Schizophrenia Bulletin vol. 35 no. 5 pp.

10121021, 2009
doi:10.1093/schbul/sbn040
Advance Access publication on May 20, 2008

Major Self-mutilation in the First Episode of Psychosis

Matthew Large1,2, Nick Babidge3, Doug Andrews4,5,

Philip Storey6, and Olav Nielssen2,7
2

Major self-mutilation (MSM) is a rare but catastrophic

complication of severe mental illness. Most people who inflict
MSM have a psychotic disorder, usually a schizophrenia
spectrum psychosis. It is not known when in the course of
psychotic illness, MSM is most likely to occur. In this study,
the proportion of patients in first episode of psychosis (FEP)
was assessed using the results of a systematic review of published case reports. Histories of patients who had removed an
eye or a testicle, severed their penis, or amputated a portion
of a limb and were diagnosed with a schizophrenia spectrum
psychosis were included. A psychotic illness was documented
in 143 of 189 cases (75.6%) of MSM, of whom 119 of 143
(83.2%) were diagnosed with a schizophrenia spectrum psychosis. The treatment status of a schizophrenia spectrum
psychosis could be ascertained in 101 of the case reports,
of which 54 were in the FEP (53.5%, 95% confidence interval
43.7%63.2%). Patients who inflict MSM in FEP exhibited
similar symptoms to those who inflict MSM later in their
illness. Acute psychosis, in particular first-episode schizophrenia, appears to be the major cause of MSM. Although
MSM is extremely uncommon, earlier treatment of psychotic illness may reduce the incidence of MSM.
Key words: self-mutilation/schizophrenia/first-episode
psychosis

Introduction
Self-mutilation has been defined as the direct and deliberate self-destruction of a part of a persons own body
1

To whom correspondence should be addressed; Private Practice,

Paddington, PO Box 110, Double Bay, NSW 1360, New South
Wales, Australia; e-mail: mmbl@bigpond.com.

The Author 2008. Published by Oxford University Press on behalf of the Maryland Psychiatric Research Center. All rights reserved.
For permissions, please email: journals.permissions@oxfordjournals.org.

1012

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Private Practice, Paddington, New South Wales, Australia;

Sutherland Hospital, Kingsway, Caringbah 2229, New South
Wales, Australia; 4Rural Clinical School, Faculty of Medicine
University of New South Wales, Australia; 5North Coast Area
Health Service, New South Wales, Australia; 6Rotary Ambassadorial Scholar, Evanston, IL; 7Clinical Research Unit for Anxiety
Disorders, School of Psychiatry, UNSW, Darlinghurst, New South
Wales, Australia
3

without the intention of suicide.1 Minor self-mutilation

is quite common, does not usually cause significant disability, and may even be part of recognized cultural practices. In contrast, major self-mutilation (MSM) is rare,
usually only occurs in association with serious mental illness and often results in permanent loss of an organ or its
function.2 The 3 main forms of MSM are ocular, genital,
and limb mutilation. Patients who have removed an eye
or cut off a limb are almost always psychotic, as are three
quarters of patients who severely injure their genitals.3
The published accounts of MSM are almost all either
single-case histories or small case series, from which it is
difficult to make valid causal inferences. Even quite recent publications sometimes explain MSM in terms of
the patients reaction to passages in religious texts or unconscious sexual conflicts. The few authors who have
reviewed more than a small number of cases have attributed MSM to the direct effects of psychotic illness.37 In
a recent review of self-inflicted eye injures, we found that
almost all cases of serious self-inflicted eye injuries result
from schizophrenia spectrum psychosis and half the injuries that caused permanent loss of vision occurred in the
first episode of psychosis (FEP).7
Studies that have examined prior treatment at the time
of another uncommon and catastrophic complication of
psychosis, homicide, generally report that between 30%
and 50% occur during the FEP,8 usually in response to
frightening symptoms.9 These studies also provide sufficient information to demonstrate that the risk of homicide in FEP may be as much as 20 times the subsequent
annual risk after treatment.10 The hypothesis for this
study was that MSM is similar to homicide in psychosis
and may also be more likely to occur during the FEP.
We aimed to use published case histories to estimate
what proportion of the MSM that is associated with
schizophrenia spectrum psychosis occurs in the FEP.
Our hypothesis was that there may be a greater proportion of MSM in the FEP than would be expected by
chance, assuming that the risk of MSM is equal in
FEP and previously treated psychosis (PTP). We included case reports of patients who had a diagnosis of
schizophrenia spectrum psychosis and who had completely removed an eye or a testicle or severed their penis
or a limb. The findings were used to examine the extent to
which psychosis meets epidemiological criteria for causation of MSM.

Major Self-mutilation

Methods
Search Terms
The [Medline], [Embase], and [Psychlit] databases were
searched from 1960 to February 2008 using the terms psychosis OR schizophrenia OR mental disorder AND self
mutilation OR self-enucleation OR enucleation OR selfinflicted eye injuries OR eye injuries OR oedipism OR
castration OR emasculation OR orchidectomy OR penile
amputation OR penile injury OR amputation OR limb
amputation OR mutilation OR self-mutilation OR deliberate self-harm. The references of the located articles
were hand searched for further cases.

We included published case histories of patients with

a schizophrenia-related diagnosis in which prior treatment status could be ascertained and who had completely
removed (1) an eye (2) or a testicle or severed (3) their
penis (proximal to the glans) or (4) a limb (proximal
to the hand or foot). We also collected data on cases
that met our severity criteria for MSM but were not diagnosed with a schizophrenia spectrum disorder or did
not provide sufficient information to establish prior
treatment status. The latter 2 groups were excluded
from the main analysis. Cases of patients with mutilation
that did not meet our injury severity criteria or removed
other body parts were not considered. Case histories of
people who required an amputation or enucleation for
medical purposes after self-injury and cases of fatal
self-amputation were also excluded.
A narrow definition of MSM was used to ensure that
all the cases were of similar severity. There was great variation in the severity of other forms of mutilation, and in
some cases, the severity was difficult to assess. Moreover,
many cases of limb, neck, chest, abdomen, and penetrating orbital injures occurred in failed suicide attempts. The
other forms of self-injury inflicted by the patients included in this study, in addition to their MSM, were
broadly defined as any self-inflicted physical injury
that did not meet our definition of MSM.

Statistics
Definitions of Psychosis and Treatment Status
The diagnoses used by the authors of the case histories
were accepted at face value, and no attempt was made
to standardize the diagnostic criteria. Psychosis was defined as any diagnosis characterized by hallucinations,
delusions, or thought disorder and included psychosis
secondary to medical conditions or substance use, affective psychosis, and schizophrenia spectrum psychosis.
The category of schizophrenia spectrum psychosis
included schizophrenia, delusional disorder, schizoaffective disorder, schizophreniform disorder, brief psychotic
disorder, or psychosis not otherwise specified (NOS).
Cases diagnosed with affective psychosis or psychosis

The mean and 95% confidence intervals (CIs) for the proportion of case histories documenting each characteristic
were calculated for the whole group. The secondary hypothesis of possible clinical difference between FEP and
PTP groups was examined with a chi-square test for categorical variables or a Fishers exact test if N was less
than 5 in any cell. A 2-tailed Students t-test was used
to compare age in FEP and PTP groups.
Female cases were excluded for penis amputation and
castration. A Bonferroni correction was calculated to be
0.0024 for the 21 comparisons that were independent of
treatment status. Age and current treatment with antipsychotics were not regarded as independent of treatment
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Definition of MSM

secondary to substance abuse were not included in the

main analysis, even if it appeared that the patient had persisting hallucinations or delusional beliefs.
We classified a case as FEP if the case history stated
that the patients had never received antipsychotic treatment or if they had only recently commenced treatment
for the first time, had not had a remission from symptoms
and were still in hospital. Few of the case histories provided sufficient information to use the preferred definition of the FEP, which is the period from the onset of
psychotic symptoms until the patient had a remission
after receiving an adequate trial of antipsychotic treatment. Hence, patients were also classified as being in
the FEP if an otherwise comprehensive history did not
report previous treatment or if the case history clearly
stated that the diagnosis of psychosis was not made until
after the injury.
Few case histories documented the dosages and duration of prior treatment. Patients were classified as having
PTP if the case history reported that the patient had been
treated before, if the diagnosis had been made before the
injury, or if the patient was described in the history as
having chronic schizophrenia. Patients who had received
prior treatment were included in the PTP group even if
they had not been adherent to antipsychotic medication
for many years. Cases in which the treatment status was
not described or could not be determined and borderline
cases of patients who had received 3 or 4 weeks of antipsychotic treatment were excluded from the analysis of
treatment status.
We relied on the information provided by the authors
for almost all the data points, including the presence of
particular symptoms. If a paper failed to mention a specific psychotic symptom, the symptom was rated as not
having been present. In cases that provided little information beyond the diagnosis and previous treatment status,
absent data points were left blank. Some inferences about
psychomotor agitation and the patients indifference to
the injury were made on the basis of the descriptions
of the patients behavior.

M. Large et al.

status. SPSS version 15.0 was used for all statistical

analysis.
Estimation of the Incidence of MSM

Results
Results of the Searches and Case Selection
One hundred and ninety six publications were examined
after the exclusion of case histories describing superficial
mutilation, finger amputation, cases of MSM of the
tongue, breasts, or nose and 4 histories of fatal amputation. These publications contained a total of 305 case histories of probable MSM, including 189 case histories in
which there was complete amputation or removal of an
organ. The case histories were then coded according to
whether (1) injuries met inclusion criteria, (2) the psychiatric diagnosis, and (3) if there was an adequate account
of previous treatment.
First, case reports of patients who had self-inflicted
injuries that were not reported in association with a diagnosed psychotic illness were excluded. This included
39 cases of less severe eye or genital injuries and 44 cases
of major genital injury. The major genital injury cases included 25 patients who had amputated one or both testicles, 13 patients who had amputated their penis, and
6 cases of complete genital amputation. Seventeen of
the 44 genital mutilation cases were reported to have gender identity disorder, 6 alcohol dependence, 12 other nonpsychotic disorders, and there were 9 cases in specialist
surgical journals that only reported the presence of psychiatric disorder without providing any further details.
There were several cases of men from Asian backgrounds
who believed that penile amputation would result in
death and who were regarded by the authors to be suicidal but not necessarily psychotic.12 There were 2 cases
of upper limb self-amputation in patients who were not
diagnosed with a psychotic illness but there were no case
histories of nonpsychotic patients who had performed an
enucleation or a lower limb amputation. A total of
42 publications were excluded on this basis.
1014

Reliability of Data Collection

M.L. and N.B. independently examined the cases for inclusion or exclusion. There were two disagreements about the
inclusion of cases, 1 due to accidental double counting by
one author and the second as a result of multiple publications about the same patient.
M.L. and N.B. also independently collected clinical
data using spreadsheets of data points versus case histories. There were no disagreements about the injuries, the
psychiatric diagnosis, demographic details, or the setting
of the injury. There was 1 disagreement about the rating
of previous treatment in a history that was subsequently
excluded because of the uncertainty on this point. Disagreement about 5% of other data points was resolved
by a further review of the cases.
Results of Examination of the Cases
We located 189 cases of patients who had removed an eye
or testicle or had severed their penis or limb. A psychotic
illness was diagnosed in 143 of 180 (79.4%) cases of MSM
in which a specific psychiatric diagnosis was mentioned.
Of these, 119 (83.2%) were diagnosed with a schizophrenia spectrum psychosis. Treatment status could be
ascertained in 101 of 119 (84.9%) schizophrenia spectrum

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An attempt to estimate the incidence of MSM and proportion of cases that were the subject of a published
report was made using acquaintance chains, in a method
that was loosely adapted from that described by Stanley
Milgram.11 The authors initially contacted 8 senior clinicians in key positions in mental health, forensic psychiatry, rehabilitation, and hospital-based ophthalmic
services in New South Wales (NSW), a state of Australia
with a current population of just under 7 million. Each
clinician was asked about their knowledge of patients
with MSM and was also asked to nominate other clinicians who may know of cases. The nominated clinicians
were then contacted in a sequence that continued until
no new names were suggested.

Second, cases of patients with psychosis whose selfinflicted injuries did not meet the inclusion criteria
were excluded. These consisted of 53 case histories of penetrating, superficial, and blunt force self-inflicted injury
to the eye, 19 reports of genital self-injury that fell short
of amputation, 2 reports of genital mutilation by females,
and 3 reports of incomplete upper limb amputation by
psychotic patients. As a result, a further 38 publications
were excluded.
Third, 24 case histories of patients with a nonschizophrenia spectrum psychosis who had inflicted MSM
were excluded. These consisted of 5 self-enucleating
patients with psychosis that were secondary to various
medical conditions, 12 cases in which the diagnosis
was affective psychosis, and 7 cases of psychosis reported
to be secondary to substance use. Exclusion of these cases
resulted in the exclusion of a further 22 publications.
Finally, we excluded case histories of 18 patients with
a schizophrenia spectrum psychosis who had amputated
or removed an eye, limb, or genital part but did not provide sufficient information to establish if the patient had
been previously treated. These comprised 18 cases in
15 publications and included 2 patients who inflicted
MSM after several weeks of hospital treatment with antipsychotic medication but who remained unwell.
One hundred and one casesof MSM from 79 publications
were included.3,1390 There were 42 cases that met our criteria for MSM and psychosis but did not document the treatment status or the presence of a schizophrenia spectrum
psychosis and were excluded from the main analysis.

Major Self-mutilation

Table 1. Characteristics of Patients With Psychosis and Self-enucleation, Limb, or Genital Amputation
Schizophrenia Spectrum Psychosis With
Documented Treatment Status

Excluded
Casesa
95% CI

N = 42

Age (mean)
Age range

32.7
(1667)

30.834.7

34.0
(1960)

Male
FEP
Enucleation
Limb amputation
Penis amputation
Castration
Other self-injury
On antipsychotics
In hospital, jail, or care facility

Frequency
90
54
40
9
40/90
30/90
27
20
21

%
89.1
53.5
39.6
8.9
44.4
33.3
26.7
19.8
20.8

83.095.2
43.763.2
30.049.2
3.314.5
34.754.2
24.142.6
18.135.4
12.027.6
12.828.7

%
90.2
89.5b
37.5
9.5
52.6
18.4
31.6
5.7
5.7

Cases with more complete data

Any delusion
Delusion about the amputated organ
Religious delusions
Reference to a religious text
Command hallucinations to remove an organ
Visual hallucinations
Thought disorder
Behavior indicating indifference to injury
Psychomotor agitation
Habitual substance abuse
Intoxicated at the time
Previous stimulant or hallucinogen use
Violence toward others
Suicidal thoughts or acts

N = 89
82
62
61
30
28
10
41
44
35
20
7
20
10
18

92.1
69.7
68.5
33.7
31.5
11.2
46.1
49.4
39.3
22.5
7.9
22.5
11.2
20.2

86.597.8
60.179.3
58.878.3
23.843.6
21.741.2
4.617.9
35.656.5
38.959.9
29.149.6
13.731.2
2.213.5
13.731.2
4.617.9
11.828.7

N = 24
100
62.5
62.5
28.0
29.2
10.7
24.0
41.7
33.3
36.1
16.7
33.3
8.0
32.1

5 cases of psychosis secondary to a medical condition, 12 cases had affective psychosis, 7 cases of psychosis secondary to substance
abuse, and in 18 cases a schizophrenia spectrum psychosis with undocumented treatment status.
b
Proportion in FEP based on an N of 19 cases only.

psychosis cases, of which 54 were classified as FEP

(53.5%, 95% CI = 43.7%63.2%).
Table 1 is a summary of the characteristics of the included and excluded cases.
The sample has a predominance of younger men, most
of whom were in the FEP. Most of the cases were of genital amputation or enucleation, with comparatively few
cases of limb amputation. The excluded cases were similar in characteristics to the included cases. However,
cases that were excluded on the grounds of diagnosis
were significantly more likely to be in FEP (17 of 19 in
FEP, chi-square = 8.582, P = 0.003)
Eighty nine of 101 cases were diagnosed with schizophrenia, 4 schizophreniform psychosis, 2 schizoaffective disorder, 2 with a brief psychotic disorder, 1 with delusional
disorder, and 3 were considered to have psychosis NOS.
Eighty-seven of 101 histories made a specific mention
of at least 1 psychotic symptom and 82 described the presence of a delusional belief. The most common delusions
involved a false belief about the amputated organ including that the organ was evil (43%), that the organ had a spe-

cial, usually threatening supernatural powers such as the

ability to spread evil (28%), or that it needed to be sacrificed in order to save the patient or others (20%).
The FEP and PTP groups and the sample of excluded
patients had high proportion of patients with religious
delusions, disorganized thinking, and behavior, and
many patients were indifferent to their injuries (Tables
1 and 2). A third of PTP patients were reported to be taking antipsychotic medication at the time of MSM.
Table 2 compares those with FEP to those with PTP.
Patients in their FEP were, as expected, younger and less
likely to be taking antipsychotic medication. Habitual
substance use was more common in the FEP group,
and more patients in the PTP group reported command
hallucinations, but neither finding was significant after
a Bonferroni correction. It may be that FEP patients
were less able to identify voices as hallucinations.
Excluded Cases
The psychosis cases that were excluded because their eye
injuries were not severe enough to meet our inclusion
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N = 101

M. Large et al.

Table 2. Comparison of Previously Treated Psychosis and First-Episode Psychosis Patients

PTP

FEP

54
29.7
27.432.0
1653
50
19
6
22 (50)c
16 (50)c
15
4
12

Psychotic symptoms, substance use, and evidence of dangerousness, n = 89

N
39
Any delusion
37
Delusion about the amputated organ
29
Religious delusions
29
Referred to religious textd
14
Command hallucinations to remove an organ
17
Visual hallucinations
5
Thought disorder
18
Behavior indicating indifference to injury
19
Psychomotor agitation
15
Habitual substance abuse
5
Intoxicated at the time
2
Previous stimulant or hallucinogen use
8
Violence toward others
5
Suicidal thoughts or acts
7

50
45
33
32
16
11
5
23
25
20
15
5
12
5
11

Chi-Square

0.006a

99
1
1
1
1
1
1
1
1

1
1
1
1
1
1
1
1
1
1
1
1
1
1

0.947
0.009
0.090
0.065
0.144

0.724
1.090
0.149
4.736
0.175
0.000
0.014
0.022
3.712
0.153
0.175
0.223

0.338b
0.330
0.498b
0.924
0.764
0.799
0.001b
0.704

0.461b
0.395
0.296
0.700
0.029
0.676
0.989
0.904
0.883
0.054
0.461b
0.696
0.676
0.637

Unpaired 2-tailed t-test, t = 2.8.

Fishers exact test.
c
Reduced n in brackets.
d
No patient was reported to have mentioned any religious text other than the gospel of Matthew.
b

criteria are reported elsewhere.7 A lower proportion of

the patients with schizophrenia spectrum psychosis and
less serious genital or limb injuries were thought to be
in FEP, but the histories were less detailed than the
case histories of more severe MSM.
There were 42 cases that met inclusion criteria for
MSM, but were excluded because they were not diagnosed with a schizophrenia spectrum psychosis (24
cases), because treatment status was not documented
(18 cases) or for both of these reasons (5 cases). The nonschizophrenia spectrum psychosis cases included 5 cases
of psychosis secondary to a medical condition (including
systemic lupus erythematosus, hypothyroidism, and epilepsy), 12 cases diagnosed with an affective psychosis,
and 7 cases of psychosis secondary to substance abuse.
Overall, the excluded patients had symptoms that were
similar to the included cases. Patients reported to have
affective psychosis were just as likely to have a bizarre
organ-specific delusion but were more likely to have amputated part of the genitals, were older, with a mean age
of 40 years, and 9 of 12 were considered to have pathological guilt.
1016

Estimate of the Number of Cases of MSM in NSW

Between 1990 and 2007
The acquaintance chain method enabled us to locate
long-serving clinicians in every mental health service in
NSW after only 3 steps, and after 5 steps, no new doctors
were suggested. A total of 38 clinicians or administrators
were contacted directly by telephone or email. Many of
the clinicians we contacted spoke to colleagues before
responding, but no new cases were identified from secondary contacts.
We were able to obtain corroborated accounts of 6
enucleations and 3 completed upper limb amputations
in NSW between 1990 and 2007. Cases of genital amputation were not remembered in as much detail as the ocular and limb cases, but there were at least 11 cases, all
but one of which was in association with a psychotic illness. Of the 13 cases in which treatment status was clearly
remembered, 6 were thought to have occurred in the FEP
and several occurred soon after the first admission to hospital. The clinicians also described a number of severe
cases that fell short of amputation, including 3 almost

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Demographics and circumstances of the injury, n = 101

N
47
Age
36.2
95% confidence interval
33.239.3
Range
2167
Male
40
Enucleation
21
Limb amputation
3
Penis amputation
18 (40)c
Castration
14 (40)c
Other self-injury
12
On antipsychotics
16
In hospital, jail, or care facility
9

df

Major Self-mutilation

complete hand amputations, 4 self-inflicted eye injuries

that resulted in blindness, and a self-amputation of
a breast. A number of less severe genital cases, several
failed enucleations, a case of psychotically motivated
nonsuicidal self-evisceration, 3 additional cases of penetrating injury to the brain via the orbit or nose, and
numerous cases of finger amputation were excluded. In
total, we found 28 cases of MSM resulting in significant
disability in NSW in the 17 years, including 20 cases that
would have met the inclusion criteria for our study.
Discussion

Possible Sources of Error and Bias

The predominance of ocular and genital MSM in published cases may be because these injuries are easier to
perform, that the genitals and the eyes have special psychological significance, or because of a publication bias
as these injuries may be seen as being of greater interest.
Some of the cases of penile and limb amputation were
reported because the surgical replantation methods
and some ocular cases concerned ophthalmic complications. Hence, although we found a similar ratio of injury
types in our informal survey in NSW to the ratio in
the published cases, the ratio of MSM injuries in publications may not accurately reflect their incidence in the
community.
A more important consideration is whether diagnostic
error, the methods used to define FEP, or publication
bias influenced the proportion of cases that we counted
as FEP.
The number of FEP cases may have been overestimated if the authors of some case histories were unaware
of previous treatment. However, 44 of 54 (81%) of the
first-episode case histories included a psychiatric and
personal history, and the remaining 10 histories clearly
stated that the patient had not been diagnosed or treated
prior to the injury. On the other hand, the proportion of
PTP patients may have been overestimated because
8 cases were classified as PTP on the basis of a diagnosis
of chronic schizophrenia, and 3 further patients were
classified as PTP because of a preexisting diagnosis of

The Relative Risk of MSM in FEP and PTP

We found that half the cases of MSM and a schizophrenia
spectrum psychosis had never received antipsychotic
treatment and more than half were in the FEP. This is
a notable finding because schizophrenia is a lifelong condition that usually begins in early adult life.
The majority of published studies of the incidence of
schizophrenia report that between 0.01% and 0.03% of
the population will develop schizophrenia in any given
year and about 0.5% have a preexisting schizophrenic illness.96 If one assumes an incidence of 0.02% per annum
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Based on reports of 28 cases in NSW between 1990 and

2007, we estimated the incidence of very extreme severe
forms of MSM to be at least 1 case per 4 million of population per year during that period. The true figure may
have been greater because it is likely that we did not have
information about every case, and we did not include
cases of less severe MSM. Only 2 of these cases were
the subject of a published case report.19 Hence, despite
its rarity and confronting nature, published case reports
of MSM are only a limited and semirandom sample of
MSM events, and the results of this review of published
cases should be treated with some caution, because biases
and some types of error are possible.

schizophrenia. Previous treatment was assumed but

not documented in these 11 cases.
We may also have underestimated the proportion of
patients in FEP if some cases were wrongly classified
as nonpsychotic. Firstly, there were men who castrated
themselves for religious reasons,91,92 a teenage student
who amputated his penis so he could concentrate on
his studies,93 a man who castrated himself to treat alopecia, although his doctors thought he did not suffer from
this condition,94 and a traumatized woman with gustatory hallucinations who amputated her hand because
she believed it had done bad things.95 Second, all
but one of the 7 cases of drug-induced psychosis with
known treatment status was in a FEP and several had
persistent symptoms, which suggested the correct diagnosis was schizophrenia. Third, few of patients who were
given a diagnosis of affective psychosis had received prior
treatment but many had bizarre delusions that are more
typical of a schizophrenia spectrum psychosis. If the inclusion criteria had included all psychotic illnesses, 71 of
120 (59.2%) cases would have been classified as FEP.
If the 18 case histories of schizophrenia spectrum psychosis that did not document treatment status were
assumed to have received treatment, 54 of 119 (45.4%)
of the cases could have been classified as FEP. However,
some of these cases were almost certainly in the FEP because 6 patients were in their teens or early 20s and as
a group they had a mean age that was slightly younger
than the mean age of the included cases.
The number of FEP cases did not appear to have been
overestimated by reporting or publication bias. Cases in
published series of 2 or more patients, which it can be
assumed included all the cases known to the author,
had a similar chance of being in the FEP (29 of 56,
52%) than cases reported as a single case history (25 of
45, 55%).
Although patients who enucleate both eyes in FEP
cannot do so later in their illness, it is unlikely that the
proportion of patients in FEP was increased by a survivor
effect because patients with MSM make up a tiny proportion of all cases of schizophrenia. Furthermore, only
a few FEP patients, including those who underwent
limb, penile, or testicular replantation surgery, inflicted
further MSM.

M. Large et al.

Psychosis and Causation of MSM

The authors of some case histories have attributed the
MSM to unconscious sexual conflicts26,32,55 or knowledge of the Bible.75 More recently, it has been suggested
that limb self-amputation may be the result of a nonpsychotic disorder provisionally named body integrity identity disorder,97 and there is also a recent series of patients
with nonpsychotic self-emasculation.98 These 2 series
must be viewed with some caution because they are based
on contact via the Internet and telephone-based assessment rather than personal interview. However, they do
raise the question of how the causation of MSM should
be considered.
We used the criteria for epidemiological causation as
set down by Austin Bradford Hill to guide a consideration
of whether psychosis can be considered to be a cause of
MSM.99 Hills criteria (in italics) and our conclusions are
Strength of association: There is a very strong association
between psychosis and MSM because at least 143 of 180
(79.4%) patients (excluding 9 cases with no with no mention of the persons mental state) had a psychotic illness,
despite chronic psychotic illness affecting less than 1% of
the population. Consistency of association: Published
reports of MSM in association with psychosis have
come from all parts of the world with increasing frequency since the first case was reported in the 19th century. Specificity of association: MSM is specifically
reported in association with schizophrenia spectrum psychosis. Reports of MSM in association with substance
abuse and affective psychosis are rare, but the characteristics of these patients are similar to those with a schizophrenia spectrum psychosis. Temporality: We only found
2 cases in which MSM may have preceded over psychosis,28,62 although it is also possible that some of the cases
in which psychosis was not diagnosed were in the prodromal phase of psychotic illness. Biological gradient: The
cases examined in this study had severe psychotic illness
with numerous acute symptoms. Many were so ill that
they were indifferent to pain and to the loss of their
1018

organs. Most had religious delusions, which may be

a marker of the severity of psychosis.100 Plausibility: A
reaction to a bizarre delusion leading the patient to
remove an organ that they believed was threatening or
an experience of hallucinations directing them to remove
their organs are plausible links between the psychosis and
MSM. Coherence: The presence of MSM among differing
types of psychosis suggests a coherent link between psychosis and MSM. Experiment: The lower risk of MSM in
treated psychosis represents what Hill described as
a semiexperiment in which a modification of the cause
reduces the effect. Analogy: The risk in first-episode psychosis, the threatening psychotic symptoms,101 and a predominance of male patients with schizophrenia with an
average age in the early 30s are features that MSM
patients have in common with psychotic patients who
commit homicide.
Reasons for the Decline in MSM After Treatment
Although psychosis, particularly first-episode schizophrenia, seems to meet epidemiological criteria for causation of MSM, the reason for the decline in MSM after
a period of initial treatment is uncertain.
The lower incidence of MSM in PTP patients may be
partly explained by ongoing treatment with antipsychotic
medication. However, this is unlikely to be the sole explanation because adherence to medication by patients with
schizophrenia is known to be poor102 and about a third of
the PTP patients in this review were reported to be adherent to treatment.
The most obvious explanation is that once patients
have experienced a remission and have received a medical
explanation of their symptoms, they are less likely to act
in such a drastic way when the symptoms return, regardless of the severity of their symptoms or their apparent
loss of insight. Other possible explanations are that bizarre organ-specific delusions are less common in PTP
or that the intensity of delusional beliefs declines later
in the illness. The differences may even be due to changes
in the brain after treatment with antipsychotic medication. Other factors such as an ongoing relationship
with a treating team or awareness of the patients illness
by family and friends may result in some protective measures such as encouraging treatment when the patient
becomes unwell. Although we found no major clinical
differences between those who self-mutilated in FEP
and those who did so in PTP, it would have been of
interest to compare possible protective factors such as
engagement with a treating team and the level of insight
of MSM and non-MSM patients in FEP and PTP.
Risk and Management
Males in a first episode of a schizophrenic illness that is
characterized by delusions associated with a body part or
religious delusions are at the greatest risk for MSM.

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and a prevalence of 0.5%, there are 25 times more previously treated schizophrenia spectrum patients than firstepisode patients in a typical population in any given year.
If the risks of MSM were evenly spread over the course of
the illness, then the ratio of PTP to FEP patients with
MSM would be about 25 to 1. In fact, previously treated
patients and those in FEP were found in about equal in
numbers, indicating that that the risk of MSM in FEP is
as much as 25 times greater than the subsequent annual
risk after initial treatment.
The finding supports the hypothesis that the risk of
MSM is significantly greater in the FEP compared
with subsequent episodes of psychosis, a finding that is
similar to recently published studies of homicide in
psychosis.

Major Self-mutilation

Conclusions
The importance and limitations of published case histories
are highlighted by this study. Research about MSM
requires new methods because case histories are inevitably
subject to publication bias, and valid inferences cannot be
made from the observation of small numbers of cases. The
reliance on memory and retrospective ways of case finding, such as we used to make an estimate of the incidence
of MSM, are also unsatisfactory. Ideally, state health authorities should collected data about MSM events. An alternative is for networks of clinicians to gain ethical
approval to create an electronic registry of rare psychiatric
cases. A representative sample of cases could be derived by
this method and a priori hypotheses could be tested.
Future research along these lines could make a more
conclusive estimate of the risk of MSM in FEP. We
have not been able to show that the risk of MSM
increases over time if treatment is delayed. However,
this article provides the basis for a convincing argument
that earlier intervention in psychosis may reduce the
chance of MSM because adequate antipsychotic treatment appears to be protective.
Funding
Rotary Foundation, Evanston to P.S.
Acknowledgments
We would like to thank Ilona Harsanyi and Fang Fang of
the Winston Library at the Sydney Eye Hospital for their

assistance locating the publications and the many

clinicians in NSW who shared their knowledge of cases
of MSM. Declaration of interests: None.

References
1. Favazza A. Bodies Under Siege. Baltimore: John Hopkins
University Press; 1987.
2. Favazza A, Rosenthal R. Diagnostic issues in self-mutilation. Hosp and Community Psychiatry. 1993;44:134140.
3. Nakaya M. On background factors of male genital self-mutilation. Psychopathology. 1996;29:242248.
4. Tapper CM, Bland RC, Danyluk L. Self-inflicted eye injuries and self-inflicted blindness. J Nerv Ment Dis. 1979;167:
311314.
5. Clark RA. Self-mutilation accompanying religious delusions: a case report and review. J Clin Psychiatry. 1981;42:
243245.
6. Sweeny S, Zamecnik K. Predictors of self-mutilation in
patients with schizophrenia. Am J Psychiatry. 1981;138:
10861089.
7. Large M, Andrews D, Babidge N, Hume F, Nielssen O. Selfinflicted eye injuries in first-episode and previously treated
psychosis. Aust N Z J Psychiatry. 2008;42:183191.
8. Large M, Nielssen O. Evidence for a relationship between
the duration of untreated psychosis and the proportion of
psychotic homicides prior to treatment. Soc Psychiatry Psychiatr Epidemiol. 2008;43:3744.
9. Nielssen OB, Westmore BD, Large MM, Hayes RA. Homicide during psychotic illness in New South Wales between
1993 and 2002. Med J Aust. 2007;186:301304.
10. Large M, Nielssen O. Treating the first episode of schizophrenia earlier will save lives. Schizophr Res. 2007;92:276277.
11. Travers J, Milgram S. An experimental study of the small
world problem. Sociometry. 1969;32:425443.
12. Tharoor H. A case of genital self-mutilation in an elderly
man. Prim Care Companion J Clin Psychiatry. 2007;9:
396397.
13. Agoub M, Battas O. Male genital self-mutilation in patients
with schizophrenia. Can J Psychiatry. 2000;45:670.
14. Ananth J, Kaplan HS, Lin KM. Self-inflicted enucleation of
an eye: two case reports. Can J Psychiatry. 1984;29:145146.
15. Aung T, Yap EY, Fam HB, Law NM. Oedipism. Aust N Z J
Ophthalmol. 1996;24:153157.
16. Becker M, Hofner K, Lassner F, Pallua N, Berger A. Replantation of the complete external genitals. Plast Reconstr
Surg. 1997;99:11651168.
17. Blacker KH, Wong N. Four cases of autocastration. Arch
Gen Psychiatry. 1963;8:169176.
18. Borenstein A, Yaffe B, Seidman DS, Kaplan HY, Tsur H.
Successful microvascular replantation of an amputated penis. Isr J Med Sci. 1991;27:395398.
19. Buhrich N, Hayman J. Self-inflicted enucleation of both
eyes. Aust N Z J Psychiatry. 1994;28:337341.
20. Brown BZ. Self-inflicted injuries of the eye. Ann Ophthalmol.
1972;4:147149.
21. Brown BZ. Self-inflicted injuries of the eye. Trans Pac Coast
Otoophthalmol Soc Annu Meet. 1970;51:267276.

1019

Downloaded from http://schizophreniabulletin.oxfordjournals.org/ by guest on November 7, 2016

However, MSM of this severity is so rare that it cannot be

predicted accurately unless there has been a previous
attempt at self-injury or the patient has spoken about
wanting to remove or injure an organ.
Threatened ocular mutilation deserves special mention
because it may occur in a hospital setting, and the case
histories suggest that one to one nursing is not always
be sufficient to prevent enucleation.7 All MSM requires
urgent medical attention because genital and limb MSM
may cause exsanguination, and subarachnoid hemorrhage, meningitis, and pituitary failure are potentially
fatal complications of self-enucleation. Replantation of
amputated genitals and limbs can be performed with
some return of function, and testicular replantation
may avoid the requirement for long-term testosterone
therapy. Repeated MSM has been reported but seems
to be uncommon. Hence, emergency limb and genital replantation can be justified in the presence of severe psychosis that renders the patient incapable of informed
consent. Patients with MSM warrant a detailed assessment for the presence of psychosis and even patients
who are not initially forthcoming about the reason for
MSM should be regarded as suffering from a psychosis
until proved otherwise.

M. Large et al.

1020

46. Kobayashi T, Osawa T, Kato S. Upper-extremity selfamputation in a case with schizophrenia. Eur Psychiatry.
2002;17:172173.
47. Koh KG, Lyeo BK. Self-enucleation in a young schizophrenic patient-a case report. Singapore Med J. 2002;43:
159160.
48. Krauss HR, Yee RD, Foos RY. Autoenucleation. Surv Ophthalmol. 1984;29:179187.
49. Kumar AV, Geist CE. A case report of bilateral autoenucleation and its prevention. Orbit. 2007;26:309313.
50. Kushner AW. Two cases of auto-castration due to religious
delusions. Br J Med Psychol. 1967;40:293298.
51. Landstrom JT, Schuyler RW, Macris GP. Microsurgical
penile replantation facilitated by postoperative HBO treatment. Microsurgery. 2004;24:4955.
52. Lazarou EE, Catalano G, Catalano MC, Leon YC, Gorman
JM. The psychological effects of leech therapy after penile
auto-amputation. J Psychiatr Pract. 2006;12:119123.
53. Leslie J, Taff ML, Patel I, Sternberg A, Fernando MM. Selfinflicted ocular injuries. A rare form of self-mutilation. Am J
Forensic Med Pathol. 1984;5:8388.
54. Lidman D, Danielsson P, Abdiu A, Fahraeus B. The functional result two years after a microsurgical penile replantation. Case report. Scand J Plast Reconstr Surg Hand Surg.
1999;33:325328.
55. MacLean G, Robertson BM. Self-enucleation and psychosis. Report of two cases and discussion. Arch Gen Psychiatry. 1976;33:242249.
56. Martin T, Gattaz WF. Psychiatric aspects of male genital
self-mutilation. Psychopathology. 1991;24:170178.
57. Mendez R, Kiely WF, Morrow JW. Self-emasculation.
J Urol. 1972;107:981985.
58. Money J, De Priest M. Three cases of genital self-surgery
and their relationship to transexualism. J Sex Res.
1976;12:283294.
59. Mora W, Drach GW. Self-emasculation and self-castration:
immediate surgical management and ultimate psychological
adjustment. J Urol. 1980;124:208209.
60. Moskovitz RA, Byrd T. Rescuing the angel within: PCPrelated self-enucleation. Psychosomatics. 1983;24:402406.
61. Murphy M, Nathan M, Lee E, Parsons B, Gunasekera L.
Oedipism: auto-enucleation in a schizophrenic patient. Ir
J Psychol Med. 2006;23:159160.
62. Myers WC, Nguyen M. Autocastration as a presenting
sign of incipient schizophrenia. Psychiatr Serv. 2001;52:
685686.
63. Novak-Grubic V, Tavcar R. Autocastration and schizophrenia. Psychiatr Serv. 2002;53:485486.
64. Pantuck AJ, Lobis MR, Ciocca R, Weiss RE. Penile replantation using the leech Hirudo medicinalis. Urology.
1996;48:953956.
65. Riffle MD. Lower extremity amputation: conclusion. Air
Med J. 2007;26:202204.
66. Riffle MD, Ross DW, Wichman C, MacKinnon M. Lower
extremity amputation. Air Med J. 2007;26:164.
67. Rogers T. Self-inflicted eye-injuries. Br J Psychiatry. 1987;
151:691693.
68. Romilly CS, Isaac MT. Male genital self-mutilation. Br
J Hosp Med. 1996;55:427431.
69. Rosen DH, Hoffman AM. Focal suicide: self-enucleation by
two young psychotic individuals. Am J Psychiatry. 1972;128:
10091012.

Downloaded from http://schizophreniabulletin.oxfordjournals.org/ by guest on November 7, 2016

22. Brown R, al-Bachari MA, Kambhampati KK. Self-inflicted

eye injuries. Br J Ophthalmol. 1991;75:496498.
23. Carson DI, Lewis J. Ocular auto-enucleation while under
the influence of drugs: case report. Adolescence. 1971;6:
397403.
24. Conacher GN, Westwood GH. Auto-castration in Ontario
federal penitentiary inmates. Br J Psychiatry. 1987;150:
565566.
25. Cooper AJ, Swamy GN. The effect of testosterone on psychopathology and sexual function in a paranoid schizophrenic self-castrate. Can J Psychiatry. 1994;39:436438.
26. Davidson SI. Auto-enucleation of the eye: a study of self
mutilation. Acta Psychother Psychosom. 1962;10:286300.
27. Dilly JS, Imes RK. Autoenucleation of a blind eye. J Neuroophthalmol. 2001;21:3031.
28. Duggal HS, Jagadheesan K, Nizamie SH. Acute onset of
schizophrenia following autocastration. Can J Psychiatry.
2002;47:283284.
29. Eisenhauer GL. Self-inflicted ocular removal by two psychiatric inpatients. Hosp Community Psychiatry. 1985;36:
189191.
30. Erdur B, Turkcuer I, Herken H. An unusual form of selfmutilation: tongue amputation with local anesthesia. Am
J Emerg Med. 2006;24:625628.
31. Evins SC, Whittle T, Rous SN. Self-emasculation: review of
the literature, report of a case and outline of the objectives of
management. J Urol. 1977;118:775776.
32. Feldshuh B, Zasloff M, Frosch WA. If thy right eye offend
thee. A case of bilateral self-enucleation. Isr Ann Psychiatr
Relat Discip. 1977;15:145155.
33. Fisch RZ. Genital self-mutilation in males: psychodynamic
anatomy of a psychosis. Am J Psychother. 1987;41:453458.
34. Goldstein JL, Gerdis JE, Whitman LJ, Novins DK. A case
of genital self-amputation in which reconstruction was proposed. Gen Hosp Psychiatry. 1990;12:401402.
35. Goldenberg E, Sata L. Religious delusions and self mutilation. Curr Concepts Psychiatry. 1978;4:25.
36. Goldwyn RM, Cahill JL, Grunebaum HU. Self-inflicted injury to the wrist. Plast Reconstr Surg. 1967;39:583589.
37. Gorin M. Self-inflicted bilateral enucleation. Arch Ophthalmol. 1964;72:225226.
38. Greilsheimer H, Groves JE. Male genital self-mutilation.
Arch Gen Psychiatry. 1979;36:441446.
39. Hall DC, Lawson BZ, Wilson LG. Command hallucinations
and self-amputation of the penis and hand during a first psychotic break. J Clin Psychiatry. 1981;42:322324.
40. Halo HH, Jordan TA, Stewart RM, Apshaga SE. Selfenucleation: pathology and treatment. R I Med J. 1990;73:
117120.
41. Hingorani M, Singh A, Williams H. Oedipism and ocular
self mutilation. Ir J Psychol Med. 1995;12:144146.
42. Ishida O, Ikuta Y, Shirane T, Nakahara M. Penile replantation after self-inflicted complete amputation: case report.
J Reconstr Microsurg. 1996;12:2326.
43. Jimenez-Cruz JF, Garcia-Reboll L, Alonso M, Broseta E,
Sanz S. Microsurgical penis replantation after self-mutilation. Eur Urol. 1995;27:246248.
44. Jones NP. Self-enucleation and psychosis. Br J Ophthalmol.
1990;74:571573.
45. Kalin NH. Genital and abdominal self-surgery. A case report. JAMA. 1979;241:21882189.

Major Self-mutilation

88. Yang JG, Bullard MJ. Failed suicide or successful male genital self-amputation? Am J Psychiatry. 1993;150:350351.
89. Young LD, Feinsilver DL. Male genital self-mutilation combined surgical and psychiatric care. Psychosomatics.
1986;27:513517.
90. Yucel B, Ozkan S. A rare case of self-mutilation: self-enucleation of both eyes. Gen Hosp Psychiatry. 1995;17:310311.
91. Master V, Santucci R. An American hijra: a report of a case
of genital self-mutilation to become Indias third sex.
Urology. 2003;62:1121.
92. Bhatia MS, Arora S. Penile self-mutilation. Br J Psychiatry.
2001;178:8687.
93. Shimizu A, Mizuta I. Male genital self-mutilation: a case report. Br J Med Psychol. 1995;68:187189.
94. Gleeson MJ, Connolly J, Grainger R. Self-castration as
treatment for alopecia. Br J Urol. 1993;71:614615.
95. Brenner I. Upper-extremity self-amputation in a case of
dissociative identity disorder. J Clin Psychiatry. 1999;60:
705706.
96. Saha S, Chant D, McGrath J. Meta-analyses of the incidence
and prevalence of schizophrenia: conceptual and methodological issues. Int J Methods Psychiatr Res. 2008;17:5561.
97. First MB. Desire for amputation of a limb: paraphilia, psychosis, or a new type of identity disorder. Psychol Med.
2005;35:919928.
98. Johnson TW, Brett MA, Roberts LF, Wassersug RJ.
Eunuchs in contemporary society: characterizing men who
are voluntarily castrated (part I). J Sex Med. 2007;(4 Pt 1):
930945.
99. Hill AB. The environment and disease: association or causation? Proc R Soc Med. 1965;58:295300.
100. Siddle R, Haddock G, Tarrier N, Faragher EB. Religious
delusions in patients admitted to hospital with schizophrenia. Soc Psychiatry Psychiatr Epidemiol. 2002;37:130138.
101. Link BG, Stueve A, Phelan J. Psychotic symptoms and violent behaviors: probing the components of threat/controloverride symptoms. Soc Psychiatry Psychiatr Epidemiol.
1998;33:5560.
102. Ward A, Ishak K, Proskorovsky I, Caro J. Compliance with
refilling prescriptions for atypical antipsychotic agents and
its association with the risks for hospitalization, suicide,
and death in patients with schizophrenia in Quebec and
Saskatchewan: a retrospective database study. Clin Ther.
2006;28:19121921.

1021

Downloaded from http://schizophreniabulletin.oxfordjournals.org/ by guest on November 7, 2016

70. Schlozman SC. Upper-extremity self-amputation and replantation: 2 case reports and a review of the literature.
J Clin Psychiatry. 1998;59:681686.
71. Schweitzer I. Genital self-amputation and the Klingsor syndrome. Aust N Z J Psychiatry. 1990;24:566569.
72. Schweitzer I, Rosenbaum MB, Sharzer LA, Strauch B. Liaison consultation psychiatry with patients who have replantation surgery to the upper limb. Aust N Z J Psychiatry.
1986;20:3843.
73. Sharpe JR, Belsole RJ, Keesal RW, Sadlowski RW, Finney
RP. A preplanned approach to the amputated penis. South
Med J. 1979;72:1634.
74. Shirodkar SS, Hammad FT, Qureshi NA. Male genital selfamputation in the Middle East. A simple repair by anterior
urethrostomy. Saudi Med J. 2007;28(5):791793.
75. Shiwach RS. Autoenucleationa culture-specific phenomenon: a case series and review. Compr Psychiatry. 1998;39:
318322.
76. Shore D, Anderson DJ, Cutler NR. Prediction of self-mutilation in hospitalized schizophrenics. Am J Psychiatry. 1978;
135:14061407.
77. Stunell H, Power RE, Floyd M Jr, Quinlan DM. Genital
self-mutilation. Int J Urol. 2006;13:13581360.
78. Strain JJ, DeMuth GW. Care of the psychotic self-amputee
undergoing replantation. Ann Surg. 1983;197:210214.
79. Tang WN. The first case of autocastration from east Asia.
Can J Psychiatry. 1996;41:607608.
80. Tapper CM, Bland RC, Danyluk L. Self-inflicted eye injuries and self-inflicted blindness. J Nerv Ment Dis. 1979;167:
311314.
81. Tavcar R, Dernovsek MZ, Zvan V. Self-amputation of left
hand: a case report. J Clin Psychiatry. 1999;60:793794.
82. Thomas RB, Fuller DH. Self-inflicted ocular injury associated with drug use. J S C Med Assoc. 1972;68:202203.
83. Thompson JN, Abraham TK. Male genital self mutilation
after paternal death. Br Med J. 1983;287:727728.
84. Volkmer BG, Maier S. Successful penile replantation following autoamputation: twice!. Int J Impot Res. 2002;14:197198.
85. Walter PJ, Krauss DJ, Nsouli IS. Repeat male genital selfmutilation precipitated by urinary complications of prior repair. J Urol. 1993;149:15511552.
86. Waugh AC. Autocastration and biblical delusions in schizophrenia. Br J Psychiatry. 1986;149:656658.
87. Wei FC, McKee NH, Huerta FJ, Robinette MA. Microsurgical replantation of a completely amputated penis. Ann
Plast Surg. 1983;10:317321.