The Open Neurology Journal




ISSN: 1874-205X ― Volume 13, 2019
CASE REPORT

Brain Tumor in Differential Diagnosis of Seizures in Puerperium: Case Report



Luíza Guazzelli Pezzali3, *, Gabriela Francoes Rostirolla1, 3, Carina Bauer Luiz3, Lúcio Brandão Gomes4, Raquel Camara Rivero5, 6, Edimárlei Gonsales Valério1, 2, 3, Janete Vettorazzi1, 2, 3
1 Postgraduate Program in Health Sciences: Gynecology and Obstetrics, Universidade Federal do Rio Grande do Sul (UFRGS), Porto Alegre, Brasil
2 Department of Gynecology and Obstetrics, Faculty of Medicine (FAMED), Universidade Federal do Rio Grande do Sul, Porto Alegre - Rio Grande do Sul (RS), Brasil
3 Service of Obstetrics and Gynecology, Hospital de Clínicas de Porto Alegre, Porto Alegre/RS, Brasil
4 Service of Neurosurgery, Hospital de Clínicas de Porto Alegre, Porto Alegre/RS, Brasil
5 Department of Pathology, Faculty of Medicine (FAMED), Universidade Federal do Rio Grande do Sul, Porto Alegre - Rio Grande do Sul (RS), Brasil
6 Service of Pathology, Hospital de Clínicas de Porto Alegre, Porto Alegre/RS, Brasil

Abstract

Convulsive crisis is a transient disturbance of cerebral function, and the etiology of which may be manifold. Its clarification is essential for establishing adequate therapy and seizure control. In the pregnancy-puerperal cycle, eclampsia is the most common cause of it, but brain tumor, epilepsy and other disorders are part of the differential diagnosis. We report a case of seizure triggered by tumor in a primigest, 22 years old, at 38 weeks’ gestation with premature rupture of membranes. Cesarean section was performed due to non-reassuring fetal condition. On the second day of puerperium, she presented recurrent episodes of generalized tonic-clonic seizures and elevated blood pressure. She received magnesium sulfate for 24 hours and persisted with severe headache. Magnetic resonance imaging of the brain showed an expansive lesion in the left frontoparietal region. The patient underwent intracranial microsurgery on the eighth day after cesarean section, with complete resection of the lesion and anatomopathological diagnosis of schwannoma. She went through good postoperative evolution, without neurological deficits, and stayed on phenobarbital, with no seizures until 6 months of follow-up. From the case we conclude that although the first diagnosis in pregnant or puerperal women with seizures should be eclampsia, there are other causes, such as brain tumor and Posterior Reversible Encephalopathy Syndrome (PRES). Careful evaluation is required, especially in refractory cases and not responding to usual treatment.

Keywords: Seizure, Puerperium, Brain tumor, Eclampsia, Case report, Cerebral function.


Article Information


Identifiers and Pagination:

Year: 2019
Volume: 13
First Page: 67
Last Page: 71
Publisher Id: TONEUJ-13-67
DOI: 10.2174/1874205X01913010067

Article History:

Received Date: 25/10/2018
Revision Received Date: 13/03/2019
Acceptance Date: 04/04/2019
Electronic publication date: 30/04/2019
Collection year: 2019

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© 2019 Pezzali et al.

open-access license: This is an open access article distributed under the terms of the Creative Commons Attribution 4.0 International Public License (CC-BY 4.0), a copy of which is available at: https://creativecommons.org/licenses/by/4.0/legalcode. This license permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.


* Address correspondence to this author at the Service of Obstetrics and Gynecology, Hospital de Clínicas de Porto Alegre, Porto Alegre/RS, Brasil;
E-mail: luizapezzali@gmail.com





1. INTRODUCTION

Neurological disorders manifested in pregnancy may be directly associated with it (pre-eclampsia, eclampsia) or may be related to preexisting conditions (epilepsy, multiple sclerosis, myasthenia gravis, brain tumor), which may be exacerbated during pregnancy and the puerperium due to physiological changes associated with this period [1Paula JCR, Aquino LO, Padua BJ, Leite HV, Cabral ACV, Brandão AHF. Neurological syndromes during pregnancy. Femina 2015; 43(3): 119-24.]. Convulsive crisis is a transient disturbance of cerebral function. Multiple factors can be involved in the etiology of this disorder, and its clarification is essential for establishing adequate therapy and seizure control. During the pregnancy-puerperal cycle, eclampsia is the most frequent cause of the first convulsive manifestation [2Beach RL, Kaplan PW. Seizures in pregnancy: diagnosis and management. Int Rev Neurobiol 2008; 83: 259-71.[http://dx.doi.org/10.1016/S0074-7742(08)00015-9] [PMID: 1892 9087] ]. Its incidence has declined over the years in areas with increased health care, which includes adequate prenatal care. The incidence rate reported in Latin America (0.14%) is approximately three times greater compared to the one found in the United States (range of 0.03% to 0,05%) [3Saftlas AF, Olson DR, Franks AL, Atrash HK, Pokras R. Epidemiology of preeclampsia and eclampsia in the United States, 1979-1986. Am J Obstet Gynecol 1990; 163(2): 460-5.[http://dx.doi.org/10.1016/0002-9378(90)91176-D] [PMID: 2386132] , 4Moura JL. Preeclampsia-eclampsia. Rev Latinoam Hipertens 2007; 2(4): 117-27.]. However, other causes of convulsive crisis should be investigated or excluded, especially in refractory cases that do not respond to usual treatment. Although the incidence of brain tumor does not increase during pregnancy, it may become symptomatic and voluminous due to water retention and hormonal action resulting from this period [5Simon RH. Brain tumors in pregnancy. Semin Neurol 1988; 8(3): 214-21.[http://dx.doi.org/10.1055/s-2008-1041380] [PMID: 3057552] , 6Michelsen JJ, New PF. Brain tumour and pregnancy. J Neurol Neurosurg Psychiatry 1969; 32(4): 305-7.[http://dx.doi.org/10.1136/jnnp.32.4.305] [PMID: 4980021] ]. This report aims to describe a patient with convulsive crisis in the puerperium triggered by brain tumor, occurred in Hospital de Clínicas de Porto Alegre (HCPA), public-private hospital linked to educational institution Federal University of Rio Grande do Sul (UFRGS).

2. CASE REPORT

A 22-year-old, primigest, caucasian, and obese (BMI: 36.3) woman was admitted at 38 weeks’ gestation due to premature rupture of membranes. On admission, normal and stable vital signs (blood pressure: 128/79 mmHg; heart rate: 80 bpm) were observed. During induction of labor with oxytocin, patient stayed assintomatic, evolving into cesarean delivery due to non-reassuring fetal condition. Patient presented convulsive crisis 26 hours after cesarean section, and laboratory test revealed urinary protein/creatinine ratio of 0.91. Patient was diagnosed with eclampsia, and received magnesium sulphate for 24 hours. She persisted with headache, and evaluation with anesthesiologist was performed due to suspicion of post-rachi headache. A new seizure was reported on the fourth day of hospitalization. The evolution of the puerperium is presented in Table 1.

Table 1
Evolution of the case in puerperium.


Fig. (1)
Brain MRI at 7th PO Caesarean - frontoparietal expansive lesion at left with perilesional edema, probable neoplasia.


Fig. (2)
Intracranial Microsurgery.


Fig. (3)
Brain MRI at 8th month of follow up.


Fig. (4)
Nuclear palisade. HE 200X.


Fig. (5)
Antoni A area with Verocay Bodies. HE 50X.


Fig. (6)
CD34 positivity. Immunohistochemistry 200X.


Pathology identified an encapsulated lesion composed entirely of well-differentiated Schwann cells, with eosinophilic cytoplasm, indistinct cell membranes and tapered nuclei. Areas of compact spindle cells (Antoni A) with nuclear palisading and Verocay Bodies were alternated with hypocellular less orderly areas (Antoni B) (Figs. 4 and 5). Mitoses were not observed. The blood vessels have hyalinized walls. The lesion presented SOX-10 and CD34 immunoreactivity (Fig. 6).

At follow-up of 6 months, patient had no neurological deficits or new generalized tonic-clonic seizures in the use of phenobarbital. Brain MRI at 8th month of follow up showed complete remission of the lesion (Fig. 3).

3. DISCUSSION

Clinical manifestation of neurological diseases is often reported during gestation and puerperium due to physiological changes observed in these periods. It is necessary to understand those conditions in order to better assist their diagnosis and treatment. For example, increasing levels of procoagulant factors and fibrinogen in contrast to decreased production of fibrinolytic agents leads to a state of hypercoagulability, which results in a greater risk of venous thromboembolism. Other changes related to renal clearance and hepatic metabolism also influence some drugs pharmacokinetics, such as anticonvu-lsants, and may lead to an increase in the number of seizures if there is no dose adjustment [1Paula JCR, Aquino LO, Padua BJ, Leite HV, Cabral ACV, Brandão AHF. Neurological syndromes during pregnancy. Femina 2015; 43(3): 119-24., 7Haider B, von Oertzen J. Neurological disorders. Best Pract Res Clin Obstet Gynaecol 2013; 27(6): 867-75.[http://dx.doi.org/10.1016/j.bpobgyn.2013.07.007] [PMID: 23988426] ].

In addition to preexisting diseases that may be exacerbated in the pregnancy-puerperal period, Eclampsia, a pathology directly related to pregnancy, is usually the first diagnosis to be considered in a pregnant or puerperal woman who presents with acute neurological symptoms [8Edlow JA, Caplan LR, O’Brien K, Tibbles CD. Diagnosis of acute neurological emergencies in pregnant and post-partum women. Lancet Neurol 2013; 12(2): 175-85.[http://dx.doi.org/10.1016/S1474-4422(12)70306-X] [PMID: 2333 2362] ]. A detailed anamnesis of the patient's symptoms is essential to support the diagnostic investigation.

Headache is the most common neurological symptom in pregnancy. Tension and migraine are the most frequent types among pregnant and postpartum women [8Edlow JA, Caplan LR, O’Brien K, Tibbles CD. Diagnosis of acute neurological emergencies in pregnant and post-partum women. Lancet Neurol 2013; 12(2): 175-85.[http://dx.doi.org/10.1016/S1474-4422(12)70306-X] [PMID: 2333 2362] ]. The patient reported in this case is among the 40% who present this symptom in the first week of puerperium [9Goldszmidt E, Kern R, Chaput A, Macarthur A. The incidence and etiology of postpartum headaches: a prospective cohort study. Can J Anaesth 2005; 52(9): 971-7.[http://dx.doi.org/10.1007/BF03022061] [PMID: 16251565] -11Von Wald T, Walling AD. Headache during pregnancy. Obstet Gynecol Surv 2002; 57(3): 179-85.[http://dx.doi.org/10.1097/00006254-200203000-00023] [PMID: 1188 9417] ]. She characterized the pain as pressure in the frontal and occipital regions, with improvement towards the decubitus and worsening towards the orthostatism. No other symptoms were reported. When headache is related to preeclampsia, it is usually throbbing, bilateral, and may be associated with scotomas, hypertension, epigastralgia, edema and proteinuria [12Steegers EA, von Dadelszen P, Duvekot JJ, Pijnenborg R. Pre-eclampsia. Lancet 2010; 376(9741): 631-44.[http://dx.doi.org/10.1016/S0140-6736(10)60279-6] [PMID: 2059 8363] -14Bushnell C, Chireau M. Preeclampsia and Stroke: Risks during and after Pregnancy. Stroke Res Treat 2011; 2011858134[http://dx.doi.org/10.4061/2011/858134] [PMID: 21331165] ]. However, some warning signs suggest a secondary cause. Cases of abrupt headache, severe intensity, and with a different pattern from usual should be promptly investigated [15Schwedt TJ, Matharu MS, Dodick DW. Thunderclap headache. Lancet Neurol 2006; 5(7): 621-31.[http://dx.doi.org/10.1016/S1474-4422(06)70497-5] [PMID: 1678 1992] ]. Causes such as acute subarachnoid hemorrhage, reversible posterior encephalopathy syndrome, cerebral venous sinus thrombosis, reversible cerebral vasoconstriction syndrome, and cervical-cranial artery dissections should be excluded by computed tomography, magnetic resonance and/or lumbar puncture. Patients who underwent spinal anesthesia (subarachnoid block or spinal anesthesia) during labor and/or cesarean section may experience headache after accidental puncture of the dura mater. It occurs between 1 and 7 days postpartum, and it is often located in the nuchal and occipital regions, with more severe pain towards orthostatism region, and improvement after 10-15 minutes of decubitus. Neurolo-gical symptoms, nausea, vomiting, and intense incapacitation may also be observed [10Klein AM, Loder E. Postpartum headache. Int J Obstet Anesth 2010; 19(4): 422-30.[http://dx.doi.org/10.1016/j.ijoa.2010.07.009] [PMID: 20833030] ]. In the aforementioned case, there were no records of accidental puncture or difficulty in performing both anesthesia of labor and cesarean section. Besides, the non-classical clinical presentation makes this etiology less likely for the headache.

Women with seizures in the pregnancy-puerperal cycle can be divided into 3 categories: the first includes the majority of patients, who have established epilepsy prior to gestation; the second involves women who present their first crisis in this period, but it is not directly related to gestation and puerperium (for example, due to brain tumor or hypoglycemia), and the third group is represented by patients who also present the first crisis in this period, but as a manifestation of a pathology directly related to gestation (e.g, eclampsia, reversible posterior encephalopathy syndrome, intracranial hemorrhage, venous sinus thrombosis, among others) [8Edlow JA, Caplan LR, O’Brien K, Tibbles CD. Diagnosis of acute neurological emergencies in pregnant and post-partum women. Lancet Neurol 2013; 12(2): 175-85.[http://dx.doi.org/10.1016/S1474-4422(12)70306-X] [PMID: 2333 2362] , 16Stead LG. Seizures in pregnancy/eclampsia. Emerg Med Clin North Am 2011; 29(1): 109-16.[http://dx.doi.org/10.1016/j.emc.2010.09.005] [PMID: 21109107] ].

Epilepsy is the most common chronic neurological disorder, affecting 0.7% of the population. The majority of patients remain without seizures or with no changes in the previous pattern. However, due to the metabolic and hormonal changes of this period, there may be a need for larger doses of anticonvulsants for this to occur [7Haider B, von Oertzen J. Neurological disorders. Best Pract Res Clin Obstet Gynaecol 2013; 27(6): 867-75.[http://dx.doi.org/10.1016/j.bpobgyn.2013.07.007] [PMID: 23988426] ]. Symptoms associated with seizures include the presence of aura, focal or generalized motor movements, changes in breathing pattern, loss of consciousness and function of the bowel and bladder. The crisis can be followed by amnesia, lethargy, headache, muscle pain and transient focal weakness [17Hart LA, Sibai BM. Seizures in pregnancy: Epilepsy, eclampsia, and stroke. Semin Perinatol 2013; 37(4): 207-24.[http://dx.doi.org/10.1053/j.semperi.2013.04.001] [PMID: 23916020] ]. The onset of epilepsy in pregnancy is less common, as its incidence peaks are in childhood and old age. Therefore, acute seizures in this period require urgent investigation [1Paula JCR, Aquino LO, Padua BJ, Leite HV, Cabral ACV, Brandão AHF. Neurological syndromes during pregnancy. Femina 2015; 43(3): 119-24.].

It has been reported that 44% of seizures related to eclampsia occur in the puerperium, 38% prepartum and 18% intrapartum [7Haider B, von Oertzen J. Neurological disorders. Best Pract Res Clin Obstet Gynaecol 2013; 27(6): 867-75.[http://dx.doi.org/10.1016/j.bpobgyn.2013.07.007] [PMID: 23988426] ]. It may be preceded by symptoms such as persistent frontal or occipital headache, blurred vision, photopsia, photophobia, and altered level of consciousness lasting about 1 minute [1Paula JCR, Aquino LO, Padua BJ, Leite HV, Cabral ACV, Brandão AHF. Neurological syndromes during pregnancy. Femina 2015; 43(3): 119-24.]. It is related to hypertension, edema and proteinuria [2Beach RL, Kaplan PW. Seizures in pregnancy: diagnosis and management. Int Rev Neurobiol 2008; 83: 259-71.[http://dx.doi.org/10.1016/S0074-7742(08)00015-9] [PMID: 1892 9087] ]. Magnetic resonance imaging of the brain may reveal cortical and subcortical hyperintense lesions, predominantly in the posterior region, classifying the condition as Posterior Reversible Encephalopathy Syndrome (PRES) [7Haider B, von Oertzen J. Neurological disorders. Best Pract Res Clin Obstet Gynaecol 2013; 27(6): 867-75.[http://dx.doi.org/10.1016/j.bpobgyn.2013.07.007] [PMID: 23988426] ].

The state of hypercoagulability associated with pregnancy and puerperium contributes to an increase in the incidence of cerebral venous thrombosis in this period. Headache occurs in 95% of the patients, with about 50% of them presenting convulsions or focal neurological deficits, and papilledema or mental status changes can be observed in a third of the cases. There is also a greater occurrence of subarachnoid hemorrhage in pregnancy and puerperium due to an increase in cardiac output and to hormonal effects in the vasculature, being one of the differential diagnoses to be considered [2Beach RL, Kaplan PW. Seizures in pregnancy: diagnosis and management. Int Rev Neurobiol 2008; 83: 259-71.[http://dx.doi.org/10.1016/S0074-7742(08)00015-9] [PMID: 1892 9087] ].

Convulsive crisis affect 50% to 80% of patients with a primary brain tumor, so one should pay attention to this diagnostic hypothesis [18Wong ET, Wu JK. Overview of the clinical features and diagnosis of brain tumors in adults 2017. Available from: www.uptodate.com/ contents/overview-of-the-clinical-features-and-diagnosis-of-brain-tumors-in-adults?topicRef=4838&source=see_link]. There is no increase in its incidence during pregnancy, however, some tumors may grow and become symptomatic due to water retention and hormonal changes arising from this period [19DeAngelis LM. Central nervous system neoplasms in pregnancy. Adv Neurol 1994; 64: 139-52.[PMID: 8291463] ]. Tumor-related seizures may be focal, with secondary generalization, repetitive, associated with post-ictal status, and focal neurological findings on imaging tests (preferably contrast magnetic resonance imaging) [18Wong ET, Wu JK. Overview of the clinical features and diagnosis of brain tumors in adults 2017. Available from: www.uptodate.com/ contents/overview-of-the-clinical-features-and-diagnosis-of-brain-tumors-in-adults?topicRef=4838&source=see_link, 20Lee MJ, Hickenbottom S. Neurologic disorders complicating pregnancy 2017. Available from: www.uptodate.com/contents/ neurologic-disorders-complicating-pregnancy?search=brain%20 tumor%20pregnant&source=search_result&selectedTitle=1~150&usage_type=default&display_rank=1#H18].

Regarding the patient in our report, despite the evidence of hypertension and proteinuria from the first convulsive crisis, the persistence of the episodes despite the standard treatment for eclampsia led to the finding of the intracranial lesion in the imaging exam. After its complete removal, the patient remained without new generalized seizures or neurological deficits following a 6-month follow-up.

CONCLUSION

Although the first diagnosis in pregnant or postpartum women with seizures is usually eclampsia, there are other causes for their occurrence that should be investigated or excluded. In cases of neurological symptoms with acute onset in this period, the investigation should also cover causes not related to gestation, such as brain tumor. Careful assessment is required, individualizing the patient's clinical condition, especially in refractory cases and not responding to usual treatment.

FUNDING

None.

CONSENT FOR PUBLICATION

Not applicable.

CONFLICT OF INTEREST

The authors declare no conflict of interest, financial or otherwise.

ACKNOWLEDGEMENTS

Declared none.

REFERENCES

[1] Paula JCR, Aquino LO, Padua BJ, Leite HV, Cabral ACV, Brandão AHF. Neurological syndromes during pregnancy. Femina 2015; 43(3): 119-24.
[2] Beach RL, Kaplan PW. Seizures in pregnancy: diagnosis and management. Int Rev Neurobiol 2008; 83: 259-71.[http://dx.doi.org/10.1016/S0074-7742(08)00015-9] [PMID: 1892 9087]
[3] Saftlas AF, Olson DR, Franks AL, Atrash HK, Pokras R. Epidemiology of preeclampsia and eclampsia in the United States, 1979-1986. Am J Obstet Gynecol 1990; 163(2): 460-5.[http://dx.doi.org/10.1016/0002-9378(90)91176-D] [PMID: 2386132]
[4] Moura JL. Preeclampsia-eclampsia. Rev Latinoam Hipertens 2007; 2(4): 117-27.
[5] Simon RH. Brain tumors in pregnancy. Semin Neurol 1988; 8(3): 214-21.[http://dx.doi.org/10.1055/s-2008-1041380] [PMID: 3057552]
[6] Michelsen JJ, New PF. Brain tumour and pregnancy. J Neurol Neurosurg Psychiatry 1969; 32(4): 305-7.[http://dx.doi.org/10.1136/jnnp.32.4.305] [PMID: 4980021]
[7] Haider B, von Oertzen J. Neurological disorders. Best Pract Res Clin Obstet Gynaecol 2013; 27(6): 867-75.[http://dx.doi.org/10.1016/j.bpobgyn.2013.07.007] [PMID: 23988426]
[8] Edlow JA, Caplan LR, O’Brien K, Tibbles CD. Diagnosis of acute neurological emergencies in pregnant and post-partum women. Lancet Neurol 2013; 12(2): 175-85.[http://dx.doi.org/10.1016/S1474-4422(12)70306-X] [PMID: 2333 2362]
[9] Goldszmidt E, Kern R, Chaput A, Macarthur A. The incidence and etiology of postpartum headaches: a prospective cohort study. Can J Anaesth 2005; 52(9): 971-7.[http://dx.doi.org/10.1007/BF03022061] [PMID: 16251565]
[10] Klein AM, Loder E. Postpartum headache. Int J Obstet Anesth 2010; 19(4): 422-30.[http://dx.doi.org/10.1016/j.ijoa.2010.07.009] [PMID: 20833030]
[11] Von Wald T, Walling AD. Headache during pregnancy. Obstet Gynecol Surv 2002; 57(3): 179-85.[http://dx.doi.org/10.1097/00006254-200203000-00023] [PMID: 1188 9417]
[12] Steegers EA, von Dadelszen P, Duvekot JJ, Pijnenborg R. Pre-eclampsia. Lancet 2010; 376(9741): 631-44.[http://dx.doi.org/10.1016/S0140-6736(10)60279-6] [PMID: 2059 8363]
[13] Martin SR, Foley MR. Approach to the pregnant patient with headache. Clin Obstet Gynecol 2005; 48(1): 2-11.[http://dx.doi.org/10.1097/01.grf.0000153208.93620.39] [PMID: 1572 5852]
[14] Bushnell C, Chireau M. Preeclampsia and Stroke: Risks during and after Pregnancy. Stroke Res Treat 2011; 2011858134[http://dx.doi.org/10.4061/2011/858134] [PMID: 21331165]
[15] Schwedt TJ, Matharu MS, Dodick DW. Thunderclap headache. Lancet Neurol 2006; 5(7): 621-31.[http://dx.doi.org/10.1016/S1474-4422(06)70497-5] [PMID: 1678 1992]
[16] Stead LG. Seizures in pregnancy/eclampsia. Emerg Med Clin North Am 2011; 29(1): 109-16.[http://dx.doi.org/10.1016/j.emc.2010.09.005] [PMID: 21109107]
[17] Hart LA, Sibai BM. Seizures in pregnancy: Epilepsy, eclampsia, and stroke. Semin Perinatol 2013; 37(4): 207-24.[http://dx.doi.org/10.1053/j.semperi.2013.04.001] [PMID: 23916020]
[18] Wong ET, Wu JK. Overview of the clinical features and diagnosis of brain tumors in adults 2017. Available from: www.uptodate.com/ contents/overview-of-the-clinical-features-and-diagnosis-of-brain-tumors-in-adults?topicRef=4838&source=see_link
[19] DeAngelis LM. Central nervous system neoplasms in pregnancy. Adv Neurol 1994; 64: 139-52.[PMID: 8291463]
[20] Lee MJ, Hickenbottom S. Neurologic disorders complicating pregnancy 2017. Available from: www.uptodate.com/contents/ neurologic-disorders-complicating-pregnancy?search=brain%20 tumor%20pregnant&source=search_result&selectedTitle=1~150&usage_type=default&display_rank=1#H18

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