Neurocase, 2015 Vol. 21, No. 2, 268–270, http://dx.doi.org/10.1080/13554794.2014.892620

The right amygdalar tumor presenting with symptoms of separation anxiety disorder (SAD): a case report Huang-Chen Chena*, Chun-Fu Linb and Ying-Chiao Leec a

Department of Psychiatry, Veterans General Hospital, Taipei, Taiwan; bDepartment of Neurosurgery, Neurological Institute, Veterans General Hospital, Taipei, Taiwan; cDivision of Community & Rehabilitation of Psychiatry, Department of Psychiatry, Veterans General Hospital, Taipei, Taiwan (Received 16 January 2014; accepted 28 January 2014) A patient with an astrocytoma of the right-sided amygdala developed symptoms of separation anxiety disorders (SADs). These symptoms significantly subsided after tumor resection. The temporal relationship suggested that the amygdalar tumor could result in the specific symptoms. To our knowledge, this is the first report of SAD as one manifestation of the amygdalar tumor. The tumorigenesis of amygdala resulted in impaired regulation and abnormal activity associated with anticipating anxiety and conditioning. It deserves clinical attention to early detection and intervention. Keywords: amygdala; amygdalar tumor; temporal lobe tumor; separation anxiety disorder (SAD); anxiety

Case report A 21-year-old Taiwanese female patient had a normal developmental history. Her elder sister was diagnosed with bipolar II disorder. One of her aunts had a history of brain tumor and another had a history of schizophrenia. She first had fear and resistance about leaving her parents when she was 10 years old, but had no focal neurological signs. As soon as she received a cue about leaving her parents, she presented disabling fear, distress, and the anxiety of anticipated separation from her parents. She cried uncontrollably when the anxiety overwhelmed her and she became euthymic instantly after having a family reunion. She also had persistent and excessive worry that an untoward event would lead to separation from her parents. Therefore, she had difficulty functioning academically and refused to go to school or attend cram school classes. She first visited our psychiatry outpatient department when she was 11 years old. Imipramine (10 mg/day) was prescribed initially under the diagnosis of separation anxiety disorder (SAD). The dosage of imipramine was titrated to 25 mg/day 2 weeks later. Sertraline (25 mg/day) was added for augmentation due to persistently high anticipating anxiety 1 month later. The combination brought significant improvement after 6 weeks of treatment. Her anticipation anxiety and fear remitted and she was able to go to school and perform fairly well academically. Psychiatric medication was gradually discontinued after 1 year of maintenance treatment. She was relatively stable for 2.5 years. However, another episode of separation anxiety occurred when she was 15 years old. The frequency and severity increased *Corresponding author. Email: taufi[email protected] © 2014 Taylor & Francis

in the following 1 month and she came back to our psychiatry outpatient department. She received sertraline (100 mg/day), imipramine (50 mg/day), and (milnacipran) 50 mg/day with partial improvement. She began experiencing a sudden onset of smelling a familiar odor when she was 18 years old. The quality of the odor was similar to dust and a déjà vu phenomenon was reported. She felt numbness from her head to her whole body with a general hot sensation a few seconds after smelling the odor. She suffered from extreme fear and anxiety in the wake of the odor. The duration of entire attack was 10 seconds. There was no loss of consciousness, motor presentation, focal weakness, urination, or defecation incontinence during this period. The frequency of the attacks was positively associated with stressful events and was up to five times a day at most. She was referred to our neurology outpatient department, where levetiracetam (1,000 mg/day) was prescribed under the impression of simple partial seizure, olfactory type. Electroencephalography revealed right mesial temporal spikes. Brain positron emission tomography revealed hypo-metabolism at the right anterior temporal lobe, right basal ganglion, and thalamus. Brain magnetic resonance imaging revealed an ill-defined tumor in the right amygdala with compression to the right hippocampal head and choroid fissure (Figure 1). She underwent right-side anterior temporal lobectomy, resection of amygdalar tumor, and the pathology report indicated a low-grade astrocytoma. She continued to take the same medication, including sertraline (100 mg/day), imipramine (25 mg/day), (milnacipran) 50 mg/day, and levetiracetam (1,000 mg/day) after surgery. Anxiety symptoms subsided subjectively and objectively after surgical

Neurocase

Figure 1. Axial magnetic resonance imaging of the brain shows a heterogeneous signal with a lesion with an ill-defined margin at the right head of the hippocampus (arrow).

intervention. The total Hamilton Anxiety Rating Scale (Hamilton, 1959) and Beck Anxiety Inventory scores (Beck, Steer, & Beck, 1993) 3 months after surgery were 1 and 9, respectively, which were lower than the values (19 and 22) before surgery. She came back to school as a college freshman with no difficulty. We tapered medication gradually and maintained only sertraline (25 mg/day). She had no significant cognitive impairment or anxiety symptoms 2 years after surgery.

Discussion Fear is an affective symptom and observed in about onetenth of patients with temporal lobe epilepsy (Cendes et al., 1994). Studies on fear conditioning in animals and humans have illuminated the neural mechanisms underlying cued and contextual fears, which are associated with amygdala responsivity (Indovina, Robbins, NúñezElizalde, Dunn, & Bishop, 2011). Our patient was diagnosed as having SAD, according to the Diagnostic and Statistical Manual of Mental Disorders Fourth Edition, text revision (American Psychiatric Association, 2000). Her symptoms persisted and were partially responsive to medication, even with good drug compliance, and an adequate dosage and duration since she was 15 years old. She maintained all medication before and after surgery. Refractory fear and anxiety resolved significantly 3 months after tumor removal, which also brought dramatic functional improvement. She needed less average daily dose of medication than that required before surgery

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after 1 year of follow-up. The temporal relationship indicated that the amygdala tumor contributed to the possible etiology of clinical symptoms meeting the criteria of SAD in our case. Another possible etiology is that our case, with a premorbid anxiety disorder, developed simple partial epilepsy that had anxiety as a component. However, the refractory course, dramatic improvement after tumor surgery, and decreasing dosage of maintenance medication during the 2 years of clinical follow-up also favored the association of specific anxious symptoms with amygdalar tumor. The amygdala consists of a number of different regions. Auditory inputs to the lateral amygdala come from both the auditory thalamus and the auditory cortex, which can mediate auditory fear conditioning (LeDoux, Farb, & Ruggiero, 1990). The projection of the ventral hippocampus to the basal and accessory basal nuclei of the amygdala is involved in contextual fear conditioning (Majidishad, Peli, & LeDoux, 1996). The amygdala is widely believed to possess plasticity during conditioning. In the event of tumorigenesis occurring in the amygdala, the resulting abnormal activity could not be mediated by other regions of the brain. The output from the central nucleus of the amygdala to the brainstem brought repetitive, disabling, and specific fear reactions, resulting in clinical symptoms similar to SAD. Mona Sazgar et al. reported five patients with right temporal lobe epilepsy presenting episodic symptoms characterized by panic attacks and feelings of intense fear (Sazgar, Carlen, & Wennberg, 2003). Assefa et al. reported a case with meningioma compressing the left amygdala had anticipating anxiety and fear symptoms (Assefa, Haque, & Wong, 2012). Our demonstration suggested that a cluster of symptoms resembling SAD may be one form of semiology in patients with amygdala tumor. The slow progression of low-grade astrocytoma of our case is one of the explanations for these specific presentations. It needs further investigations to elucidate the correlation of tumor pathology and symptoms of SAD. In our case, antidepressants, including selective serotonin reuptake inhibitors, serotonin–norepinephrine reuptake inhibitors, and tricyclic antidepressants, have partial efficacy. Combination treatment with different classes of antidepressants seems to have a better effect than single treatment. The most effective solution is tumor resection, whenever feasible. Nevertheless, it is essential for physicians to be aware of this clinical phenomenon and arrange surveys and neuroimaging for early detection.

References Assefa, D., Haque, F. N., & Wong, A. H. (2012). Case report: Anxiety and fear in a patient with meningioma compressing the left amygdala. Neurocase: The Neural Basis of Cognition, 18(2), 91–94. doi:10.1080/13554794.2011.556126

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Indovina, I., Robbins, T. W., Núñez-Elizalde, A. O., Dunn, B. D., & Bishop, S. J. (2011). Fear-conditioning mechanisms associated with trait vulnerability to anxiety in humans. Neuron, 69(3), 563–571. doi:10.1016/j.neuron.2010.12.034 LeDoux, J. E., Farb, C., & Ruggiero, D. A. (1990). Topographic organization of neurons in the acoustic thalamus that project to the amygdala. The Journal of Neuroscience: The Official Journal of the Society for Neuroscience, 10(4), 1043–1054. Majidishad, P., Peli, D. G. & LeDoux, J. E. (1996). Disruption of fear conditioning to contextual stimuli but not to a tone by lesions of the accessory basal nucleus of the amygdala. Social Neuroscience Abstracts, 22, 11–16. Sazgar, M., Carlen, P. L., & Wennberg, R. (2003). Panic attack semiology in right temporal lobe epilepsy. Epileptic Disorders: International Epilepsy Journal with Videotape, 5(2), 93–100.

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The right amygdalar tumor presenting with symptoms of separation anxiety disorder (SAD): a case report.

A patient with an astrocytoma of the right-sided amygdala developed symptoms of separation anxiety disorders (SADs). These symptoms significantly subs...
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