Abstracts
Schwannoma is a rare cause of benign tumors of the larynx. The first-choice treatment is surgical resection. The objective of this paper is to report on a rare case of a young female patient who suffered severe intraoperative hemorrhaging during surgical resection of a laryngeal Schwannoma and needed emergency embolization.
therapeutic embolization; schwannoma; larynx
O schwanoma representa etiologia rara de tumor benigno de laringe, tendo como principal tratamento a ressecção cirúrgica. O objetivo deste trabalho é relatar um caso raro de paciente jovem submetido à ressecção cirúrgica de schwanoma laríngeo, evoluindo, no transperatório, para complicação hemorrágica grave e necessitando de embolização de urgência.
embolização terapêutica; schwanoma; laringe
INTRODUCTION
Schwannomas are benign tumors that typically grow slowly, are encapsulated and have
extensive vascularization. They get their name from the fact that they originate in the
Schwann cells of motor, sensory or cranial nerves, but do not contain nervous elements.
The most common site is the side of the neck, originating in the cervical nerve roots
and the cervical sympathetic trunk11. Fini-Storchi I, Frosini P. Laryngeal neurinomas (a case report and
review). ORL. 1997;59:182-5. PMid:9186976.
http://dx.doi.org/10.1159/000276935
http://dx.doi.org/10.1159/000276935...
. They are rarely found in the larynx and there are only sporadic reports of this
presentation in the literature. Laryngeal Schwannomas account for less than 0.5% benign
tumors of the larynx and the first choice treatment is surgery, despite the fact that
degeneration to malignancy is extremely rare22. Palva T, Jokinem K, Karja J. Neurilemmoma (schwannoma) of the larynx.
J Laryngol Otol. 1975;89:203-7. PMid:1123570.
http://dx.doi.org/10.1017/S0022215100080270
http://dx.doi.org/10.1017/S0022215100080...
3. Phang WK, Raman R, Jayalaksmi E. Neurogenous tumour of the larynx (a
case report). J Laryngol Otol. 1987;101:1209-10. PMid:3694036.
http://dx.doi.org/10.1017/S0022215100103512
http://dx.doi.org/10.1017/S0022215100103...
4. Takumida M, Taira T, Suzuki M, Yajin K, Harada Y. Neurilemmoma of the
larynx (a case report). J Laryngol Otol. 1986;100:847-50. PMid:3734607.
http://dx.doi.org/10.1017/S0022215100100180
http://dx.doi.org/10.1017/S0022215100100...
5. Barnes L, Ferlito A. Soft tissue neoplasms. In: Ferlito A. Neoplasms
of the larynx. Edinburgh: Churchill Livingstone; 1993. p. 277-9.
6. Supance JS, Quenelle DJ, Crissman J. Endolaryngeal neurofibromas.
Otolaryngol Head Neck Surg. 1980;88:74-8. PMid:7393604.
-
77. Delozier HL. Intrinsic malignant schwannoma of the larynx: a case
report. Ann Otol Rhinol Laryngol. 1982;91:336-8. PMid:7092059. .
CASE DESCRIPTION
The patient was a 29-year-old female (S.R.X.) with a history of progressive solids dysphagia and a sensation of pharyngeal bolus. After initial evaluation by the otorhinolaryngology team, videolaryngoscopy and computed tomography of the neck identified an encapsulated solid tumor of the larynx with an intimate relationship with the right external and internal carotid arteries (Figure 1).
Computed tomography with venous contrast and axial cross-section, showing laryngeal tumor with an intimate relationship with the right-side carotid arteries.
The patient was operated on by the otorhinolaryngology team under general anesthetic. A transoral approach was used to achieve total resection of the tumor. However, after removal of the tumor, the patient exhibited massive and persistent bleeding via the oral cavity, presenting hemodynamic instability, even after careful review of hemostasis and tests for intraoperative coagulation disorder. The vascular surgery team were called and requested to make an emergency assessment.
After hemodynamic stabilization, selective arteriography of the right carotid arteries was conducted, showing injury to branches of the right external carotid artery, with considerable contrast leakage (Figure 2).
Selective arteriography with digital subtraction, showing contrast leaking via branches of the right external carotid artery.
Superselective catheterization was performed using an Echelon 14(r) microcatheter (eV3 Neurovascular, Inc.) in a coaxial system, and embolization was achieved using controlled release 2 mm × 2 mm and 2.5 mm × 2 mm Micrus(r) microcoils (Micrus Endovascular Corporation. CA/USA), with immediate control of bleeding (Figure 3).
The patient recovered in the intensive care unit for 24 hours without further intercurrent conditions and without requiring blood transfusion, with hemodynamic stability maintained and no further bleeding. She was discharged from hospital 48 hours after embolization. The material removed was sent for pathoanatomical assessment and histological analysis confirmed a laryngeal Schwannoma. The patient attended outpatients follow-up for 30 days with the vascular surgery team and showed no sign of relapse of bleeding, laryngeal symptoms exhibited improvement and there was no evidence of neurological damage.
DISCUSSION
Schwannomas of the larynx present as tumors with extensive vascularization and slow
growth and symptomology is caused by their growth and mass effects. The treatment of
choice is surgical resection and access can be intraoral, by direct laryngoscopy, or via
an external approach (lateral pharyngotomy or laryngofissure)88. Schaeffer BT, Som PM, Biller HF, Som ML, Arnold LM. Schwannoma of the
larynx: review and computed tomographic scan analysis. Head Neck Surg. 1986;8:469-72.
PMid:3721890. http://dx.doi.org/10.1002/hed.2890080613
http://dx.doi.org/10.1002/hed.2890080613...
,
99. Whittam DE, Morris TMO. Neurilemmoma of the larynx. J Laryngol Otol.
1970;84:747-50. http://dx.doi.org/10.1017/S0022215100072480
http://dx.doi.org/10.1017/S0022215100072...
, depending on the tumor's anatomic characteristics. The transoral approach is
associated with a lower incidence of recurrent paralysis of the laryngeal nerve during
the postoperative period, but complete resection of the tumor can prove difficult using
this approach44. Takumida M, Taira T, Suzuki M, Yajin K, Harada Y. Neurilemmoma of the
larynx (a case report). J Laryngol Otol. 1986;100:847-50. PMid:3734607.
http://dx.doi.org/10.1017/S0022215100100180
http://dx.doi.org/10.1017/S0022215100100...
.
Arteriographic assessment and preoperative embolization are described for management of head and neck Schwannomas1010. Abramowitz J, Dion JE, Jensen ME, et al. Angiographic diagnosis and management of head and neck schwannomas. AJNR. 1991;12:977-984. PMid:1950934., but no cases of embolization of a laryngeal Schwannoma, whether preoperative or postoperative, were found in the literature, probably because of the rarity of the presentation.
This paper described a rare case of a patient with a Schwannoma of the larynx who required immediate emergency embolization to control severe hemorrhage.
We are grateful to Dr. Gustavo Jorge of the otorhinolaryngology team at the Hospital São Francisco de Assis for the referral and for following the case in conjunction.
REFERENCES
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1Fini-Storchi I, Frosini P. Laryngeal neurinomas (a case report and review). ORL. 1997;59:182-5. PMid:9186976. http://dx.doi.org/10.1159/000276935
» http://dx.doi.org/10.1159/000276935 -
2Palva T, Jokinem K, Karja J. Neurilemmoma (schwannoma) of the larynx. J Laryngol Otol. 1975;89:203-7. PMid:1123570. http://dx.doi.org/10.1017/S0022215100080270
» http://dx.doi.org/10.1017/S0022215100080270 -
3Phang WK, Raman R, Jayalaksmi E. Neurogenous tumour of the larynx (a case report). J Laryngol Otol. 1987;101:1209-10. PMid:3694036. http://dx.doi.org/10.1017/S0022215100103512
» http://dx.doi.org/10.1017/S0022215100103512 -
4Takumida M, Taira T, Suzuki M, Yajin K, Harada Y. Neurilemmoma of the larynx (a case report). J Laryngol Otol. 1986;100:847-50. PMid:3734607. http://dx.doi.org/10.1017/S0022215100100180
» http://dx.doi.org/10.1017/S0022215100100180 -
5Barnes L, Ferlito A. Soft tissue neoplasms. In: Ferlito A. Neoplasms of the larynx. Edinburgh: Churchill Livingstone; 1993. p. 277-9.
-
6Supance JS, Quenelle DJ, Crissman J. Endolaryngeal neurofibromas. Otolaryngol Head Neck Surg. 1980;88:74-8. PMid:7393604.
-
7Delozier HL. Intrinsic malignant schwannoma of the larynx: a case report. Ann Otol Rhinol Laryngol. 1982;91:336-8. PMid:7092059.
-
8Schaeffer BT, Som PM, Biller HF, Som ML, Arnold LM. Schwannoma of the larynx: review and computed tomographic scan analysis. Head Neck Surg. 1986;8:469-72. PMid:3721890. http://dx.doi.org/10.1002/hed.2890080613
» http://dx.doi.org/10.1002/hed.2890080613 -
9Whittam DE, Morris TMO. Neurilemmoma of the larynx. J Laryngol Otol. 1970;84:747-50. http://dx.doi.org/10.1017/S0022215100072480
» http://dx.doi.org/10.1017/S0022215100072480 -
10Abramowitz J, Dion JE, Jensen ME, et al. Angiographic diagnosis and management of head and neck schwannomas. AJNR. 1991;12:977-984. PMid:1950934.
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*All authors should have read and approved of the final version of the article submitted to J Vasc Bras.
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The study was carried out at Angiogyn – Service of Vascular and Endovascular Surgery, Hospital São Francisco de Assis, Goiânia-GO, Brazil.
Publication Dates
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Publication in this collection
11 Nov 2013 -
Date of issue
Oct-Dec 2013
History
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Received
11 May 2013 -
Accepted
19 May 2013