Preterm delivery in a primigravida with uterine adenomyosis

Korean Journal of Obstetrics and Gynecology Vol. 53 No. 1 January 2010 - 70 - 접 수 일：2009. 11. 2. 채 택 일：2009. 12. 1. 교신저자：황한성 E-mail：[email protected] 자궁선근증을 가진 초산모의 조산 건국대학교 의과대학 산부인과학교실 고정화 ․ 윤상희 ․ 강현정 ․ 권한성 ․ 손인숙 ․ 황한성 Preterm delivery in a primigravida with uterine adenomyosis Jung Hwa Ko, M.D., Sang Hee Yoon, M.D., Hyun Jung Kang, M.D., Han Sung Kwon, M.D., In Sook Sohn, M.D., Han Sung Hwang, M.D. Division of Maternal and Fetal Medicine, Department of Obstetrics and Gynecology, Konkuk University Medical Center, Seoul, Korea Adenomyosis has been well known to be associated with infertility, spontaneous rupture of the uterus during labor in a primipar ous woman, spontaneous preterm labor, preterm premature rupture of membranes (PPROM), and delayed postpartum hemorrhage. We recently experienced a case of preterm delivery at 29 gestational weeks in a primigravid woman with uterine adenomyosis. We report the c ase of preterm delivery accompanied by various complications such as uncontrolled pain, preterm labor, and oligohydramnios in a woman with uterine adenomyosis. Key Words: Uterine adenomyosis, Abdominal pain, Preterm birth Adenomyosis was firstly used by discovered by Frankl, and pathologically defined by Bird et al. in 1972; the benign invasion of endometrium into the myometrium, producing a diffusely enlarged uterus which microscopically exhibits ectopic, non-neo- plastic, endometrial glands and stroma surrounded by the hypertrophic and hyperplastic myometrium. 1,2 Several previous studies were reported that the risk of adenomyosis was increased after spontaneous abor- t 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 - enomyosis most commonly occurs in multiparous wom- e 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 women. Uterine adenomyosis can be classified into two categories: focal adenomyosis with lesions localized in the anterior or posterior wall; and diffuse ad- enomyosis with lesions in the entire uterus. Regarding obstetric problems related with adenomyosis, recent reports showed that adenomyosis was associated with infertility, 5,6 spontaneous rupture of the uterus during labor in a primiparous woman,7,8 spontaneous preterm labor, preterm premature rupture of membranes (PPROM), 9 and delayed postpartum hem orrhage.10 W e recently experienced a case of preterm delivery at 29 gestational weeks in a primigravid woman with uterine adenomyosis. 고정화 외 5인. 자궁선근증을 가진 초산모의 조산 - 71 - A B Fig. 2. At 24 th gestational week, shortening of uterine cervix with funneling was visible by transvaginal ultrasonography (A), and uterine adenomyosis with normal gestational sac was seen in transabdominal ultrasonography (B). Fig. 1. At 15 th gestational week, uterine adenomyosis was seen in the mainly fundal area (thick white arrow). Normal gesta- tional sac and fetus were visible in lower uterine segment area. Case Report A 35-year-old woman, gravid 1 para 0 was admitted to our hospital with acute lower abdominal pain at 15th gestational week. The patient had tried unsuccessfully to become pregnant for about 2 years. In infertility work up, bilateral tubal obstruction and adenomyosis were noted. She became pregnant after undergoing in vitro fertilization and embryo transfer (IVF-ET) to be pregnant of adenomyosis. After admission, trans- abdominal ultrasonography (Aloka Prosound alpha-10, Tokyo, Japan) revealed uterine adenomyosis including normal gestational sac in lower uterine segment (Fig. 1). Thickened uterine wall in adenomyosis was extended from fundal area to mid portion of uterus, and in this area, there was a narrow endometrial cavity without gestational sac. Anterior wall thickness of uterus was about 10 cm, and posterior was about 3 cm. Routine chemistry, ultrasonography for exclusion of the acute appendicitis, and tococardiogram for uterine con- t 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 hemoglobin 10.8 g/dL, white bleed cell count 20.19× 10 3/μL, neutrophil segmented 81%, C-reactive protein (CRP) 17.96 mg/dL (reference range: 0.01~0.3 mg/dL). With leukocytosis and elevated CRP being noted, and she had mild fever (body temperature: 37.5℃). Acute appendicitis, placental abruption, ureteral stone, and pyelonephritis were ruled out by physical examination, ultrasonography, and laboratory test. Uterine con- traction was detected at 15-minutes interval. She was diagnosed with threatened abortion, and adenomyosis. She was prescribed analgesics including acet- aminophen and codeine for pain control, and anti- biotics with ampicillin and sulbactam were taken. Uterine contraction was subsided after hydration and bed rest. At hospital day 10, uterine cervix length es- 대한산부회지 제53권 제1호, 2010 - 72 - A B Fig. 3. Uterine adenomyosis was serially decreased in size at 2 weeks (A), and 8 weeks (B) after delivery. timated by transvaginal ultrasonography was 3.1 cm, abdominal pain was subsided, and she was discharged without uterine contraction. After 24 th gestational week, she was repeatedly ad- mitted to the hospital because of severe lower ab- dominal pain, and regular uterine contraction. Ultrasonography (Accuvix, Medison, Seoul, Korea) showed that uterine cervix was shortened and fun- neled (cervix length: about 1.3 cm) (Fig. 2). Tocolytics with ritodrine and analgesics including acetamino- phen, codeine, nalbuphine, and pethidine were taken for uterine contraction and pain control. Pain control was daily performed due to extreme and frequent pain o 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 - t 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 sustained. At hospital day 9, abdominal pain was de- creased, so she was discharged. At 27 th gestational week, she was readmitted with severe abdominal pain, and regular uterine contrac- tion. Uterine cervix length estimated by transvaginal ultrasonography was 1.0 cm. In fundal area, anterior wall thickness was about 9 cm, and posterior was 3 cm. Estimated fetal weight was adequate to gestational age, but amnionic fluid index was 8 cm. There was no evidence of preterm premature rupture of membranes (PPROM). During 2 weeks, tocolytics with ritodrine and analgesics including acetaminophen, codeine, nal- buphine, and pethidine, and betamethasone were taken. At 29 th gestational week, emergency cesarean section was done, because oligohydramnios (amnionic fluid index: 4), breech presentation by ultra- sonography, uncontrolled abdominal pain, and fetal distress (moderate to severe variable deceleration d 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 weighing 1,140 g was born. Apgar scores were 3 at 1 minute and 6 at 5 minute. The baby was moved to the neonatal intensive unit (NICU) with endotracheal i 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 loss during surgery was about 500 mL. The baby had respiratory distress in the early period of NICU ad- mission but was recovered in NICU and was discharged from the hospital after 72-day stay, and so was the m oth er 6- day after th e op eration w ith ou t any other problem. Ultrasonography at 2 and 8 weeks after de- livery was performed. Uterus size was serially de- creased (Fig. 3). Discussion Because uterine adenomyosis is associated with in- 고정화 외 5인. 자궁선근증을 가진 초산모의 조산 - 73 - fertility, spontaneous uterine rupture, preterm labor, and preterm premature rupture of membranes, preg- nancy in a primigravid woman with uterine ad- enomyosis is rare and highly risky. The effects of myoma on pregnancy or pregnancy on myoma are well known, 11,12 but adenomyosis not. A re- cent case report showed that a rapidly growing ad- enomyosis was identified during the first trimester in a 35-year-old infertile woman achieving pregnancy after controlled ovarian hyperstimulation. 13 A lth o u g h the pregnancy was terminated at the 18th gestational week after premature rupture of membranes following genetic amniocentesis, severe abdominal pain by red degeneration was similar to our case. No imaging study for pregnant uterus for prepregnant and 1 st tri- m 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 whether the enlarged uterine was rapidly grown dur- ing first trimester. But, pregnancy in our case was maintained to 29 th gestational week, and the more the pregnancy progresses, the more severe the intensity of pain was. During follow up periods, there was no evi- dence of rapid growing adenomyosis. From 15 th gesta- tional to delivery, one of major problems of pregnancy maintenance was pain in our case. Various analgesics were fully used for pain control, but the pain was not satisfactorily controlled. It is well known that the most complaint associated with uterine myoma during pregnancy is pain. Although initial hypotheses sug- gested that pain was secondary to fibroid growth dur- ing pregnancy, painful episodes appear to be unrelated to absolute fibroid size or growth. 14 Prospective, lon- gitudinal studies have also failed to document sig- nificant change in fibroid size during pregnancy.15-17 A c 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 with large uterine intramural myoma was reported by Skrablin S et al. Pregnancy with about 13 cm sized in- tramural myoma in fundal area was reported. That case showed that abdominal pain did not worsen dur- ing pregnancy, and an elective cesarean section was performed at 38 weeks. 18 However, it is little known that pregnancy with adenomyosis is associated with pain. The most common cause of neonatal morbidity reported in any of these studies was preterm delivery. Women with myomas in pregnancy seem to be at in- creased risk of threatened preterm labor with reported rates of up to 21.5%, and the risk of preterm labor correlated positively with the size of the largest fibroid. And it is generally accepted that tocolytic agents used in pregnancy with myoma and preterm la- bor is not different from with only preterm labor. 19-22 In this case, the pregnant woman had low abdominal pain, mild fever, leukocytosis, and increased in- flammatory markers. These symptoms and signs were similar to that of pregnancy with red degeneration of myoma. Also, it was difficult to differentiate the pregnancy with uterine adenomyosis from other medi- co-surgical, and obstetrical problems such as appen- dicitis, placental abruption, preterm labor, ureteral stone, pyelonephritis, and chorioamnionitis. Here, we report the case of preterm delivery ac- companied by various complications such as uncon- trolled pain, preterm labor, and oligohydramnios in a woman with uterine adenomyosis. 참고문헌 1. Frankl O . A denom yosis uteri. Am J O bstet G ynecol 1925; 10: 680-4. 2. Bird CC, McElin TW, Manalo-Eslrella P. The elusive adenomyosis of the uterus - revisited. Am J Obstet Gynecol 1972; 112: 583-93. 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 , Oldani S, Crosignani PG. Risk factors for adenomyosis. Hum Reprod 1997; 12: 1275-9. 대한산부회지 제53권 제1호, 2010 - 74 - 4. Curtis KM, Hillis SD, Marchbanks PA, Peterson HB. Disruption of the endometrial-myometrial border during pregnancy as a risk factor for adenomyosis. Am J Obstet Gynecol 2002; 187: 543-4. 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 , Schlichter S, Menzel C, et al. Uterotubal transport dis- order in adenomyosis and endometriosis--a cause for infertility. BJOG 2006; 113: 902-8. 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 , Leyendecker G. Adenomyosis in endometriosis- prev- alence and impact on fertility. Evidence from mag- n 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 : 2309-16. 7. Bensaid F, Kettani F, el Fehri S, Chraibi C, Alaoui MT. Obstetrical complications of adenomyosis. Literature review and tw o case rep orts. J G yn ecol O b stet B iol Reprod (Paris) 1996; 25: 416-8. 8. Mueller MD, Saile G, Brühwiler H. Spontaneous ute- rine rupture in the 18th week of pregnancy in a primi- gravida patient with adenomyosis. Zentralbl Gynakol 1996; 118: 42-4. 9. Juang C M , C hou P, Y en M S, T w u N F, H orng H C , Hsu WL. Adenomyosis and risk of preterm delivery. BJOG 2007; 114: 165-9. 10. Wang PH, Pang YP, Chao HT, Lai CR, Juang CM, Yuan CC, et al. Delayed postpartum hemorrhage in adenomyosis: a case report. Zhonghua Yi Xue Za Zhi (Taipei). 1998; 61: 492-5. 11. Sheiner E, Bashiri A, Levy A, Hershkovitz R, Katz M, Mazor M. Obstetric characteristics and perinatal out- c 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 Reprod Med 2004; 49: 182-6. 12. Lev-Toaff AS, Coleman BG, Arger PH, Mintz MC, Arenson RL, Toaff ME. Leiomyomas in pregnancy: so- nographic study. Radiology 1987; 164: 375-80. 1 3 . K im SH , K im JK , C hae H D , K im C H , K an g B M . Rapidly growing adenomyosis during the first trimes- ter: magnetic resonance images. Fertil Steril 2006; 85: 1057-8. 14. Katz VL, Dotters DJ, Droegemueller W. Complica- tions of uterine leiomyomas in pregnancy. Obstet Gynecol 1989; 73: 593-6. 1 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 . P 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 leiomyomas. J Reprod Med 2006; 51: 671-4. 16. Aharoni A, Reiter A, Golan D, Paltiey Y, Scharf M. Patterns of growth of uterine leiomyomas during pregnancy: a prospective longitudinal study. Br J Obstet Gynaecol 1988; 95: 510-3. 17. Muram D, Gillieson M, Walters JH. Myomas of the uterus in pregnancy: ultrasonographic follow-up. Am J Obstet Gynecol 1980; 138: 16-9. 18. Skrablin S, Banović V, Kuvacić I . S u c c e s s f u l p r e g - nancy outcome in a woman with large uterine intra- m 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 2005; 118: 115-6. 19. Exacoustòs C, Rosati P. Ultrasound diagnosis of ute- rine myomas and complications in pregnancy. Obstet Gynecol 1993; 82: 97-101. 20. Qidwai GI, Caughey AB, Jacoby AF. Obstetric out- comes in women with sonographically identified ute- rine leiomyomata. Obstet Gynecol 2006; 107 (2 Pt 1): 376-82. 21. Coronado GD, Marshall LM, Schwartz SM. Complica- tions in pregnancy, labor, and delivery with uterine leiomyomas: a population-based study. Obstet Gynecol 2000; 95: 764-9. 2 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 , Sassoon D. Uterine leiomyomas in pregnancy: a pro- spective study. Obstet Gynecol 1990; 75: 41-4. = 국문초록 = 임신과 관련하여 자궁선근증은 불임, 초산임신에서의 자연적인 자궁파열, 조기진통, 조기양막파수, 산후출혈 등과 관련이 있는 것으로 알려져 있다. 최근 본원에서는 자궁선근증을 가지고 임신된 초산모를 임신 29주까지 유지한 후 조산하게 된 경험을 하게 되었다. 임신기간 중 산모는 반복적인 조기진통과 다양한 진통제에도 잘 조절되지 않는 극심한 복통을 호소하였고, 원인이 밝혀지지 않는 양수 감소 등의 증상이 나타나서 결국 임신 29주에 조산으로 분만하게 되었다. 수술 후 태아는 27일 만에, 산모는 6일 만에 특별한 합병증 없이 퇴원하였다. 저자들은 초산모에게는 드물게 생기며, 여러 가지 위험요인을 안고 있는 자궁선근증과 동반된 임신을 경험하였고, 임신 29주까지 성공적으로 임신을 유지하였기에 간단한 문헌고찰과 함께 보 고하는 바이다. 중심단어: 자궁선근증, 복통, 조산