Abstract
Objective:
Jikoshu-kyofu (JKF), a condition characterised by a fear of offending others through emitting foul body odour, was first described in Japan in the 1960s. Although initially thought to be a culture-bound syndrome, it has been described in other countries. It is well established that there are two variants, a non- delusional and a delusional variant.
Method:
We describe two cases of JKF who sought treatment from a hospital in Singapore, compare and contrast their characteristics, and in view of overlaps with other conditions, discuss differential diagnoses.
Results:
Both cases involved women with fairly similar symptomatology except that the first case was younger, had overvalued ideas (but not delusions), an earlier age of onset and good treatment response, while the other had delusions, late age of onset, became too distressed to continue working and was initially resistant to treatment.
Conclusion:
It appears that the delusional variant may have a longer symptomatic duration before presentation, poorer insight, more resistance to treatment and multiple physician consultations, as well as greater impairment of socio-occupational functioning in contrast to the non-delusional variant.
The erroneous belief that one is emitting a foul smell is not a new phenomenon. It was first described in the medical literature over a century ago.1,2 In Japan, it is referred to as Jikoshu-kyofu (JKF). 3 This is a subtype of Taijin Kyofusho (TKF), a term first used in the 1920s by Shoma Morita to describe persons who developed the fear of offending others by their physical characteristics, such as blushing, gaze or the emitting of foul body odour during the course of social interactions. 4 Although JFK has been frequently referred to as a culture-bound syndrome, cases have been described in the United States, Korea, Australia and Europe, thus challenging the notion that it is purely a culture-bound syndrome. 5
It is well established that there are two variants of TKF, general (non-delusional) and offensive (delusional). 6 General and offensive variants have also been used to subtype patients with JKF. 7 We describe two patients representing both variants of JKF, compare and contrast their characteristics and discuss their differential diagnoses.
Case reports
Ms A was a 27-year-old woman who complained of emitting a bad smell from her axillae and back for over one year. The symptoms apparently started when a passer-by coughed behind her. Ms A recalled employing a housekeeper with such a foul body odour that she felt like coughing or covering her nose whenever she was near. Afterwards, Ms A was certain that she was giving off a foul odour whenever others behaved in the same way around her.
Although her work performance was unaffected, her social functioning suffered as she stayed away from gatherings in order to avoid offending people, even though she desired interactions with them. Ms A was largely unconvinced by reassurances. She nonetheless acknowledged not being certain of the veracity of the smell itself, and arbitrarily estimated the strength of her belief as 50%.
Ms A presented as neatly dressed and with no detectable odour. She was neither anxious nor depressed. She described what appeared to be overvalued ideas of smelling foul. After treatment with Cognitive Behavioural Therapy, she made a remarkable recovery, her social anxieties diminished remarkably and she no longer avoided interacting with people.
Ms B was a 60-year-old woman who presented in her early 50s with the singular belief that she was emanating a “bad smell”. This was accompanied by intense embarrassment, avoidant behaviour and social isolation. She had a history of multiple previous specialist consultations and extensive unremarkable organic investigations. While acknowledging that she could not smell the odour herself, she had prominent delusions of reference, in that she believed the odour to be ostensibly pervasive, and speculated that it must be redolent of “fish” or “rotten eggs”, based on the reactions of others, such as turning away, rubbing their noses or coughing. She was also absolutely certain that when people gathered and whispered among themselves, they must be griping about her smell despite her not being able to hear their conversation. If anyone used perfume, she believed it was to mask her smell. Ms B was so convinced about her offensive odour that she observed strict dietary restrictions and avoided social interaction as much as possible. Eventually, she quit her 30-year job as a clerk. She never had many friends. Nonetheless, there was no evidence of separation anxiety or any other types of anxiety.
She described herself as an introverted and a proud person, who was particularly anxious in social situations. Most of her hobbies were solitary in nature, including reading and watching television.
Ms B presented as tidy in appearance. She was mildly anxious but not depressed. Her manner was prim, and her speech laconic. Her beliefs were firmly held and she was resistant to reasoning. The fact that she could not smell the odours further suggests that she was not experiencing olfactory hallucinations. Ms B reluctantly agreed to a course of low-dose antipsychotic medications. After taking a few doses she stopped her medications and eventually defaulted further treatment.
Discussion
Non delusional versus delusional: our two patients illustrate important similarities and differences between the non- delusional and delusional variants of JKF. The two cases were similar in that both women believed that seemingly innocuous behaviours were reactions to an unpleasant odour they emitted, both exhibited social avoidance and both did not experience olfactory hallucinations. Their premorbid personalities were similar in that they desired cleanliness and were self-conscious of smells.
Ms A showed several characteristics that are typical of patients with the non-delusional variant of JKF. She had overvalued ideas, had an earlier age of onset and benefitted from Cognitive Behavioural Therapy. In contrast, Ms B’s presentation was consistent with the delusional variant in that she clearly had delusions, reported a later age of onset and was difficult to engage in treatment. In addition, Ms B described schizoid traits, was unmarried and preferred more solitary activities. Her symptoms also worsened over time, resulting in her leaving a hitherto stable job.
Differential diagnoses: it has been generally agreed that the general (non-delusional) variant of JKF resembles social anxiety disorder (SAD), which is characterised by the individual being afraid of embarrassing him or herself in social or performance situations. 8 Although Ms A’s condition is obviously non-delusional, what distinguishes it from SAD is that she fears causing embarrassment to others, and is less concerned regarding embarrassing herself.
In the West a similar condition is termed Olfactory Reference Syndrome (ORS). While Bizamcer and colleagues 9 have regarded ORS as a variant of delusional disorder (somatic type), others have argued that since social avoidance is not usually associated with delusional disorder, it would not be appropriate to classify ORS under this category. 10 In support of this argument, several authors have observed that that not all cases of ORS are characterised by delusions.11,12
Suzuki et al. 10 have held that the clinical characteristics of JKF are almost identical to those of ORS and that the two conditions even share a common entity, but they also highlight an important difference. The average age of onset for patients with ORS was older (in the mid-20s), in contrast to the teenage to early adolescent age of onset of JKF patients. 10 The data in this area are not conclusive, since it has also been reported that the ages of JKF and ORS sufferers were fairly similar. 12
A discussion of certain differential diagnoses is in order. Correct diagnoses precede appropriate management, resolution of symptoms and optimise prognosis. In cases that present with somatic preoccupations, body dysmorphic disorder (BDD) should always be considered. However, BDD patients are concerned more with perceived defects in physical appearance than with unpleasant smells. Both our patients were preoccupied with the idea that they were producing smells, rather than with any defects in their appearance.
When patients present with meticulous cleaning and washing behaviour, obsessive-compulsive disorder (OCD) may also be considered. While such behaviour in OCD is usually secondary to a fear of contamination, our patients did not describe contamination fears. Some have argued that in contrast to ORS, OCD beliefs are delusional in fewer than 5% of cases, and delusions of reference seem less common in OCD than in ORS. 13 Moreover, in ORS there is a high prevalence of social avoidance, in contrast to OCD.
Cultural considerations: in seeking to understand the cultural context of this condition, it is pertinent to note that both our patients have been raised in a Southeast Asian society where shame and collectivism feature prominently. Persons from these societies typically endorse interdependence with a view of themselves as being part of a wider social group. 14 Therefore, it is important to these individuals to behave in a manner that is considered appropriate and acceptable to the rest of the community. 15 By contrast, persons from individualistic societies in the West tend to value independence and autonomy from the group. 14 While shame-prone and self-effacing behaviour are highly valued and are actively promoted in Asian societies, Western cultures often discourage such behaviours and the expression of one’s vulnerability, while encouraging demonstration of one’s autonomy and capability. 16
Conclusions
Our two cases illustrate similarities and differences between the non-delusional and the delusional variants of JKF. Careful assessment is needed to confirm the diagnosis and to exclude conditions with fairly similar features, for example, SAD, ORS, BDD and OCD. While Ms A improved with treatment, Ms B was more resistant to the idea of taking antipsychotic medications. Having resigned from her job and being more socially isolated, her prognosis is considered the less favourable of the two women.
As in the case with delusional patients, the challenge is to build a strong therapeutic alliance and to encourage medication intake that these patients would be likely to benefit from.
Footnotes
Acknowledgements
The authors would like to thank Dr Sharon Sung for providing helpful comments on the manuscript.
Disclosure
The authors report no conflict of interest. The authors alone are responsible for the content and writing of the paper.