{"paper_id":"b49e1ade-0ac5-4828-8e38-20b38b047daf","body_text":"Journal of Case Reports and Images in Oncology, Volume 9, Issue 2, 2023; Pages 19–23. ISSN: 2582-1318\nJ Case Rep Images Oncology 2023;9(2):19–23. \nwww.ijcrioncology.com\nHe et al. 19\nCASE REPORT OPEN ACCESS \nA case report of a rare endocervical-type typical polypoid \nadenomyoma\nHua He, Nuermanguli Rouzi, Ting-An Chen,  \nYu Hou, Ling Zhang, Guang-Zong Zhao\nABSTRACT\nIntroduction: Endocervical-type typical polypoid \nadenomyoma is an exceptionally rare benign tumor, \nwhich should be seldom reported to date. We aim to \nshare our clinical experience about endocervical-type \ntypical polypoid adenomyoma and review relevant \npublications to decrease the rates of misdiagnosis and \nmissed diagnosis.\nCase Report:  A 15-year-old adolescent had recurrent \nirregular vaginal bleeding persisting for six months, and \nthe vaginal mass could not be returned after toileting \nfinally. Ultrasound examination revealed a huge mass \nwith heterogeneous hypoechogenicity and prominent \nintramural blood flow, comprising multiple cystic \nareas. Magnetic resonance imaging demonstrated \nsignificant endometrial thickening, and a well-defined \npedunculated mass of varying signal intensity protruding \ninto the vaginal orifice. Hysteroscopy was performed, \nand a pathologic biopsy of the mass was conducted. \nPathological analysis revealed an endometrial epithelial \nmonolayer overlaying the tissue, with subtle glandular \nhyperplasia and partial gland expansion. The stroma \nconsisted of fibrous connective tissue with a few smooth \nmuscle fibers.\nHua He 1,#, Nuermanguli Rouzi 1,#, Ting-An Chen 2, Yu Hou 1, \nLing Zhang1, Guang-Zong Zhao1\nAffiliations: 1Department of Obstetrics and Gynecology, \nGuangyuan Central Hospital, Guangyuan, Sichuan, China; \n2Department of Pathology, Guangyuan Central Hospital, \nGuangyuan, Sichuan, China.\nCorresponding Author: Guang-Zong Zhao, Guangyuan Cen-\ntral Hospital, No. 16 Jing Alley, Lizhou District, Guangyuan \nCity, Sichuan, China; Email: wstczgz@163.com\nReceived: 24 July 2023\nAccepted: 01 September 2023\nPublished: 19 September 2023\nCASE REPORT PEER REVIEWED | OPEN ACCESS   \nConclusion: The prolapsed mass was confirmed as an \nendocervical-type typical polypoid adenomyoma. We \nsuccessfully eradicated the tumor through hysteroscopy, \nleading to the restoration of normal cervical morphology, \ncorrection of anemia, and normalization of body \ntemperature. During the procedure, we directly observed \nthe pink mass, exhibiting cystic changes, encircling the \nhypertrophied cervix. Persistent exposure to high-dose \nestrogen may potentially play a significant role in the \ndevelopment of polypoid adenomyoma.\nKeywords: Adenomyoma, Adolescent, Hysteroscopy, \nVaginal bleeding\nHow to cite this article\nHe H, Rouzi N, Chen T-A, Hou Y, Zhang L, Zhao \nG-Z. A case report of a rare endocervical-type typical \npolypoid adenomyoma. J Case Rep Images Oncology \n2023;9(2):19–23.\nArticle ID: 100126Z10HH2023\n*********\ndoi: 10.5348/100126Z10HH2023CR\nINTRODUCTION\nUterine polypoid adenomyomas, also known as \nadenomyomyoid polyps, are relatively rare in the \nclinic. The pathological features of uterine polypoid \nadenomyomas are that the polyp gland is surrounded \nby a large number of smooth muscle fibers rather than \nthe normal polyp stroma. According to the type of \nglandular epithelium, it can be divided into two types: \ntypical polypoid adenomyoma (TPA) and atypical \npolypoid adenomyoma (APA) [1, 2]. Although uterine \npolypoid adenomyoma has been referred widely in \nthe literature, typical polypoid adenomyoma in the #Hua He and Nuermanguli Rouzi contributed equally to this work.\n\nJournal of Case Reports and Images in Oncology, Volume 9, Issue 2, 2023; Pages 19–23. ISSN: 2582-1318\nJ Case Rep Images Oncology 2023;9(2):19–23. \nwww.ijcrioncology.com\nHe et al. 20\ncervix, containing endometrioid-type glands, has been \noccasionally mentioned and discussed previously [3–\n5]. In addition, due to its uncommon occurrence, it is \noften underdiagnosed or misdiagnosed, thus delaying \ntreatment.\nIn this report, we present a unique case of a 15-year-old \nadolescent with recurrent irregular vaginal bleeding, who \nwas ultimately diagnosed with endocervical-type typical \npolypoid adenomyoma, in the absence of tamoxifen and \nhormone therapy. To the best of our knowledge, this is \nthe first case report of a patient in the youngest age of \nonset. We aim to share our clinical experience about \nendocervical-type typical polypoid adenomyoma and \nreview relevant publications to decrease the rates of \nmisdiagnosis and missed diagnosis.\nCASE REPORT\nA 15-year-old patient, gravida 0, para 0, had irregular \nvaginal bleeding, and the amount of menstruation was \nnormal, without dysmenorrhea. Six months before \nadmission, the patient was able to touch the vaginal \norifice mass (approximately 2 cm) during toileting. \nHowever, the prolapsed mass could only be returned in \nthe supine position or standing position. In addition, \nafter forced defecation, she had irregular vaginal \nbleeding that lasts for about two days and loses fresh \nblood similar in volume to those as during common \nmenstruation. On the day before admission, symptoms \nwere exacerbated and the prolapsed mass could not \nbe returned after toileting, with abnormal increments \nof vaginal bleeding and elevated body temperature \n(38.5°C). By admission, she had very low hemoglobin \nlevel (57 g/L) due to the blood-loss anemia. The vaginal \nmass was pedunculated and composed of two parts. \nOne part appeared light pink and was located on the \nsuperior side, while the other part exhibited a purplish-\nblack color with an unpleasant odor, which may be the \nprimary cause of the fever.\nTransabdominal ultrasound revealed that the \nendometrial bilayer had an approximate thickness of 23 \nmm, exhibiting a heterogeneous echo with small cyst-\nlike echogenicity. Furthermore, the solid mass displayed \nan inhomogeneous hypoechogenicity, with abundant \nblood flow signals observed internally (Figure 1A). The \nmass consisted of multiple cystic areas measuring 87×31 \nmm (Figure 1B) and could be traced from the cervical \ntube to the body surface, suggesting a possible origin \nfrom the endometrium. However, determining the exact \nderivation of the mass proved challenging. Consequently, \nwe conducted further examination of the lesion using \nmagnetic resonance imaging (MRI).\nAn MRI examination revealed evident thickening of \nthe endometrium, along with a well-defined pedunculated \nmass exhibiting inhomogeneous intensity and protruding \ninto the vaginal orifice. Sagittal T2-weighted images \ndisplayed a cordlike hyperintensity, possibly originating \nfrom the cervix or uterine cavity, connecting irregular \nand perplexing clump signals that entirely occupied the \nlower reproductive tract. Within the vaginal canal, the \nprolapsed mass divided into two parts, each displaying \nmultiple cystic hyperintensities surrounded by a grid-like \npattern of isointense signals (Figure 2A). Furthermore, \nthese hyperintense features on T2-weighted images \nremained unattenuated on fat-suppressed T2-weighted \nimaging (Figure 2B). Additionally, axial post-contrast \nT1-weighted imaging revealed T1-hyperintense signals, \nindicating the presence of vascularity (Figure 3A). \nNotably, the uterine cavity exhibited hyperproliferative \nendometrium without a distinct demarcation line between \nthe mass and the endometrium. This enhancement was \nobserved on coronal post-contrast T1-weighted imaging \n(Figure 3B). To establish their relationship, hysteroscopy \nwas performed.\nThe patient underwent hysteroscopy in the lithotomy \nposition. We directly observed that the stalk of the \npink mass, performing cystic changes, grew around \nthe hypertrophy cervix. Due to the presence of tissue \nfilling the cervical ostium, accessing the uterine cavity \nduring hysteroscopy was challenging prior to the \nsurgery. The mass was carefully pulled caudally and \nrotated using tissue forceps. After removing major \ntissue, the remained mass was completely cut away \nalong the peduncle through hysteroscopy, with the \ncervical morphology returning to normal. Grossly, \nthe pedunculated mass presented florid appearance, \nwith a rough surface measuring approximately 80×36 \nmm. Then, images of uterine cavity were obtained \nunder the hysteroscopy, while the endometrium was \nextensively thickened and looked rough with small \ncystic changes. The hysteroscopic finding verified that \nthe mass indeed derived from cervix rather than uterine \ncavity. To further identify the vaginal mass, pathologic \nbiopsy was conducted. Hematoxylin and eosin (HE) \nstaining demonstrated that the tissue was overlaid by \nan endometrial epithelial monolayer, with unapparent \ngland hyperplasia and partial gland expansion. The \nstroma is composed of fibrous connective tissue and a \nfew smooth muscle fibers (Figure 4).\nFigure 1: Transabdominal ultrasonography from the uterine to \nthe body surface. (A) Ultrasonography showed the endometrium \nwas obviously thickened and the abundant blood flow signals \nderived from cervix (arrow indicates the mass). (B) The part of \nthe mass revealed a complex heterogeneous echogenicity (arrow \nindicates the cyst-like echogenicity).\n\nJournal of Case Reports and Images in Oncology, Volume 9, Issue 2, 2023; Pages 19–23. ISSN: 2582-1318\nJ Case Rep Images Oncology 2023;9(2):19–23. \nwww.ijcrioncology.com\nHe et al. 21\nDISCUSSION\nTypical polypoid adenomyoma (TPA) is a benign \nspace-occupying lesion originating from the uterine \ncavity, with extremely rare cases reported in the uterine \ncervix, making them infrequently encountered in clinical \npractice [6]. Generally, the symptoms of TPA and APA, \ninclusive of menorrhagia, vaginal mass, anemia, and \nabnormal bleeding are nonspecific. Furthermore, there \nis no significant disparity in the diagnostic performance \nof ultrasound and MRI [7, 8]. Therefore, distinguishing \nbetween TPA and APA poses a challenge. Sajjad et al. \npointed out that prolapsed uterine tissue, with blood-\ncontaining cystic spaces on MRI, should be considered \nas typical polypoid adenomyoma [9]. However, this \nviewpoint lacks persuasiveness. The histopathological \nstaining still remains the most effective method for \ndistinguishing between TPA and APA. Empirically, \nAPA exhibited characteristic lesions under microscopic \nobservation. Hyperplasia disordered glands frequently \nshow branch-like or lobular arrangement, which are \nsimilar to endometrial complex atypical hyperplasia \n[10]. Concisely, glandular epithelial cells have mild \nand moderate atypicality, accompanied by squamous \nmetaplasia. The gland may appear as differentiated \nadenocarcinoma-like structures in focal lesions. Finally, \nthe prolapsed mass was confirmed as TPA through \nhistopathological staining. The treatment of polypoid \nadenomyomas includes curettage, hysterectomy, \nand hysteroscopic resection. Curettage is an invisible \noperation that is hampered by its residual lesions and \nhigh recurrence rate in the clinic. Although hysterectomy \ncan completely eradicate the lesion, it has a large trauma \nrange and serious complications, which is just suitable \nfor postmenopausal or no fertility intention patients \nwith atypical polypoid adenomyomas. Hysteroscopy is a \nmultifunctional tool that integrates imaging and treatment \ncapabilities. It enables visible lesion removal, providing \nadvantages such as accurate localization, complete \nresection, minimal tissue trauma, and a low recurrence \nrate. These results underscore the irreplaceable value of \nhysteroscopy in the diagnosis of location for polypoid \nadenomyomas when compared to ultrasound and \nMRI. Consequently, hysteroscopic resection should be \nrecommended as the preferred treatment modality for \nyoung patients seeking fertility preservation [11].\nThe pathogenesis and mechanism of TPA have \nnot received extensive research attention to date. \nHowever, there are scholars who propose that polypoid \nadenomyomas could potentially arise from endometrial \nstromal progenitor cells. These cells possess the ability \nto differentiate into smooth muscle cells, a process \nthat is potentially triggered by prolonged exposure to \nestrogen [12]. Perimenopausal women experience a \ndecline in estrogen levels, which has significant effects \non the hypothalamic feeding centers and neurons. These \neffects result in a range of metabolic changes, including \na decrease in metabolic rate, an increase in appetite, \nFigure 2: Magnetic resonance sagittal images. (A) Sagittal \nT2-weighted image of the patient. A large, well-defined, \nheterogeneous signal derived from cervical ostium, protruding \ninto the vaginal orifice. Within the vaginal canal, the prolapsed \nmass divided into two parts, each displaying multiple cystic \nhyperintensities surrounded by a grid-like pattern of isointense \nsignals. (B) Sagittal T2-fat-suppression sequences. Compared \nto sagittal T2-weighted image, the signal of the mass was little \naltered (arrow indicates the mass).\nFigure 3: Magnetic resonance axial and coronal images. (A) \nAxial post-contrast T1-weighted image of the patient. The mass \nexhibited grid-like hyperintense signals, enveloping multiple \nhypointense signals. Meanwhile, the signal of vaginal wall was \nalso enhanced. (B) Coronal post-contrast T1-weighted image \nof the patient. The myometrium, endometrium, and the mass \nshowed hyperintense signals. There is no obvious demarcation \nline between the mass and endometrium (arrow indicates the \nmass).\nFigure 4: Photomicrograph of the mass section. The tissue \nwas overlaid by an endometrial epithelial monolayer and \nendometrial-like glands were surrounded by stroma cells and \nsmooth muscle cells (hematoxylin and eosin staining, 100×).\n\nJournal of Case Reports and Images in Oncology, Volume 9, Issue 2, 2023; Pages 19–23. ISSN: 2582-1318\nJ Case Rep Images Oncology 2023;9(2):19–23. \nwww.ijcrioncology.com\nHe et al. 22\nand the accumulation of fat in visceral organs. These \nalterations can potentially lead to increased estrogen \nsynthesis and disturbances in glucose-lipid metabolism. \nMoreover, the decline in ovarian function, coupled \nwith factors such as obesity-induced insulin resistance, \ncan contribute to ovulation disorders. Consequently, \nthese factors contribute to a sustained single estrogen \naction on the endometrium [13]. For postmenopausal \nwomen, several cases reported that patients diagnosed \nwith adenomyomyoid polyps received treatment with \ntamoxifen for breast cancer [14, 15]. Tamoxifen is a \nclass of agents known as selective estrogen receptor \nmodulators (SERMs), which have both antiestrogen \neffects and estrogen-like effects on different tissues and \norgans [14]. In contrast to the antagonistic estrogen \neffect on the mammary gland, tamoxifen shows estrogen-\nlike action on female reproductive organs. Therefore, \ntamoxifen may cause benign or malignant lesions, \nsuch as endometrial hyperplasia, endometrial polyps, \nendometrial cancer and uterine sarcoma. Adolescents \nwho are within 2–3 years of their first menstruation \n(postmenarche) experience endometrial stimulation \ndue to the continuous production of estrogen without \nthe counterbalancing effect of progesterone. This occurs \nbecause their hypothalamic-pituitary-ovarian (HPO) axis \nis still immature [16]. Consequently, adolescent girls are \nsusceptible to dysfunction caused by excessive mental \nstress, malnutrition, and anxiety, which can disrupt the \nregulation of the HPO axis and result in vaginal bleeding. \nTherefore, when formulating treatment plans, it is crucial \nto consider individualized and effective approaches that \nprioritize safety, fertility needs, and psychological and \nsocial ethical factors following surgery. Wiktor et al. \nreported a woman incidentally diagnosed with polypoid \nadenomyoma who received prolonged combination \ntherapy for irregular, heavy abnormal uterine bleeding \nfrom the age of 15 [17]. Hence, we speculated that \npersistent high-dose estrogen may be an important factor \nin inducing polypoid adenomyoma. This conjecture can \nalso be evidenced by the fact that the patient had abnormal \nendometrial thickening, with biopsy-proven endometrial \nhyperplasia and small foci of complex hyperplasia.\nCONCLUSION\nWe described one case in which an adolescent \nsuffered from a huge vaginal mass that was confirmed as \nan endocervical-type typical polypoid adenomyoma. We \nsuccessfully eradicated the tumor, and directly observed \nthe pink mass, exhibiting cystic changes, encircling the \nhypertrophied cervix through hysteroscopy. We surmise \nthat persistent exposure to high-dose estrogen may \npotentially play a significant role in the development of \npolypoid adenomyoma. As the environment changes \nand social stresses increase, the incidence of polypoid \nadenomyoma may present a growing and younger trend. \nConsequently, it is crucial for gynecologists to heighten \ntheir vigilance in this area to ensure prompt diagnosis \nand treatment for affected patients.\nREFERENCES\n1. 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Pathol Res Pract 2020;216(7):152998.\n*********\nAuthor Contributions\nHua He – Conception of the work, Design of the work, \nDrafting the work, Revising the work critically for \nimportant intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nNuermanguli Rouzi – Conception of the work, Design of \nthe work, Drafting the work, Revising the work critically \nfor important intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nTing-An Chen – Acquisition of data, Analysis of data, \nRevising the work critically for important intellectual \ncontent, Final approval of the version to be published, \nAgree to be accountable for all aspects of the work in \nensuring that questions related to the accuracy or integrity \nof any part of the work are appropriately investigated and \nresolved\nYu Hou – Acquisition of data, Revising the work critically \nfor important intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nLing Zhang – Acquisition of data, Revising the work \ncritically for important intellectual content, Final approval \nof the version to be published, Agree to be accountable for \nall aspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nGuang-Zong Zhao – Conception of the work, Analysis of \ndata, Interpretation of data, Drafting the work, Revising \nthe work critically for important intellectual content, \nFinal approval of the version to be published, Agree to be \naccountable for all aspects of the work in ensuring that \nquestions related to the accuracy or integrity of any part \nof the work are appropriately investigated and resolved\nGuarantor of Submission\nThe corresponding author is the guarantor of submission.\nSource of Support\nNone.\nConsent Statement\nWritten informed consent was obtained from the patient \nfor publication of this article.\nConflict of Interest\nAuthors declare no conflict of interest.\nData Availability\nAll relevant data are within the paper and its Supporting \nInformation files.\nCopyright\n© 2023 Hua He et al. 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