Special Article

Psychother Psychosom 2008;77:209–218 Published online: April 16, 2008

DOI: 10.1159/000126072

Management of Factitious Disorders:

A Systematic Review
Sarah Eastwood Jonathan I. Bisson
Department of Psychological Medicine, Cardiff University, Cardiff, UK

Key Words strong evidence regarding the effectiveness of different

Factitious disorder ⴢ Munchausen’s syndrome ⴢ Global management approaches. Conclusions: There is an absence
Improvement Scale of sufficient robust research to determine the effectiveness
of any management technique for FD. The establishment of
a central reporting register to facilitate the development of
Abstract evidence-based guidelines is recommended.
Background: The literature regarding the management of Copyright © 2008 S. Karger AG, Basel
factitious disorder (FD) is diverse and generally of case re-
ports or case series. To date there has been no systematic
review of the effectiveness of management techniques. Introduction
Methods: Systematic review of all evidence reporting the
management and subsequent outcome in FD. Data were ex- Factitious disorder (FD), although a psychiatric condi-
tracted and outcomes were assessed using an adaptation of tion, challenges practitioners in most fields of medicine.
the Global Improvement Scale. Results were analysed by Categorised as an Axis I DSM-IV condition, it is diag-
parametric statistical tests; a meta-analysis was not possible. nosed when there is intentional production or feigning of
Results: Thirty-two case reports and 13 case series were eli- physical or psychological signs or symptoms where the
gible for inclusion. Analysis of the case reports found no sig- incentive is to assume the sick role and external incen-
nificant difference in outcomes between confrontational tives for the behaviour are absent [1]. In the ICD-10 (F68.1)
and non-confrontational approaches [t(29) = 0.72, p = 0.48], it is defined as repeated and consistent feigning of symp-
between treatment with psychotherapy compared to no toms with obscure motivation for the behaviour and best
psychotherapy [t(30) = 0.69, p = 0.48], and when psychiatric interpreted as a disorder of illness behaviour and the sick
medication had been prescribed compared with not [t(30) = role [2]. Once an organic cause has been excluded, the
0.35, p = 0.73]. A trend was observed that a longer length of main differential diagnoses are malingering where exter-
treatment lead to better outcomes, but this was not signifi- nal incentives are present for the intentional behaviour,
cant [F(5, 26) = 1.17, p = 0.35]. The consecutive case series and somatoform disorders in which both unconscious
demonstrated that many FD sufferers were not engaged in symptom production and unconscious motivations are
treatment and were lost to follow-up but did not provide any present. Distinction between these disorders can be very
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© 2008 S. Karger AG, Basel Dr. Jonathan I. Bisson

0033–3190/08/0774–0209$24.50/0 Clinical Senior Lecturer in Psychiatry, Department of Psychological Medicine
Fax +41 61 306 12 34 Monmouth House, University Hospital of Wales
E-Mail karger@karger.ch Accessible online at: Heath Park, Cardiff, CF14 4XW (UK)
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www.karger.com www.karger.com/pps Tel. +44 29 2074 4534, Fax +44 29 2074 7839, E-Mail BissonJI@cardiff.ac.uk
difficult and the diagnosis of FD can only be wholly con- ‘factitious disorder’ or ‘Munchausen’s syndrome’ and ‘treatment’
firmed if observation of symptom-producing behaviour or ‘management’ or ‘therapy’ or ‘psychotherapy’ or ‘pharmaco-
therapy’. Additional papers were found by hand searching the ref-
occurs or it is admitted. As neither of these scenarios is erences of retrieved articles. The search was restricted to papers
frequent, diagnosis usually remains only a high index of with English language abstracts.
suspicion [3].
A community study of 2,363 people in Italy found the Study Selection
lifetime prevalence of FD to be 0.1% [4]. Estimated prev- All systematic reviews, randomised controlled trials, con-
trolled trials, case series or case reports which appeared in peer-
alence rates of FD have varied in other studies from 0.6% reviewed journals were eligible for inclusion. To be included,
of 15,000 psychiatric consultations [5] to 9.3% of referrals studies had to be of FD or Munchausen’s syndrome that had been
to the National Institute for Allergy and Infectious Dis- confirmed or was considered the most likely diagnosis in patients
ease with fever of unknown origin lasting over 1 year [6]. of either gender aged over 16 years; cases of Munchausen’s by
It is generally acknowledged that the disorder is under- proxy were excluded. Papers were only included if they described
the management techniques used and subsequent outcomes. The
reported and constitutes a real problem to medical ser- searching and selection were done by the authors. Any disagree-
vices [7]. Munchausen’s syndrome [8] is an extreme form ments with regard to inclusion or exclusion of a study were re-
of FD and is estimated to account for around only 10% of solved by discussion.
the factitious population [9], despite its overrepresenta-
tion in the literature. In Munchausen’s by proxy the feign- Quality Assessment
All papers deemed eligible for assessment were assessed for
ing of symptoms is in another person for the purpose of quality using a standardised form. This form incorporated factors
assuming the sick role by proxy and will not be consid- used in systematic reviews within psychiatry [17] but was spe-
ered further in this paper. The phenomenon of autode- cifically designed for this study. The criteria included clarity of
structive behaviour has also attracted interest, encom- the description of techniques used, evidence of selection bias (e.g.
passes a broader spectrum of disorders than FD alone selected versus consecutive selected cases), whether follow-up was
reported for all patients in the series, and whether there was in-
and reported prevalence rates range from 0.032 to 9.36% dependent assessment of outcome using a standardised scale.
[10].
The literature regarding management of FD is scarce Data Extraction
and of poor quality. Many different techniques have been All included papers were scrutinised and relevant data were
reported, primarily focussing on confrontational [11–13] extracted using a specially designed form. The data were extract-
ed in the categories of demographic information, details of the
versus non-confrontational [14] approaches. Various FD, the method of diagnosis, the occurrence and form of any con-
forms of psychological, medical, surgical and conserva- frontation, therapies used, the duration of therapies and the pa-
tive treatment methods have also been reported. Several tient condition at the last reported follow-up. The accuracy of data
publications have provided useful reviews of the manage- extraction was double checked by the second author.
ment of FD [15, 16] but to date there has been no system-
Assessment of Outcomes
atic review of the management of FD or any clear evi- As a result of the absence of the use of standardised outcome
dence to suggest that one treatment method is beneficial measures, the authors objectively assessed the studies using an
above and beyond others. adaptation of the Global Improvement Scale (GIS) from the Clin-
Given the limited existing literature, we decided to ical Global Impression scale [18] to quantify the change in overall
perform a systematic review of published evidence re- condition of reported patients. The papers were reviewed and
scored by both authors, there was good concordance between the
garding the management of FD and then use this evi- scores of each assessor and any disagreements were resolved by
dence base to hypothesise an effective management strat- discussion.
egy which can be implemented in clinical practice and
tested in future research. Data Synthesis
An initial narrative synthesis was undertaken to describe the
characteristics, quality and outcomes of the studies. There were
insufficient trials of good quality to perform a true meta-analysis
Method of the results. SPSS 12 was used for statistical analysis of extracted
data and GIS scores. Case reports and case series were analysed
Data Sources separately due to their heterogeneity. For the case report data
A systematic bibliographic search was undertaken to identify parametric statistical testing was used to compare the mean dif-
all evidence regarding the treatment and management of FD from ferences in outcomes across different variables. Independent t
databases (EMBASE, Medline, PsycINFO and CINAHL), and the tests (two tailed) were used to analyse the mean difference in out-
Cochrane Library with each database being searched from incep- come variables with two conditions (e.g. confrontation and no
tion to December 2005. A search string of keywords was used; confrontation) and one-way ANOVAs were used to analyse the
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mean difference in outcomes across variables with more than two Management
conditions, all tests were applied at a 5% level of significance. The Confrontation
distribution of the data for the sample size of 32 was close to nor-
mal and therefore the sample size of 30 or more and normal dis- Seventeen (53.1%) patients were confronted with their
tribution preconditions were satisfied for the use of parametric diagnosis, 14 (43.8%) were not and confrontation was not
tests. The data from the case series could not be synthesised in reported once (3.1%). Of those who were confronted, 14
this way. had a non-punitive approach (82.4%), two had an accusa-
tive approach (11.8%) and the exact method was not re-
ported in one (5.9%).
Results Different non-punitive confrontational techniques
were used. In some, the patient was presented with diag-
Trial Flow nostic test results and it was explained that as they did not
match the clinical picture it was believed that symptoms
The search identified 132 papers for potential inclu- were being feigned [27]. Another technique was to tell the
sion in the review, no systematic reviews, randomised patient that whilst doctors believed the symptoms were
controlled trials or controlled trials were identified. A to- being produced by the patient treatment was available
tal of 45 papers were eligible for inclusion in the review; [26, 28–30]. One accusative confrontation occurred in an
32 case reports and 13 case series. The main reasons for angry, non-therapeutic atmosphere; the authors conclud-
exclusion of papers were poor reporting of management ed that this was critical in the subsequent success of treat-
strategies and outcomes or that they were anecdotal case ment [31]. One patient was repeatedly and vigorously
histories focussed on presentation and diagnosis. Some confronted with the diagnosis whilst in a locked psychi-
large case reviews were excluded because there was insuf- atric ward from which she was unable to escape [12].
ficient detail to determine which treatment methods gave A variety of scenarios were reported in those cases
more successful outcomes than others [13]. where confrontation did not take place. In some cases
confrontation was not required as the patient had admit-
ted to fabricating illness [33–36]. Some authors reported
Case Reports that confrontation was not used for fear of damaging the
therapeutic relationship [37, 38]. For one patient, a ver-
Study Characteristics sion of inexact interpretation was adopted in which there
Table 1 includes the characteristics of the case reports. was free access to a hospital bed for a year and the patient
There were 8 males and 24 females with an age range of was in control of when they came to hospital but a re-
19–64 years (mean = 32.0, SD = 10.7). Seventeen patients sponse to treatment was expected [19]. Jamieson et al. [39]
had been diagnosed with Munchausen’s syndrome, of reported a technique where 2 psychiatrists collaborated;
these 5 were male. Duration of factitious illness ranged one acted as a helper and one as a confronter and through
from 2 months to 40 years (mean = 6.5 years, SD = 7.5). alternate interactions with the patient, enabled her to ac-
Psychiatric comorbidity was common; 50% had another knowledge the diagnosis.
Axis I diagnosis and 40% had an Axis II diagnosis.
Other Interventions
Quality Assessment Nineteen patients (59.4%) received only one type of
Case reports are flawed in terms of proving the effi- treatment, the others (40.6%) received multiple therapies
cacy of an intervention, in particular because of the risk as outlined in table 1. Psychotherapy was the only form
of positive reporting bias and the absence of any form of of therapy in 16 patients (50.0%) but in many the exact
control. There was considerable variation in the amount regimen was not fully described or techniques had been
of information provided regarding demographic details combined. Six (18.8%) cases reported the concurrent de-
of patients and the management techniques used. Only livery of psychotherapy and medication. Other reported
10 (31.3%) reports made reference to diagnostic criteria. combinations were psychiatric interventions with phys-
Only 6 papers (18.8%) reported follow-up at 12 months or iotherapy and occupational therapy [24, 29], and surgical
more post-discharge from treatment. No papers reported treatment of a wound fistula followed by psychotherapy
an independent assessment of outcome or the use of a and both antidepressant and antipsychotic medication
standardised assessment scale. [47]. One case reported monitoring of the patient but no
specific therapy was mentioned [28]. Two cases reported
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Table 1. Case report characteristics and outcomes

Authors Confrontation technique Single/ Treatment description Treatment Treatment GIS

multiple location length score
therapies (0–7)

Spiro, 1968 [35] No confrontation (self-admission) Single Psychotherapy (not described) Inpatient then 6 months 5
outpatient
Fras and Coughlin, Repeated non-punitive Multiple Psychodynamic psychotherapy + anti- Mostly 2 years 2
1971 [44] confrontation psychotic + social worker involvement outpatient
Stone, 1977 [12] Accusative confrontation Multiple Psychotherapy + antidepressant Inpatient then 10 months 2
outpatient
Yassa, 1978 [41] Not reported Single Dynamic behavioural approach and Inpatient 3 years 2
supportive psychotherapy
Tucker et al., Carefully planned non-punitive Single Intensive psychotherapy Inpatient 11 months 2
1979 [31] confrontation.
Jamieson et al., Non-punitive confrontation Single Behavioural psychotherapy Outpatient 4 months 3
1979 [39]
Serafin et al., Accidental confrontation, with Multiple Supportive psychotherapy + Inpatient 2 months 3
1983 [47] unconscious motivation emphasised antidepressants + antipsychotics and
surgery to repair fistula
Klonoff et al., No confrontation (previous attempts Multiple Behavioural therapy delivered by 2 Outpatient 15 months 2
1983 [22] unsuccessful) psychologists + biofeedback and
relaxation techniques
Mayo and Haggerty, No confrontation (self-admission) Single Psychoanalytic psychotherapy Outpatient 16 months 4
1984 [34]

Batshaw et al. No confrontation (previous attempts Single Insight-oriented psychotherapy and Inpatient then 12 months 2
1985 [33] unsuccessful) behaviour modification outpatient

Gordon and Chrys, Non-punitive confrontation with Single Psychotherapy and stopping of Inpatient 5 weeks 4
1985 [25] supporting evidence medications
Earle and Folks, Non-punitive confrontation by Multiple Cognitive/supportive psychotherapy + Inpatient then 17 months 2
1986 [42] physician antidepressant nursing home
Kallen et al., No confrontation; aimed to educate Single Symptomatic treatment focussed on Inpatient then 5 months 3
1986 [40] patient through alternative method speech articulation and relaxation outpatient
techniques
Simmons et al., No confrontation Single Behavioural therapy Inpatient 2 months 4
1987 [64]
Johnson et al., Non-punitive confrontation Single Supportive psychotherapy Outpatient 6 months 2
1987 [30]

Schoenfeld et al., Angry, accusative confrontation by Single Psychotherapy focussed on life coping- Outpatient 4 years 1
1987 [32] psychiatrist skills

Savard et al., Non-punitive confrontation (had Single Psychotherapy (initially refused) Outpatient 2 months 7
1988 [49] been confronted previously)
Harrington et al., Non-punitive confrontation Single Monitoring of patient only Outpatient 12 months 3
1988 [28]

Schlesinger et al., Non-punitive confrontation Single Psychotherapy and behaviour Inpatient 5 months 3
1989 [23] modification

Higgins, No confrontation, establishment of Multiple Supportive authoritarian approach Outpatient (GP 8 years 1
1990 [48] alliance with GP adopted by GP + antidepressants and co-ordinated)
antipsychotics
Christensen and Non-punitive confrontation with Single Psychotherapy + withdrawal from Outpatient 2 months 2
Szlabowicz, diagnostic evidence antipsychotics.
1991 [27]
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Table 1 (continued)

Authors Confrontation technique Single/ Treatment description Treatment Treatment GIS

multiple location length score
therapies (0–7)

Parker, 1993 [36] No confrontation (self-admission) Single Psychotherapy Outpatient 8 months 3

Schwarz et al., Inexact interpretation; Multiple Behavioural psychotherapy + Patient choice 12 months 2
1993 [19] previous confrontation unsuccessful antidepressant + open access to hospital
bed
Arya, 1993 [45] No confrontation Multiple Psychiatric treatment (not specified) + Inpatient 2 years 2
analgesia for pain
Guziec et al., Non-punitive confrontation Single Supportive and insight-oriented Outpatient 12 months 3
1994 [26] psychotherapy
Feldman and Duval, Non-punitive confrontation with Multiple Psychological counselling + Outpatient 5 months 2
1997 [24] family present physiotherapy + OT

Kwan et al., Non-punitive confrontation Multiple CBT + physiotherapy + OT Inpatient 6 weeks 4

1997 [29]
Leonardou et al., No confrontation for fear of Single Psychoanalytic psychotherapy and Inpatient 2 months 1
2002 [37] damaging therapeutic relationship mileu therapy
Hirayama et al., Non-punitive confrontation Multiple Psychotherapy + antipsychotic and oral Outpatient 15 months 7
2003 [21] iron
Kubota et al., No confrontation for fear of Single Levothyroxine as day-patient to treat Outpatient 3 years 2
2003 [38] damaging therapeutic relationship feigned hypothyroidism
Yanik et al., No confrontation Multiple Psychiatric consultations + Outpatient 2 months 2
2004 [43] antidepressant
Oh et al., No confrontation Multiple Antidepressant + treatment for Inpatient 2 months 3
2005 [46] panniculitis

long-term, multi-faceted innovative approaches to treat- Confrontation

ment. In one, the patient was given open access to a hos- Analysis of the difference in mean GIS between pa-
pital bed for a year whilst receiving behavioural therapy tients who had been confronted (mean = 3.0, SD = 1.6)
and antidepressants [19]. In the other, a GP took a consis- and not confronted (mean = 2.6, SD = 1.2) (reported for
tent supportive yet authoritarian approach to a patient, n = 31) gave a non-significant result [t(29) = 0.72, p =
who was also prescribed antidepressant and antipsychot- 0.48].
ic medication [48].
Other Interventions
Quantitative Data Synthesis A single categorisation system for the different treat-
The frequencies of GIS scores across the reviewed pa- ments was difficult due to the heterogeneity of the treat-
pers are included in table 1. The mean GIS score was 2.81 ments described. Therefore, different aspects of the treat-
(SD 1.45) suggesting a small improvement at follow-up. ments have been analysed. An independent samples t test
Only seven (21.9%) had a score of 4 or higher representing (two tailed) comparing single or multiple treatments gave
no change or a worsening of condition. Two (6.3%) were a non-significant difference in mean GIS (single treat-
assigned a value of 7; in both of these cases, the patient ment mean = 2.8, SD = 1.4; multiple treatments mean =
died [18, 48] through a successful suicide. Three had a 2.8, SD = 1.5) t(30) = 0.14, p = 0.89. Analysis comparing
GIS score of 1 [32, 37, 49] but no common themes emerged the difference in mean outcome between the interven-
from these cases. tions which incorporated psychotherapy (n = 27, mean =
2.9, SD = 1.5) and interventions with no psychotherapy
(n = 5, mean = 2.4, SD = 0.9) gave a non-significant result,
t(30) = 0.69, p = 0.48.
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 An independent samples t test comparing mean dif- which may confound the evidence that they present. Two
ference in GIS outcome following treatment where psy- (15.4%) reported outcomes as assessed by another clinician
chiatric medication had been prescribed (n = 9, mean = [52, 55], and one paper used a standardised scale (the Ma-
2.7, SD = 1.7) compared with those where it had not (n = lan scale) to assess the change in patient condition [56].
23, mean = 2.9, SD = 1.4) gave a non-significant result,
t(30) = 0.35, p = 0.73. Management
There was a trend of greater improvement with longer Confrontation
treatment (!2 months mean = 3.0, SD = 1.8; 2–6 months Confrontation techniques were not always reported
mean = 3.0, SD = 1.0; 6–12 months mean = 2.6, SD = 0.6; and were generally poorly described but included non-
12–18 months mean = 3.8, SD = 2.4; 18–24 months punitive approaches [51, 58, 59], a mixture of confronta-
mean = 2.0, 124 months mean = 1.5, SD = 0.6), but this was tional and non-confrontational strategies [53, 60], and a
not significant [F(5, 26) = 1.17, p = 0.35]. The mean differ- double-binded face-saving technique [54, 61].
ence in GIS between treatment as an inpatient (n = 15,
mean = 2.9, SD = 1.4) or as an outpatient (n = 16, mean = Other Interventions
2.8, SD = 1.5) was non-significant [t(29) = 0.34, p = 0.73]. Table 2 details the wide range of treatments reported
across the case series with some papers describing differ-
ent techniques in different groups of patients. It was not
Case Series always clear how the outcomes related to individual treat-
ments across the case series [52]. The larger case series
Study Characteristics demonstrated that only a small proportion of patients
The characteristics of the 13 case series are presented who were identified with FD engaged in treatment [9, 53].
in table 2, they varied in size from 2 to 93 patients. A total Many reported techniques were multidisciplinary in na-
of 284 patients were reported; 193 (68.0%) were female, ture [56, 61]. Plassmann [56] described a long-term psy-
67 (23.6%) were male and in 24 (8.4%) gender was not chotherapeutic approach which had its foundations in
documented. Patient ages ranged from 14 to 70 years the treatment of borderline personality disorder; patients
(mean = 33.7, SD = 7.6). The duration of factitious illness initially received inpatient clinical psychotherapy fol-
was only reported for 218 patients, the mean for these was lowed by outpatient psychotherapy which had lasted up
6.31 (SD = 9.4) years. Most papers did not distinguish be- to 5 years at the time of reporting.
tween FD and Munchausen’s syndrome; only 7 (2.5%) pa-
tients were reported as having a diagnosis of Munchau- Quantitative Data Synthesis
sen’s syndrome. Seven papers (53.8%) made reference to A range of outcomes was seen, the GIS scores are in-
co-morbid psychiatric diagnoses; from those papers (195 cluded in table 2. The median value for the GIS was 0 (not
patients), 113 patients (57.9%) were reported as having co- reported) as the majority of patients (60.6%) were not fol-
morbid psychiatric disorders (Axis I or Axis II). Other lowed up. Four patients (3.6%) were reported deceased. In
papers referred to co-morbidities with depression, schizo- the 7 papers where follow-up was reported for over 80%
phrenia and personality disorders. of patients (a total of 40 patients), the median GIS was 2
(improvement). Further analysis was not possible due to
Quality Assessment the wide variation in reporting between the case series.
There was much variation in the quality of reporting.
Only six (46.2%) described a consecutive series of patients
[9, 50–54], the other seven (53.8%) described a group of Discussion
selected patients which had been treated by the authors.
Five (38.5%) referred to diagnostic criteria. Patient demo- Principal Findings
graphics and histories, the management techniques used There is an absence of robust research to determine
and the duration of follow-up were reported with wide the effectiveness of any management technique for FD.
variation in detail. Follow-up information was only re- The best level of evidence was level III [62] indicating an
ported for 112 (39.4%) patients. The duration of follow-up absence of randomised controlled trials or controlled tri-
was only reported in 9 papers (56 patients) and ranged als. The consecutive case series demonstrated that many
from 1 month to 15 years (mean = 45 months, SD = 22.9). FD sufferers are not engaged in treatment and are lost to
Seven (53.8%) did not follow-up all patients in the series, follow-up.
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Table 2. Case series characteristics and outcomes

Authors Cases Series selection Confrontation technique Treatment descriptions Outcome descriptions GIS score1

O’Shea and 2 Patients Not reported Informal psychotherapy with Improvement (n = 2) (2–3)
McGennis, behavioural approach focussed
1982 [57] on social skills
Maurice- 14 Cases of simulated Conveyed that nature of Amyteal abreaction (n = 6), Improvement (n = 2), 0 (0–5)
Williams and paralysis, 10 years behaviour was understood hypnosis (n = 2), psychotherapy reoccurrence of symptoms
Marsh, neurosurgical (n = 1) pharmacotherapy (n = 2) (n = 3), no follow-up
1985 [52] admissions. placebo and reassurance (n = 7) (n = 9)
Grunberger 10 Cases of factitious Non-punitive with Not described in detail, Dead (n = 2), much 3 (0–7)
et al., 1988 [51] hypoglycaemia in persistence until patient multidisciplinary approach improved (n = 3),
13 year period acknowledgement minimal improvement
(n = 3), lost to follow-up
(n = 1)
Solyom and 2 Patients None; informed that they CBT and physiotherapy Good improvement (1–3)
Solyom, would be treated (faradic massage) (n = 2)
1990 [55] accordingly
Sutherland and 10 Cases of FD referred 9 confronted; exact Outpatient psychotherapy (NOS) No reoccurrence of 0 (0–7)
Rodin, to consultation- technique unknown (n = 2) symptoms (n = 2), death
1990 [50] liaison service, (n = 1), no follow-up
3 years (n = 7)
Spivak et al., 2 Patients Non-punitive Psychotherapy focussing on Physical improvement, 3 (3–3)
1994 [58] emotions and experiences (n = 2), still psychosocial
ECT (n = 1), antipsychotic problems
medication (n = 1) (n = 2)
Teasell and 3 Patients Double-binded, face-saving Strategic-behavioural Rapid good improve- 2 (1–3)
Shapiro, interpretation intervention involving ment (n = 2), partial
1994 [61] psychotherapy, physiotherapy, improvement (n = 1)
OT and nursing with specific
goals for patients
Plassmann, 24 Patients Not described; flexible, Inpatient clinical psychotherapy Variable improvement 0 (0–4)
1994 [56] non-aggressive approach then long-term psychotherapy (n = 12), refusal of therapy
(n = 12)
Freyberger 70 Hospitalised FD Accusative (n = 13), indirect Psychodynamic psychotherapy Improvement (n = 9), 0 (0–4)
et al., 1994 [53] patients, 9 years seen (n = 58), no confrontation inpatient/outpatient (n = 17) no change/worse (n = 3),
by psychosomatic (n = 12) continuing treatment
consultants (n = 1)
Shapiro and 24 Consecutively Double-binded, face-saving Strategic-behavioural Complete improvement 0 (0–4)
Teasell, admitted patients interpretation intervention (physiotherapy); (n = 15), significant
1997 [54] rehab ward, psychological counselling in some improvement (n = 3),
9 years patients minimal improvement
(n = 2)
Al-Qattan, 28 All patients FD Various techniques; Psychotherapy, pharmacotherapy Successful treatment 0 (0–5)
2001 [60] upper limb, 6 years confrontational and non- or no treatment beyond (not qualified) (n = 11)
confrontational confrontation but numbers not
reported
de Fontaine 2 Patients Non-punitive, by surgeon Plastic surgery (n = 2); Wounds healed (n = 2) (2–4)
et al., 2001 [59] and psychiatrist additionally psychotherapy
(n = 1), monitoring in pain clinic
(n = 1)
Krahn et al., 93 Computer list, FD Confrontation (technique Psychiatric treatment (n = 19), Ongoing care (n = 29), 0 (0–7)
2003 [9] diagnosis, 20 years not specified) (n = 71); ongoing care (n = 29) known deceased (n = 2)
psychiatric consultation unknown (n = 62)
(n = 80)
1
Values are given as median (range).
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Table 3. FD management recommendations logical therapy in FD seems likely to be in treatment of
co-morbid psychiatric disorder [42, 48]. The reported
a A comprehensive psychiatric assessment should be completed successes of both confrontational [32] and non-confron-
to identify comorbid psychiatric illnesses, and suicide risk
b One person should have primary therapeutic responsibility tational approaches [37, 48] along with treatments that
c The multidisciplinary team should be involved with all being focused on the presenting symptoms [38, 49] suggest that
aware of the psychiatric assessment, risk assessment and treat- various strategies may be helpful but do not really help
ment plan the clinician to select a particular management plan.
d If confrontation takes place, this should be non-punitive and In two of the four case reports where a negative out-
supportive in nature
e A treatment plan should be individualised to the patient come was documented, suicide had occurred. Many au-
f Comorbid illnesses should be treated appropriately thors have highlighted the unfavourable prognosis and
g Long-term therapy or support should be provided to aid recov- risk of suicide in patients with FD [65]. Clinicians should
ery and transition back into a ‘normal’ life remain vigilant to this and routinely assess suicide risk in
suspected FD sufferers. The results of this review do not
allow for the development of an evidence-based manage-
ment pathway for FD but do allow some tentative man-
agement recommendations (table 3) to be made that re-
Strengths and Weaknesses of the Study quire further evaluation.
This review was conducted systematically using com-
prehensive searches with predetermined inclusion and Future Research
exclusion criteria, and standardised assessment tools. There remain many unanswered questions. In order
Only limited quantitative data synthesis was possible. A to improve the current evidence base, we believe that a
formal meta-analysis could not be performed, but this network of clinicians with an interest in this area should
study has provided what is likely to be an accurate picture be established, a central reporting register developed and
of the current evidence base and should form the basis for standardised patient data routinely collected and anal-
the development of a strategy to improve knowledge in ysed. Such systems are already in place for other rare con-
this area. Despite the absence of evidence for effective- ditions such as Huntington’s disease [66] and CJD [67]
ness of the management techniques considered, the risk with the emergence of better quality research than could
of bias towards positive outcomes is very high with au- have been produced without their development. Rothwell
thors being more likely to present their successes than [68] recently highlighted the general neglect of good ob-
their failures and a similar risk of publication bias. The servational research within medical academia leading to
very variable lengths of follow-up also compromise inter- a failing in effective clinical practice. This is true for FD
pretation. The paucity of good research in this area is and if conducted would form a foundation for the phased
likely to have been contributed to by FD being a relative- development and testing of complex interventions that
ly rare and difficult to detect condition that is notori- have the potential to be effective for FD in the future.
ously difficult to manage with engagement often being a
major problem [63].

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