Journal of Obstetrics and Gynaecology

ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20

Conservative management of a choledochal cyst during a twin pregnancy A. Kaponis, Ch. Vitsas & G. O. Decavalas To cite this article: A. Kaponis, Ch. Vitsas & G. O. Decavalas (2015) Conservative management of a choledochal cyst during a twin pregnancy, Journal of Obstetrics and Gynaecology, 35:6, 646-647, DOI: 10.3109/01443615.2014.991293 To link to this article: http://dx.doi.org/10.3109/01443615.2014.991293

Published online: 29 Dec 2014.

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646  Obstetric Case Reports Declaration of interest:  The authors report no declarations of interest. The authors alone are responsible for the content and writing of the paper.­­­­

References

Chandru S, Srinivasan J, Roberts AD. 2007. Acute uterine cervical prolapsed in pregnancy. Journal of Obstetrics & Gynaecology 27:423–424. Daskalakis G, Lymberopoulos E, Anastasakis E, et  al. 2007. Uterine prolapse complicating pregnancy. Archives of Gynecology and Obstetrics 276: 391–392. Eddib A, Allaf MB, Lele A. 2010. Pregnancy in a woman with uterine procidentia: a case report. Journal of Reproductive Medicine 55:67–70. Lau S, Rijhsinghani A. 2008. Extensive cervical prolapse during labour. Journal of Reproductive Medicine 53:67–69 Mohamed-Suphan N, Ng RK. 2012. Uterine prolapse complicating pregnancy and labor: a case report and literature review. International Urogynecology Journal 23:647–650.

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Poor neonatal adaptation following in-utero exposure to quetiapine and lamotrigine C. Lau1, H. Watson2 & J. Cheong2,3,4 1Department of Pharmacy, Royal Women’s Hospital, Parkville,

Australia, 2Neonatal Services, Royal Women’s Hospital, Parkville, Australia, 3Victorian Infant Brain Studies, Murdoch Children’s Research Institute, Parkville, Australia, 4Department of Obstetrics & Gynaecology, University of Melbourne, Parkville, Australia DOI: 10.3109/01443615.2014.991292 Correspondence: Charis Lau, Department of Pharmacy Level 1, Royal Women’s Hospital, Corner Grattan Street and Flemington Road, Parkville, Victoria 3052, Australia. E-mail: [email protected]

With psychotropic medicines used by an estimated 3.5% of pregnant women in the Western world, it is essential for clinicians to be aware of their possible effects on newborns (Kieviet et al. 2013). Poor neonatal adaptation (PNA) has been described in newborns following in-utero exposure to psychotropic medications and is characterised by jitteriness, abnormal muscle tone, tremors, sleeping and feeding difficulties, agitation and respiratory distress (Kieviet et  al. 2013). Polypharmacy is becoming increasingly common in the management of mental health disorders, and has been associated with an increased risk of PNA (Sadowski et al. 2013). We report a case of PNA following maternal use of quetiapine and lamotrigine during pregnancy. A 31-year-old Caucasian female with a history of bipolar disorder and hypothyroidism was treated with lamotrigine: 400 mg/day, quetiapine: 600 mg/day and thyroxine: 100 mg/day during pregnancy. At 30 weeks’ gestation, her lamotrigine dose was increased to 500 mg/ day. She was a smoker of 2 cigarettes per day during pregnancy, with no other substance use. Pregnancy progressed uneventfully and she decided antenatally not to breastfeed. A male infant weighing 3610 g was delivered by forceps-assisted delivery at term. Birth was complicated by shoulder dystocia and he received mask ventilation, chest compressions and 2 doses of intramuscular adrenaline as no heart rate and respiratory effort were noted. His Apgar scores were 0 at 1 min, 3 at 5 min and 3 at 10 min. He was assessed as having stage-1 (mild) hypoxic–ischaemic encephalopathy. Cranial ultrasound was normal, and clinically he had a mild subgaleal haematoma. Over the next 2 days, he demonstrated signs out of keeping with stage-1 hypoxic–ischaemic encephalopathy, with severe irritability and feeding difficulties which did not improve with analgesia (paracetamol: 15 mg/kg 6 hourly). On examination, he had normal movement of all 4 limbs but was generally tense. Given the maternal use of psychotropic medicines, phenobarbitone (10 mg/ kg loading dose, followed by 5 mg/kg daily) was given to treat possible withdrawal. Within 24 h of phenobarbitone administration, his

symptoms improved (as assessed using a modified Finnegan scoring tool) and phenobarbitone was ceased after 5 days. Infant levels of quetiapine and lamotrigine were not assayed before treatment with phenobarbitone as management was guided clinically. Symptoms of PNA following maternal use of antipsychotics usually develop within 48 h of birth and last for 2–6 days (Kieviet et al. 2013). A systematic review (search last updated July 2008) of antipsychotic use during pregnancy identified 227 cases of quetiapine use; PNA was not reported (Gentile 2010). Two subsequent case reports have also not described PNA (Grover and Madan 2012; Tenyi et al. 2013). Although PNA secondary to maternal antipsychotic use is well documented, there is less evidence for PNA resulting from use of quetiapine specifically. Quetiapine demonstrates less placental passage compared with other antipsychotics which may explain the apparent lower incidence of PNA (Gentile 2010). There is little information available to characterise PNA following in-utero exposure to lamotrigine; however, symptoms such as decreased muscle tone, poor feeding and sedation have been described (Kieviet et al. 2013; Takcı et al. 2013). This case highlights the possibility of withdrawal in infants of mothers treated with quetiapine and/or lamotrigine, as early diagnosis can minimise unnecessary investigations and allow for more timely treatment if required. Declaration of interest:  The authors report no declarations of interest. The authors alone are responsible for the content and writing of the paper.

References Gentile S. 2010. Antipsychotic therapy during early and late pregnancy: a systematic review. Schizophrenia Bulletin 36:518–544. Grover S, Madan R. 2012. Successful use of quetiapine in two successive pregnancies. Journal of Neuropsychiatry and Clinical Neurosciences 24:E38. Kieviet N, Dolman KM, Honig A. 2013. The use of psychotropic medication during pregnancy: how about the newborn? Neuropsychiatric Disease and Treatment 9:1257–1266. Sadowski A, Todorow M, Yazdani Brojeni P et al. 2013. Pregnancy outcomes following maternal exposure to second-generation antipsychotics given with other psychotropic drugs: a cohort study. BMJ Open 3:e003062. doi:10.1136/bmjopen-2013-003062. Takcı Ş, Bayhan C, Çelik T et al. 2013. Hypotonia and poor feeding in an infant exposed to lamotrigine and valproic acid in utero. The Turkish Journal of Pediatrics 55:546–548. Tenyi T, Nagy A, Herold R et al. 2013. Extended release quetiapine fumarate and pregnancy. Neuropsychopharmacologia Hungarica 15:49–50.

Conservative management of a choledochal cyst during a twin pregnancy A. Kaponis, Ch. Vitsas & G. O. Decavalas Department of Obstetrics and Gynecology, Patras University School of Medicine, Greece DOI: 10.3109/01443615.2014.991293 Correspondence: Apostolos Kaponis, M.D., Department of Obstetrics and Gynecology, Patras University School of Medicine, Rio, 26504 Peloponnisos, Greece. Tel:  30-6972233270;  30-2613603893. E-mail: kaponisapostolos@ hotmail.com; [email protected]

Case report

We present the case of a 36-year-old woman, gravida 2, para 1, with a twin pregnancy at 26 weeks’ gestation, who was admitted to the outpatient clinic with clinical and laboratory manifestation of acute cholangitis. Clinical presentation included right-upper-quadrant pain, jaundice, fever up to 40o C with chills and uterine contrac-

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Obstetric Case Reports  647 tions. The pain radiated to the lumbus and the urine was hyperpigmented. Laboratory findings were white blood cell count of 24,700 cells/mm3, total bilirubin of 4.5 mg/dL, amylase of 375 U/L, alkaline phosphatase of 180 IU/L and gamma-glutamyl transpeptidase of 91 IU/L. Ultrasonography confirmed a twin, dichorionic, diamniotic pregnancy and revealed a cyst formation in the bile duct with a maximum diameter of 8 cm. A magnetic resonance cholangiopancreatogram (MRCP) revealed a cyst with a maximum diameter of 11.5 cm with dilated choledochal duct and several intra-hepatic biliary dilatations. Intra-venous meropenem and tocolysis with oxytocin receptor antagonist were administered. However, the maximum diameter of the choledochal duct was being increased and liver laboratory examinations continued to deteriorate. Percutaneous trans-choledochal cyst puncture was performed and a catheter was placed for external drainage under abdominal ultrasonography guidance. About 1800 mL of green bile was excreted from the cyst and about 700–800 mL of green bile was excreted daily. Her clinical condition and laboratory findings improved, gradually. The catheter’s position and the anatomy of the biliary tract was monitored periodically by ultrasonography. Her pregnancy progressed normally till the 36 weeks’ gestation when elective caesarean section was performed without complications and two healthy female babies were delivered. Post-partum, a percutaneous trans-hepatic cholangiogram confirmed the diagnosis of choledochal cyst type IVa (Figure 1). Four weeks post-partum, the patient underwent surgical excision of the choledochal cyst, cholecystectomy and formation of Rouxen-Y hepaticojejunostomy. Treatment of a cyst type IV could be difficult and patients often continue to have symptoms because of intra-hepatic disease (Singham et al. 2010). Pathological examination of the surgical specimen revealed typical features of choledochal cyst and chronic cholecystitis.

peritonitis, whereas foetal complications include pre-term labour and foetal demise (Wig et al. 1997). Abdominal ultrasound is the initial radiologic approach for evaluation of a choledochal cyst. The biliary tree, gallbladder, liver and pancreas are usually well visualised with ultrasound. There may be difficulties later in pregnancy due to distortion of the normal abdominal anatomy and gravid uterus. MRCP allows visualisation of the size and extent of choledochal cysts, and clearly demonstrates continuity between the cysts and the biliary tree. Magnetic resonance imaging and ultrasonography are particularly advantageous during pregnancy without exposing the mother and the growing foetus to ionising radiation (Shanley et al. 1994). Conservative management with puncture and drainage of the cyst is a reasonable approach till the attainment of foetal lung maturity. Percutaneous trans-hepatic drainage has been also performed successfully (Nasu et al. 2004). In uncomplicated cases, hydration, bowel rest and antibiotic treatment could be adequate to prevent clinical manifestation of acute cholangitis (Grotz et al. 1991). Postpartum, due to high risk of malignancy, surgical excision of the choledochal cyst is the only therapeutic option (Shi et al. 2001). Choledochal cysts can be a great challenge to the clinician with the prospective of a favourable maternal and foetal outcome. In the current case, although the patient was admitted with symptoms of acute cholangitis, conservative management of the cyst was adequate to maintain a twin pregnancy till the 36 weeks’ gestation. Therefore, this approach could be considered the first-line treatment for choledochal cyst’s management, even if complications are present.­

Discussion

References

Choledochal cysts are congenital cystic dilatations of the extra-hepatic or intra-hepatic portion of the biliary tree, with an incidence of 1:100,000–150,000 live births in Western countries to 1:1000 in Asian populations (Jablonska 2012). The diagnosis is, usually, made during childhood; however, 25% of cysts are diagnosed during adulthood (Conway et  al. 2009). Clinical symptoms during pregnancy are non-specific including intermittant abdominal pain, mass in the right-upper abdomen or jaundice. Pregnancy itself can provoke clinical manifestation of a previous asymptomatic choledochal cyst due to compression of the common bile duct and cyst by the gravid uterus and due to increased intra-abdominal pressure. Maternal complications can be cholangitis, pancreatitis and cystic rupture with biliary

Declaration of interest: The authors report no declarations of interest. The authors alone are responsible for the content and writing of the paper. Conway WC, Campos GM, Gagandeep S. 2009. Choledochal cyst during pregnancy: the patient’s first pregnancy was complicated by a congenital anomaly. American Journal of Obstetrics and Gynecology 200:588 e1–2. Grotz RL, Macdermid RG, Orlando R et al. 1991. Choledochal cyst diagnosed in pregnancy. Connecticut Medicine 55:262–266. Jablonska B. 2012. Biliary cysts: etiology, diagnosis and management. World Journal of Gastroenterology 18:4801–4810. Nasu K, Matsuki S, Kawano Y et al. 2004. Choledochal cyst diagnosed and conservatively treated during pregnancy. American Journal of Perinatology 8:463468. Shanley DJ, Gagliardi JA, Daum-Kowalski R. 1994. Choledochal cyst complicating pregnancy: antepartum diagnosis with MRI. Abdominal Imaging 19:61–63. Shi LB, Peng SY, Meng XK et  al. 2001. Diagnosis and treatment of congenital choledochal cyst: 20 years’ experience in China. World Journal of Gastroenterology 7:732–734. Singham J, Yoshida EM, Scudamore CH. 2010. Choledohal cysts. Part 3 of 3: Management. Canadian Medical Association 53:51–56. Wig JD, Goenka MK, Chawla YK et  al. 1997. Cholangitis secondary to choledochal cyst in pregnancy and puerperium. Journal of Clinical Gastroenterology 25:489–491.

Foetal axillary lymphangioma with ipsilateral pes equinovarus: Pitfalls in sonographic differential diagnosis (Axillary Lymphangioma & Pes Equinovarus) Yesim Bulbul Baytur, Burcu Artunc Ulkumen & Halil Gursoy Pala Figure 1. Percutaneous trans-hepatic cholangiogram performed post-partum showed dilated common bile duct and several intra-hepatic biliary dilatations. A: cyst formation, B: bile duct, C: right intra-hepatic biliary dilatations, D: left intrahepatic biliary dilatations, E: catheter, F: stomach, G: duodenum.

Division of Perinatology, Department of Obstetrics and Gynecology, Celal Bayar University School of Medicine, Manisa, Turkey­ DOI: 10.3109/01443615.2014.992872

Conservative management of a choledochal cyst during a twin pregnancy.

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