{"paper_id":"59c36b8c-b4a0-4a62-a199-ae0bed407110","body_text":"Korean Journal of Obstetrics and Gynecology\nVol. 53  No. 1  January 2010\n- 70 -\n접 수 일：2009. 11.  2.\n채 택 일：2009. 12.  1.\n교신저자：황한성\nE-mail：hwanghs@kuh.ac.kr\n자궁선근증을 가진 초산모의 조산\n건국대학교 의과대학 산부인과학교실\n고정화 ․ 윤상희 ․ 강현정 ․ 권한성 ․ 손인숙 ․ 황한성\nPreterm delivery in a primigravida \nwith uterine adenomyosis\nJung Hwa Ko, M.D., Sang Hee Yoon, M.D., Hyun Jung Kang, M.D., \nHan Sung Kwon, M.D., In Sook Sohn, M.D., Han Sung Hwang, M.D.\nDivision of Maternal and Fetal Medicine, Department of Obstetrics and Gynecology, \nKonkuk University Medical Center, Seoul, Korea\nAdenomyosis has been well known to be associated with infertility, spontaneous rupture of the uterus during labor in a primipar ous woman, \nspontaneous preterm labor, preterm premature rupture of membranes (PPROM), and delayed postpartum hemorrhage. We recently \nexperienced a case of preterm delivery at 29 gestational weeks in a primigravid woman with uterine adenomyosis. We report the c ase \nof preterm delivery accompanied by various complications such as uncontrolled pain, preterm labor, and oligohydramnios in a woman with \nuterine adenomyosis.\nKey Words: Uterine adenomyosis, Abdominal pain, Preterm birth\nAdenomyosis was firstly used by discovered by \nFrankl, and pathologically defined by Bird et al. in \n1972; the benign invasion of endometrium into the \nmyometrium, producing a diffusely enlarged uterus \nwhich microscopically exhibits ectopic, non-neo-\nplastic, endometrial glands and stroma surrounded by \nthe hypertrophic and hyperplastic myometrium.\n1,2 \nSeveral previous studies were reported that the risk of \nadenomyosis was increased after spontaneous abor-\nt i o n s  a n d  d i l a t i o n  a n d  c u r e t t a g e .3,4 U t e r i n e  a d -\nenomyosis most commonly occurs in multiparous wom-\ne n  i n  t h e i r  l a t e  3 0 s  o r  4 0 s ,  b u t  r a r e  i n  n u ll i g r a v i d \nwomen. Uterine adenomyosis can be classified into two \ncategories: focal adenomyosis with lesions localized in \nthe anterior or posterior wall; and diffuse ad-\nenomyosis with lesions in the entire uterus. Regarding \nobstetric problems related with adenomyosis, recent \nreports showed that adenomyosis was associated with \ninfertility,\n5,6 spontaneous rupture of the uterus during \nlabor in a primiparous woman,7,8 spontaneous preterm \nlabor, preterm premature rupture of membranes \n(PPROM),\n9 and delayed postpartum  hem orrhage.10 W e \nrecently experienced a case of preterm delivery at 29 \ngestational weeks in a primigravid woman with uterine \nadenomyosis. \n\n고정화 외 5인. 자궁선근증을 가진 초산모의 조산\n- 71 -\nA\n B\nFig. 2. At 24 th gestational week, shortening of uterine cervix with funneling was visible by transvaginal ultrasonography (A), and \nuterine adenomyosis with normal gestational sac was seen in transabdominal ultrasonography (B).\nFig. 1. At 15 th gestational week, uterine adenomyosis was seen \nin the mainly fundal area (thick white arrow). Normal gesta-\ntional sac and fetus were visible in lower uterine segment area. \nCase Report\nA 35-year-old woman, gravid 1 para 0 was admitted \nto our hospital with acute lower abdominal pain at 15th \ngestational week. The patient had tried unsuccessfully \nto become pregnant for about 2 years. In infertility \nwork up, bilateral tubal obstruction and adenomyosis \nwere noted. She became pregnant after undergoing in \nvitro fertilization and embryo transfer (IVF-ET) to be \npregnant of adenomyosis. After admission, trans-\nabdominal ultrasonography (Aloka Prosound alpha-10, \nTokyo, Japan) revealed uterine adenomyosis including \nnormal gestational sac in lower uterine segment (Fig. 1). \nThickened uterine wall in adenomyosis was extended \nfrom fundal area to mid portion of uterus, and in this \narea, there was a narrow endometrial cavity without \ngestational sac. Anterior wall thickness of uterus was \nabout 10 cm, and posterior was about 3 cm. Routine \nchemistry, ultrasonography for exclusion of the acute \nappendicitis, and tococardiogram for uterine con-\nt r a c t i o n  w a s  p e r f o r m e d .  L a b o r a t o r y  r e s u l t s  s h o w e d  \nhemoglobin 10.8 g/dL, white bleed cell count 20.19× \n10\n3/μL, neutrophil segmented 81%, C-reactive protein \n(CRP) 17.96 mg/dL (reference range: 0.01~0.3 mg/dL). \nWith leukocytosis and elevated CRP being noted, and \nshe had mild fever (body temperature: 37.5℃). Acute \nappendicitis, placental abruption, ureteral stone, and \npyelonephritis were ruled out by physical examination, \nultrasonography, and laboratory test. Uterine con-\ntraction was detected at 15-minutes interval. She was \ndiagnosed with threatened abortion, and adenomyosis. \nShe was prescribed analgesics including acet-\naminophen and codeine for pain control, and anti-\nbiotics with ampicillin and sulbactam were taken. \nUterine contraction was subsided after hydration and \nbed rest. At hospital day 10, uterine cervix length es-\n\n대한산부회지 제53권 제1호, 2010\n- 72 -\nA\n B\nFig. 3. Uterine adenomyosis was serially decreased in size at 2 weeks (A), and 8 weeks (B) after delivery.\ntimated by transvaginal ultrasonography was 3.1 cm, \nabdominal pain was subsided, and she was discharged \nwithout uterine contraction.\nAfter 24\nth gestational week, she was repeatedly ad-\nmitted to the hospital because of severe lower ab-\ndominal pain, and regular uterine contraction. \nUltrasonography (Accuvix, Medison, Seoul, Korea) \nshowed that uterine cervix was shortened and fun-\nneled (cervix length: about 1.3 cm) (Fig. 2). Tocolytics \nwith ritodrine and analgesics including acetamino-\nphen, codeine, nalbuphine, and pethidine were taken \nfor uterine contraction and pain control. Pain control \nwas daily performed due to extreme and frequent pain \no n  l o w  a b d o m e n .  A t  h o s p i t a l  d a y  6 ,  u t e r i n e  c o n -\nt r a c t i o n  w a s  d i s a p p e a r e d ,  b u t  a b d o m i n a l  p a i n  w a s  \nsustained. At hospital day 9, abdominal pain was de-\ncreased, so she was discharged. \nAt 27\nth gestational week, she was readmitted with \nsevere abdominal pain, and regular uterine contrac-\ntion. Uterine cervix length estimated by transvaginal \nultrasonography was 1.0 cm. In fundal area, anterior \nwall thickness was about 9 cm, and posterior was 3 cm. \nEstimated fetal weight was adequate to gestational \nage, but amnionic fluid index was 8 cm. There was no \nevidence of preterm premature rupture of membranes \n(PPROM). During 2 weeks, tocolytics with ritodrine \nand analgesics including acetaminophen, codeine, nal-\nbuphine, and pethidine, and betamethasone were \ntaken. At 29\nth gestational week, emergency cesarean \nsection was done, because oligohydramnios (amnionic \nfluid index: 4), breech presentation by ultra-\nsonography, uncontrolled abdominal pain, and fetal \ndistress (moderate to severe variable deceleration \nd u r i n g  f e t a l  m o n i t o r i n g )  w e r e  n o t e d .  A  m a l e  b a b y  \nweighing 1,140 g was born. Apgar scores were 3 at \n1 minute and 6 at 5 minute. The baby was moved to \nthe neonatal intensive unit (NICU) with endotracheal \ni n t u b a t i o n  a n d  o x y g e n  s u p p l y .  T h e  e s t i m a t e d  b l o o d  \nloss during surgery was about 500 mL. The baby had \nrespiratory distress in the early period of NICU ad-\nmission but was recovered in NICU and was discharged \nfrom the hospital after 72-day stay, and so was the \nm oth er 6- day after th e op eration  w ith ou t any  other \nproblem. Ultrasonography at 2 and 8 weeks after de-\nlivery was performed. Uterus size was serially de-\ncreased (Fig. 3).  \nDiscussion \nBecause uterine adenomyosis is associated with in-\n\n고정화 외 5인. 자궁선근증을 가진 초산모의 조산\n- 73 -\nfertility, spontaneous uterine rupture, preterm labor, \nand preterm premature rupture of membranes, preg-\nnancy in a primigravid woman with uterine ad-\nenomyosis is rare and highly risky. \nThe effects of myoma on pregnancy or pregnancy on \nmyoma are well known,\n11,12 but adenomyosis not. A re-\ncent case report showed that a rapidly growing ad-\nenomyosis was identified during the first trimester in \na 35-year-old infertile woman achieving pregnancy \nafter controlled ovarian hyperstimulation.\n13 A lth o u g h \nthe pregnancy was terminated at the 18th gestational \nweek after premature rupture of membranes following \ngenetic amniocentesis, severe abdominal pain by red \ndegeneration was similar to our case. No imaging \nstudy for pregnant uterus for prepregnant and 1\nst tri-\nm e s t e r  p e r i o d  w a s  p e r f o r m e d ,  s o  w e  d o n’t know \nwhether the enlarged uterine was rapidly grown dur-\ning first trimester. But, pregnancy in our case was \nmaintained to 29\nth gestational week, and the more the \npregnancy progresses, the more severe the intensity of \npain was. During follow up periods, there was no evi-\ndence of rapid growing adenomyosis. From 15\nth gesta-\ntional to delivery, one of major problems of pregnancy \nmaintenance was pain in our case. Various analgesics \nwere fully used for pain control, but the pain was not \nsatisfactorily controlled. It is well known that the \nmost complaint associated with uterine myoma during \npregnancy is pain. Although initial hypotheses sug-\ngested that pain was secondary to fibroid growth dur-\ning pregnancy, painful episodes appear to be unrelated \nto absolute fibroid size or growth.\n14 Prospective, lon-\ngitudinal studies have also failed to document sig-\nnificant change in fibroid size during pregnancy.15-17 A \nc a s e  f o r  s u c c e s s f u l  p r e g n a n c y  o u t c o m e  i n  a  w o m a n  \nwith large uterine intramural myoma was reported by \nSkrablin S et al. Pregnancy with about 13 cm sized in-\ntramural myoma in fundal area was reported. That \ncase showed that abdominal pain did not worsen dur-\ning pregnancy, and an elective cesarean section was \nperformed at 38 weeks.\n18 However, it is little known \nthat pregnancy with adenomyosis is associated with \npain. The most common cause of neonatal morbidity \nreported in any of these studies was preterm delivery. \nWomen with myomas in pregnancy seem to be at in-\ncreased risk of threatened preterm labor with reported \nrates of up to 21.5%, and the risk of preterm labor \ncorrelated positively with the size of the largest \nfibroid. And it is generally accepted that tocolytic \nagents used in pregnancy with myoma and preterm la-\nbor is not different from with only preterm labor.\n19-22\nIn this case, the pregnant woman had low abdominal \npain, mild fever, leukocytosis, and increased in-\nflammatory markers. These symptoms and signs were \nsimilar to that of pregnancy with red degeneration of \nmyoma. Also, it was difficult to differentiate the \npregnancy with uterine adenomyosis from other medi-\nco-surgical, and obstetrical problems such as appen-\ndicitis, placental abruption, preterm labor, ureteral \nstone, pyelonephritis, and chorioamnionitis. \nHere, we report the case of preterm delivery ac-\ncompanied by various complications such as uncon-\ntrolled pain, preterm labor, and oligohydramnios in a \nwoman with uterine adenomyosis.\n참고문헌\n 1. Frankl O . A denom yosis uteri. Am  J O bstet G ynecol \n1925; 10: 680-4.\n 2. Bird CC, McElin TW, Manalo-Eslrella P. The elusive \nadenomyosis of the uterus - revisited. Am J Obstet \nGynecol 1972; 112: 583-93.\n 3 . P a r a z z i n i  F ,  V e r c e l l i n i  P ,  P a n a z z a  S ,  C h a t e n o u d  L ,  \nOldani S, Crosignani PG. Risk factors for \nadenomyosis. Hum Reprod 1997; 12: 1275-9. \n\n대한산부회지 제53권 제1호, 2010\n- 74 -\n 4. Curtis KM, Hillis SD, Marchbanks PA, Peterson HB. \nDisruption of the endometrial-myometrial border \nduring pregnancy as a risk factor for adenomyosis. Am \nJ Obstet Gynecol 2002; 187: 543-4.\n 5 .K i s s l e r  S ,  H a m s c h o  N ,  Z a n g o s  S ,  W i e g r a t z  I ,  \nSchlichter S, Menzel C, et al. Uterotubal transport dis-\norder in adenomyosis and endometriosis--a cause for \ninfertility. BJOG 2006; 113: 902-8.\n 6 . K u n z  G ,  B e i l  D ,  H u p p e r t  P ,  N o e  M ,  K i s s l e r  S ,  \nLeyendecker G. Adenomyosis in endometriosis- prev-\nalence and impact on fertility. Evidence from mag-\nn e t i c  r e s o n a n c e  i m a g i n g .  H u m  R e p r o d  2 0 0 5 ;  2 0 :  \n2309-16.\n 7. Bensaid F, Kettani F, el Fehri S, Chraibi C, Alaoui MT. \nObstetrical complications of adenomyosis. Literature \nreview  and  tw o case rep orts. J G yn ecol O b stet B iol \nReprod (Paris) 1996; 25: 416-8.\n 8. Mueller MD, Saile G, Brühwiler H. Spontaneous ute-\nrine rupture in the 18th week of pregnancy in a primi-\ngravida patient with adenomyosis. Zentralbl Gynakol \n1996; 118: 42-4.\n 9. Juang C M , C hou P, Y en M S, T w u N F, H orng H C , \nHsu WL. Adenomyosis and risk of preterm delivery. \nBJOG 2007; 114: 165-9.\n10. Wang PH, Pang YP, Chao HT, Lai CR, Juang CM, \nYuan CC, et al. Delayed postpartum hemorrhage in \nadenomyosis: a case report. Zhonghua Yi Xue Za Zhi \n(Taipei). 1998; 61: 492-5.\n11. Sheiner E, Bashiri A, Levy A, Hershkovitz R, Katz M, \nMazor M. Obstetric characteristics and perinatal out-\nc o m e  o f  p r e g n a n c i e s  w i t h  u t e r i n e  l e i o m y o m a s .  J  \nReprod Med 2004; 49: 182-6.\n12. Lev-Toaff AS, Coleman BG, Arger PH, Mintz MC, \nArenson RL, Toaff ME. Leiomyomas in pregnancy: so-\nnographic study. Radiology 1987; 164: 375-80.\n1 3 . K im  SH , K im  JK , C hae H D , K im  C H , K an g  B M . \nRapidly growing adenomyosis during the first trimes-\nter: magnetic resonance images. Fertil Steril 2006; 85: \n1057-8.\n14. Katz VL, Dotters DJ, Droegemueller W. Complica-\ntions of uterine leiomyomas in pregnancy. Obstet \nGynecol 1989; 73: 593-6.\n1 5 .N e i g e r  R ,  S o n e k  J D ,  C r o o m  C S ,  V e n t o l i n i  G .  \nP r e g n a n c y - r e l a t e d  c h a n g e s  i n  t h e  s i z e  o f  u t e r i n e  \nleiomyomas. J Reprod Med 2006; 51: 671-4.\n16. Aharoni A, Reiter A, Golan D, Paltiey Y, Scharf M. \nPatterns of growth of uterine leiomyomas during \npregnancy: a prospective longitudinal study. Br J \nObstet Gynaecol 1988; 95: 510-3. \n17. Muram D, Gillieson M, Walters JH. Myomas of the \nuterus in pregnancy: ultrasonographic follow-up. Am \nJ Obstet Gynecol 1980; 138: 16-9.\n18. Skrablin S, Banović V, Kuvacić I .  S u c c e s s f u l  p r e g -\nnancy outcome in a woman with large uterine intra-\nm u r a l  m y o m a .  E u r  J  O b s t e t  G y n e c o l  R e p r o d  B i o l  \n2005; 118: 115-6. \n19. Exacoustòs C, Rosati P. Ultrasound diagnosis of ute-\nrine myomas and complications in pregnancy. Obstet \nGynecol 1993; 82: 97-101.\n20. Qidwai GI, Caughey AB, Jacoby AF. Obstetric out-\ncomes in women with sonographically identified ute-\nrine leiomyomata. Obstet Gynecol 2006; 107 (2 Pt \n1): 376-82.\n21. Coronado GD, Marshall LM, Schwartz SM. Complica-\ntions in pregnancy, labor, and delivery with uterine \nleiomyomas: a population-based study. Obstet \nGynecol 2000; 95: 764-9.\n2 2 . D a v i s  J L ,  R a y - M a z u m d e r  S ,  H o b e l  C J ,  B a l e y  K ,  \nSassoon D. Uterine leiomyomas in pregnancy: a pro-\nspective study. Obstet Gynecol 1990; 75: 41-4.\n= 국문초록 =\n임신과 관련하여 자궁선근증은 불임, 초산임신에서의 자연적인 자궁파열, 조기진통, 조기양막파수, 산후출혈 등과 관련이 있는 \n것으로 알려져 있다. 최근 본원에서는 자궁선근증을 가지고 임신된 초산모를 임신 29주까지 유지한 후 조산하게 된 경험을 \n하게 되었다. 임신기간 중 산모는 반복적인 조기진통과 다양한 진통제에도 잘 조절되지 않는 극심한 복통을 호소하였고, \n원인이 밝혀지지 않는 양수 감소 등의 증상이 나타나서 결국 임신 29주에 조산으로 분만하게 되었다. 수술 후 태아는 27일 \n만에, 산모는 6일 만에 특별한 합병증 없이 퇴원하였다. 저자들은 초산모에게는 드물게 생기며, 여러 가지 위험요인을 안고 \n있는 자궁선근증과 동반된 임신을 경험하였고, 임신 29주까지 성공적으로 임신을 유지하였기에 간단한 문헌고찰과 함께 보\n고하는 바이다. \n중심단어: 자궁선근증, 복통, 조산","source_license":"CC0","license_restricted":false}