European Journal of Obstetrics & Gynecology and Reproductive Biology 149 (2010) 117–123 Contents lists available at ScienceDirect European Journal of Obstetrics & Gynecology and Reproductive Biology journal homepage: www.elsevier.com/locate/ejogrb LETTERS TO THE EDITOR—BRIEF COMMUNICATION Massive ascites, pleural effusion, and diaphragmatic implants in a patient with endometriosis Dear Editor, We report the case of a 38-year-old nulliparous young black woman, with primary infertility having massive ascites and a left adnexal cyst. She has a previous surgical history of diagnostic laparoscopy for endometriosis 2 years ago. In March 2008, she presented a thoracic endometriosis with right pleural effusion treated by thoracoscopy and pleurodesis. In December 2008, she presented a rapidly enlarging abdominal distension, dysmenorrhea, and chronic pain, without hematuria or dyschezia. Upon physical examination we found a massive ascites, decreased vesicular breath sounds on the right lung base, and a nodule on the left uterosacral ligament. Hemogram, hepatic, renal laboratory findings were normal. Thorax radiography showed a small atelectasia on the right side with no pleural effusion. Abdominal ultrasound and pelvic magnetic resonance imaging (MRI) showed a voluminous ascites, a simple 24 mm left adnexal cyst, uterus fibromas and sigmoid diverticulosis. Ca 125 was 50 U/ml. We performed a laparoscopic approach, extracting seven liters of brown fluid. Cytology studies of the ascites fluid were performed. The exploration of the abdomen and pelvis showed: an inflammatory peritoneum with intestinal adhesions to the pelvic wall, a left uterosacral nodule, a small left endometrioma, endometriotic implants on the diaphragm, and a normal right ovary. The uterus had several small fibromas and the Douglas pouch was occluded. A resection of the uterosacral endometriotic nodule, an ovarian cystectomy, coagulation of diaphragmatic implants and adhesiolysis were performed. The biopsy confirmed non-malignant endometriotic lesions. Drain was left in place for 2 days till drainage stopped. The patient’s evolution was satisfactory and she was discharged on the fourth day after surgery. Histocytological results confirmed the endometriotic lesions and an inflammatory peritoneal fluid. She had received gonadotropinreleasing hormone (GnRH) analogs for 3 months, starting 1 month before the surgery. Fertility is not an issue for the patient right now, her most important concern is to avoid ascites recurrence; she therefore remains under GnRH and an add-back therapy will be started. Eight months post-operatively, the patient is asymptomatic, and has no ascites, but a small right pleural effusion was detected on ultrasound. Endometriosis associated with ascites was first described in 1954 [1]. This unusual type of presentation involves deep, extrapelvic and severe endometriosis. To this day, 55 cases have been published on ascites and endometriosis. Including ours, 24 of the 56 cases had an additional history of pleural effusion [2,3]. We found it interesting that our patient had a history of pleural effusion due to thoracic endometriosis 9 months before the 0301-2115/$ – see front matter ß 2009 Elsevier Ireland Ltd. All rights reserved. massive ascites, and that diaphragmatic endometriotic implants were found during laparoscopy. To our knowledge, the association of ascites and pleural effusion with diaphragmatic endometriotic implants is infrequent, and had been only published by Brew [1]. In the largest series published [4,5], we could not find other cases with this association. The endometrial tissue migration mechanism from the pelvis to the thoracic cavity remains uncertain. Some possible routes include microembolization through pelvic veins, and trans-diaphragmatic movement through diaphragmatic defects [4]. The physiopathology of these large ascites is not completely understood. Some theories state that reactive peritoneal irritation and the production of exudates, could explain its occurrence [6]. Remarkably, only very few women with endometriosis have ascites. GnRH treatment for ascites and endometriosis, was first described in 1991 [7], and has been used in 11 cases since 1991 [2,3]. After 6 months of treatment, the ascites remits. However, in many cases it recurred after the treatment was stopped. In fact, most of the cases described in the literature recurred [2,3]. Many had problems with the infertility treatments and ascites recurred after ovarian stimulation [2,3,6,8], though; we think that this case has a high probability for recurrence. The diagnosis of this infrequent endometriosis presentation is rarely done preoperatively. The laparoscopic approach seems unusual since these patients could be managed as suspicions of ovarian cancer [2,3]. As its presentation can simulate malignancy, surgeons prefer laparotomy as an initial approach [8]. We performed laparoscopic surgery because we knew the previous history of endometriosis, and because the preoperative study did not allow suspecting a malignancy. We found that the advantages of using laparoscopy are important, adding magnified exploration of the abdominal and pelvic cavity, and the excision of most of the macroscopic disease, in a conservative and minimally invasive manner. We do not think that laparoscopic excision of endometriosis alone will solve the problem, but in this case the approach gave us more information through the exploration of the diaphragm, which could be interesting to study in the context of a patient with thoracic and pelvic endometriosis. These advantages could help us better understand this disease in the future. We think that these rare and severe cases must be evaluated carefully searching for extra pelvic endometriosis. References [1] Brew A. Endometriosis including endometriosis of the diaphragm and Meigs syndrome. Proc R Soc Med 1954;47:461. [2] Muneyyirci-Delale O, Neil G, Serur E, Gordon D, Maiman M, Sedlis A. Endometriosis with massive ascites. Gynecol Oncol 1998;69:42–6. 118 Letters to the Editor / European Journal of Obstetrics & Gynecology and Reproductive Biology 149 (2010) 117–123 [3] Ussia A, Betsas G, Corona R, De Cicco C, Koninckx PR. Pathophysiology of cyclic hemorrhagic ascites and endometriosis. J Minim Invasive Gynecol 2008;15: 677–81. [4] Redwine DB. Diaphragmatic endometriosis: diagnosis, surgical management, and long-term results of treatment. Fertil Steril 2002;77:288–96. Review. [5] Nezhat C, Seidman DS, Nezhat F, Nezhat C. Laparoscopic surgical management of diaphragmatic endometriosis. Fertil Steril 1998;69:1048–55. [6] El-Newihi HM, Antaki JP, Rajan S, Reynolds TB. Large bloody ascites in association with pelvic endometriosis: case report and literature review. Am J Gastroenterol 1995;90:632–4. [7] Yu J, Grimes D. Ascites and pleural effusion with endometriosis. Obstet Gynecol 1991;78:553–4. [8] Goumenou A, Matalliotakis I, Mahutte N, Koumantakis E. Endometriosis mimicking advanced ovarian cancer. Fertil Steril 2006;86:219.e23–2. Miranda-Mendoza Ignacio* Nassif Joseph Ferreira Hélder IRCAD/EITS and Strasbourg University Hospitals, Faculty of Medicine, Strasbourg, France Kone Mamourou Obstetrics and Gynecology Department, Clinique Fatima, Ivory Coast Wattiez Arnaud Obstetrics and Gynecology Department, IRCAD/EITS and Strasbourg University Hospitals, Faculty of Medicine, Strasbourg, France *Corresponding author at: IRCAD/EITS and Strasbourg University Hospitals, 1 Place de l’hopital, CP: 67091, Strasbourg, France. Tel.: +33 388119201; fax: +33 388119028 E-mail address: ignaciomir@med.uchile.cl (Miranda-Mendoza Ignacio) 26 May 2009 doi:10.1016/j.ejogrb.2009.10.017 Wernicke encephalopathy complicating hyperemesis gravidarum: A case report Dear Editor, We report an unusual case of Wernicke’s encephalopathy (WE), a disorder due to thiamine deficiency associated with alcoholism and malnutrition but that can also arise during the first trimester of pregnancy, due to hyperemesis gravidarum [1]. A 31-year-old woman, G3P1, presented at 14 weeks of gestation with uncontrollable vomiting and major weight loss. She was hospitalized and received glucose and saline, antiemetic therapy (metoclopramide) and parenteral nutrition. She was discharged ten days later on an oral diet. Two weeks later she developed neurologic disorders including diplopia and memory impairment. Physical examination showed normal consciousness, sinoauricular tachycardia, anxiety, horizontal multidirectional nystagmus and diplopia due to ophthalmoplegia. Laboratory studies showed elevated aspartate and alanine aminotransferase levels (144 IU/l [normal range <47] and 302 IU/l [normal range <65], respectively). Fetal sonography was normal. Cerebral magnetic resonance imaging showed hyperintense areas in the posterior part of the thalami, the mesencephalic tegmentum and the mammillary bodies (Fig. 1). Fig. 1. Note the hyperintensity of the mamillary bodies (white arrow) on T2weighted magnetic resonance imaging. WE was diagnosed on the basis of her clinical and radiological findings, and was confirmed by vitamin B1 assay (22.7 nmol/l, normal range 28–85 nmol/l). She was given intravenous vitamin B1 (1 g per day), vitamin B6 (pyridoxine, 500 mg per day), vitamin B3 (nicotinamide), metoclopramide and omeprazole. Most of the disorders gradually subsided over days or weeks, but nystagmus persisted. She gave birth at 38 weeks to a healthy boy weighing 3160 g. Wernicke’s encephalopathy is a rare complication of severe hyperemesis gravidarum, severe thiamine deficiency resulting from a combination of poor dietary intake, frequent vomiting, and the increased metabolic demands of pregnancy. It complicates 0.1– 0.5% of pregnancies [2]. The clinical features are non-specific but include the classical triad of ocular abnormalities, confusion and ataxia. Although WE is reversible, major complications can arise in the pregnant woman and her unborn child. On the maternal side, without active management, WE can lead to permanent neurologic lesions and the Korsakoff syndrome, which is fatal in 10–20% of cases [1]. On the fetal side, WE can lead to miscarriage, preterm birth and intrauterine growth retardation [3]. Magnetic resonance imaging is particularly important for initial diagnosis and for eliminating other diagnoses such as vascular lesions and thrombophlebitis. The lesions of WE are bilateral and symmetric, and involve the mamillary bodies, hypothalamic nuclei, periaqueductal gray matter, and superior cerebellar vermis [4]. Serum vitamin B1 assay does not precisely reflect total vitamin B1 status and is not routinely performed. The best diagnostic test is the response to vitamin B1 administration. Prevention is crucial for women with hyperemesis gravidarum, restoring glucose homeostasis should be systemically associated with thiamin supplementation (250 mg per day intravenous in normal saline or intramuscular) in combination with vitamins B3 and B6, dietary and behavioral modifications. Intravenous vitamin B1 500 mg three times per day should be given immediately if neurologic disorders occur [5]. Most of the associated disorders take several weeks to resolve. In conclusion, it should be remembered that women with severe hyperemesis gravidarum are at risk of WE. Urgent treatment is necessary when clinical signs and symptoms occur. Effective preventive measures are available and should be applied in situations at risk.