Kovalenko YA, Zharikov YO, Kiseleva YV, Goncharov AB, Shevchenko TV, Gurmikov BN, Kalinin DV, Zhao AV. Rare primary mature teratoma of the liver: A case report. World J Hepatol 2021; 13(12): 2192-2200 [PMID: 35070019 DOI: 10.4254/wjh.v13.i12.2192]
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05841184
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January 10, 2022, 02:16
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Reader Comments:
I read with great interest the case report by Kovalenko YA et al. about the primary liver teratoma. The case report provides detailed information about the incidence, pathogenesis and classification of teratoma and primary liver teratoma. The article makes a clear distinction between mature and immature teratoma in terms of growth rate, clinical course, malignant potential and prognosis. It also explains the pathogenesis and clinical significance of the Growing teratoma syndrome.
However, there are few issues that should be discussed. It is well known that mature teratoma is diagnosed by the presence of well-differentiated histologic elements derived from at least two of the three germinal layers [1]. When discussing about the diagnosis of extragonadal teratoma, it seems that there are two crucial issues that have to be considered in order to establish the proper diagnosis: exclude metastasis from a primary gonadal teratoma and distinguish it from another type of poorly differentiated neoplasm. It seems that in this case both issues can be discussed. The patient was previously diagnosed with benign ovarian teratoma. Although there were no signs of malignancy and dissemination, still the diagnosis of primary liver teratoma comes in a specific clinical setting. According to the literature, the diagnosis of mature liver teratoma is mainly suspected during the gross pathologic examination of the tumor sections detecting sebaceous material, hair, cartilage, teeth etc [2-5]. The definitive histological diagnosis in most cases was made on H&E stain confirming the presence of mature elements within epidermal structures, bone, fat, thyroid or neural tissue [2-5]. In this case, the H&E stain did not detect any of the previously stated structures and could not conform the diagnosis of teratoma. Moreover, the initial histological diagnosis was biliary hamartoma. When performed, the immunohistochemical tests usually provide a definitive diagnosis. But still, in this case, the assay only confirmed an immunophenotype corresponding to thyroid follicular epithelium, potentially opening the possibility for thyroid neoplasm, despite the normal appearance of the thyroid gland on ultrasound.
I am not a pathology expert, but considering all the above, I think that several other condition must be discussed. Taking into account the immunophenotype for thyroid follicular epithelium, the differential diagnosis of ectopic thyroid tissue [6,7] must be seriously considered, especially if we have in mind that the patient was previously diagnosed with mature ovarian teratoma . We do not have the information whether an immunohistochemical assay was performed on the ovarian teratoma, but in this clinical setting, the current findings justify the suspicion for previous struma ovarii in the differential diagnosis. According to the literature, struma ovarii comprises around 1% of all ovarian tumors and 2–4% of all ovarian teratomas, with the thyroid tissue usually being present in more than 50% of the total tissue [8-10]. Despite the low frequency of detecting ectopic thyroid tissue [11], in this case I think that a scintigraphy could potentially provide useful information that would help for additional clarification of the case. I think that all of these aspects of the case should have been more thoroughly discussed in the article.
In summary, I would conclude that this is a well written, interesting and informative case report that should remind us about the importance of the comprehensive clinical approach during the demanding, challenging and creative diagnostic process.
References
1. Ronchi A, Cozzolino I, Montella M, Panarese I, Zito Marino F, Ros- setti S, et al. Extragonadal germ cell tumors: not just a matter of lo- cation. A review about clinical, molecular and pathological features. Cancer Med 2019;8:6832-6840
2. Gupta R, Bansal K, Manchanda V, Gupta R. Mature cystic teratoma of liver. APSP J Case Rep. 2013 May 2;4(2):13. PMID: 24040591; PMCID: PMC3754398.
3. Rahmat K, Vijayananthan A, Abdullah B, Amin S. Benign teratoma of the liver: a rare cause of cholangitis. Biomed Imaging Interv J. 2006 Jul;2(3):e20. doi: 10.2349/biij.2.3.e20. Epub 2006 Jul 1. PMID: 21614237; PMCID: PMC3097637.
4. Jaklitsch M, Sobral M, de Figueiredo AAFP, Martins A, Marques HP. Rare giant: mature cystic teratoma in the liver. J Surg Case Rep. 2019 Dec 9;2019(12):rjz347. doi: 10.1093/jscr/rjz347. PMID: 31832137; PMCID: PMC6900339.
5. García-Ríos LE, García-Ávila AK, Luna-Castillo M, Anda-González JG, Medrano-Guzmán R. Giant liver teratoma with gliosis peritonei treated by right extended hepatectomy: Overview and case report. Ann Hepatobiliary Pancreat Surg. 2021 Nov 30;25(4):544-550. doi: 10.14701/ahbps.2021.25.4.544. PMID: 34845129; PMCID: PMC8639313.
6. Noussios G, Anagnostis P, Goulis DG, Lappas D, Natsis K. Ectopic thyroid tissue: anatomical, clinical, and surgical implications of a rare entity. Eur J Endocrinol. 2011 Sep;165(3):375-82. doi: 10.1530/EJE-11-0461. Epub 2011 Jun 29. PMID: 21715415.
7. Jamshidi M, Kasirye O, Smith DJ. Ectopic thyroid nodular goiter presenting as a porta hepatis mass. Am Surg. 1998 Apr;64(4):305-6. PMID: 9544138.
8. Kim SJ, Pak K, Lim HJ, Yun KH, Seong SJ, Kim TJ, Lim KT, Jung HW, Park IS, Shim JU, Park CT & Lee KH. Clinical diversity of struma ovarii. Korean Journal of Obstetrics and Gynecology 2002 45 748–752.
9. Yoo SC, Chang KH, Lyu MO, Chang SJ, Ryu HS & Kim HS. Clinical characteristics of struma ovarii. Journal of Gynecologic Oncology 2008 19 135–138. (doi:10.3802/jgo.2008.19.2.135)
10. Ciccarelli A, Valdes-Socin H, Parma J, Khoo SK, Schoumans J, Colao A, Hamoir E & Beckers A. Thyrotoxic adenoma followed by atypical hyperthyroidism due to struma ovarii: clinical and genetic studies. European Journal of Endocrinology 2004 150 431–437. (doi:10.1530/eje.0.1500431)
11. Lim-Dunham JE, Feinstein KA, Yousefzadeh DK & Ben-Ami T. Sonographic demonstration of a normal thyroid gland excludes ectopic thyroid in patients with thyroglossal duct cyst. American Journal of Roentgenology 1995 164 1489–1491
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