Abstract
Chief Complaint and Presenting Problem
History of Present Illness
In the past 5 years N. had been hospitalized 10 times for acute exacerbation of symptoms related to his OCD, bipolar disorder NOS, and PDD-NOS. At the time of the current admission, N. had reportedly been experiencing increased intrusive sexual thoughts and urges precipitated by watching a television show about a child molester. According to his mother, N. had experienced severe guilt and frustration regarding these intrusive thoughts. As a result of the guilt, he experienced suicidal thoughts and engaged in self-injurious behavior. In addition, N. was reported to be masturbating 25 to 30 times per day, which resulted in penile ulcers. Furthermore, he had tried to stab his penis with tweezers to punish himself. During this period, N. had attempted suicide multiple times with lethal means, including overdose on multiple medications and attempts of stabbing and hanging himself. He reported intense hopelessness and helplessness regarding his sexual urges. These symptoms developed in the context of recent history notable for pressured speech, tangentiality, hypersexuality, grandiosity, easy distractibility, mood lability and sleep disturbance, thought to be consistent with bipolar disorder NOS.
Another major concern was N.'s sexual preference and gender role. He had expressed conflict regarding his sexual preference for younger children. Most recently, N. had expressed intermittent homicidal ideation toward his mother's boyfriend, G., as he was worried that G. might not approve of or might interfere with a future relationship with a boyfriend.
According to mother, N. had experienced a severe fear of contamination starting at age 6 years; he was reportedly worried about germs, cleanliness and getting sick. He used disinfectant multiple times a day, washed his hands up to 20 times per day—which resulted in frequent fungal infections—and was adamant about avoiding hand shaking with others because of a risk of contamination. N. had been treated with multiple medications for his OCD; according to mother and N., his current treatment with clomipramine was beneficial for his thoughts and rituals.
In addition, mother reported that N. exhibited repetitive behaviors such as blowing air from his mouth and performing magic tricks, which may have been self-soothing. She denied any past history of simple or complex motor or vocal tics suggestive of Tourette's disorder. Mother reported that N. had recently developed some abnormal body movements, such as frequent head turning, lip smacking, grimacing and tongue rolling, which she associated with abrupt discontinuation of risperidone just prior to the referral for hospitalization. In addition, N. was reported to be sensitive to loud noises and specific textures of clothes.
Finally, N. was reported to be very rigid, to need routine, and to want things done in a certain way. He would reportedly get upset if things were not done his way, and this would escalate to episodes of physical and verbal aggression. He also was reported to have poor social boundaries, lacked social skills, and struggled to follow conventional rules. N. was greatly interested in crystals, stones, and different scientific theories and had in-depth knowledge about them. Mother reported that he spent long hours on the computer conducting research about different stones. During interaction with peers and clinicians, N. was reported to frequently divert the topic of discussion toward different scientific theories or stones.
Mother denied that N. had had any significant history of hyperactivity, inattention, fidgetiness, or restlessness. She stated that his focus and concentration were good. N. was described as eating and sleeping well. There was no history of auditory, visual or olfactory hallucinations.
Past Psychiatric History
N. had been hospitalized 10 times in the past five years for suicidal and homicidal thoughts, and exacerbations of OCD symptoms.
Developmental History
N. was the product of a full term, uncomplicated pregnancy and vaginal delivery. Birth weight is not known. He reached all milestones within five months of the normal range and received speech therapy for a speech delay in early childhood.
Neuropsychological testing conducted between ages eight and nine years showed a major delay in social and communication skills. His overall IQ was 59.
Educational History
N. has been enrolled in a special education program since early childhood and currently attends a special education class for children with autism spectrum disorders. He receives speech, occupational, and physical therapy at school. He also participates in a group to improve his social and communication skills.
Social History
N. lives with his mother and her boyfriend. At time of N.'s hospitalization, mother was pregnant and worried about the well-being of the new baby, given N.'s aggression, sexual urges, and sexual preference for children.
Biological parents had been divorced for the past four years, and biological father was not in contact with N. As a result, N. was reported to feel rejected and angry toward his biological father.
N. reportedly did not have friends and was mostly interested in scientific theories and magic tricks.
There was no history of physical, sexual, or emotional abuse. There was no history of legal charges.
Family History
Family history is significant for bipolar disorder in both biological parents. The details of their treatment are not known. As per mother, there was no family history of tics, Tourette's disorder, or any other known psychiatric problem.
Medical History
N. has reportedly had no serious medical problems, hospitalizations, or surgery. He had no history of any major childhood illnesses and had received all his appropriate vaccinations. His weight, nutrition, and metabolic status were within the normal range. No allergies were reported.
Medication History
N. had been treated with multiple medications including quetiapine, risperidone, sertraline, citalopram, fluvoxamine, fluoxetine, valproic acid, clomipramine, guanfacine, clonazepam, and clonidine. Unfortunately, mother was unable to provide the details of the dosage, outcome, and duration of the medication trials. Most recently, N. had been treated with risperidone 6 mg, which had been discontinued as a result of concerns regarding increased aggressive behavior several weeks prior to hospitalization.
Current medications upon admission included sertraline 250 mg daily, guanfacine 4.5 mg daily, clomipramine 225 mg daily, and clonazepam 1 mg daily.
Mental Status Examination
N. was alert and oriented and appeared his stated age. He had poor eye contact, and exhibited abnormal movements, including frequent head turning, lip smacking, grimacing, and tongue rolling. Speech was pressured and monotonous with odd prosody but well articulated. Mood was irritable and affect was restricted. Thought process was egocentric; N. changed the topic, wanting to discuss sex, crystals and scientific theories, suggestive of derailment and some tangentiality. He denied any auditory or visual hallucinations; delusions or paranoia were not noted. N. reported some passive suicidal thoughts and homicidal ideation toward mother's boyfriend. Insight and judgment were poor.
Hospital Course
Once hospitalized, N. engaged in sexually inappropriate behavior on the unit and showed desire to have sex with other male peers. During his stay in the hospital, N. exhibited lip rolling, shoulder shrugging, and facial grimacing. The movements resolved after N. was started on aripiprazole. During his stay, sertraline and clomipramine were gradually tapered, as mother believed that they were not helpful and was also concerned about the risk of serotonin syndrome from the high dosage of medications. N. was continued on guanfacine and clonazepam, as mother reported benefits from these medications. Aripiprazole 5 mg twice a day (bid) and lithium 300 mg bid were added to the medication regimen. Aripiprazole was increased to 10 mg bid. Lithium was increased to therapeutic range (serum level 1 mEq/l), and he responded positively, showing an improvement in his mood symptoms. In addition, he denied suicidal or homicidal ideation, and his sexual obsessions and thoughts decreased significantly. After 14 days, N. was stabilized and discharged to the care of his mother and outpatient treatment.
Brief Formulation
In summary, N. was a 17-year-old adolescent who presented with a complicated history of aggressive, hypersexual, and disruptive behavior in the context of developmental and speech delay, behavioral rigidity, decreased social reciprocity, and restrictive interests best understood in the context of an autism spectrum disorder (ASD). He was preoccupied with intrusive sexual and contamination thoughts leading to ritualistic behaviors. In addition, N. had suicidal and homicidal ideation, mood lability, and tangential thought processing. He had attempted suicide with potentially lethal means, and expressed homicidal ideation toward his mother's boyfriend in the context of his mother's pregnancy, putting both N. and the family in a very high-risk situation.
From a biological perspective, N. was at strong genetic risk for bipolar disorder or other major mental illness because of the significant family history of bipolar disorder on both maternal and paternal pedigrees. Although there were no apparent medical conditions that might have contributed to his current exacerbation of mood and anxiety symptoms, he appeared to have withdrawal dyskinesia as a result of risperidone discontinuation. From a psychosocial perspective, N. had experienced losses and inconsistency in his social environment, as biological father was not invested in him; mother's boyfriend and the pending birth of a sibling were undoubtedly precipitants of his current deterioration. Furthermore, he struggled in school because of learning difficulties, sexual preference, abnormal movements, and poor social and communication skills. A strength was his mother's investment, support, and concern for him.
Multi-Axial Diagnoses
Discussion
This case illustrates the challenges and complexity of diagnosis and treatment of an adolescent presentation in the context of three major intersecting psychiatric disorders with overlapping clinical features. N.'s clinical features must be viewed in the context of his PDD, the overarching and underlying psychopathology that both colored and crafted his overall development and the unfolding of symptoms over time. This challenging case illustrates the limitations of categorical diagnosis when overlapping symptoms of several Axis I disorders are the primary target for intervention.
N.'s sexual obsessions and compulsive masturbation could be viewed as signs/symptoms of hypersexuality of bipolar disorder, manifestations of OCD, his maladaptive cognitive and emotional response to developmental trauma precipitated by concerns about child molestation, and/or conflicts over sexuality. The sexual symptoms were initially thought to be a component of OCD, but the patient did not respond to high dosages of selective serotonin reuptake inhibitors (SSRIs), including clomipramine, and psychotherapy. These symptoms were also viewed as maladaptive developmental features of PDD, and perhaps superimposed trauma. Interestingly, and not surprisingly given the strong family history of bipolarity, when lithium was added, reduction of these symptoms occurred. As a result, in retrospect, the conclusion was that the hypersexuality was primarily driven by bipolar disorder.
There is remarkably little research on the sexuality of adolescents with ASD, both in terms of their knowledge of sexuality and sexual preferences. As adolescents with ASD experience major difficulty with social interactions and perhaps lack of sexual knowledge, these youth often experience impairment in the ability to appropriately interact in the sexual world of adolescence; inappropriate sexual behavior may result. There are no studies that have examined the role of hypersexuality, sexual obsessions, or sensory states and sexuality in youth with ASD. Given the clinical observation of sensory dysfunction in youth with ASD, for example, tactile sensitivities, it is possible that perception of pleasure may also be dysregulated, resulting in excessive masturbation.
From the perspective of OCD, there are very few studies that have examined the prevalence and clinical correlates of sexual obsessions, either in adults or youth. Williams et al. (2010) studied sexual obsessions in 409 adults with OCD. They reported that 8% (n=33) had current sexual orientation obsessions, and 11.9% (n=49) endorsed lifetime symptoms. Subjects with a history of sexual orientation issues were twice as likely to be male than female, and have moderate OCD severity. Regarding treatment, in a study of the effects of citalopram, sexual obsessions were a predictor of positive response (Stein et al. 2007); however, in another SSRI study, individuals with sexual obsessions had poorer long-term outcomes (Mataix-Cols et al. 2005).
Grant et al. (2006) also studied sexual symptoms in OCD adult patients. They reported that in 296 patients, 13.3% had current sexual obsessions, whereas 24.9% reported symptoms in the past. They also found that subjects with sexual obsessions had an earlier age of OCD onset, an earlier entry into treatment, higher rates of aggressive and religious obsessions, as well as a trend for greater depressive symptoms, longer duration of treatment, and higher rates of impulse control disorder. More research is clearly needed in youth with ASD and/or OCD.
Another issue was N.'s major concern regarding sexual orientation and preference. A conflict regarding sexual orientation can occur in ASD, as patients may misinterpret normal homosexual curiosity in the context of being less attuned to social rules and norms. Exposure and response prevention and cognitive behavioral therapy remain the treatment of choice for sexual obsessions, including for individuals with ASD (Purdon 2004).
Given the patient's positive response to lithium, and strong family history of bipolar disorder in mother and father, the conclusion was that the sexual preoccupations and masturbation were a manifestation of the hypersexuality of bipolar disorder. Although N. lacked some classical features of bipolar disorder such as grandiosity, euphoria, and decreased need for sleep, his intense sexual obsessions, tangentiality, loose associations, pressured speech, and distractibility were convincing. Interestingly, one study reported significantly higher rates of sexual obsessions in individuals with bipolar disorder and co-morbid OCD, and lower rates of ordering rituals (Perugi et al. 2002). They also reported more frequent co-morbidity with panic disorder-agoraphobia and substance abuse in co-morbid bipolar disorder and OCD, and a substantial impact on clinical characteristics and treatment outcome. Another study reported that patients with episodic OCD and co-morbid bipolar disorder do not respond to tricyclic antidepressants or SSRIs, but show full and persistent recovery in response to lithium or an acute course of electroconvulsive therapy (Swartz & Shen 1999).
The challenges faced in the diagnosis and management of complex patients such as N. indicate that additional research on this topic is needed. Future investigation should also be directed at potential pubertal and neurobiological factors that may contribute to the etiology and pathophysiology of these particular obsessions and behaviors in children with ASD.
Footnotes
Disclosures
Dr. Singh has no conflicts of interest or financial ties to disclose. Dr. Coffey has received research support from Eli Lilly Pharmaceutical, NIMH, NINDS, Tourette Syndrome Association, Otsuka, Shire, Bristol-Myers Squibb, Pfizer, and Boehringer Ingelheim.
Acknowledgment
We would like to acknowledge and thank Resham Gellatly for her assistance in review and preparation of the manuscript.