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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 8  |  Issue : 1  |  Page : 11-15

Papillary carcinoma thyroid evolving within struma ovarii: An unusual occurrence


Department of Pathology, Jawaharlal Nehru Medical College (JNMC), Aligarh Muslim University (AMU), Aligarh, Uttar Pradesh 202002, India

Date of Submission17-Aug-2021
Date of Decision20-Aug-2021
Date of Acceptance29-Sep-2021
Date of Web Publication22-Apr-2022

Correspondence Address:
Shagufta Qadri
Department of Pathology, Jawaharlal Nehru Medical College (JNMC), Aligarh Muslim University (AMU), Aligarh, Uttar Pradesh 202002
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijhi.IJHI_2_21

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  Abstract 

Struma ovarii is a monodermal variant of ovarian teratoma. It is a very rare, germ cell tumor of the ovary, constituting less than 1% of all ovarian tumors. Thyroid tissue is the predominant element, constituting more than 50% of the total. Thyroid-type carcinoma arising in struma ovarii is a rare finding, of which papillary carcinoma being more common, followed by follicular carcinoma and a newer entity termed as highly differentiated follicular carcinoma of ovarian origin. A case of 48-year-old female presented with complaints of acute lower abdominal pain and vaginal bleeding. Imaging showed a solid cystic mass in left adnexa whereas right adnexa was unremarkable. Left salpingo-oophorectomy was performed. Histopathological examination and subsequent immunohistochemistry revealed struma ovarii with neoplastic transformation into papillary thyroid carcinoma. Malignant struma ovarii is difficult to diagnose clinicoradiologically and is mostly discovered incidentally, with only handful of cases published in the literature.

Keywords: Malignant, ovary, PTC, struma ovarii, teratoma


How to cite this article:
Qadri S, Shams A, Alam K, Irfan S. Papillary carcinoma thyroid evolving within struma ovarii: An unusual occurrence. Int J Histopathol Interpret 2019;8:11-5

How to cite this URL:
Qadri S, Shams A, Alam K, Irfan S. Papillary carcinoma thyroid evolving within struma ovarii: An unusual occurrence. Int J Histopathol Interpret [serial online] 2019 [cited 2022 May 24];8:11-5. Available from: https://www.ijhi.org/text.asp?2019/8/1/11/343725




  Introduction Top


Struma ovarii (“goiter of the ovary”) is an uncommon monodermal variant of teratoma and constitutes less than 1% of all ovarian tumors and 2–5% of ovarian teratomas.[1],[2] It was initially described by Bottlin in 1888[3] and later by Pick in 1901 who suggested that ovarian goiters are teratomas in which thyroidal tissue has exceeded the rest of the tissue.[4] This term is used when the thyroidal element constitutes more than 50% of the ovarian tumor.[1] Most cases are benign; however, 5–10% of struma ovarii undergo malignant transformation.[5] Distinction between the two is difficult as there are no uniform criteria and the tumor is rare.[5] The most common tumor is papillary thyroid carcinoma (PTC) with an incidence of 70%, of which 44% are classical type and 26% are follicular variant of PTC.[6] Recently, a new entity, highly differentiated follicular carcinoma of ovarian origin (HDFCO) which shows extra ovarian dissemination and resemblance to non-neoplastic thyroid tissue, has been described.[7]


  Case Presentation Top


A 48-year-old female (Para 2 Live 2) presented with lower abdominal pain, off and on vaginal spotting for a duration of 5 months. She previously had normal menstrual cycles and no medical illness. Her vitals were stable and general physical examination within normal limits except for mild pallor. Complete blood count revealed hemoglobin 10.5%, and thyroid function tests were normal. Transvaginal ultrasound showed the presence of a small solid and cystic mass with a diameter of 4 cm in the left adnexa. Contrast-enhanced computed tomography revealed a well-defined mass with slight enhancement in the left adnexa, without any lymphadenopathy or peritoneal dissemination. CA 125 was found to be elevated to be 455 IU/L.

The patient underwent laparotomy with left salpingo-oophorectomy. Intraoperatively, the left ovary appeared as a unilocular cystic mass with a solid component. Findings in the uterus and right adnexa were unremarkable. There were no enlarged lymph nodes, metastasis, or disseminated lesions in the intraperitoneal organs. Clinically, a diagnosis of complex hemorrhagic cyst or left adnexal cyst was made.

On gross examination, left ovarian mass measured 4 × 3.5 × 1.2 cm and contained both cystic and solid components. The cystic component measured 1.5 × 1.5 cm with the solid component measuring 2.5 × 1.8 cm. Histological sections demonstrated stratified squamous lining with the adnexa [Figure 1] and [Figure 2]. The underlying tissue showed variable sized thyroid follicles filled with pink staining, homogeneous colloid, lined with cuboidal or columnar epithelium and separated by fibrous septa along with a solid nodular proliferation in the center [Figure 2] and [Figure 3], comprising more than 50% of the total tumor area characterized by papillae formation along with small follicles and solid areas consisting tumor cells that were exhibiting increased N/C ratio, irregular nuclear membrane with pale chromatin, nuclear grooves, and intranuclear cytoplasmic invaginations [Figure 4]. Mitotic figures were <1 per 10 high-power fields. No evidence of necrosis or marked cell atypia was seen. No lymphovascular invasion was seen. Diagnosis of PTC developing in struma ovarii was made. Immunohistochemical staining was done and CK-19 [Figure 5] and galectin-3 [Figure 6] were positive in the tumor cells, further confirming the diagnosis of malignant struma ovarii.
Figure 1: Normal appearing skin and adnexa of dermoid cyst along with numerous colloid-filled follicles along with solid areas comprising PTC (extreme left) [H&E ×40]

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Figure 2: Numerous colloid-filled follicles along with solid areas comprising PTC [H&E ×40]

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Figure 3: Tumor cells arranged in follicles, solid nodule, and complex papillary structures [H&E ×100]

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Figure 4: PTC: showing solid areas and small follicles lined by cells exhibiting nuclear clearing, grooving, and overcrowding [H&E ×400]

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Figure 5: Cytoplasmic and membrane positivity of CK-19 in the tumor cells of PTC [IHC ×400]

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Figure 6: Cytoplasmic positivity of galectin-3 in the tumor cells of PTC [IHC ×400]

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  Discussion Top


Struma ovarii is a very rare tumor representing less than 1% of all ovarian tumors.[1] Histologically, it can be benign or malignant.[8] Malignancy is seen in less than 5% of the cases[9] and the rate of metastasis is still very low (0.3–0.5%).[10] Average age of presentation is in the fifth decade[3] as was the case in our patient. The common presenting symptoms are pain abdomen, palpable pelvic mass, bleeding per vaginum, or maybe an incidental finding on ultrasound.[11] Ascites and occasionally Meigs syndrome can be associated.[3]

Matsuda et al.[12] reported a case of a 48-year-old woman who presented with symptoms of hyperthyroidism and an ovarian mass. They also observed that approximately 92% of the patients are euthyroid, whereas hyperthyroidism is seen in the remaining 8%. DeSimone et al.[9] found that approximately 94% of the thyroid-type carcinomas arising in struma ovarii are unilateral and the left ovary is more commonly involved than the right ovary with an average size of 10.5 ± 4.69 cm. Grossly, tumors as small as 2.5 cm and as large as 11 cm[12] have been reported, and an analysis of large series of 68 patients showed a mean tumor size of 5.28 cm.[13] Roth and Talerman[3] found papillary carcinoma to be the most common thyroid-type carcinoma to occur in struma ovarii. No definite and uniform criteria for diagnosis have been established. They also outlined that the criteria for diagnosis of conventional papillary carcinoma include a true papillary architecture with a central fibrovascular core and variable sized thin-walled blood vessels in loose stroma. These papillae are lined by one to several layers of neoplastic cells, with crowded, round, or oval nuclei that show overlapping and have clear “ground glass” chromatin. Mitoses are rarely present. Intranuclear inclusions of cytoplasm and nuclear grooves are often found. All these histopathological features were present in our case. Similar nuclear features in the absence of papillary architecture or with minimal papillae are present in the follicular variant of PTC.

Roth and Karseladze[7] also described a new variant of follicular carcinoma—HDFCO, a rare entity, known to arise in the struma ovarii that resembles non-neoplastic thyroid tissue. The clinical presentation and radiological findings have great resemblance to cystic ovarian neoplasms.

Thyroid-type carcinoma arising in struma ovarii may mimic other primary ovarian tumors, such as granulosa cell tumor, Brenner tumor, papillary serous cystadenoma, or cystadenocarcinoma. Distinction from these primary ovarian tumors is crucial as misdiagnosis would lead to improper treatment. Cytomorphological features of neoplastic cells on histopathological section combined with the combination of immunohistochemistry staining with thyroglobulin, TTF-1, help to confirm the thyroid origin, whereas CK-19, HBME-1, and galectin-3 further confirm the PTC. The malignant thyroid carcinoma of ovary are also positive for RET/PTC rearrangement, whereas benign struma ovarii are negative for CK-19, HBME-1, and RET/PTC rearrangement.[14] PTC arising from struma ovarii is associated with a good prognosis. The 5- and 25-year survival rates were observed to be 92% and 79%, respectively.[15]


  Conclusion Top


A multimodal approach to the treatment is therefore mandatory in order to maximize the disease-free survival and decrease the risk of recurrence. Surgical removal of the ovarian mass remains the main stay of treatment; however because of its rare occurrence, the diagnosis and management of this tumor are yet to be clearly defined.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Devaney K, Snyder R, Norris HJ, Tavassoli FA. Proliferative and histologically malignant struma ovarii: A clinicopathologic study of 54 cases. Int J Gynecol Pathol 1993;12:333-43.  Back to cited text no. 1
    
2.
Yoo SC, Chang KH, Lyu MO, Chang SJ, Ryu HS, Kim HS. Clinical characteristics of struma ovarii. J Gynecol Oncol 2008;19:135-8.  Back to cited text no. 2
    
3.
Roth LM, Talerman A. The enigma of struma ovarii. Pathology 2007;39:139-46.  Back to cited text no. 3
    
4.
Gruhn J. A selective historical survey of ovarian pathology emphasizing neoplasms. In: Roth LM, Czernobilsky B, editors. Tumors and Tumor-like Conditions of the Ovary. Vol. 6. New York: Churchill Livingstone; 1985. p. 269-85.  Back to cited text no. 4
    
5.
Kostoglou-Athanassiou I, Lekka-Katsouli I, Gogou L, Papagrigoriou L, Chatonides I, Kaldrymides P. Malignant struma ovarii: Report of a case and review of the literature. Horm Res 2002;58:34-8.  Back to cited text no. 5
    
6.
Makani S, Kim W, Gaba AR. Struma ovarii with a focus of papillary thyroid cancer: A case report and review of the literature. Gynecol Oncol 2004;94:835-9.  Back to cited text no. 6
    
7.
Roth LM, Karseladze AI., Karseladze AI. Highly differentiated follicular carcinoma arising from struma ovarii: A report of 3 cases, a review of the literature, and a reassessment of so-called peritoneal strumosis. Int J Gynecol Pathol 2008;27:213-22.  Back to cited text no. 7
    
8.
Kraemer B, Grischke EM, Staebler A, Hirides P, Rothmund R. Laparoscopic excision of malignant struma ovarii and 1 year follow-up without further treatment. Fertil Steril 2011;95:2124.e9-12.  Back to cited text no. 8
    
9.
DeSimone CP, Lele SM, Modesitt SC. Malignant struma ovarii: A case report and analysis of cases reported in the literature with focus on survival and I131 therapy. Gynecol Oncol 2003;89:543-8.  Back to cited text no. 9
    
10.
McGill JF, Sturgeon C, Angelos P. Metastatic struma ovarii treated with total thyroidectomy and radioiodine ablation. Endocr Pract 2009;15:167-73.  Back to cited text no. 10
    
11.
Al Hassan MS, Saafan T, El Ansari W, Al Ansari AA, Zirie MA, Farghaly H, et al. The largest reported papillary thyroid carcinoma arising in struma ovarii and metastasis to opposite ovary: Case report and review of literature. Thyroid Res 2018;11:10.  Back to cited text no. 11
    
12.
Matsuda K, Maehama T, Kanazawa K. Malignant struma ovarii with thyrotoxicosis. Gynecol Oncol 2001;82:575-7.  Back to cited text no. 12
    
13.
Goffredo P, Sawka AM, Pura J, Adam MA, Roman SA, Sosa JA. Malignant struma ovarii: A population-level analysis of a large series of 68 patients. Thyroid 2015;25:211-5.  Back to cited text no. 13
    
14.
Boutross-Tadross O, Saleh R, Asa SL. Follicular variant papillary thyroid carcinoma arising in struma ovarii. Endocr Pathol 2007;18:182-6.  Back to cited text no. 14
    
15.
Barrera JR, Manalo LA. Papillary thyroid-type carcinoma arising from struma ovarii. Case Reports 2012;2012:bcr0320126145.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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