Case Report: Maternal death due to frontal lobe meningioma mimicking postpartum depression

Introduction

Meningiomas are the most common type of central nervous system (CNS) neoplasm, accounting for about 37% of all primary CNS neoplasms.¹ Although the majority of benign intracranial tumors are typically located at the base of the skull or over the cerebral convexity, their position can result in significant morbidity and mortality.^2,3 They are usually benign and slow-growing but can cause neurological signs and symptoms depending on their location and size.^1,4 Although neurological signs and symptoms are thought to be the most predominant manifestations, psychiatric symptoms such as depression can occasionally be the initial symptoms of meningiomas.¹ Alterations in mental status are a hallmark of psychiatric diseases, but they can also be caused by a number of clinical conditions, including brain tumors, metabolic disorders, or infections.⁵ Therefore, a thorough evaluation and diagnostic workup should be an essential component of every psychiatric diagnosis that includes neuroimaging in atypical cases.

We present a case of a 38-year-old woman who died of frontal lobe meningioma that was misdiagnosed before death as postpartum depression. The diagnosis of frontal lobe meningioma as a cause of maternal death was confirmed by autopsy and histopathology.

Case report

We present a case of a 38-year-old Ethiopian woman living in an urban area, who died of frontal lobe meningioma that was misdiagnosed as postpartum depression. She was a para III woman who had delivered a healthy baby by cesarean section 2 months before her death. She had no previous medical or psychiatric history. There is no family history of psychiatric illnesses. She had been suffering from headaches and occasional nausea since a week after delivery, which was not relieved by medications prescribed repeatedly by her postpartum follow-up institution. Her condition worsened over time and she progressively started to experience hopelessness, depressed mood, fatigue, apathy, withdrawal from social activities, and insomnia. Her family took her to a holy water site hoping for a cure, but her condition worsened. She was eventually admitted to a nearby primary hospital, where she was diagnosed with postpartum depression and died after 8-hour of hospital stay.

The body was sent to our mortuary by a police officer for postmortem examination. External examination revealed a moderately built and light-brown complexion woman with a cesarean section scar in the lower abdomen. The examination of the brain, weighing 1,600 g, was soft and edematous with flattening of cerebral gyri (Figure 1). A large spherical, well-circumscribed, firm, mobile mass overlying the cribriform plate, measuring 6.5×5.5×5.0 cm with a stalk-like attachment to the undersurface of the frontal lobe was found (Figure 2). The mass was well demarcated from the brain and white-grey on cut sections. There was no hemorrhage. No other significant findings were noted. All other organs were normal, and no other abnormality was found. Histopathological examination showed capsulated tissue consisting of meningothelial whorls and syncytial growth of uniform round cells featuring round nuclei, powdery chromatin, and eosinophilic cytoplasm; at areas, there were fascicular growth patterns with more spindled nuclei and collagen deposition (Figure 3). The tumor was a bifrontal transitional meningioma (WHO grade I).

Figure 1. A photograph of the brain showing marked cerebral edema and congestion.

Figure 2. Photograph of a large meningioma that is located at the base of the frontal lobe.

Figure 3. Histological picture of meningioma (WHO grade I).

Discussion

Meningiomas are benign, slow-growing CNS tumors. Meningioma incidence during pregnancy is unknown but is believed to be the same as in nonpregnant women of childbearing age, which has been estimated to be 4.5 per 100,000.⁶ They represent 18% of all brain neoplasms and are believed to develop from arachnoid cells.⁷ The vast majority of meningiomas are asymptomatic or barely symptomatic, and they grow very slowly. As a result, the diagnosis of meningiomas is usually made when they are accidentally discovered during neuroimaging or postmortem examinations.^1,8 For those that are symptomatic, the location of the tumor and the duration of its growth primarily determine the symptoms observed at the time of diagnosis.⁹

Meningiomas can have different levels of hormone receptors, such as progesterone and androgen receptors, and to a lesser extent, estrogen receptors.^7,10,11 The sudden and potentially life-threatening growth of meningiomas during pregnancy can be caused by hormonal exposure, fluid retention, or vascular engorgement.^11–13 Accelerated growth of meningiomas during pregnancy can lead to clinical symptoms and signs either during pregnancy or shortly after delivery. Initial manifestations can present during pregnancy and the postnatal period.¹⁴ Early signs and symptoms of intracranial meningioma can be confused with hyperemesis gravidarum in early pregnancy, preeclampsia in late pregnancy or even postpartum depression or psychosis after delivery.^4,5,13–17

Psychiatric symptoms are frequently observed in patients with brain tumors or lesions. Studies have shown that the incidence of psychiatric presentations in individuals with brain tumors ranges from 50% to 78%.^17,18 However, it is uncommon for psychosis to be caused by a brain tumor, as only a small percentage of patients with psychiatric symptoms have an organic cause.¹⁷ Meningiomas may not cause neurological symptoms and instead commonly present with psychiatric symptoms only.^1,4,17–19 A study showed that 21% of cases with meningiomas experienced psychiatric manifestations without any neurological signs.¹⁹

In our specific case study, a bifrontal meningioma was discovered on post-mortem examination. The location of the tumor in the frontal lobe is significant because frontal lobe tumors often manifest with mental status and personality changes, dementia, apathy, disinhibition, impulsivity, and other symptoms. It is worth mentioning that while there is no definitive link between psychiatric presentations and tumor type or location, frontal lobe involvement typically exhibits characteristics such as depression, apathy, disinhibition, or impulsivity.¹⁷

Our particular case relied on non-professional sources of help and died without proper diagnosis and treatment. Previous studies conducted in Ethiopia indicated that factors such as limited access to healthcare services, attribution of mental distress to psychosocial factors or preexisting problems rather than to an illness, and help-seeking preferences are barriers to help-seeking from formal healthcare services for postpartum depression episodes.^20,21 However, the deceased was an urban resident and had received postnatal care service twice indicating the accessibility of the maternal health service. It is crucial to address the lack of professional help-seeking behavior to minimize the negative impact of postpartum depression on both maternal and infant outcomes. There is a need to create public awareness about postpartum depression, its causes and consequences, and the need for help-seeking.

The case reported here highlights the importance of considering frontal lobe meningioma as a possible cause of psychiatric symptoms in postpartum women. It is imperative to acquire a detailed history and perform a thorough neurologic examination including fundoscopic evaluation in patients who present with psychiatric manifestations during pregnancy or postpartum period. The basic psychiatric diagnostic evaluation typically includes physical examination and blood testing are a part of basic psychiatric differential diagnostics.²² However, the use of neuroimaging in the assessment of psychiatric disorders remains a topic of controversy. Neuroimaging is indicated for patients with neurologic deficit, dementia, delirium, anorexia nervosa, or first-episode psychosis. Imaging studies are also required for patients older than 50 years who show personality changes or experience a new onset of depression or mania.^4,5 In contrast, in young patients with the first episode of postpartum depression, such as the one in this study, neuroimaging is not indicated.

It is well-established that headaches, nausea, visual disturbances, and focal deficits, are signs of intracranial tumors or lesions.^1,15 But initial symptoms such as headache, nausea as in our case could be subtle, or even could be attributed to other conditions and may warrant a heightened sense of awareness on the part of the clinician. A review of the literature also revealed a patient with meningioma who initially presented with nausea and severe headaches, which were initially attributed to preeclampsia.¹⁵ Our patient had occasional nausea and headaches, which could have justified a neuroimaging study, but it was not performed. These somatic symptoms can be associated with depression and thus can be misleading. Symptoms such as persistent headaches should alert clinicians to suspect organic causes of psychiatric symptoms. The case reported here signifies the importance of considering frontal lobe meningioma as a possible cause of psychiatric symptoms in postpartum women. Besides, classic symptoms of such slowly growing intracranial tumour could be subtle, or even could be attributed to other conditions and may require a high index of suspicion. The routine use of brain imaging in evaluating psychiatric disorders in younger individuals has always been a topic of controversy. However, this case clearly emphasizes the importance of obtaining a detailed history, conducting a thorough physical examination, and further investigating abnormal findings.

In conclusion, our case reported here emphasizes the importance of considering frontal lobe meningioma as a possible cause of psychiatric manifestations in postpartum women. Clinicians should be aware of this possibility and perform neuroimaging studies in patients with postpartum depression who do not respond to conventional treatment or who have atypical features. Early diagnosis and management may improve the survival and quality of life of these patients. It also underscores the need for early diagnosis and intervention of frontal lobe meningiomas to prevent fatal complications.

Consent

Written informed consent for publication was obtained from the authorized representative of the decedent.