Psychosomatics 49:77-81, January-February 2008
doi: 10.1176/appi.psy.49.1.77
© 2008 Academy of Psychosomatic Medicine
Olfactory Reference Syndrome
Aurelia N. Bizamcer, M.D., Ph.D.,
William R. Dubin, M.D., and
Bernadette Hayburn, Psy.D.
Received November 13, 2006; revised February 13, 2007; accepted February 21, 2007. From the Dept. of Psychiatry and Behavioral Sciences, Temple University Hospital–Episcopal Campus, Philadelphia, PA. Send correspondence and reprint requests to Aurelia N. Bizamcer, M.D., Dept. of Psychiatry and Behavioral Sciences, Temple University Hospital–Episcopal Campus, 100 E. Lehigh Ave., Suite 105, Philadelphia, PA 19125. e-mail: bizamcer{at}temple.edu
© 2008 The Academy of Psychosomatic Medicine
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INTRODUCTION
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Olfactory reference syndrome (ORS) is a variant of delusional disorder of somatic type in which one believes that his or her body emits a foul odor that makes people react in a negative way to their body.1 Which category ORS fits into diagnostically is still being disputed. One of the most important corollaries of the diagnostic debate is the choice of a therapeutic agent; some psychiatrists treat the disorder with antipsychotic medication, others with antidepressants, primarily serotonin reuptake inhibitors (SSRIs), and some clinicians use both.2–9 The authors present a case of ORS and then discuss the literature with regard to diagnosis and treatment and then talk about how the they approached the treatment of this patient with ORS.
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Case Report
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"Ms. T" is a 26-year-old single woman who has been followed in the Psychiatry Outpatient Department at Temple University Hospital for 32 months because of the belief that she smells like "garbage" and that people are repulsed by her odor. She interpreted somebody sneezing, wiping their nose, scratching their head, or covering their face, as a response to her unpleasant body odor. Intermittently, she also claimed to smell this odor. She believed that lots of animals follow her because of her body odor.
She also reported mild-to-moderate anxiety symptoms, related to fear of heights, bridges, elevators, airplanes, and anxiety related to public speaking. However, she never reported or gave evidence of avoidant behavior related to these issues. The patient expressed discomfort regarding eating in public but was not disturbed by this. Ms. T had no history of panic attacks, free-floating anxiety, or obsessive-compulsive symptoms. She was comfortable with her appearance.
Ms T’s preteen years were unremarkable. She had good grades and friends and was on the honor roll in school until she started thinking that people were repulsed by her body odor. She started menstruating at age 14, the time of the onset of her symptoms. She recalls that, while riding a bus home from school, she heard a man making an observation about wearing deodorant that she believes was directed at her. She became preoccupied with the thought that she smells bad, increased her showering, watched her diet, and even purchased herbal supplements to "detoxify" her body. However, she continued to notice behaviors in people around her that made her believe she emits a foul odor. She became isolated at school, where most of the incidents occurred, started missing classes, and her grades dropped. She did not tell her mother of these beliefs, but, instead, every morning, left home and pretended that she went to school; instead, she often hid in backyards or in a park until the usual time to return home. She was first admitted to a psychiatric hospital when she was in the 11th grade for 5 days on an involuntary commitment because of her behavior. The patient had no psychiatric follow-up after the discharge. She dropped out of high school in 12th grade, but managed to get a GED and went to college, where she obtained passing grades for the first semester but was dismissed soon afterward because of absenteeism and poor grades. Her pattern of behavior in college was similar to that in high school, with problems in large classes and performing in public.
She eventually found a job as a secretary. Six months after starting the job she stopped going to the office without notifying the boss or her own mother. At home, she would just stay in bed and watch TV. Her employer thought that she might be depressed and recommended that she see a psychiatrist. She was admitted to a psychiatric hospital where she spent 5 days, was diagnosed with Psychosis, Not Otherwise Specified, and referred to our clinic without being placed on psychotropic medication.
Ms. T has no significant medical history, and her laboratory studies were within normal limits. An MRI scan of the brain was normal. She has no significant family, medical, psychiatric, or substance-abuse history.
She received a battery of psychological tests that included the Wechsler Intelligence Scale for Adults, 3rd Edition (WAIS–III), the Faces I and II of the Wechsler Adult Memory Scale, 3rd Edition (WMS–III), the Bender Visual-Motor Gestalt Test, the Benton Visual Retention Test, 5th Edition, the Controlled Oral Word Association Test (COWAT), the Hooper Visual Organization Test, the Manual Finger-Tapping Test, the Rey Auditory-Verbal Learning Test, the Trail-Making Test A and B, the Lateral Dominance Examination, the Minnesota Multiphasic Personality Inventory (MMPI–2), the House–Tree–Person Test, and the Thematic Apperception Test. The results were within normal-average on the verbal component but in the lower range on the performance component. Her visual-spatial organization skills were mildly deficient. Among the personality traits, alienation, inadequacy, helplessness, passivity, and avoidance were prominent, suggesting a Cluster C personality type.
Initial mental status examination was remarkable for nonspontaneous, low-volume, aprosodic speech, blunted affect, occasional referential delusions as described above, and occasional olfactory hallucinations, although these were far less frequent than the referential thinking. Ms. T scored 30 out of 30 on the Mini-Mental State Exam. She exhibited partial insight, and her judgment was adequate.
The working diagnosis was delusional disorder, somatic type, and mixed personality traits, including discomfort in social situations, dependence on her mother, and sensitivity to rejection, isolation of affect, social isolation, and persistent odd beliefs.
At the time that Ms T was transferred to the care of one of the authors (ANB) 2 years ago, she had been in supportive psychotherapy for 7 months with another psychiatrist, with no change in symptoms. She was started on aripiprazole 10 mg daily. Although some supportive therapy was offered, cognitive-behavior therapy elements were also introduced; these included role-playing, analyzing behavioral scenarios, and establishing and testing behavioral schedules. Automatic thoughts related to other people’s reactions and core beliefs regarding feelings of inadequacy were identified and discussed, as well.
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Discussion
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Dr. Dubin (WRD): It seems to me, Dr. Bizamcer, that ORS appears to be a delusional disorder. From the sparse literature that exists, however, there still seem to be questions regarding the diagnostic classification of this disorder.
Dr. Bizamcer (ANB): The first paper about ORS was published by Pryse-Philips in 1971.1 He identified 100 cases of patients who experienced olfactory hallucinations of unpleasant personal body odor and the belief that people reacted negatively toward them because of it. All but 36 cases met diagnostic criteria for schizophrenia, depression, or epilepsy. For these cases, he introduced the term "olfactory reference syndrome."
The remaining literature on ORS is represented by case reports. These papers include the following diagnoses: "olfactory paranoid syndrome,"10 delusional disorder,11 bipolar disorder,2 major depressive disorder,9 body dysmorphic disorder,6 obsessive-compulsive disorder with poor insight,4 stimulants-induced disorder,7 and alcohol dependence.5
WRD: I am aware that some clinicians consider this a culturally-bound syndrome.
ANB: Some researchers have drawn parallels between ORS and certain syndromes believed to be culturally specific, such as the taijin kyofu described in Japan as a form of social phobia, ranging from simple social avoidance to a delusional form, caused by beliefs about having physical characteristics that may repel others.12–15 Taijin kyofu has a subtype, jiko-shu-kyofu, that involves a belief that one has an unpleasant body odor.9,16 Kobayashi and Kato,9 reviewing Japanese psychiatric literature, report that some authors consider this Japanese variant of ORS as a psychopathological entity that falls between psychosis and neurosis.
WRD: Is there any evidence that ORS is a neurological disorder with psychiatric manifestations?
ANB: In a few cases of ORS, researchers have been able to establish a link to central nervous system pathology. Toone17 reported on a patient with ORS who had an arteriovenous malformation in the right temporal lobe, and seizures. Devinsky et al.18 described ORS found in frontotemporal lobe epilepsy, and Konuk et al.19 showed frontotemporal hypoperfusion in one case of ORS.
WRD: Do personality factors have any role in this disorder?
ANB: Although most of the literature focuses on diagnostic classification or pharmacological treatment, several authors have noted specific personality traits in patients with ORS. Pryse-Philips1 observed that the ORS patients had a tendency toward self-criticism, feelings of inferiority, shyness, and problems expressing emotions. Videbech10 also found that certain personality traits were characteristic of the "olfactory paranoid syndrome." These traits included: insecurity, perfectionism, inferiority, and "sensitiveness."
WRD: Given the wide array of possible diagnoses, what type of treatment is recommended in the literature?
ANB: Not all papers have reported treatment. However, in those that have, some clinicians treated the disorder with antipsychotic medication, others with antidepressants, primarily SSRIs, and some used both.2–9 There are sparse data on the outcome of these treatments (Table 1). With the three cases that were presented as having neurological etiologies, treatment was not addressed.
WRD: Given the lack of evidence for both diagnosis and treatment, how did you decide to approach this case using a combination of aripiprazole and cognitive-behavior therapy (CBT)?
ANB: Ms. T’s referential thoughts were undeniably delusional, and the olfactory hallucinations were intermittent. Therefore, we decided to try aripiprazole because of its reported low side-effect burden and the fact that the patient liked the idea of once-daily dosing.20 The patient had been offered but refused medication by her previous therapist, always citing the need for additional information.
WRD: Well, I can understand why you chose an antipsychotic. Many psychiatrists would have just focused on the delusions, and it is realistic to conceive that this patient might have become a medication management-only patient. Why did you decide to use CBT as well?
ANB: She was initially ambivalent about long-term medication treatment. She actually requested to come to therapy biweekly because she felt that this would enable her to stay focused on and pursue her goals. I decided to pursue therapy with CBT because of the patient’s lack of insight and her inability to view the world in abstract terms, which made her less appropriate for insight-oriented psychotherapy. She was a concrete thinker, who would identify problems, but was unable to develop strategies to resolve them. She had very clear goals that that included getting through a job interview, having social interactions with coworkers, having daily lunch in the cafeteria, and developing alternatives to her time-wasting behaviors. I felt that this type of thought process was well suited for CBT.
WRD: You have now been treating the patient for 25 months. How is she doing?
ANB: She currently works full-time and was recently promoted to a senior position. She has been at her current job for over a year and is attending evening undergraduate courses and receiving good grades. Her goal is to become a certified financial planner and purchase her own home. She also recently began expressing an interest in dating. Although the referential thoughts continue, they are less intense, and Ms. T sometimes expresses doubt regarding the veracity of her perceptions. She does not experience the foul odor herself. However, she continues to interpret gestures made by other people; for instance, coworkers waving to colleagues or scratching their head, as reactions toward her body odor. Although we suggested increasing the dose of aripiprazole, the patient stated that she preferred achieving her professional and educational goals, rather than full remission of symptoms, and she did not want the dose to be increased.
WRD: There is one final point that is so important to achieving therapeutic success and is not often talked about in the current "medication-management" era. It is clear to me that the therapeutic gains were accomplished because of the therapeutic alliance that you formed with the patient, which allowed you to effectively engage her in treatment. In contrast, the patient repeatedly refused medication over a 7-month period with her previous psychiatrist. Horvath21 observed that the quality of the therapeutic alliance is a robust predictor of outcome regardless of the type of therapy or diagnosis:21 "the development of a good alliance with clients includes not only a positive, emphatic disposition by the therapist, but also a collaborative framework, a partnership in which clients see themselves as active, respected participants." What do you think were the ingredients that enabled you to form a successful therapeutic alliance?
ANB: There were several elements that fostered the relationship. In contrast to her previous feeling of having no control in her life, and her sense of inadequacy, she was empowered to shape her own treatment. She was allowed to establish the treatment goals, determine the frequency of visits, and the dose and frequency of medication. This led to a sense of trust in the therapist, and, thus, she was willing to experiment with new behaviors, including taking medication.
WRD: ORS remains a poorly-defined symptom, with ambiguity as to diagnosis and treatment. Yet, despite these significant limitations, this case demonstrates that judicious medication use, combined with psychotherapy, in the context of a therapeutic alliance can yield significant therapeutic success. Thank you for presenting this case.
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REFERENCES
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- Pryse-Philips W: An olfactory reference syndrome. Acta Psychiatr Scand 1971; 47:484–509[Medline]
- Davidson M, Mukherjee S: Progression of olfactory reference syndrome to mania: a case report. Am J Psychiatry 1982; 139:1623–1624[Free Full Text]
- Dominguez RA, Puig A: Olfactory reference syndrome responds to clomipramine but not to fluoxetine: a case report. J Clin Psychiatry 1997; 58:497–498[Medline]
- Stein DJ, Le Roux L, Bouwer C, et al: Is olfactory reference syndrome an obsessive-compulsive spectrum disorder? two cases and a discussion. J Neuropsychiatry 1998; 10:96–99[Abstract/Free Full Text]
- Warnecke M, Bahlmann M, Soyka M: Olfactory delusional syndrome and alcoholism. Psychiatrische Praxis 2002; 29:211–213[CrossRef][Medline]
- Lochner C, Stein DJ: Olfactory reference syndrome: diagnostic criteria and differential diagnosis. J Postgraduate Med 2003; 49:328–331
- Luckhaus C, Jacob C, Zielasek J, et al: Olfactory reference syndrome manifests in a variety of psychiatric disorders. Int J Psychiatry Clin Pract 2003; 7:41–44[CrossRef]
- Sayar K, Aksu G: Olfaktor referans sendromu: bir olgu sunumu. Bull Clin Psychopharmacol 2003; 13:183–187
- Kobayashi T, Kato S: Senile depression with olfactory reference syndrome: a psychopathological review. Psychogeriatrics 2005; 5:55–63[CrossRef]
- Videbech T: Chronic olfactory paranoid syndromes: a contribution to the psychopathology of the sense of smell. Acta Psychiatr Scand 1966; 42:183–213[Medline]
- Kopecek M, Horáîcek J, Motlová L, et al: Paranoidní syndrom vlastního zápachu v obraze 18FDG PET (kazuistika). Psychiatrie 2003; 7:214–217
- Tanaka-Matsumi J: Taijin kyofusho: diagnostic and cultural issues in Japanese psychiatry. Culture Med Psychiatry 1979; 3:231–245[CrossRef]
- Kleinknecht RA, Dinnel DL, Kleinknecht EE, et al: Cultural factors in social anxiety: a comparison of social phobia symptoms and taijin kyofusho. J Anxiety Disord 1997; 11:157–177[CrossRef][Medline]
- Matsunaga H, Kiriike N, Matsui T, et al: Taijin kyofusho: a form of social anxiety disorder that responds to serotonin reuptake inhibitors? Int J Neuropsychopharmacol 2001; 4:231–237[CrossRef][Medline]
- Tarumi S, Ichimyia A, Yamada S, et al: Taijin kyofusho in university students: patterns of fear and predisposition to the offensive variant. Transcultural Psychiatry 2004; 41:533–546[Abstract]
- Suzuki K, Takei N, Iwata Y, et al: Do olfactory reference syndrome and jiko-shu-kyofu (a subtype of taijin-kyofu) share a common entity? Acta Psychiatr Scand 2004; 109:150–155[CrossRef][Medline]
- Toone BK: Psychomotor seizures, arteriovenous malformation, and the olfactory reference syndrome: a case report. Acta Psychiatr Scand 1978; 58:61–66[CrossRef][Medline]
- Devinsky O, Khan S, Alper K: Olfactory reference syndrome in a patient with partial epilepsy. Neuropsychiatry Neuropsychol Behav Neurol 1998; 11:103–105[Medline]
- Konuk N, Atik L, Atasoy N: Birol ugur M: frontotemporal hypoperfusion detected with (99m) Tc HMPAO spect in a patient with olfactory reference syndrome. Gen Hosp Psychiatry 2006; 28:174–177[CrossRef][Medline]
- Practice Guideline for the Treatment of Patients with Schizophrenia, 2nd Edition. American Psychiatric Association Practice Guidelines. Washington, DC, American Psychiatric Publishing, Inc., 2004, pp 15-16
- Horvath AO: The therapeutic relationship: from transference to alliance. J Clin Psychol 2000; 56:163–173[CrossRef][Medline]
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INTRODUCTION
Case Report
