Secondary mania caused by olfactory groove meningioma: A case report

INTRODUCTION

Orbitofrontal circuit dysfunction can cause impaired impulse control, and obsessive and antisocial behaviour.¹ Secondary mania is an emotional disorder that is caused by an organic brain lesion, drug addiction and delayed anoxic encephalopathy.² Some patients with an olfactory groove meningioma may present with only psychiatric symptoms, such as depression, panic attacks and personality changes.³ We report a patient with a large olfactory groove meningioma who presented with manic symptoms, which disappeared after removal of the tumour.

THE CASE

A 69-year-old female, who was right-handed and a high school graduate, was referred due to abnormal behaviours, such as hyperactivity, insomnia and aggressiveness for 1 year. She denied any history of hypertension, diabetes or psychiatric illness. She had no family history of psychiatric illness. She had been well but began to express strange and unusual thoughts 1 year before visiting our hospital. Subsequently, she quit her job as a result of frequent quarrels and poor sales performance. She also started to have excessive alcohol consumption.

On presentation, she was euphoric and confused. She also had reduced concentration and disinhibited thoughts. She spoke loudly and laughed excessively and had an irritable mood, psychomotor agitation and decreased need for sleep. She had a blood pressure of 130/80 mmHg, heart rate of 90 beats/minute, respiratory rate of 21 breaths/minute and body temperature of 36 °C. Her physical examination showed unremarkable findings. Neurological examination revealed clear consciousness but her speech was somewhat rapid and she was physically aggressive towards her family and the medical staff. The cranial nerves, motor, sensory, cerebellar and reflex function tests were all unremarkable. A psychiatric interview which was performed for the systematic assessment of the patient’s behaviour revealed manic state. The interview revealed elevated or irritable mood, increased energy, decreased need for sleep and grandiosity in the patient.

We performed the Korean version of the Mini-mental status examination (K-MMSE),⁴ neuropsychiatric inventory (NPI) and a standardized neuropsychological battery called the Seoul Neuropsychological Screening Battery (SNSB)⁵ for a detailed cognitive evaluation. The SNSB contains tests for language, visuospatial ability, memory and frontal executive functions.⁵

Cognitive examination revealed disorientation to place and time, deficits in short-term recall and serial concentration tasks, and constructional apraxia related to visuospatial impairments. Abstract thinking was concrete, and judgment ability in response to hypothetical scenarios was poor. Her K-MMSE and NPI scores were 11 and 45 points, respectively (Table I). The Young Mania Scale (YMS)⁶ was used to assess her manic condition, which revealed severe mania with 24 points.⁷

The differential diagnoses considered were bipolar disorder, substance-induced mania, medical conditions such as hyperthyroidism and neurological conditions such as multiple sclerosis, stroke, brain tumours or traumatic brain injury.

Magnetic resonance imaging (MRI) scans of the brain revealed a large, midline extradural well-circumscribed homogeneously enhancing lobulated mass (5×6.2×6 cm; Fig 1) in both frontal lobes based on the planum sphenoidale and tuberculum sellae. Prominent displacement and oedema in the bifrontal area were observed. A major mass effect with cortical buckling and lateral displacement of the medial aspects of the frontal lobes with posterior displacement of the genu of the corpus callosum was noted. An olfactory groove meningioma was suspected.

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FIG 1.

Magnetic resonance imaging of the brain revealed a large, midline extradural well-circumscribed homogeneously enhancing lobulated mass (5×6.2×6 cm) of both the frontal lobes based on the planum sphenoidale and tuberculum sellae. The T1 isointense extra-axial lesion in the anterior cranial fossa shows diffusion restriction and vivid contrast enhancement arising from the olfactory groove involving the nasal cavity and ethmoidal sinuses. Prominent displacement and oedema of the bifrontal area were seen. A mass effect with cortical buckling and lateral displacement of the medial aspects of the frontal lobes with posterior displacement of the genu of the corpus callosum was noted.

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She received a clinical diagnosis of organic manic disorder (F06.30) as per the International Classification of Diseases, Tenth revision diagnostic criteria.

She was transferred to the neurosurgery department, where she underwent uneventful resection of the mass lesion through a bifrontal craniotomy. After interrupting the blood supply, the tumour was debulked and removed. Histopathological examination of the resected tissue showed moderately hypercellular meningothelial meningioma with a well-developed syncytial architecture. Tumour cells had uniform nuclear features, without mitotic figures or necrotic areas.

After brain surgery, her cognitive function and psychiatric symptoms slowly returned to normal, and her elevated mood was stabilized. Ten months after surgery, the follow-up brain MRI showed a postoperative porencephalic defect in the frontal lobe and 1.3×1.2×1.1 cm size residual meningiomatous lesion in the operated bed area of the anterior falx (Fig 2). The patient’s psychiatric symptoms were resolved and she stopped consuming alcohol. Her follow-up cognitive and psychiatric evaluation showed improved cognitive functions in visuospatial, language, memory and frontal domains. The follow-up scores of K-MMSE, NPI and YMS were 24, 8 and 5 points, respectively (Table I).

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FIG 2.

Postoperative porencephalic defect in the right frontal lobe. Residual meningioma, approximately 1.3×1.2×1.1 cm in size, in the operated bed area of the anterior falx (arrow).

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DISCUSSION

Anatomical location of tumours is an important factor in determining the nature and severity of neuropsychiatric symptoms.^3,8,9 Typically, a dorsolateral prefrontal lesion leads to executive function deficits, a medial-frontal lesion leads to apathy or abulia and an orbitofrontal lesion leads to disinhibition.^2,8,9 Some patients with olfactory groove meningioma can present with psychiatric symptoms, including depression, poor attention, personality changes and dementia, without any localising signs until they are large enough.^3,10

Our patient presented with progressive symptoms of mania for 1 year without focal or lateralising neurological signs. Symptoms, such as increased activity, decreased sleep, faster speech and aggressive behaviour, met the criteria for severe mania using the DSM-V and YMS, respectively.^6,7

In this patient, the frontal-subcortical circuit dysfunction caused secondary mania. Damage to the base of the frontal lobe, with which the orbitofrontal meningioma is intimately involved, caused manic symptoms, such as euphoria, or disinhibition. In contrast, depression and apathy have been linked to damage of the dorsal and lateral aspects of the frontal lobe.^3,8–10 Individual differences have been found between the onset of the frontal lobe lesion and the occurrence of secondary mania.

Individual differences in the brain network exist; however, the fronto-temporal pathways that connect the orbitofrontal area to the basal temporal regions of the limbic system are known to play a critical role in producing secondary mania. The disruption of these fronto-temporal pathways disconnects the prefrontal cortex from the limbic system, thereby releasing inhibitions of the frontal limbic system. Thus, the basal frontal compression in our patient might have prevented frontal inhibitions from the limbic system, resulting in mania, which manifested itself as a burst of limbic activities.^2,8,11

This patient is an example of secondary mania caused by orbitofrontal compression caused by olfactory groove meningioma.