Arduino, Bruno

D'Alessandro, Pietro

Della Corte, Luigi

Giudicepietro, Antonia

IACOBELLI, ALESSANDRO

Insabato, Luigi

Saccone, Gabriele

Zullo, Fulvio

Archivio della ricerca - Università degli studi di Napoli Federico II

European Journal of Obstetrics & Gynecology and Reproductive Biology xxx (2019) xxx–xxxG ModelEURO 10735 No. of Pages 2Short communicationContents lists available at ScienceDirectEuropean Journal of Obstetrics & Gynecology andReproductive Biologyjournal homepage: www.elsevier .com/ locate /e jogrbA case of gastric-type mucinous endocervicaladenocarcinoma in presence of nabothian cystsDear Editor,A group of benign and malignant endocervical glandularlesions unrelated to human papillomavirus (HPV) and showinggastric differentiation, has been described in the last years [1].Minimal deviation adenocarcinoma (MDA) and lobular endo-cervical glandular hyperplasia (LEGH) exhibit a gastricphenotype and immunophenotype [2]. Gastric-type adenocar-cinoma (GAS) has been recently described as a subtype ofcervical adenocarcinoma [3].These neoplasms have aggressiveclinical behavior. There is a likely LEGH-GAS sequence, and anabsence of HPV in cases of LEGH, and MDA suggested that thissequence represents an HPV-independent pathway of carcino-genesis [1]. Morphologically deﬁned GAS exhibits immunore-activity for MUC6 and/or HIK1083, representative markers forpyloric gland differentiation. Notably, GAS is negative forp16INK4a, a marker for high-risk HPV-driven neoplasm [4].Clinical manifestations include watery vaginal discharge,although women can be asymptomatic [5]. Surgery remainsthe main treatment for patients with early-stage lesions.Radiotherapy and/or chemotherapy are recommended forwomen with advanced disease.We described a case ofgastric-type mucinous endocervical adenocarcinoma in awoman with positive Pap smear tests for atypical glandularcells of undetermined signiﬁcance (AGUS) but negative HPV-DNA test. A 46 years old nulliparous woman gave a medicalexamination for hydrorrhea. The gynecological examinationshowed plentiful mucinous secretions from vagina withvaginal fornix preserved. The specular examination showedan eroded and enlarged cervix. The transvaginal ultrasound(US) scan showed multiple Naboth cysts, as well as an unevenappearance and a plentiful vascularization of the uterus, butregular ovaries. The last two Pap smear tests, performed sixmonths and one year before respectively, showed the presenceof atypical glandular cells of undetermined signiﬁcance(AGUS), so patient underwent colposcopy that conﬁrmed theeroded and enlarged aspect of cervix but turned out to benegative for HPV-related lesions. After 3 months, the patientredid Pap smear test which showed the persistence of AGUS,whereas HPV-DNA test was negative. For this reason, thewoman underwent a cervical conization: the histologicexamination of cervical sample, measuring 4  4  2.5 cm,showed a mucinous adenocarcinoma (NOS) moderately differ-entiated (G2) inﬁltrating endocervical margin and part ofsquamous cervical epithelium tissue, with vascular inﬁltration.Her cancer markers (α-FP, CEA, CA 19-9, CA 15-3, CA 125)https://doi.org/10.1016/j.ejogrb.2019.02.0240301-2115/© 2019 Published by Elsevier B.V.Please cite this article in press as: P. D’Alessandro, et al., A case of ganabothian cysts, Eur J Obstet Gynecol (2019), https://doi.org/10.1016/j.eturned out to be all negative. Abdomen and Pelvic MRI showeda tumor that invaded the cervix for all its thickness withoutinvolvement of nearby lymph nodes. The patient was referredto our institution and underwent a laparotomy procedure.Uterus and both annexes appeared macroscopically regular.She underwent a total hysterectomy, with bilateral salpingo-oophorectomy and pelvic and obturator lymphadenectomy.Deﬁnitive pathologic examination showed a mucinous adeno-carcinoma gastric-type, moderately differentiated (G2), inﬁl-trating cervical canal for all its thickness, with a microscopicmetastasis in a lymph node isolated in the right parametriumand in two of pelvic and obturator lymph nodes (pT1B1N1)(Fig. 1A–B). Immunohistochemical analysis turned out to benegative for estrogen receptor (ER) and progesterone receptor(PR) (Fig. 1C) and positive for MUC6 (Fig. 1D). The patient iscurrently being treated with pelvic EBRT (external beamradiation therapy) and concurrent chemotherapy with cisplat-in. The diagnosis of GAS is established primarily based onmorphology: tumor cells with abundant eosinophilic cyto-plasm, distinct cell borders, and a greater degree of cytologicalatypia characterize it [2,4]. However, immunohistochemistrymay contribute to the diagnosis. GAS shows the gastricphenotype, as demonstrated by HIK1083, MUC6, or carbonicanhydrase type IX staining, negativity for p16, as well as afrequent mutant pattern of p53 staining [2]. Unlike MDA andLEGH, the MRI ﬁndings of GAS have not been clearly describedyet, although image analysis based on T2-WI have recentlyshowed a tumor shape classiﬁed as type II, inﬁltrative [5]. GASis associated with aggressive behavior and a poor prognosis,including a possible propensity for peritoneal and adnexaldissemination [5]. Indeed, since the ﬁrst description of GAS in2007, it has been reported its poorer outcomes than those ofusual-type adenocarcinomas (UEA), tipically associated tohigh-risk HPV in over 90% of cases. GAS is rather common inJapan, accounting for up to 20%–25% of all endocervicaladenocarcinomas while it is considered rare in Westerncountries [2]. Kojima et al. showed that GAS had a 5-yeardisease-speciﬁc survival of 30% compared to 77% for UEA [5].The mean age of presentation ranges between 45–48 years. It’srelated to high rate of lymphovascular invasion (LVI) andregional lymph node metastases [2].Here, we discussed thediagnostic difﬁculties for endocervical glandular lesions withgastric differentiation and the possible relationship with thenabothian cysts. This uncommon entity needs a particularattention for its aggressive behavior and because it is not HPVrelated. Screening methods for cancer control for the usualendocervical type like HPV DNA testing are ineffective and thismay result in a probable delay in diagnosis and a worseprognosis. It would be really interesting to evaluate the role ofnabothian cysts in the pathogenesis of this particular tumor.stric-type mucinous endocervical adenocarcinoma in presence ofjogrb.2019.02.024Fig.1. A–D: A. High view of the adenocarcinoma that resemble the gastric adenocarcinoma, this particular ﬁeld highlight a lymphovascular invasion; B. It is possibile to noticethe inﬁltrative pattern of the adenocarcinoma gastric-type. The glands are composed by cells with high clear cytoplasm and distinct borders; C. Immunohistochemicalanalysis: negativity for the estrogen and progesterone receptors that are commonly negative in this type of cervix adenocarcinoma; positive controls stated by the ﬁbroblastsin the stroma; D. Immunohistochemical analysis: positivity for MUC6.2 Short communication / European Journal of Obstetrics & Gynecology and Reproductive Biology xxx (2019) xxx–xxxG ModelEURO 10735 No. of Pages 2Declaration of interest statementThe authors declare that they have no conﬂicts of interest andnothing to disclose.References[1] Park CM, Koh HM, Park S, Kang HS, Shim SS, Kim SY. Gastric type mucinousendocervical adenocarcinoma of the uterine cervix: very rare and interestingcase. Obstet Gynecol Sci 2018;61(January (1)):165–9.[2] Asaka S, Nakajima T, Momose M, Miyamoto T, Uehara T, Ota H. Trefoil factorfamily 2 protein: a potential immunohistochemical marker for aiding diagnosisof lobular endocervical glandular hyperplasia and gastric-type adenocarcinomaof the uterine cervix. Virchows Arch 2019;474(January (1)):79–86.[3] Chung T, DO SI, Na K, Kim G, Jeong YI, Kim YW, et al. Stromal p16 overexpressionin gastric-type mucinous carcinoma of the uterine cervix. Anticancer Res2018;38(January (6)):3551–8.[4] Yamanoi K, Ishii K, Tsukamoto M, Asaka S, Nakayama J. Gastric gland mucin-speciﬁc O-glycan expression decreases as tumor cells progress from lobularendocervical gland hyperplasia to cervical mucinous carcinoma, gastric type.Virchows Arch 2018;473(September (3)):305–11.[5] Park KJ, Kim MH, Kim JK, Cho KS. Gastric-type adenocarcinoma of the uterinecervix: magnetic resonance imaging features, clinical outcomes, and prognosticfactors. Int J Gynecol Cancer 2018;28(July (6)):1203–10.Pietro D’AlessandroAntonia GiudicepietroLuigi Della Corte*Please cite this article in press as: P. D’Alessandro, et al., A case of ganabothian cysts, Eur J Obstet Gynecol (2019), https://doi.org/10.1016/j.eBruno ArduinoGabriele SacconeDepartment of Neuroscience, Reproductive Sciences and Dentistry,School of Medicine, University of Naples Federico II, Naples, ItalyAlessandro IacobelliLuigi InsabatoAnatomic Pathology Unit, Department of Advanced BiomedicalSciences, School of Medicine, University of Naples Federico II, Naples,ItalyFulvio ZulloDepartment of Neuroscience, Reproductive Sciences and Dentistry,School of Medicine, University of Naples Federico II, Naples, Italy* Corresponding author at: Department of Neuroscience, Repro-ductive Sciences and Dentistry, School of Medicine, University ofNaples “Federico II”, Via Pansini 5, Naples, Italy.E-mail address: dellacorte.luigi25@gmail.com (L. Della Corte).Received 28 January 2019Available online xxxstric-type mucinous endocervical adenocarcinoma in presence ofjogrb.2019.02.024

A case of gastric-type mucinous endocervical adenocarcinoma in presence of nabothian cysts

https://www.iris.unina.it/retrieve/handle/11588/752457/251074/148%20Case%20report%20letter%20gastric%20type%20adenocarcinoma%20-%20European%20DOI%20-%20Dalessandro.pdf

A case of gastric-type mucinous endocervical adenocarcinoma in presence of nabothian cysts

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