{"paper_id":"da43abbb-04e3-4b1b-a700-b3ce79289b3e","body_text":"~ 221 ~ \nInternational Journal of Clinical Obstetrics and Gynaecology 2021; 5(2): 221-223 \n \nISSN (P): 2522-6614 \nISSN (E): 2522-6622 \n© Gynaecology Journal \nwww.gynaecologyjournal.com \n2021; 5(2): 221-223 \nReceived: 25-01-2021 \nAccepted: 28-02-2021 \n \nDr. Yeshli Thakur \n3rd Year Resident, Sri Aurobindo \nInstitute of Medical Sciences \nIndore, Madhya Pradesh, India \n \nDr. Neeta Natu \nProfessor and Head, Sri Aurobindo \nInstitute of Medical Sciences \nIndore, Madhya Pradesh, India \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \n \nCorresponding Author: \nDr. Yeshli Thakur \n3rd Year Resident, Sri Aurobindo \nInstitute of Medical Sciences \nIndore, Madhya Pradesh, India \n \nAn unusual case of adenomyosis reported in Central \nIndia \n \nDr. Yeshli Thakur and Dr. Neeta Natu \n \nDOI: https://doi.org/10.33545/gynae.2021.v5.i2d.892 \n \nAbstract \nAdenomyosis is the presence of endometrial glands and stroma within the myometrium. A 40 years old \nnullipara presented with complaints of heavy bleeding during menses with passage of clots and white \ndischarge with pain i n abdomen more during menses for 3 years. On per abdomen examination a globular \nmass seen in lower abdomen below umbilical. It was about 20 weeks uterus size, firm, smooth with regular \nmargins. Ultrasound was done which suggested bulky uterus of 9.6x11.9x8 .5 cm, with heterogeneous \nmyometrium globular fundus with increased vascularity and indistinct endomyometrial junction. \nEndometrial thickness of 4mm and normal ovaries. Mild hydronephrosis on both sides. MRI done \nsuggested solid vascular mass lesion of 14. 1x12x5.2cm with smooth margin in posterior wall of uterus \nextending to anterior wall of fundus with multiple cystic degeneration. Patient was not desirous of child in \nfuture and wanted permanent cure. So total abdominal hysterectomy with bilateral DJ stent ing was done.  \nDiffuse Adenomyosis was eventually confirmed in histopathology report. \n \nKeywords: adenomyosis, myometrium, hysterectomy \n \nIntroduction  \nAdenomyosis is a condition that affects the uterus. In women with adenomyosis, the \nendometrial tissue (which typically lines the uterus) moves into the outer, muscular walls of the \nuterus. Some women may have no signs or symptoms of the condition. When present, features \nof the condition include heavy menstrual bleeding, painful menstrual periods, and  pelvic pain \nduring intercourse. Some women may also develop an adenomyoma, which is a mass or growth \nwithin the uterus. The underlying cause of the condition is currently unknown [1]. \nThe modern definition of adenomyosis was provided in 1972 by Bird who stated: “Adenomyosis \nmay be defined as the benign invasion of endometrium into the myometrium, producing a \ndiffusely enlarged uterus which microscopically exhibits ectopic non -neoplastic, endometrial \nglands and stroma surrounded by the hypertrophic and hyperplastic myometrium” [2]  \nIn simple words, Adenomyosis is the presence of endometrial glands and stroma within the \nmyometrium.3 \n70 to 80 % of women undergoing hysterectomy for adenomyosis are in their fourth and fifth \ndecade of life and are multiparous; several studies have reported a mean age over 50 years for \nwomen undergoing hysterectomy for adenomyosis [4-13]  \nA high percentage of women with adenomyosis are multiparous [8-10, 14, 15].  \n \nCase Report \nHere we report a case of unusual presentation of adenomyosis in a 40 -year nullipara with 20 -\nweek size uterus not associated with fibroid. \nA 40 years old nullipara presented with compl aints of heavy bleeding during menses with \npassage of clots and white discharge with pain in abdomen more during menses for 3 years. \nPatient also had lump in lower abdomen noticed 1 year back. Menstrual cycles were regular, \nbleeding continued for 8 - 10 day s. There were no urinary complaints. Patient was previously \ngiven Gonadotropin releasing hormone (GnRH) agonist but was not relieved. She never \nconceived and did not take any treatment for the same. There was no past surgical history. \nFamily history was insignificant.  \nOn examination patient was vitally stable and clinically pale. On per abdomen examination a \nglobular mass seen in lower abdomen below umbilical. It was about 20 weeks uterus size, firm, \nsmooth with regular margins, lower margin could not be reached, freely mobile transversely and  \n\n\nInternational Journal of Clinical Obstetrics and Gynaecology http://www.gynaecologyjournal.com \n~ 222 ~ \nlimited mobility upside down. Per speculum examination \nsuggestive of grossly healthy cervix, pulled up with minimal \nwhite discharge. On Per -vaginal examination same mass could \nbe palpated, mobile with movement of cervix. \nUltrasound was done which suggested bulky uterus of \n9.6x11.9x8.5 cm, with heterogeneous myometrium globular \nfundus with increased vascularity and indistinct endomyometrial \njunction. Endometrial thickness of 4mm and normal ovaries. \nMild hydronephrosis on both sides. \nMRI done suggested solid vascular mass lesion of \n14.1x12x5.2cm with smooth margin in posterior wall of uterus \nextending to anterior wall of fundus with multiple cystic \ndegeneration suspecting Adenomyoma.  \nPatient was not desirous of child in future and wanted permanent \ncure. So total abdominal hysterectomy with bilateral DJ stenting \nwas done. Ovaries were healthy and preserved. Uterus was \n22x16x16cm, symmetrically enlarged with 1.5kg weight. \nOn cut section no localized mass could be seen, my ohyerplasia \nobserved in walls with cystic degeneration and indistinct \nendomyometrial junction. \nDiffuse Adenomyosis was eventually confirmed in \nhistopathology report. \n \nDiscussion  \nThis case report brings light to the fact that a lot more needs to \nbe discovered and studied about adenomyosis. Adenomyosis can \npresent in unique ways. As the causation of adenomyosis is \nlinked with breach in endomyometrial junction due to trauma \nduring delivery or any intervention, which results in \ninternalization of endometrial g lands. In this case no cause for \nbreach in endomyometrial junction was observed. It favours \ntheory of genetic predisposition. According to Parrot et.al nerve \ngrowth factor -alpha, preadipocyte factor -1, and insulin -like \ngrowth factor-2 genes may play an imp ortant role in regulating \ndifferentiation and development of the myometrium, these data \nsuggest that adenomyosis may be caused primarily by defects in \nthe formation of the myometrium [16].  \nThis case is an exception to the fact that adenomyosis uterus \ndoes not grow beyond 14weeks size. Over 14 weeks growth is \nassociated with fibroid in about 60% cases. In this case uterus \nsymmetrically enlarged to 20 weeks size with 1.5kgs weight. \nCut section shows granular pattern with no circumscribed area \nwith capsule, d enying coexistence of fibroid. This case is of \nsevere adenomyosis according to Bird et al grading and grade III \nby Molitor's criteria  [17, 18] Based on depth it is Deep \nAdenomyosis. \nThe accuracy of investigation is decreased in women with \ncoexisting fibroid or focal adenomyosis. It may be misdiagnosed \nin ultrasound as it may be taken as multiple leiomyoma or \nendometrial thickening. Pathological confirmation can be made \nat the time of hysterectomy. Histopathology remains \nconfirmatory for diagnosis. \nAdenomyosis is less commonly associated with infertility. Its \nrole in infertility is still debatable but frequency of infertility has \nnot been assessed. It has been seen to be associated with \npregnancy which favors invagination of nasal endometrium into \nmyometrium.  \nAdenomyosis unlike endometriosis, is refractory to hormone \ntreatment. GnRH agonists are efficient in reducing the \nadenomyosis uterus size and may facilitate fertility. Recurrence \nof adenomyosis is common after discontinuation of GnRH \nagonists. Conservati ve surgical treatment is available but \nhysterectomy holds the principal diagnostic, therapeutic and \ndefinitive treatment for deep adenomyosis. \nConclusion  \nThis case draws attention to rare presentation of adenomyosis. \nAssociation with infertility, growth b eyond 14weeks uterus size \nwithout association with fibroid and newer theory of causation. \nMRI is superior to ultrasound in diagnosis of adenomyosis. \nGnRH agonists have limited role in providing relief in deep \nadenomyosis. Hysterectomy is still the definiti ve treatment of \nchoice in such a case. \n \nReferences \n1. https://rarediseases.info.nih.gov/diseases/8156/adenomyosis\n#:~:text=Summary,-\nListen&text=Adenomyosis%20is%20a%20condition%20th\nat,or%20symptoms%20of%20the%20condition. Accessed \non 22-02-2021 \n2. Benagiano G, Brosens I. History of adenomyosis. Best Pract \nRes Clin Obstet Gynaecol 2006;20:449-463. \n3. https://www.cdc.gov/des/hcp/resources/materials/clinician_i\nns_materials.pdf Accessed on 22-02-2021 \n4. Garcia L, Isaacson K. Adenomyosis: review of the \nliterature. J Minim Invasive Gynecol 2011;18:428-437. \n5. Azziz R. Adenomyosis: current perspectives.  Obstet \nGynecol Clin North Am 1989;16:221-235. \n6. Vercellini P, Vigano P, Somigliana E et al. Adenomyosis: \nepidemiological factors.  Best Pract Res Clin Obstet \nGynaecol 2006;20:465-477.  \n7. Parazzini F, Mais V, Cipriani S et al . Determinants of \nadenomyosis in women who underwent hysterectomy for \nbenign gynecological conditions: results from a prospective \nmulticentric study in Italy.  Eur J Obstet Gynecol Reprod \nBiol. 2009;143:103-106. \n8. Taran FA, Weaver AL, Coddington CC et al . \nUnderstanding adenomyosis: A case control study.  Fertil \nSteril 2010;94:1223-1228. \n9. Taran FA, Weaver AL, Coddington C C et al . \nCharacteristics indicating adenomyosis coexisting with \nleiomyomas: a case -control study.  Hum \nReprod. 2010;25:1177-1182. \n10. Taran FA, Wallwiener M, Kabashi D et al .Clinical \ncharacteristics indicating adenomyosis at the time of \nhysterectomy: A retrospective study in 291 patients.  Arch \nGynecol Obstet 2012;285:1571-1576. \n11. Siedentopf P. Chronic pain syndromes  in gynaecological \npractice: endometriosis and fibromyalgia.  Geburtsh \nFrauenheilk. 2012;72:1092-1098. \n12. Burghaus S, Klingsiek P, Fasching PA et al. Risk factors for \nendometriosis in a German case –control study.  Geburtsh \nFrauenheilk 2011;71:1073-1079. \n13. Wölfler MM, Stadermann M, Rath W et al. Anamnestisches \nScreening bei symptomatischen Patientinnen mit und ohne \nEndometriose. Geburtsh Frauenheilk. 2011;71:53-58. \n14. Templeman C, Marshall S F, Ursin G et al . Adenomyosis \nand endometriosis in the California Teachers Stu dy. Fertil \nSteril 2008;90:415-424.  \n15. Weiss G, Maseelall P, Schott L L et al . Adenomyosis a \nvariant, not a disease? Evidence from hysterectomized \nmenopausal women in the Study of Womenʼs Health Across \nthe Nation (SWAN) Fertil Steril 2009;91:201-206. \n16. Parrott E, Butterworth M, Green A, White IN, Greaves P. \nAdenomyosis--a result of disordered stromal differentiation. \nAm J Pathol. 2001 ;159(2):623-30. doi: 1 0.1016/S0002-\n9440(10)61733-6. PMID: 11485920; PMCID: \nPMC1850567. \n17. Vora IM, Raizada RM, Rawal MY, Chadda JS. \n\nInternational Journal of Clinical Obstetrics and Gynaecology http://www.gynaecologyjournal.com \n~ 223 ~ \nAdenomyosis. J Postgrad Med 1981;27:7-11 \n18. Malcolm G. Munro Classification and Reporting Systems \nfor Adenomyosis, Jmig (Journal of minimally invasive \ngynecology) 2 7, 2, P296 -308, FEBRUARY 01, 2020 \nhttps://doi.org/10.1016/j.jmig.2019.11.013","source_license":"CC0","license_restricted":false}