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MEDICINE Diagnosis and Treatment of the Factitious Disorder on Another, Previously Called Munchausen Syndrome by Proxy M. Lopez-Rico 1 & J. J. Lopez-Ibor Jr 1 & D. Crespo-Hervas 1 & A. Muñoz-Villa 1 & J. L. Jimenez-Hernandez 1 Accepted: 21 February 2019 /Published online: 26 March 2019 # Springer Nature Switzerland AG 2019 Abstract In the factitious disorder (FD) on another, formerly called Munchausen syndrome by proxy (MSbP), one of the parents, the mother in 95% of the cases, fabricates or invents clinical symptoms in her child with the intention of convincing doctors and pediatricians that the child is sick. In the chapter on addictive disorders of the 5th Edition of Diagnostic and Statistical Manual of Mental Disorders, the gambling disorder was included, opening up the possibility of incorporating other problematic addictive risk behaviors in the same chapter. In this work, we intend to provide current data for improving the detection and treatment of MSbP as addictive disorder. Four clinical cases of MSbP studied by the authors are described. Retrospective analysis and semi- structured interviews were carried out to assess the psychopathological profile of MSbP. The four perpetrating mothers in the cases studied had a common and characteristic psychopathological profile. These mothers falsified diseases in their victimized children, obtaining rewards from the hospitalization of their children. The continuous alteration of the mother-to-child attachment with a pathological bond, which is at the basis of therapeutic intervention together with the mothersemotional confrontation with reality, is verified. It is proposed that the psychopathological profile of the perpetrator of FD on another corresponds to an addictive disorder, in which the hospital context is compulsively sought, providing a high degree of gratification and reward. Keywords Munchausen syndrome by proxy . Addictive disorder . Mother-to-child attachment . Child abuse Introduction Virtually, all of society is aware of the existence of abused children and the emotional consequences that this abuse pro- duces [30]. Exposure to maltreatment during vulnerable pe- riods of life may be a critical risk factor for the emergence of suicidal behavior in adolescence and early adulthood [60]. In the diagnostic framework that covers child abuse, Roy Meadow added, in 1977, the Munchausen syndrome by proxy (MSbP). In this disease, one of the parents, the mother in 95% of the cases, fabricates or invents the presence of clinical symptoms in their child, turning them into a patient in the eyes of doctors and pediatricians [8]. The recent publication of two extensive reviews in Lancet (2014) dealing with the etiology, diagnosis, treatment, and prevention of MSbP have reignited interest in this complex syndrome [9, 10]. Knowledge regarding MSbP is consolidat- ed on the detailed study of published clinical cases and the information evidence-based medicine provides [40, 53, 74]. In the latest Diagnostic and Statistical Manual of Mental Disorders (5th Ed., [DSM-5], [3]), factitious disorder (FD) can be imposed on oneself or on another person. The latter situa- tion is identified as MSbP. The diagnosis of FD is not common in current clinical prac- tice [21], as the difficulty in detection lies in its particular position at the boundaries between medicine, psychiatry, and law [8, 34]. After reviewing the chapter in the DSM-5 on FD, Hamilton et al. [27] recommended an in-depth review regarding the reasons that underlie the infrequent diagnosis of MSbP, as well as insisting on the need to draw clear lines that facilitate its early detection. The DSM-5 proposes two diagnoses in MSbP: one that po- sitions the child as a victim of child abuse and the other that positions the perpetrator as a psychiatric patient of MSbP. The specific criteria for diagnosing MSbP should be applied to the J. J. Lopez-Ibor Jr. passed away in 2015. This article is part of the Topical Collection on Medicine * M. Lopez-Rico [email protected] 1 Depto. de Fisiología y Farmacología, Facultad de Medicina, Universidad de Salamanca, 37005 Salamanca, Spain SN Comprehensive Clinical Medicine (2019) 1:419433 https://doi.org/10.1007/s42399-019-00057-6

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Page 1: Diagnosis and Treatment of the Factitious Disorder on ... · perpetrator of Munchausen syndrome by proxy (MSbP) 1. Genetic and biological vulnerability 2. Alterations in the mother-to-child

MEDICINE

Diagnosis and Treatment of the Factitious Disorder on Another,Previously Called Munchausen Syndrome by Proxy

M. Lopez-Rico1& J. J. Lopez-Ibor Jr1 & D. Crespo-Hervas1 & A. Muñoz-Villa1 & J. L. Jimenez-Hernandez1

Accepted: 21 February 2019 /Published online: 26 March 2019# Springer Nature Switzerland AG 2019

AbstractIn the factitious disorder (FD) on another, formerly called Munchausen syndrome by proxy (MSbP), one of the parents, themother in 95% of the cases, fabricates or invents clinical symptoms in her child with the intention of convincing doctors andpediatricians that the child is sick. In the chapter on addictive disorders of the 5th Edition of Diagnostic and Statistical Manual ofMental Disorders, the gambling disorder was included, opening up the possibility of incorporating other problematic addictiverisk behaviors in the same chapter. In this work, we intend to provide current data for improving the detection and treatment ofMSbP as addictive disorder. Four clinical cases of MSbP studied by the authors are described. Retrospective analysis and semi-structured interviews were carried out to assess the psychopathological profile of MSbP. The four perpetrating mothers in thecases studied had a common and characteristic psychopathological profile. These mothers falsified diseases in their victimizedchildren, obtaining rewards from the hospitalization of their children. The continuous alteration of the mother-to-child attachmentwith a pathological bond, which is at the basis of therapeutic intervention together with the mothers’ emotional confrontation withreality, is verified. It is proposed that the psychopathological profile of the perpetrator of FD on another corresponds to anaddictive disorder, in which the hospital context is compulsively sought, providing a high degree of gratification and reward.

Keywords Munchausen syndrome by proxy . Addictive disorder . Mother-to-child attachment . Child abuse

Introduction

Virtually, all of society is aware of the existence of abusedchildren and the emotional consequences that this abuse pro-duces [30]. Exposure to maltreatment during vulnerable pe-riods of life may be a critical risk factor for the emergence ofsuicidal behavior in adolescence and early adulthood [60]. Inthe diagnostic framework that covers child abuse, RoyMeadow added, in 1977, the Munchausen syndrome by proxy(MSbP). In this disease, one of the parents, the mother in 95%of the cases, fabricates or invents the presence of clinicalsymptoms in their child, turning them into a patient in the eyesof doctors and pediatricians [8].

The recent publication of two extensive reviews in Lancet(2014) dealing with the etiology, diagnosis, treatment, andprevention of MSbP have reignited interest in this complexsyndrome [9, 10]. Knowledge regarding MSbP is consolidat-ed on the detailed study of published clinical cases and theinformation evidence-based medicine provides [40, 53, 74].

In the latest Diagnostic and Statistical Manual of MentalDisorders (5th Ed., [DSM-5], [3]), factitious disorder (FD) canbe imposed on oneself or on another person. The latter situa-tion is identified as MSbP.

The diagnosis of FD is not common in current clinical prac-tice [21], as the difficulty in detection lies in its particular positionat the boundaries between medicine, psychiatry, and law [8, 34].

After reviewing the chapter in theDSM-5 on FD, Hamiltonet al. [27] recommended an in-depth review regarding thereasons that underlie the infrequent diagnosis of MSbP, aswell as insisting on the need to draw clear lines that facilitateits early detection.

The DSM-5 proposes two diagnoses in MSbP: one that po-sitions the child as a victim of child abuse and the other thatpositions the perpetrator as a psychiatric patient of MSbP. Thespecific criteria for diagnosing MSbP should be applied to the

J. J. Lopez-Ibor Jr. passed away in 2015.

This article is part of the Topical Collection on Medicine

* M. [email protected]

1 Depto. de Fisiología y Farmacología, Facultad de Medicina,Universidad de Salamanca, 37005 Salamanca, Spain

SN Comprehensive Clinical Medicine (2019) 1:419–433https://doi.org/10.1007/s42399-019-00057-6

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person performing the fabrication, where attention is focused onthe false behavior and on the absence of external rewards [3, 9].

According to the DSM-5, the characteristic persistence ofthe behavior and the intentional efforts to conceal the patho-logical behavior by deception are similar in factitious disor-ders (FD) and in substance-related addictive disorders. In theDSM-5 diagnostic criteria for FD, the repeated behavior offalsification of physical or psychological signs and symptomsis emphasized and deception is evident even in the absence ofobvious external rewards, circumventing a possible underly-ing internal motivation [3, 29].

Likewise, the addition of the gambling disorder in the newchapter of substance-related and addictive disorders in theDSM-5 helps to support the inclusion of other types of prob-lematic behavior not directly related to substance use, such asthe Internet and shopping, as addictive diseases. From ourpoint of view, we suggest that MSbP also be included in thischapter, since the perpetrator exhibits an addiction toward thehospital context and medical issues, with their child as a proxypatient ([39], p. 43).

We have established seven factors that will help us contex-tualize our proposal of MSbP as an addictive disease. Theseseven factors will allow us to check the psychiatric profilecharacteristic of MSbP perpetrators (Table 1).

The first factor to analyze is genetic vulnerability and/orthe possible biological substratum that may be at the base of afuture addictive behavior. Although the DSM-5 has not beenable to specify the inclusion of specific genetic markers for thediagnosis of addictive disorders, it has exposed several genesrelated to the structure of receptors that are supposed to berelated to the problem, such as nicotinic receptors and μ opi-oid receptors (CHRNA5 and OPRM1). Variations in both,such as the SNP (single nucleotide polymorphism) A118Gof OPRM1, have been detected in relation to a possible anom-alous response to endogenous opioid peptides that could con-fer greater vulnerability to addiction [28]. In the general pop-ulation, behavior, perception, reward, and mood are clearly

connected through the hypothalamic–pituitary–adrenal axisthat modulates their interactions and in turn is downregulatedby the opioid system. This system is altered in addictive dis-orders [17]. When studying the MSbP in the context of anaddictive disease, genetic associations are probably found thatare currently impractical for reaching a diagnosis but may helpto validate it in the future.

Neuroimaging techniques have allowed us to verify a disrup-tion in the prefrontal cortex (PFC) that occurs not only duringthe intake of substances but also during problematic behaviorsassociated with addictive risk [25]. In the functional magneticresonance imaging (fMRI) performed on amother perpetrator ofMSbP, a pronounced activation of the ventrolateral PFC and theanterior cingulate cortex (ACC) was evident in association withsignificantly longer response times during the process of con-frontation with her compulsive deception behavior, which tookplace as part of her treatment [64].

Among the multiple brain neurochemical changes that oc-cur in addiction, the concentrations of dopamine, serotonin,and norepinephrine are especially affected [19]. The noradren-ergic input from the locus coeruleus (LC) is responsible for thecontrol of midbrain dopaminergic neurons originating in theventral tegmental area (VTA) previously described as the neu-ral substrates underlying individual vulnerability to drug ad-diction [36]. To know the functional modulation of the VTA iskey to understand how craving and withdrawal symptoms areproduced in addiction [71]. The hyperactivity of the LC pro-motes most of the symptoms that occur in all addictive disor-ders when withdrawal syndrome is induced [12]. This factmust be taken into account so that the confrontation of themother perpetrator of an MSbP with reality can occur in thesafe environment required when dealing with a withdrawalsyndrome. The confrontation leaves the mother in a situationof withdrawal, which increases risk to the victim, where shemay try to continue the farce in other hospitals and is alsomore at risk of suicide [10].

A second factor assesses the altered bonding patterns ofmothers exhibiting MSbP. An unsafe mother-to-child attach-ment is most often found among parents who mistreat theirchildren, including those mothers who falsify or induce illnesson their own family. Adshead and Bluglass [1] study 67 per-petrators of MSbP using a semi-structured interview to assessthe type of mother-to-child attachment and show that 85% ofmothers have unsafe relationships with their child. Thesemothers exhibited nervousness and anxiety when thinkingabout the act of caring or their own care during childhood.

The third factor analyzes the emergence of situations ofviolence, neglect, abuse, or emotional deprivation experi-enced by perpetrators. Of the 67 mothers studied byAdshead and Bluglass [1], 31 (46%) had histories of child-hood trauma and 19 (28%) of adult trauma, predominantlyrape, or domestic violence. Thirtymothers (45%) had historiesof illness or of having had an accident during childhood, and

Table 1 Factors that can contribute to the psychiatric study of theperpetrator of Munchausen syndrome by proxy (MSbP)

1. Genetic and biological vulnerability

2. Alterations in the mother-to-child attachment

3. Early traumas experienced by the perpetrator (violence, abuse,deprivation)

4. Intergenerational transmission of behaviors of medical falsificationwithin the family network

5. Learned positive experiences regarding the disease

5.1 Receiving attention for being sick

5.2 Losing reinforcement of non-somatic expressions of discomfort

6. Motivational forces behind the perpetrator’s actions

7. Psychiatric comorbidity

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16 (24%) remembered how somemembers of their family hadsuffered from a serious illness while they were young girls oradolescents. Thirty-six mothers (54%) had experienced painowing to the loss of loved ones throughout their lives.

The proposed study of the intergenerational transmission ofthe behavior of feigning illness within the family context ofMSbP is in fourth place. Symptom-inventing behaviors aretaught and learnedwithin the family. It is a fact that the differentforms of factitious disorder (FD) are presented in a cyclicalmanner, going from being performed on oneself to being per-formed on another person, and vice versa. Very little is knownabout the long-term impact of being a victim of MSbP [59].Most probably many victims are not even identified, and othersare lost within the follow-up when they are no longer super-vised by child protection agencies [62]. Palmer and Yoshimura[50] described a 9-year-old boy whose mother falsified stoolsamples. Upon reviewing the anamnesis, it was found that inthe mother’s childhood and adolescence, there were severalantecedents of FD, where her pediatric history was full of un-explained fractures, short stature caused by emotional depriva-tion, cyclic vomiting, and numerous questionable surgical pro-cedures. This case represents a complete generational cycle:perpetrator of MSbP who was victimized by her mother duringher childhood and who suffered from FD in her youth [50].

As a fifth factor in the study of factitious disorder, it isproposed that early learning experiences related to any diseasethat lead to positive reinforcement and also the acceptance ofthe role of a sick person during childhood are the basis offuture factitious behaviors. The learned reward is based onthe attention and parental care obtained from being orappearing sick and from being exempt from domestic, social,or school tasks [34]. BBeing sick^ becomes a desired statebecause of the emotional gains that are achieved along withit. The perpetrator achieves these benefits as she becomes theBmother of a sick child^ [37]. Gradually, the child victim ofMSbP becomes more vulnerable to the pressure exerted by themother [65] and then starts a collaboration with her, whichpasses from the first phase of being Bnaïve^, to the secondphase of passive acceptance, to the third with active participa-tion, and then, in some cases, reaching the fourth phase ofauthentic active self-harm [38].

The sixth factor seeks to identify the unconscious motiva-tion of MSbP perpetrators [57]. The suggestions proposed inthe literature are varied, and the different motives that arepresented do not result in a single behavioral profile thatwould allow a psychiatric diagnosis to be established, identi-fying all perpetrators of MSbP [10]. The need to obtain acommon psychopathological profile of all mothers who fabri-cate or falsify diseases in their children is of great interest toimprove the diagnosis and treatment of the syndrome. Asregards, we have added a semi-structured interview, obtainedafter the study of 15 perpetrators of MSbP, to the recent pro-posal by Bass and Glaser [9] concerning the mechanisms that

explain the motives of these mothers. The interviews outlinethe relevant aspects of the mothers’ addictive behaviors withinthe hospital context and the topics related to medicine(Table 2), constituting a common behavioral profile that char-acterizes these women ([39], p. 93).

The seventh and last factor analyzed, in the attempt to placeMSbP and addiction in the same context, is comorbidity [11].An addictive disease often occurs in the context of dual pa-thology, or psychiatric comorbidity, together with other char-acteristic disorders such as behavioral disorders with loss ofimpulse control, depression, and personality disorders, espe-cially of the border-line subtype [67]. Many specific symp-toms of addictive disease are associated with comorbid disor-ders, suggesting the interaction of different mechanisms thatunite the gambling disorder, recently recognized as an addic-tion, with the appearance of comorbid psychopathology. Asregards, MSbP has been related to hospital addiction for sometime [7]. Comorbidity is often present in association withMSbP and is similar to that presented in the gambling disorder[51]. Bass and Jones [11] show in their study of 28 perpetra-tors that 18 (64%) have factitious disorder and 17 (61%) pres-ent pathological lying as a habitual form of communicationduring adolescence and adult life, enshrouding their child in aweb of deceit and abuse [11]. Bass and Glaser [9] report thatmore than 50% of perpetrators have factitious disorder im-posed on self, and more than 75% have comorbid personality

Table 2 Semi-structured interview for studying the behavioral profileof the perpetrator in the hospital context

1. Fascination with the hospital environment and its situational context orwith relevant medical issues

2. Concern and persistent ideas about the disease and the medicalenvironment, recalling rewarding situations previously experienced inhospitals

3. Recurring thoughts about planning new hospital stays which occupyalmost all of their time

4. Self-awareness of the progressive and growing need to seek hospitalcontexts, especially when faced with stressful circumstances

5. The perpetrator recognizes she searches for the hospital context to helpher escape everyday problems, which alleviates the dysphoric feelingsof despair, guilt, anxiety, and depression

6. Repeated failure to control or disrupt addictive behavior of abuse with arecurrent difficulty in overcoming the impulse to take the victim to thehospital

7. The emergence of depression, irritability, dysphoria, or anxiety whentrying to control the behavior of searching for hospitals

8. The increased feeling of emotional tension before going to the hospital

9. Experiencing gratification, stress relief, and well-being during hospitalstay

10. The perpetrator deceives the family and doctors to hide her level ofinvolvement and carries out punishable acts to achieve the desiredgoals such as the invention and fabrication of symptoms in the child

Adapted from Lopez-Rico and Jimenez-Hernandez [39]. Munchausensyndrome by proxy. Factitious Disorder in Pediatrics. SalamancaUniversity Press, Spain (pp. 93–94)

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disorders, particularly of the sociopathic, borderline, or histri-onic subtype [9].

In summary, the main study hypothesis tries to demonstratethat MSbP can be included in the category of addictive disor-ders as a condition for further study, showing the perpetratoran addiction to specific context. The proposal of a commonpsychopathological profile for perpetrators will be useful tofacilitate the identification and management of this complexform of child abuse.

Materials and Methods

Four clinical cases of MSbP are described, providing newinformation that could be useful in the research on this syn-drome and help to facilitate intervention that is both safe andappropriate. These four cases were among a series of 19 vic-tims belonging to the 15 families included within the authors’study ([39], p. 111). These families were referred to us by apediatric service to confirm the diagnosis of MSbP. In thepediatric service, there were 54 patients suspected of beingvictims of this syndrome, since all exhibited the associatedwarning signs defined by Roy Meadow in 1985 (Table 3).Of these 54 patients, 19 children were diagnosed as beingvictims of MSbP, corresponding to 15 perpetrators.

Clinical Case 1

This case involves a 6-year-old girl with 10 successive admis-sions to the pediatric intensive care unit (PICU), where shewas always arrived in a comatose state. She was admitted topediatrics for studying the possibility of an inborn error ofmetabolism that might have explained the clinical picture.The mother reported that the girl had presented paroxysmalepisodes from 7 months of age. The mother’s description waspeculiar, associating a set of symptoms to define the episodes

and using medical terms as if she were a health professional.Apparently, episodes were becoming more complex and themother stated that Bthe girl begins with a fever of 41°C, poly-uria and dehydration, and then continues with seizures and aprogressive decrease in the level of consciousness, sometimesbecoming comatose^. According to the mother’s story, thissymptomatology had always been refractory to all types oftreatment.

The mother’s description of the episodes did not coincidewith the 25 discharge reports obtained from 5 hospitals, wherethe child had been admitted on a total of 60 occasions(Table 4). The study of the chronology of the reports showedthat the symptoms described could be grouped by their orderof appearance: febrile syndromes, between 7 and 10months ofage; polyuria and dehydration, between 11 months and 2 anda half years of age; and convulsions, between 3 and 5 years ofage. The convulsions led to repeated episodes of gradual lossof consciousness, prior to entering into coma, symptoms thevictim had been experiencing for 2 years.

It was confirmed that during the period of experiencingconvulsions, the victim had been repeatedly assessed on 42occasions in various neurological services due to epilepticseizures identified as grand mal subtype. However, the gener-al examinations carried out in the emergency room were al-ways absolutely normal, with no signs or post-critical statusobserved. This contradicted the dramatic clinical picture de-scribed by the mother. The first time the child was admitted toneuropediatrics, and according to the victim’s medical history,she was diagnosed as having Atypical Epilepsy and begantreatment with Valproate®. One month later, the girl was re-admitted due to irritability, incoordination and ataxia, at whichtime Valproate® was replaced by Phenobarbital®. During fol-low-up, toxic sanguineous levels of both drugs were discov-ered in the child’s blood (69 mcg/ml of Phenobarbital®; nor-mal level between 10 and 30 mcg/ml and 190 mcg/ml ofValproate®; normal level between 50 and 100 mcg/ml) andit was confirmed that the mother was administering the drugs

Table 3 Warning signs of Munchausen syndrome by proxy (MSbP)

1. Presence of fictitious or factitious symptoms and signs

2. Unexplained chronic illness

3. Discrepancies (anamnesis/medical history)

4. Symptoms and signs are extraordinary

5. Symptoms and signs are only present when mother is present

6. Overly involved or overfriendly mother

7. Treatments are poorly tolerated

8. Mother is indifferent about the prognosis

9. The same illness is experienced by the mother or siblings

10. History of similar illness and unexplained deaths in the family

Adapted from [43], by Lopez-Ibor & Jimenez-Hernandez, in Jimenez-Hernandez et al. [33] Munchausen syndrome by proxy: a special type ofchild abuse. Actas Luso-Españolas de Neurología, Psiquiatría y cienciasafines, 15, 333–343

Table 4 Diagnosesfound in the 25 hospitaldischarge reportsassociated with case 1

1. Congenital hip dysplasia

2. Unexplained fever (4 reports)

3. Hypokalemia type II

4. Congenital polyuria

5. Hyperhidrosis (7 reports)

6. Functional inhibition of ADH due toexcessive water intake

7. Diabetes insipidus (3 reports)

8. Neurological process of the centralnervous system

9. Atypical epilepsy (2 reports)

10. Recurrent comas (3 reports)

11. Urea cycle disorders

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over the prescribed dose. Among the mother’s psychiatricantecedent, it was verified that she had a histrionic personalitydisorder. During one of the victim’s last hospital stays, withoutthe mother present, the child had four strange episodes lasting2 to 3 min in which she knelt, looked away, and did not re-spond to any type of external stimuli; she appeared to beBabsent.^ During these episodes, there were signs of oscilla-tions in muscle tone, successively going from hyper to gener-alized muscular hypotonia. But the child retained posturalreflexes and demonstrated a full awareness of what was goingon around her. In the Bpost-critical^ period, the victim af-firmed that she had seen the BVirgin Mary .̂

Due to the discovery of the toxic blood levels ofValproate® and Phenobarbital®, to the evidence supportingthe victim’s fictitious epileptic-like crises and to the chrono-logical analysis of the factitious symptoms and signs sufferedby the patient from the age of 7 months to the present, detailedand proven by multiple disciplines, it was possible to confrontthe mother with reality. The mother admitted the compulsivenature of her problematic use of hospitals, the handling ofthermometers, the invention of seizures, the administrationof Furosemide® to produce polyuria and dehydration, andthe administration of non-prescribed doses of Valproate®and Phenobarbital® to cause confusion and coma in the girlthat resembled epilepsy. The confrontational interview lasted6 h, during which time a child protection agency took chargeof the victim in order to prevent relapses in the form of newfactitious episodes. From this point on, the child abuse andfictitious visits to the doctor ceased.

The joint follow-up with the child’s pediatrician ensuredher eventual return to normality. The girl remained asymptom-atic and attended school, developing an adequate relationalmaturity that coincided with her emotional separation andindividuation from the mother figure.

Clinical Case 2

The victim of the second case of MSbP was 7 years old at thetime of consultation and was the third of four siblings. Thepediatrician who received her in the emergency room hadpreviously attended to her two brothers due to manifestationsof paroxysmal metabolic acidosis, dysarthria, ataxia,somnolence, and coma. This coincidence prompted the reviewof the family’s medical history. The two brothers had beenrepeatedly examined in four hospitals in two Spanish autono-mous communities; however, no explanation for these epi-sodes could be found. The pediatrician suspected the possibil-ity of MSbP and admitted the girl for her own protection,assessment, and treatment.

At the time of admission, the patient was in coma which wascomplicated by aspiration pneumonia. The nursing staff ob-served the mother trying to administer yoghurt to the victimwhile she was in the comatose state. In the PICU, the girl

presented two cardiorespiratory arrests. During the second epi-sode of cardiac arrest, themotherwas trying to calm the situationby saying Bdoctors, don’t rush about so much, nothing is goingto happen to my daughter .̂ After recovery from coma, the girlwas assessed within the same clinical picture as experienced byher brothers: dysarthria, ataxia and somnolence. Apparently,the girl had been suffering the aforementioned symptoms formore than 3 years, where they had progressively become morecomplex including seizures and coma.

After reading the available medical reports and talking withthe doctors who had treated the girl and her siblings, we identi-fied significant discrepancies between the anamnesis providedby the mother and the real facts. Moreover, the patient hadentered into a comatose state more than 27 times, but only themother had witnessed the convulsions that supposedly precededcoma. The description provided by the mother concerning theseizures varied and became more elaborate from one report tothe next, where the mother was able to incorporate new infor-mation that she had received from the doctors during previousvisits. Also, the patient underwent complementary examinations(laparotomies, liver biopsy, sural nerve, quadriceps, and rectalbiopsies) that were not free of risk. The child was sent to see achild psychiatrist because a brain biopsy had been scheduled,which caused the head of pediatrics to become concerned.

Furthermore, the mother always accompanied her daughterwhile she was in the hospital. During the hospital stays, shedisplayed behaviors of satisfaction and Boverfriendliness^,which can be characteristic of MSbP perpetrators [59]. Shehad also been diagnosed as having a borderline personalitydisorder and kleptomania.

The sudden death of the maternal grandmother and theappearance of episodes of unexplained apnea in the patient’ssister fulfilled all of the 10 warning signs associated withMeadow’s syndrome [45] (Table 3). The confirmed diagnosisof MSbP led us to initiate urgent treatment and temporaryphysical separation by court order.

During the therapeutic process, which lasted for 2 years, thevictim presented two relapses that included ataxia, dysarthria,and drowsiness lasting 24 h. Subsequently, toxic levels ofPhenobarbital® (59.2 mcg/ml in the first and 96.2 mcg/ml inthe second) were detected after the timely shipment of serumsamples by the family doctor. When the mother was confrontedwith these findings, she confessed to causing the toxicity andbeing the fabricator of the non-existent convulsions.

The psychological examination showed the victim to besignificantly affected on both personal and relational level,where she demonstrated ambivalent behavior. It was observedthat her personal relationships ranged from Bclinging^ to thepeople she trusted to rejection, which was expressed in theform of psychomotor agitation when faced with the slightestof frustration, toward strangers. She maintained a differenttype of relationship with the mother, which was cold andfearful, but mainly symbiotic.

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Once the mother had been confronted with reality and dy-adic psychotherapy concerning the mother-to-child attach-ment was initiated, the victim began to resume her normaldaily activities and was able to return back to school. Lateron, the child protection agency that supervised the case indi-cated the mother and child were being treated separately inindividualized and comprehensive programs for emotional re-habilitation. No other factitious symptoms have appeared inthe patient, in her siblings, or in any of the other members ofthe nuclear family.

Clinical Case 3

The third case is an example of MSbP with the subtypeBdoctor shopping with the child as proxy patient^ involvinga 9-year-old girl. This subtype is characterized by the mothertaking the child on rigorous pilgrimages to visit outpatientclinics and emergency rooms, and involves a milder form offactitious behavior regarding the child [73].

The case was referred to us by neurology as the child hadbeen suffering repeated and untreatable paroxysmal episodesof syncope for 3 years. She was also obese and was beingtreated by a dietician, and neurologists had begun to assessher for possible epilepsy. Additionally, she was being assessedfor delayed growth. When the family was referred to us, themother had requested allergy treatment as the child was alleg-edly suffering from asthma. The mother had also sought helpprivately from psychologists, claiming that her daughter wassuffering from forced right-handedness, dyslexia, dyscalculia,and supposed maturational, mental, and academic delays,which were never proven. Upon examining their medical his-tories, going back practically to birth, one comment made bythe patient’s mother stood out as she boasted about the factthat both her and her daughter had suffered Ball the diseases inthe world^.

The 35 hospital discharge reports that we collected camefrom 6 hospitals and virtually involved all pediatric special-ties. The data obtained from the reports did not coincide withthe anamnesis reported by the mother, and in addition, theseverity and complexity of the alleged signs and symptomscontradicted with the girl’s good clinical appearance. Uponexamining the reports, it was concluded the most commonreason for the interminable number of visits to the doctorwas always the same recurrent episodes of syncope, which iswhy the family had been referred to us. In 13 of the 35 reports,the manifestation of a special loss of consciousness whichoccurred at the end of the Bsyncopal episodes^ was added tothe emerging picture. The pediatricians who had witnessedthis loss of consciousness described these events asBlightheadedness similar to the fainting of film actresses^.

In addition, Bartter syndrome, Fanconi syndrome, a possi-ble ovarian cyst, surgically treated appendicitis with a normalanatomopathological outcome, and generalized photo

paroxysmal epileptic seizures were among the pediatric diag-noses that figured in the 35 reports. Furthermore, previouslycarried out treatments had always proven ineffective.

The diagnoses, made by the nine psychiatrists who hadpreviously attended to the mother, covered a broad psycho-pathological spectrum including depressive disorder, gam-bling disorder, and borderline personality disorder. We wereable to identify the existence of a pathological and almostsymbiotic mother-to-child attachment. On some occasions,the symptoms of the mother were transmitted to the daughterand vice versa. The victim represented this relationship ofconnection in her spontaneous drawings (Fig. 1). Both themother and the daughter seemed to compete with each otherfor the number of clinical studies conducted on them. Also,the mother sought medical attention for paroxysmal episodesof syncope and dizziness similar to that experienced by thevictim. The medical consultations of the mother and daughterwere simultaneous; the two always accompanied each other inthe waiting rooms.

It was known that the mother had studied nursing.However, during her first 10 years of life, she had sufferedfrom renal tuberculosis, which in her opinion Bhad not beendiagnosed in time^ and Bhad been riddled with unfortunatemedical interventions^. In the end, the kidney disease led to aright renal nephrectomy that the mother described as beingpsycho-traumatic in her own medical history.

As the case showed 9 out of the 10 warning signs proposedby Meadow [45] (Table 3), a multidisciplinary treatment in-volving pediatrics and a child protection agency was set up. Inthe confrontation with reality, the mother would come to ac-cept to having invented almost all the signs and symptoms andwe made her understand that her profound need of medicalconsultations had actually caused the sudden episodes offeigned syncope. It was also explained to the mother that her

Fig. 1 In this spontaneous drawing, the patient in case 3 representedherself in bed with her mother at her bedside. The child stated that Bthisis a girl who has eaten cold ice creams and has a fever and dizziness. Shehas to be with her mother in a hospital room to be cured, she needs somemedical tests and maybe surgery like her mother had”

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daughter colluded with her, exhibiting the same symptoms, inorder to maintain the bond between them. The psychothera-peutic treatment, which focused on making the girl autono-mous, acting as an individual, was complemented withmother-to-child group therapy sessions. This was done togeth-er with other mother and daughter pairs seeking to readjusttheir social relationships to improve mother and child interac-tions and to adopt healthier behaviors that eliminated thesearch for hospital contexts.

In subsequent evaluations, it was observed that the symp-toms experienced by the mother and daughter disappeared.The child went on to regain a normal body weight and inte-gration at school, without further fictitious visits to the doctor.

Clinical Case 4

The fourth case of MSbP also involved the Bdoctor shoppingwith the child as proxy patient^ subtype [73]. The victim wasa 9-year-old boy who was referred from pediatric nephrologydue to recurrent and paroxysmal Babdominal pain^, whichwas often accompanied by hematuria. These symptoms hadcaused the child and his mother to seek emergency room at-tention on several occasions, at which time the child had re-ceived medical assistance from almost all types of pediatricspecialists. The exhaustive studies carried out did not revealan organic cause which could explain the pain. The abdominalpain was described as being psychological and no treatmentwas provided. Faced with this situation, the mother increasedher factitious behavior by proxy and appeared with a containerof macrohematuric urine that she claimed was collected thelast time the patient had urinated. Later on, during confronta-tion, the mother would come to admit to having altered thesample with her own menstrual blood.

In order to assess the hematuria, the patient was admitted tohospital and another extensive study was repeated, includinglaboratory tests, several abdominal magnetic resonances, gas-troscopies, and laparoscopies. The results showed noetiopathogenic organic alteration. When no new episodes ofBhematuria^ were observed, and in view of the patient’s goodgeneral condition, the child was discharged from hospital withthe same functional diagnosis as upon admission. This thenleads to more insistent and dramatic pilgrimages to other hos-pital centers. The mother provoked the abdominal pain whenin the presence of doctors by slightly squeezing the child’sstomach, which seemed to act as an Bon–off^ switch thattriggered the victim to complain in a colluding manner. Therecurrent and paroxysmal abdominal pain led to five surgicalinterventions being carried out, all of which were unnecessary.The various surgeries performed included an appendectomy,three interventions to treat alleged sequelae flanges, and final-ly an operation for unilateral cryptorchidism that the patientactually suffered from and lead with the unnecessary ablationof his left testicle.

The patient was an only child and lived with his mother andstepfather. According to the mother, her husband was beingtreated for paranoid schizophrenia with depot antipsychotics.The mother complained about her husband’s constant imbal-ances, which included delusions, threats, frequent familyabuse, and alcohol consumption. However, in the few inter-views, we conducted he seemed normal and we were unableto confirm the diagnosis. The mother’s psychiatric anteced-ents included being admitted to psychiatric units on severaloccasions and being diagnosed with major depressive disor-der. In the anamnesis, it was recorded that the mother hadexperienced physical and sexual abuse during childhood. Atthe time of our intervention, she had abandoned, on her ownaccord, the outpatient treatment prescribed by the psychiatristwho had previously treated her.

Essentially, the techniques for treating the mother-to-childattachment were made possible owing to the child’s high levelof emotional and intellectual development, as shown in hisseries of drawings about Bthe illness^, starting from his ad-mission (Fig. 2) up until discharge (Fig. 3). The patient wasable to achieve individuation without the mother being pres-ent, as she had been admitted to the psychiatric unit of ourhospital for the treatment of her depression.

This case involved 8 of the 10 warning signs attributed toMeadow’s syndrome [45] (Table 3). The intervention provid-ed by the child protection agency temporarily separated thevictim from his mother and disrupted further factitious visitsto the doctor. The recovery of the mother-to-child attachmentfacilitated the mother’s confrontation with reality, where shewas able to partially understand the process that gives rise toMSbP and to accept her own psychiatric treatment.

Results

Symptoms and Signs

Two of the four cases (cases 1 and 4) exhibited 8 of the 10Meadow warning signs associated with the presence of MSbP(Table 3). Case 3 showed 9 of the 10 signs, since a similarillness was experienced by the mother, which can be added tothe list of 8 warning signs common to all four cases providedbelow. Case 2 presented all 10 warning signs, with the twoadditional signs of siblings experiencing a similar illness andunexplained deaths in the family. This particular family was atthe greatest risk of the presence of MSbP.

The following is a list of the eight warning signs identifiedin all four cases: 1—the presence of fictitious or factitioussymptoms and signs (fever, polyuria and dehydration in case1, drowsiness, dysarthria, ataxia, seizures, and coma in cases 1and 2, lightheadedness/dizziness and syncope in case 3, ab-dominal pain and hematuria in case 4); 2—the illness is de-fined as being unexplained and chronic (in all four cases the

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illnesses were prolonged, recurrent and unexplained); 3—dis-crepancies between the anamnesis provided by the motherand the child’s actual medical history (in all four cases); 4—the symptoms and signs are extraordinary (the symptoms andsigns did not correspond to any known medical disease in anyof the four cases); 5—symptoms and signs always displayedwhen mother is present (in all four cases, although the victimin case 1 also experienced feigned epileptic seizures when themother was absent); 6—the mother is overly involved andfriendly with the doctors and nurses (all four mothers werehelpful and overly involved but rejected and commented onall staff decisions); 7—all treatments employed are poorlytolerated (all four victims experienced a surprising series ofuncommon side effects to the treatment they were provided);and 8—the mother is incomprehensibly indifferent about theprognosis (in cases 1 and 2, the mothers showed no concern

over the life-threatening symptoms experienced by their chil-dren, while in cases 3 and 4, the mothers showed littleconcern).

Clinical Presentations

In cases 1 and 2, the presentation was Bnon-accidentalintoxication^ leading to Bfictitious epilepsy^ with neurologi-cal symptoms of loss of consciousness and coma. In cases 3and 4, the presentations were included within the Bdoctorshopping with the child as proxy patient^ subtype; in case 3,the presentation was Bsyncope^ leading to Blethargy^ and incase 4, Babdominal pain^ leading to Bhematuria^.

Methods of Deception Employed

It was confirmed that in cases 1 and 2, the signs and symptomswere actively falsified by the administration of drugs over theprescribed dose. In cases 3 and 4, the mothers did not exhibitbehavior of active fabrication; they only used methods of in-vention and falsification. In general, the mothers in all the fourcases exhibited behaviors of simulation and pretense.

The Mothers’ Psychopathological Profile

The mothers of all four cases had psychiatric antecedents. Themother in case 1 had been diagnosed with histrionic person-ality disorder. The second mother was diagnosed as havingborderline personality disorder, as well as displaying klepto-maniac behavior inside the hospital. The third mother wasdiagnosed with depressive disorder, gambling disorder, andborderline personality disorder. It was also confirmed thatshe had factitious disorder imposed on self, as she also soughtmedical attention for medical conditions similar to those shehad fabricated in her daughter. The fourth mother was onceagain admitted to the psychiatric unit due to the exacerbationof major depressive disorder, which she had previously beendiagnosed with.

Fig. 2 The patient in case 4 drewa Bdrawing about the disease^ atthe onset of the consultations andrelated his perception regardingthe drawing. The child stated thatBone time a man found out thatthe appendix was very dangerousand had killed 1.000.000 people.When he got home and becameill, his wife called an ambulanceand they took him to theEmergency Room where theytold him they had to remove hisappendix, but half an hour later hedied^

Fig. 3 The patient in case 4 drew a new Bdrawing about the disease^ atthe end of the treatment and related his improved perception regarding thedrawing. He recounts a story with a better ending: BIt’s a boy who is 9who was losing all of his teeth. They didn’t know what the disease wasbut what was happening was he wasn’t eating fruit. His father drives himto the Emergency Room where the pediatrician tells him he has scurvyand that they cure this with vitamin C. The doctor gave him vitamin C andlittle by little the scurvy was cured^. It was observed that the child wasprogressing favorably, which is reflected in his noticeably improvedprognostic opinion

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The existence of childhood abuse and trauma was con-firmed for the mother in case 4, who had a history of physicaland sexual abuse. She had also been the victim of domesticviolence as an adult. The mother in case 3 retold a traumaticexperience related to kidney disease that she had suffered fromsince childhood. For the mothers in cases 1 and 2, no evidenceof abuse or trauma could be verified.

To study the behavioral psychopathological profile of the 4mothers affected by MSbP, a semi-structured clinical inter-view, involving 10 items, was carried out to analyze theirbehavior during hospital stays (Table 2). The results werethe following: (1) the four mothers were attracted to hospitalsand their situational context; (2) they had concerns and per-sistent ideas regarding illness and the medical environment;(3) all recalled having lived gratifying situations in the hospi-tals and that they occupied a great deal of their time planningnew hospital stays; (4) the four mothers expressed a growingneed for the search of the hospital context, which was aggra-vated by stressful circumstances; (5) in all four cases, themothers recognized that their search for the hospital contexthelped them to escape everyday problems; (6) all confirmedthe repeated failure of controlling their impulse to take thechild to the hospital; (7) the four mothers reported experienc-ing dysphoria, irritability, and anxiety when trying to suppresstheir factitious behaviors; (8 and 9) they also stated that theyfelt excitement immediately before going to the hospital andwell-being during their hospital stay; and (10) all admitted thatin order to achieve their goal, they were capable of misleadingtheir family and doctors by inventing, falsifying, or fabricatingsymptoms and signs in their children.

The Mother’s Healthcare Training

It was observed that all mothers indirectly acquired clinicalknowledge during each successive consultation with the doc-tors. This was especially noticeable in cases 1 and 2, where themothers’ descriptions of the symptoms regarding the fictitiousepilepsy became more elaborate and detailed with each hos-pital stay. The mothers incorporated new knowledge that thedoctors had transmitted to them. In case 3, it was known thatthe mother had studied nursing.

The Mother-to-Child Attachment

The relationship between mothers and children was cold andsymbiotic in all four cases, where the victims presented sig-nificant difficulties in the process of separation–individuation.The children in the presence of the mothers appeared to beBover attached^ to them, maintaining the extreme relationalpatterns of Bclingy^ to rejection, without any intermediatestep. The victim in case 2 was totally unaware of what washappening to her. The victims in cases 1, 3, and 4 showedbehaviors of cooperation and collusion with the mother. In

case 1, the girl actively feigned epileptic seizures in the ab-sence of the mother, whereas in cases 3 and 4, the victimsexhibited a type of passive collaboration when the motherwas present.

Treatment

In all four cases, confrontation with reality and mother-to-child attachment therapy were undertaken. The mother in case1 was confronted during a 6-h session, which produced posi-tive results that caused the factitious symptoms to stop. Themother in case 2 took part in a process of confrontation lasting2 years, which included periods of partial acceptance of thedeception and two repeats of factitious behavior. The confron-tation that took place in case 3 required the presence of thedaughter in order for the mother to admit to having a factitiousdisorder. In case 4, the mother accepted her own psychiatrictreatment after confrontation.

The mother-to-child attachment therapy, which sought toachieve autonomy and individuation of the victims, showedthe best results with respect to case 4, producing the highestdegree of separation–individuation. For the rest of the victims,an adequate level of individuation was also achieved. In case3, sessions of mother-to-child attachment group therapy wereprovided to help improve the bond between mother anddaughter.

Follow-up and Protection

In all four cases, child protection agencies were notified.However, in case 2, there was judicial intervention for thesafeguard and necessary protection of the victim and her sisterand brothers. Also, collaboration with general pediatricianswas an essential element to ensure the follow-up of the vic-tims. In case 2, the syndrome went on to affect not only twoother siblings but also the victim until MSbPwas detected andthere was contact between the various health services.

Of the four cases described, the process of rehabilitationand recovery leads to physical separation between the motherand the victim in cases 2 and 4.

Discussion

Among the studies published on MSbP, involving a broadrange of cases, are those by Rosenberg [55] with 117 casesand Sheridan [63] with 451 cases. Feldman and Brown [20]reviewed 59 articles from 24 countries and found 122 cases, ofwhich 86% had the mother as the sole perpetrator. In theRosenberg review (1987), all the perpetrators were mothersof the victims, and in the Sheridan survey [63], the motherswere perpetrators in 76.5% of the cases. A systematic searchfor case reports and series published since 1965 by Yates and

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Bass [74] yielded a sample of 796 perpetrators, of which95.6% were the victims’ mothers. Regarding the four casespresented here, all the perpetrators were the biologicalmothers.

Reviews focused on understanding the psychopathology ofperpetrator and their significance in the etiology of MSbP areless frequent [9, 11, 14, 58]. Bools et al. [13] found that out of47 mothers studied, 41 had a previous psychiatric history andthe remaining 6 acquired one after factitiously seeking medi-cal treatment for their children. Themain personality disordersinclude those of the borderline subtype, factitious disorders onthemselves, depressions, and problems with addiction. Bassand Jones [11] found that 64% of the 28 women diagnosedwith MSbP had factitious disorder. These data coincide withthe high level of psychopathology that is suspected in thesemothers [74] and with the clinical cases of the four perpetra-tors of MSbP presented in this article. Personality disorderswere a common diagnosis in all four cases, two borderlines,one dependent, and the other histrionic. Depressive disorderswere detected in two of the mothers, and addictive disorderswere found in other two, being kleptomania and gamblingdisorder. Finally, we came to discover that only the motherperpetrator of MSbP in case 3 had sought medical attention onher own accord for factitious disorder imposed on self.

With respect to proposing a common behavioral profile ofperpetrators, which would help to identify them, we have ver-ified by use of a semi-structured clinical interview that theirlives revolve around the hospital context ([32], p. 199). In ourfour cases, the mothers were confident and self-assured withinthe hospital, appearing to be satisfied and rewarded by theirrole as Bthe mother of a sick child^. A behavior similar to thatof addicts was observed in all four cases, where the objects ofaddiction were the hospital context and the issues related tomedicine. All four mothers believed that their child was sickand needed medical attention and believed that their lies werenecessary in order for doctors to accept their children’s illness.The four mothers interacted with the health professionals as ifthey were Bcolleagues^ and Bequals^ and sought to participatein the usual dialog among doctors. Moreover, they ventured tocomment on the reports, demanded additional tests be carriedout, new admissions to hospital or treatments, and offeredtheir opinion and help. They wanted to form part of theBhospital game^ because this offered them reward; they tookpride in the social recognition they received for their role asBself-sacrificing^mothers. None of the four mothers was fullyaware of the damage they were causing ([39], p. 99).Relationships with these mothers become difficult from a pro-fessional perspective, because beneath their apparent normalappearance is a serious psychopathology [11, 74].

The ways in whichMSbP is manifested are varied, rangingfrom being quite straightforward to extraordinarily astonish-ing [4, 15, 68]. The psychopathology of the perpetrator under-lies the presentation of the syndrome, its symptomatology, and

the severity of the method used to deceive [9]. Our first twocases presented long-term fictitious epilepsy, a particularlyserious subtype of MSbP [22, 44]. In both presentations, theclinical picture progressed to coma. The last two cases, involv-ing the subtype Bdoctor shopping with the child as proxypatient^ tended to have a more non-threatening appearance,although the impact on the victim with respect to variouscontexts, such as schooling, affective, emotional, physical,or relational, cannot be considered any less serious ([32, 73],p. 197).

In MSbP, there are three levels of deception: invention(Bmy child has had an epileptic seizure^), falsification(Badding maternal blood to the child’s urine^), and fabrication(Badministering drugs over the prescribed dose^). It is consid-ered that cases involving fabrication have a worse prognosis[9, 47]. In our study, the four mothers invented and falsifiedsymptoms, although actively fabricated symptoms only oc-curred in cases exhibiting fictitious epilepsy. Throughout theprogression of the syndrome and owing to the situational stim-uli that intensifies the mother’s behavior, it could becomepossible that the perpetrator of fictitious epilepsy may switchfrom inventing convulsions to fabricating them [44, 47]. Alsoin the Bdoctor shopping with the child as proxy patient,^ sit-uations of stress or frustration could generate changes in themethod used to deceive, where the mother goes frominventing an abdominal pain to falsifying a hematuria, as incase 4 [33]. The perpetrator in case 4 increases the degree ofdanger caused by her actions to overcome the frustration thatwas caused by not being able to continue carrying out testsand treatments on her child.

Whichever the method used by the perpetrator of MSbP,the victim can show different degrees of understanding withrespect to what the mother is actually doing, ranging fromabsolute unawareness, as in case 2, to colluding with her.The latter can progress to the victim themselves, inventing apain, a convulsion, or paralysis, which can make diagnosiseven more complex [5, 38]. With the mother, the victim usu-ally appears cold, fearful, and quiet, but extremely dependenton the mother’s Bmonologue^. The patients in cases 3 and 4referred to and simulated what was being proposed by theirmothers in the presence of the pediatrician, exhibiting an atti-tude of passive mother-to-child collaboration. In case 3, thevictim colluded with her mother, which was confirmed bysimultaneously examining both of their clinical reports.They appeared to act as Bcommunicating vessels^ transmit-ting episodes of fictitious lightheadedness, the reason for theconsultation, from mother to daughter and vice versa. It wasnoted in the study of the family medical histories, going backto two previous generations, that there were often atypicalclinical pictures involving dizziness. These episodes had oc-curred starting from, at least, the maternal great-grandmother,suggesting a family Blegacy^ learned from the role of Bmotherof someone ill^ [37, 39, 56]. In the absence of the perpetrating

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mother, the victim in case 1 feigned fictitious epilepticseizures, in which the girl claimed to see Bthe Virgin Mary .̂The mother was unable to directly induce the seizures, as shewas not present when they occurred. The strength of thechild’s bond with the mother and the difficulties in separatingthem reflect the level of alteration of the mother-to-child at-tachment ([1, 32], p. 196). The percentage of cases where thevictim actively colludes with the mother is greater than half ofthe number of cases of MSbP reported in the literature [20, 55,63].

We have been able to confirm that some victims have anintuitive idea of the process in which they are involved. Thisperception provides them with a view of how the syndrome isprogressing, which can be used in some circumstances to as-sess whether the condition is improving or getting worse [33].Regardless of what it meant to be the victim of MSbP in case4, the child’s opinion about the curative capacity of the doctorsat the beginning of the consultations was quite negative, as wecan see in his first interpreting drawing of Bthe disease^ (Fig.2). At the end of the therapeutic process, his prognostic per-ception was more favorable and less disabling, as reflected inthe analysis of the new drawing he made regarding the disease(Fig. 3). The comparison of both drawings showed us how thepatient could, at last, avoid the effects that the mother’s psy-chopathology had on him.

Owing to the special characteristics of MSbP, it was neces-sary for us to use atypical and different clinical methods thanthose usually employed in medical practice for diagnosis([39], p. 139). The customary relationship between the pedia-trician and the patient’s family becomes much more complex,because the professional has to doubt the mother instead ofBtotally^ believing her, which is normally the case [61]. Thesuspicion of the pediatrician is what leads the case to psychi-atry [35, 66]. Before the psychiatric approach, all the signs andsymptoms provided by the mother and all those found in theantecedents of each case must be formulated in a precise andchronological manner. Also, it is recommendable that data beverified by the doctors signing the reports collected [23, 24].

The confirmed detection of MSbP requires a series of pre-ventive measures to protect the victim and/or siblings. As arule, and for victims of any age [16, 18], limits should be setregarding the complementary tests so as to not repeat thosealready performed or those that are potentially harmful, suchas the brain biopsy that was proposed in case 2, which oc-curred before we were consulted. In Spain, it is mandatory bylaw that a child protection agency is notified of any case ofMSbP [41, 69]. In the four cases of MSbP presented in thiswork, the guidelines for victim protection were adopted joint-ly between the pediatricians and the child protection agencies.The need for a higher level of protection was evident in thefirst two cases of MSbP, because both mothers used non-accidental poisoning which is a very dangerous method forharming the physical integrity of their children [58]. The

mortality rate among the victims of MSbP has been shownto vary, 6% according to Sheridan [63], 9% according toRosenberg [55], and 33.3% as reported by Meadow [46],which unite the most serious presentations of recurrent apneaby suffocation and fictitious epilepsies due to non-accidentalpoisoning [44].

Once the victims of the MSbP are safe, the therapeuticapproach to treat the perpetrators can gradually be initiated.To achieve a safe and effective intervention, action must betaken on three levels: (1) directing the confrontation (Table 5),(2) the continuation of mothers’ psychotherapy (Table 6), and(3) the completion of mother-to-child attachment therapy(Table 7). The first step in treating MSbP is to progressivelyestablish a therapeutic alliance between the mother and thepsychiatrist based on obtaining the mother’s confidence in thatwe can cure what is happening to her child ([39], p. 141).These mothers would be helped to discover their real capaci-ties for forming social relationships by strengthening theirself-esteem and providing positive reinforcement for behaviorunrelated to hospital contexts ([32, 49], p. 201).

Additionally, the entire family needs to receive treatment,since most often the fathers of the victims of MSbP are char-acterized as being absent from family life [48]. The personal-ities of the fathers in cases 1 and 2 were considered deficient,in case 3 there was no data regarding the father, and in case 4,the father had been diagnosed with Bparanoid schizophrenia^.Although the mothers in these three cases described their hus-bands as being passive and in need of help, the reaction totherapy of all three fathers was positive and each adequatelysupported the integration of the mothers in the socialenvironment.

Table 5 Therapeutic approach used on the confrontation with reality inMunchausen syndrome by proxy (MSbP)

1. Establish a therapeutic alliance based on trust and bonding

2. Help the perpetrator to discover their real capacity for experiencingempathy and social relationships, as well as their ability to handlestressful situations

3. Help to improve self-esteem and self-confidence

4. Promote the gradual modification of the perpetrator’s reward systemand self-awareness of the reasons why they search for the hospitalcontext

5. Family intervention, especially with the non-perpetrator father, tominimize undesirable side effects of the confrontation

6. Perform confrontation that is gradual, allowing the perpetrator torecognize the compulsivity of their factitious behaviors

7. Ensure that any associated psychiatric pathology detected receivespharmacological and psychotherapeutic treatment, as well as theconsequences of the abrupt suppression of the addictive behaviortoward hospitals

Adapted from Lopez-Rico and Jimenez-Hernandez [39]. Munchausensyndrome by proxy. Factitious Disorder in Pediatrics. SalamancaUniversity Press, Spain (p. 141)

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To achieve the emotional confrontation of the perpetratingmother with reality and her acceptance of the addictive behav-ior she is displaying toward the hospital context, individualpsychotherapy is essential ([39], p. 142). During therapy, themothers of all four cases described having difficult childhoodsand current problems with dependency, topics which werebroached by means of the therapeutic bond.

Confrontation is a gradual process in which progress isslowly made [10]. The messages that are initially transmittedto the mothers affected by MSbP are Bwe know the type ofillness your child is suffering from and we know how to cureit^ and Bwe are your friends and we will help you in any waywe can^ ([32], p. 200).

Knowledge of all possible clinical evidence, including se-cretly filmed videos, is absolutely necessary for structuringand discussing the confrontation [26]. In cases 1 and 2, toxiclevels of phenobarbital were detected in plasmawell above theprescribed doses. In case 4, the mother acknowledged that theurine sample given to the pediatricians presenting hematuriawas falsified using her own menstrual blood. In all four cases,the invention of symptoms was exposed.

When the diagnosis of MSbP is confirmed, a strategy isneeded that is established in conjunction with pediatriciansand periodically reviewed during the process. The informationgathered from other sources must be compared with themother’s anamnesis to compile a more accurate and detailedmedical history that helps to confront the mother with reality[6]. In Table 4, we detail the list of diagnoses encountered in the25 successive reports of all the doctors who had treated thevictim in case 1. Over time, themother combined the symptomsand signs appearing in these reports, forming a single episodicclinical picture, which she repeatedly used when consulting us.The episodes described by themother did not correspond to anyknown disease, and the analysis of the 25 reports allowed us tocluster the symptoms into four groups in a chronological order:first fever, second polyuria, third convulsions, and fourth coma.Later on, during confrontation, we discovered that the fever wasfictitious and fabricated by manipulating and changing the ther-mometer, polyuria was induced because the mother had giventhe child furosemide, the seizures were invented, and coma wasinduced when the mother administer an excessive dose of phe-nobarbital, exceeding the dose prescribed by the pediatricians,to her daughter.

In our experience, confronting the perpetrator with realitymust be oriented toward the mother’s acceptance of both herdeceits and her own psychopathology of being addicted tohospital contexts. The initial partial recognition of the addic-tive behavior is effective because it allows us to continueworking on the subsequent acceptance of false behavior. Inaddition, it is necessary that the perpetrator becomes aware ofwhat her behavior implies, so that her acknowledgement ofthe deception can have a favorable effect on how the syn-drome progresses and can stop the abuse and factitious behav-ior ([39], p. 141). In case 3, for example, the mother’s emo-tional confrontation with reality allowed the mother to under-stand that her Bhypochondriacal^ fears for the health of the girlwere the cause that ended up precipitating the episodes offeigned syncope. It was also explained to the mother that herdaughter was colluding with her, exhibiting the same symp-toms in order to maintain the bond between them.

Table 6 Characteristics of the individual psychotherapy of theperpetrator in Munchausen syndrome by proxy

1. Review of the traumatic processes experienced during the perpetrator’schildhood, including a history of sexual abuse, neglect, or physicalabuse

2. The verification of the existence of altered relational bonds between theperpetrator and her own mother, which should be remediated as muchas possible

3. Emphasis placed on improving the perpetrator’s ability to deal withprimary traumatic fears and feelings of guilt caused by abusivebehaviors

4. The use of cognitive-behavioral therapy to improve the perpetrator’ssocial skills and for changing their adaptive mechanisms, allowingtheir emotional needs and rewards to be met outside of the hospitalcontext

5. The psychotherapeutic environment provides the perpetrator with thepossibility of expressing their feelings of pain without having to resortto any extreme compulsive action against their child or againstthemselves

6. Finally, work is done on reforming and reconstructing the perpetrator’spersonal identity

Adapted from Jimenez-Hernandez and Lopez-Rico [32]. Munchausensyndrome by proxy. In D. Crespo-Hervas & Muñoz-Villa (Eds.).Psychopathology in the Pediatric Clinic. Majadahonda (Madrid): Ergon.(p. 202)

Table 7 Intervention for child victims of Munchausen syndrome byproxy

1. Pediatric care of secondary somatic problems associated with thesyndrome

2. Comprehensive psychological, maturational, school, and psychosocialevaluation

3. Individual psychotherapy complemented with the adequate school,family, and social rehabilitations.

4. Play therapy centered on topics such as fear of the destruction ofoneself, the loss of basic trust in the people loved by the child, thealtered relationship of mother-to-child attachment, and the mother’sdistorted image, which the child perceives as being full of contradic-tions

5. Cognitive-behavioral therapy aimed at reducing anxiety and the ac-quisition of skills necessary for coping with the stress caused by theunnecessary surgical interventions and painful exploration methods

6. Mother-to-child dyadic therapy to repair the altered relationship withthe mother that exacerbates anxiety and the feelings of stress

Adapted from Jimenez-Hernandez and Lopez-Rico [32]. Munchausensyndrome by proxy. In D. Crespo-Hervas & Muñoz-Villa (Eds.).Psychopathology in the Pediatric Clinic. Majadahonda (Madrid): Ergon(p. 202)

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The victim or victims should be well protected during con-frontation [65]. In the event that physical separation of themother and child is necessary, the child protection agency ofeach country would be responsible for carrying this out [6,74]. In cases 2 and 4, physical separation measures were nec-essary to protect the victim. The psychotherapeutic environ-ment must provide the possibility for the mother to describeher painful feelings without having to resort to any extremecompulsive action against the child or against herself [72].The characteristics of the confrontation of perpetrators shouldbe studied from the perspective of identifying standard proto-cols for all types of patients [42].

The recovery process of each victim ([52], p. 14) is basedon mother-to-child attachment therapy and must also be su-pervised from a multidisciplinary perspective to reinforce theachievements obtained [62]. Regarding the mother-to-childattachment therapy, one of the basic steps taken to re-establish the appropriate relationship between the mother suf-fering fromMSbP and the victim is to get the mother to acceptthe proposal of starting or continuing individualized psycho-therapy [1, 9, 49]. In case 2, a judicial sentence was required inorder for this to happen, and in case four, this was achieved byhaving the mother admitted to a psychiatric unit. The separatetreatment of each victim permitted the individuation and psy-chological restructuring in all four cases. The four victimsprogressively understood that their mother had a disease thatled her, unknowingly, to invent and even to fabricate symp-toms in them because of the needed to be in a hospital context.

One of the foundations for success in these extremely com-plex cases is their timely detection, generating the need for aninterhospital registry to be established [6]. Other approachesinvolve the creation of committees or groups of experts thatcould collaborate to solve cases of MSbP once they have beendetected, as well as the development of programs to adequate-ly prevent and treat this syndrome [23, 66]. For 30 years, it hasbeen known that there is a need in Spain for the multidisci-plinary treatment of MSbP [31, 54, 61]. Moreover, this mul-tidisciplinary approach could be implemented in all countriesand include research, the prevention, and global treatment ofMSbP [2, 20, 70, 74].

Conclusion

MSbP or factitious disorder on another must be identifiedusing a psychopathological, emotional, and behavioral pro-files shared among perpetrators. These mothers present a typeof behavior that is in constant search for the hospital contextand topics related to medicine, which for them produces greatreward. Their motivational force revolves around visiting andstaying at hospitals, and in order to achieve this, they arecapable of inventing, falsifying, and fabricating diseases intheir own children. The common and basic behavior of

perpetrators of MSbP presents a high degree of psychiatriccomorbidity with other addictions, personality disorders, anddepression. The therapeutic approach to MSbP uses similarmethods employed to treat addictive behaviors, where theobject of reward is focused on attending medical consultationsor on hospital stays. During confrontation with reality, themother must accept her deceptive and addictive behavior inorder to commence the psychotherapeutic process aimed atdisrupting factitious behavior. In MSbP, there is serious pa-thology regarding the mother-to-child attachment where theprocess of individuation–separation is altered and, in somecases, the victims actively participate in the deception believ-ing they will become more united with their mothers.Attachment therapy begins by using tactics to bond with boththe victim and the perpetrator and is completed by using dy-adic therapy. In all cases of MSbP, it is necessary to carry outan extensive psychotherapeutic follow-up to assess the recov-ery of both the victim and their mother, focusing on theirability to gain family and social rewards which are totallyhealthy. This conclusion is supported by four clinical casesthat suggest some hypothetical correlations that need to befurther investigated.

Compliance with Ethical Standards

Conflict of Interest The authors declare that there is no conflict ofinterest.

Ethical Approval Not applicable as no new clinical data was collectedafter 1987.

Informed Consent At the time of the retrospective analysis carried outhere, we no longer had access to the patients.

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