Shared Psychotic Disorder with Sexual Delusions

Shared psychotic disorder (SPD) is perceived as a relatively rare and poorly understood psychiatric phenomenon. Patients sharing sexual delusions may refer to sex therapists looking for treatment of an alleged sexual pathology. This might cause significant diagnostic and therapeutic challenges. The aim of this article was to discuss diagnostic and management difficulties of SPD with special interest in patients sharing sexual delusions. PubMed selective search was provided for publications with keywords including SPD, induced delusional disorder, folie à deux, and induced psychosis. One case is presented and discussed according to recent diagnostic criteria and the medical and legal issues of the therapy.


Diagnostic Considerations
Contemporary diagnostic criteria for SPD according to DSM-IV-TR and induced delusional disorder (IDD) according to ICD-10 are shown in Table 1. Guidelines for diagnosing SPD and IDD do notdiffersignificantly.Itisonewhereonlyonepersonsuffersfrom a genuine, established psychotic disorder (inducer), most commonly schizophrenia or delusional disorder. The other person (recipient) is often highly suggestible,younger,less intelligent, more passive, and with lower self-esteem. Both usually live in some kind of isolation from other people combined with a longstanding and very close relationship to each other (Silveira & Seeman, 1995;Wehmeier et al., 2003). Another factor predis-posingtoreceivedelusionalinductionisDependentPersonality Disorder, like in the case of folie à deux and incubus syndrome presented by Petrikis et al. (2003). It is important to distinguish whether the disclosed delusional symptoms are truly psychotic and not representing special cultural beliefs (Gaines, 1995). In M. Lew-Starowicz (&) III Department of Psychiatry, Institute of Psychiatry and Neurology, ul. Sobieskiego 9, 02-957 Warsaw, Poland e-mail: michallew-starowicz@wp.pl affected individuals, psychotic symptoms are clearly believed regardless of evidence to the contrary and usually impair their social, occupational or interpersonal functioning. Distinguishing between the inducer and the recipient can be difficult due to the circular character of IDD where role reversal (when the recipientbecomestheinducer)sometimesoccurs (Mentjox,van Houten, & Kooiman, 1993). However, in such cases, endogenous etiology in both individuals (more likely when consanguineous) mustberuledoutifdiagnosinginducedpsychosis.Thiswasargued by Arnone, Patel, and Tan (2006), who pointed out that the diagnosticcriteriaoffolie à deux areinsufficientanddo notaccountfor the high rate of psychiatric comorbidity in the recipients. A recent reviewofcourtcasesinvolvingfolieà deuxpublishedbyNewman and Harbit (2010) showed that the diagnosis of SPD often causes confusion among mental health experts and legal professionals and needs further investigation.

Case Report
Mr. D, a 64-year old male with higher technological education, a retired academic professor, married for 39 years, was admitted to theDepartmentofPsychiatrywithadiagnosisofSPD.Hewaspreviously hospitalized for 4 months in another hospital with the same diagnosis, treated with perazine and signed himself out after 12 days. He continued ambulant treatment, changed later to risperidone without any substantial improvement. On his wife's (Mrs. E, 61 years-old) initiative, before any psychiatric treatment,theyhadturnedforhelptoapsychologist,anexorcist,and finallytoasexologist(whowasalsoapsychiatrist)whoreferred them at that time and now once again to another hospital.
Mr. D agreed to hospitalization as he declared he wanted to undergo civil commitment to prevent him from making unfavorable financial decisions connected with being''exploited by a mistress,'' The patient and his wife claimed that he would go out at nights to his female lover who would''stimulate him with Viagra and narcotics,'' transport him in her car, have sex with him, and finally the patient would wake up at home and not remember anything that had happened. He got all the information of what was going on from his wife that ''examined him with drug tests'' and offered that he could sleep tied to the bed with handcuffs to prevent going out at night. The woman that was accused to be the mistress wasoneoftheteachersemployedwithMr.Dinthesameuniversity. Mrs. E claimed that she had installed a camera at home and convinced her husband that he was persecuted and tracked by his mistress and he had also had many''contacts''with other women in the meantime.
Mr. D declared that he had''a double life''for 6 years and had beensexuallyabusedand coerced by hismistress''tosignfinancial documents in her favor''although he could not remember any of these events. He claimed that he was convinced that situation was truebecausehiswifetoldhimso,andhefullytrustsherastheyhave a very strong romanticrelationship. Talking to hiswife,hecameto theconclusionthathewasmentallyillandneededprofessionalcare andnowevenacivilcommitment.Mrs.Eclaimedthattheprevious hospitalizationwasunsuccessfulandhadtobeterminatedbecause they discovered that hospital staff were cooperating with her husband's mistress and released him at night to meet her secretly. She maintained that the woman had even followed them during their holiday trip to Greece. The patient could not remember this but he agreedthatitmusthavehadcertainlyhappened.Hewascompletely uncriticalin his narration. Theybothtried to intervene bycontacting Table 1 Diagnostic criteria for Folie à deux-Induced Delusional Disorder in ICD-10 (World Health Organization, 1994) and Shared Psychotic Disorder in DSM-IV-TR (American Psychiatric Association, 2000) ICD-10 DSM-IV-TR Induced delusional disorder Shared psychotic disorder A. A subject must develop a delusion or delusional system originally held by someone else with a disorder classified in F2-F23 A. Delusion develops in an individual in the context of a close relationship with another person(s), who has an already-established delusion B. The two people must have an unusually close relationship with one another, and be relatively isolated from other people B. The delusion is similar in content to that of the person who already has the established delusion C. The subject must not have held the belief in question prior to contact with the other person, and must not have suffered from any other disorder classified in F20-F23 in the past C. The disturbance is not better accounted for by another psychotic disorder (e.g., schizophrenia) or a mood disorder with psychotic features and is not due to the direct physiological effects of a substance (e.g., a drug of abuse, a medication) or a general medical condition ''the mistress,''who finally wrote to the chancellor of the university and stated that she did not know and had no relationship with Mr. D andcomplainedthatshewasintrudedonbythecouple.Shortlyafterwards, the patient retired. Since then, Mr. D and Mrs. E were spendingalmostalltheirtimetogetherandbothperceivedtheirrelationship very well apart from the presented problem. Mrs. E was interviewed but she refused answering any questions about herself, pretending they were''strictly confidential.''She was not at all motivated to discuss her feelings and expected only an intervention that would prevent her husband from seeing''the mistress''andmakingfinancialdecisionsinherfavor(theywerenot able to present any document as evidence of it). Mr. D was routinely diagnosed during his stay in the ward. No previous psychiatric morbidity could be found; both their family historywasalsonegative(asstated).Hehadcomorbidarterialhypertension, coronary artery disease, and diabetes, all properly controlled pharmacologically. Due to an untreated erectile dysfunction (ED), he had not had sexual intercourse with his wife or other women for a few years. He was upset about this and did not know that effective treatments of ED were available. He consented to eventualmedicaltreatmentthatcouldimprovehissexualfunction. Psychological evaluation made during previous hospitalization (Minnesota Multiphasic Personality Inventory,Benton Test, Wisconsin Card Sorting Test) showed no relevant disorder apart froma decrease in abstract reasoning. A recent brain Computed Tomographyscanshowedonlyslightventricleextensionandsclerotic plaques in internal carotid arteries. Other standard examinations were not significant.
In the course of hospitalization, the patient was calm, slightly depressed,oncebecomingambivalentabouthisdelusionalthoughts but after each of his wife's daily visits reassured about their correctness. The dose of risperidone was lowered. After only a few days of being in theward, Mrs. Eclaimedthat''themistress''wasseen in the hospitalbythepeopleshehiredtoobserveherhusbandandshetook him out of the ward. Mr. D initially denied the insinuation but later admitted it musthave happened if his wifesaid so. Mrs. E refused to bringtherecordingsdocumentingherhusbandescapingfromhome with''the mistress''as''the tapes were left in another city and hard to obtain.''Duringeveryconversation,shewasextremelywatchfuland suspicious. After initial objections, the couple finally permitted me to contact their son who lived in another country. In a telephoneconversation,hestatedthatformanyyearshehadhadvery poor contact with his mother and recently the relations with his father had also become much worse. He perceived his mother as definitely ill and remembered that in the past she used to take some kind of psychotropic medication. He was also not motivatedforanyinvolvementinhisparents'therapyanddidnotwant tomaintainanycloserrelationswiththem.Mr.DandMrs.Eliveda relatively solitary life and had no close relations with other people.
Afterseveralexaminations,thenatureofdelusionalbeliefswas discussed with the patient, and he did not resist the suggestion that his wife should be simultaneously treated. However, the next day, Mr. D and Mrs. E declared that Mr. D had to leave the hospital immediately due to his wife's''unexpected business trip''that was ''strictly confidential'' and ''would last several months.'' Mrs. E refused any offer of treatment and convinced her husband to sign himself out of the psychiatric ward. No premises for involuntary hospitalizationcouldbefound,sotheirrequesthadtoberespected.

Discussion
Why isthereso limitedresearchon SPDandthe prevalenceofthis phenomenon is so hard to obtain? One possible explanation is that SPD is an extremely rare condition. The other, more convincing reason is that people sharing psychotic disorder are not likely to seekpsychiatrictreatmentduetolackofinsightanddefendingtheir intimate thoughts unless they break the law or their behavior is exposedtothepublic.However,couplesaffectedbySPDwithsexual delusions may seek for sexual therapy of an alleged sexual abnormality.
This is the first report in the literature on patients with SPD attending a sexologist's office sharing a delusional belief of one's infidelity or deviant behavior. The prognosis for treating people withSPDisusuallypoorbecauseoflackofmotivation,poorinsight, and defending against separation of affected individuals. If not treated involuntarily, patients often abandon therapy, asin this case.
Presentingnegativeoutcomesallowsustoconsidertherapeutic failures that could be avoided, possible negative consequences as well as legal issues that should be improved in the future. I would like to concentrate on several questions: Was the diagnosis of SPD appropriate and what would be the advice for sexologists seeing such patients? What could underlie the development of such a unique type of delusional system? What limited my approach and did I exhaust therapeutic possibilities in this case? Were there premises for involuntary treatment?
As noted above, most individuals who share psychotic symptoms (folie à deux), both inducers and recipients, do not seek professional care or treatment due to lack of insight. This may be different for individuals with SPD with sexual delusions. In my case, the patient persistently looked for treatment on the inducer's initiative, related to the delusional belief of sexual pathology of an induced partner. In other words, the belief of the induced partner's exploitation and unaccepted sexual behavior was one of the core symptoms of delusional disorder. This must be distinguished from Munchausen-by-proxy Syndrome (MbPS), where patients simulate physical or psychological symptoms in a related person. The importantdifferencebetweenMbPSandSPDistheintentionalproduction of symptoms (factitious disorder) in another person and motivation to assumethe sickroleby proxy in theformer, whereas in the latter the symptoms are of clearly psychotic origin.
Inthepresentedcase,thepsychoticbackgroundofreportedpartners'behaviorseemstobeevidentandthecouplemetthecriteriaof SPD or IDD according to DSM-IV-TR or ICD-10, respectively. Nohistoryofpsychiatricillnesscouldbefoundintherecipient,but some evidence on previous psychiatric treatment of Mrs. E was given by her son. Additionally, the origins of psychotic illness in theinducerwerehardtoobtainduetolackofobjectivedata.Unlike other case reports on SPD, this one contains many more details about the recipient while the information on the inducer was very limited. The explanation for that is that other case reports rely on involuntary treatment conditions or court investigations. Mrs. E was strongly concentrated on sharing her delusional beliefs and allowed to perform all the examinations of her husband, while she was getting very suspicious and resistant to answering questions about herself. She also did not allow Mr. D to disclose any information concerning her. Finally, Mrs. E stated that she worked for the secret service and that her private data were strictly confidential.Thiswaslikelytobeapartofherdelusionalsystembut,infact, it limited the possibilities of obtaining more data on the inducer's case.Ialsodidnotsucceedinestablishingtherapeuticrelationwith Mrs. E as she refused to discuss her emotions (apart from expressing anger against''the mistress''and worries about her husband's mental state) and jumped quickly to talk about possible interventions focused on Mr. D.
Inthepresentedcase,thediagnosisofSPDwassupportedbyan unusually close relationship between these individuals, one being dominant (Mrs. E) and other submissive (Mr. D), and their relative isolation from others, including family and non-relatives. Mr. D's symptomsstartedtoremitwhenseparatedfromMrs.E(evenwhen therisperidonedosewaslowered),whichfurtherconfirmstheexistence of induced psychosis. I therefore predict that a longer separation would benefit the induced individual in the presented casethiscouldliberatehimfrominduceddelusionalthoughtsandimprove hiscognitivefunction.Itisconsistentwithacommonobservationthat inducedpartner'sdelusionalsymptomsmaydiminishordisappear when the relationship with the inducer is interrupted. However, according to Shiwach and Sobin (1998), in most cases of affected monozygotic twins, there was only transient or no improvement aftertheseparation.Theyconcludedthatblood-relatedfamilymembers who improve on separation are worth following up over time. McNiel, Verwoerdt, and Peak (1972) found that positive outcome may be also less likely in elderly pairs with shared psychosis. This maybeattributedalsotoGralnick'ssubtypeCofSPD-foliecommuniqué (Table 2), where the induced partner develops psychosis aftera longperiod ofresistanceand symptoms maintain evenafter separationfromtheinducer.Thepresentedcasewasrathercloserto Gralnick'ssubtypeA,apartfromtheinducedpartnerbeingherenot the younger but the older person (from Gralnick's classification, only subtypes A and C meet recent diagnostic criteria for SPD as subtypesBandDattributeendogenouspsychosistobothpartners). Mr. D also revealed other common risk factors of being the recipientofhispartner'sdelusionalbeliefs,likesuggestibilityandpassiveness. This might be compared to mild cognitive impairment that was found in his case, quite common for older people with cardiovascular risk factors.
It can be supposed that some delusional statements of patients seeingasexologistcouldinitiallyseembelievable.Untilmoreinformation comes to light or delusions become more evident or irrational, these cases can be treated as disturbed sexual behavior. It indicates a critical need for establishing the probability of patients' complaints in similar cases. It is always important to rule out the possibility that events thought to be delusional indeed happened. In the presented case, clearly there was no evidence for any of the sexual practices suspected by the inducing partner. Mr. D suffered from ED that prevented sexual activity with his wife. This could be explained most probably by his comorbidities-cardiovasculardisease anddiabetes thatare commonriskfactors for male sexual dysfunction (Apostoloet al., 2009;El-Sakka,2007).
It is interesting whether sexual dysfunction could act here as a ''triggerpoint''forinducingthepartners'delusionalideation about patients' infidelity, exploitation or deviant behavior. This would correspond with psychodynamic and cognitive reasoning of delusional creation in predisposed individuals. So, a hypothesis about the origins of Mrs. E's specific psychotic symptoms would be as follows:Shehadprobablybeensufferingfromdelusionaldisorder for many years. Mr. D's sexual dysfunction combined with Mrs. E's state of''psychotic readiness''could be a trigger to develop a new delusional system. It is very common that a woman perceives herpartner'sEDasasignthatsheisnotattractivetohimanymoreor even that he is having an affair with someone else. In the case of a womansufferingfrompsychoticdisorder,thiscouldbeeasilyinterpretedandincorporatedintoadelusionalsystem.Thesewomenwill be concentrated on stopping an alleged affair rather than treating theirpartner'sED.Inthecaseofdelusionaljealousy,Mrs.E'sbelief thatherhusbandwasexploitedbythemistressagainsthiswillmight have protected their loving relationship-''he is not unfaithful, but mentally ill, insane.''It is only an assumption, of course. But, in fact, Mrs.Ewasconvincedthattheycouldhaveasuccessfulmarriageand sexual life if only her husband would not meet the mistress (even if confronted with his medical situation as a background of sexual dysfunction). She opposed the idea of treating Mr. D's ED as it was Table 2 Classification of Shared Psychotic Disorder by Gralnick (1942)

Subtype of SPD Description
A (folie imposée) The dominant person with delusions imposes his or her delusions on a younger and more submissive person. Both persons are intimately associated, and the delusions of the recipient disappear after separation B (folie simultanée) The simultaneous appearance of an identical psychosis occurs in two intimately associated and morbidly predisposed individuals C (folie communiguée) The recipient develops psychosis after a long period of resistance and maintains the symptoms even after separation D (folie induite) New delusions are adopted by an individual with psychosis who is under the influence of another individual with psychosis perceived only as the consequence of sexual exploitation by the mistress. Mrs. E did not blame her husband for the situation, but simultaneouslysheaccuratelycontrolledhisstayinthehospitaland later even accused the staff of admitting the mistress to meet the patient during the night, which remains consistent with her delusional jealousy. Mrs.Eresistedanykindofexaminationorinterventionfocused on her. If she had consented, her therapy could combine antipsychotics with psychotherapeutic approach focused on emotions of sexual dissatisfaction, jealousy, lack of trust and relationship with her husband, who could be simultaneously treated for sexual dysfunction. In the case of Mrs. E's recovery, separation from Mr. D and his antipsychotic treatment would probably not be necessary. However, if Mrs. E refused therapy, separation would be justified and could benefit her husband's therapy. It is recommended that separationshouldlastforatleast6 months.Itisusuallyveryhardto obtain as both partners are tightly linked to each other and tend to defend the isolation that makes them relatively happy. This was also found in the presented case. Separation would also demand applyingtheprocedureofinvoluntaryhospitalizationofoneorboth partners.Then,onlytheinducer(Mrs.E)wouldneedantipsychotic therapy.Fortherecipient(Mr.D),separationandpsychoeducation would probably be sufficient for recovery. Similar to many other countries, in Poland involuntary hospitalization can be applied onlywhenapersonthreatensdirectlytheirownoranotherperson's lifeorhealthorisincapableofmeetingbasicvitalneeds.Inthepresented case, there was no evident life nor health threat. Legal regulation does not determine whether psychotic induction can be seen as harming someone's health when the victim's insight is lacking.Involuntaryhospitalizationcouldbeappliedwhenatleast one of the patients (inducer or induced partner) would break the law. Joshi et al. (2006) presented a case of folie à trois where three affected sisters were involuntarily hospitalized and discharged to outpatient care under the provision (ordered by the court) of living inseparatecountiesandprohibitedfromvisitingeachotherwithout supervision. However, in that case, the court order was supported by the fact that the affected individuals had committed a penalized crime-burglary, assault and battery with intent to kill, and they were adjudicated not guilty by reason of insanity. Another case of involuntary treatment of a couple sharing psychotic disorder that waschargedwiththeabductionofawomanwasdescribedbyNewman and Harbit (2010). In other, less threatening cases (like the presentedone),suchasolution maybeimpossiblebecauseoflegal regulations. Moreover, even if separation was applied in the presented case, the prognosis would still be uncertain. If Mrs. E would prove to be resistant to therapy (which often happens in patients with delusional disorder), prolonging the separation could also be harmful, as both partners were living in a relatively happy relationship, even if overwhelmed by a shared delusional system. Further investigation is needed to establish a long-term efficacy of different therapeutic settings in patients sharing delusional beliefs.

Conclusion
SPD is one of the most confusing and poorly validated psychiatric disorders. Couples sharing delusions that concern sexual behavior are referred to sexual health professionals. The literature on such cases is lacking. This might be due to weak understanding of this relatively rare phenomenon, diagnostic difficulties, and challeng-ingtherapyofSPDwithfrequentlynegativeoutcome.SPDinnonconsanguineous partners sharing sexual delusions referred to a sexualmedicine professionalwas presented intheliteratureforthe first time. Therapeutic reversal in the presented case was strongly connectedwithlackofinsight,inducer'sresistance,andlegallimitations preventing involuntary treatment and separation of the affected individuals. Further research is needed to evaluate the epidemiology and nosological status of SPD. The possibility of implementingmoreefficient therapyinthefuturewouldrelyon changes in legal regulations and validation of therapeutic settings.