Self-Amputation in Patient with Body Integrity Dysphoria in Comorbidity with Gender Dysphoria

Introduction

The loss of limb(s) or becoming disabled is an undesirable state which most people would try to avoid. The existence of people for whom limb loss/disability leads to feelings of happiness or improving well-being is therefore difficult to comprehend and is commonly recognized as clearly pathological. The cases of individuals who harbor a longstanding desire to amputate or disable their disowned body parts have been described in the literature [1^-4]. Those people recognize themselves, often according to internet-based forums, as having body integrity identity disorder (BIID), now described as BID. The current understanding of this condition has shifted from a traditional psychological explanation of its etiology as paraphilic motivation. The two main approaches remain a matter of debate in the conceptualization and understanding of BID. One of these approaches relates to a neural correlate, such as alteration in the functional connectivity in certain cortical hubs, as well as in the concentration of gray matter in a neurodevelopmental perspective [5]. Consequently proposing that this condition arises from congenital dysfunction of the right superior parietal lobule and its neural connections with the insular cortex [6^-10]. Another explanation of BID is to regard the condition in terms of identity disorder, especially gender identity disorder. There is an indication that the disruption of the identification with one’s own body is not only limited to limbs but also affects the gender identity of individuals. Scupin et al. [11] tested the features of gender identity on a group of 50 BID people (25 male and 25 female) compared with the same size control group of non-BID individuals. The results show that for the BID group, gender identity was less pronounced and not as defined as the control group. Furthermore, 8% of the sample met cutoff scores for gender dysphoria (GD), thus indicating that the disruption of the identity in an individual with BID is not always confined to a particular body area. This points to a connection between BID and GD. Previously, First [2] reported a higher than expected comorbidity rate for BID and GD. In a published series of 52 cases with BID, he described “feeling of being in the body of opposite sex” (transgender experience) in 19% of the sample. These findings are significant, not only in terms of higher frequency of co-occurrence but also other clinical characteristics [12^-14]. Lawrence [13] argues also that these parallels between the desire for amputation and transsexualism are more numerous, as well as being more clinically and theoretically significant, in the subgroup of nonhomosexual male-to-female transsexuals. Other psychiatric comorbidity in the BID population does not differ much from the general population, except for an increase in depressive and anxiety symptoms [7^, 12^, 15]. These conditions are considered to be secondary due to the distress generated by BID. In an online-based survey of 54 people with identified BID [15] symptoms of mood disorders were identified in almost 25% of the responders, 2 people had eating disorder or anxiety disorder and 1 patient met the criteria for psychotic disorder.

As demonstrated in the case below, BID can be comorbid with GD and other psychiatric conditions. This can make diagnostics problematic.

Case Presentation

A 24-year-old transgender male1 presented to the emergency department after self-amputation of the left hand using an electric saw. This act was carefully planned for several months. He booked a hotel room, wrote a note requesting medical care, took a ritual bath listening to his favorite music, took nonsteroid analgetics, cut the skin on his wrist with a particular knife, and then using an electric saw cut through tissue and bones, severing his hand. He then deliberately placed the amputated hand into a caustic agent, to destroy tissue and so preventing surgical reattachment. Throughout the amputation, he took photographs and communicated online with other members of an online peer group identifying themselves as having BIID. When asked to justify his actions, he explained that he did not want his hand, as since childhood he dreamed about waking up without his left hand. He felt the hand was alien and he did not identify his left hand as being a part of his own body.

Furthermore, the patient identifies “herself” as a man and wishes to be addressed with a male name, pronoun, and treated as a male. This condition has been present in the patient’s life since early adolescence. He plans to apply for legal gender reassignment sometime in the future.

Previous History

The patient was raised as the only child by his parents in a conservative and isolated community in a small central European country. His father was successfully treated for alcohol dependence. His parents divorced. The patient lived alternately with his mother or father. He studied at Art School. His early development was reportedly normal. His parents described him as emotionally distant and detached, with hobbies and skills more typical for boys (repairing toys, creating knives, etc.). Around the age of 10 years, he started to make comic book-style drawings, in which the heroes had a prosthetic implant instead of a left hand (Fig. 1). During secondary school, the patient was treated for attention deficit hyperactivity disorder with atomoxetine. At the age of 15 years, he disclosed to his parents that he identified as a man and requested to be treated as male. At the age of 19 years, he was admitted to a psychiatric clinic due to severe dysphoria associated with gender incongruence. At that time, no further treatment was recommended. He did not reveal his feelings concerning his hand to doctors or his parents but later admitted that these feelings were present at that time. During his college studies, he was treated for a depressive episode with escitalopram. At that time, he started to intensively play a multiplayer online role play game known as Wakfu. He created his avatar without the left hand. He described being obsessed with the game, failed in school, was not able to concentrate on daily activities, did not eat properly, and lost significant weight. He appreciated this because it helped him to “get rid of” his breasts. While playing Wakfu, the desire to cut off his left hand was reduced, as he could realize his desires through a virtual online character. However, shortly after he stopped playing, this desire to self-amputate returned and became more intense, almost unbearable. It was then that he started to plan to amputate his left hand. He described the support of other members of the BIID internet forum as being very supportive and helpful on this matter.

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In-Patient Care

After admission to the psychiatric clinic, mood swings were the dominant psychopathological symptom. In his elevated, euphoric mood, he described with marked excitement the process of amputation to other patients, collecting photographs of the amputation. This elevation did not have a significant influence on the observed behavior, which was neither hyperactive nor disinhibited. The patient was cooperative. Later, transient depressive mood with anxiety occurred, but no other depressive signs and symptoms were present. The patient described relief from “getting rid of the block” (unwanted hand). Social acceptance of his behavior, and how he was perceived by his family and peers, has always been very important to him. Although he was satisfied with the outcome of his act, the disappointment of his parents and the reactions of medical staff concerning his self-amputation aroused remorse in him and feelings of guilt toward his family. Throughout, the patient had no doubts about his gender, identifying, and referring to himself as male. Living as he did in a place with little cultural variety, or understanding of difference, he faced difficulties with social acceptance of his gender identity. This discouraged him from expressing his preferred identity.

Due to the inadequate and bizzare explanation offered by the patient, the motivation for the amputation was initially described as delusional. Reduced contact with reality was also described. However, no other disturbances of thinking, in terms of structure and content, nor disturbances of perception were found. Examination (blood count, biochemistry, electroencephalography, magnetic resonance imaging) showed no pathological signs. Because the patient was considered psychotic at this time, he was treated with antipsychotic medication. Risperidone 3 mg was replaced with after 3 weeks to olanzapine 20 mg daily due to the development of depressive symptoms with massive anxiety. The patient engaged in individual psychotherapy and attended ergotherapeutical, musicotherapeutical, and art therapeutical groups. At discharge, the patient showed no signs of depression. His attitude toward the loss of his limb he described as a “relief,” associated with the absence of an estranged body part, and satisfaction with his new body image. However, he identified a negative impact on the quality of his interpersonal relationships toward his parents. The initial diagnosis of a psychotic disorder was later revised, as the desire for left hand amputation was not finally evaluated as psychotic, due to the fact that no convincing psychotic symptoms were found during his hospitalization, and given that the idea of removing his unwanted left hand had accompanied him from a young age, and he did not regret his autoamputation at all. He was diagnosed as BID on discharge. Other diagnoses were other acute predominantly delusional psychotic disorders (F23.3), schizoid personality disorder (F60.1), and gender identity disorder – female to male transsexualism, which was to be further evaluated and clinically addressed.

Follow-Up Period

Outpatient treatment followed hospital discharge. Due to diurnal somnolence, olanzapine was reduced to 15 mg pro die. Two months after discharge, the patient developed a severe depressive episode that was treated with sertraline. During this depression, the patient experienced feelings of guilt and blamed himself for upsetting his parents by cutting his hand. Yet, he showed absolutely no remorse for the loss of his hand. He described his forearm stump as “cute, sweet and adorable,” and considered other amputees as “complete” and “nice.” The authors find this of diagnostic importance. However, those feelings were not evidenced as sexual and not associated with sexual arousal. He felt sorry for his father, who was upset about the whole situation and was worried that self-amputation could ruin or postpone his plans for future gender reassignment. He was not able to study, and so dropped out his studies, with plans to continue when feeling better.

Later, while watching TV with his father, he suddenly developed intense obsessive thoughts about killing his father, which he shared with him. To begin with, they both discussed this, and the father turned it into a joke. As the intensity of these fantasies increased over time, the thoughts became more intrusive and complex, with designs of planned killing. The patient was again admitted to psychiatric hospital. These intrusive thoughts were markedly aggressive and directed only against his father. With increasing doses of olanzapine, these thoughts faded and disappeared within a few days of hospitalization.

Two psychiatric hospitalizations were then recommended – one to a psychiatric clinic and the other to a daily psychiatric care unit. The reason for admission was the worsening of depression with reappearing obsessive thoughts. The patient was treated with olanzapine long-acting injectable 405 mg monthly, cariprazine 3 mg, valproate 500 mg, and escitalopram 20 mg daily. Due to ongoing mood swings, the diagnosis was changed to bipolar affective disorder. Intrusive thoughts with homicidal content disappeared usually shortly after admission but tended to reappear when antipsychotic medication was decreased.

Even during these hospitalizations, his transgender experiences and the alienation of his left hand preceding its self-amputation were not assessed as psychotic or obsessive. They fully met the diagnostic requirements of GD and BID.

The patient’s attitude toward the loss of his hand was less positive, as he realized that “it’s not practical in everyday life,” but overall he continued to show satisfaction with it. His plans for gender reassignment were postponed. He had transient doubts about expressing his gender identity in terms of male-female, and transitionally contemplated agender identity. His uncertainty was mainly due to his low self-esteem, weak communication, and social skills, as well as critical attitudes from loved ones who were unable to support him.

For practical reasons due to problems arising from the incongruence between legal status as female and male appearance, he presented himself as female during this period, but he described it as a “forced and rational behavior.” However, with the help of his psychiatrist, he finally gained the courage to openly present himself as a male transgender person and to accept, not hide, his amputee status. At present, the patient is striving for transition, at least in terms of hormonal therapy. Gender identity problems are perceived by the patient himself as different from problems with his left hand, although it seems strange to him that he suffered from both problems at the same time. When asked to give his explanation for his feelings, he describes it simply as “it is how it is, I was born this way.” After amputation, the patient’s left-hand dysphoria was resolved and he feels satisfied. He denies the need to cut his hand or other parts of his body further. He is happy not to have his hand, but “wouldn’t do it again this way,” because of the experienced pain. However, he sees the solution for GD only in the distant future, as, according to him, he must gather enough courage to solve it openly. He never mentioned sexual arousal in relation to amputees or in relation to idea of himself after amputation. However, the stub after the amputation, he considers as aesthetically attractive and as a real and positive part of his body. He is not sexually aroused by the idea of being male and he denies that the motives for his bodily refiguration were sexually motivated in any way.

Discussion

This rare phenomenon, an unusual psychiatric condition characterized by a persistent desire to be physically disabled, has been described mostly in case reports [2^, 3^, 16]. Money and Simcoe [16] coined the term apotemnophilia as paraphilia that pertains to self-amputation in contrast to acrotomophilia that can be understood as paraphilic interest in amputation and amputees. First and Fisher [7] proposed the term BIID as well as broadening its meaning to incorporate individuals with a persistent desire to acquire significant disabilities such as paraplegia, blindness, as well as amputation. As yet, this condition has not been included in The Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition, but has been added to the forthcoming International Classification of Diseases, Eleventh Revision, as BID under the code 6C21 [17]. It is characterized by an intense and persistent desire to become physically disabled in a significant way (e.g., major limb amputee, paraplegic, blind), with onset by early adolescence accompanied by persistent discomfort, or intense feelings of inappropriateness concerning the current nondisabled body configuration. The desire to become physically disabled can result in grievous consequences, as manifested by either the preoccupation with the desire to become disabled (including time spent pretending to be disabled), significantly interfering with productivity as well as leisure activities, or with social functioning (e.g., the person is unwilling to have close relationships as the pretense of disability would be difficult to maintain), or by attempts to actually become disabled that have resulted in the person putting his or her health or life in significant jeopardy.

Our patient met the proposed diagnostic requirements of BID. Moreover, this condition was comorbid with GD. First [2^, 7] noted parallels between BID and a GD. These include a feeling of discomfort with a given anatomical identity, onset usually during childhood, successful treatment being affected by mimicking the desire and paraphilic sexual arousal obtained by fantasies of the desired identity. Other evidence of a connection between BID and GD is that the co-occurence of those conditions is higher than would be expected by chance [7]. The conceptual relationship with transsexualism was already noted by Money et al. [3], although, unlike First [2^, 7], he considered sexual motivation and attraction as primary at this point, in parallel with paraphilias.

A leading point in diagnosis was the question whether the desire for self-amputation was part of a psychotic disorder, or was a separate symptom of another condition. According to Money et al. [3] apotemnophilia obsession represents more an “ideé fixe” than paranoid delusion. First and Fischer [7] also maintain the idea of a non-delusional origin to the patient’s motivation. They state that if anything, such beliefs might be considered overvalued ideas [7]. The main characteristics in our patient were the onset in early adolescence; preoccupation with the idea; the impact on social activities; persistence with the oscillation of intensity according to external factors (desire was diminished while intensely playing a computer game, in which he adopted a virtual amputee identity); resistance to antipsychotic or antidepressant treatment (no core changes in an amputee or transgender identity); and what we find crucial, the completely resolved desire after self-amputation, the patient’s satisfaction with the acquired body-scheme – in terms of his hand loss. Additionally, the patient would like to have a mastectomy, as part of gender reassignment surgery sometime in the future.

From this point of view, the diagnosis of BID could be considered “ex-post” diagnosis. There are still questions concerning how long the patient’s relief will last. Systematic data relating to the postamputation period are not available, so it is hard to assume whether the relief from “being complete again” will last and whether it will overcome the inevitable negative practical consequences of amputation. In a study of 21 BID subjects who achieved their desired amputation, the authors stated that most of the study participants reported that they had suffered more from BID than from any disadvantages in life as a disabled person and that those effects are solid even for a longer term [18]. In a follow-up of our patient, a year after initial hospitalization, it was observed that he was more critical to the practical consequences associated with the loss of his hand but not to the hand loss or amputation per se.

Apart from the diagnosis of BID and GD, the case of our patient was complicated with other psychopathological symptoms which made diagnosis nebulous. The biggest concern was dealing with the recurrent obsessive thoughts that could imply the diagnosis of obsessive-compulsive disorder. The patient was not by now diagnosed with obsessive-compulsive disorder due to the fact that those thoughts seemed to be present only during the worsening of bipolar disorder, mainly as a part of depressive syndrome. These thoughts tended to disappear when the patient was in remission.

The global outcome continues to be influenced by other interrelated conditions. These include a gaming disorder with episodes of abstaining from playing as well as relapses, personality traits arising from schizoid personality disorder, a life led predominantly online, all leading to the deepening of his social isolation. Symptoms of ADHD are not present at the moment.

Conclusion

We present a patient with a rare condition of BID. Unlike other published cases, this is atypical due to other presented comorbidities such as GD, gaming, and personality disorders as well as bipolar disorder. This made the diagnostics problematic, mostly in evaluating the patient’s motivation for amputation. Although the patient’s attitude to his own self-amputation varies slightly over time, he does not regret it and finds the new body configuration attractive. Moreover, this self-amputation has clearly resolved his BID.

During the described period, high variability in the type and the intensity of other psychopathological symptoms were observed and are still present, requiring treatment. The authors are aware of the need for longer observation in such cases and wish to show that even severe automutilation, such as self-amputation, may not always be an isolated condition. As practicing clinicians, such rare cases highlight the need for continued vigilance and dialog. Our knowledge and understanding increase with each case.

Statement of Ethics

The study was conducted ethically in accordance with the World Medical Association Declaration of Helsinki. Our study was approved by The Ethics Committee of The University Teaching Hospital in Bratislava (Ethics Committee Statement No. EC/122/2021). Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent and The Ethics Committee Statement is available for review by the Editor-in-Chief of this journal.

Conflict of Interest Statement

The authors declare that they have no conflict of interest.

Funding Sources

The authors received no financial support for the research, authorship, and/or publication of this article.

Author Contributions

All four authors have treated the patient in different clinical settings. B.V. initiated the case report. All four authors wrote the first draft, each in part, as treating psychiatrist. All authors contributed equally in reviewing and revising the manuscript and approved the final and submitted version of the manuscript.

Data Availability Statement

All data generated or analyzed during this study are included in this article. Further inquiries can be directed to the corresponding author.