{"paper_id":"08025446-3b8a-43aa-ac65-805baab63729","body_text":"Obstetrics and \nGynaecology Cases - Reviews\nAfaneh et al. Obstet Gynecol Cases Rev 2020, 7:162\nVolume 7 | Issue 2\nDOI: 10.23937/2377-9004/1410162\nISSN: 2377-9004\nOpen Access\nAfaneh et al. Obstet Gynecol Cases Rev 2020, 7:162\n• Page 1 of 3 •\nCitation: Afaneh H, Moses K, Young O, Abuzeid M (2020) Surgical Management of a Juvenile Cys-\ntic Adenomyoma: A Unique Presentation of Adenomyosis. Obstet Gynecol Cases Rev 7:162. doi.\norg/10.23937/2377-9004/1410162\nAccepted: April 01, 2020: Published: April 03, 2020\nCopyright: © 2020 Afaneh H, 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.\nSurgical Management of a Juvenile Cystic Adenomyoma: A Unique \nPresentation of Adenomyosis\nHuda Afaneh, MD 1*, Karen Moses, MD 1, Omari Young, MD 2 and Mostafa Abuzeid, \nMD, FACOG, FRCOG3,4,5\npatients. In this case report, we focus on the surgical \nmanagement of a juvenile cystic adenomyoma and en-\ndometrioma within the uterine wall. The surgical tech-\nniques used to excise the cystic adenomyoma closely \nresemble the techniques used during a myomectomy \nalong with unique surgical challenges when considering \nthe presence of underlying adenomyosis.\nCase\nOur patient is a 16-year-old, gravida 0, with a his -\ntory of chronic pelvic pain and dysmenorrhea. Alter -\nnative etiology of her pain was ruled out including an \ninfectious, genitourinary and thorough gastrointestinal \nworkup. She did not attempt treatment with hormonal \ntherapy prior to surgery. Initial CT scan of abdomen/pel-\nvis performed prior to surgery suggested a cystic mass \nseparate from the endometrial cavity measuring 2.2 \ncm × 2.2 cm. Upon initial diagnostic laparoscopy, she \nwas found to have two small endometriotic implants on \nthe left ovary as well as an endometrioma of the uterus \ndiagnosed via needle aspiration. Pathologic evaluation \ndescribed the aspirated fluid as containing “hemosid -\nerin-laden macrophages and blood, with no evidence \nof malignancy”. She also had a hormonal intrauterine \n*Corresponding author:  Huda Afaneh, Clinical Instructor, Department of Obstetrics and Gynecology, Hurley Medical \nCenter, Michigan State University College of Human Medicine, Flint MI, USA\n1Clinical Instructor, Department of Obstetrics and Gynecology, Hurley Medical Center, Michigan State University College \nof Human Medicine, USA\n2Assistant Professor, Department of Obstetrics and Gynecology, IVF Michigan Rochester Hills and Flint PC, Hurley Medical \nCenter, Michigan State University College of Human Medicine, USA\n3Director, Reproductive Endocrinology and Infertility Hurley Medical Center, USA \n4Professor, Michigan State University College of Human Medicine, USA\n5Practice and Medical Director, IVF Michigan Rochester Hills and Flint PC, USA\nCAse RepORt\nCheck for\nupdates\nIntroduction\nEndometriosis is characterized by the presence of \nendometrial glands and stroma in ectopic locations \nwithin the peritoneal cavity. More commonly, the le-\nsions can be found on areas such as the cul-de-sac and \nother pelvic parietal surfaces. Endometrial glands and \nstroma infiltrating the myometrium characterizes Ad-\nenomyosis [1,2]. Adenomyosis is usually characterized \nby diffuse foci of endometrial glands scattered through-\nout the myometrium. Meanwhile, a less common type \nof adenomyosis, in which the lesions are focal or local -\nized in nature is referred to as anadenomyoma [3, 4]. \nAn adenomyoma is described as a circumscribed nod -\nule of hypertrophic and distorted endometrium within \nthe myometrium [3,5]. Only a few cases in the literature \nhave been reported with histologically confirmed endo-\nmetrioma within the myometrium of the uterus [4, 6]. \nSuch patients may primarily present with chronic pelvic \npain among the wide spectrum of clinical syndromes in \npatients with endometriosis. On the other hand, there \nare a few case reports in the literature describing juve-\nnile cystic adenomyoma as a unique uterine pathology \n[4,6,7]. Juvenile cystic adenomyoma is basically an ade-\nnomyoma with a cystic component that occurs in young \n\nISSN: 2377-9004\nDOI: 10.23937/2377-9004/1410162\n• Page 2 of 3 •\nAfaneh et al. Obstet Gynecol Cases Rev 2020, 7:162\n \nFigure 1: Ultrasound view of juvenile cystic adenomyoma.\n \nFigure 2: MRI view of juvenile cystic adenomyoma.\n \nFigure 3: Chocolate-like material exiting from the juvenile \ncystic adenomyoma encased on the posterior aspect of \nuterus.\ndevice placed intraoperatively. About one month fol-\nlowing her surgery, her pain recurred. Repeat imag-\ning with transvaginal ultrasound scan (TVUS) and MRI \n(Figure 1 and Figure 2) suggested recurrence of her \nendometrioma. MRI suggested a clearly demarcated \napproximately 3 cm × 3 cm mass located within the \nmyometrium. Decision was made for a second sur-\ngery in conjunction with a Reproductive Endocrinol-\nogy and Infertility specialist. On second laparoscopy, \nthe uterine endometrioma was excised and tech-\nniques included the use of monopolar energy, sharp \nand blunt dissection (Figure 3 and Figure 4). Choco-\nlate-like material was noted coming out of the cavity \nof the endometrioma (Figure 3). The uterine cavity \nwas ultimately not entered (Figure 5). Repair of the \ndefect in the myometrium was closed in a two-layer \nclosure using 2-0 and 3-0 V-lock suture (Figure 6). The \n \nFigure 4: Excision of juvenile cystic adenomyoma.\n \nFigure 5: Adenomyoma cyst bed, following excision, with \nno evidence of entry of the endometrial cavity. \n\nISSN: 2377-9004\nDOI: 10.23937/2377-9004/1410162\n• Page 3 of 3 •\nAfaneh et al. Obstet Gynecol Cases Rev 2020, 7:162\n< 1 cm in diameter independent of the uterine lumen \nand covered by hypertrophic myometrium on diag-\nnostic images; and 3) associated with severe dysmen-\norrheal” [5 ]. Kriplani A, et al. (2011) described lapa-\nroscopic management of 4 patients of juvenile cystic \nadenomyoma and reviewed the other case reports \non the topic [3 ]. Our case report and supplementary \nvideo aims to increase awareness for such an unusu-\nal pathology and options for minimally invasive sur-\ngical management. In view of a report of 2 patients \nwho had rupture uterus at 32 and 37 week gestation, \nmeticulous uterine closure and avoid use of cautery \nshould be emphasized [9 ]. In addition, such patients \nshould be managed with maternal Fetal Medicine \nspecialist when pregnant with a plan for an elective \ncesarean section.\nReferences\n1. Pelvic Mass. In: Hoffman BL, Schorge JO, Bradshaw KD, \nHalvorson LM, Schaffer JI, et al. Williams Gynecology. (3 rd \nedn), McGrawHill, New York.\n2. Endometriosis. In: Jones Howard, Rock JA (2015) Te-\nLindes Operative Gynecology. (11\nth edn), Lippincott, Wil-\nliams & Wilkins, Philadelphia.\n3. Kriplani A, Mahey R, Agarwal N, Bhatla N, Yadav R, et al. \n(2011) Laparoscopic management of juvenile cystic adeno-\nmyoma: Four cases. JMIG 18: 343-348.\n4. Osama Zaghmout, Omar Abuzeid, John Hebert, Mostafa \nAbuzeid (2017) Endometrioma embedded within the myo-\nmetrium. American Journal of Obstetrics & Gynecology \n(AJOB) 34.\n5. Takeuchi H, Kitade M, Kikuchi I, Kumakiri J, Kuroda K, et \nal. (2010) Diagnosis, laparoscopic management, and his-\ntopathologic findings of juvenile cystic adenomyoma: A re-\nview of nine cases. Fertil Steril 94: 862-868.\n6. Abhishek Trehan (2014) Endometrioma contained within \nthe broad ligament. BMJ Case Rep.\n7. Younes G, Tulandi T (2018) Conservative surgery for ade-\nnomyosis and results: A systematic review. JMIG 25: 265-\n276.\n8. Tamura M, Fukaya T, Takaya R, Ip CW, Yajima A (1996) \nJuvenile adenomyotic cyst of the corpus uteri with dysmen-\norrhea. Tohoku J Exp Med 178: 339-344.\n9. Koo YJIK, Kwon YS (2011) Conservative surgical treatment \ncombined with GnRH agonist in symptomatic uterine ade-\nnomyosis. Pak J Med Sci 27: 365-370.\nduration of surgery was 90 minutes and the estimat-\ned blood loss was approximately 40 cc. The patient \nwas discharged home on postoperative day 1 and her \npost-operative period was uneventful. The pathology \nreport confirmed the presence of fragments of myo-\nmetrium and adenomyosis. Therefore, the diagnosis \nof juvenile cystic adenomyoma was considered as a \nprimary diagnosis. Post-operatively, the patient did \nhave relief of her pain. Six weeks later, repeat trans-\nvaginal ultrasound revealed no evidence of recur-\nrence of the juvenile cystic adenomyoma. She had a \nfollow-up appointment 8 months following her sec-\nond surgery, and she continues to have relief of her \nprevious symptoms.\nDiscussion\nJuvenile cystic adenomyoma of the uterus is a rare \nentity of focal adenomyoma that occurs in young pa-\ntients. It usually causes severe and debilitating pain. \nThe first report describing juvenile cystic adenomy-\noma was published by Tamura M, et al. in 1996 [8 ]. \nTakeuchi H, et al. (2010) indicated that 30 cases of \njuvenile cystic adenomyoma (including those in their \nseries) have been reported in the Japanese-language \npublications [5 ]. They suggested the following diag-\nnostic criteria: “1) age < 30 years; 2) Cystic lesion of \n \nFigure 6: Two-layer repair was carried 2-0 V-lock suture.","source_license":"CC0","license_restricted":false}