Obstetric case reports Alvarez M, Lockwood CJ, Ghidini A et al. 1992. Prophylactic and emergent arterial catheterization for selective embolisation in obstetric hemorrhage. American Journal of Perinatology 9:441 – 444. Badawy SZA, Etman A, Singh M et al. 2001. Uterine artery embolisation: the role in obstetrics and gynecology. Clinical Imaging 25:288 – 295. Brown BJ, Heaston DK, Poulson AM et al. 1979. Uncontrollable postpartum bleeding: a new approach to hemostasis through angiographic arterial embolisation. Obstetrics and Gynecology 54:361 – 365. Chou YJ, Cheng YF, Shen CC et al. 2004. Failure of uterine arterial embolisation: placenta accreta with profuse postpartum haemorrhage. Acta Obstetricia Gynecologica Scandinavica 83:688 – 690. Combs CA, Murphy EL, Laros RK. 1991. Factors associated with postpartum haemorrhage with cesarean deliveries. Obstetrics and Gynecology 77:77 – 82. Dildy GA. 2002. Postpartum haemorrhage: new management options. Clinical Obstetrics and Gynecology 45:330 – 344. Greenwood LH, Glickman MG, Schwartz PE et al. 1987. Obstetrics and nonmalignant gynecological bleeding: treatment with angiographic embolisation. Radiology 164:155 – 159. Hansch E, Chitkara U, McAlpine J et al. 1999. Pelvic arterial embolisation for control of obstetric hemorrhage; a five year experience. American Journal of Obstetrics and Gynecology 180:1454 – 1460. Maier RC. 1993. Control of post partum hemorrhage with uterine packing. American Journal of Obstetrics and Gynecology 169:317 – 321.
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Mitty H, Sterling K, Alvarez M et al. 1993. Obstetric haemorrhage: prophylactic and emergency arterial catheterization and embolotherapy. Radiology 188:183 – 187. Nikolic B, Spies JB, Lundsten MJ et al. 2000. Patient radiation dose associated with uterine artery embolisation. Radiology 214:121 – 125. Ojala K, Pera¨la¨ J, Kariniemi J et al. 2005. Arterial embolisation and prophylactic catheterization for the treatment for severe obstetric haemorrhage. Acta Obstetricia Gynecologica Scandinavica 84:1075 – 1080. Pelage JP, Le Dref O, Mateo J et al. 1998. Life threatening postpartum haemorrhage: treatment emergency selective arterial embolisation. Radiology 208:359 – 362. Salomon LJ, De Tayrac R, Castaigne-Meary V et al. 2003. Fertility and pregnancy outcome following pelvic arterial embolisation for severe postpartum haemorrhage. A cohort study. Human Reproduction 18:849 – 852. Seror J, Allouche C, Elhaik S. 2005. Use of Sengstaken – Blakemore tube in massive post partum haemorrhage: a series of 17 cases. Acta Obstetricia Gynecologica Scandinavica 184:660. Silver RM, Landon MB, Rouse DJ et al. 2006. Maternal morbidity associated with multiple repeat caesarean deliveries. Obstetrics and Gynecology 107:1226. Vedantham S, Goodwin SC, McLucas B et al. 1997. Uterine artery embolisation: an underused method of controlling pelvic hemorrhage. American Journal of Obstetrics and Gynecology 176:938 – 948. Yamashita Y, Harada M, Yamamoto H et al. 1994. Transcatheter arterial embolisation of obstetric and gynecological bleeding: efficacy and clinical outcome. British Journal of Radiology 67:530 – 534.
Correspondence: S. Mushtaq, Department of Obstetrics and Gynecology, King Faisal Specialist Hospital and Research Centre, MBC 52, PO Box 3354, Riyadh 11211, Saudi Arabia. E-mail:
[email protected] DOI: 10.1080/01443610701748658
Pregnancy following Whipple’s procedure
A. E. MADU & O. OSOBA Department of Obstetrics and Gynaecology, Yeovil District Hospital NHS Foundation Trust, Yeovil, UK
Introduction
Case history
Neoplasms of the endocrine pancreas may be functional (secretory) and thus can cause clinically recognisable syndromes. They may also be non-functional (non-secretory) causing mainly pressure effects. Whatever the type of tumour, the treatment is basically the same, that is pancreaticoduodenectomy (Whipple’s Operation or Whipple’s procedure). The procedure was first performed by Alessandro Codivilla in 1898, however, it is widely associated with Allan Whipple (who performed it in 1935), after whom it was named. Early procedures were associated with very high mortality, up to 25% in the 1970s but this is now 54%. The pancreaticojejunostomy is the Achilles’ heels of the procedure, because in the best surgical hands, the leak rate is 10 – 20% (USC 2002; Boonnuch et al. 2005; Chabot 1993). When the procedure is performed during pregnancy or shortly before pregnancy, the emphasis would focus on the survival of the fetus irrespective of the functional state of the tumour. Here, we present a case of fetal survival and normal delivery at 36 weeks’ gestation following Whipple’s procedure.
A 28-year-old primigravida booked at our unit for antenatal care at 8 weeks’ gestation. She had pancreaticoduodenectomy for a pancreatic tumour about 8 weeks before her booking. She had a 1-year history of epigastric pain and occasional nausea and vomiting. The pain had increased in severity, occurred mainly after food and mostly in the early hours of the morning. Her GP who thought it was peptic ulcer had prescribed omeprazole (proton pump inhibitor), which gave her some relief from time to time. She also had weight loss but her appetite remained unchanged. Her symptoms however had nocturnal exacerbations. There was associated moderate vertigo for which she was given betahistine. She smoked 15 cigarettes/day but did not take alcohol. She was known to suffer from irritable bowel syndrome and was on mebeverine. She had a family history of hypertension. Abdominal ultrasound showed a benign-looking pancreatic mass measuring (3 6 3 cm) at the head of the pancreas, probably a pseudocyst. The liver, kidneys and gallbladder were normal.
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Her GP referred her to the physician/gastroenterologist for assessment. The proton inhibitor was stopped and the assessment included a test for insulin C-peptide to rule out insulinoma; tests for gut hormones; urgent gastroscopy to look for duodenal ulcers that may be caused by a gastrinoma and a CT scan. The C-peptide and gut hormone profile tests were all normal. A gastroscopy was attempted but the procedure was abandoned because she was unable to tolerate it. A CT scan showed a large mass of 3.7 cm, round and well defined in the inferior head of the pancreas and the uncinate process of the pancreas. There was some associated calcification but no dilatation or obstruction of either the bile or pancreatic duct. The tumour was in contact with the first jejunal branch of the superior mesenteric vein but not with the main vein. No lymphadenopathy or intrahepatic lesion was seen. The mass was not particularly vascular and thus unlikely to be a gastrinoma or insulinoma. She was then referred for review by a hepatobiliary surgeon who after review concluded that at her age, the tumour was most likely to be neuroendocrine in origin and non-secreting. She later had a pancreaticoduodenectomy (Whipple’s resection). She had no intraoperative complications but the postoperative period was complicated by intermitted diarrhoea (steatorrhea) and occurred after intake of any fatty foods. Her stool tended to float on water. She was then given pancreatic enzyme supplementation 10,000 units, two tablets, three times daily, to be adjusted according to her fat intake. She also suffered nocturnal reflux symptoms postoperatively and was given omeprazole 40 mg daily. She was recommended to have higher doses of the proton pump inhibitors in the long term because her duodenum was removed but as a temporary measure she was given esomeprazole to provide a better control of her symptoms. Histology confirmed a pancreatic tumour measuring 3.5 6 3 cm, benign and neuroendocrine in origin. It had a small possibility of local recurrence with low-grade malignant potential. Immunohistochemistry studies show tumour cells to be positive for neurone specific enolase and negative for cytokeratin. There was demonstrable immunoreactivity for chromogranin and synaptophysin. She was discharged, with follow-up arranged to take place every 3 months with the hepatobiliary unit. However, 8 weeks following the operation, she went to her GP for routine checks and complained she had not had a period. A urinary pregnancy test was positive and vaginal examination revealed a 6 – 8-week sized uterus. A transvaginal scan later confirmed a viable intrauterine of 6 weeks’ gestation. She was then referred for early obstetric care. Concerns were raised about her medication following the operation. She continued taking folic acid, esomeprazole, Creon and Gaviscon but mebeverine was discontinued. Her body mass index (BMI) on booking was 26. Routine booking investigations were normal, including the triple test at 16 weeks and anomaly scan at 19 weeks. She developed significant glycosuria at 27 weeks and glucose tolerance test confirmed gestational diabetes, which was later controlled by diet. She was seen every 2 weeks in the antenatal clinic and had shared care with input from the local surgical team and the surgical team that performed the Whipple’s procedure. A serial growth scan at 30 and 34 weeks showed growth in the 50 – 95th centile. At 23 and 26 weeks, she was admitted for upper abdominal pain and was treated symptomatically, as no cause was found after clinical and biochemical investigations. At 34 weeks, she was admitted for feeling unwell, vomiting and upper abdominal pain. She had pyrexia, a raised white cell count (17.7 6 109/l and neutrophilia (16.8 6 109/l. Alanine transaminase (128 m/l) was also raised and she was treated for cholangitis with intravenous Augmentin. At 36 weeks, she was admitted with another episode of severe right upper abdominal pain. A decision was then made to induce labour. On vaginal examination, Bishop’s score was 7 and an amniotomy was performed; the liquor was clear. She had an epidural anaesthetic for pain relief in labour. Labour was
spontaneous but was later augmented with oxytocin to correct incoordinate uterine contractions. She however progressed to normal delivery of a healthy male baby weighing 2,635 g in direct occipitoposterior position following episiotomy. The baby had Apgar scores of 9 at 1 min and 9 at 5 min. Labour had lasted for 8 h 40 min and the total blood loss was 350 ml. Mother and baby had no problems in the immediate puerperium and the latter needed no admission to the special baby care unit. She was discharged with a follow-up arranged with the medical, surgical and obstetric teams.
Discussion The occurrence of a benign pancreatic tumour in pregnancy is very rare. A prolonged and extensive literature search did not yield any article on pregnancy following Whipple’s operation. There were no articles on the incidence of Whipple’s procedure in pregnancy or the incidence of benign neuroendocrine pancreatic tumour in pregnancy for citation. Pancreatic tumours in pregnancy should be surgically removed. Even those classed as benign could grow rapidly in pregnancy due to the influence of the female sex hormones (Ganepola et al. 1999), causing greater pressure symptoms. The procedure of choice, Whipple’s procedure, is a major surgical operation. For the recuperating woman who is in the first 13 weeks of pregnancy, one of the significant risks is miscarriage. Many would consider termination of pregnancy on the grounds of maternal well-being. Since our patient did not have any period before the pregnancy, we calculated retrospectively that our patient may have become pregnant in 52 weeks following the procedure. Thus, her drug use in pregnancy had occurred in the pre-embryonic and embryonic phases of organogenesis. Of the seven cases Sciscione et al. (1996) studied, that had the operation in pregnancy, six resulted in live births and one had a maternal postoperative complication of pseudomembranous colitis caused by Clostridium difficile. Despite the effective treatment given, the fetus was compromised at 27 weeks and suffered in-utero intracranial haemorrhage and did not survive despite prompt caesarean section. The authors advised consideration of mother and fetus before such a major procedure. In other isolated malignant cases reported (Ruano et al. 2001; Haddad et al. 2005), the authors advocated radical surgery and special prenatal care to improve pregnancy outcome. Our patient had her postoperative period complicated by steatorrhea which apparently had no significant adverse effect on the fetus. There had been concerns about her medication prior to the discovery of her pregnancy; possible nutritional deficiency following the operation (and in association with steatorrhea) and subsequent development of gestational diabetes, which was controlled by diet. These are risks factors for pregnancy loss and growth restriction, and thus made fetal surveillance imperative. Also, our patient had cholangitis at 34 weeks, risking pre-term labour and delivery. Her pregnancy was complicated by recurrent upper abdominal and epigastric pain which can be common in pregnancy but simulates post-Whipple’s complaints. The hypochondrial and epigastric pain persisted during the intra-partum period, thus simulating uterine rupture or placental abruption. She was also very strong-willed and went on to have a normal vaginal delivery. Thus our experience and that of others (Ruano et al. 2001; Levy et al. 2004; Fernandez et al. 2005) strongly suggests that pregnancy after Whipple’s procedure does not have a significant harmful effect on the fetus if adequate prenatal care is in place.
References Boonnuch W, Akaraviputh T, Lohsiriwat D. 2005. Whipple’s operation with an operative mortality in 37 consecutive patients: Thai surgeons’ experience. Journal of the Medical Association of Thailand 88:467 – 472.
Obstetric case reports Chabot JA. 1993. The Whipple Procedure 1935 – 1993. PandS Medical Review 1:1. Fernandez EML, Malagon AM, Gonzalez IA, Montes RM, Luis HD, Hermoso FG et al. 2005. Mucinous cystic neoplasm of the pancreas during pregnancy: the importance of proper management. Journal of Hepatobiliary Surgery 12:494 – 497. Ganepola GAP, Gritsman AY, Asimakopulos N, Yiengpruksawan A. 1999. The American Surgeon 65:105 – 111. Haddad O, Porcu-Buisson G, Sakr R, Guidicelli B, Letreut YP, Gamerre M. 2005. Diagnosis and management of adenocarcinoma of the ampulla of Vater during pregnancy. European Journal of Obstetrics, Gynaecology and Reproductive Biology 119:246 – 249.
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Levy C, Pereira L, Dardarian T, Cardonick E. 2004. Solid pseudopapillary pancreatic tumor in pregnancy. A case report. Journal of Reproductive Medicine 49:61 – 64. Ruano R, Hase EA, Bernini C, Steinman DS, Birolini D, Zugaib M. 2001. Pancreaticoduodenectomy as treatment of adenocarcinoma of the papilla of Vater diagnosed during pregnancy. Journal of Reproductive Medicine 46:1021 – 1024. Sciscione AC, Villeneuve JB, Pitt HA, Johnson TR. 1996. American Journal of Perinatology 13:21 – 25. USC. 2002. Whipple Operation. Los Angeles: University of Southern California, Center for Pancreatic and Biliary Disease. pp 1 – 4.
Correspondence: A. E. Madu, Yeovil District Hospital NHS Foundation Trust, Yeovil, Somerset, UK. E-mail:
[email protected] DOI: 10.1080/01443610701800319
Negative pregnancy test: Could it be a molar pregnancy?
O. OFINRAN1, S. PAPAIOANNOU1, V. KANDAVEL1, S. SHRIVASTAVA1, S. HALL1 & J. TZAFETTAS2 1
Department of Obstetrics and Gynaecology, Heart of England NHS Foundation Trust, Princess of Wales Women’s Unit, Heartlands Hospital, Birmingham, UK and 2Department of Obstetrics and Gynecology, Aristotle University, Ippokration University Hospital, Thessaloniki, Greece
Introduction The urine pregnancy test is the standard instrument used in the triage of reproductive age women presenting with lower abdominal pain and/or vaginal bleeding. A positive result directs the diagnostic process towards complications of early pregnancy (miscarriage, ectopic, molar) that need immediate attention. A negative pregnancy test on the other hand essentially excludes the above. The case presented illustrates a noteworthy exception to the above common sense emergency gynaecology algorithm. Failure to take account of this exception might lead to delays in the diagnosis of a molar pregnancy, as well as in the appropriate management and prognosis of women with this potentially malignant condition.
Case report A 17-year-old woman presented to the gynaecology emergency department with a 2-week history of vaginal bleeding, suprapubic pain and persistent vomiting. A home urine pregnancy test had been negative. Her last menstrual period was reported to be about 2 weeks previously and her menstrual cycles had been regular. On examination, she was tachycardic, while her blood pressure was normal. Her abdomen was soft. A non-tender, smooth and mobile lower abdominal mass was palpable. Pelvic examination revealed a closed cervix and an enlarged uterus equivalent to 14 weeks’ gestation size. However, the repeat urine pregnancy test was again negative. The gynaecology registrar on-call felt there was no urgency, thus the patient was discharged home and given an appointment for a gynaecology outpatient clinic for investigation of the enlarged uterus. She collapsed at home 2 weeks later and at the Accident and Emergency department, she gave a history of heavy vaginal bleeding with passage of clots for 8 days, persistent vomiting and increasing abdominal distention and pain. She recalled that she last had unprotected sexual intercourse about 5 months before,
however four pregnancy tests in a row had been negative. Pelvic examination this time revealed a closed cervical os with minimal bleeding and a 20-week size uterus. A urine pregnancy test was faintly positive on this occasion and an urgent pelvic ultrasound scan showed a mass in the uterine cavity with solid areas interspaced with cystic areas consistent with the presence of molar tissue. Serum beta human chorionic gonadotrophin (bhCG) was in excess of 500,000 IU/l and her haemoglobin level was 10.6 g/dl. A suction evacuation of the uterus under ultrasound guidance was performed the same day, during which 1.3 l of blood-stained soft vesicular tissue was evacuated. Her haemoglobin on the third postoperative day was 7.0 g/dl. She declined blood transfusion and was discharged home on oral iron. Histopathology confirmed a complete hydatidiform mole and she was registered with the Trophoblastic Tumour Screening and Treatment Centre at Charing Cross Hospital, London, in accordance with current guidelines. When she was seen in the clinic 2 months later, she felt well and reported that she had her periods as usual.
Discussion The incidence of gestational trophoblastic neoplasia in the UK is 1 in 714 live births (Tham et al. 2003). The majority of women with complete hydatidiform mole present with vaginal bleeding, an enlarged uterus with a size larger than dates and abnormally high serum bhCG levels. Medical complications such as hyperemesis, anaemia, early pregnancy induced hypertension and hyperthyroidism are relatively rare (Jauniaux 1998). A positive urine pregnancy test plays a pivotal role in the early diagnosis by prompting further investigations in these patients. In countries with restricted facilities, severe genital tract haemorrhage, pulmonary embolism and high output cardiac failure are still potentially lethal complications that affect women with molar pregnancies (Mehra et al. 2005; Flam et al. 1998). Characteristic ultrasound appearances
