Robert King, M.D.
Application of behavioral methodologies to overcome challenges to the diagnosis of bipolar disorder (BAD) in individuals with developmental disabilities (DD) are now well established (Lowry and Sovner, 1992). Obstacles to the determination of the prevalence of this disorder in individuals with DD remain to be surmounted (Sovner, 1990). Successful outcomes arising from the application of state-of-the-art approaches to typical and atypical BAD in individuals with DD (Sovner, 1989) including those with a rapid-cycling course (Sovner, 1991) have been documented.
Similarly, the presence of obsessive-compulsive disorder (OCD) in individuals with DD has been established (McNally & Calamari, 1989) leading to successful pharmacological interventions (Cook Terry, Heller, Leventhal, 1990). Initial prevelence studies (Vitello, Spreat & Behar, 1989, Bodfish Crawford, Powell & Parker, 1995) have suggested that the presence of DD may place individuals at increased lifetime risk of the emergence of OCD.
Gordon and Rasmussen (1989) presented the first detailed case report of OCD symptoms varying with mood, noting a previous emphasis in the literature on a belief that ego-dystonic OCD symptoms and mania appeared not to occur together. Keck, Lipinski & White (1986) documented an inverse relationship between mania and OCD, noting in a case report an inability to achieve control of one disorder without induction of the other.
Chen et. al. (1995) used Epidemiologic Catchment Area data (Robins, Helzer, Weissman et al, 1984) to determine lifetime rates of comorbidity for OCD among individuals with BAD. The need to carefully evaluate the latter group for OCD comorbidity was emphasized through the documentation of a lifetime OCD rate of 21% (8.1 x the rate in the general population) among individuals with BAD. Berk, Koopowitz & Szabo (1996) presented a case series discussing management issues related to OCD/BAD comorbidity. The recording of a 15.7% incidence of BAD/OCD comorbidity in a large consecutive series lead Pergui, Askiskal, Pfanner, Presta, et. al. (1997) to suggest BAD should take precedence in diagnosis, course and treatment considerations in this context.
Clinical characteristics and unique aspects of the treatment outcomes in five individuals with DD and comorbid BAD and OCD are described in the following case reports. All five individuals were referred as outpatients to the Developmental Disabilities Program of the North Bay Psychiatric Hospital in North Bay, Ontario, Canada. Diagnostic formulations, assessment and treatment outcomes were based on caregiver and client reports, clinical interviews, a retrospective review of previous assessments and the completion of standardized screening instruments. Treatment outcomes were assessed through behavioral data arising from objective monitoring systems, based on the application of Lowrys Assessment of Symptomatic Behaviors (Lowry, 1997) and previously reported methodologies (Sovner and Lowry, 1992).
Complex issues complicate decision making regarding pharmacological intervention and treatment outcome in this context. These include the following:
The documented potential for anticompulsive agents (selective serotonin reuptake inhibitors [SSRIs] and Clomipramine) to precipitate hypomania (Steiner, 1991).
The documented potential for Risperidone, an atypical neuroleptic, if used to supplement anticompulsive agents, to precipitate hypomania (Schnierow & Graeber, 1996).
The documented potential for Risperidone, if used as an acute maintenance mood stabilizer in the context of BAD, to exacerbate the frequency and intensity of compulsions (Remington and Adams, 1994).
The documented potential for risperidone to augment SSRIs in obsessive-compulsive and related disorders (Stein, Bauwer, Hawkridge, Emsley, 1997).
Similarities in the recurrent nature of the natural histories of OCD and BAD; both often chronic lifetime disorders and characterized by remission, relapses and reoccurrences.
The need to treat potentially life-threatening compulsions, including self-injury equivalents, despite the risk of exacerbating a known concurrent BAD.
Case #1 - SSRI-Induced Hypomania While Treating OCD
Ms A was a 52-year old single woman living in a group home setting. She was previously diagnosed as having a moderate degree of DD. She presented with a chronic history of self-injurious behavior, impaired frustration tolerance and repetitive behaviors which included the shaving of her pubic hair and the plucking of her eyebrows. A previous history of Nortriptyline-induced hypomania had lead to its discontinuation and the prescription of Lithium Carbonate. Intolerance of lithium was followed by the prescription of Valproic Acid. Persistent ordering, cleaning and completeness compulsions were treated with Paroxetine, again resulting in the induction of hypomania and the discontinuation of this medication. Aggressive pharmacological treatment of the acute and subsequent maintenance phase of her BAD with Valproic Acid and Risperidone has lead to mood stabilization. A reassessment of her compulsions demonstrated a significant decrease in their intensity and frequency. A trial with further anticompulsive agents was judged not to be necessary by the treatment team.
Case #2 - Life-Threatening Compulsions and SSRI-Induced Hypomania
Mr. B was a 31-year old male living in a group home setting. He was previously diagnosed as having a moderate degree of DD. He was noted to have an expressive dysphasia with echolalia, as well as a severe bilateral visual impairment.
Mr B was born to a mother who abused alcohol during her pregnancy and he was exposed to severe environmental deprivation during his childhood. Mr. B was known to have at least two siblings with DD and mental health concerns (schizophrenia and an atypical BAD). He presented with a history of repeatedly inserting his finger into electrical outlets in a compulsive manner, removing light bulbs and dismantling electrical wall outlets. He was also noted to have a preoccupation with specific toys and a history of plugging toilets with household objects resulting in property damage. Multiple episodes of self-induced electrocution had been recorded, necessitating 24-hour per day supervision and a severe restriction of his access to community social and recreational opportunities.
Previous episodes of presumed hypomania, characterized by overactivity and inappropriate affect, interpersonal aggression and sleep disturbance, precipitated by Fluvoxamine and Fluoxetine when prescribed as anticompulsive agents, had been documented. The prescription of Valproic Acid as a mood stabilizer allowed the introduction of Clomipramine as an anticompulsive agent with a documented 50% reduction in attempted and completed acts of electrocution. Two break-through episodes of hypomania were treated acutely with the addition of Risperidone while maintaining both Clomipramine and Valproic Acid.
Case #3 - Comorbid BAD and OCD and Baseline Exaggeration
Mr. C was a 22-year old male living in a group home setting subsequent to repatriation from an institutional setting. He had previously been diagnosed as having an autistic disorder and a severe degree of DD. He was reported to have no verbal expressive language by his caregivers.
A psychiatric consultation was requested to address the etiology and management of aggression and a concern that he may be psychotic. This concern arose from allegations he had reportedly made, through a facilitated communication process, (Sovner, 1990) regarding sexual assaults perpetrated by staff members. The latter issue was addressed systemically and educationally with his caregivers and was judged to be unfounded. His aggression appeared correlated with the interruption of multiple compulsive behaviors. A trial with Fluvoxamine precipitated an increase in his activity level and inappropriate affect, masturbation in public places, distractibility and an increase in aggression, as well as a sleep disturbance. Cross-sectionally this change in his behavior and mental status was attributed to akathesia and resolved with the discontinuation of Fluvoxamine and acute treatment with a neuroleptic and benzodiazepines. Several years later, social withdrawal, hypersomnia, episodes of tearfulness, hyperphasia and a decrease in adaptive skills (urination in public places) was diagnosed as a major depression. Clomipramine, chosen to concurrently address persistent compulsive behaviors, was prescribed and again precipitated a second episode similar to the changes observed following the prescription of Fluvoxamine. A subsequent retrospective chart review and the initiation of a prospective data collection system, confirmed a diagnosis of a comorbid rapid-cycling BAD. This was supported by a history of previous mood deteriorations. The episodes of Fluvoxamine-induced akathesia was reformulated retrospectively as an episode of SSRI-induced hypomania.
Treatment with the eventual triad of Carbamazepine, Valproic Acid and Risperidone resulted in a decrease in cycling frequency. Of significance, this correlated with an anecdotal significant decrease in the frequency and intensity of compulsive behaviors. The historical increase in the intensity and frequency of these behaviors when Mr. C was hypomanic or depressed, was judged to be an example of baseline exaggeration (Sovner, 1986).
Over the past few years the absence of further depressions and the persistence of improved control of Mr. Cs compulsions has allowed the withholding of any further anticompulsive medications.
Case #4 - Unrecognized Comorbid BAD Unmasked by OCD Charting
Mr. D was a 34-year old man, living in a group home setting. He has previously been diagnosed as having a severe degree of DD and has two brothers with a moderate and profound degree of DD respectively. A sister also has a learning disability. All siblings have been assessed genetically, in the absence of an identified syndrome or chromosomal abnormality.
In 1989 Mr. D had been diagnosed as having BAD with subsequent trials of Carbamazepine, Lithium and Modecate, with only a marginal response. At the time of his initial consultation, his current caregivers were not able to support the diagnostic hypothesis of a BAD based upon their more recent observations of his behavior. The psychiatric consultation instead was requested to address the etiology and management of ordering, completeness/incompleteness, deviant grooming and cleaning compulsions, documented by his support team following the completion of Gedyes (1992) Compulsive Behavior Checklist for Clients with Mental Retardation. No history of previous depressions was elicited. A charting mechanism was established to determine the correlation between the frequency and intensity of aggression and the interruption of compulsive behaviors.
While continuing to include the possibility of a comorbid BAD in the differential diagnosis, a trial with Buspirone was initiated, acknowledging the risk of SSRI-induced hypomania, while maintaining Carbamazepine and low-dose Methotrimeprazine. Over a nine month period, Buspirone was judged to be ineffective. Of significance, however, monitoring systems demonstrated clear evidence of cycling with respect to the frequency and intensity of compulsions. This allowed Mr. Ds support team to re-explore retrospectively and prospectively signs and symptoms of hypomania, mania and depression. This process confirmed a diagnosis of comorbid BAD. The team is now initiating aggressive management of BAD with a refined perspective monitoring system in place.
Case #5 - Use of Buspirone as an Anticompulsive Agent Combined with Nonpharmacological Approaches to OCD
Mr. E was a 48-year old male, living in a group home setting after many years of institutionalization. He has been diagnosed as a having a severe degree of DD, has no verbal expressive language and is hearing impaired. An admission in September 1993, to a psychiatric facility was precipitated by an acute history of aggression, hypersexuality, a decrease in his adaptive living skills, emotional lability and a sleep disturbance. A diagnosis of BAD and OCD had been established one year prior to this, on an outpatient basis, with a combination of Clomipramine, Tegretol and Lithium prescribed over a 10-month period. Completion of the Compulsive Behavior Checklist (1992) documented compulsions which included layering of clothing, hoarding of lint, papers, scraps of material, as well as checking and grooming rituals. An objective monitoring system was established to monitor his mood. Clomipramine was discontinued to address an admitting diagnosis of a hypomanic phase of BAD. In March 1994, though euthymic, Mr. E demonstrated an increase in hoarding, complicated by the stealing of hoarded objects. Buspirone, as an adjunctive anticompulsive agent, (Blier & Bergeron, 1996) was prescribed and Carbamazepine and Lithium maintained. A person-centered planning process created new opportunities for flexible access to preferred activities and friendships. Buspirone appeared to modulate Mr. Es previously documented negative responses to limits established by his caregivers regarding his previous unlimited access to clothing to allow layering (judged to be a health hazard secondary to a risk of heat stroke) and hoarding (judged to be a fire risk). Buspirone was discontinued in June 1995 in association with a euthymic mood and a significant improvement in the frequency and intensity of compulsive behaviors. An episode of hypomania in February 1996 responded to the addition of Risperidone, which was tapered and discontinued in May 1997. In May 1997 with sustained mood stability on Lithium and Carbamazepine, hoarding and layering were not observed and Mr. E enjoyed independent community access, several sustained friendships, attended music lessons and enjoyed a job delivering catalogues in his community.
This case series illustrates the complex interplay which develops while addressing comorbid BAD and OCD in individuals with DD. The following observations and recommendations are offered with respect to the assessment and treatment phases involved in supporting individuals experiencing both these disorders:
a)An objective monitoring system is critical to allow an interdisciplinary team to concurrently measure the intensity and frequency of observed compulsions as well as monitor the signs and symptoms of depressed, hypomanic and manic phases of BAD (Cases 1 to 5).
Charting of either BAD or OCD initially may offer diagnostic clues to the presence of the second previously undiagnosed, comorbid condition (Cases 3 and 4).
Clomipramine and SSRIs as anti-compulsive agents, have the potential to precipitate hypomania and mania and contribute to the development of a rapid-cycling BAD pattern (Baker, Chengappa, Baird, Steingard, Christ, Schooler, 1992) (Cases 1 and 2).
Risperidone is useful as an acute hypomanic agent, (Ghaemi & Nessir, 1997) has been demonstrated to have mood stabilizing properties (Jacobson, 1995) and can function in an adjunctive role in combination with a primary anti-compulsive agent (Stein, Bouwer, Hawkridge, Emsley, 1997). Given its reported capacity to both paradoxically precipitate hypomania (Schnierow & , Graeber, 1996) and exacerbate OCD (Remington &Adams, 1994), however, clinical vigilance is necessary during trials with risperidone.
The aggressive pharmacological treatment of BAD may minimize the impact of comorbid OCD symptoms, on the quality of life of the individual and negate the need for pharmacological treatment of the latter disorder (Case 3).
The concurrent treatment of both disorders may be necessitated by the life-threatening nature of some compulsions or the severe intensity and/or frequency of compulsions despite mood stabilization (Case 2).
Adjunctive anticompulsive agents, such as Buspirone may play a role as primary anticompulsive agents if the intensity of a comorbid BAD prevents the use of an SSRI or Clomipramine (Jenike, Baer, Buttolph, 1991) (Case 5).
Signs and symptoms of OCD may improve with habilitative changes, person-centered planning and specific behavioral intervention plans (Low, 1995), while concurrently achieving stabilization of a BAD (Case 5).
There remains a need to establish the prevalence of comorbid BAD and OCD in large series of individuals with DD and mental health concerns. Accurate and concise documentation of clinical observations regarding outcomes in response to pharmacological treatment of these comorbid disorders holds promise regarding improved understanding of their respective etiologies. The establishment of an objective monitoring system incorporating operationalized definitions of observable behavior, judged to be overt expressions of the signs and symptoms of these diorders, allows the safe, efficacious prescription of state of the art pharmacological interventions. Through this process quality of life in individuals with DD, OCD and BAD can be optimized.
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For further information contact:
Robert King, MD
PO Box 3010
Hwy. 11 North
North Bay, ON P1B 8L1