Manabu Ogoyama

Decidualization of Ovarian Endometriosis during Pregnancy Mimicking Malignancy : Report of Three Cases with a Literature Review

Background: Intracystic papillary excrescence is a characteristic morphological feature of ovarian malignancy. A few recent reports have demonstrated that ovarian endometriotic cysts, undergoing decidualization during pregnancy, occasionally show excrescence, necessitating surgery during pregnancy; however, this phenomenon is not well recognized among clinicians. Cases: Three pregnant women with decidualized ovarian endometriosis showed excrescence. Both ultrasound and magnetic resonance imaging (MRI) preoperatively suggested the presence of underlying ovarian endometriotic cysts in 2 women, but not in the other. Intracystic papillary excrescence prompted us to perform laparotomy at 14, 14, and 19 weeks of pregnancy, respectively, with 1 woman aborting in the 21st week, and with 2 delivering healthy term infants. Histological examination confirmed the diagnosis of decidualized ovarian endometriotic cysts in all 3 patients. Conclusions: We provide the first report of pregnant women in whom excrescence occurred from ovarian endometriotic cysts without preoperative evidence. Decidualized ovarian endometriosis, even without preoperative morphological features of endometriosis, should be added to the differential diagnosis of ovarian malignancy during pregnancy.

Uterine artery pseudoaneurysm: its occurrence after non-traumatic events, and possibility of “without embolization” strategy

OBJECTIVES Uterine artery pseudoaneurysm (UAP) has been considered to occur very rarely after traumatic delivery/abortion, and is usually detected after its rupture, yielding massive bleeding. Our hypothesis is: some UAP may be undetected without massive bleeding and may spontaneously resolve, and, thus, may not require transarterial embolization (TAE). We attempted: (1) to detect both ruptured and non-ruptured UAP, thereby characterizing candidates of spontaneously resolving UAP, and (2) to confirm that UAP is not rare and not always associated with traumatic events. STUDY DESIGN This was a retrospective observational study of 50 women with angiographically confirmed UAP and treated by TAE. Angiograms and medical charts were retrieved to examine the associations among symptoms, ultrasound findings, and extravasation. Gray-scale ultrasound was performed for all women after delivery or abortion as our routine practice. RESULTS UAP occurred in 3-6/1000 deliveries and 40% occurred after non-traumatic deliveries/abortion. While 36% had active vaginal bleeding at admission, 64% did not. While 100% of patients with current active bleeding showed extravasation from the pseudoaneurysmal sac, patients without it showed a varied incidence of extravasation depending on the bleeding pattern/history and ultrasound findings. Interestingly, all patients with current bleeding (-), bleeding history (+), and ultrasound-discernable-intrauterine low echoic mass (-) were devoid of extravasation, suggesting that UAP may show progression to thrombosis and, thus, resolve spontaneously. CONCLUSIONS UAP may not be so rare and not associated with traumatic delivery/abortion. Some UAP may resolve, and, thus, may not require TAE, at least immediately.

Postpartum hemorrhage: is angiographically detectable “sac” mandatory for diagnosis of ruptured pseudoaneurysm?

Postpartum hemorrhage (PPH) continues to threaten mothers’ lives: articles in this journal repeatedly dealt with this issue [1]. Rupture of a uterine artery pseudoaneurysm (UAP) causes PPH. Contradictory to previous belief, UAP can occur more frequently (2–3/1000 deliveries) [2] and can no less frequently occur after non-traumatic than traumatic deliveries [2], and, thus, UAP has attracted wider attention. The symptoms and signs of UAP change in patient-by-patient manner, deceiving obstetricians [3], leading our team to refer to UAP as ‘‘chameleon’’ in obstetric practice [4]. Although ultrasound and enhanced computed tomography facilitates a pretreatment diagnosis, pelvic angiography is a gold standard for diagnosing ‘‘ruptured’’ UAP, namely, a parent artery (usually the uterine artery), narrow connecting portion, pseudoaneurysmal sac, and extravasation from the sac: many obstetricians consider this as a ‘‘typical’’ angiographic image of ruptured UAP. Actually, Lipere et al. [5] recently reported PPH caused by uterine artery injury showing ‘‘extravasation without sac’’, with a context that this was a new clinical entity and not ruptured UAP. The presence of angiographically detectable ‘‘sac’’ may be considered mandatory for ruptured UAP diagnosis. We are concerned about ‘‘too much’’ emphasis being placed on the presence of a sac. In UAP rupture, a sac may not be as evident as expected. In our previous analysis of 22 consecutive angiographically confirmed UAP cases [2], some had only a small ‘‘sac’’, and at its extreme end, ‘‘no discernable sac’’ may be the result. We consider four scenarios regarding the relationship between the presence/absence of a sac and extravasation in ruptured UAP. First, a pseudoaneurysmal sac is initially occupied only by swirling blood flow. If it ruptures, the round ‘‘sac’’ and extravasation from it may be visible on angiography, showing a typical ruptured pseudoaneurysm, with which obstetricians are well accustomed. Second, with the time course, an intra-sac thrombus may be formed: centripetal formation of an intra-sac thrombus has already been demonstrated in a femoral artery pseudoaneurysm [6]. If rupture occurs at this stage, the sac, partly occupied by the thrombus, may become irregularly shaped and angiography may not show the typical image of a ‘‘round sac with extravasation’’. Third, at the extreme end of this phenomenon, the aneurysmal sac may mostly become occupied by the thrombus and there remains a small free blood-flow space. When a pseudoaneurysm ruptures at this stage, angiography may indicate extravasation without a sac. Fourth, the thrombosed sac may be broken and detached altogether at the rupture: the aneurysmal structure may no longer exist and, thus, it is undiscernable. In the latter two scenarios, angiography may show ‘‘extravasation without sac’’, similarly to in Lipere et al.’s patient [5]. & Shigeki Matsubara matsushi@jichi.ac.jp

Markedly higher sFlt-1/PlGF ratio in a woman with acute fatty liver of pregnancy compared with HELLP syndrome: sFlt-1/PlGF ratio in AFLP and HELLP

To compare serum levels of angiogenesis‐related factors between 14 women with HELLP (hemolysis, elevated liver enzymes and low platelet count) syndrome and a woman with acute fatty liver of pregnancy (AFLP).

Galectin-1 as a novel risk factor for both gestational hypertension and preeclampsia, specifially its expression at a low level in the second trimester and a high level after onset

Our aim was to evaluate whether the serum level of galectin-1 (Gal-1) at 18–24 and 27–31 weeks of gestation is a risk factor for predicting the later occurrence of not only preeclampsia (PE) but also gestational hypertension (GH). We measured serum levels of soluble fms-like tyrosine kinase 1 (sFlt-1), placental growth factor (PlGF), and Gal-1 using an enzyme-linked immunosorbent assay in 81 and 73 normal pregnant women, 22 and 16 women with a later onset of GH, and 37 and 29 women with a later onset of PE at 18–24 and 27–31 weeks, respectively. We also measured Gal-1 in 33 women with GH and 78 women with PE after the onset. The levels of Gal-1 after the onset of GH, late-onset PE (onset at ⩾34 weeks), and early-onset PE (onset at <34 weeks) were significantly higher than those in normal pregnant women at 27–31 weeks. However, the low levels of Gal-1 (<8.1 ng ml−1) at 18–24 weeks, but not at 27–31 weeks, predicted the later occurrence of not only early-onset PE and late-onset PE but also GH. The low level of Gal-1 at 18–24 weeks was an independent risk factor for the later occurrence of GH and PE, after adjusting for the effects of a high BP and increased sFlt-1/PlGF ratio at 18–24 weeks. In conclusion, the serum level of Gal-1 is a novel risk factor for both GH and PE, specifically its expression at a low level in the second trimester and a high level after onset.

Ritodrine-induced rhabdomyolysis, infantile myotonic dystrophy, and maternal myotonic dystrophy unveiled

A primiparous pregnant woman in remission of myositis suffered very acute‐onset ritodrine‐induced rhabdomyolysis. At 29 gestational weeks, ritodrine was administered for threatened preterm labor. Just 3 h later, she complained of severe limb muscle pain, with serum creatinine phosphokinase elevated to 32 019 U/L and myoglobinuria. The muscle pain disappeared immediately after ceasing administration of ritodrine. At 31 weeks, premature rupture of the membranes occurred, necessitating cesarean section, yielding a baby with weak tonus, and the presence of infantile muscle diseases was suspected. Genetic analysis of the infant confirmed myotonic dystrophy (dystrophia myotonica, DM), which prompted us to perform maternal genetic analysis, confirming maternal DM. Ritodrine can induce rhabdomyolysis even in the prodromal phase with a mild phenotype of DM. A literature review suggested that ritodrine‐induced rhabdomyolysis may be likely to occur more acutely after ritodrine administration in DM compared with non‐DM mothers.

Contents Vol. 66, 2008

M.A. Belfort, Provo, Utah J. Bornstein, Nahariya H.L. Brown, Durham, N.C. C. Chapron, Paris P.G. Crosignani, Milan J. de Haan, Maastricht G.A. Dekker, Adelaide J.A. Deprest, Leuven K. Hecher, Hamburg S. Kahhale, São Paulo H. Kliman, New Haven, Conn. T.F. Kruger, Tygerberg J.A. Kuller, Raleigh, N.C. M.J. Kupferminc, Tel Aviv H. Minkoff , Brooklyn, N.Y. J. Moodley, Congella J.M. Mwenda, Nairobi H. Odendaal, Tygerberg J.T. Repke, Hershey, Pa. G.R. Saade, Galveston, Tex. Founded 1895 as ‘Monatsschrift für Geburtshilfe und Gynäkologie’, continued 1946–1969 as ‘Gynaecologia’ and 1970–1977 as ‘Gynecologic Investigation’