Amelioration of trichotillomania with onabotulinumtoxinA for chronic migraine

Background

Trichotillomania (TTM) is a rare disorder whose name was derived from Greek words: thrix (hair), tillein (to pull) and mania (madness), and is categorised as an obsessive-compulsive-related mental disorder. Characteristics of TTM include repeated attempts to pull one’s hair due to abnormal urges which typically progresses to secondary alopecia. Feelings of tension or anxiety are often reported as triggers for TTM, and relief of the negative feeling is achieved through the act of hair pulling.1

TTM affects roughly 0.5%–2.0% of the population and can have significant psychological morbidity, especially among adolescent and female individuals where the disorder is more prevalent.2 Observations among children and adolescents found that TTM was accompanied by a high incident of co-occurring disorders such as depression and anxiety, which may mediate the severity of TTM and functional impairment.3 4

The current mainstay treatment for TTM is behavioural therapy and pharmacotherapy. Among pharmacotherapy options, antidepressants such as tricyclic antidepressant agents (TCAs) and selective serotonin reuptake inhibitors (SSRIs) have been the subject of the most randomised controlled trials.5 However, a lack of efficacy is a concern in the treatment of TTM. Results from a meta-analysis evaluating 298 adults from 11 studies found little to no evidence of beneficial response to treatment or reduction of TTM symptom severity reported for antioxidants, cell signal transducers, opioid antagonists or SSRIs, and much of the discontinuation reported especially for SSRIs and TCAs was linked to tolerability issues.6 While SSRIs and clomipramine are considered a first-line therapy for TTM, a recent Cochrane review concluded that although clomipramine has demonstrated some benefit for TTM, there is no strong evidence of a treatment effect for the SSRIs.7 Given the issues with lack of efficacy and tolerability with the TTM pharmacotherapies available, it is important to investigate alternative treatment options.

Botulinum toxin received Food and Drug Administration approval for the prophylaxis of chronic migraine in 2010. The recommended dose range is 155 units, with the option to increase to 195 units for the condition of TTM.8 Botulinum toxin has a longstanding history of clinical benefit in focal dystonia, in part due to alteration of the sensorimotor feedback loop mediated by the basal ganglia.9 For this reason, we postulated that onabotulinumtoxinA (OBTA) may be clinically beneficial in a female patient with a history of TTM who was receiving 155 units of OBTA every 12 weeks for chronic migraine. In this case report, we summarise a case of TTM with a promising response to OBTA.

Case presentation

Our patient is a woman in her 30s with a history of chronic migraine and ulcerative colitis (on mesalamine therapy) who was initially seen at our clinic 5 years ago for her migraines. Initial therapy included sumatriptan and erenumab-aooe, but was discontinued. Approximately a year after her initial visit, she was started on OBTA, 155 units, per the Phase III REsearch Evaluating Migraine Prophylaxis Therapy (PREEMPT) protocol for chronic migraine in which 31 injections of 5 units each are placed just under the skin in the superficial muscle of seven specific head and neck muscle areas.10 The procerus muscle receives one injection (5 units) midline, while all other muscles are injected bilaterally to the left and right side of the neck. Muscles injected bilaterally and dose for each side include the corrugator, 5 units; frontalis, 10 units; temporalis, 20 units; occipitalis, 15 units; cervical paraspinal, 10 units; and trapezius, 15 units.10

Three months after treatment initiation of OBTA, the patient’s migraines had notably improved, with migraine days decreasing from 20 days per month to 4 days per month. During her visit, we discussed in more detail her lifetime history of TTM and the patient disclosed that TTM greatly impaired her quality of life, and she had profound alopecia for which a wig was worn regularly (figure 1).

The patient reported that TTM began in childhood after her father died from brain cancer. Soon thereafter, she developed the compulsion to pull out the hair on her scalp. She had attempted behavioural therapy and has tried SSRIs multiple times with her last SSRI treatment being 7 years prior, but has been unable to control these urges.

Treatment

The history of failed conventional treatments for TTM along with the treatment success of OBTA for her migraines motivated the patient to consent to OBTA treatment for her TTM with the understanding that it was an ‘off-label’ treatment. Informed consent was obtained to administer the ‘unavoidable wastage’, which consisted of the remaining 45 units from the OBTA 200-unit vial to her scalp. Three months after treatment initiation with OBTA for migraine, with the patient’s consent for change in dose, the OBTA treatment was expanded to include injections for TTM with 155 units administered according to the PREEMPT protocol for migraine and an additional 45 units injected, 5 units per site, in diffuse regions of her scalp, primarily in the centre of affected areas of TTM-induced alopecia.

Outcome and follow-up

During the patient’s regularly scheduled follow-up visits every 12 weeks evaluating the use of OBTA for her migraines, she also reported remarkable improvement in her TTM signs and symptoms. Specifically, 12 weeks after her first TTM treatment, she reported being symptom-free for approximately 8 weeks and without any urges to pull out her hair. There was also notable hair regrowth as shown in figure 2. Treatment effects were maintained, as reported during her regularly scheduled 12-week follow-up appointments. Additional hair regrowth was observed at the 1-year post-treatment initiation for TTM, which equated to approximately four cycles of treatment (figure 3).