Malignant struma ovarii with synchronous primary papillary thyroid cancer in the neck: A case report and literature review

Li,Sijian; Hong,Ruping; Yang,Jiaxin

doi:10.3892/ol.2024.14307

Malignant struma ovarii with synchronous primary papillary thyroid cancer in the neck: A case report and literature review

Authors:
- Sijian Li
- Ruping Hong
- Jiaxin Yang
View Affiliations

Affiliations: National Clinical Research Center for Obstetric and Gynecological Diseases, Department of Obstetrics and Gynecology, Peking Union Medical College Hospital, Beijing 100730, P.R. China, Department of Pathology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences, Peking Union Medical College, Beijing 100730, P.R. China
Published online on: February 23, 2024 https://doi.org/10.3892/ol.2024.14307
Article Number: 174

Metrics: Total Views: 0 (Spandidos Publications: | PMC Statistics: )

Metrics: Total PDF Downloads: 0 (Spandidos Publications: | PMC Statistics: )

Cited By (CrossRef): 0 citations Loading Articles...

Abstract

Malignant struma ovarii (MSO) with synchronous primary thyroid cancer in the neck is extremely rare and lacks a treatment consensus. A 44‑year‑old woman presenting with a left ovarian cyst was admitted to Peking Union Medical College Hospital (Beijing, China) Ultrasonography showed a 6 cm solid‑cystic left ovarian mass with plentiful blood signals. Other notable findings were an elevated CA125 level and a suspected malignant thyroid nodule. A unilateral salpingo‑oophorectomy (USO) was conducted, and the surgical pathology was papillary thyroid cancer (PTC) arising in a struma ovarii. The patient underwent a total thyroidectomy and cervical lymph node dissection, and the pathology of the right lobe nodule was follicular‑variant PTC without capsule invasion or lymph node metastasis (5 mm; pT1aN0M0). No further adjuvant therapy was administered. The serum thyroglobulin value was normal before surgery and was undetectable after thyroidectomy. During regular follow‑up examinations over 4 years, the patient remained well with no evidence of disease (NED). In a literature review, another 13 cases of MSO coexisting with cervical thyroid cancer that had reported outcomes were found. The MSO was confined to the ovary in all cases. A total of nine patients received radioiodine therapy (RAI) treatment after total thyroidectomy. Two patients relapsed and were successfully cured with RAI after the initial surgery Only one patient died due to another disease, while 11 patients showed NED and the remaining patient was alive with the disease after a median follow‑up time of 2 years. This data suggests that USO with personalized RAI may be a preferred option for MSO confined to the ovary plus synchronous primary thyroid cancer due to the conferred satisfactory prognosis.

View Figures

View References

1	Devaney K, Snyder R, Norris HJ and Tavassoli FA: Proliferative and histologically malignant struma ovarii: A clinicopathologic study of 54 cases. Int J Gynecol Pathol. 12:333–343. 1993. View Article : Google Scholar : PubMed/NCBI
2	Outwater EK, Siegelman ES and Hunt JL: Ovarian teratomas: Tumor types and imaging characteristics. Radiographics. 21:475–490. 2001. View Article : Google Scholar : PubMed/NCBI
3	Hanby A and Walker C: Pathology and genetics: Tumours of the breast and female genital organs. WHO classification of tumours series-volume IV. Lyon, France: IARC Press. Breast Cancer Res. 6:1332004. View Article : Google Scholar
4	Goffredo P, Sawka AM, Pura J, Adam MA, Roman SA and Sosa JA: Malignant struma ovarii: A population-level analysis of a large series of 68 patients. Thyroid. 25:211–215. 2015. View Article : Google Scholar : PubMed/NCBI
5	Li S, Yang T, Xiang Y, Li X, Zhang L and Deng S: Clinical characteristics and survival outcomes of malignant struma ovarii confined to the ovary. BMC Cancer. 21:3832021. View Article : Google Scholar : PubMed/NCBI
6	Li S, Yang T, Li X, Zhang L, Shi H, Cheng N and Lang J: FIGO stage IV and age over 55 years as prognostic predicators in patients with metastatic malignant struma ovarii. Front Oncol. 10:5849172020. View Article : Google Scholar : PubMed/NCBI
7	Shrimali RK, Shaikh G and Reed NS: Malignant struma ovarii: The west of Scotland experience and review of literature with focus on postoperative management. J Med Imaging Radiat Oncol. 56:478–482. 2012. View Article : Google Scholar : PubMed/NCBI
8	Shaco-Levy R, Peng RY, Snyder MJ, Osmond GW, Veras E, Bean SM, Bentley RC and Robboy SJ: Malignant struma ovarii: A blinded study of 86 cases assessing which histologic features correlate with aggressive clinical behavior. Arch Pathol Lab Med. 136:172–178. 2012. View Article : Google Scholar : PubMed/NCBI
9	Li S, Kong S, Wang X, Zhang X, Yin M and Yang J: Survival outcomes and prognostic predictors in patients with malignant struma ovarii. Front Med (Lausanne). 8:7746912021. View Article : Google Scholar : PubMed/NCBI
10	Tessler FN, Middleton WD, Grant EG, Hoang JK, Berland LL, Teefey SA, Cronan JJ, Beland MD, Desser TS, Frates MC, et al: ACR thyroid imaging, reporting and data system (TI-RADS): White paper of the ACR TI-RADS committee. J Am Coll Radiol. 14:587–595. 2017. View Article : Google Scholar : PubMed/NCBI
11	Amin MB, Greene FL, Edge SB, Compton CC, Gershenwald JE, Brookland RK, Meyer L, Gress DM, Byrd DR and Winchester DP: The eighth edition AJCC cancer staging manual: Continuing to build a bridge from a population-based to a more ‘personalized’ approach to cancer staging. CA Cancer J Clin. 67:93–99. 2017. View Article : Google Scholar : PubMed/NCBI
12	Dardik RB, Dardik M, Westra W and Montz FJ: Malignant struma ovarii: Two case reports and a review of the literature. Gynecol Oncol. 73:447–451. 1999. View Article : Google Scholar : PubMed/NCBI
13	Janszen EW, van Doorn HC, Ewing PC, de Krijger RR, de Wilt JH, Kam BL and de Herder WW: Malignant struma ovarii: Good response after thyroidectomy and I ablation therapy. Clin Med Oncol. 2:147–152. 2008.PubMed/NCBI
14	Marti JL, Clark VE, Harper H, Chhieng DC, Sosa JA and Roman SA: Optimal surgical management of well-differentiated thyroid cancer arising in struma ovarii: A series of 4 patients and a review of 53 reported cases. Thyroid. 22:400–406. 2012. View Article : Google Scholar : PubMed/NCBI
15	Leite I, Cunha TM, Figueiredo JP and Félix A: Papillary carcinoma arising in struma ovarii versus ovarian metastasis from primary thyroid carcinoma: A case report and review of the literature. J Radiol Case Rep. 7:24–33. 2013.PubMed/NCBI
16	Krishnamurthy A, Ramshankar V, Vaidyalingam V and Majhi U: Synchronous papillary carcinoma thyroid with malignant struma ovarii: A management dilemma. Indian J Nucl Med. 28:243–245. 2013. View Article : Google Scholar : PubMed/NCBI
17	Leong A, Roche PJ, Paliouras M, Rochon L, Trifiro M and Tamilia M: Coexistence of malignant struma ovarii and cervical papillary thyroid carcinoma. J Clin Endocrinol Metab. 98:4599–4605. 2013. View Article : Google Scholar : PubMed/NCBI
18	Brusca N, Del Duca SC, Salvatori R, D'Agostini A, Cannas P, Santaguida MG, Virili C, Bianchi L, Gargano L and Centanni M: A case report of thyroid carcinoma confined to ovary and concurrently occult in the thyroid: Is conservative treatment always advised? Int J Endocrinol Metab. 13:e182202015.PubMed/NCBI
19	Ma D, Guseva NV, Dahmoush L and Robinson RA: Struma ovarii with malignant transformation and germline KIT mutation: A case report with review of the literature. Int J Gynecol Pathol. 35:442–447. 2016. View Article : Google Scholar : PubMed/NCBI
20	Middelbeek RJW, O'Neill BT, Nishino M and Pallotta JA: Concurrent intrathyroidal thyroid cancer and thyroid cancer in struma ovarii: A case report and literature review. J Endocr Soc. 1:396–400. 2017. View Article : Google Scholar : PubMed/NCBI
21	Gomes-Lima CJ, Nikiforov YE, Lee W and Burman KD: Synchronous independent papillary thyroid carcinomas in struma ovarii and the thyroid gland with different RAS mutations. J Endocr Soc. 2:944–948. 2018. View Article : Google Scholar : PubMed/NCBI
22	Tzelepis EG, Barengolts E, Garzon S, Shulan J and Eisenberg Y: Unusual case of malignant struma ovarii and cervical thyroid cancer preceded by ovarian teratoma: Case report and review of the literature. Case Rep Endocrinol. 2019:79641262019.PubMed/NCBI
23	Donato S, Simões H and Leite V: Malignant struma ovarii with concurrent thyroid cancer: Outcomes during and after pregnancy. Eur Thyroid J. 10:523–527. 2021. View Article : Google Scholar : PubMed/NCBI
24	Seo GT, Minkowitz J, Kapustin DA, Fan J, Minkowitz G, Minkowitz M, Dowling E, Matloob A, Asti D, Dhar M, et al: Synchronous thyroid cancer and malignant struma ovarii: Concordant mutations and microRNA profile, discordant loss of heterozygosity loci. Diagn Pathol. 18:472023. View Article : Google Scholar : PubMed/NCBI
25	Boyd JC, Williams BA, Rigby MH, Kieser K, Offman S, Shirsat H, Trites JRB, Taylor SM and Hart RD: Malignant Struma ovarii in a 30-year old nulliparous patient. Thyroid Res. 10:32017. View Article : Google Scholar : PubMed/NCBI
26	Lim ST, Jeong HJ, Chung MJ, Yim CY and Sohn MH: Malignant struma ovarii demonstrated on post-therapy radioiodine scan after total thyroidectomy for papillary thyroid cancer. Clin Nucl Med. 33:429–431. 2008. View Article : Google Scholar : PubMed/NCBI
27	Kitahara CM and Sosa JA: The changing incidence of thyroid cancer. Nat Rev Endocrinol. 12:646–653. 2016. View Article : Google Scholar : PubMed/NCBI
28	Poli R, Scatolini M, Grosso E, Maletta F, Gallo M, Liscia D, Nelva A, Cesario F, Forte G, Metovic J, et al: Malignant struma ovarii: Next-generation sequencing of six cases revealed Nras, Braf, and Jak3 mutations. Endocrine. 71:216–224. 2021. View Article : Google Scholar : PubMed/NCBI
29	Moon S, Song YS, Kim YA, Lim JA, Cho SW, Moon JH, Hahn S, Park DJ and Park YJ: Effects of coexistent BRAF ^V600E and TERT promoter mutations on poor clinical outcomes in papillary thyroid cancer: A meta-analysis. Thyroid. 27:651–660. 2017. View Article : Google Scholar : PubMed/NCBI
30	Siegel MR, Wolsky RJ, Alvarez EA and Mengesha BM: Struma ovarii with atypical features and synchronous primary thyroid cancer: A case report and review of the literature. Arch Gynecol Obstet. 300:1693–1707. 2019. View Article : Google Scholar : PubMed/NCBI
31	Ayhan S, Kilic F, Ersak B, Aytekin O, Akar S, Turkmen O, Akgul G, Toyran A, Turan T and Kimyon Comert G: Malignant struma ovarii: From case to analysis. J Obstet Gynaecol Res. 47:3339–3351. 2021. View Article : Google Scholar : PubMed/NCBI
32	Filetti S, Durante C, Hartl D, Leboulleux S, Locati LD, Newbold K, Papotti MG, Berruti A, ESMO Guidelines Committee and Electronic address: simpleclinicalguidelines@esmo.org: Thyroid cancer: ESMO clinical practice guidelines for diagnosis, treatment and follow-up. Ann Oncol. 30:1856–1883. 2019. View Article : Google Scholar : PubMed/NCBI
33	Yassa L, Sadow P and Marqusee E: Malignant struma ovarii. Nat Clin Pract Endocrinol Metab. 4:469–472. 2008. View Article : Google Scholar : PubMed/NCBI
34	Prat J: FIGO's staging classification for cancer of the ovary, fallopian tube, and peritoneum: abridged republication. J Gynecol Oncol. 26:87–89. 2015. View Article : Google Scholar : PubMed/NCBI