Braz J Otorhinolaryngol. 2018;84(4):519---522

www.bjorl.org

Brazilian Journal of

OTORHINOLARYNGOLOGY

CASE REPORT

A rare laryngeal tumor in a patient with thyroidpapillary cancer: granular cell tumor�

Um raro tumor de laringe em paciente com carcinoma papilíferoda tireoide: tumor de células granulares

Sheng-Yao Chenga, Li-Hsiang Chenga,∗, Yi-Shu Liaob, Wen-Sen Laic

a National Defense Medical Center, Tri-Service General Hospital, Department of Otolaryngology---Head and Neck Surgery, Taipei,Taiwanb National Defense Medical Center, Tri-Service General Hospital, Department of Pathology, Taipei, Taiwanc Taichung Armed Forces General Hospital, Department of Otolaryngology-Head and Neck Surgery, Taichung, Taiwan

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Received 11 September 2015; accepted 8 December 2015Available online 29 March 2016

Introduction

Granular cell tumor (GCT), Abrikossoff’s tumor, is a raretumor, especially that occurring in the larynx. It may sharesimilar appearance and symptoms with laryngeal granu-loma, polyp, or nodule although they have different etiologyand features (Table 1). The diagnosis must be made byhistological examination. In the past decades, the originof GCT remained controversial but current mainstreamopinion regarded it as of neuroectodermal origin accord-ing to immunohistochemical staining.1 Here we presenteda patient with a history of papillary thyroid carcinomaunder regular follow-up who was diagnosed recently witha GCT of the larynx. The related literatures were alsoreviewed.

� Please cite this article as: Cheng S-Y, Cheng L-H, Liao Y-S, Lai W-

S. A rare laryngeal tumor in a patient with thyroid papillary cancer:granular cell tumor. Braz J Otorhinolaryngol. 2018;84:519---22.

∗ Corresponding author.E-mail: m1041000@mail.ndmctsgh.edu.tw (L.-H. Cheng).Peer Review under the responsibility of Associacão Brasileira de

Otorrinolaringologia e Cirurgia Cérvico-Facial.

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https://doi.org/10.1016/j.bjorl.2015.12.0071808-8694/© 2016 Associacao Brasileira de Otorrinolaringologia e Cirurgiaaccess article under the CC BY license (http://creativecommons.org/lic

ase report

35-year-old female presented with a 10 month historyf progressive painless hoarseness and easy voice fatigue.hree years ago, she was diagnosed with papillary thyroidancer and received total thyroidectomy and Iodine-131herapy. Since then, she has been under daily levothyrox-ne supplement (0.1 mg per day) and regular follow-up.he has been smoking 20 cigarettes per day over 10 yearsut without voice abuse history. There was no palpableeck mass on physical examination. Due to the persistentymptom, she came to our clinic where indirect laryn-oscope showed a white tumor lesion at right posteriorhird vocal fold with mucosal cover and unclear borderhich led to failure of glottis closure during phonation

Fig. 1). Because of her history and the appearance ofumor, primary laryngeal malignancy or recurrent papillaryhyroid carcinoma metastasis was suspected. The tumoras completely excised through microlaryngeal surgery withold knife under general anesthesia. The lesion measuredbout 0.5 cm × 0.2 cm in size and was totally embedded forections. Histopathologic examination revealed one nodu-

ar lesion with pseudoepitheliomatous hyperplasia. Underematoxylin-eosin (H&E) staining, polygonal cells withbundant eosinophilic granular cytoplasm and small uniformuclei arranged in nests was impressed (Fig. 2). There was no

Cervico-Facial. Published by Elsevier Editora Ltda. This is an openenses/by/4.0/).

520 Cheng S-Y et al.

Table 1 Vocal fold lesions.

Vocal fold lesions Features Common causes Treatment

NoduleUsually bilateral Chronic voice abuse Speech therapySmaller Excision

PolypOften unilateral Acute voice abuse Behavior modification (quit

smoking, speech therapy)Obvious surface Chronic irrigation (For example,smoking, gastroesophageal reflux)Blood vessel Excision

GranulomaOften bilateral Repeated trauma (For example,

intratracheal intubation)Excision

Larger

Granular cell tumorSingle

Unknown ExcisionUnilateralMost common at

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oiaalfGoilump sensation in the throat, dry cough, hemoptysis, andodynophagia can also be present.5 Grossly, laryngeal GCTsare characterized as being firm, round, mucosa-covered

posterior glottis

vidence of malignancy due to the absence of pleomorphismith small nuclei.

Immunohistochemical staining showed positive for S-00 protein (Fig. 3A), vimentin and neuron-specific enolaseFig. 3B). The tumor was uniformly negative for thyroglob-lin, TTF1, and HBME-1. According to the above pathologicesults, the final diagnosis was laryngeal granular cell tumor.fter 6 months of follow-up, she was satisfied with her voiceutcome and no local tumor recurrence was noted.

iscussion

ost cases of papillary thyroid cancer had laryngealnvolvement through direct invasion and were regarded asdvanced stage. Although papillary thyroid carcinoma tendso spread via the lymphatic system, manifesting as laryngeal

etastatic nodules, hematogenous spread had also been

eported before.2 The patient had a thyroid cancer historynd was under regular follow-up after total thyroidectomy.

igure 1 Preoperative indirect laryngoscope finding: a whiteumor lesion was at right vocal fold with mucosal cover andnclear border which led to failure of glottis closure duringhonation.

Fan

hus, metastatic malignancy should be considered for dif-erential diagnosis of laryngeal tumors.

GCTs are uncommon neoplasms and can be found in anyrgan of the whole body. About half of the cases are foundn the head and neck, with the tongue being the site mostffected in this region. The larynx being affected by GCT iss rare as 3---10% of all cases.3 Different from other commonaryngeal lesions such as polyp or nodule, GCT showed slightemale predominance, and no causal relationship betweenCT and voice abuse had been established.4 The symptomsf GCTs vary with tumor size and localization. Weaken-ng hoarseness is the most common symptom. However,

igure 2 The tumor comprised round or polygonal cells withbundant eosinophilic granular cytoplasm and small uniformuclei arranged in nest and sheet patterns (H&E, 400×).

Laryngeal granular cell tumor in a patient with thyroid cancer 521

Figure 3 (A) Positive S-100 immunostain. (B) Positive neuron-specific enolase immunohistochemical staining denoted its neuroec-

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todermal origin.

masses located most frequently in the posterior two-thirdsof the vocal folds although other sites such as arytenoid,posterior cricoid region, supra- or subglottic areas have alsobeen described.6

They often resemble vocal fold granulomas, polyps, andeven malignant lesions. Consequently, the definite diagnosiswas made by histopathological examination which demon-strated polygonal cells with thickening of the cell membraneand abundant eosinophilic intracytoplasmic granules. Theorigin of GCT remained controversial. It was first describedby Abrikossof, in 1926, who named it as myoblastoma thatwas considered from skeletal muscle cells base on their cyto-logic picture.7 However, recent immunochemistry studieshave provided better evidences of the origin of this tumorwhich show positive for S-100 protein, neuron-specific eno-lase, and CD68 but negative for muscular markers such asmyoglobin, keratin, and desmin. These staining characteris-tics suggested that they originate from the neuroectodermaltissue or Schwann cells rather than muscle cells.4 In recentimmunohistochemical study, GCTs have a component ofendomesenchymal origin suggested by Simona Gurzu et al.8

Most GCTs are benign with slow growth, only 1---2% ofall cases occur as malignant tumors which showed morenuclear pleomorphism, high nuclear to cytoplasmic ratio,and increased mitotic activity on histologic examination. Inaddition, malignant tumors tend to exceed 4 cm in size withinvasion of adjacent structures or metastases.1 The coexis-tence of GCT and other malignant neoplasms in the sameorgan has been reported in multiple organ including tongueand larynx.9 Sometimes, biopsy or subtotal resection was toosuperficial to distinguish pseudoepitheliomatous hyperplasia(PEH) which appears on the mucosal layer in half of all cases

of laryngeal GCTs from squamous cell carcinoma.10 In viewof these problems, the treatment aimed at complete exci-sion using laser or cold instruments and minimal functionaldamage. After complete resection, most cases can be cured,

ut recurrence still presents in 2---21% of all cases, usually athe primary site.5 Therefore, the patients need to be underollow-up with laryngoscopy to confirm complete recovery.

onclusion

n conclusion, laryngeal GCT is a rare and benign tumor. How-ver, the possibility of coexistence with malignant neoplasmnd voice disturbance were of concern. Complete resectionith laser or cold knife for pathologic examination shoulde performed. To our best knowledge, this is the first casef glottic GCT in patients receiving levothyroxine supple-ent after total thyroidectomy. In this patient, althoughe cannot provide evidence that tumor growth is medi-ted by levothyroxine stimulation; more cases should beollected for analysis to understand the etiology of this rareumor.

onflicts of interest

he authors declare no conflicts of interest.

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