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Could 20 years of treatment for migraine have prompted a spontaneous absence of speech after this patient stopped taking her medication?
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CHALLENGING CASE
A 45-year-old woman was suddenly unable to speak for two weeks. Her husband says that she has not talked since she returned from work one day. She works as a bookkeeper, and a few days before her current speech difficulties, she had a sore throat due to a cold. She understands language and communicates by email, texting, or writing notes.
She was taking amitriptyline prophylactically for migraine diagnosed 20 years ago, but she stopped when she became concerned with weight gain. She had an unexplained 50-pound weight gain about 10 years ago. She also had bouts of insomnia. Her thyroid tests were normal at the time. After dieting, she lost about 20 pounds and has remained at a stable weight. She also suffers from chronic rhinitis, and has considered having an ear, nose, and throat evaluation, but did not “get around to it.”
She is still working, and according to her husband, she is performing well at work despite her absence of speech. The patient’s husband explains that they have had some anxiety lately. Their daughter has recently given birth to twins and has an 18-month-old child. Their daughter is having marital problems and plans to move in with her parents. The patient and her husband are dependent on both of their salaries, but the idea of the patient cutting back on work in order to help care for the children was discussed about one month ago. Since her recent symptoms, logistical issues about work and childcare have not been discussed.
Physical examination
She had a normal appearance, with no skin lesions, bruises or injuries. She was quiet but cooperative and appeared to have intact comprehension. Her pulses were normal. Her hearts sounds were regular, without murmurs, and her breath sounds were regular and clear. Abdominal examination was normal, without rigidity, distension or any indication of pain.
On her neurological examination, the patient was alert and demonstrated that she is oriented x 3 by writing her answers on paper. She had no complaints and when asked if she is concerned about anything, she wrote down, “I can’t talk.” Her cranial nerve examination was normal, and muscle strength was 5/5 in all extremities. Reflexes were normal except for patellar reflexes, which were brisk bilaterally and slightly anticipatory. Her sensation was intact to light touch, pinprick, vibration, and position, and she indicated her responses to her sensory examination with appropriate gestures.
Her coordination was normal, as tested by finger to nose and rapid alternating movements. She had a normal gait and she could do heel-to-toe walking without difficulty. A Romberg test was negative.
Diagnostic testing
Blood tests, including CBC with differential, electrolytes and thyroid studies, showed no abnormalities. A brain MRI was completely normal. She subsequently had a brain and neck MRA, both of which were normal. She then had an echocardiogram to rule out valve disease or abnormal heart rhythm for embolic stroke risk, and the study was completely normal. She had an EEG that was unremarkable. Throughout her workup, she was compliant, and her symptoms remained unchanged.
Preliminary treatment
Initially, the patient was diagnosed with atypical migraine headaches due to anxiety. She was started on sertraline for migraine prophylaxis and was prescribed sumatriptan for abortive therapy. She was instructed to take sertraline every other day for one week, with no change.
When her symptoms did not improve, she was sent for brain imaging. Despite the absence of changes on her brain MRI, this patient’s doctors performed an EEG to evaluate her for abnormal cerebral cortical activity, such as a postictal slowing or any alteration in activity that may have been caused by an inflammatory etiology.
Diagnosis: Psychogenic mutism
Given the persistence of her symptoms and the absence of any other symptoms or diagnostic findings, she was sent for a psychiatry consultation, and the diagnosis of psychogenic mutism was made.
The patient declined psychological counseling and pharmacological treatment with antidepressants, both of which were recommended by her neurologist.
This patient continued to function well at work and was able to carry out activities such as shopping, food preparation, housework, and helping with her grandchildren throughout her period of mutism. She experienced a sudden resolution of her symptoms 2 years after onset and, when asked about her period of mutism, she referred to it as a "resolved stroke."
Discussion
Mutism is a condition that has been described as psychogenic mutism, selective mutism, and psychogenic voice loss. This is distinct from aphasia, which occurs as the result of a cerebral damage such as a cerebral infarct.
Mutism is more common in children, particularly among those who have autism.1 It can worsen and improve over time, often in response to stress or major changes in life, but sometimes without any identifiable cause or trigger. This condition often occurs along with developmental disorders, but the diagnosis of mutism does not require the presence or absence of developmental features. It has been reclassified in DSM-5 as an anxiety disorder,2 and it had previously been classified as a “disorder of childhood and adolescence.”
The most common adult-onset of mutism is described in the postpartum period. At the current time, there is no specific identified physiological mechanism for postpartum mutism, and it is considered a psychogenic response to situational life changes.3
A recent multicenter study of patients with psychogenic voice loss showed that patents had increased activity in the medial prefrontal region and decreased emotional activity in the amygdala using fMRI.4 It is unclear whether the changes occur as a result of the clinical manifestations or whether they cause the clinical manifestations.
Take home points
• Even if there is a history of migraine headaches, it is important to consider other possibilities, especially if a patient does not respond to prophylactic and abortive headache treatment
• Mutism can be the only manifestation of a psychogenic state
• Patients may improve without treatment when they are told that their symptomss are a manifestation of stress, but they may do so at a pace that is not easy for clinicians to predict
1. Ãstergaard KR. Treatment of selective mutism based on cognitive behavioural therapy, psychopharmacology and combination therapy - a systematic review. Nord J Psychiatry. 2018;72:240-250.
2. Holka-Pokorska J, Piróg-Balcerzak A, Jarema M. The controversy around the diagnosis of selective mutism - a critical analysis of three cases in the light of modern research and diagnostic criteria. Psychiatr Pol. 2018;52:323-343.
3. Nahar A, Kondapuram N, Desai G, Chandra PS. Catatonia among women with postpartum psychosis in a Mother-Baby inpatient psychiatry unit. Gen Hosp Psychiatry. 2017;45:40-43.
4. Spengler FB, Becker B, Kendrick KM, et al. Emotional Dysregulation in Psychogenic Voice Loss. Psychother Psychosom. 2017;86:121-123.