J Med Cases
Journal of Medical Cases, ISSN 1923-4155 print, 1923-4163 online, Open Access
Article copyright, the authors; Journal compilation copyright, J Med Cases and Elmer Press Inc
Journal website https://www.journalmc.org

Case Report

Volume 12, Number 2, February 2021, pages 71-73


Thyroid Warthin-Like Cancer Concurrent With Multiple Sclerosis: A Case Report

Lucia Stella Curtoa, d, Rita Gervasib, Valeria Zuccalac, Nadia Innarob

aUnit of Clinical Surgery, Department of Medical and Surgical Sciences, Magna Graecia University Medical School, Catanzaro, Italy
bUnit of Endocrine Surgery, A.O.U. Mater Domini, Catanzaro, Italy
cPathology Unit, “Pugliese Ciaccio” Hospital, Catanzaro, Italy
dCorresponding Author: Lucia Stella Curto, Unit of Clinical Surgery, Department of Medical and Surgical Sciences, Magna Graecia University Medical School, Catanzaro, Italy

Manuscript submitted November 24, 2020, accepted December 8, 2020, published online December 30, 2020
Short title: A Case of WL-PTC With MS
doi: https://doi.org/10.14740/jmc3625

Abstract▴Top 

Warthin like-papillary thyroid cancer (WL-PTC) is a rare malignancy; it is difficult to distinguish preoperatively a WLPTC from a classic PTC. Often it is associated with Hashimoto thyroiditis (HT) that determines a better prognosis with a very low probability of recurrence. The case concerns a 43-year-old female with a single thyroid nodule suspected for cancer; and she had multiple sclerosis (MS) from the age of 19. Thyroid hormone levels were normal such as thyroid antibodies. Total thyroidectomy with lymphadenectomy of central compartment was performed. Histological examination revealed a Warthin like-PTC without Hashimoto thyroiditis. The mechanisms involved in pathogenesis of thyroid cancer in patients with autoimmune disease are not completely clear. We hypothesized that not only a local autoimmune response, such as HT, could contribute to the determination of this type of cancer but also a systemic autoimmune disease such as MS.

Keywords: Thyroid Warthin-like carcinoma; Autoimmune disease; Multiple sclerosis; Hashimoto thyroiditis

Introduction▴Top 

Thyroid cancer (TC) is the most common endocrine malignancy. Among differentiated TCs, the papillary variety (PTC) is more frequently diagnosed. The last edition of World Health Organization (WHO) classification of thyroid tumors considers different variants such as papillary microcarcinoma, encapsulated, follicular, diffuse sclerosing, tall cell, columnar cell, cribriform-morular, hobnail (micro papillary/discohesive), fibromatosis/ fasciitis-like stroma, solid/trabecular, oncocytic, spindle cell, clear cell and Warthin-like [1].

In 1995 Apel described for the first time thyroid Warthin-like cancer (WLPTC). This name comes from the near morphological similarity to the major salivary glands cancer. This variety is characterized by papillary architecture, oxyphil cytology and lymphocytic infiltrates in papillary stalks. He recognized a strength association with Hashimoto thyroiditis (HT) [2]. It seems that patients with WLPTC had similar demographic, clinical, pathologic, and molecular characteristics to those with classic PTC coexisting with HT [3]. Generally, its prognosis is similar to classical papillary cancer, but when associated with HT it seems to be less aggressive. The role of autoimmune systemic disease is still under evaluation; some studies reveal a relationship between thyroid disease and multiple sclerosis (MS) [4-7].

We report a singular case of thyroid Warthin-like carcinoma in a patient with MS, but without HT. Although it is clear the association with autoimmune thyroiditis, we want to underlines probably strength relationship with systemic autoimmune disease even without a local autoimmune thyroiditis.

Case Report▴Top 

Investigations

A 43-year-old woman was admitted to the the Department of Endocrine Surgery of A.O.U. Mater Domini in Catanzaro for a cancer suspected thyroid single nodule. When she was 19, she was diagnosed with MS.

Diagnosis

During the follow-up for this disease, in 2019, she was subjected to a complete thyroidal evaluation. The ecographic sonography revealed the presence of single nodule of 10 × 11 × 10 mm with hyperechogenic spots, in upper sinister lobe of the thyroid gland. No lymphadenomegaly was found. On February 2020, she underwent fine needle aspiration cytology (FNAC) evaluation. It was positive for TIR4 according to SIAPEC classification [8]. Thyroidal hormone and antibody dosage was normal.

Treatment

The patient underwent total thyroidectomy and biopsy of two lymph nodes of central compartment. IOMN was used during surgical procedure.

Postoperative period was uneventful and the patient was discharged on the second postoperative day. Calcium levels were always normal and patient never had dysphonia.

Pathological examination was compatible with papillary carcinoma Warthin-like variety; all lymph nodes were cancer-free. Dexter lobe was 3.5 × 2 × 1.5 mm; sinister lobe was 4 × 2 × 2.5 mm. Dexter lobe had parenchymatous texture and appearance. Sinister lobe compounds a nodule within margins, white color, and size of 0.8 cm. Cytological characteristic was as follow: papillary architecture, oxyphilic cytoplasm and stroma with dense lymphoplasma cellular infiltrate (Fig. 1).

Figure 1.
Click for large image
Figure 1. Cytological characteristic of WLPTC (10 ×). The tumor cells lining the papillae are eosinophilic (lower arrow) and large, the cores of the papillae contain a prominent lymphoplasmacytic infiltrate (upper arrow). This variant shares histological features with Warthin tumor of salivary gland origin. WLPTC: Warthin like-papillary thyroid cancer.

However, there was no evidence of any extra-thyroidal extension or any nodal metastasis.

Follow-up and outcomes

After 7 months, no signs of local recurrence or metastases were observed.

Discussion▴Top 

Warthin-like PTC is a rare entity; there was described only about 160 cases in the world. It is more common in women and median age of diagnosis is 47 years [9].

There are no specific sonographic characteristics or preoperative cytological patterns that could recognize earlier this type of cancer. However, surgical strategy is the same of a classic PTC. Only definitive pathology and immunochemistry can differentiate a PTC from a WLPTC. Often, changes seen in Warthin-like PTC are associated to autoimmune thyroiditis (88%) [10]; its presence is considered a favorable prognostic factor. WLPTC in AT has lower rates of lymph node metastasis, extra-thyroidal extension and recurrence [11].

The lymphocytic infiltration may suggest a not well-defined role for immunological mechanisms in the pathogenesis of thyroid tumors. According to different study, Hashimoto’s disease and MS are autoimmune disorders that may share similar mechanisms for the pathogenesis of the dysregulation of their self-immunity [4].

TCs have been described in immunosuppressed patient, especially with autoimmune disease [5]. The role of systemic autoimmune disease in the pathogenesis of TC was partially studied. There is a strength connection between systemic lupus erythematosus (SLE) and TC, even if the molecular mechanisms are not totally defined [6].

Therefore, we hypothesized a role of MS in the pathogenesis of WLPTC regardless of HT coexistence.

The mechanism involved is unclear; probably immunosuppressive treatments play an important role in association with Epstein-Barr virus (EBV) infection [7].

This case report is characteristic for its presentation: in our case, WLPTC is not associated with HT but with MS. So probably not only a local autoimmune response could contribute to the determination of this type of cancer but also a systemic autoimmune disease. Due to limited literature on this topic, our evaluation is based only on few studies, so further studies are needed to explain the molecular mechanism involved.

Acknowledgments

None to declare.

Financial Disclosure

None to declare.

Conflict of Interest

None to declare.

Informed Consent

The patient has provided permission to publish these features of her case and the identity of the patient has been protected.

Author Contributions

NI, VZ and LSC contributed to think up the design of manuscript. RG and LSC contributed to literature research and language revision. All authors wrote the manuscript.

Data Availability

The data described during the current study are available from the corresponding author on reasonable request.


References▴Top 
  1. Kakudo K, Bychkov A, Bai Y, Li Y, Liu Z, Jung CK. The new 4th edition World Health Organization classification for thyroid tumors, Asian perspectives. Pathol Int. 2018;68(12):641-664.
    doi pubmed
  2. Apel RL, Asa SL, LiVolsi VA. Papillary Hurthle cell carcinoma with lymphocytic stroma. "Warthin-like tumor" of the thyroid. Am J Surg Pathol. 1995;19(7):810-814.
    doi pubmed
  3. Yeo MK, Bae JS, Lee S, Kim MH, Lim DJ, Lee YS, Jung CK. The Warthin-like variant of papillary thyroid carcinoma: a comparison with classic type in the patients with coexisting Hashimoto's thyroiditis. Int J Endocrinol. 2015;2015:456027.
    doi pubmed
  4. Sloka JS, Phillips PW, Stefanelli M, Joyce C. Co-occurrence of autoimmune thyroid disease in a multiple sclerosis cohort. J Autoimmune Dis. 2005;2:9.
    doi pubmed
  5. Milosevic Z, Tanic N, Bankovic J, Stankovic T, Buta M, Lavrnic D, Milovanovic Z, et al. Genetic alterations in quadruple malignancies of a patient with multiple sclerosis: their role in malignancy development and response to therapy. Int J Clin Exp Pathol. 2014;7(4):1826-1833.
  6. Zhang M, Li XM, Wang GS, Qian L, Tao JH, Ma Y, Li XP. Thyroid cancer in systemic lupus erythematosus: a meta analysis. Int J Clin Exp Pathol. 2014;7(9):6270-6273.
  7. Sirbu AM, Sirbu CA, Eftimie L, Soare AM, Ghinescu MC, Ionita-Radu F. Multiple sclerosis, human herpesvirus 4 and thyroid collision tumor: A case report. Exp Ther Med. 2020;20(4):3458-3461.
    doi
  8. Nardi F, Basolo F, Crescenzi A, Fadda G, Frasoldati A, Orlandi F, Palombini L, et al. Italian consensus for the classification and reporting of thyroid cytology. J Endocrinol Invest. 2014;37(6):593-599.
    doi pubmed
  9. Kim EY, Lee KH, Park YL, Park CH, Kim Dae-Hyun, Chae SW, Yun JS. Warthin-like papillary thyroid carcinoma accompanying classical papillary carcinoma: Report of three cases and systematic review of the literature. International Journal of Clinical and Experimental Pathology. 2017;10:7223-7231.
  10. Paker I, Kokenek TD, Yilmazer D, Seker GE, Alper M. Oncocytic variant of papillary thyroid carcinoma with lymphocytic stroma (Warthin-like variant): report of a case with fine needle aspiration cytology and review of the literature. Cytopathology. 2012;23(6):408-410.
    doi pubmed
  11. Gonzalez-Colunga KJ, Loya-Solis A, Cecenas-Falcon LA, Barboza-Quintana O, Rodriguez-Gutierrez R. Warthin-like papillary thyroid carcinoma associated with lymphadenopathy and Hashimoto's thyroiditis. Case Rep Endocrinol. 2015;2015:251898.
    doi pubmed


This article is distributed under the terms of the Creative Commons Attribution Non-Commercial 4.0 International License, which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.


Journal of Medical Cases is published by Elmer Press Inc.

 

Browse  Journals  

 

Journal of Clinical Medicine Research

Journal of Endocrinology and Metabolism

Journal of Clinical Gynecology and Obstetrics

 

World Journal of Oncology

Gastroenterology Research

Journal of Hematology

 

Journal of Medical Cases

Journal of Current Surgery

Clinical Infection and Immunity

 

Cardiology Research

World Journal of Nephrology and Urology

Cellular and Molecular Medicine Research

 

Journal of Neurology Research

International Journal of Clinical Pediatrics

 

 
       
 

Journal of Medical Cases, monthly, ISSN 1923-4155 (print), 1923-4163 (online), published by Elmer Press Inc.                     
The content of this site is intended for health care professionals.
This is an open-access journal distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 International License, which permits unrestricted
non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Creative Commons Attribution license (Attribution-NonCommercial 4.0 International CC-BY-NC 4.0)


This journal follows the International Committee of Medical Journal Editors (ICMJE) recommendations for manuscripts submitted to biomedical journals,
the Committee on Publication Ethics (COPE) guidelines, and the Principles of Transparency and Best Practice in Scholarly Publishing.

website: www.journalmc.org   editorial contact: editor@journalmc.org
Address: 9225 Leslie Street, Suite 201, Richmond Hill, Ontario, L4B 3H6, Canada

© Elmer Press Inc. All Rights Reserved.