Compulsive Spitting in a Child with Pervasive Developmental Disorder and Tourette's Disorder: Tic or Compulsion?

Chief Complaint and Presenting Problem

G. is an 11 ½-year-old, right-handed, fifth grade student in a special education school who carries a diagnosis of pervasive developmental disorder-not otherwise specified (PDD-NOS) and Tourette's disorder (TD). G. was referred for recent onset of repetitive, worsening spitting symptoms.

History of Present Illness

G.'s parents initially became concerned about him within the first few years of life. Parents report that G. had been slow to develop language. He did not talk very much until age 2 ½ years. Echolalia was present when G. was a young child. As he grew older, his language improved, but language pragmatics and prosody remained issues. His conversations tended to be quite tangential and focused on restricted areas of interests such as his Pokémon cards and video sports games. His prosody was abnormal; his voice could be too soft, too monotonic, too mechanical, or too loud. Socially, G. had a preference for being alone but did interact with other children, albeit in a socially awkward fashion. Eye contact was a problem but could be improved when he was pressed to do it. He averted gaze during conversation. He had a history of some stereotypic movements as a young child, including flapping and finger flicking, but these had virtually disappeared by age 5 years. When G. was first evaluated by a pediatric neurologist at age 8 years, he carried a diagnosis of PDD-NOS. His chromosomal and metabolic workup was negative.

G. first developed tics at about 6 years of age, which waxed and waned. His initial tics primarily included eye blinking, grimacing, head turning and shoulder shrugging. Vocal tics included blowing, slurping, throat clearing, and gagging. G. was first evaluated for the tics at age 8 years because he moved so much it was hard to pay attention in class and other children were commenting about them. Nose picking was particularly prominent at that time, and his classmates kept telling him to wash his hands.

The tics were of sufficient severity that treatment was indicated. G. was treated first with clonidine (0.05 mg bid), which initially reduced the tics. Clonazepam (0.5 mg bid) was added when he had an exacerbation that did not respond to increasing his clonidine to 0.1 mg bid. This combination resulted in a significant decrease in tics for several months. G. returned at age 8 ½ relatively tic-free but complaining that he had a strong urge to spit, and he was continually worried that he would have to do so. Because of his constant worry about spitting and his generally inflexible and perseverative behaviors, a small dose of fluoxetine hydrochloride (5 mg) was added to his regimen. Whenever either the clonidine or the clonazepam was decreased, he experienced an exacerbation of his motor and/or vocal tics, which subsequently responded to an increase in medication.

Approximately one year later, at age 9 ½ years, G. returned because of a tic exacerbation. Head nodding was particularly problematic at that point and was interfering with everyday activities like playing the guitar, which he enjoyed. Baclofen (30 mg bid) was added in an effort to decrease this particular motor tic. Again, whenever any of his medications was decreased, he experienced an exacerbation of tics that responded to a return to the previous dose of medication.

Six months later at age 10 years, G. returned with the tics relatively well controlled but with significant problems of distractibility, hyperactivity, and impulsivity. A decision was made at the parents' request to monitor his progress in a new classroom placement rather than to introduce stimulants. Approximately one month later, mother reported that he was pacing back and forth, counting on his fingers words in the sentences he was saying, hoarding little candies, and tapping and touching various surfaces in a rhythmic fashion. Because of the obsessive qualities of these behaviors, G.'s fluoxetine hydrochloride was increased to 10 mg with moderate benefit. At age 10 ½ years, six months later, G. began chewing on his shirt and blinking, but these symptoms were not interfering with everyday activity. Six months later, at age 11, he had another brief exacerbation during which he would lick and sniff mother's face and hair. He remained on baclofen, clonazepam, clonidine, and fluoxetine hydrochloride.

Shortly thereafter, G.'s vocal tics, including throat clearing and slurping became more prominent, followed months later by beginning to spit repetitively. G. would spit when he had a hair in his mouth or when he had the feeling of a hair in his mouth. Parents reported that G. had recently begun spitting very frequently, as often as several times an hour. G. reported that most often before he spit he would see one of three different ugly faces which he described in some detail. Sometimes seeing something “gross” prompted the spitting, and sometimes having a hair, or the feeling of a hair, in his mouth prompted him to spit. He spit on his shirt sleeve if necessary, but if he had a tissue available (which he usually did) he spit into the tissue; he then held it in a particular way to avoid touching the saliva and would fold it very carefully.

Mother described his tics at that point as the worst they had been since he started medication. Clonidine was discontinued, and risperidone 0.25 mg bid was added to the clonazepam, baclofen and fluoxetine. All of his tics decreased significantly in severity after risperidone was started. However, after about one month, the spitting recurred with the premonitory symptoms. An increase in risperidone to 0.5 mg bid promptly eliminated virtually all his tics.

Past Psychiatric History

There was no past psychiatric history other than described above. Psychoeducational testing at age 9 years documented an average IQ. G. had difficulties with right hemisphere tasks like block design and attention to visual detail. His memory for rote information was excellent, but his visual memory was relatively poor. He also had problems with cognitive flexibility and executive function. G. was diagnosed with a nonverbal learning disability at that time.

Developmental History

G. was the product of an uncomplicated pregnancy and delivery. Both motor and language milestones were delayed. He walked at about 18 months and was described as hypotonic. He began speaking when he was about 2 ½ years old.

Educational History

G. attended special education programs from age 2 years onward in which he received speech and language therapy as well as occupational therapy, which focused both on his fine motor problems and tactile hypersensitivities.

Social History

G. had problems with social relatedness and reciprocity and the development of peer relationships appropriate to his developmental level consistent with his PDD-NOS diagnosis.

Family History

G. has a 7-year-old sibling with a developmental language disorder, a developmental coordination disorder, and attention-feficit/hyperactivity disorder (ADHD). Several other relatives had ADHD and several had anxiety disorders.

There was no known family history of obsessive-compulsive disorder (OCD), TD, or autistic spectrum disorders.

Medical History

G. had no serious medical problems, hospitalizations or surgery. He had no history of any major childhood illnesses and had received all his appropriate vaccinations.

Medication History

Current medications included clonazepam 0.5 mg bid, baclofen 30 mg bid, fluoxetine hydrochloride 10 mg qd, and risperidone 1 mg bid.

Mental Status Examination

Mental status examination revealed a cooperative child, with no dysmorphic features, whose response to questions about everyday life were accurate but brief. G. lay draped over a chair during much of the visit. No tics or other abnormal movements were apparent during the examination. During the interview, eye contact was fleeting, and G. tended to avert the examiner's gaze. His prosody was unusual in that it had a singsong quality at times and a mechanical quality at other times. G. was oriented to time, day, place, and situation. During a half hour office visit, he had approximately 10 spitting episodes. During each of these, he was alert and cognizant of what was happening, disturbed by it, and methodical in his management of the tissue and saliva. He was obviously distressed by the strong urge to spit and very involved in managing the tissue into which he spit. He was capable of describing all the circumstances that surrounded his strong urge to spit.

G. was able to abstract. His memory was intact. G. denied any auditory or visual hallucinations other than the gross faces that preceded the spitting. His drawing of a person was immature, and his pencil grip was below age expectation.

His neurological examination was within normal limits, although he was hypotonic; his coordination on stressed gait (heel, toe, and tandem walk) was poor and running was awkward.

Brief Formulation

In summary, G. is an 11 ½ year old boy who meets diagnostic criteria for PDD-NOS and TD. Notable were linguistic and pragmatic language problems and difficulties with social relatedness and reciprocity consistent with autistic spectrum disorders. G. had an early history of stereotypic movements typical of children on the spectrum, including flapping and finger flicking when he was young. He developed motor and vocal tics about age 6 years that responded moderately well to medication. At about age 8 years, G. developed onset of compulsive rituals including touching, counting, and hoarding. His spitting symptom, which developed at about age 11 years, was both quite problematic but intriguing; the symptom developed after G. had an urge for several years without compulsion. The spitting occurred at a time of life-time peak severity of the tics. Whether the spitting is more appropriately categorized as a complex motor tic or a compulsion is an interesting question.

Multi-Axial Diagnoses

Discussion

This case represents an interesting and not uncommon interface of autistic spectrum disorders and TD. Children with TD referred to clinical settings often meet diagnostic criteria for other psychiatric disorders, such as ADHD and OCD. Less widely described in the literature but very common in clinical practice is the co-morbidity between autistic spectrum disorders and TD/tic disorders. A recent review suggested that approximately 6% of autistic patients meet criteria for TD (Swain et al. 2007), and the prevalence is likely higher if chronic tics are included.

Another interesting area of phenomenological overlap in both TD and spectrum patients that is not widely understood are repetitive behaviors. Motor tics are categorized as either simple or complex; simple motor tics are brief, sudden, repetitive, non-stereotyped movements involving primarily one muscle group such as eye blinking, whereas complex motor tics are slower, more purposeful, coordinated movements involving several muscle groups and performed in a more stereotyped manner (American Psychiatric Association 1994). Compulsions are repetitive behaviors or mental acts that the patient feels driven to perform in response to an obsession, the goal of which is to prevent or reduce anxiety or distress. However, to the “naked eye” of the observer, it may be difficult to differentiate complex motor tic or compulsion without subjective information from the patient regarding premonitory experiences. Children on the spectrum and with TD often have repetitive behaviors; disentangling the phenomenology may be difficult, but important, in that complex motor tics are usually treated with tic reducing medication such as neuroleptics or alpha adrenergic agonists, and compulsions with serotonin reuptake inhibitors.

G.'s spitting clearly falls somewhere on the spectrum of complex motor tic or compulsion. It is interesting that the symptom is precipitated by a visual image (ugly faces) or tactile-perceptual sensation (hair or feel of hair in mouth). Contamination concerns (something “gross”) also triggered the spitting, suggestive of obsessive compulsive phenomena. Miguel and colleagues (1997) reported that repetitive behaviors could be disentangled in adult patients with TD, OCD, or TD + OCD by their premonitory experiences. Patients with TD had predominately sensorimotor experiences prior to their repetitive behaviors, whereas patients with OCD had primarily cognitive premonitory experiences or autonomic anxiety if prevented from performing their repetitive behaviors (Miguel et al. 1997). Palumbo and Kurlan (2007), in a review of complex obsessive compulsive and impulsive symptoms in TD, noted that there are some repetitive behaviors in TD patients that are impossible to classify as either tic or compulsion since they have features of both; they suggest categorizing these behaviors as compulsive tics. Given shared anatomic sites in the cortico-striatal-thalamic cortical tracts is likely in developmental dysfunction of the basal ganglia and limbic system in both TD and OCD, overlapping symptoms and phenomenology is not surprising (Palumbo et al. 1997).

Clonidine, an alpha adrenergic agonist, was initiated to address G.'s simple motor and vocal tics and was found to be effective; augmentation with a benzodiazepine was also at least initially helpful in reducing tics. Additional augmentation with baclofen was beneficial when increasing the clondine and/or benzodiazepine was limited by adverse effects such as sedation. Baclofen, although not widely prescribed, is a GABA derivative used to treat muscle disorders and spasticity (Silay and Jankovic 2005). Awaad (1999) compared the efficacy of baclofen and botulinum toxin in more than 200 patients with TD, and 95% were reported to experience reduction in tics.

Fluoxetine was added to target compulsive behaviors such as hoarding, touching, tapping, and counting and was also helpful. Interestingly, risperidone, a tic reducing medication, was helpful in reducing the spitting, although it is possible that it was the combination of fluoxetine and risperidone that was most beneficial, given the spitting has both complex motor tic and OCD phenomenology. Augmentation of selective serotonin reuptake inhibitors with typical or atypical neuroleptics has been described as helpful in both adults and children with comorbid OCD and tic disorders (McDougle et al. 2000; McDougle et al. 1995; McDougle et al. 1990).

More studies of the phenomenology of repetitive behaviors in children and adolescents with autistic spectrum disorder and comorbid TD are indicated, given the overlap of symptomatology and the dearth of studies in this area.