Introduction
Obsessive-compulsive disorder (OCD) is characterized by (i) recurrent and persistent thoughts, urges, or images that, at some point during the disturbance, are experienced as intrusive, unwanted, and overwhelming, leading to anxiety and/or distress (i.e., obsessions) and/or (ii) excessive or senseless repetitive behaviours, mental acts, or rituals that the individual feels driven to perform to decrease resulting anxiety and/or distress or according to rigid rules (i.e., compulsions) 1. Epidemiological studies suggest OCD to be both prevalent, affecting up to 3.1% of the general population, and disabling 2. It is now accepted, however, that a sizable proportion (up to 36%) of patients with OCD may be unable to recognize that their beliefs are false (OCD with poor or absent insight/delusional beliefs) 3. A systematic review found increased severity of obsessive, compulsive, depressive, and anxiety symptoms among OCD patients showing low levels of insight, suggesting poor insight OCD to be simply more severe OCD rather than a standalone condition 3. There are also reports of OCD patients who develop symptoms that closely resemble “true” auditory, visual, tactile, olfactory, and bodily hallucinations 4. Despite some dispute based on earlier longitudinal studies 5, OCD has emerged as a risk factor for schizophrenia in a prospective cohort study with 45 million person-years of follow-up 6.
However, it is unclear how patients with OCD develop other symptoms typical of schizophrenia (such as prominent apathy and avolition) as their illness progresses. One of the first clinicians to tackle this issue was Cabaleiro Goas (1918-1977), probably the most important figure in Galician psychiatry in the second half of the 20th century and very influential in Spanish and Portuguese-speaking countries at his time 7. Goas based his description of a “Genuine Obsessional Psychosis” (GOP) 8 on the work of Jaspers 9 who, using philosophical concepts, differentiated between “understandable” developments characterizing personality disorders and “un-understandable” psychological or “organic” processes typical of schizophrenia and dementia, respectively 8,9. Although Jaspers himself was skeptical about the processual nature of “compulsive neurosis” 9, Goas argued that OCD might sometimes “start as ‘development’ and end as a ‘process’”, meaning that, in GOP, there may be a typical and insidious OCD onset but also an “end state” that is almost indistinguishable from the one described in schizophrenia 8. According to Goas, individuals with GOP always display some degree of premorbid meticulosity, prolixity, perseveration, stubbornness, scrupulosity, or stern and rigid self-criticism that, contingent on some external, somatic, or “intrapsychic” problem 8, can be followed by an obsessive “thrust”* (in Jaspers’ terms) 9 and the onset of a “process-like” downfall **.
In fact Lopez-Ibor, another Spanish author who has greatly influenced the work of Goas on GOP, had also described a “limiting crack to the vital progression” 10 or “a threat to the continuity of the self” 11 in some OCD patients. This concept is consistent with “the appearance of a new factor that can be localized within a brief span of time,” 9 a criterion needed for the characterization of a “process”, according to Jaspers. Goas described GOP as an illness that progressively isolates the patient from problems that are not their own and whose contents they are inclined to “absorb compulsively” 8. According to Goas, the postmorbid personality of GOP forces individuals to severely limit their professional and social activities, often leading to hopelessness and suicidal thoughts 8. Resistance to treatments available at Goas’ time, including “anxiolytics, thymoleptics, or neuroleptics,” given alone or together, electroconvulsive therapy, and psychotherapy, were frequent 8. To Goas, in the later stages of the illness, obsessions and compulsions slowly fade, or there is an adaptation to the presence of symptoms. In any case, the GOP patient would develop a limiting “residuum,” i.e., an accentuation of obsessive traits that already existed in the premorbid personality 8. Although the personality of individuals with GOP is not “devastated” as in schizophrenia, Goas argued that their psychological activity would become “blurred or constrained” to the point of generating an inability to face one’s vital activities 8.
It is difficult to ignore the resemblance between the “processual” characteristics described in GOP and the “negative symptoms” that are so typical of schizophrenia. Although the more recent Anglo-Saxon literature has expanded the use of the term “Obsessional Psychosis” to include patients who do not display negative symptoms but only lower insight toward their obsessions and compulsions, including delusion- or hallucinations-like experiences 12, it remains unclear whether GOP represents a unique standalone condition or simply an end state of severe OCD. Indeed, despite all studies investigating the multiple borders between OCD and schizophrenia 13, we are not aware of any report addressing whether some OCD patients may develop “negative symptoms” classically seen in schizophrenia in the long term, including apathy, avolition, asociality, anhedonia, alogia, or restricted affect 14. We do speculate, however, that these “processual” forms of OCD are overrepresented among treatment-resistant cases who are unable to engage in mobilizing exposure and response prevention (ERPs), are resistant to high-doses of (selective or non-selective) serotonin reuptake inhibitors (SRIs) and are potential candidates for neurocircuitry interventions (including deep brain stimulation [DBS] or ablative surgeries). In the following report, we provide the clinical vignettes of three cases seen in our OCD clinic that resemble the GOP patients described by Goas by developing severe and limiting negative symptoms. These descriptions are based on a chart review complemented by discussions with attending physicians. This report has been approved by the local ethics committee.
Case reports
Case 1
Mr. A, a 64-year-old Caucasian male, is single and currently unemployed, relying on financial support from his two brothers. He has an incomplete higher education and lives alone. The onset of his obsessive-compulsive disorder (OCD) occurred at the age of seven, primarily manifesting as contamination fears and compulsive washing. However, the severity of his symptoms escalated significantly around the age of 20. Mr A contends that the intensity of his OCD symptoms compelled him to discontinue his pursuit of higher education (law school) due to challenges in attendance and academic performance. Notable additional OCD symptoms include fears of harm, characterized by distressing images of “machine guns” and “mutilated bodies”, as well as sexual and religious obsessions. His compulsions encompass mental rituals, checking behaviours, symmetry-related actions, and repetitive rituals. For a description of his negative symptoms, see Table I.
During his initial assessment, Mr. A scored a total of 37 on the Yale-Brown Obsessive Compulsive Scale (YBOCS) and a 7 on the Clinical Global Impression (CGI) scale. His symptoms have significantly impaired various aspects of his life, leading to the abandonment of academic, professional, and recreational pursuits. Ritualized and exhaustive showers, lasting up to four hours daily, have resulted in self-injurious washing behaviours. In addition to his OCD, Mr. A is currently experiencing a major depressive disorder characterized by symptoms such as sadness, anhedonia, irritability, hopelessness, suicidal thoughts, and psychomotor retardation. Regular medication is necessary for him to achieve restful sleep. Rage attacks have led to four psychiatric admissions, although there is no evidence of manic or hypomanic episodes. Mr. A has a measured IQ of 150. Furthermore, he has struggled with alcohol abuse, consuming a bottle of gin every two weeks, and has faced issues related to gambling.
Mr. A has been followed in our clinic for a span of 23 years. Throughout this period, he has been prescribed therapeutic doses of different serotonin reuptake inhibitors (e.g. fluoxetine, clomipramine) augmented by atypical antipsychotics (risperidone, olanzapine, quetiapine, and ziprasidone), typical antipsychotics (thioridazine, levomepromazine and promethazine) and glutamate agents (i.e. topiramate), always with minimal results. He has performed 16 sessions of exposure and response prevention (ERP) but was non-adherent to the exposures and often missed meetings.
Case 2
Mr. B, a 47-year-old individual of mixed ethnicity, is single and unemployed, relying on permanent social security benefits since the age of 22 due to persistent mental health challenges. His initial experience with obsessive-compulsive disorder (OCD) began at the age of eight, but an official diagnosis was only made at age 14 when he reported poorly articulated contamination fears and washing compulsions. During this period, he dealt with distress generated by having OCD by punching his head, biting his arms, and vocalizing his distress. Mr. B’s current struggles encompass fears of self-harm involving knives and forks, sexual/religious obsessions related to fear of performing inappropriate behaviours, and ongoing contamination concerns. Prominent compulsive behaviours include tic-like compulsions, specifically grimacing to assess the physical integrity of his face, in addition to other washing, checking, and mental compulsions. For a description of his negative symptoms, see Table II.
Upon initial assessment, Mr. B scored 38 on the Yale-Brown Obsessive Compulsive Scale (YBOCS) and a 6 on the Clinical Global Impression (CGI) scale. He withdrew from his job as a fast-food cashier two decades ago due to the impact of his symptoms and the social challenges associated with constant grimacing. While he currently denies depressive symptoms, historical records indicate a severe major depressive episode almost 20 years ago, marked by prolonged periods in bed, notable sadness, anhedonia, thoughts of self-harm, and decreased appetite leading to weight loss. Mr. B has an IQ of 114.
Over the course of more than 20 years at our clinic, Mr. B has undergone treatment involving therapeutic doses of fluoxetine and sertraline combined with either risperidone or olanzapine. The combination of sertraline (200 mg/day) and olanzapine (10 mg/day) has proven the most effective, resulting in complete remission of depressive symptoms and partial remission of his OCD. While he engaged in some exposure and response prevention (ERP) sessions that contributed positively to his symptoms, negative aspects of his condition persist unchanged.
Case 3
Mr. C, a 41-year-old Caucasian male, is single and unemployed, relying on permanent social security benefits since the age of 30 due to enduring mental health problems. The onset of Mr. C’s symptoms began at the age of 20, and involved the emergence of peculiar behaviours, including jumping, hitting, kicking, freezing, slamming doors, and an insistence on touching objects or people. He struggled to articulate a cognitive rationale for these actions, merely stating that he felt compelled to perform them. Currently, Mr. C’s primary obsessive-compulsive (OC) symptoms fall within the miscellaneous dimension of the Dimensional Yale-Brown Obsessive-Compulsive Scale (D-YBOCS). These symptoms predominantly manifest as tic-like compulsions involving the need to touch, tap, or rub.
However, Mr. C also exhibits ritualistic repetition, contamination concerns, washing compulsions, sexual/religious obsessions, fear of harm, and checking compulsions. His Clinical Global Impression (CGI) score is 6, demonstrating a very severe level of illness. His symptoms significantly impede his ability to sustain professional and social relationships. At home, he requires continuous supervision from his parents as some of his rituals occasionally result in broken objects, and he may exhibit other impulsive or “unpredictable” behaviours. There are concerns about his suboptimal cognitive functioning, exacerbated by his lack of insight into his problematic actions. IQ was rated at 57. He adamantly believes that he does not require psychiatric medications. Additionally, he has a history of traumatic brain injury at the age of 11. For a description of his negative symptoms, see Table III
Throughout his follow-up, Mr. C underwent treatment with therapeutic doses of fluoxetine and paroxetine, supplemented by risperidone, olanzapine, pimozide, and thioridazine. Unfortunately, these interventions yielded limited results, with high-dose SRIs eventually leading to treatment-emergent psychosis. Consequently, electroconvulsive therapy (ECT) was administered, resulting in the remission of positive symptoms but a persistence of compulsive and negative symptoms. Despite multiple attempts at exposure and response prevention (ERP) sessions, Mr. C struggled to consistently adhere to the recommendations. Following a diagnostic discussion, a decision was made to maintain Mr. C on a combination of clozapine and lithium. While this approach prevented further deterioration of his obsessive-compulsive disorder (OCD) symptoms, Mr. C remains severely incapacitated to date.
Summary of cases
As indicated in Table IV, all our cases exhibited varying degrees of avolition and asociality. Additionally, one case displayed anhedonia, attributed to a current major depressive disorder. Two cases demonstrated restricted affect, and all individuals showed a reduction in the variety of emotions expressed during consultations. Furthermore, two cases encountered difficulties articulating their internal experiences beyond specific inquiries, indicative of alogia. Notably, avoidance emerged as a significant factor in all negative symptoms, particularly impacting the avolition-apathy domain. However, it remained unclear whether avoidance alone could account for the entirety of experiential negative symptoms (i.e. avolition, asociality and anhedonia). Delusionality did not serve as an explanation for negative symptoms in any patient. Despite all patients being on antipsychotics, the absence of clear-cut parkinsonism or noticeable sedation attributed to the medication suggested that side effects were not a predominant factor in generating negative symptoms.
Discussion
In this case series, we describe three patients who exhibited a multiplicity of negative symptoms consistent with a “processual” type of OCD or GOP. Our results can be summarized as follows: Firstly, all cases exhibited varying degrees of avolition, asociality, and anhedonia that could be attributed, at least partially, to underlying OCD avoidance or depressive symptoms. Secondly, all cases also showed expressive deficits that are less likely to be secondary, including restricted affect, diminished range of emotional expression, and alogia. Finally, there was no evidence that delusionality or the use of therapeutic doses of antipsychotics played any role in the negative symptoms exhibited by our OCD patients.
The fact that the experiential domains of negative symptoms (avolition, asociality, anhedonia) could be partially ascribed to other symptoms has some interesting implications. From the perspective of traditional phenomenological psychopathology, because the origin of negative symptoms is traceable (or “understandable” in Jaspers’ terms) 9, describing GOP as a “genuine” obsessive psychosis or a “processual” form of OCD seems inadequate. Although we cannot exclude the existence of cases in which avolition, asociality, and anhedonia are unequivocally “primary” in some people with OCD, it is important to underline that these three patients were selected from individuals attending our OCD clinic specifically for having significant negative symptoms.
From a diagnostic point of view, our series suggests that severe OCD cases can be overlooked or misdiagnosed as individuals with schizophrenia if (i) too much importance is given to negative symptoms as a core and pathognomonic component of schizophrenia, (ii) negative symptoms are poorly assessed and assumed to be primary at face value, or (iii) the role of avoidance and depressive symptoms as a cause of negative symptoms in other non-schizophrenia conditions is downplayed. From a therapeutic standpoint, our findings also suggest that approaches other than traditional strategies focused on the use of clozapine may be useful for managing negative symptoms in the context of disorders other than schizophrenia.
In contrast to our findings on the experiential domains of negative symptoms, the presence of expressive deficits across our OCD patients with GOP is consistent with the findings of a systematic review 16. This review described the emotional reactions of individuals with OCD in response to social emotion-inducing stimuli, ranging from a slower initial velocity of involuntary laughing movements in response to a humorous movie clip of Mr. Bean 17 to more incongruent emotive feelings and facial expressions than healthy participants to video clips thought to generate amusement, fear, surprise, anger, sadness, disgust, or neutrality 18,19. Other studies have described OCD to be associated with increased apathy 20 and bradykinesia 21. Curiously, “psychic akinesia”, a condition resulting from basal ganglia (particularly pallidal) lesions, involves both decreased motivation and compulsive behavior 22.
Because the roles of avoidance and depression seem to be more prominent in the genesis of negative symptoms in OCD patients than delusional beliefs and the co-prescription of dopamine blockers, our findings at least provide some preliminary evidence supporting the minor role played by decreased insight into the experiential component of the negative syndrome and the safety of therapeutic doses of antipsychotics usually employed in augmentation of SRIs in the treatment of OCD 23. Also, although both SRIs 24 and antipsychotics 25 have been implicated to different degrees in the appearance of apathy and other negative symptoms, it is important to note that the negative symptoms reported in the present series appeared before the introduction of medications and remained unaltered during several years of follow-up (and numerous dose adjustments).
Because we have been following these patients for more than two decades, it is tempting to suggest that some of their clinical characteristics, including negative symptoms, are partially contingent on greater duration of illness, as in schizophrenia 26. Indeed, there is increasing evidence that both the brain and the phenotypical expression of OCD patients may show changes with the progression of illness 27. This trend has prompted some to advocate for a neuroprogressive model of OCD, akin to what’s observed in other neuropsychiatric conditions 27,28. Neuroprogression in the context of OCD may be associated with changes in the motivations underlying compulsive behaviours (e.g., from fear or reward to excessive habit formation) 29,30, alterations in white matter integrity 31, changes in brain volume (such as increased striatal volume) 32-35, the emergence of other psychiatric 36,37 and medical comorbidities 38, and increased mortality 39. However, it’s worth acknowledging that our initial assessments did not specifically address the presence of negative symptoms. Consequently, it’s plausible that we may have overlooked some negative symptoms and other symptoms rooted in neurodevelopment during the early stages of the illness 40.
Our paper has some significant limitations. Firstly, it was based on a syndrome (GOF) whose description combined complex philosophical concepts that are difficult to objectify and may not be fully captured by the concept of “negative symptoms”. Secondly, it included a small case series of OCD patients with prominent negative symptoms that were “cherry-picked” from a larger sample seen in a public OCD clinic in Brazil. Therefore, the exact rate and generalizability of this phenomenon to OCD patients in other settings are impossible to establish. Thirdly, negative symptoms were qualitatively assessed with a checklist based on a chart review complemented by discussions with other team members who have regularly seen these patients for some period. Therefore, future studies could employ valid measures to assess negative symptoms, their correlates, and their impact on treatment outcomes in regular OCD patients. It is also possible that we have overlooked more “ordinary” autistic traits associated with OCD 40 in our initial assessments, only to consider them as present in our more recent evaluations. Finally, because no systematic evaluation of treatments was employed, it is difficult to provide precise data on the outcomes of these patients.
Conflict of interest statement
The authors declare no conflicts of interest.
Funding
The first author is currently receiving grant # 313162/2023-9 from Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq), and the grant # E-26 200.950/2021 from Fundação de Amparo à Pesquisa do Estado do Rio de Janeiro (FAPERJ). The funder had no role in the design, data collection, data analysis, and reporting of this study.
Authors’ contributions
The first author provided substantial contributions to the conception and interpretation of data for the work; drafted the first version of the manuscript; approved the version to be published, and agreed to be accountable for all aspects of the work; The second and third authors significantly contributed to the acquisition and analysis of data for the work; reviewed it critically for important intellectual content; approved the version to be published, and agreed to be accountable for all aspects of the work. The fourth and fifth authors contributed to the conception of the work, reviewed it critically for important intellectual content, approved the version to be published, and agreed to be accountable for all aspects of the work.
Ethical consideration
Study approval statement: this study protocol was reviewed and approved by the Institute of Psychiatry of the Federal University of Rio de Janeiro Ethics Committee, approval number 77672724.9.0000.5263.
Consent to participate statement: written informed consent was obtained from participants to participate in the study.
Figures and tables
| Mr. A was a tall and overweight man with a long, greyish beard and dishevelled hair. He frequently wore oversized, perspiring t-shirts, contributing to an unhygienic and somewhat unpleasant appearance. During regular meetings, an awkward and stiff emotional expression was evident, accompanied by dramatic gesticulations indicative of anger and desperation. In various situations, Mr. A displayed manneristic behaviour, such as long-windedly and robotically praising female doctors and attempting to kiss their hands during consultations. While his spontaneous speech was not lacking in quantity, it tended to be repetitive and monothematic, focusing either on one-sided expressions of distress related to OCD symptoms or reminiscences of past sexual encounters. |
| It is noteworthy that Mr. A adamantly denied any romantic or intimate relationships for the past 20 years. Unemployed for almost the same time, he leads a completely sedentary lifestyle, resulting in Grade III obesity. He lacked initiative, often sleeping throughout the afternoon. To circumvent prolonged showers, Mr. A already refrained from bathing for up to six months, confining himself to his living space most of the time. He disavowed engaging in any pleasurable or recreational activities aside from alcohol consumption, gambling, and food. Although he expressed an interest in forming close relationships with women, his lack of consistent and proactive initiative hindered this pursuit. His brothers had to monitor his behaviour daily, as he would frequently cease responding to calls due to being either intoxicated or depressed. |
| Mr. B is a short, slender man with grown mutton chops. Upon initial interaction, a noticeable decrease in the expression of affect is apparent in his facial expressions, vocal tone, and gestures. Remarkably, he is consistently accompanied by his father during consultations, giving the impression that he is unable to speak for himself. While initially considered a symptom of family accommodation, Mr. B himself was often laconic. Despite his willingness to help, he struggled to provide even most basic personal information, such as his own phone number. |
| For nearly 20 years, Mr. B has been unemployed. His social circle is limited to immediate family members, and he describes a lack of motivation to form connections with others. He avoids using knives and forks, cutting his nails, entering rooms with tools, and regularly showering due to the fear of encountering razor blades in the bathroom. Despite having been a successful karate fighter in the past, he abandoned the practice out of concern about potential success resulting from a pact with the devil. Consequently, his interest, motivation, and active participation in recreational activities are severely restricted. |
| Mr. C is a man of medium stature who appears younger than his actual age. During interactions with his attending psychiatrist, he consistently engages in a complex and socially awkward ritual, firmly gripping the doctor’s hand and shaking it for an unusually prolonged period. His facial expressions often reveal a stiff emotional demeanour, accompanied by manneristic gestures. Mr. C struggles to address his symptoms spontaneously, frequently diverting conversations to topics like soccer and TV programs. Elaborating on his internal experiences, reasons for treatment, and long-term plans proves challenging. His discourse tends to be circumstantial and superficial, and he exhibits some degree of puerility. |
| According to Mr. C’s family, they often need to bring him food in bed as he remains uncommunicative in his room. He frequently urinates in his pants due to being stuck in the same position for extended periods, making it difficult for him to reach the toilet promptly. The family reports that he remains in a dirty state for prolonged periods, seemingly indifferent to the smell of urine in his clothes. During the day, Mr. C leaves the house and spends extended periods in a nearby bush. His approach to people is often odd and unconventional. He is also quite impersistent for frequently neglecting or abandoning small chores requested by his parents and sisters at home. |
| Avolition-Apathy | CASE 1 | CASE 2 | CASE 3 | |
|---|---|---|---|---|
| Avolition | Reduced initiation and persistence of goal-directed activity due to reduced motivation | ✓ | ✓ | ✓ |
| Asociality | Reduced social interactions and initiative due to decreased motivation for and interest in forming and maintaining relationships with others | ✓ | ✓ | ✓ |
| Anhedonia | Reduced experience of pleasure for a variety of activities or events, during or for future anticipated activities or events | ✓ | ||
| Expressive deficit | ||||
| Restricted affect | Reduction in the intensity of a specific emotional expression during conversation | ✓ | ✓ | |
| Diminished range of emotions | Reduction in the number of different emotional expressions during conversation | ✓ | ✓ | ✓ |
| Alogia | Reduction in quantity of words spoken and in spontaneous elaboration (i.e., amount of information spontaneously given beyond what is needed) | ✓ | ✓ | |
| Potential causes of negative symptoms | ||||
| Obsessions/avoidance | Negative symptoms that are caused by fear related to obsessions, leading to avoidant behaviours | ✓ | ✓ | ✓ |
| Depression | Negative symptoms caused by sadness and anergia emerging or not from OCD | ✓ | ||
| Psychosis | Negative symptoms caused by primary paranoid delusions leading for instance, to barricading behaviours | |||
| Drug-induced | Negative symptoms that are side effects of medication, such as neuroleptic induced Parkisonism or sedation | |||
| GOP = Genuine Obsessive Psychosis; Definitions based on National Institute of Mental Health-Measurement and Treatment Research to Improve Cognition in Schizophrenia (NIMH–MATRICS) consensus statement 15 and on 14. | ||||
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