Metastatic Bilateral Strumal Carcinoid: A Case Report and Review of the Literature

Borghese, Martina; Razzore, Paola; Ferrero, Annamaria; Daniele, Lorenzo; Mariani, Luca; Sgrò, Luca; Rosa, G.; Biglia, Nicoletta

doi:10.21873/anticanres.13697

Cited by 5 publications

(12 citation statements)

References 10 publications

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“…These tumors have a variable size that can reach up to 26 cm. The OSC are usually unilateral as in our case, although, there are bilateral cases described [8,9]. In 10% of cases, the contralateral ovary may present a tumor, which is usually a cystic teratoma, as occurred in our case [8].…”

Section: Discussionmentioning

confidence: 48%

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Strumal Carcinoid Focus in Mature Cystic Teratoma in a Patient with Breast Cancer and Desire for Fertility Preservation

Bosquet¹,

Muniesa²,

Saborit³

et al. 2020

SCR

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Ovarian primary carcinoid tumors are very rare, accounting for 0.5-1.7% of all carcinoid tumors and 3% of ovarian teratomas. This type of tumor frequently arises from a mature cystic teratoma. The primary ovarian carcinoid tumors are divided into 4 major types, based on their histological characteristics: insular, trabecular or mucinous pattern. Strumal carcinoid refers to insular or trabecular pattern associated with struma ovarii. The strumal type is the most common histologic form, accounting for 40% of primary ovarian carcinoid tumors. Approximately 5% of carcinoid tumors of the non-mucinous type can be malignant. However, in the case of ovarian strumal carcinoid, the occurrence of metastasis is unusual. This article is based on a case of a 30-year-old nulliparous woman diagnosed with strumal carcinoid, who presented a right ovarian tumor suggestive of teratoma synchronous to a breast cancer. She was referred to our center for fertility preservation.

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Section: Discussionmentioning

confidence: 48%

“…Approximately 5% of carcinoid tumors of the nonmucinous type can demonstrate malignant behavior [5]. However, in the case of ovarian strumal carcinoid, the appearance of metastasis is unusual [1,9]. In our case, the excision of the ovarian cystic lesions was performed, as the patient was young and wanted to preserve her fertility.…”

Section: Discussionmentioning

confidence: 99%

Strumal Carcinoid Focus in Mature Cystic Teratoma in a Patient with Breast Cancer and Desire for Fertility Preservation

Bosquet¹,

Muniesa²,

Saborit³

et al. 2020

SCR

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“…A second report described a bilateral case of strumal carcinoid with metastases originating from the neuroendocrine component. CDX2 immunoreactivity was found both in the putative primary tumors and their metastases [10]. Since CDX2 is generally considered as a marker of intestinal neuroendocrine tumors (we were unable to immunelocalize CDX2 in the strumal carcinoids of our series), it also remains questionable whether this case constitutes a genuine metastatic strumal carcinoid or rather represents a case of metastatic (intestinal) neuroendocrine tumor in an ovarian teratoma with thyroid tissue.…”

Section: Discussionmentioning

confidence: 68%

“…The vast majority of strumal carcinoids are biologically indolent and curable by oophorectomy [1]. Only very few cases with metastatic disease were described [9,10]. Only one tumorassociated death has been published [11].…”

Section: Introductionmentioning

confidence: 99%

Immunohistochemical Profile and 47-Gene Next-Generation Sequencing (NGS) Solid Tumor Panel Analysis of a Series of 13 Strumal Carcinoids

et al. 2020

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Strumal carcinoid is an extraordinary rare tumor of the ovary consisting of thyroid tissue intermixed with neuroendocrine tumor component. The cellular origin of strumal carcinoids has been an area of debate. There is also little data on detailed immunohistochemical and molecular characteristics of these neoplasms. For this reason, this series investigated the characteristics of a series of 13 strumal carcinoids using immunohistochemical markers and a 47-gene next-generation sequencing (NGS) solid tumor panel analysis. Both cellular components showed thyroglobulin expression in all tumors. TTF-1 expression was noted in both cellular components of 11 cases. Chromogranin A was positive in both components of most tumors (n = 12, 92.3% in the neuroendocrine component and n = 10, 76.9% in the thyroid follicular component). Synaptophysin stained the neuroendocrine component of all cases, and it was also identified in the follicular thyroid component of a single case. All tumors were negative for CDX2 and calcitonin. ISLET1 was positive in the neuroendocrine component of 8 cases (6.5%). With the exception of one case, all tumors were positive for SSTR2a. The tumors were associated with a low Ki67 labeling index. All cases were microsatellite stable and no pathogenic mutations were identified using a 47-gene NGS solid tumor analysis. This series underscored that strumal carcinoids are distinct neuroendocrine tumors. The synchronous expression for thyroid follicular epithelial and neuroendocrine differentiation biomarkers may suggest a precursor cell origin displaying mixed-amphicrine differentiation. While strumal carcinoids can be diagnosed by their typical morphology and immunohistochemical profile, frequent SSTR expression may serve as a potential theranostic biomarker in the management of affected patients. In addition, the absence of common driver mutations in the NGS solid tumor panel may suggest that these neoplasms seem to be genetically unrelated to follicular epithelial-derived thyroid tumors and potentially different than other commonly identified well-differentiated neuroendocrine neoplasms. Therefore, further studies focusing on molecular characteristics of this entity are still needed.

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“…Four patients developed distant metastases involving peritoneum (carcinosis), breast, bone and liver; one patient underwent recurrence in the homolateral ovary 7 years after surgery and subsequently a second strumal carcinoid in the contralateral ovary almost 30 years later ( 26 ). Disease recurrence was asymptomatic (detected by follow-up imaging) in one patient ( 26 ) and symptomatic in the other four patients. The symptoms associated with the disease recurrence were: lumbago; recrudescence of constipation; amenorrhea; abdominal pain and weight loss ( Table 4 ).…”

Section: Resultsmentioning

confidence: 99%

Ovarian Strumal Carcinoid: Case Report, Systematic Literature Review and Pooled Analysis

et al. 2022

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BackgroundOvarian strumal carcinoid is a rare tumor in which thyroid (struma) and carcinoid components coexist. The disease is generally considered to be a borderline malignancy, however, cases with metastatic disease have been described. No data in the literature are available to guide diagnosis and therapy.MethodsWe performed a pooled analysis and a systematic review of histopathological-confirmed strumal carcinoid cases published in the literature using the following keywords: “strumal carcinoid of the ovary”, “strumal carcinoid case report”. A case of strumal carcinoid tumor diagnosed and followed-up at the Medical Oncology Unit of Spedali Civili (Brescia, Italy) was also described and included.ResultsSixty-six eligible publications were identified, providing data from one hundred and seventeen patients, plus a case diagnosed at our institution. At presentation, among the eighty-eight patients with symptomatic disease, 37% of patients suffered from abdominal distention and 49% from pain due to a growing abdominal tumor mass, 37% from constipation (peptide YY was analyzed in only nine of them, resulting above the physiologic range). Surgery was the primary therapy in 99% of the patients. Three patients had metastatic disease at diagnosis and five patients underwent recurrence after radical surgery. Histology at disease recurrence concerned the thyroid component in two patients, the carcinoid component in two patients, both histologies in one patient. Median disease-free survival and overall survival in this series were not attained.ConclusionStrumal carcinoid of the ovary generally presents a benign behavior and surgery is curative in most cases. However, a small group of patients with this disease can undergo disease recurrence due to both the thyroid and the neuroendocrine (carcinoid) components. A follow-up in radically operated patients is therefore needed, particularly in those with a voluminous disease at diagnosis.

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Metastatic Bilateral Strumal Carcinoid: A Case Report and Review of the Literature

Cited by 5 publications

References 10 publications

Strumal Carcinoid Focus in Mature Cystic Teratoma in a Patient with Breast Cancer and Desire for Fertility Preservation

Strumal Carcinoid Focus in Mature Cystic Teratoma in a Patient with Breast Cancer and Desire for Fertility Preservation

Immunohistochemical Profile and 47-Gene Next-Generation Sequencing (NGS) Solid Tumor Panel Analysis of a Series of 13 Strumal Carcinoids

Ovarian Strumal Carcinoid: Case Report, Systematic Literature Review and Pooled Analysis

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