Pollakiuria as a complex motor tic in a patient with Tourette syndrome

Dear Editor,

Tourette syndrome (TS) is a condition with onset in childhood, characterised by motor and phonic tics, and such associated psychiatric disorders as obsessive-compulsive disorder (OCD) and attention-deficit/hyperactivity disorder; patients with TS present a wide variety of phenotypes. It is believed to be caused by a dysfunction of different cortical circuits, especially in the fronto-parietal cortex and basal ganglia, which explains the clinical heterogeneity of the syndrome. The current theory is that TS is caused by alterations in the brain's development and maturation.¹ Pollakiuria is defined as an abnormal urinary frequency, and has occasionally been described as a tic in children with TS or paediatric autoimmune neuropsychiatric disorders associated with streptococcus.² There are no reported cases of pollakiuria in adults diagnosed with TS. We describe an adult case of TS and pollakiuria.

Our patient was a 46-year-old man with OCD, hypothyrodism, and TS diagnosed according to the DSM-IV criteria since childhood, with simple and complex motor and phonic tics. He responded well to tetrabenazine (125 mg/day). He had been treated for depression or anxiety disorder by the psychiatry department; at some point during this treatment, he presented benzodiazepine overuse. In January 2012, as he presented good emotional control and no tics, the patient himself decided to progressively discontinue all medication (tetrabenazine at 125 mg/day and dipotassium clorazepate at 30 mg/day). In November 2012, he consulted due to a history of pollakiuria of several months’ duration, reporting a urinary frequency of up to 50 times a day, which incapacitated him. The patient reported perineal and left inguinal pain, which was alleviated when he urinated. Pain was diffuse, dull, of short duration, and paroxysmal, and was exacerbated by sitting. The symptoms incapacitated him fully, preventing him from going outdoors or living independently. He reported no symptoms suggesting depression or anxiety recurrence. Months before his follow-up appointment, he had already consulted with the urology department, which requested the following tests: lumbar MRI scan, urinary tract and abdomen and pelvis ultrasounds, cystography, urine culture, blood analysis, urodynamic study, and EMG of the pelvic wall; all tests yielded normal results. At the time of consultation, he reported having used antimuscarinic drugs with no benefits. The neurological examination identified no abnormalities. This was a case of pollakiuria as a response to several months’ history of genital pain with no bladder, prostate, or genital lesion. Since no alterations were found in the neurological examination, lumbar MRI scan, or EMG, we also ruled out lumbosacral neuromuscular disease. Considering the reported symptoms, we considered the possibility of a motor tic or compulsion. We believed the pain to be a premonitory sensation and urination to be the motor pattern alleviating pain; therefore, our initial diagnostic hypothesis was a complex motor tic, to be treated with aripiprazole at 10 mg/day. One month after treatment onset, the patient was asymptomatic and reported no adverse effects.

Obsessive-compulsive disorder, or anakastic personality disorder, shares many features with TS: both mainly affect young patients, present a remitting course and premonitory sensation prior to motor tic, and may be associated with complex motor rituals. Between 25% and 50% of patients with TS present OCD and 20%–30% of OCD patients have tics. Therefore, in a patient with TS presenting complex motor rituals, a differential diagnosis of tics and compulsion should be performed.³ The function of compulsion is to alleviate an obsession or the anxiety caused by an unpleasant intrusive thought. However, tics involve a premonitory sensation which can be defined as a sense of urgency to perform the movement, although it may also be a physical symptom localised in the area where the tic occurs. In our patient, the genital pain, alleviated by urination, may be considered the premonitory sensation that appears by definition before the tic. Urinating would be the complex motor tic.

Interoception is the capacity to perceive sensations originating in the internal organs of the body. Increased perception of the need to urinate may also be considered as heightened interoception in our patient. Increased interoceptive capacity has been related to increased perception of the premonitory sensation of tics, which in turn has been associated with more severe tics.¹ Our patient's pollakiuria may be considered a severe complex motor tic causing a significant limitation to his daily life.

It is important to identify the symptoms of patients displaying complex motor rituals in order to select an appropriate therapeutic approach, since compulsion and tic require different treatments. Aripiprazole is a well-tolerated drug which achieves adequate control of tics.⁴

Pollakiuria in an adult patient with TS should raise suspicion of the possibility of a complex motor tic, which would require an adjustment of the therapeutic approach.