Central JSM Clinical Case Reports

Case Report *Corresponding author Papa Dasari, Senior Professor, Department of and Gynecology, JIPMER, Pondicherry, India, Tel: Hypoxic Ischemic 9442566883; Email: [email protected] Submitted: 29 June, 2020 Encephalopathy and Persistent Accepted: 02 August, 2020 Published: 04 August, 2020 Copyright © 2020 Dasari P, et al. Vegetative state in Obstetrics ISSN: 2373-9819 OPEN ACCESS Papa Dasari* and Haritha Sagili Department of Obstetrics and Gynecology, JIPMER, India Keywords • Severe gestational • Eclampsia Abstract • Cardiac arrest Background: Hypoxic ischemic encephalopathy (HIE) most commonly results due • Cerebral ischaemia to Cardiac arrest secondary to Cardiac diseases. Global ischemia occurs due to a drop • Hypoxic ischaemic encephalopathy in cerebral perfusion pressure and if the insult is for a brief period it results in selective neuronal damage and not brain . Persistent Vegetative state develops due to diffuse cortical, thalamic or combined neuronal loss with intact brain stem. This is rarely encountered in Obstetrics. It is important to diagnose HIE early and there is a great role for imaging in Arriving at the diagnosis so that prognosis can be explained to the family members. Case Series: The 3 women with HIE were in their 3rd decade of life and all 3 suffered from hypertension during and developed complications that led to cardiac arrest and were successfully revived. Though advances in resuscitation resulted in survival, persistent vegetative state occurred due to the resultant hypoxia to the brain. CT was normal but MRI -FLAIR showed the insult as evidenced by bilateral diffuse hyper-intensity of cerebral cortex and hyper-intensities along head of bilateral caudate nucleus, putamen and occipital lobes. Delayed termination of Pregnancy is the preventable factor identified. Mortality occurred due to secondary reasons in 2 of them after prolonged ICU stay. Conclusion: Early decision for termination of Pregnancy is the key for prevention of morbidity and mortality in women with severe hypertension developing complications. MRI (DWI) should be the neuroimaging of choice to find out the extent of brain damage and prognosticate.

INTRODUCTION Case 1 Hypoxic ischemic encephalopathy (HIE) most commonly Mrs. X 35 year old referred to our emergency services at 28 results due to Cardiac arrest secondary to Cardiac diseases. Global weeks of pregnancy as she had BP of 180/120 mmHg. She was ischemia occurs due to a drop in cerebral perfusion pressure and married for 15 years, conceived on 2nd cycle of IVF; morbidly if the insult is for a brief period it results in selective neuronal obese (120kg) received regular antenatal care at a private damage and not . Persistent Vegetative state develops hospital. She was diagnosed to have hypertension during her last due to diffuse cortical, thalamic or combined neuronal loss visit at 26 weeks of pregnancy and was treated with Tab. Aldomet with intact brain stem. This is rarely encountered in Obstetrics. (Alpha methyl dopa) 500mg 8th hourly and Tab, Frusemide20 It is important to diagnose HIE early and there is a great role mg once daily. She had no history of blurring of vision, epigastric, for imaging in arriving at the diagnosis so that prognosis can pain, and headache. Her past history included management for be explained to the family members. Case series of Vegetative infertility during which she underwent laparoscopic ovarian state following transient cardiac arrest in pregnant women are drilling and treatment for last 3 years on 50 µg reported as it is very rare and may be frequently encountered of levothyroxine. Her family history revealed that both parents in future with the recent advances in resuscitation techniques were suffering from Diabetes and Hypertension. On general resulting in increased survival of such women. examination, there was no palor and BP was 160/110 mm Hg. Respiratory and cardiovascular systems were normal. Knee jerk CASE SERIES was normal. Abdominal examination showed 28 week uterus and The main features are presented in table 1 and description is clinically FHS not localized due to obesity and abdominal wall given below. edema. USG revealed a single live in breech presentation with

Cite this article: Dasari P, Sagili H (2020) Hypoxic Ischemic Encephalopathy and Persistent Vegetative state in Obstetrics. JSM Clin Case Rep 8(2): 1178. Dasari P, et al. (2020) Central

Table 1: Clinical Presentation, Diagnosis and outcome of women with HIE. MRS.G MRS. G MRS.K Characteristics (April 2015) (September 2015) (August 2015) AGE 35 yrs 36 yrs 32 yrs Obstetric Status Primigravida G3 P1 L1 A1 ; Prior LSCS G4P3L2 Morbid Obesity (120 kg) Pre-Conception Hypothyroidism-3 yrs Hypothyroidism-4 yrs NIL Morbidity infertility(15yrs); IVF Chr.Hypertension-31/2 yrs conception Tertiary Care Centre; Hospitalization Antenatal Care Private Nursing Home for Primary Health Centre BP control at 25 weeks GDM-28 weeks Hypertension-26 weeks Hypertension; Abruption Post-Conceptional Morbidity 34 weeks Imminent features GDM-28 weeks Em. LSCS; Post-Partum Eclampsia HELLP Syndrome At Delivery & 30 weeks 35 weeks term Neonatal Outcome 1 kg Alive Apgar 8/10-NICU Alive ; Apgar 9/10 --NICU IUD Hypertensive crisis Cardiac arrest on arrival Status At Admission Imminent eclampsia Imminent Eclampsia GCS – E1 V1 M3 Prophylactic MgSo4 ABG-pH:7.433 Conservative Mx-6 days Intensive care Eclampsia room Em .CS with Sterilisation Emergency. LSCS Management Prior To The Event Labetalol withheld; hyponatraemia, hyperkalaemia AKI; Magnesium sulphate Standard protocol not followed Antihypertensive withheld as BP was Transport problem normal Conservative management Events Leading To -13days Haemodialysis Postoperative Day-4 Eclampsia Cardiac Arrest Inj. Labetalol stopped Hypertensive crisis MgSO4 Pulmonary oedema CPR Management After CPR pH:7.1 CPR The Cardiac Event pH:6.9 Emergency. LSCS after 48 hrs Diffuse cerebral oedema Neuro-Imaging PRES HIE (Fig 1-5) (Fig 6&7) Hyponatraemia Hypoglycaemia NIL Uraemia Sheehan’s Syndrome hyperkalaemia Resp. &Metabolic alkalosis Metabolic Profile DVT prophylaxis Hypertensive crisis Ionotropes; Ventilation; Late postpartum eclampsia VAP (Ventilation Associated 30 days ; ventilator; VAP Ventilation; VAP(Ventilation Pneumonia) ICU Care & Events tracheostomy Associated Pneumonia) Tracheostomy CCU Stay:42 days Tracheostomy; stay: 58 days Recc. Hypoglycaemia CCU Stay 21 days

Supportive Care Ileostomy Gastrostomy Gastrostomy Short Term -Outcome HIE HIE HIE; Sheehan's Syndrome Persistent vegetative state Persistent vegetative state Persistent Vegetative State Long Term Outcome Living Death later at hospital-Aspiration; and Death later at home Hospital stay 55 days hypoglycaemia

EFW (Estimated Fetal Weight) of 1kg and AFI was 5. Umbilical management she developed hypertensive crisis again and had artery PI was 1.2. It was decided to manage conservatively. She features of pulmonary oedema with acute onset dyspnoea. was given prophylactic magnesium sulphate for 24 hours and Labetalol was stopped due to pulse rate of 60 /min and oral Tab Labetalol 100 mg 12 hourly. Her Investigations were normal Lasix was only continued. She was in ICU and after 24 hours except for platelets which were 1.5 lakhs and few crenated she suddenly became comatosed and her pulmonary edema RBCs on peripheral smear. Fundus examination showed Grade worsened and sustained cardiac arrest at 4.30 am. Heart rate was I hypertensive Retinopathy and GDM (Gestational Diabetes 30 per minute after chest compressions and SpO2 24% and BP Mellitus) was diagnosed as single as 1st hour value was 190 not recordable. She was given injection atropine and adrenaline mg%. Her BP was controlled and it was maintained between 150 and successfully revived and BP has shot up to 190/120mm Hg /90mm Hg and 140/100 mmHg. After 13 days of conservative with tachycardia. She was treated with injection mannitol as

JSM Clin Case Rep 8(2): 1178 (2020) 2/5 Dasari P, et al. (2020) Central per Neurosurgery opinion and injection Magnesium sulphate and was on ventilator SIMV mode. ABG showed PH of 7.1pCO2- 48.9, pO2-193.2, O2sat-99% Na+/K+ - 104/3.72, Calcium 0.46 mmol/L. Corrections were undertaken she developed decerebrate rigidity and neurology opinion sought. Neurological examination revealed GCS: E3M1VT. Pupillary reaction reaction were of extensor. A provisional diagnosis of ICH or HIE was made and– sluggish, CT revealed Doll’s eye only reflex features + All DTR’s of cerebral – absent, edema. bilateral Emergency plantars LSCS done on day 3 of the event of cardiac arrest at 30+1 day. An alive fetus with Apgar of 8/10 was delivered weighed 1 kg. Atonic PPH managed medically and she developed hypertensive crisis again BP was 216/126 managed with intravenous labetalol and magnesium sulphate .GCS: E4VTM5 after 6 hours of LSCS and she was shifted to RICU. Started on DVT Prophylaxis and underwent Figure 1 AXIAL FLAIR -Fluid Attenuation and Inversion Recovery. antibiotics for ventilator associated pneumonia. Later required NTGtracheostomy (Nitroglycerine) on 7th infusion postoperative for control day andof hypertensive received specific crisis and later on 3 antihypertensives. She underwent ileostomy and recovered after 60 days and was discharged. Neurological evaluation at the time of discharge was as follows. GCS-E4M3V1 pupils were reactive to light. Decerebrate postures, Deep Tendon

After 2 weeks of discharge she expired at home. Reflexes were diminished, bilateral plantar response-withdrawal. Case 2 Mrs. G, 36 years Gravida 3 para 1, abortion1, previous LSCS who was a known case of hypothyroidism and hypertension was admitted for control of BP at 25 weeks of gestation. Developed GDM at 28 weeks and HELLP syndrome and imminent features of eclampsia at 34 weeks but conservatively managed for a week and emergency LSCS with sterilization was done at 35 weeks. Postoperatively antihypertensives were not given. Developed hyponatraemia, hyperkalaemia and AKI (Acute Kidney Injury). Developed hypertensive crisis on 4th Postoperative day and Figure 2 T2 Weighted coronal images shows diffuse hyper intensity of bilateral required hemodialysis but sustained cardiac arrest during cerebral cortex and basal ganglia with effacement of cerebral sulci. haemodialysis PH was 6.9 revived. On prolonged ventilation for 30 days. Underwent Tracheostomy, and gastrostomy. ICU stay therapy was given. Magnesium sulphate was withheld and started was 42 days and she developed VAP and treated for the same. on Ionotropes. By 2 hours her BP was normalised but her GCS Neuroimaging (Figure 1 & 2). Short term outcome was HIE 3 did not improve. Fundus examination did not show papilledema. and long term outcome was persistent vegetative state when Her investigations revealed moderate anaemia for which she discharged.

Case 3 mcg/dl,received Prolactin 2 packed – cell 21.32 transfusions. ng/ml ;CSF Her – glucose freeT3 -54 - 0.43(mIU/l) mg/dl, Protein (↓), freeT4 – 0.26(mIU/l) (↓), TSH was– 1.84 (mIU/l) ; S. Cortisol 6.35 Para 4 live 2, post LSCS day 1 was refereed as a case of -75 mg/dl, Cl-112 meq/l, No WBC; Spot Urine Protein was 6.5mg/ postpartum eclampsia after administration of magnesium dl. After 48 hours, Neurological assessment reported E2 M1 sulphate. She underwent LSCS for abruption IUD (Intrauterine Death) due to hypertension in a peripheral health centre. Her Plantar extensor, Decorticate extensor posturing. Her blood M3. Pupils sluggishly reacting to light, Knee reflex exaggerated, previous pregnancy was also IUD and macrosomic baby of 4.2 culture and CSF Culture were sterile and tracheal aspirate grew kg delivered normally. She was given phenytoin, mannitol at acenetobacter boumani resistant to all drugs and urine culture the time of seizure. On arrival to Tertiary care centre sustained grew E. coli moderately sensitive to nitrofurantoin and resistant Cardiac arrest and was resuscitated. GCS- E1 V1 M3 and was to all other drugs. She underwent Recurrent hypoglycemic transferred to ICU and was put on ventilator. ABG (Arterial Blood attacks called for endocrinology opinion and disgnosis of Sheehans syndrome was established. She was gradually weaned Gases) showed PH of 7.4 and blood glucose was 15 gm% and was administered 25% dextrose and maintained with 5% dextrose. off the ventilator and shifted to the dept. of OBG , ICU (Intensive BP was 90/60 mmHg. Central line was secured and goal directed Care Unit) and was ambulating and taking feeds orally but unable

JSM Clin Case Rep 8(2): 1178 (2020) 3/5 Dasari P, et al. (2020) Central to speak and recognize the relatives. She developed recurrent episodes of hyponatraemia and hypoglycaemia and sustained cardiorespiratory arrest in the middle of night 2 am and died of aspiration. DISCUSSION Cardiac arrest during Pregnancy is a rare event (1 in 33,000 births) and results in high maternal and perinatal mortality and only 15% of women survive. The most common cause is pulmonary embolism and hypertensive disorders are reported to be responsible for 8%. Diminished maternal physiological reserve is responsible for rapid maternal deterioration and concern for fetus usually alters the decision for termination of was at 28 weeks of gestation, conception following IVF (Invitro- fertilization)Pregnancy [1]. leading This is to what substandard has happened care in in the the management first case who of Hypertensive emergency ultimately resulted in cardiac arrest. In Figure 3 Plain CT – Normal. the second case also there was a delay in termination of pregnancy beyond 34 weeks. The third women suffered from complications of hypertension, abruption, PPH (Postpartum Hemorrhage), shock and cardiac arrest. HIE is usually diagnosed by history and clinical examination but there is great role for neuroimaging in recent years. Different regions of the grey matter may be affected in HIE. These include isolated cortical injury, isolated deep grey nuclei injury (including caudate nucleus, putamen, and thalamus), and mixed cortical and deep grey nuclei involvement. Among the cortical injuries the Rolandic (precentral and postcentral),medial occipital, and parietal cortices are more frequent sites often sparing the frontal and temporal cortices. The mixed pattern of injury involving both cortex and deep grey nuclei often correlates with the most severe brain injury of postcardiac arrest survivors and is likely predictive of an unfavorable outcome. Other sites that may be involved include the cerebellum, pons, and the subcortical white matter. These are associated with poor chances Figure 4 Contrast CT –Normal. of recovery [2]. This was true when applied to all these 3 women. CT is generally the initial imaging study performed when Early detection of brain damage is essential to guide the management and predict long term prognosis Clinical effacement of the CSF-containing spaces, decreased cortical gray improvement appears to be associated with a decrease in matterbrain injury attenuation is suspected. with loss CT of findings normal include gray-white diffuse differentiation, edema with the extent of signal intensity abnormalities , but residual and decreased bilateral basal ganglia attenuation and are not abnormalities can persist beyond 18 months Surviving patients very sensitive to pick up the damage3. Plain and contrast CT were may go on to develop diffuse atrophy at follow-up imaging. The normal (Figure 3&4) in Mrs K. survivor of these series showed the same on follow up. Diffusion-weighted MR imaging is the earliest imaging Cerebral edema is the cause for adverse outcomes in women with hypertensive crisis and eclampsia and it may be vasogenic or cytotoxic and cerebral edema alone will not produce severe diffusion-weightedmodality to become imaging positive, may usually demonstrate within the increased first few signalhours neurological manifestations until intracranial pressure increases intensityafter a hypoxic-ischemicin the cerebellar hemispheres, event. During basal the ganglia, first or 24 cerebral hours, the levels that result in cerebral ischaemia. Altered Consciousness, cortex in particular, the perirolandic and occipital [2,3]. Figures abnormal breathing patterns, bradycardia, inequal pupil size, 5 to 7 show the changes detected by using MRI Flair in Mrs K . extensor plantar should raise the suspicion of cerebral oedema. Cerebral edema develops soon after ischaemic stokes and peaks can occur after the insult can be acute, subacute and chronic phase at 26 -94 hours. It causes compression of the adjacent areas and Figure 6&7 show the findings of MRI in MrsPlain G. The and changes contrast that CT worsens the ischaemic stoke and causes rapid deterioration and were normal (Figure 1&2) in Mrs K. death [4]. Hence anticipating and managing cerebral oedema and imaging findings vary accordingly [2].

JSM Clin Case Rep 8(2): 1178 (2020) 4/5 Dasari P, et al. (2020) Central

timely is important to prevent worsening of ischaemic stroke . Osmotherpay instituted early can prevent worsening of cerebral ischaemia due to cerebral edema. Instituting timely management is essential for good neurological outcome. A non- pregnant woman who sustained cardiac arrest and hypoxic ischaemic brain injury was managed optimally in addition with hypothermia and she was able to lead useful life later with the family as reported by Maxmilian and his collegue [5]. He later proved the effects of therapeutic hypothermia in 56 of comatosed survivors of whom 32% had good neurological [6]. CONCLUSION Figure 5 MRI FLAIR - AXIAL: Hyper intensities along head of bilateral caudate Early decision for termination of Pregnancy is the key for nucleus. prevention of morbidity and mortality in women with severe hypertension during Pregnancy. MRI (DWI) should be the

and prognosticate. neuroimaging of choice to find out the extent of brain damage REFERENCES 1. Rodrigues AA, Clode N, Graca LM. Cardiac arrest in pregnancy: Best Practices are needed. Acta Obstet Gynecol Port. 2014; 8: 164-168. 2. Bhatia M. Hypoxic ischaemic injury in adult’s post-cardiac arrest: A pictoral essay. European Society of Radiology. 3. Huang BY, Castillo M. Hypoxic Ischaemic Brain injury: Imaging

417-440. findings from birth to adulthood. Radiographics. 2008; 28: Figure 6 MRI FLAIR - AXIAL: Hyper intensities along head of bilateral Putamen. 4. SK Jha. Cerebral oedema and its management. MJAFI. 2003; 59: 326-331. 5. Mulder M, Geocadin RG. Hypoxic Ischaemic Encephalopathy in adults. Neurocritical Society Course Review. Chapter 13; 276- 299. 6. Mulder M, Gibbs HG, Smith SW, Stephen W Smith, Ramnik Dhaliwal, Nathaniel L Scott, Mark D Sprenkle, et al. Awakening and withdrawal of life-sustaining treatment in cardiac arrest survivors treated with therapeutic hypothermia. Crit Care Med. 2014; 42: 2493-2499.

Figure 7 MRI FLAIR - AXIAL: Hyper intensities along head of bilateral occipital lobes.

Cite this article Dasari P, Sagili H (2020) Hypoxic Ischemic Encephalopathy and Persistent Vegetative state in Obstetrics. JSM Clin Case Rep 8(2): 1178.

JSM Clin Case Rep 8(2): 1178 (2020) 5/5