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| CASE REPORT |
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| Year : 2020 | Volume
: 9
| Issue : 3 | Page : 150-153 |
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An ultralate female growing teratoma syndrome: 19 years after aggressive treatment for advanced ovarian immature teratoma
Tanitra Tantitamit1, Ala U'wais2, Kuan- Gen Huang3
1 Department of Obstetrics and Gynecology, Faculty of Medicine, Srinakharinwirot University, Nakhonnayok, Thailand; Department of Obstetrics and Gynecology, Linkou Chang Gung Memorial Hospital, Al-Karak, Jordan
2 Department of Obstetrics and Gynecology, Mutah University, Al-Karak, Jordan
3 Department of Obstetrics and Gynecology, Linkou Chang Gung Memorial Hospital, Al-Karak, Jordan; Department of Obstetrics and Gynecology, Chang Gung University College of Medicine, Kweishan, Taoyuan, Taiwan
| Date of Submission | 29-Jun-2018 |
| Date of Decision | 20-Sep-2019 |
| Date of Acceptance | 16-Jun-2020 |
| Date of Web Publication | 1-Aug-2020 |
Correspondence Address:
Dr. Kuan- Gen Huang
Department of Obstetrics and Gynecology, Chang Gung University College of Medicine, No. 5, Fu-Hsin Street, Kwei-Shan, Tao-Yuan 333
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/GMIT.GMIT_70_18
We report a rare case with the late occurrence of growing teratoma syndrome (GTS). A 24-year-old woman with Grade 3 immature teratoma of ovary underwent complete surgery and chemotherapy. Nineteen years later, she developed hematuria and pelvic mass that was completely resected and pathology revealed mature cystic teratoma. She has regularly followed up with tumor marker and computed tomography every three months. No evidence of disease has been detected throughout 14 years. In addition, we present a brief review of literature of ovarian GTS in the last decade. We have found that advanced stage, high grade, or early recurrence of germ cell tumor (GCT) could be the risk factors of GTS. It tends to appear within 1 year if the patients had the incomplete resection of primary disease. We stress the importance of long-term follow-up after treatment GCT to early recognition and treatment.
Keywords: Growing teratoma syndrome, immature teratoma, late occurrence, neoplasm, ovary
How to cite this article:
Tantitamit T, U'wais A, Huang KG. An ultralate female growing teratoma syndrome: 19 years after aggressive treatment for advanced ovarian immature teratoma. Gynecol Minim Invasive Ther 2020;9:150-3 |
How to cite this URL:
Tantitamit T, U'wais A, Huang KG. An ultralate female growing teratoma syndrome: 19 years after aggressive treatment for advanced ovarian immature teratoma. Gynecol Minim Invasive Ther [serial online] 2020 [cited 2023 Mar 24];9:150-3. Available from: https://www.e-gmit.com/text.asp?2020/9/3/150/291264 |
| Introduction | |  |
Growing teratoma syndrome (GTS) of the ovary is uncommon which is characterized by enlargement of mature teratoma mass, combined with the normal tumor marker. It can appear at any time during or after the treatment of germ cell tumor (GCT). This condition should be differentiated from recurrence disease. Early recognition is a key to curative resection and good outcome. We report on a case of GTS, diagnosed 19 years after primary treatment of immature teratoma.
| Case Report | | |
A 24-year-old woman, G3P2, was diagnosed with ovarian tumor during pregnancy. She underwent ovarian cystectomy at the local hospital. Six months later, she came to our hospital due to pelvic discomfort and pelvic mass. On exploration, frozen section pathology reported immature teratoma. Due to advanced cancer status, total hysterectomy and bilateral salpingo-oophorectomy were performed with no residual disease. Final pathology confirmed the diagnosis of immature teratoma Grade 3. Postoperative adjuvant chemotherapy was arranged with 3 cycles of bleomycin, etoposide and cisplatin, 3 cycles of PE (etoposide and cisplatin), and 6 cycles of vincristine, adriamycin, and cyclophosphamide. After chemotherapy, the serum tumor markers (alpha-fetoprotein [AFP], CA 125) fell within normal range. Unfortunately, the patient was lost to follow-up and returned to our clinic due to abdominal discomfort and hematuria 19 years later. Computed tomography (CT) showed huge cystic mass occupying the entire pelvic cavity with bilateral hydronephrosis. The multiple small cystic lesions at mesenteric, perihepatic, and subphrenic region were also noted. The level of the AFP was found to be within normal limit (4.19 ng/ml) but the CA-125 was raised to 147.54 U/ml. She was arranged for debulking of the tumor including pelvic mass excision, bilateral ureterolysis, omentectomy, retroperitoneal lymph node resection, small bowel resection, and Hartmann's procedure. During operation, there was a huge multicystic mass filling the entire pelvis and abdomen. An isolated mass invaded ileum 40 cm in length and multiple cystic mass, ranging from 0.2 to 5 cm in size, over peritoneum, intestinal serosa, and intestinal mesentery were also found. The huge tumor compressed bladder, rectum, and bilateral ureter resulting in bilateral hydroureters and hydronephrosis. Histology revealed a mature teratoma, confirming the diagnosis of GTS of the ovary. No further chemotherapy was administered. The patient has regular follow-up with tumor marker and CT at the 3-month interval. At the time of this report, she remains free of disease 14 years after the primary GTS was diagnosed.
| Discussion | | |
GTS is an unusual condition characterized by (1) enlargement of tumors during or after chemotherapy for GCT, (2) normalization of tumor markers, and (3) the absence of any germ cell component other than mature teratoma. The median age at diagnosis of GCT ranges from 22 to 26 years.[1],[2],[3] We reviewed the literature of ovarian GTS reported in the last decade [Table1]. The etiology, pathogenesis, and risk factor remain unclear. The previous study reported that the presence of mature teratoma in the first tumor, the incomplete resection, and the absence of volume mass reduction during chemotherapy were predictors.[4] Recent studies revealed that the patient who developed GTS tended to have more advanced stage and were more likely to have received chemotherapy.[1],[3] The median interval from diagnosis of GCT to development GTS was 7–26.6 months.[2],[5] | Table 1: Reported cases of ovarian growing teratoma syndrome; literature search during 2008-2017 period
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Our case exhibits a late occurrence of GTS, 228 months after chemotherapy. Differential diagnosis includes recurrent IT and GTS. Because of the growing mass with normal tumor marker, in the context of regression GCT after chemotherapy, GTS should be considered. The advanced stage of GCT may be the risk factor of GTS in this case. From the literature reviews, most of the primary GCT cases were in Stage III. There were three cases reported of patients with Stage I GCT. Two in three cases had high-grade histology. Another patient with Grade 1 had tumor recurrence at 4 months. This could be explained by a tendency for understaging from fertility-sparing surgery.[6],[7],[8] It seems possible that the advanced stage, high-grade histology, and early recurrent of GCT might be the risks of GTS. The most frequent metastatic site was the site involved in primary tumor.[1] Other sites involved are large bowel, small bowel, omentum, abdominal wall, and trocar port site. Despite its benign nature, GTS leads to serious morbidity including mechanical complication and malignant transformation which occurred in 3%–5%. For this reason, it is essential for early diagnosis and treatment as much as possible. The problem is some locations of disease are not easy to be completely resected. Several types of chemotherapy and immunotherapy have been investigated to treat unresectable masses.[3],[9] However, the evidence is far from being confirmed. Even after complete resection, long-term recurrence GTS may occur in 0%–4%.[5] Therefore, lifelong follow–up is mandatory. Close follow-up with serial tumor marker and imaging by CT or magnetic resonance imaging are highly recommended. In properly treated, the overall prognosis of GTS is good.
| Conclusion | | |
Unusual presentations and complications of GCT should be kept in mind.[10] GTS nodule can occur anytime. There is no prevention strategy for this condition. It highlights that the clinicians should be aware of this condition even in patients without definite risk. Long-term and regular follow-up is crucial. Early diagnosis and early treatment can increase chances of complete resection which is associated with good outcomes and minimal morbidity.
Ethical approval
This study was approved by the institutional review board of Chang Gung Medical Foundation (approval no. 202000995B0 obtained on June 15, 2020) and the IRB approved the waiver of the participants' consent.
Acknowledgment
We would like to address special thanks to Dr. Chi-Ju Yeh and his colleagues at the Department of Pathology, Chang Gung Memorial Hospital for their efforts in pathological diagnosis and keeping the archives.
Financial support and sponsorship
Nil
Conflicts of interest
There are no conflicts of interest.
| References | | |
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| 4. | Lorusso D, Malaguti P, Trivellizzi IN, Scambia G. Unusual liver locations of growing teratoma syndrome in ovarian malignant germ cell tumors. Gynecol Oncol Case Rep 2011;1:24-5. |
| 5. | Kato N, Uchigasaki S, Fukase M. How does secondary neoplasm arise from mature teratomas in growing teratoma syndrome of the ovary? A report of two cases. Pathol Int 2013;63:607-10. |
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| 7. | Shigeta N, Kobayashi E, Sawada K, Ueda Y, Yoshino K, Hori Y, et al. Laparoscopic excisional surgery for growing teratoma syndrome of the ovary: Case report and literature review. J Minim Invasive Gynecol 2015;22:668-74. |
| 8. | Soufi M, Lupinacci RM, Godiris-Petit G, Vignot S, Genestie C, Menegaux F, et al. Growing teratoma syndrome of the ovary presenting with liver metastasis: Report of a case. Eur J Gynaecol Oncol 2015;36:473-6. |
| 9. | Daher P, Riachy E, Khoury A, Raffoul L, Ghorra C, Rehayem C. Growing teratoma syndrome:First case report in a 4-year-old girl. J Pediatr Adolesc Gynecol 2015;28:e5-7. |
| 10. | Terada A, Tasaki S, Tachibana T, Sakamoto Y, Yokomine M, Shimomura T, et al. Two cases of acute limbic encephalitis in which symptoms improved as a result of laparoscopic salpingo-oophorectomy. Gynecol Minim Invasive Ther 2017;6:34-7. |
[Table 1]
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