- Email: [email protected]
Encephalitis due to ovarian dermoid
256
Letters to the Editor – Brief Communications / European Journal of Obstetrics & Gynecology and Reproductive Biology 194 (2015) 249–263
Dantrolene reversibly decreased the strength of the detrusor responses to acetylcholine and histamine in a guinea pig bladder [3]. Dantrolene has been shown to be neuroprotective by reducing neuronal apoptosis after spinal cord injury. Other potential modes of action include anti-inflammatory and antioxidant properties with improved voiding, once this drug acts on the external urethral sphincter relaxation [4]. Whatever the mode of action, it seems to depress the detrusor muscle of the bladder as it does the other smooth muscles [3]. Importantly while on Dantrolene regular monitoring of liver functions is imperative due to the small risk of hepatotoxicity [5]. Despite conflicting views on the effect of Dantrolene on the detrusor muscle, in our patient it appeared to work synergistically with an anti-muscarinic and a beta 3-receptor agonist. This combination therapy may prove to be beneficial for refractory NDO. The treatment duration is uncertain but is likely to be beneficial long term in keeping with experience with Dantrolene in the management of limb spasticity. References [1] De Murphy KP, Boutin SA, Ide KR. Cerebral palsy, neurogenic bladder, and outcomes of lifetime care. Med Child Neurol 2012;54:945–50. http:// dx.doi.org/10.1111/j.1469-8749.2012.04360.x. [2] Harrison JD, Benson GS. Effect of Dantrolene sodium on canine bladder contractility. Urology 1980;16:229–31. [3] Lograno MD, Conte-Camerino D, De Filippis F. The effect of sodium dantrolene on the detrusor urinae muscle of the bladder isolated from the guinea pigs. Boll Societa Ital Biol Sper 1979;55:484–8. [4] Torres B, Serakides R, Caldeira F, Gomes M, Melo E. The ameliorating effect of dantrolene on the morphology of urinary bladder in spinal cord injured rats. Pathol Res Pract 2011;207:775–9. http://dx.doi.org/10.1016/j.prp.2011.10.004. [5] Cornette M, Gillard C, Borlee-Hermans G. Fatal progressive liver disease after a week course of dantrolene. Acta Neurol Belg 1980;80:336–47.
Supriya Bulchandani* Philip Toozs-Hobson Birmingham Women’s NHS Foundation Trust, Birmingham, UK Angus Kennedy Chelsea and Westminster Hospital, London, UK Steve Sturman Queen Elizabeth University Hospital, Birmingham, UK *Corresponding author at: Birmingham Women’s NHS Foundation Trust, Mindelsohn Way, Edgbaston, Birmingham B15 2TG, UK. Tel.: +44 121 472 1377 E-mail address: [email protected] (S. Bulchandani). 14 June 2015 http://dx.doi.org/10.1016/j.ejogrb.2015.08.012
Encephalitis due to ovarian dermoid Dear Editors, A 32 years woman was admitted in a small hospital as she had insomnia, headaches and convulsions for 15 days. When she developed additional symptoms like irrelevant talk, psychiatric disturbances and drowsiness, she was transferred to a tertiary centre, private hospital in Mumbai, under the care of a neurophysician (AS). The patient’s Glasgow coma scale was 10/ 15. She was treated with IV immunoglobulins; Plasma exchange
and plasmapheresis were also carried out. Routine investigations for encephalitis and MRI of the brain were normal. Searching for an unusual cause to explain her neurological status, serum N-methylD-aspartate (NMDA) receptor antibody test was done first carried out. This was positive and led to a diagnosis of Paraneoplastic Limbic Encephalitis (PLE) [1]. Since PLE is known to be associated with ovarian dermoid, transvaginal-sonography was done. Sonography revealed a 4 3 cm dermoid cyst in the right ovary which was confirmed by MRI scan. She was then referred to a gynaecologist (SSS) for an opinion. He advised Laparoscopic right salpingo-oophorectomy. By this time, the patient had become unconscious and was on ventilatory support with a tracheostomy. For surgery, General anaesthesia was induced with fentanyl (1 mcg/kg), midazolam (0.05 mg/kg) and propofol (2–3 mg/kg) and atracurium (35 mg) and then maintained with oxygen (1 L/ min) and compressed air (1.5 L/min), isoflurane (1.5–2%) and fentanyl (0.03 mcg/kg). Neuromuscular blockade was maintained with atracurium. Laparoscopic removal of the right ovarian dermoid cyst was done (SSS-KP) by right salpingo-oophorectomy. On gross appearance, the ovarian dermoid specimen contained lipid, hair tuft and a tooth. The Uterus, left fallopian tube and left ovary as well as the rest of the abdomen and pelvis were normal. Histopathology confirmed the diagnosis of a mature teratoma. Her neurological symptoms gradually improved after the tumour was removed. She was in the Intensive Care Unit (ICU) for totally 32 days including ventilatory support for 10 days before it was withdrawn and tracheostomy was closed after 13 days. She was discharged from the hospital after total stay of 36 days with necessary instructions. NMDA receptor antibodies directed against the NMDA receptor affects the nervous system in young women, who present with subacute encephalopathy which is characterised by psychosis, movement disorder and refractory siezures, Autoimmune encephalitis manifests with psychiatric symptoms and inflammatory CSF abnormalities [2,3]. Nearly 90% of cases of anti-NMDA receptor encephalitis occur in women, with a median age of 23. The male: female ratio of this disease is 9:91 [3]. Teratoma cells produce anti NMDA receptor antibodies against limbic brain receptors leading to NMDA receptor antibody positivity which is confirmed by the detection of antibodies to NR1/NR2 heteromers of NMDA in serum and CSF [4]. Out of 100 patients with anti-NMDA receptor encephalitis, who had received early treatment, Dalmau [3] reports that 75 recovered or had mild deficits and 25 had severe deficits or died. Various anaesthetic drugs like ketamine, propofol, opiods like methadone and inhaled agents like nitrous oxide, sevoflurane, xenon behave unpredictably in the presence of NMDA-R. Ketamine binds to the phencyclidine site of the ion channel of NMDA-R, acting as an antagonist to inhibit the influx of sodium and calcium [5], causing similar clinical features as the disease itself – such as hallucinations, psychosis, and tachycardia. Anaesthesia using nondepolarizing neuromuscular blocking agents, benzodiazepines and opioids should preferably be used in such patients as they do not interact with the NMDA pathway. The Lesson in this case for the neurophysician or a general physician is that abdomino-pelvic sonography should be considered in a woman with the encephalitis, which cannot be otherwise explained. Identifying a gynaecological cause of the neurological condition in a young woman and prompt treatment of it leads to improvement in clinical condition and can be life saving. Though rarely, ovarian teratoma (Dermoid) can cause serious illness and not only be restricted to pathology in the pelvis. The prime mover is a neurophysician in recognizing that dermoids can cause much more than pathology in the pelvis and crux lies in advising
Letters to the Editor – Brief Communications / European Journal of Obstetrics & Gynecology and Reproductive Biology 194 (2015) 249–263
abdominal-pelvic sonography after positive CSF NMDA receptor antibody test. Conflicts of interests No author has any potential conflicts of interest. Contribution to authorship All authors equally participated in writing the article and operating on patient. References [1] Gultekin SH, Rosenfeld MR, Voltz R, Eichen J, Posner JB, Dalmau J. Paraneoplastic limbic encephalitis: neurological symptoms, immunological findings and tumour association in 50 patients. Brain 2000;123:1481–94. [2] Vincent A, Buckley C, Schott JM, et al. Potassium channel antibody-associated encephalopathy: a potentially immunotherapy-responsive form of limbic encephalitis. Brain 2004;127:701–12. [3] Dalmau J, Gleichman AJ, Hughes EG, et al. Anti-NMDA-receptor encephalitis: case series and analysis of the effects of antibodies. Lancet Neurol 2008;7:1091–8. [4] Pru¨ss H, Dalmau J, Harms L, et al. Retrospective analysis of NMDA receptor antibodies in encephalitis of unknown origin. Neurology 2010;75: 1735–9. [5] Anis NA, Berry SC, Burton NR, Lodge D. The dissociative anaesthetics, ketamine and phencyclidine, selectively reduce excitation of central mammalian neurons by N-methyl-aspartate. Br J Clin Pharmacol 1983;79:565–75.
Arun Shaha,b,c TN Medical College, Mumbai, India b Saifee Hospital, Mumbai, India c Breach Candy Hospital, Mumbai, India a
Shirish Shetha,b,c,d Breach Candy Hospital, Mumbai, India b Saifee Hospital, Mumbai, India c Sir Hurkisondas Hospital, Mumbai, India d Sheth Maternity and Gynecological Nursing Home, Mumbai, India
highlighting the complexity of managing pregnancy after uterine surgery. A 39-year-old lady was diagnosed with an incomplete miscarriage. She underwent surgical management of miscarriage and histology confirmed products of conception. Two months later, she conceived again. She had a transvaginal ultrasound scan which demonstrated a left interstitial ectopic pregnancy. She underwent a laparoscopy, which confirmed a left interstitial ectopic pregnancy. A laparoscopic wedge resection and left salpingectomy were performed. Repair of the defect was in three layers with no1 polysorb sutures (PolysorbTM, Covidien, UK). Estimated blood loss was 200 ml and she had an uncomplicated post-operative recovery. She was counselled about the risks of recurrent ectopic pregnancy (10%) and advised to wait three–six months before conceiving (Fig. 1). She conceived again within six months. Antenatal care was uncomplicated and ultrasound scans showed a left lateral placenta. Despite having previously delivered vaginally, she was advised to deliver by elective Caesarean section in view of previous uterine surgery. After delivery of the placenta by controlled cord traction, a defect in the left cornu of the uterus was identified when checking the cavity. The uterus was exteriorized and placenta percreta was noted, with a defect in the uterine wall and decidualisation of the uterine serosa. This was repaired continuously with no1 vicrylTM. Estimated blood loss was 800 ml. In the absence of histological confirmation from Caesarean hysterectomy, placenta percreta was clinically diagnosed during surgery, based on the uterine wall defect with decidualisation of surrounding uterine serosa. It is acknowledged that the incidence of clinically defined placenta accreta/percreta is seen more frequently than when the disorder is pathologically defined, as not every case will proceed to hysterectomy. Furthermore, in
a
Kurush Paghdiwallaa,b Saifee Hospital, Mumbai, India b Breach Candy Hospital, Mumbai, India a
Dipti Shende* Sheth Maternity and Gynecological Nursing Home, Mumbai, India M. Aslam Saifee Hospital, Mumbai, India *Corresponding author at: 2/2, Navjivan Society, Lamington road, Mumbai 400008, India. Tel.: +91 9022052483 E-mail address: [email protected] (D. Shende).
6 February 2015 http://dx.doi.org/10.1016/j.ejogrb.2015.08.002
Placenta percreta after laparoscopic excision of intersitial ectopic pregnancy: a case for elective caesarean section after cornual resection? Dear Editors, We present a case of undiagnosed placenta percreta after laparoscopic management of interstitial ectopic pregnancy,
257
Fig. 1. Placenta percreta at Caesarean section.
Letters to the Editor – Brief Communications / European Journal of Obstetrics & Gynecology and Reproductive Biology 194 (2015) 249–263
Dantrolene reversibly decreased the strength of the detrusor responses to acetylcholine and histamine in a guinea pig bladder [3]. Dantrolene has been shown to be neuroprotective by reducing neuronal apoptosis after spinal cord injury. Other potential modes of action include anti-inflammatory and antioxidant properties with improved voiding, once this drug acts on the external urethral sphincter relaxation [4]. Whatever the mode of action, it seems to depress the detrusor muscle of the bladder as it does the other smooth muscles [3]. Importantly while on Dantrolene regular monitoring of liver functions is imperative due to the small risk of hepatotoxicity [5]. Despite conflicting views on the effect of Dantrolene on the detrusor muscle, in our patient it appeared to work synergistically with an anti-muscarinic and a beta 3-receptor agonist. This combination therapy may prove to be beneficial for refractory NDO. The treatment duration is uncertain but is likely to be beneficial long term in keeping with experience with Dantrolene in the management of limb spasticity. References [1] De Murphy KP, Boutin SA, Ide KR. Cerebral palsy, neurogenic bladder, and outcomes of lifetime care. Med Child Neurol 2012;54:945–50. http:// dx.doi.org/10.1111/j.1469-8749.2012.04360.x. [2] Harrison JD, Benson GS. Effect of Dantrolene sodium on canine bladder contractility. Urology 1980;16:229–31. [3] Lograno MD, Conte-Camerino D, De Filippis F. The effect of sodium dantrolene on the detrusor urinae muscle of the bladder isolated from the guinea pigs. Boll Societa Ital Biol Sper 1979;55:484–8. [4] Torres B, Serakides R, Caldeira F, Gomes M, Melo E. The ameliorating effect of dantrolene on the morphology of urinary bladder in spinal cord injured rats. Pathol Res Pract 2011;207:775–9. http://dx.doi.org/10.1016/j.prp.2011.10.004. [5] Cornette M, Gillard C, Borlee-Hermans G. Fatal progressive liver disease after a week course of dantrolene. Acta Neurol Belg 1980;80:336–47.
Supriya Bulchandani* Philip Toozs-Hobson Birmingham Women’s NHS Foundation Trust, Birmingham, UK Angus Kennedy Chelsea and Westminster Hospital, London, UK Steve Sturman Queen Elizabeth University Hospital, Birmingham, UK *Corresponding author at: Birmingham Women’s NHS Foundation Trust, Mindelsohn Way, Edgbaston, Birmingham B15 2TG, UK. Tel.: +44 121 472 1377 E-mail address: [email protected] (S. Bulchandani). 14 June 2015 http://dx.doi.org/10.1016/j.ejogrb.2015.08.012
Encephalitis due to ovarian dermoid Dear Editors, A 32 years woman was admitted in a small hospital as she had insomnia, headaches and convulsions for 15 days. When she developed additional symptoms like irrelevant talk, psychiatric disturbances and drowsiness, she was transferred to a tertiary centre, private hospital in Mumbai, under the care of a neurophysician (AS). The patient’s Glasgow coma scale was 10/ 15. She was treated with IV immunoglobulins; Plasma exchange
and plasmapheresis were also carried out. Routine investigations for encephalitis and MRI of the brain were normal. Searching for an unusual cause to explain her neurological status, serum N-methylD-aspartate (NMDA) receptor antibody test was done first carried out. This was positive and led to a diagnosis of Paraneoplastic Limbic Encephalitis (PLE) [1]. Since PLE is known to be associated with ovarian dermoid, transvaginal-sonography was done. Sonography revealed a 4 3 cm dermoid cyst in the right ovary which was confirmed by MRI scan. She was then referred to a gynaecologist (SSS) for an opinion. He advised Laparoscopic right salpingo-oophorectomy. By this time, the patient had become unconscious and was on ventilatory support with a tracheostomy. For surgery, General anaesthesia was induced with fentanyl (1 mcg/kg), midazolam (0.05 mg/kg) and propofol (2–3 mg/kg) and atracurium (35 mg) and then maintained with oxygen (1 L/ min) and compressed air (1.5 L/min), isoflurane (1.5–2%) and fentanyl (0.03 mcg/kg). Neuromuscular blockade was maintained with atracurium. Laparoscopic removal of the right ovarian dermoid cyst was done (SSS-KP) by right salpingo-oophorectomy. On gross appearance, the ovarian dermoid specimen contained lipid, hair tuft and a tooth. The Uterus, left fallopian tube and left ovary as well as the rest of the abdomen and pelvis were normal. Histopathology confirmed the diagnosis of a mature teratoma. Her neurological symptoms gradually improved after the tumour was removed. She was in the Intensive Care Unit (ICU) for totally 32 days including ventilatory support for 10 days before it was withdrawn and tracheostomy was closed after 13 days. She was discharged from the hospital after total stay of 36 days with necessary instructions. NMDA receptor antibodies directed against the NMDA receptor affects the nervous system in young women, who present with subacute encephalopathy which is characterised by psychosis, movement disorder and refractory siezures, Autoimmune encephalitis manifests with psychiatric symptoms and inflammatory CSF abnormalities [2,3]. Nearly 90% of cases of anti-NMDA receptor encephalitis occur in women, with a median age of 23. The male: female ratio of this disease is 9:91 [3]. Teratoma cells produce anti NMDA receptor antibodies against limbic brain receptors leading to NMDA receptor antibody positivity which is confirmed by the detection of antibodies to NR1/NR2 heteromers of NMDA in serum and CSF [4]. Out of 100 patients with anti-NMDA receptor encephalitis, who had received early treatment, Dalmau [3] reports that 75 recovered or had mild deficits and 25 had severe deficits or died. Various anaesthetic drugs like ketamine, propofol, opiods like methadone and inhaled agents like nitrous oxide, sevoflurane, xenon behave unpredictably in the presence of NMDA-R. Ketamine binds to the phencyclidine site of the ion channel of NMDA-R, acting as an antagonist to inhibit the influx of sodium and calcium [5], causing similar clinical features as the disease itself – such as hallucinations, psychosis, and tachycardia. Anaesthesia using nondepolarizing neuromuscular blocking agents, benzodiazepines and opioids should preferably be used in such patients as they do not interact with the NMDA pathway. The Lesson in this case for the neurophysician or a general physician is that abdomino-pelvic sonography should be considered in a woman with the encephalitis, which cannot be otherwise explained. Identifying a gynaecological cause of the neurological condition in a young woman and prompt treatment of it leads to improvement in clinical condition and can be life saving. Though rarely, ovarian teratoma (Dermoid) can cause serious illness and not only be restricted to pathology in the pelvis. The prime mover is a neurophysician in recognizing that dermoids can cause much more than pathology in the pelvis and crux lies in advising
Letters to the Editor – Brief Communications / European Journal of Obstetrics & Gynecology and Reproductive Biology 194 (2015) 249–263
abdominal-pelvic sonography after positive CSF NMDA receptor antibody test. Conflicts of interests No author has any potential conflicts of interest. Contribution to authorship All authors equally participated in writing the article and operating on patient. References [1] Gultekin SH, Rosenfeld MR, Voltz R, Eichen J, Posner JB, Dalmau J. Paraneoplastic limbic encephalitis: neurological symptoms, immunological findings and tumour association in 50 patients. Brain 2000;123:1481–94. [2] Vincent A, Buckley C, Schott JM, et al. Potassium channel antibody-associated encephalopathy: a potentially immunotherapy-responsive form of limbic encephalitis. Brain 2004;127:701–12. [3] Dalmau J, Gleichman AJ, Hughes EG, et al. Anti-NMDA-receptor encephalitis: case series and analysis of the effects of antibodies. Lancet Neurol 2008;7:1091–8. [4] Pru¨ss H, Dalmau J, Harms L, et al. Retrospective analysis of NMDA receptor antibodies in encephalitis of unknown origin. Neurology 2010;75: 1735–9. [5] Anis NA, Berry SC, Burton NR, Lodge D. The dissociative anaesthetics, ketamine and phencyclidine, selectively reduce excitation of central mammalian neurons by N-methyl-aspartate. Br J Clin Pharmacol 1983;79:565–75.
Arun Shaha,b,c TN Medical College, Mumbai, India b Saifee Hospital, Mumbai, India c Breach Candy Hospital, Mumbai, India a
Shirish Shetha,b,c,d Breach Candy Hospital, Mumbai, India b Saifee Hospital, Mumbai, India c Sir Hurkisondas Hospital, Mumbai, India d Sheth Maternity and Gynecological Nursing Home, Mumbai, India
highlighting the complexity of managing pregnancy after uterine surgery. A 39-year-old lady was diagnosed with an incomplete miscarriage. She underwent surgical management of miscarriage and histology confirmed products of conception. Two months later, she conceived again. She had a transvaginal ultrasound scan which demonstrated a left interstitial ectopic pregnancy. She underwent a laparoscopy, which confirmed a left interstitial ectopic pregnancy. A laparoscopic wedge resection and left salpingectomy were performed. Repair of the defect was in three layers with no1 polysorb sutures (PolysorbTM, Covidien, UK). Estimated blood loss was 200 ml and she had an uncomplicated post-operative recovery. She was counselled about the risks of recurrent ectopic pregnancy (10%) and advised to wait three–six months before conceiving (Fig. 1). She conceived again within six months. Antenatal care was uncomplicated and ultrasound scans showed a left lateral placenta. Despite having previously delivered vaginally, she was advised to deliver by elective Caesarean section in view of previous uterine surgery. After delivery of the placenta by controlled cord traction, a defect in the left cornu of the uterus was identified when checking the cavity. The uterus was exteriorized and placenta percreta was noted, with a defect in the uterine wall and decidualisation of the uterine serosa. This was repaired continuously with no1 vicrylTM. Estimated blood loss was 800 ml. In the absence of histological confirmation from Caesarean hysterectomy, placenta percreta was clinically diagnosed during surgery, based on the uterine wall defect with decidualisation of surrounding uterine serosa. It is acknowledged that the incidence of clinically defined placenta accreta/percreta is seen more frequently than when the disorder is pathologically defined, as not every case will proceed to hysterectomy. Furthermore, in
a
Kurush Paghdiwallaa,b Saifee Hospital, Mumbai, India b Breach Candy Hospital, Mumbai, India a
Dipti Shende* Sheth Maternity and Gynecological Nursing Home, Mumbai, India M. Aslam Saifee Hospital, Mumbai, India *Corresponding author at: 2/2, Navjivan Society, Lamington road, Mumbai 400008, India. Tel.: +91 9022052483 E-mail address: [email protected] (D. Shende).
6 February 2015 http://dx.doi.org/10.1016/j.ejogrb.2015.08.002
Placenta percreta after laparoscopic excision of intersitial ectopic pregnancy: a case for elective caesarean section after cornual resection? Dear Editors, We present a case of undiagnosed placenta percreta after laparoscopic management of interstitial ectopic pregnancy,
257
Fig. 1. Placenta percreta at Caesarean section.