{"paper_id":"53df44aa-e6c4-4b16-b58c-f03c1f68fc62","body_text":"Obstetrics and \nGynaecology Cases - Reviews\nVidal et al. Obstet Gynecol Cases Rev 2018, 5:121\nVolume 5 | Issue 2\nDOI: 10.23937/2377-9004/1410121\nISSN: 2377-9004\nOpen Access\nCitation: Vidal IS, Soares LS, Dutra CA, Jovino MJ, Gonçalves AK, et al. (2018) Adenomyosis in an \n18-Year-Old Adolescent: A Case Report. Obstet Gynecol Cases Rev 5:121. doi.org/10.23937/2377-\n9004/1410121\nReceived: February 25, 2018: Accepted: March 29, 2018: Published: March 31, 2018\nCopyright: © 2018 Vidal IS, et al. This is an open-access article distributed under the terms of the \nCreative Commons Attribution License, which permits unrestricted use, distribution, and reproduction \nin any medium, provided the original author and source are credited.\nVidal et al. Obstet Gynecol Cases Rev 2018, 5:121\n• Page 1 of 4 •\nAdenomyosis in an 18-Year-Old Adolescent: A Case Report\nIaponira da Silva Vidal 1*, Lilian Santos Soares 1, Cássio Aurélio Dutra 2, Mara Juliane \nJovino2, Ana Katherine Gonçalves3 and Ricardo Ney Cobucci4\nuteri and often associated with the diffuse subtype, and \nin a juvenile form seen in nulliparous women between \n13 and 20 years of age, for which the true incidence \nis not known, since few cases have been reported. Al-\nthough adenomyosis seems very rare before age 20, a \ncystic form has mainly been reported in young women \n[3,4].\nA variant of adenomyosis that seems specific to \nyoung women is called myometrial cystic adenomyosis, \nin which young patients present with nonresponsive \nsevere dysmenorrhea. Adenomyosis was commonly \nthought to be a condition confined to adulthood, with \nlimited clinical cases in adolescents, but can be a cause \nof primary dysmenorrhea in this population. Howev -\ner, further research is needed in adolescent girls, par-\nticularly those whose pain is refractory to treatment \nwith non-steroidal anti-inflammatories (NSAID) and/or \ncombined oral contraceptives (COC) pills, to determine \nwhether some cases of primary dysmenorrhea may ac-\ntually be early stages of adenomyosis [5,6].\nWe report a case of primary dysmenorrhea in an \nadolescent with magnetic resonance imaging (MRI) re -\nvealing adenomyosis.\nCase Report\nAn 18-year-old nulliparous woman presented with \na history of severe and worsening dysmenorrhea with \ncramps and irregular menstrual cycle with excessive \n*Corresponding author: Iaponira da Silva Vidal, MD, Federal University of Rio Grande do Norte (UFRN), Nilo Peçanha Av., \n259, Natal-RN, 59012-310, Brazil, E-mail: iaponiravidal277@gmail.com\nAbstract\nAdenomyosis is a rare cause of chronic pelvic pain or se-\nvere dysmenorrhea that presents in the adolescent popula-\ntion. Here we describe an 18-year-old nulliparous woman \nwho presented with a history of severe and worsening dys-\nmenorrhea with cramps and increased menstrual flow since \nthe menarche occurred 4-years-ago. A magnetic resonance \nimaging (MRI) described a poorly defined junctional zone of \nthe endometrium, suggestive of adenomyosis, associated \nwith the discrete heterogeneity of the adjacent myometri-\num. The patient underwent a course of GnRH agonists for \n6 months, followed by continuous combined oral contra-\nceptives (COC) pills with pain resolution. In this population, \nfertility preservation is a goal and therefore initial therapy \nfor focal adenomyosis involves hormonal suppression with \nCOC.\nKeywords\nAdenomyosis, Pelvic pain, Dysmenorrhea, Adolescent\n1Federal University of Rio Grande do Norte (UFRN), Brazil\n2Potiguar University, Brazil\n3Department of Gynecology and Obstetrics, Federal University of Rio Grande do Norte (UFRN), Brazil\n4Department of Gynecology and Obstetrics, Potiguar University, Brazil\nCASe RepoRt\nCheck for\nupdates\nIntroduction\nAdenomyosis is a benign condition of the uterus de-\nfined by the presence of endometrial glands and stroma \n> 2.5 mm in depth in the myometrium and a variable \ndegree of adjacent myometrial hyperplasia, causing \nglobular and cystic enlargement of the myometrium, \nwith some cysts filled with extravasated, hemolyzed red \nblood cells, and siderophages [1,2].\nCystic adenomyosis can be present in both an adult \nform, which is present in 5-7% of hysterectomy fibroid \n\nISSN: 2377-9004\nDOI: 10.23937/2377-9004/1410121\n• Page 2 of 4 •\nVidal et al. Obstet Gynecol Cases Rev 2018, 5:121\nal-myometrial interface, such as that caused by sur-\ngery or pregnancy may create a predisposition towards \nendometrial invasion of myometrium, and that local \nhyperestrogenism or dysperistalsis facilitates the pro-\ncess. However, evidence of adenomyosis early in repro-\nductive life and in the absence of previous surgery or \npregnancy suggests that invasion of endometrial tissue \nis not necessarily mediated mechanically; it may occur \nfrom endometrial-myometrial dysfunction [8]. Evidence \nshows that endometriosis and adenomyosis have in \ncommon an endometrial dysfunction involving both eu-\ntopic and heterotopic endometrium. Although anoma-\nlies are not identical, they share the common feature \nof leading to increased invasiveness. In both conditions, \nthere is also a reaction of the inner myometrium that, \nalthough more pronounced in the case of adenomyosis, \nis nonetheless also present in endometriosis [6].\nThe symptoms of adenomyosis are variable and the \nproportion of women with adenomyosis who have ab-\nnormal uterine bleeding and/or painful symptoms, as \nwas reported by the adolescent patient, is unclear. A \nconsensus on the association between dysmenorrhea \nand adenomyosis is lacking, with some studies support-\ning this relationship, and others finding only a weak as-\nsociation [8].\nAdenomyosis is a rare cause of chronic pelvic pain \nor severe dysmenorrhea in the adolescent population. \nFor patients who have symptoms of dysmenorrhea or \nchronic pelvic pain refractory to NSAIDs or COC therapy, \nadditional investigation with imaging may reveal this di-\nagnosis as happened in this case. MRI is more indicated \nthan TVUS, since it has shown potential superiority over \nTVUS in some situations, and because defined diagnos -\ntic criteria (Table 1) have made inter-observer variability \nbleeding since the menarche occurred 4-years-ago, \nthese were insufficiently relieved by NSAID and COC. \nShe reported having started using COC two-years-ago \nwhen her sex life began, and she never underwent sur-\ngery, had an abortion or suffer any trauma. Her family \nhistory is unremarkable.\nPelvic examination revealed a normal vagina, vulva \nand adnexae and a normal-sized anteverted uterus. \nTransvaginal ultrasound (TVUS) demonstrated a uterus \nof normal volume and irregular contours, homogeneous \nmyometrium and endometrium and without change of \nthickness. Ovaries were enlarged at the expense of mul-\ntiple follicles. A follow-up MRI obtained 1 month later \ndescribed a poorly defined junctional zone of the endo-\nmetrium, suggestive of myometrial cystic adenomyosis, \nassociated with the discrete heterogeneity of the adja -\ncent myometrium and, in addition, a small increase in \nthe thickness of the posterior uterine wall (Figure 1).\nThe patient underwent a course of GnRH agonists \nfor 6 months, followed by continuous COCs with pain \nresolution.\nDiscussion\nThis case demonstrates that adenomyosis is a rare \nbut possible cause of dysmenorrhea and pelvic pain in \nadolescent patients, despite the disease being typically \npresent in adult women in the third or fourth decade of \nlife. Although endometriosis may be the most common \ncause of secondary dysmenorrhea in younger patients, \nadenomyosis should remain in the differential diagnosis \n[7].\nPrevious pathogenetic theories for adenomyosis \nhave proposed that traumatization of the endometri-\nFigure 1: An asymmetric widening of the posterior junctional zone is shown in magnetic resonance imaging (MRI), with ill-de-\nfined border, presenting an elliptical shape and bright foci, with no mass effect on the endometrial cavity (arrows).\n\nISSN: 2377-9004\nDOI: 10.23937/2377-9004/1410121\n• Page 3 of 4 •\nVidal et al. Obstet Gynecol Cases Rev 2018, 5:121\nAn example of drugs with higher costs and more trou-\nblesome side effects are GnRH agonists. They are very \neffective against adenomyosis related pain, and there -\nby also contribute to the occurrence of many successful \npregnancies and deliveries, however their use is associ-\nated with frequent and intolerable hypoestrogenic side \neffects, including vasomotor syndrome, genital atrophy \nand mood instability, and the use of these drugs should \nbe for a short period of time among adolescents who \ndo not improve their symptoms with continuous use of \nCOC, which is what happened in our case. GnRH ago-\nnists also have a negative impact on bone health and a \npossible negative influence on cardiovascular health [2].\nSurgical management is necessary in cases where \ndysmenorrhea is medication resistant. Some authors \nargued in favor of the laparoscopic approach. Other \nsurgical approaches have been proposed such as abla -\ntion after insertion of a radiofrequency needle under \nultrasound guidance, the use of a single-incision with \nmonopolar cautery or the use of robotic surgery. In the \ncase of a focal lesion or adenomyotic cyst not involving \nthe endometrial cavity, safe surgical resection is possi -\nble [4].\nConclusion\nIn conclusion, this case draws attention to a little \nknown but not uncommon cause for both primary and \nsecondary dysmenorrhea, which can affect young nul -\nliparous women. MRI may be useful in establishing a \ndiagnosis even when TVUS is normal. In this population, \nfertility preservation is the primary goal and, therefore \ninitial therapy for focal adenomyosis involves hormonal \nsuppression with COC. Further research is needed due \nto limited reports in the literature regarding manage -\nment of adenomyosis in the adolescent.\nConflicts of Interest\nThe authors declare no potential conflicts of interest \nand no sources of support.\nAcknowledgements\nAll authors equal contributed to the design and im-\nplementation of the research, to the analysis of the case \nand to the writing of the manuscript.\nReferences\n1. Naftalin J, Hoo H, Nunes N, Holland T, Mavrelos D, et al. \n(2016) Association between ultrasound features of adeno -\nmyosis and severity of menstrual pain. Ultrasound Obstet \nGynecol 47: 779-783.\n2. Tsui KH, Lee WL, Chen CY, Sheu BC, Yen MS, et al. (2014) \nless significant [9]. Although adenomyosis has tradition-\nally been confirmed by histopathologic diagnosis from \nsurgery, MRI can allow for accurate diagnosis of adeno-\nmyosis in most cases. Several studies have demonstrat-\ned sensitivity rates ranging from 70 to 88% and specific-\nity as high as 91%. This is significantly better sensitivity \nand specificity of MRI compared with TVUS in the diag -\nnosis of these abnormalities [9,10].\nCareful review of the literature reveals very few case \nreports, case series or small cohorts addressing adeno -\nmyosis or adenomyotic cysts presenting in the female \nadolescent [3, 4,7,11,12]. A variety of imaging modali -\nties were utilized in diagnosis. All cases presented with \nsevere dysmenorrhea or pelvic pain, as reported by the \npatient in this case. In most cases, as in our patient and \nin the cases of two adolescents reported by Itam, et al. \n[7], the diagnosis was only made when patients did not \nrespond to initial empirical treatment including the use \nof COC and NSAID.\nMedical treatments for adenomyosis always follow \nthe principles of the management of endometriosis, \nwhich are usually aimed at reducing the production of \nendogenous estrogen or inducing endometrial differen-\ntiation with progestins. Clinical evidence points to the \nclear and deleterious effect of uninterrupted ovulato -\nry cycles on the development and persistence of ade-\nnomyosis. The objectives of medical treatment are the \ninhibition of ovulation, abolition of menstruation, and \nachievement of a stable steroid hormone milieu [2].\nBoth medical and surgical treatments offer oppor -\ntunities for disease stabilization and regression. Initial \ntherapy for focal adenomyosis involves hormonal sup -\npression with COC, especially for the female adolescent \nin whom preserving fertility is paramount. Adolescents \nmay represent a subset of patients with adenomyosis \nwhose lesions are completely treatable with hormonal \ntherapy [10].\nFor adenomyosis, the goal of therapy is important, \nand can include symptom relief and possibly increased \nfertility. The therapeutic goal of medical treatment is not \nlesion resorption: Lesions survive any drug, at any dose, \nfor any period of use, and come back after treatment dis-\ncontinuation. Therefore, treatment should be tailored \nto the specific symptom or the special request of the in-\ndividual patient. The results from systematic literature \nreviews have consistently demonstrated that, if amenor-\nrhea is obtained, there are no statistical differences be-\ntween the various available drugs in terms of pain relief, \nbut tolerability, side effects, and costs vary widely [2].\nTable 1: Diagnostic criteria for adenomyosis in magnetic resonance imaging (MRI).\n1- Thickening of the junctional zone, with a thickness exceeding 12 mm\n2- Large, rand asymmetric uterus, with a maximum junctional zone thickness of at least 12 mm and punctate high-intensity myo-\nmetrial foci\n3- Bright foci on T2-weighted images, which represent foci of heterotopic endometrial tissue, cystic dilatation of endometrial \nglands or hemorrhagic foci\n\nISSN: 2377-9004\nDOI: 10.23937/2377-9004/1410121\n• Page 4 of 4 •\nVidal et al. Obstet Gynecol Cases Rev 2018, 5:121\n8. Pinzauti S, Lazzeri L, Tosti C, Centini G, Orlandini C, et al. \n(2015) Transvaginal sonographic features of diffuse ade-\nnomyosis in 18-30-year-old nulligravid women without en-\ndometriosis: Association with symptons. Ultrasound Obstet \nGynecol 46: 730-736.\n9. Agostinho L, Cruz R, Osório F, Alves J, Setúbal A, et al. \n(2017) MRI for adenomyosis: A pictorial review. Insights \nImaging 8: 549-556.\n10. Dietrich JE (2010) An update on adenomyosis in the ado-\nlescent. Curr Opin Obstet Gynecol 22: 388-392.\n11. Mansouri R, Santos XM, Bercaw-Pratt JL, Dietrich JE \n(2015) Regression of Adenomyosis on Magnetic Reso-\nnance Imaging after a Course of Hormonal Suppression in \nAdolescents: A Case Series. J Pediatr Adolesc Gynecol 28: \n437-440.\n12. Manta L, Suciu N, Constantin A, Toader O, Popa F (2016) \nFocal adenomyosis (intramural endometriotic cyst) in a \nvery young patient - differential diagnosis with uterine fibro-\nmatosis. J Med Life 9: 180-182.\nMedical treatment for adenomyosis and/or adenomyoma. \nTaiwan J Obstet Gynecol 53: 459-465.\n3. Ball E, Ganji M, Janik G, Koh C (2009) Laparoscopic re-\nsection of cystic adenomyosis in a teenager with arcurate \nuterus. Gynecol Surg 6: 367-370.\n4. Brosens I, Gordts S, Habiba M, Benagiano G (2015) Uter-\nine Cystic Adenomyosis: A disease of younger women. J \nPediatr Adolesc Gynecol 28: 420-426.\n5. Iacovides S, Avidon I, Baker FC (2015) What we know \nabout primary dysmenorrhea today: A critical review. Hum \nReprod Update 21: 762-778.\n6. Benagiano G, Brosens I, Habiba M (2015) Adenomyosis: \nA life-cycle approach. Reprod Biomed Online 30: 220-232.\n7. Itam SP, Ayensu-Coker L, Sanchez J, Zurawin R, Dietrich \nJE (2009) Adenomyosis in adolescente population: A case \nreport and review of the literature. J Pediatr Adolesc Gyne-\ncol 22: e146-e147.","source_license":"CC0","license_restricted":false}