General Hospital Psychiatry 36 (2014) 760.e1–760.e3

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Case Report

Olfactory reference syndrome: a still open nosological and treatment debate Jule Ane Ferreira, M.D. a, Renata P. Dallaqua a, Leonardo F. Fontenelle, M.D., Ph.D. b, Albina R. Torres, M.D., Ph.D. a,⁎ a b

Botucatu Medical School, São Paulo State University–Univ Estadual Paulista (Unesp), Brazil Anxiety and Depression Research Program, Institute of Psychiatry, Federal University of Rio de Janeiro & D’Or Institute for Research and Education, Brazil

a r t i c l e

i n f o

Article history: Received 20 November 2013 Revised 4 June 2014 Accepted 5 June 2014 Keywords: Olfactory reference syndrome Diagnostic criteria Differential diagnoses Nosological classification Obsessive–compulsive disorder

a b s t r a c t Objective: The objective was to report a case of olfactory reference syndrome (ORS) with several co-occurring disorders and to discuss ORS differential diagnoses, diagnostic criteria and classification. Method: Case report. Results: A 37-year-old married woman presented overvalued ideas of having bad breath since adolescence. She met current diagnostic criteria for social anxiety disorder, specific phobia, obsessive–compulsive disorder, generalized anxiety disorder, body dysmorphic disorder and major depressive disorder. ORS similarities and differences with some related disorders are discussed. Conclusion: Further studies regarding symptoms, biomarkers and outcomes are needed to fully disentangle ORS from existing depressive, anxiety and obsessive–compulsive spectrum disorders. © 2014 Elsevier Inc. All rights reserved.

1. Introduction Olfactory reference syndrome (ORS) is a condition in which a person mistakenly believes he or she exudes an unpleasant body odor. The odor is not perceived by others and is usually described by patients as originating in regions naturally associated with bad odor, like the mouth, underarms and genital/anal regions. This belief is usually accompanied by ideas of reference and repetitive behaviors, including sniffing the body, bathing excessively or attempting to mask the odor [1–5]. Sufferers also frequently seek other health professionals (e.g., dentists, dermatologists, gastroenterologists, ENT specialists), trying to solve the alleged problem [2,4–6]. Embarrassment, suffering and social avoidance are the rule, with considerable impact on the individual’s functioning [4,7,8]. ORS has received numerous denominations throughout history, including olfactory paranoid syndrome, chronic olfactory delusional syndrome, monosymptomatic hypochondriasis, delusional or psychosomatic halitosis, olfactory hallucination and parosmia [3,4]. The Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition, Text Revision (DSM-IV-TR) [9] classified ORS as a delusional disorder, but in DSM, Fifth Edition (DSM5) [10], it is under the heading of other obsessive–compulsive and related disorders, where it is mentioned as a synonym of Jikoshu-kyofu, a condition described in eastern cultures.

⁎ Corresponding author. Departamento de Neurologia, Psicologia e Psiquiatria, Faculdade de Medicina de Botucatu (FMB)–UNESP, Distrito de Rubião Jr., Botucatu-SP (18.618-970), Brazil. Tel.: +55 14 38116260, +55 14 38801220. E-mail addresses: [email protected], [email protected] (A.R. Torres). http://dx.doi.org/10.1016/j.genhosppsych.2014.06.001 0163-8343/© 2014 Elsevier Inc. All rights reserved.

We report a case of ORS in which symptoms of various disorders overlap, generating a complex diagnostic picture. By describing this challenging scenario, we specifically wanted to map ORS frontiers with other more clearly defined disorders, thus identifying areas of controversial (or at least blurred) boundaries that indicate areas for future research. 2. Case report Mrs. A, a 37-year-old married Caucasian woman and mother of two daughters, sought help at our university in December 2011. Since age 7, she reported being afraid of water and extremely careful when drinking or bathing for fear of drowning. She also avoided going to dentists for fear of water and needles. At age 11, she was sexually abused by a cousin. During adolescence, her self-esteem worsened, and she started feeling ashamed of her physical appearance and especially of her “bad breath.” Mrs. A quit school in the eighth grade because she thought she “offended/harmed” people with her halitosis and ugliness, becoming increasingly isolated. She also presented ideas of reference; compulsive washing/cleaning of the hands, teeth, home utensils and clothes; excessive worrying about her daily routine; insomnia; irritability; periods of depressive mood and anhedonia. She married at age 21, but the relationship with her husband is troubled. She presented panic attacks during her second pregnancy. Mrs. A initially sought treatment at age 19 and, since then, has used several medications (amitriptyline, clomipramine, fluoxetine, sertraline, paroxetine, citalopram, diazepam, risperidone and lithium) prescribed by different services. However, these were mostly used irregularly and at subtherapeutic doses since

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Mrs. A often developed side effects, with no improvement. In her first psychiatric examination, Mrs. A was well dressed and showed no evidence of halitosis, but presented long-winded speech, phobic and obsessive thoughts, depressive and anxious mood, hypobulia and overvalued ideas concerning her appearance and breath (main complaint). We attempted to reintroduce different selective serotonin reuptake inhibitors (SSRIs; paroxetine, citalopram, sertraline), but Mrs. A discontinued them due to side effects. For 8 months, she used fluoxetine (maximum tolerated dose: 40 mg/day) and underwent psychotherapy, but showed no improvement. For 10 weeks now, she has been taking escitalopram 10 mg/day, so far with no response. Neuroleptics were not prescribed so far because of their poor tolerability profile (compared to SSRIs) and because a previous trial with risperidone was described as being ineffective. In the Structured Clinical Interview for DSM-IV Axis I Disorders [11], she met current diagnostic criteria for social anxiety disorder, specific phobia, obsessive–compulsive disorder (OCD), generalized anxiety disorder (GAD), body dysmorphic disorder (BDD) and major depressive disorder (MDD) and past criteria for panic disorder. In the first evaluation, her total score in the Brown Assessment of Beliefs Scale [12] was 21 (maximum: 24), indicating ideas of reference and poor insight. 3. Discussion Whereas in the DSM-IV-TR [9] ORS was considered a somatic delusional disorder, also mentioned in the social phobia section, in the DSM5 [10] it is classified along with other OCD-related disorders. In fact, it overlaps with several conditions but also presents important differences from them all, including symptoms profile, comorbidity and treatment response [3,4,7,13]. To address the diagnostic dilemma that this case poses, the main clinical features of the most important differential diagnoses of ORS are briefly discussed below. 3.1. Delusional disorder Patients with ORS are not always delusional; they may present overvalued ideas and even good insight [6]. Furthermore, many patients respond to SSRI monotherapy, but not to antipsychotics [3,5]. However, as delusional BDD [14], delusional ORS might be impossible to differentiate from delusional disorder, somatic type. Whether delusional ORS responds to SSRIs similarly to nondelusional ORS is still unclear. 3.2. Social anxiety disorder (SAD) Despite common concerns about social disapproval, shame, ideas of reference and social avoidance, in SAD, the level of insight is usually better [3], and the main fear is of presenting a bad performance in social situations, not of exhaling a bad odor. 3.3. OCD Some ORS avoidant and repetitive behaviors (e.g., checking the odor, excessive hygiene and reassurance seeking) are similar to OCD [2] and can be seen as “safety seeking” in response to obsessions concerning the odor [3,15]. Nevertheless, in OCD, other symptoms are usually present and tend to change over time, insight is better, and ideas of reference are rare [3,15].

consider ORS as an unusual symptom or a subtype of BDD [2,17] since the core belief is not of a defect in physical appearance [16]. 3.5. Hypochondriasis Somatic preoccupations, poor insight, repetitive checking of body functions and the search for medical treatments are common features. However, in hypochondriasis, the central fear/belief is of having a serious illness [9,15], while ideas of reference and social avoidance are rarely observed [3]. 3.6. Depression Poor self-esteem, negative thoughts and social isolation are common manifestations, but major depression has an episodic course, whereas ORS is chronic and stable. Depression is the most frequent comorbidity in ORS [4] and may be secondary, due to the distress and negative impact in all life domains. In this case, the patient had presented impairing ORS symptoms for over 20 years and met current diagnostic criteria for SAD, specific phobia, OCD, GAD, BDD and MDD. It is important to emphasize that, although presenting several psychiatric symptoms, the patient’s main complaint has always been the “halitosis.” In the appointments, she repeatedly argued that almost all her problems (e.g., low self-esteem, insecurity, irritability, social isolation, ideas of reference, compulsive teeth brushing, anxious and depressive symptoms) were due to her bad breath. Therefore, we believe that ORS is indeed a central aspect in her psychopathological manifestations. In fact, prior to the onset of “halitosis,” the patient presented specific phobias (water, needles) that led to the avoidance of dentists and are, therefore, also related to her main dysfunctional belief. The sexual abuse she suffered when she was 11 years old probably had a negative impact on her self-esteem, which is a key aspect not only of ORS but also of BDD and SAD. Despite some overlapping phenomenological characteristics with other disorders, the literature suggests that ORS is a distinct clinical entity. It has been described in different parts of the world, presents a well-defined symptom profile and generates significant distress and impairment [3]. However, given the secrecy of symptoms, it can be missed in clinical settings and inappropriately treated [3]. Although no consensus exists on the best treatment strategies for ORS, a recent review of 84 case reports [5] concluded that it responds to antidepressant and psychotherapy more frequently than to neuroleptics. Although the high number of comorbid disorders can weaken the concept of ORS [18], its allocation under a new diagnostic category in DSM5 [10] may have clinical utility and should stimulate more specific research in terms of nosology and treatment response. 4. Conclusion Since the literature on ORS is based exclusively on case reports/ series, we believe that the nosological debate regarding this impairing condition remains open. More epidemiological and clinical studies are needed to determine ORS prevalence and diagnostic status, which is the first step to achieving evidence-based treatment approaches. Although difficult to conduct, given ORS’ secrecy and high comorbidity, controlled trials are warranted to enable better pharmacological and psychotherapeutic management of these patients. In summary, further studies regarding symptoms, biomarkers and outcomes are needed to fully disentangle ORS from existing depressive, anxiety and obsessive–compulsive spectrum disorders.

3.4. Body dysmorphic disorder Disclosure Similarities include a body-related concern, checking behaviors, attempts to disguise or “solve” the imaginary problem, poor insight and the search for alternative medical treatments [16]. Some authors

L.F. is a member of the WHO ICD Revision Working Group on the Classification of Obsessive–Compulsive Related Disorders, reporting

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to the International Advisory Group for the Revision of ICD-10 Mental and Behavioural Disorders. The views expressed in this article are those of the authors and do not represent the official policies or positions of the International Advisory Group, the Working Group on Obsessive–Compulsive Related Disorders or the WHO. References [1] Pryse-Phillips W. An olfactory reference syndrome. Acta Psychiatr Scand 1971;47:484–509. [2] Prazeres AM, Fontenelle LF, Mendlowicz MV, Mathis MA, Ferrão YA, Brito NFC, et al. J Clin Psychiatry 2010;71(1):87–9. [3] Feusner JD, Phillips KA, Stein DJ. Olfactory reference syndrome: issues for DSM-V. Depress Anxiety 2010;27:592–9. [4] Phillips KA, Menard W. Olfactory reference syndrome: demographic and clinical features of imagined body odor. Gen Hosp Psychiatry 2011;33:398–406. [5] Begum M, McKenna PJ. Olfactory reference syndrome: a systematic review of the world literature. Psychol Med 2011;41:453–61. [6] Cruzado L, Cáceres-Taco E, Calizaya JR. Apropos of an olfactory reference syndrome case. Actas Esp Psiquiatr 2012;40(4):234–8. [7] Konuk N, Atik L, Atasoy N, Ugur MB. Frontotemporal hypoperfusion detected by 99mTc HMPAO SPECT in a patient with olfactory reference syndrome. Gen Hosp Psychiatry 2006;28:174–7.

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[8] Bizamcer AN, Dubin WR, Hayburn B. Olfactory reference syndrome. Psychosomatics 2008;49(1):77–81. [9] American Psychiatric Association (APA). Diagnostic and statistical manual of mental disorders: DSM-IV-TR. Washington (DC): American Psychiatric Association; 2000. [10] American Psychiatric Association (APA). Diagnostic and statistical manual of mental disorders: DSM5. Washington (DC): American Psychiatric Association; 2013. [11] First MB, Spitzer RL, Gibbon M, Williams JB. Structured clinical interview for DSM-IV axis I disorders: clinical version (SCID-CV). Washington (DC): American Psychiatric Press; 1997. [12] Eisen JL, Phillips KA, Baer L, Beer DA, Atala KD, Rasmussen SA, et al. The Brown Assessment of Beliefs Scale: reliability and validity. Am J Psychiatry 1998;155(1):102–8. [13] Singh GP. Is olfactory reference syndrome an OCD? Indian J Psychiatry 2006;48 (3):201–2. [14] Phillips KA, Hart AS, Simpson HB, Stein DJ. Delusional versus nondelusional body dysmorphic disorder. CNS Spectr 2014;19(1):10–20. http://dx.doi.org/10.1017/ S1092852913000266. [15] Lochner C, Stein DJ. Olfactory reference syndrome: diagnostic criteria and differential diagnosis. J Postgrad Med 2003;49:328–31. [16] Phillips KA, Wilhelm S, Koran LM, Didie ER, Fallon BA, Feusner J, et al. Body dysmorphic disorder: some key issues for DSM-V. Depress Anxiety 2010;27:573–91. [17] Gebara CM, Barros-Neto TP. Considerações sobre a clínica e o tratamento de uma manifestação incomum do transtorno dismórfico corporal: a síndrome de referência olfatória. J Bras Psiquiatr 2011;60(4):347–9. [18] Maj M. Psychiatric comorbidity: an artifact of the current diagnostic systems? Br J Psychiatry 2005;186:182–4.

Olfactory reference syndrome: a still open nosological and treatment debate.

The objective was to report a case of olfactory reference syndrome (ORS) with several co-occurring disorders and to discuss ORS differential diagnoses...
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