney, K. O., Ferlito, A., Rinaldo, A. <a target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?db=PubMed&cmd=Search&doptcmdl=Citation&defaultField=Title+Word&term=Endolymphatic+sac+tumor+(low-grade+papillary+adenocarcinoma)+of+the+temporal+bone.">Endolymphatic sac tumor (low-grade papillary adenocarcinoma) of the temporal bone.</a> Acta Otolaryngol. 123 (9), 1022-1026 (2003).</li></ol>

ELST are rare and locally aggressive neoplasms that arise on the posteromedial surface of the temporal bone. Due to their slow growth rate, and the initial paucity of the symptoms, the diagnosis is often delayed, resulting in an advanced tumor stage. Since the surgery morbidity as well as the oncologic outcomes are strongly correlated with the tumor dimensions and extension6,8,13,15, an early d...

Endolymphatic sac tumors (ELST) are neoplasms originating from the endolymphatic sac, a neuroectoderm-derived organ located in the posteromedial surface of the temporal bone. Histologically, ELST are characterizable as low-grade papillary adenocarcinoma1. Usually, ELST are slow-growing, with local aggressiveness and a low risk of distant metastases2,3,4. ELST can be sporadic, but they are also frequently associated with von Hippel Lindau disease (VHL)4.
Given the slow growing rate, the paucity of the clinical signs, and the late onset of the symptoms, they are difficult to diagnose in the early stages, especially when not associated with VHL5,6. The typical symptomatology at diagnosis of ELST includes hearing loss (79.8%), tinnitus (52.6%), and disequilibrium (45.5%), due to endolymphatic hydrops or intralabyrinthine hemorrhages4,7,8. Further and less frequent symptoms are facial palsy (25.6%), headache (13.8%), otalgia (5.1%), and trigeminal symptoms (3.2%)8.
The current treatment of ELST is primarily surgical resection. The tumor&#39;s residuals can be treated with stereotactic radiosurgery, which can be considered if the patient cannot tolerate the operation9,10. The surgical approach and the prognosis are mostly related to the tumor extension6. Even if there is no official staging system for ELST11,12, a recent grading system proposed by Li and colleagues13, based on imaging data and intraoperative findings, defined four main grades, according to the tumor extension. This and other previously proposed staging systems are reported in Table 1.
<table border="1" fo:keep-together.within-page="1" fo:keep-with-next.within-page="always">
	<tbody>
		<tr>
			<td>Article</td>
			<td colspan="4">ELST Staging / Grading</td>
		</tr>
		<tr>
			<td>Bambakidis et al.11</td>
			<td colspan="4">Stage I: confiled to the temporal bone and middle ear</td>
		</tr>
		<tr>
			<td></td>
			<td colspan="4">Stage II: extended to the posterior fossa</td>
		</tr>
		<tr>
			<td></td>
			<td colspan="4">Stage III: extended to the middle cranial fossa</td>
		</tr>
		<tr>
			<td></td>
			<td colspan="4">Stage IV: extended to the clivus and/or sphenoid wing</td>
		</tr>
		<tr>
			<td>Schipper et al.12</td>
			<td colspan="4">Type A: limited to the dura of the posterior cranial fossa</td>
		</tr>
		<tr>
			<td></td>
			<td colspan="4">Type B: infiltration of lateral semicircular canal and/or cochlea</td>
		</tr>
		<tr>
			<td></td>
			<td colspan="4">Type C: infiltration of sigmoid sinus and/or jugular bulb</td>
		</tr>
		<tr>
			<td>Li et al.13</td>
			<td colspan="4">Grade I: confined to the posterior petrous bone between posterior semicircular canal and posterior fossa, may involve facial nerve, no inner ear and jugular foramen invasion</td>
		</tr>
		<tr>
			<td></td>
			<td colspan="4">Grade II: extended anteriorly with invasion of semicircular canals, cochlea, IAC, middle ear, without cerebellopotine angle and jugular foramen involvement</td>
		</tr>
		<tr>
			<td></td>
			<td colspan="4">Grade IIIa: involvement of jugular foramen or internal carotid artery or clivus</td>
		</tr>
		<tr>
			<td></td>
			<td colspan="4">Grade IIIb: extensive intracranial invasion or involvement of cavernous sinus or jugular foramen</td>
		</tr>
		<tr>
			<td>Wu et al.6</td>
			<td colspan="4">Type I: confined to the endolymphatic sac region</td>
		</tr>
		<tr>
			<td></td>
			<td colspan="4">Type II: extended to at least another one anatomic structure</td>
		</tr>
	</tbody>
</table>
Table 1: Previously published tumor staging systems. An overview of previously published grading/staging systems.
To date, only ~300 cases were reported, mostly as single clinical cases or small case series5,6. Recurrence rates and survival estimates have not been clearly defined yet8. In the present paper, we report the clinical and surgical management of a single case of sporadic ELST, as well as the follow-up results, providing clinicians our experience of the handling of such a challenging otologic skull base surgery and rare disease. Here, we describe ELST in a 55-year-old woman who accessed our otologic tertiary level referral center from a peripheric hospital. The patient had been hospitalized 3 days before for a sudden worsening of the left hearing loss, which began 3 months earlier, accompanied by an episode of acute vertigo with nausea and vomiting. The anamnesis was positive for hypertension and hypercholesterolemia, both under treatment, and smoking (3 pack-years). No personal or familiar history of VHL disease or compatible symptoms were reported by the patient.

<table border="1" fo:keep-together.within-page="1" fo:keep-with-next.within-page="always">
	<tbody>
		<tr>
			<td>AC40</td>
			<td>Interacoustic</td>
			<td></td>
			<td>Clinical audiometer</td>
		</tr>
		<tr>
			<td>AT235</td>
			<td>Interacoustic</td>
			<td></td>
			<td>Clinical tympanometry</td>
		</tr>
		<tr>
			<td>Innova 3131</td>
			<td>GE Healthcare</td>
			<td></td>
			<td>Biplane angiograph</td>
		</tr>
		<tr>
			<td>LightSpeed VCT</td>
			<td>GE Healthcare</td>
			<td></td>
			<td>64 Slice CT system</td>
		</tr>
		<tr>
			<td>S2 &#960;Drive</td>
			<td>Striker</td>
			<td></td>
			<td>Drilling system for Otosurgery</td>
		</tr>
		<tr>
			<td>Signa HDX</td>
			<td>GE Healthcare</td>
			<td></td>
			<td>1.5 T MRI system</td>
		</tr>
		<tr>
			<td>SYNAPSYS VNG</td>
			<td>Inventis</td>
			<td></td>
			<td>Video nystagmograph</td>
		</tr>
	</tbody>
</table>

surgical treatment of an endolymphatic sac tumor

Endolymphatic sac tumors (ELST) are low-grade papillary adenocarcinoma originating from the endolymphatic sac. Usually slow-growing, with local aggressiveness and a low risk of distant metastases, ELST can be sporadic but also frequently associated with von Hippel Lindau disease. The current treatment of ELST is primarily surgical resection. A 55-year-old woman accessed our otologic tertiary level referral center for a sudden worsening of hearing loss in her left ear and vertigo. A magnetic resonance (MRI) and computer tomography scan study subsequently showed a mass in the petrous bone; hence, the presence of an ELST was hypothesized. After embolization of the mass, the patient underwent surgical removal of the lesion. The resection of the mass was done through a translabirinthine approach, with an uneventful procedure. No residual disease remained after surgery. After 24 months of radiologic follow up with MRI, there are no signs of recurrence disease. This paper reports the management of this sporadic ELST, as well as the follow up results, providing clinicians this protocol for the handling of such a challenging otologic skull base surgery and rare disease.

From the audiologic assessment, it was possible to identify a moderately severe left sensorineural hearing loss (Figure 1), with a reduction of word discrimination. The tympanometry was bilaterally normal, and the stapedial reflexes were evocable with ipsilateral and contralateral stimulus. At the videonystagmography , it was possible to assess a grade II horizontal spontaneous nystagmus beating on the right side, which grew in intensity after head shaking. From the clinical and audiological...

Watch this Scientific Journal Video about Surgical Treatment of an Endolymphatic Sac Tumor at JoVE.com

Surgical Treatment of an Endolymphatic Sac Tumor

Here, we discuss clinical and radiological features of endolymphatic sac tumors and report the methodology and results of a surgical removal in a case in care at our otologic referral center.

Endolymphatic sac tumors (ELST) are low-grade papillary adenocarcinoma originating from the endolymphatic sac. Usually slow-growing, with local ...

هذا البروتوكول مهم لأنه يوضح كيفية علاج ورم الكيس اللمفاوي الباطن بشكل فعال. يمكن أن يؤدي التشخيص الفوري بناء على الصورة السريرية وتقييم التصوير إلى جراحة يمكن التحكم فيها لإزالة الورم. للبدء ، قم بإجراء فحص التصوير بالرنين المغناطيسي باستخدام استعادة الانقلاب المحوري الموهن بالسوائل والتسلسلات المرجحة بصدى الدوران المحوري السريع T2.استهدف صور السبق الحرة للحالة المستقرة باستخدام تصوير سريع يستخدم تسلسل اكتساب الحالة المستقرة ، أو FIESTA ، على الأذن الداخلية. إذا ظهرت كتلة في سياق العظم الصخري ، فقم بإجراء تصوير مقطعي بالكمبيوتر عالي الدقة مع مسح ضوئي مكون من 64 شريحة على الأقل لتأكيد التشخيص. بمجرد الاشتباه في ELST ، حدد درجة الورم وخطط للنهج الجراحي للاستئصال الشامل.بعد إجراء الانصمام لفائف واستئصال الكتلة ، قم بعمل شق خلف الأذن على شكل حرف C ، وارفع رفرف العضلة الصدغية. قم بإجراء عملية استئصال الخشاء بشكل كبير باستخدام نظام الحفر Stryker S2 PiDrive ، وقم بقص الأطراف الدائرية الدقيقة المخددة والأطراف المستديرة الماسية الخشنة والدقيقة من الهياكل المحفورة. فضح الجيب السيني الخلفي والجافية من الحفرة القحفية الوسطى بشكل متفوق.أكمل بضع المفصل الوافر مع التعرض الكامل للعملية الصغيرة للسنوس. ثم قم بإعداد بضع الطبلة الخلفي عن طريق حفر جميع خلايا الخشاء خلف الوجه حتى طرف الخشاء حتى يتم تحديد المصباح الوداجي. أكمل استئصال المتاهة الخلفية عن طريق تشريح القنوات الهلالية.تشريح المتاهة العظمية الخلفية حتى تحديد القناة السمعية الداخلية. بعد ذلك ، قم بقطع الجافية وإزالتها على كتلة مع الورم لتحديد الأورام وإزالة الورم تماما. إجراء الفحص النسيجي المرتجل أثناء العملية.ختم بضع الطبلة الخلفي وبضع الشرج بالشمع الجراحي ، وملء تجويف الخشاء الواسع بدهون البطن الذاتية وغراء الفيبرين. من التقييم السمعي ، كان من الممكن تحديد فقدان السمع العصبي الحسي الأيسر الشديد بشكل معتدل مع تقليل التمييز بين الكلمات. احتلت خلايا الخشاء اليسرى مادة مخاطية عالية الكثافة ، مما تسبب في ظهور منتفخ لعظم القشرة والجانب الخلفي من الجزء الصخري مع طمس الصهريج البابوي المخيخي المماثل والحد الأدنى من البصمة على الدوران المخيخي الأيسر ، بما يتفق مع وجود ELST.أكد التصوير المقطعي المحوسب عالي الدقة للعظم الصخري مع مسح 64 شريحة وجود كتلة متساوية الكثافة على الجدار الخلفي للعظم الصخري مع تآكل في نمط تسلل تؤكل العثة. وفقا لنظام درجات Lee ، تم الاشتباه في وجود درجة 2 ELST. أظهر تصوير الأوعية الدموية بالطرح الرقمي التشخيصي ورما وعائيا مع احمرار خدود الورم بسبب إمداد الدم من فروع البلعوم الصاعدة والأذن الخلفية والقذالية للشريان السباتي الخارجي الأيسر.أظهر تصوير الأوعية الدموية بعد الانصمام طمسا كاملا لأحمر خدود الورم دون أي مضاعفات أثناء العملية وبعدها. بعد التشخيص الفوري ، يمكن إجراء استئصال كامل لورم الكيس اللمفاوي الباطن ، حتى لو كان متطلبا. أثناء الجراحة ، تتمثل النقطة الأكثر أهمية في إجراء عملية استئصال الخشاء بشكل كبير للغاية ، مما يوفر تعرضا كاملا للمتاهة الخلفية ووصولا جيدا إلى منطقة الكيس اللمفاوي الباطن.

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