Primary Sclerosing Mucoepidermoid Carcinoma with Eosinophilia of the Thyroid: Description of a Case and Review of the Literature

Song I Yang; Kwang Kuk Park; Ji Young Yoo

doi:10.11106/ijt.2017.10.2.107

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Yang, Park, and Yoo: Primary Sclerosing Mucoepidermoid Carcinoma with Eosinophilia of the Thyroid: Description of a Case and Review of the Literature

Case Report

Int J Thyroidol 2017;10(2):107-113.

Published online: 5 November 2017

DOI: https://doi.org/10.11106/ijt.2017.10.2.107

Primary Sclerosing Mucoepidermoid Carcinoma with Eosinophilia of the Thyroid: Description of a Case and Review of the Literature

Song I Yang¹, Kwang Kuk Park², Ji Young Yoo¹

¹Department of Surgery, Kosin University College of Medicine, Busan, Korea

²Department of Surgery, Hub-Hu Hospital, Busan, Korea

Correspondence: Song I Yang, MD, PhD, Department of Surgery, Kosin University College of Medicine, 262 Gamcheon- ro, Seo-gu, Busan 49267, Korea Tel: 82-51-990-6462, Fax: 82-51-246-6093, E-mail: tonybin@daum.net

Received 17 April 201727 June 2017 Accepted 3 July 2017

This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creative-commons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

초록

Primary sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE) of the thyroid gland is a very rare disease. We present the clinical and histopathologic findings of a 37-year-old woman recently diagnosed with SMECE of the thyroid gland. The patient, clinically euthyroid, who presented with a neck swelling since last 2 years along. Fine needle aspiration cytology suggested thyroid papillary carcinoma. Total thyroidectomy, central neck dissection and right selective neck dissection were performed. Although SMECE is considered to be a relatively slow growing and non-aggressive tumor, occasional metastasis does occur. We report an additional case of SMECE, with metastasis to regional lymph nodes. Physicians should be aware of extended operation, including total thyroidectomy and/or neck node dissection for metastatic lesion of the neck node. More standardized treatment is likely to evolve in the future.

Keywords: Mucoepidermoid, Carcinoma, Eosinophilia, Thyroid gland

REFERENCES

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Fig. 1.

Neck computed tomography with contrast enhance-ment demonstrating tumor invasion of the right thyroid lobe. A right supraclavicular lymph node metastasis is present (arrow).

Fig. 2.

18F-fludeoxyglucose (FDG) positron emission tomography (PET) scan shows a metastatic lymphadenopathy at right supraclavicular area (arrow).

Fig. 3.

The tumor nodule has ill-defined border and adjacent thyroid reveals lymphocytic thyroiditis (H&E staining, ×40).

Fig. 4.

The tumor is characterized by anastomosing compact cords and nests of epidermoid cells, accompanied by extensive sclerosis and by infiltration of eosinophils, lymphocytes and plasma cells (A: H&E staining, ×100; B: inlet, ×400).

Fig. 5.

(A) Tumor contains a well-formed squamous pearl. (B) Mucocytes compressed by globules of mucin is interspersed in epidermoid cells (H&E staining, ×400).

Table 1.

Literature review of sclerosing mucoepidermoid carcinoma with eosinophilia of the thyroid gland

No.	Reference	Year	Age	Sex	Operation	Other treatment	Extrathyroidal extension or metastasis
1	Chan et al.¹⁾	1991	35	F	Total thyroidectomy	External radiation	Ext. into perithyroidal tissue, nerves and esophagus
2	Chan et al.¹⁾	1991	64	F	Left lobectomy		None
3	Chan et al.¹⁾	1991	71	F	Total thyroidectomy	External radiation	Ext. into perithyroidal soft tissue
4	Chan et al.¹⁾	1991	61	F	Left lobectomy		None
5	Chan et al.¹⁾	1991	43	F	Total thyroidectomy	External radiation	Adherent to trachea; tumor recurred in trachea
6	Chan et al.¹⁾	1991	46	F	Total thyroidectomy		Nodal metastasis and ext. into perithyroidal soft tissue
7	Chan et al.¹⁾	1991	69	F	Total thyroidectomy		Ext. into perithyroidal tissue; adherence to trachea and esophagus
8	Chan et al.¹⁾	1991	69	F	Total thyroidectomy		None
9	Wenig et al.¹⁴⁾	1995	46	M	Isthmusectomy		None
10	Wenig et al.¹⁴⁾	1995	46	F	Total thyroidectomy		None
11	Wenig et al.¹⁴⁾	1995	44	F	Left lobectomy		None
12	Sim et al.⁴⁾	1997	70	F	Total thyroidectomy	Chemotherapy recommended	Trachea, bilateral lung mets. developed right humeral head mets.
13	Sim et al.⁴⁾	1997	69	F	Thyroidectomy with multiple surgeries		Tracheal and esophageal involvement
14	Geisinger et al.³⁾	1998	39	F	Right thyroidectomy followed completion thyroidectomy		Perithyroidal tissue ext. then developed lung nodules and pleural effusion
15	Geisinger et al.³⁾	1998	61	M	Total thyroidectomy	RAI then combined chemotherapy	Mediastinal ext. then 2 year later had T-spine mets. and liver mets.
16	Chung et al.¹⁵⁾	1999	57	F	Total thyroidectomy then laryngectomy and esophagectomy	RT	Lymph nodes and esophageal involvement
17	Cavazza et al.²¹⁾	1999	32	F	Total thyroidectomy		None
18	Solomon et al.¹⁶⁾	2000	39	F	Total thyroidectomy	RT	Tracheal involvement
19	Baloch et al.¹⁷⁾	2000	38	F	Total thyroidectomy		Ext. to skeletal muscles and lymph nodes
20	Baloch et al.¹⁷⁾	2000	47	F	Right thyroidectomy		None
21	Baloch et al.¹⁷⁾	2000	73	F	Right thyroidectomy		None
22	Baloch et al.¹⁷⁾	2000	64	F	Right thyroidectomy		None
23	Sharma et al.²²⁾	2003	55	F	N/D	N/D	N/D
24	Shehadeh et al.¹⁹⁾	2004	38	F	Total thyroidectomy with modified radical neck dissection then right neck and supraclavicular lymph node dissection and posterior neck surgery	RAI and concurrent chemo/RT	Perithyroidal ext., right axillary adenopathy, lung nodule
25	Hunt et al.²³⁾	2004	37	F	N/D	N/D	N/D
26	Hunt et al.²³⁾	2004	57	F	N/D	N/D	N/D
27	Hunt et al.²³⁾	2004	64	M	N/D	N/D	N/D
28	Kanat et al.²⁴⁾	2004	74	F	N/D	N/D	N/D
29	Das et al.²⁵⁾	2008	65	F	Subtotal thyroidectomy		Lymph node
29	Kim et al.²⁰⁾	2014	72	F	Total thyroidectomy with central compartment and bilateral compartment lateral neck dissection	RT	Trachea, esophagus, right recurrent laryngeal nerve invasion then 3 months later had liver, lung, bone mets.
30	Our Case	2014	37	F	Total thyroidectomy with central compartment node dissection and right selective neck dissection	Concurrent chemo/RT	Right lateral lymph nodes and right supraclavicular lymph node

Ext.: extension, F: female, M: male, mets.: metastasis, N/D: no data, RAI: radioactive iodine, RT: radiotherapy

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