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Hindawi Case Reports in Oncological Medicine Volume 2020, Article ID 2084847, 5 pages https://doi.org/10.1155/2020/2084847 Case Report Splenectomy for Solitary Splenic Metastasis in Recurrent Papillary Thyroid Cancer. A Case Report and Literature Review 1 1 1 2 Antonio Maffuz-Aziz , Gabriel Garnica , Silvia López-Hernández, Janet Pineda-Diaz, 2 1 Javier Baquera-Heredia, and Patricia López-Jiménez Department of Surgical Oncology, American British Cowdray Medical Center, Mexico City, Mexico Department of Surgical and Molecular Pathology, American British Cowdray Medical Center, Mexico City, Mexico Correspondence should be addressed to Antonio Maﬀuz-Aziz; tonomaﬀuz@yahoo.com Received 7 October 2019; Revised 18 April 2020; Accepted 22 April 2020; Published 4 May 2020 Academic Editor: Katsuhiro Tanaka Copyright © 2020 Antonio Maﬀuz-Aziz et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Thyroid cancer is the most common endocrine malignancy, presenting with 23 500 new cases per year in the United States. About 7-23% of the patients will present recurrent metastases disease during follow-up. The classic variant of papillary carcinoma is less aggressive compared to its other variants like diﬀuse sclerosing, tall cell or columnar cell, and insular variants, and the sites to which this metastasizes is already well identiﬁed. Metastasis to the spleen is an extremely rare manifestation of papillary thyroid cancer. To date, only 3 cases have been reported in the literature. Herein, we present a 52-year-old male, who developed spleen metastases, 2.4 years after total thyroidectomy and central neck dissection followed by radioactive iodine ablation and seven months after treatment with sorafenib for lung metastases. The splenic lesion was detected in surveillance studies. This case highlights that splenic metastasis, although rare, may occur even in a patient with a locoregional and systemic controlled thyroid cancer and that it can be treated safely with surgical resection. 1. Introduction reported [6–8]. In this article, we present a case of splenic recurrence of papillary thyroid cancer and a literature review. Thyroid cancer is the leading cause of endocrine cancer and represents 2.1% of all cancer cases worldwide. About 90% 2. Case Presentation are well-diﬀerentiated thyroid carcinoma (DTC); papillary cancer is the most common histology . The presence of A 52-year-old male presented with dysphonia of 2 months; distant metastases at the time of diagnosis is 4%, and 7-23% laryngoscopy was performed identifying right vocal cord during follow-up; in nearly 53% of cases, the relapse is paralysis; extension studies identiﬁed a tumour dependent reported in locoregional cancer, 28% in local relapse, and on the right thyroid lobe, with oesophageal inﬁltration and 13% distance metastasis is present; of these, 6% of cases have tracheal displacement, with no evidence of cervical lymph mixed relapses . It has been reported that a global survival nodes. Total thyroidectomy with partial resection of the at 10 years is in a range of 25–70% . oesophagus and lymphadenectomy of the central compart- The most common distant metastasis sites are the lungs ment was performed. The pathology report was classic papil- and bone with 69.0% and 7.1%, respectively . Rare metas- lary thyroid carcinoma 3.6 cm in tumour size, and mixed tasis site locations are extremely low; they have been identi- pattern, with extra thyroid extension, and 1/7 lymph nodes ﬁed in sites such as the liver, adrenal gland, central nervous with metastases. Postoperative iodine-131 dose of 200 mCi system, kidney, and skin and have been reported in 1.85% was delivered, with subsequent iodine-131 tracing that of cases . reported small remnant of functional thyroid tissue in the The presentation of spleen metastases of a primary thy- thyroid bed. He continued hormone replacement therapy roid cancer is even more rare; to date, only 3 cases have been and surveillance. 2 Case Reports in Oncological Medicine Figure 1: Abdominal CT showing cystic lesion in the spleen of 40 mm diameter. Figure 3: Pathology macroscopic picture shows the spleen with a cystic lesion of 5.5 cm. 0.2 cm, with simple papillary formations (Figure 3). In sections with haematoxylin and eosin staining, the neoplastic prolifer- ation cyst is with a papillary growth pattern, and cuboidal cells, nuclear pleomorphism, empty nuclei and abundant nuclear bars are without mitosis (Figure 4). In immunohistochemistry, diﬀuse cytoplasmic positive thyroglobulin, CK19 diﬀuse cyto- plasmic positive, TTF-1 diﬀuse nuclear positive, PAX8 diﬀuse nuclear positive, and CK5/6 negative were observed (Figure 5). The diagnosis was papillary thyroid carcinoma with a cystic pattern, of conventional type, well diﬀerentiated, with focal microcalciﬁcation and intraluminal xanthomatous response, without extracapsular extension. No areas of tall, columnar, or oncocytic cells were identiﬁed. There were no poorly diﬀer- entiated or anaplastic areas. Figure 2: PET/CT images show a 40 mm lesion without increased 3. Discussion metabolic activity. The presence of spleen metastases from solid tumours is At 16 months of surveillance, right basal pulmonary nodule extremely rare and generally exists in the context of a multi- was identiﬁed in X-ray; then, whole-body iodine-131 scan and organ disease. The presence of isolated spleen metastases has thyroglobulin levels were negative, so 18F-ﬂuorodeoxyglucose- been reported <1% in autopsy studies; however, it is associ- positron emission tomography (18F-FDG PET/CT) was per- ated in 17 to 61% with metastases in other distant organs formed which was positive for bilateral pulmonary tumour . In the present case, spleen metastases present after treat- activity; this being the only place where distant metastasis ing lung metastases. Although the frequency of metastatic was found at the time of the study, the spleen was normal. lesions of solid organs to the spleen is rare (2.3-7.1%), it is Thoracoscopy was performed, where pulmonary metastases the most common sites of origin in breast (22.9%), lung were conﬁrmed secondary to well-diﬀerentiated papillary (20.2%), colorectal (9.4%), ovary (9%), and stomach (6.9%) thyroid cancer. Treatment with sorafenib was started, cancer . The reason why this type of dissemination is rare assessing complete pulmonary control after 12 months of is still poorly understood; lack of aﬀerent lymphatic vessel, treatment. the splenic capsule, the immunological capacity of the spleen Seven months after ﬁnished sorafenib treatment, and 29 parenchyma cells (macrophages and lymphocytes), and the months from initial treatment, 18F-FDG PET/CT was per- angled and spiral shape of the splenic artery constitute bar- formed, in which there was no evidence of metabolic activity rier methods for the presence of metastases in this organ . in the lung or in any other organ; however a cystic lesion was To date, only 3 cases of thyroid metastases to the spleen found in the spleen 10 mm in diameter without metabolic have been reported. The ﬁrst case was reported by Paolini activity. A control 18F-FDG PET/CT at 6 months showed et al. ; a patient with history of follicular thyroid cancer, the lung without evidence of disease, and the splenic lesion which developed lung and spleen metastases; the patient grew to a diameter greater than 40 mm; it was observed with- was diagnosed with splenomegaly and inﬁltration to the dia- out metabolic activity (Figure 1). Due to the increment of size phragm, colon, pancreas, and stomach. The second case and risk of spontaneous rupture, it was decided to perform reported was by Mayayo et al. ; with poorly diﬀerentiated splenectomy (Figure 2). thyroid carcinoma, the patient presented abdominal pain at 6 Pathology reported a 5:5×5:5×4cm semi ovoid months of surveillance. Spleen, liver, and pancreas metasta- tumour, cystic-looking lesion, with serous content, wall cut ses were identiﬁed. The diagnosis was made by ﬁne-needle Case Reports in Oncological Medicine 3 (a) (b) Advice on equations (c) (d) Figure 4: (a) Ovoid splenic lesion of cystic appearance, 5.5 cm major axis on the wall. (b) Papillary projections are observed. (c) Photomicrograph in which simple papillae protruding from the cyst wall (haematoxylin and eosin, 4x) are observed. (d). At a higher magniﬁcation, cuboidal cells with clear nuclei and bars, characteristic of papillary thyroid carcinoma (haematoxylin and eosin, 20x) are observed. (a) (b) (c) (d) Figure 5: Photomicrographs of the immunohistochemical study performed on the splenic lesion. (a) PAX8 diﬀuse nuclear positive. (b) Diﬀuse cytoplasmic positive thyroglobulin. (c) TTF-1 diﬀuse nuclear positive. (d) CK19 diﬀuse cytoplasmic positive. 4 Case Reports in Oncological Medicine noma where the 1-year survival rate was 86.6%, and median aspiration cytology (FNA). And the last case reported by Kand et al.  was in a 50-year-old patient with a follicular survival time is 66.6 months , In metastases secondary to variant of a papillary carcinoma, who was diagnosed with melanoma, median overall survival after splenectomy is 11 months, with a survival of 23 months for the subgroup of an iodine-131 uptake study, which was captured at a diﬀuse level throughout the spleen, in addition to associating bone patients treated for a solitary lesion . lesions. The deﬁnitive diagnosis was made using FNA as well. Distant metastasis is considered an important prognostic Our patient was diagnosed incidentally in surveillance factor in papillary thyroid cancer, which aﬀects survival. The studies; he had no symptoms of abdominal pain and it 5-year survival rate is almost 100% for localized papillary, 99% for locoregional cancer and 78% for metastatic papillary seemed only a cystic lesion. Before 1990, when imaging techniques were not used thyroid cancer . eﬀectively, splenic metastasis rates were between 2.3% and For patients with only lung metastases, the survival rate 7.1% and most of them were found during autopsies or were at 10 years is 73.6%, which are signiﬁcantly higher than just encountered coincidentally , because they are mostly patients with multiple organ metastases for whom the 10-year survival rate is 34.3% . asymptomatic. Therefore, studies such as 18F-FDG PET/CT currently have an important tool for detection. In a study performed on 68 oncology patients with FDG avid malig- 4. Conclusions nancy and solid splenic masses on anatomical imaging, Papillary thyroid cancer is a very common neoplasm; there a 18F-FDG PET/CT had 100% accuracy in characterizing lot of information in articles and guides regarding its behav- lesions as benign or malignant. The sensitivity, speciﬁcity, iour and management options. However, on rare behaviour, positive predictive value, and negative predictive value of uncommon site metastases can occur, and its management 18F-FDG PET/CT in diﬀerentiating benign from malignant is not well deﬁned. solid splenic lesions in patients with and without malignant disease are 100%, 100%, 100%, and 100% versus 100%, Data Availability 83%, 80%, and 100%, respectively. It should however be kept in mind that non-FDG-avid tumours, such as some renal or The [DATA TYPE] data used to support the ﬁndings of this thyroid cancers, may metastasize to the spleen [12, 13]. study are included within the article. Although the information in the literature regarding the relationship between 18F-FDG PET/CT and the diagnosis Conflicts of Interest of metastatic spleen lesions is only for solid tumours, the probable explanation is that most well-diﬀerentiated thy- The authors declare that there is no conﬂict of interest roid carcinomas are relatively slow growing and can be regarding the publication of this paper. 18F-ﬂuorodeoxyglucose negative . Several studies have reported that it has a high sensitivity (up to 85%) and References speciﬁcity (up to 95%) for distant metastases in patients with well-diﬀerentiated thyroid cancer .  C. M. Kitahara and J. A. Sosa, “The changing incidence of Use of FNA is a useful diagnosis tool, since a sensitivity of thyroid cancer,” Nature Reviews. Endocrinology, vol. 12, 98.4%, a positive predictive value of 99.2%, and 98.1% accu- no. 11, pp. 646–653, 2016. racy for diagnosis and < 1% of complications have been  R. Cirocchi, S. Trastulli, A. 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Case Reports in Oncological Medicine – Hindawi Publishing Corporation
Published: May 4, 2020
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