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Ishikawa M. Rare malignant ovarian tumors: a review. Jpn J Clin Oncol 2025; 55:205-209. [PMID: 39877943 DOI: 10.1093/jjco/hyaf005] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/29/2024] [Accepted: 01/27/2025] [Indexed: 01/31/2025] Open
Abstract
There are many histologic types of gynecologic malignancies. I reviewed three rare ovarian tumor types that have poor prognoses. Ovarian mesonephric-like adenocarcinoma (MLA) is a newly described histological type known for its aggressive behavior. It is thought to arise from mesonephric duct remnants of the female genital tract and is typically associated with endometriosis. Although MLA has some similarities to endometrioid carcinoma, they have different prognoses. Recurrence of MLA is common, even in early stage cases, and distant metastases, especially in the lungs, are often seen. MLA is characterized by positive immunohistochemical-staining for TTF-1, GATA3, PAX2, and CD10, and negative staining for estrogen and progesterone receptors. Data on treatment for MLA are scarce, and further studies are needed. Adult granulosa cell tumors, the most common type of malignant ovarian sex cord-stromal tumors, have an indolent growth pattern. Chemotherapy, hormone therapy, and radiotherapy have all shown some efficacy. However, debulking surgery remains the most important treatment because tumor disruption or remnants are risk factors for recurrence. Late recurrence is also characteristic of this tumor. Malignant transformations of mature teratoma are suspected when the patient is relatively old and the tumor is large. Squamous cell carcinoma is the most common somatic malignancy. Treatment must be tailored to the transformed histology. Chemotherapy and radiation have shown some efficacy; however, the prognosis is extremely poor in advanced cases. Because these three types of ovarian tumors are rare, research on possible treatments has been difficult, but recent significant advances in drug therapy are expected to lead to the development of effective treatments.
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Affiliation(s)
- Mitsuya Ishikawa
- Department of Gynecology, National Cancer Center Hospital, 5-1-1 Tsukiji, Chuo-ku, Tokyo 104-0045, Japan
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Jordan H, Low G, Wilson MP. Size threshold as a risk for malignant transformation in typical ovarian dermoid lesions: a scoping review. Abdom Radiol (NY) 2024:10.1007/s00261-024-04751-5. [PMID: 39707031 DOI: 10.1007/s00261-024-04751-5] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/01/2024] [Revised: 12/04/2024] [Accepted: 12/05/2024] [Indexed: 12/23/2024]
Abstract
PURPOSE The O-RADS malignancy risk stratification of typical ovarian dermoid cysts by using a 10 cm threshold is based on expert consensus rather than analysis of objective clinical data. This comprehensive scoping review consolidated all currently available studies evaluating typical benign ovarian dermoid cyst size and risk for malignant transformation. METHODS A systematic review of MEDLINE, Embase, Scopus and the Cochrane library was performed from inception to January 14, 2024, using PRISMA-ScR guidelines. A grey literature search and forward searching of reference lists from included studies were performed. Case reports and case series evaluating dermoid cyst size and malignant transformation as defined by a pathological reference standard were included. Data synthesis was provided as a qualitative review of the existing literature. RESULTS Twenty-five studies were included in qualitative synthesis comprising 6 case reports and 19 retrospective studies with a total of 15,295 dermoid cysts. Of these, 215 lesions demonstrated malignant transformation. Studies reporting dermoid size with malignant transformation ranged from 1 to 32 cm with 46/173 (27%) total malignant transformation lesions measuring < 10 cm. Solid enhancing components were infrequently reported but all measured > 1 cm when described. CONCLUSION More than 25% of dermoid cysts with malignant transformation may be classified as "almost certainly benign" with the current 10 cm O-RADS malignancy risk threshold. Although surveillance of O-RADS 2 dermoid cysts may improve sensitivity, modifying a caveat recommendation for MRI O-RADS +/- gyn-oncologist referral when potentially solid components are present in otherwise typically benign dermoid cysts may be appropriate.
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Affiliation(s)
| | - Gavin Low
- University of Alberta, Edmonton, Canada
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3
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Mahroof S, Noori H, Asmati M, Zaki A. Intra-abdominal retroperitoneal fetus in fetu: A case report. Radiol Case Rep 2024; 19:4687-4691. [PMID: 39228932 PMCID: PMC11366918 DOI: 10.1016/j.radcr.2024.07.013] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/28/2024] [Revised: 07/02/2024] [Accepted: 07/03/2024] [Indexed: 09/05/2024] Open
Abstract
Fetus in fetu (FIF) is a rare abnormality where a vertebrate parasitic fetus develops inside the body of another normally developing fetus. It is distinct from teratomas, tumors composed of cells from multiple germ layers and have malignant potential. Symptoms of FIF arise from the mass effect, causing abdominal distension, feeding difficulties, and pressure effects on organs. FIF is commonly found in the retroperitoneal region but can also occur in other locations. It often includes certain organs such as the vertebral column, limbs, central nervous system, gastrointestinal tract, vessels, and genitourinary tract. Early diagnosis of FIF by ultrasound, computed tomography, and magnetic resonance imaging can improve patient outcomes. Surgical resection is the primary treatment approach, aiming to alleviate symptoms, and molecular analysis helps differentiate FIF from malignant teratomas. Regular follow-up is necessary due to the potential recurrence of teratomas.
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Affiliation(s)
- Sahar Mahroof
- Radiology Department, French Medical Institute for Mothers and Children (FMIC), Kabul, Afghanistan
| | - Hasina Noori
- Pediatric Surgery Department, French Medical Institute for Mothers and Children (FMIC), Kabul, Afghanistan
| | - Mamoonullah Asmati
- Pediatric Surgery Department, French Medical Institute for Mothers and Children (FMIC), Kabul, Afghanistan
| | - Adil Zaki
- Radiology Department, French Medical Institute for Mothers and Children (FMIC), Kabul University of Medical Sciences (KUMS), Kabul, Afghanistan
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Luo W, Zheng J, Su B. Low-Grade Glioma within Mature Cystic Teratoma in a Patient with Anti-N-Methyl-D-Aspartate Receptor Encephalitis: A Case Report. Case Rep Oncol 2024; 17:430-437. [PMID: 38449876 PMCID: PMC10917429 DOI: 10.1159/000535708] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/20/2023] [Accepted: 11/21/2023] [Indexed: 03/08/2024] Open
Abstract
Introduction Mature cystic teratoma (MCT) is a common type of ovarian tumors that can, in rare cases, undergo malignant transformation. It has been discovered that MCT patients may experience psychiatric symptoms due to the presence of anti-N-methyl-D-aspartate receptor (NMDAR) antibodies, which is the underlying cause of autoimmune encephalitis. Here, we present the first documented case of a patient with anti-NMDAR encephalitis who also had a morphology of low-grade glioma within MCT. Case Report A 45-year-old woman presented with seizures, altered consciousness, abnormal NMDAR antibody IgG titers, and abnormal brain MRI findings confirm the diagnosis of anti-NMDAR encephalitis. Physical examination revealed an oval mixed echo mass measuring 54 × 37 mm in the left adnexal area on ultrasound of the uterine appendage. The patient underwent laparoscopic left ovarian and fallopian tube resection. The pathological gross examination revealed a pile of grayish-red cystic and solid fragmented tissue measuring 7 × 6 × 2.2 cm. Histological examination revealed characteristic components of MCT. Furthermore, the solid component of the gross tissue showed proliferative and densely arranged astrocytes with cellular atypia, which were positive for GFAP and Olig-2, negative for IDH1 and EMA. And the Ki67 index was approximately 10%, suggesting the presence of low-grade glioma lesions. The patient was diagnosed with malignant transformation of MCT into a morphology of low-grade glioma, not otherwise specified. After the removal of the ovarian tumor, the patient's psychiatric symptoms improved. Conclusions Low-grade glioma within MCT is a rare occurrence, and the presence of this malignant transformation in patients with anti-NMDAR encephalitis is even more uncommon.
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Affiliation(s)
- Wenwen Luo
- Department of Pathology, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, PR China
| | - Jinyue Zheng
- Department of Pathology, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, PR China
| | - Bojin Su
- Department of Pathology, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, PR China
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Arab M, Ashtiani AJ, Faghih N, Ghavami B, Nouri B, Ardabili SN. Malignancy Arising in Dermoid Cysts: A Case Report and Literature Review. J Family Reprod Health 2023; 17:264-268. [PMID: 38807623 PMCID: PMC11128734 DOI: 10.18502/jfrh.v17i4.14599] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 05/30/2024] Open
Abstract
Objective Malignant transformation in dermoid cysts is rare, and Squamous Cell Carcinoma (SCC) is the most common form. This event often occurs in large tumors and middle-aged women. Case report In this study, two cases are presented. They were menopause, and abdominal pain and adnexal mass was a common manifestation in both. Case 1 with adenocarcinoma arising in mature cystic teratoma had abnormal tumor markers and was diagnosed with a frozen section during surgery, but case 2 with SCC transformation had normal tumor markers, and the frozen section was not helpful in the first surgery. Both underwent complete staging surgery, and due to stage IC1 in case 1, she received chemotherapy, and in case 2, no adjuvant treatment was needed because of stage IA. Conclusion Considering the rarity of malignant transformation in the dermoid cyst, the best surgical approach and adjuvant therapy indications need further research.
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Affiliation(s)
- Maliheh Arab
- Department of Gynecology Oncology, Imam Hossein Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Iran
| | - Azadeh Jafari Ashtiani
- Department of Obstetrician and Gynecologist, Shahid Beheshti University of Medical Sciences, Tehran, Iran
| | - Nafiseh Faghih
- Department of Obstetrician and Gynecologist, Shahid Beheshti University of Medical Sciences, Tehran, Iran
| | - Behnaz Ghavami
- Department of Obstetrics and Gynecology, Tehran University of Medical Sciences, Tehran, Iran
| | - Behnaz Nouri
- Department of Obstetrician and Gynecologist, Shahid Beheshti University of Medical Sciences, Tehran, Iran
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Al Harthi S, Al Kalabni M, Al Qassabi B, Al Ajmi R. Mucinous Adenocarcinoma and Carcinoid Tumor Arising Within an Ovarian Mature Cystic Teratoma: A Case Report. Oman Med J 2023; 38:e538. [PMID: 37587986 PMCID: PMC10425607 DOI: 10.5001/omj.2023.40] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/07/2022] [Accepted: 08/30/2022] [Indexed: 08/18/2023] Open
Abstract
The coexistence of both mucinous adenocarcinoma and carcinoid tumor within an ovarian mature cystic teratoma is extremely rare. We report a case of an 18-year-old Omani woman with a left ovarian cyst. After laparotomy with left salpingo-oophorectomy, omentectomy, and peritoneal washings, the excised cyst had the typical morphology of mature cystic teratoma with grade I mucinous adenocarcinoma and a focus of carcinoid tumor.
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Affiliation(s)
- Sara Al Harthi
- Departments of Pathology, Sultan Qaboos University Hospital, Muscat, Oman
| | - Moza Al Kalabni
- Department of Obstetrics and Gynecology, Sultan Qaboos University Hospital, Muscat, Oman
| | - Badriya Al Qassabi
- Department of Radiology, Sultan Qaboos University Hospital, Muscat, Oman
| | - Radiya Al Ajmi
- Departments of Pathology, Sultan Qaboos University Hospital, Muscat, Oman
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Wei CH, Shehayeb S, Santiago NL, Kruper L, Han E, Wang E, Cristea M, Rodriguez-Rodriguez L, Yost SE, Stewart D. BRCA germline mutations in multiethnic gynecologic patients: A 10-year retrospective analysis from a single cancer institute. PLoS One 2023; 18:e0286998. [PMID: 37310942 PMCID: PMC10263324 DOI: 10.1371/journal.pone.0286998] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/07/2023] [Accepted: 05/29/2023] [Indexed: 06/15/2023] Open
Abstract
Histologic and genetic mutation information from racially and ethnically diverse populations is warranted to better inform future cancer predisposition and promote health equity. A single institutional, retrospective capture of patients with gynecologic conditions and genetic susceptibilities to malignant neoplasms of the breast or ovaries was performed. This was achieved with manual curation of the electronic medical record (EMR) from 2010-2020 with the use of ICD-10 code searches. Among 8983 consecutive women identified with gynecologic conditions, 184 were diagnosed with pathogenic/likely pathogenic (P/LP) germline BRCA (gBRCA) mutations. Median age was 54 (22-90). Mutations included insertion/deletion (majority frameshift, 57.4%), substitution (32.4%), large structural rearrangement (5.4%), and alteration in splice site/intronic sequence (4.7%). A total of 48% were non-Hispanic White, 32% Hispanic or Latino, 13% Asian, 2% Black, and 5% Other. The most common pathology was high grade serous carcinoma (HGSC, 63%), followed by unclassified/high grade carcinoma (13%). Additional multigene panels led to the detection of 23 additional BRCA-positive patients with germline co-mutations and/or variants of uncertain significance in genes functionally involved in DNA repair mechanisms. Hispanic or Latino and Asian individuals comprised 45% of patients with concomitant gynecologic condition and gBRCA positivity in our cohort, confirming that germline mutations are represented across racial and ethnic groups. Insertion/deletion mutations, the majority of which led to a frameshift change, occurred in approximately half of our patient cohort, which may have prognostic implication for therapy resistance. Prospective studies are needed to unravel the significance of germline co-mutations in gynecologic patients.
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Affiliation(s)
- Christina H. Wei
- Department of Pathology, City of Hope Comprehensive Cancer Institute, Duarte, California, United States of America
| | - Susan Shehayeb
- Department of Population Sciences, City of Hope Comprehensive Cancer Institute, Duarte, California, United States of America
| | - Nicole Lugo Santiago
- Department of Surgery, City of Hope Comprehensive Cancer Institute, Duarte, California, United States of America
| | - Laura Kruper
- Department of Surgery, City of Hope Comprehensive Cancer Institute, Duarte, California, United States of America
| | - Ernest Han
- Department of Surgery, City of Hope Comprehensive Cancer Institute, Duarte, California, United States of America
| | - Edward Wang
- Department or Medical Oncology & Therapeutics Research, City of Hope Comprehensive Cancer Institute, Duarte, CA, United States of America
| | - Mihaela Cristea
- Department or Medical Oncology & Therapeutics Research, City of Hope Comprehensive Cancer Institute, Duarte, CA, United States of America
| | - Lorna Rodriguez-Rodriguez
- Department of Surgery, City of Hope Comprehensive Cancer Institute, Duarte, California, United States of America
| | - Susan E. Yost
- Department or Medical Oncology & Therapeutics Research, City of Hope Comprehensive Cancer Institute, Duarte, CA, United States of America
| | - Daphne Stewart
- Department or Medical Oncology & Therapeutics Research, City of Hope Comprehensive Cancer Institute, Duarte, CA, United States of America
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Lai PH, Ding DC. Ruptured teratoma mimicking a pelvic inflammatory disease and ovarian malignancy: A case report. World J Clin Cases 2023; 11:3852-3857. [PMID: 37383124 PMCID: PMC10294172 DOI: 10.12998/wjcc.v11.i16.3852] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/16/2023] [Revised: 04/20/2023] [Accepted: 05/06/2023] [Indexed: 06/02/2023] Open
Abstract
BACKGROUND We report a case of ruptured ovarian teratoma mimicking pelvic inflammatory disease (PID) and ovarian malignancy. The case indicates the need for reviewing the information on ovarian teratomas, as the symptoms are vague, and, therefore, diagnosis and treatment had to be structured accordingly.
CASE SUMMARY A 60-year-old woman was admitted to the emergency department with acute lower abdominal pain. She experienced weight loss and increased abdominal girth. Pelvic ultrasound and computed tomography revealed a 14-cm pelvic tumor. Laboratory examination revealed leukocytosis (white blood cell count: 12620/μL, segment: 87.7%) and high levels of C-reactive protein (18.2 mg/dL). Elevated levels of the tumor marker cancer antigen 19-9 (367.8 U/mL, normal value < 35 U/mL) were also noted. Due to the impression of a ruptured tubo-ovarian abscess or a tumor with malignancy, she immediately underwent an exploratory laparotomy. A ruptured ovarian tumor with fat balls, hair strands, cartilage, and yellowish fluid was observed on the right side. Right salpingo-oophorectomy was performed. A pathological examination revealed a mature cystic teratoma. The patient recovered after surgery and was discharged on post-operative day three. No antibiotics were administered.
CONCLUSION This case illustrates the differential diagnosis of an ovarian tumor. Therefore, surgery is the mainstay for treating a ruptured teratoma.
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Affiliation(s)
- Pei-Hsuan Lai
- Department of Obstetrics and Gynecology, Hualien Tzu Chi Hospital, Tzu Chi University, Hualien 970, Taiwan
| | - Dah-Ching Ding
- Department of Obstetrics and Gynecology, Hualien Tzu Chi Hospital, Tzu Chi University, Hualien 970, Taiwan
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Morita K, Yoshioka H, Tanaka K, Sugiura J, Yamamoto R, Goto Y, Yamaki K, Kato W. Intrapericardial bronchogenic cyst extirpated through median sternotomy: a case report. GENERAL THORACIC AND CARDIOVASCULAR SURGERY CASES 2023; 2:20. [PMID: 39516935 PMCID: PMC11533643 DOI: 10.1186/s44215-023-00041-6] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 11/15/2022] [Accepted: 01/22/2023] [Indexed: 11/16/2024]
Abstract
BACKGROUND Bronchogenic cyst sometimes occurs in the mediastinum and rarely in the intrapericardial space. When located in the intrapericardial space, the main vessels or the heart can be compressed. In addition, if it is difficult to deny malignancy or malignancy transformation potential using any modality, surgical resection should be performed. CASE PRESENTATION The patient was a 21-year-old woman with persistent symptoms similar to a cold. Enhanced computed tomography confirmed a 51 × 36 × 35-mm intrapericardial cystic structure with partial calcification. The lesion was large enough to compress and interfere with the venous return of the superior vena cava. Thrombus formation was suspected upstream of the compression site. We performed utter extirpation through median sternotomy. A histopathological examination of the surgical specimen revealed a bronchogenic cyst. The postoperative course was uneventful, and she was discharged on postoperative day 9. CONCLUSION We experienced a case of total extirpation of an intrapericardial bronchogenic cyst complicated with compression of the superior vena cava. Long-term follow-up will be necessary.
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Affiliation(s)
- Koji Morita
- Department of Cardiothoracic Surgery of Japanese Red Cross Aichi Medical Center Nagoya Daini Hospital, Nagoya, Japan.
| | - Hiromu Yoshioka
- Department of Thoracic Surgery of Japanese Red Cross Aichi Medical Center Nagoya Daini Hospital, Nagoya, Japan
| | - Keisuke Tanaka
- Department of Cardiothoracic Surgery of Japanese Red Cross Aichi Medical Center Nagoya Daini Hospital, Nagoya, Japan
| | - Junya Sugiura
- Department of Cardiothoracic Surgery of Japanese Red Cross Aichi Medical Center Nagoya Daini Hospital, Nagoya, Japan
| | - Ryota Yamamoto
- Department of Cardiothoracic Surgery of Japanese Red Cross Aichi Medical Center Nagoya Daini Hospital, Nagoya, Japan
| | - Yuki Goto
- Department of Cardiothoracic Surgery of Japanese Red Cross Aichi Medical Center Nagoya Daini Hospital, Nagoya, Japan
| | - Koshi Yamaki
- Department of Cardiothoracic Surgery of Japanese Red Cross Aichi Medical Center Nagoya Daini Hospital, Nagoya, Japan
| | - Wataru Kato
- Department of Cardiothoracic Surgery of Japanese Red Cross Aichi Medical Center Nagoya Daini Hospital, Nagoya, Japan
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Kamala LH, Abraham S, Radhakrishnan N, Mony RP. Somatic Malignant Transformation in Ovarian Mature Cystic Teratoma - Case Series from a Tertiary Cancer Centre in India. Indian J Surg Oncol 2023; 14:150-154. [PMID: 36891411 PMCID: PMC9986185 DOI: 10.1007/s13193-022-01630-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/14/2021] [Accepted: 08/18/2022] [Indexed: 10/14/2022] Open
Affiliation(s)
| | - Sherry Abraham
- Department of Medical Oncology, Regional Cancer Centre, Trivandrum, 695011 Kerala India
| | | | - Rari P. Mony
- Department of Pathology, Regional Cancer Centre, Trivandrum, 695011 Kerala India
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Muacevic A, Adler JR, Mohamed AA, Alkouz Y, Bahlawan IH. Fetus-in-Fetu: A Differential Diagnosis of Neonatal Fetiform Encysted Abdominal Mass. Cureus 2023; 15:e33725. [PMID: 36793819 PMCID: PMC9925022 DOI: 10.7759/cureus.33725] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 01/12/2023] [Indexed: 01/15/2023] Open
Abstract
Fetus-in-fetu (FIF) is a rare embryological anomaly in which an encysted fetiform mass develops within the infant or adult host body. It mainly occurs intraabdominal. There are embryo-pathogenetic debates over whether it belongs to the spectrum of highly differentiated teratomas or is a parasitic twinning from a monozygotic monochorionic diamniotic pregnancy. The presence of vertebral segments and an encapsulating cyst can reliably distinguish FIF from teratoma. The diagnosis may be initially made by imaging modalities such as computed tomography (CT) and magnetic resonance imaging (MRI), and a confirmatory diagnosis made by histopathology of the excised mass. Our center experienced a case of a male neonate presented after emergency cesarean delivery at 40-week gestation with the suspicion of an intraabdominal mass identified antenatally. Antenatal ultrasonography at 34 weeks gestation suggested the presence of an intraabdominal cystic mass measuring 6.5 cm with a hyperechoic focus. A follow-up MRI performed after the delivery showed a well-defined mass with the cystic formation in the left abdominal region with a centrally located fetiform structure. Vertebral bodies and long limb bones were visualized. The diagnosis of FIF was initially made preoperatively by the characteristic findings of imaging studies. Laparotomy was scheduled on day 6, revealing a large encysted mass with fetiform content. FIF should be considered a possible differential diagnosis of neonatal encysted fetiform mass. Routine antenatal imaging permits more frequent antenatal detection with earlier workup and management.
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Djokic M, Hadzialjevic B, Rankovic B, Dezman R, Tomazic A. Neuroendocrine Tumor Arising within Mature Cystic Teratoma of the Pancreas: Literature Review and Case Report. Curr Oncol 2022; 29:4717-4724. [PMID: 35877234 PMCID: PMC9322391 DOI: 10.3390/curroncol29070374] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/20/2022] [Revised: 06/29/2022] [Accepted: 07/04/2022] [Indexed: 11/16/2022] Open
Abstract
Cystic teratomas are germ cell tumors most commonly found in the ovaries and testes. The pancreas, however, is very rare as a site of occurrence. Moreover, only two cases of cystic teratoma with concomitant neuroendocrine tumor have been reported to date. We report the case of a 33-year-old female who presented with abdominal pain. Computed tomography and magnetic resonance imaging of the upper abdomen revealed an 85 mm cystic tumor in the head of the pancreas. Cystic teratoma and mucinous cystadenoma were suggested as differential diagnoses. Cytopathologic analysis of endoscopic ultrasound-guided fine needle aspiration was consistent with mucinous cystadenoma. Therefore, the patient underwent surgical resection. Histologic analysis revealed a mature cystic teratoma of the pancreas with a concomitant neuroendocrine tumor. The patient is in great condition at 8 months follow-up. Cystic teratoma of the pancreas with a concomitant neuroendocrine tumor is an extremely rare condition. Surgical resection remains the mainstay of treatment as it provides a definitive diagnosis and no recurrences have been reported to date.
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Affiliation(s)
- Mihajlo Djokic
- Department of Abdominal Surgery, Ljubljana University Medical Center, Zaloska Cesta 7, 1000 Ljubljana, Slovenia; (M.D.); (B.H.)
- Department of Surgery, Faculty of Medicine, University of Ljubljana, Zaloska Cesta 7, 1000 Ljubljana, Slovenia
| | - Benjamin Hadzialjevic
- Department of Abdominal Surgery, Ljubljana University Medical Center, Zaloska Cesta 7, 1000 Ljubljana, Slovenia; (M.D.); (B.H.)
| | - Branislava Rankovic
- Institute of Pathology, Faculty of Medicine, University of Ljubljana, Korytkova Ulica 2, 1000 Ljubljana, Slovenia;
| | - Rok Dezman
- Department of Radiology, Ljubljana University Medical Center, Zaloska Cesta 7, 1000 Ljubljana, Slovenia;
| | - Ales Tomazic
- Department of Abdominal Surgery, Ljubljana University Medical Center, Zaloska Cesta 7, 1000 Ljubljana, Slovenia; (M.D.); (B.H.)
- Department of Surgery, Faculty of Medicine, University of Ljubljana, Zaloska Cesta 7, 1000 Ljubljana, Slovenia
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Neuroendocrine Tumors: Clinical, Histological and Immunohistochemical Perspectives and Case Report-Mature Teratoma in a 16-Year-Old Girl. PATHOPHYSIOLOGY 2021; 28:373-386. [PMID: 35366281 PMCID: PMC8830444 DOI: 10.3390/pathophysiology28030025] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/22/2021] [Revised: 08/22/2021] [Accepted: 08/25/2021] [Indexed: 11/22/2022] Open
Abstract
A mature teratoma is a germinal neoplasm that differentiates from embryonic multipotent cells into three germ layers. There may also be glandular tissue. The literature describes a total of 658 cases of ovarian neuroendocrine neoplasms, mainly in women over 40 years of age. The authors, together with a systemic review, present a case of a 16-year-old girl diagnosed with and treated for a neuroendocrine tumor. Case description: A 16-year-old girl visited the Paediatric Gynaecology Outpatient Clinic because of abdominal pains that intensified during menstruation. Standard painkillers and diastolic drugs were ineffective. An ultrasound examination revealed a large tumor with a heterogeneous structure in her right ovary. A sparing operation was carried out. During laparotomy, the lesion was enucleated, leaving healthy tissue. Histopathological examination revealed the typical features of teratoma, as well as the coexistence of a G1 neuroendocrine tumor. Immunohistochemical examination (IHC) showed the presence of markers characteristic for this type of tumor. The patient requires constant monitoring in the Endocrinology and Oncological Gynaecology Clinic. Conclusion: Tissue of neuroendocrine neoplasm within a teratoma is rare in this age group of patients; thus, there are currently no standards for long-term follow-up. This case adds to the body of evidence and demonstrates a possible good prognosis with non-aggressive behavior in G1 neuroendocrine tumors and teratomas in young patients.
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Ono R, Ueno H, Yoshida K, Takahashi S, Yoshihara H, Nozaki T, Suzuki K, Nakazawa A, Saiki R, Seki M, Takita J, Ogawa S, Manabe A, Hasegawa D. Clonal evidence for the development of neuroblastoma with extensive copy-neutral loss of heterozygosity arising in a mature teratoma. Cancer Sci 2021; 112:2921-2927. [PMID: 33934450 PMCID: PMC8253283 DOI: 10.1111/cas.14931] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/09/2021] [Revised: 04/16/2021] [Accepted: 04/19/2021] [Indexed: 11/27/2022] Open
Abstract
Mature teratomas are usually benign tumors that rarely undergo malignant transformation. We report an advanced neuroblastoma arising in a mature teratoma of the ovary. Whole-exome sequencing identified extensive copy-neutral loss of heterozygosity (LOH) in both neuroblastoma and teratoma elements, suggesting that the neuroblastoma evolved from the teratoma. In addition, several truncating germline heterozygous variants in tumor suppressor genes, including RBL2 and FBXW12, became homozygous as a result of LOH. Collectively, we speculate that extensive LOH in teratoma cells may force heterozygous germline variants to become homozygous, which, in turn, may contribute to the development of neuroblastoma with the acquisition of additional chromosomal changes.
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Affiliation(s)
- Rintaro Ono
- Department of Pediatrics, St. Luke's International Hospital, Tokyo, Japan
| | - Hiroo Ueno
- Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan
| | - Kenichi Yoshida
- Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan
| | - Satoko Takahashi
- Department of Pediatrics, St. Luke's International Hospital, Tokyo, Japan.,Department of Pediatrics, Japanese Red Cross Narita Hospital, Chiba, Japan
| | - Hiroki Yoshihara
- Department of Pediatrics, St. Luke's International Hospital, Tokyo, Japan
| | - Taiki Nozaki
- Department of Diagnostic Radiology, St. Luke's International Hospital, Tokyo, Japan
| | - Koyu Suzuki
- Department of Pathology, St. Luke's International Hospital, Tokyo, Japan
| | - Atsuko Nakazawa
- Department of Clinical Research, Saitama Children's Medical Center, Saitama, Japan
| | - Ryunosuke Saiki
- Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan
| | - Masafumi Seki
- Department of Pediatrics, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Junko Takita
- Department of Pediatrics, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan.,Department of Pediatrics, Graduate School of Medicine, Kyoto University, Kyoto, Japan
| | - Seishi Ogawa
- Department of Pathology and Tumor Biology, Graduate School of Medicine, Kyoto University, Kyoto, Japan
| | - Atsushi Manabe
- Department of Pediatrics, St. Luke's International Hospital, Tokyo, Japan.,Department of Pediatrics, Hokkaido University Graduate School of Medicine, Sapporo, Japan
| | - Daisuke Hasegawa
- Department of Pediatrics, St. Luke's International Hospital, Tokyo, Japan
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15
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Tehranian A, Ghahghaei-Nezamabadi A, Seifollahi A, Kasraei S, Dehghani-Nejad H, Maleki-Hajiagha A. Ovarian mature cystic teratoma with malignant transformation: two case reports. J Med Case Rep 2021; 15:23. [PMID: 33499917 PMCID: PMC7839193 DOI: 10.1186/s13256-020-02594-4] [Citation(s) in RCA: 15] [Impact Index Per Article: 3.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/15/2020] [Accepted: 11/19/2020] [Indexed: 11/10/2022] Open
Abstract
BACKGROUND Mature Cystic Teratoma (MCT) is a benign tumor that can lead to malignant transformation (MT) in 1-3% of cases. Management of MT is a big challenge for gynecologic oncologists due to the lack of specific diagnostic and treatment protocols. CASE PRESENTATION We reported two Iranian cases of MT of MCT with two different stages and prognosis. Our both cases presented the same symptoms, including chronic abdominal pain and distention, loss of appetite, and weight loss. In case number 1, despite the large size of the tumor, the disease was at stage Ia and had a good prognosis; while, case number 2 was at stage IIIc of the disease with a poor prognosis. CONCLUSION The stage of the disease is the most important prognostic factor, and early diagnosis and treatment are very critical for better survival.
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Affiliation(s)
- Afsaneh Tehranian
- Department of Obstetrics and Gynecology, Arash Women's Hospital, Tehran University of Medical Sciences, Rashid Ave, Resalat Highway, Tehranpars, P.O Box: 1653915981, Tehran, Iran. .,Research Development Center, Arash Women's Hospital, Tehran University of Medical Sciences, Tehran, Iran.
| | - Akram Ghahghaei-Nezamabadi
- Department of Obstetrics and Gynecology, Arash Women's Hospital, Tehran University of Medical Sciences, Rashid Ave, Resalat Highway, Tehranpars, P.O Box: 1653915981, Tehran, Iran
| | - Akram Seifollahi
- Pathology Department, Arash Women's Hospital, Tehran University of Medical Sciences, Tehran, Iran
| | - Sara Kasraei
- Department of Obstetrics and Gynecology, Arash Women's Hospital, Tehran University of Medical Sciences, Rashid Ave, Resalat Highway, Tehranpars, P.O Box: 1653915981, Tehran, Iran
| | - Hamideh Dehghani-Nejad
- Department of Obstetrics and Gynecology, Arash Women's Hospital, Tehran University of Medical Sciences, Rashid Ave, Resalat Highway, Tehranpars, P.O Box: 1653915981, Tehran, Iran
| | - Arezoo Maleki-Hajiagha
- Research Development Center, Arash Women's Hospital, Tehran University of Medical Sciences, Tehran, Iran
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16
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Tomita Y, Saito T, Okadome M, Sonoda K, Ariyoshi K, Shimamoto K, Nagayama R, Kitade S, Maenohara S. A Glint of Hope for Treatment of Advanced Malignant Transformations of Ovarian Mature Cystic Teratomas: A Retrospective Analysis of 9 Cases. J Gynecol Surg 2020. [DOI: 10.1089/gyn.2019.0134] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022] Open
Affiliation(s)
- Yui Tomita
- Gynecology Service, National Kyushu Cancer Center, Fukuoka, Japan
| | - Toshiaki Saito
- Gynecology Service, National Kyushu Cancer Center, Fukuoka, Japan
| | - Masao Okadome
- Gynecology Service, National Kyushu Cancer Center, Fukuoka, Japan
| | - Kenzo Sonoda
- Gynecology Service, National Kyushu Cancer Center, Fukuoka, Japan
| | - Kazuya Ariyoshi
- Gynecology Service, National Kyushu Cancer Center, Fukuoka, Japan
| | - Kumi Shimamoto
- Gynecology Service, National Kyushu Cancer Center, Fukuoka, Japan
| | - Rina Nagayama
- Gynecology Service, National Kyushu Cancer Center, Fukuoka, Japan
| | - Shoko Kitade
- Gynecology Service, National Kyushu Cancer Center, Fukuoka, Japan
| | - Shoji Maenohara
- Gynecology Service, National Kyushu Cancer Center, Fukuoka, Japan
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17
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Evaluation of adnexal tumours in the International Ovarian Tumor Analysis system in reference to histopathological results. MENOPAUSE REVIEW 2020; 18:141-145. [PMID: 31975980 PMCID: PMC6970421 DOI: 10.5114/pm.2019.90812] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 06/11/2019] [Accepted: 11/07/2019] [Indexed: 11/21/2022]
Abstract
Aim of the study To retrospectively evaluate how the International Ovarian Tumor Analysis (IOTA) simple rules used in ultrasound examinations estimate the probability of malignant and benign tumour occurrence in the studied population. Material and methods The study was performed on a group of 425 patients with ovarian tumours operated in the Clinic of Surgical and Oncological Gynecology at the Medical University of Lodz in the years 2014-2015. Adnexal tumours were rated according to IOTA simple rules, classifying them as probably malignant, probably benign, or unclassified. The results of the study were compared with final histopathological results. The statistical analysis was performed using STATISTICA 13 PL with Medical Pack. Results We analysed data on n = 43 (11%) patients with malignant, n = 346 (86%) patients with benign, and n = 12 (3%) patients with borderline tumours, respectively. Malignant tumour patients were significantly older (mean age 61.0 ±11.6 vs. 43.6 ±16.2 years, p< 0.001), had higher BMI (mean 27.3 ±7.0 vs. 25.2 ±5.2, p< 0.05), more pregnancies (median 2 vs. 1, p = 0.001), and higher cancer antigen 125 (CA 125) concentrations (median 251.5 vs. 18.5, p< 0.001) than patients with a benign tumour. Also, they more often suffered from diabetes mellitus (19% vs. 8%, p = 0.02) and arterial hypertension (60% vs. 42%, p< 0.01) than benign tumour patients. Conclusions In our study, IOTA performance in predicting or ruling out a malignant tumour was highly satisfactory and similar to that of CA 125. Both the methods may be complementary and used to assess the risk of malignant vs. benign ovarian neoplasm, although the context of other clinical variables may also be important.
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18
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Bužinskienė D, Mongirdas M, Mikėnas S, Drąsutienė G, Andreika L, Sakalauskaitė I. Chemical peritonitis resulting from spontaneous rupture of a mature ovarian cystic teratoma: a case report. Acta Med Litu 2019; 26:217-226. [PMID: 32355460 DOI: 10.6001/actamedica.v26i4.4207] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/21/2022] Open
Abstract
Background Mature cystic teratomas (dermoid cysts) are the most common germ cell tumours with 10-25% incidence of adult and 50% of paediatric ovarian tumours. The aetiology of dermoid cysts is still unclear, although currently the parthenogenic theory is most widely accepted. The tumour is slow-growing and in the majority of cases it is an accidental finding. Presenting symptoms are vague and nonspecific. The main complication of a dermoid cyst is cyst torsion (15%); other reported complications include malignant transformation (1-2%), infection (1%), and rupture (0.3-2%). Prolonged pressure during pregnancy, torsion with infarction, or a direct trauma are the main risk factors for a spontaneous dermoid rupture that can lead to acute or chronic peritonitis. The diagnosis of mature cystic teratoma is often made in retrospect after surgical resection of an ovarian cyst, because such imaging modalities as ultrasound, computer tomography, or magnetic resonance imaging cannot yet accurately and reliably distinguish between benign and malignant pathology. Materials and methods We present a report of a clinical case of a 35-years-old female, who was referred to the hospital due to abdominal pain spreading to her feet for three successive days. She had a history of a normal vaginal delivery one month before. Abdominal examination revealed mild tenderness in the lower abdomen; no obvious muscle rigidity was noted. Transvaginal ultrasound showed a multiloculated cystic mass measuring 16 × 10 cm in the pelvis. In the absence of urgency, planned surgical treatment was recommended. The next day the patient was referred to the hospital again, with a complaint of stronger abdominal pain (7/10), nausea, and vomiting. This time abdominal examination revealed symptoms of acute peritonitis. The ultrasound scan differed from the previous one. This time, the transvaginal ultrasound scan revealed abnormally changed ovaries bilaterally. There was a large amount of free fluid in the abdominal cavity. The patient was operated on - left laparoscopic cystectomy and right adnexectomy were performed. Postoperative antibacterial treatment, infusion of fluids, painkillers, prophylaxis of the thromboembolism were administered. The patient was discharged from the hospital on the seventh postoperative day and was sent for outpatient observation. Results and conclusions Ultrasound is the imaging modality of choice for a dermoid cyst because it is safe, non-invasive, and quick to perform. Leakage or spillage of dermoid cyst contents can cause chemical peritonitis, which is an aseptic inflammatory peritoneal reaction. Once a rupture of an ovarian cystic teratoma is diagnosed, immediate surgical intervention with prompt removal of the spontaneously ruptured ovarian cyst and thorough peritoneal lavage are required.
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Affiliation(s)
- Diana Bužinskienė
- Clinic of Obstetrics and Gynaecology, Institute of Clinical Medicine, Faculty of Medicine, Vilnius University, Vilnius, Lithuania.,Centre of Obstetrics and Gynaecology, Vilnius University Hospital Santaros Klinikos, Vilnius, Lithuania
| | | | - Saulius Mikėnas
- Clinic of Gastroenterology, Nephro-Urology and Surgery, Centre of Urology, Institute of Clinical Medicine, Faculty of Medicine, Vilnius University, Vilnius, Lithuania.,Centre of Urology, Vilnius University Hospital Santaros Klinikos, Vilnius, Lithuania
| | - Gražina Drąsutienė
- Clinic of Obstetrics and Gynaecology, Institute of Clinical Medicine, Faculty of Medicine, Vilnius University, Vilnius, Lithuania.,Centre of Obstetrics and Gynaecology, Vilnius University Hospital Santaros Klinikos, Vilnius, Lithuania
| | - Linas Andreika
- Centre of Obstetrics and Gynaecology, Vilnius University Hospital Santaros Klinikos, Vilnius, Lithuania
| | - Indrė Sakalauskaitė
- Clinic of Rheumatology, Orthopaedics Traumatology and Reconstructive Surgery, Institute of Clinical Medicine, Faculty of Medicine, Vilnius University, Vilnius, Lithuania.,Centre of Plastic and Reconstructive Surgery, Vilnius University Hospital Santaros Klinikos, Vilnius, Lithuania
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