Successful treatment of body integrity dysphoria with amputation: A case report

Abstract

Key Clinical Message

In select cases of body integrity identity disorder or body integrity dysphoria where noninvasive treatments prove ineffective and the patient's distress is substantial, elective amputation may serve as a viable and highly satisfying intervention, aligning the individual's physical self with their perceived identity.

Abstract

This case report presents an illustration of body integrity identity disorder (BIID), wherein a 20 years old ambidextrous male experiencing profound distress over his left hand's fourth and fifth fingers sought elective amputation after noninvasive treatments proved unsuccessful. Despite ethical concerns and limited literature on BIID, the decision to proceed with elective surgery was based on the patient's sustained desire, potential risks of self‐harm, and the distinct presentation involving two fingers rather than a complete limb. Following amputation, the patient experienced immediate relief, with nightmares ceasing, emotional distress subsiding, and improved functionality. This case highlights the potential efficacy and patient satisfaction associated with elective amputation in specific BIID presentations, shedding light on the unique challenges faced by affected individuals and emphasizing the importance of understanding, support, and inclusive healthcare practices.

1. INTRODUCTION

Body integrity dysphoria (BID) or body integrity identity disorder (BIID) causes a distressing belief that a particular body part should not exist, leading some to attempt self‐amputation, risking their lives. Ethical concerns about patient autonomy arise when contemplating the elective amputation of non‐diseased functional body parts, such as limbs and, in this case, digits. The limited literature on this condition poses challenges in establishing clear guidelines and recommendations. Patients often hesitate to seek help from healthcare professionals, turning to internet forums for advice, complicating the assessment of BID's actual prevalence. ¹ Nevertheless, surgery as a treatment option for this lesser‐known disorder should be carefully considered.

2. CASE HISTORY & EXAMINATION

A 20 years old ambidextrous male with a preference for his right hand desires to have his left hand's fourth and fifth fingers amputated. He recognizes their ownership but perceives they should not belong to his body, a feeling present since childhood. He hides his fingers, keeps them flexed, leading to impaired dexterity, localized pain, irritability, and anger. Nightmares featuring his fingers rotting or burning disturb his sleep. He reports daily intrusive thoughts concerning his fingers, provoking a distressing feeling that they do not belong to him, that they should not be there and that they encroach upon his being, traumatizing him repeatedly. He appears reserved initially but seriously discusses the surgical implications of his demand. He has not shared his distress with family due to embarrassment about the unusual nature of his request. He asked for amputation as a mean to align his physical self with its image he has in his mind. He was seeking relief from the overwhelming distress caused by incessant thoughts about his fingers, and the sensation they didn't belong to him, even if he knew they were indeed his own. He also pursued surgical intervention to avoid having to carry out the act on his own, aiming to alleviate his intense suffering. Experiencing recurring trauma due to the presence of his fingers on his hand, his determination grew to find a method to get rid of those fingers he perceived as intrusive, foreign, unwanted. He had contemplated asking a friend to watch over him and be prepared to call emergency services in case his attempt led to a need for resuscitation. Working in a sawmill, he considered building a small guillotine to cut his fingers. He was aware self‐harm wasn't a safe solution and could have repercussions on his relationships, reputation, and health. He couldn't imagine himself living for the years to come with those fingers.

3. METHODS

Brain imaging was normal, and no significant family history or substance use is found. The diagnosis was established at a general hospital's psychiatric outpatient clinic. The consultation was requested by his primary care physician, who had known him since childhood as a typically healthy patient. Diagnostic interviews by a general psychiatrist and chart reviews, which included imaging, were conducted. No specific questionnaire was administered. Follow‐up visits served to confirm the diagnosis and document the patient's ongoing condition and his unchanged desire, capacity, which remained unchanged during the entire process. His hope to get a solution for his suffering was strong, he complied with high motivation during the entire process, consistently presenting himself seriously at every visit.

4. OUTCOME & FOLLOW‐UP

He was referred for cognitive‐behavioral therapy, he tried Fluoxetine up to 80 mg and Aripiprazole 15 mg daily, tolerating it well with mild sedation for 7 months. He remained convinced that amputation was ideal but agreed to try first a noninvasive relief. Distress increased during therapy sessions involving exposure, mirror exercises, and desensitization. He related his condition to gender dysphoria.

Deemed capable of requesting amputation, he was referred to orthopedics and stopped his psychotropic medications in a collaborative decision with the treating psychiatrist. While he felt relief about getting the referral for orthopedics consultation, normal doubts emerged about the potential outcomes. Six months later, he underwent elective amputation performed by an orthopedic surgeon at his local hospital, while retaining his consent and desire for the surgery. Post‐surgery, nightmares stopped immediately, along with the emotional distress. Postoperative surgical pain subsided in 1 week, and no phantom pain occurred at the one‐month follow‐up. Functionality was not meaningfully affected. In fact, he reported that without the two fingers he was able to use his hand as he had it mapped in his mind without having the fingers bothering him. Previously, those fingers would accidentally catch onto everything when he had them flexed. He won arm‐wrestling games, was able to drive his four wheels, kept working with his hands without any problem. He had constructive life plans, reduced anger, and improved well‐being with family and at work. No regrets were expressed. He feared judgment in romantic relationships but adapted his discourse to disclose as desired. During follow‐up visits with the physicians involved in his care, he reported that he was now able to enjoy a normal life.

The patient adhered to the treatment plan, beginning with noninvasive methods. He stated, once this process done, that successfully overcoming this intense suffering about his two fingers has given him confidence to tackle any future challenges.

With the patient's good collaboration, absence of comorbidities, and documented similar cases in the literature, recommending surgery for this young individual was straightforward. The involvement of two fingers compared to a complete normal limb made the decision easier for the medical team to go forward. The patient's emotions and reactions throughout the entire process, combined with the formal evaluation, provided evidence for the clinicians evaluating his capacity to decide for himself. Despite doubt and uncertainty about the outcomes of the intervention, he remained determined and collaborated with the orthopedic team. It resulted to quick access to treatment based on a clear psychiatric recommendation and referral, and prior unsuccessful attempts involving less invasive treatments with medication and psychotherapy.

The tolerability of psychotherapy was lower, resulted in more distress and a longer duration compared to surgery's side effects. Psychotherapy sessions exacerbated the dysphoria, despite the patient and therapist persisted in the hope for extinction of the distress toward the two undesired fingers. Postoperative pain didn't last months, quickly resolved in a week and solved the main problem of the patient, enabling him to pursue the of life he envisioned as a complete human being without those two fingers bothering him. Pharmacotherapy did not help and caused sedation. Long‐term risks of metabolic and movement disorders, along with sexual dysfunction disadvantages, outweighed the benefits for the young man with clear BID presentation.

5. DISCUSSION

One of the earliest described cases of BID was termed apotemnophilia by Money in 1977. ² This syndrome is characterized by intrusive and intense thoughts related to a desire to amputate a healthy body part, such as an arm or a leg. As a hypothesis to explain the disorder, it has been suggested that his could stem from a disparity between the perceived body schema and reality, resulting in the conviction that amputation is essential for one's identity. ³

Not enough evidence and long‐term efficacy studies currently exist to make surgery a first‐line treatment option, but it has been considered effective for treating BID, with high satisfaction rates in reported cases. ⁴ , ⁵ , ⁶ Patients aren't delusional and can make their own decisions. They recognize the limb as theirs, but feel it does not align with their body schema or identity since childhood. ³ , ⁴ , ⁶ , ⁷ , ⁸ A variety of presentations has been reported in the literature which could be categorized as cases of BID, a variant or a subset of it, or an entirely distinct entity. Some patients pretend not having the limb or prefer paralysis instead of amputation, which is more common among women, ⁶ , ⁸ although violent presentations, such as attempts at self‐amputation, might be more frequent in men and influence reported cases. ³

Taking into account that the patient's characteristics, as reported in this paper, correspond to those documented in the existing literature, and acknowledging the potential harmful consequences of delaying or not offering the surgery, including the risks of death due to self‐harm in an attempt to self‐amputate, opposed to the potential benefits he could have with the amputation, the decision was made to move forward with the surgery, even though there was no guarantee of symptoms remission. A significant distinction from a lot of reported cases in the literature, which eased the decision‐making process for the medical team, was that two digits were the distressing body part, as opposed as a complete limb in most typical forms of BID reported. This could raise the question about the possibility of a selection bias and more reports and awareness of BID with a complete limb presentation because of the extent of the impairment or impact on the body of the sufferer and the implications for the medical team when contemplating invasive intervention.

Gray matter atrophy correlates with compensatory behaviors, but it's unclear if it's a cause or effect of symptom intensity. ⁴ , ⁹ The dysphoria‐causing region's boundaries are clear, ⁸ with reduced skin conductance responses under the desired amputation site. ⁹ , ¹⁰ In contrast to body dysmorphic disorder, in which surgery is not typically recommended, these patients do not usually perceive their limb as ugly or diseased. ⁵ , ¹¹ Even if they do, aesthetic reasons are not a primary motivating factor for their desire for amputation. In this specific case, the patient considered the cosmetic change and being an amputee as a potential obstacle in relationships, at work or in sports and leisure. The importance to live in accordance with his perceived body image was a stronger inner motivation to seek amputation and outweighed those concerns. Aesthetic reasons or people's opinion and judgment that might have discouraged him from discussing his condition or proceed with the intervention weren't strong enough to dissuade him from seeking a way to get rid of those fingers. This process also taught him that he was accepted by people around him as he truly is.

According to Edwards & et al., parallels have been drawn between BID and conditions such as anorexia, gender dysphoria, schizophrenia, depression, depersonalization, fixed dystonias, complex regional pain syndromes and body dysmorphic disorder. ⁹ In these disorders, surgery is not recommended and can lead to various complications. ⁹ The patient described in this paper felt as he exhibited features of post‐traumatic stress disorder. His research led him to draw comparisons between his overall state and that of people undergoing surgery for gender dysphoria.

Amputating a healthy limb for psychological distress is uncommon. ⁴ The Hippocratic principle of “First, do no harm” is sometimes cited due to concerns about regret, disability, or financial burden. ⁷ However, BID sufferers endure significant distress and may seek self‐amputation or black‐market amputations. ³ Risks include death. ³ , ⁴ , ⁵ , ⁷ , ¹² Pathology arises when adaptive capacities are exceeded. Being outside the norm doesn't necessarily imply dysfunction or disease. ¹² If amputation improves function and alleviates identity or body image suffering, it aligns with medical practice's purpose of promoting health and adaptation. ¹³ Resisting changes may hinder health promotion. Health can be defined by normal biological functioning or an individual's ability to adapt to circumstances. ¹² Individuals with BID request changes with informed consent, being capable of decision‐making and are seeking this to improve their overall functioning. The surgical intervention can, in some case, effectively relieve their suffering. Theoretically, it could be the case especially when their envisioned lifestyle aligns with the desired appearance, making the transition smoother than a traumatic, unplanned amputation. ¹⁴ Empirical data confirming this as evidence is scarce, as is documentation of long‐term outcomes.

For delusional patients, amputation could violate non‐maleficence principle if it is not in their best interest. However, for BID patients who have psychological distress and do not use their anatomy part as it is, but rather as they perceive it, amputation could theoretically alleviate suffering without additional harm or disability. ¹² In such instances, it could harmonize their physical body with their conceptualization of it and the efforts they already exert, as well as the consequences of using, abstaining from using, or pretending the absence of the body part they feel don't belong to them, thus facilitating adaptation.

Getting more evidence about high satisfaction rates and improved quality of life could lead many patients and physicians to consider earlier amputation, factoring in the costs of living with depression, dysfunction associated with BID and less invasive supportive treatments like pharmacotherapy and psychotherapy. ⁴ , ⁶ , ⁷ , ¹⁵ In BID, different limb perception and lack of use may cause limb weakness. Pretending to have amputation or paralysis can negatively impact productivity, leisure, and social interactions, increasing injury risk and risky behaviors. Yet, in some case, pretending can align their perceived body representation with the desired state, easing distress. ⁴ For this patient, attempting to pretend resulted in localized pain. Coping strategies did not successfully alleviate his distress. Surgery proved to be a highly satisfying curative treatment for him.

6. CONCLUSION

This case is the first described about digits amputation and serves as a straightforward illustration of a clinical BID presentation that engaged in noninvasive treatments without success, then clearly benefited from elective surgery. He is now living a life free from distressing preoccupations about his fingers, with all his symptoms related to BID resolved. The amputation enabled him to live in alignment with his perceived identity. It increased existing insight regarding the extent of his suffering and allowed him to share this with the medical team. Disseminating knowledge about BID can benefit affected individuals, fostering understanding and support by the medical teams. It gives an opportunity to evolve and make the healthcare system more inclusive with this type of presentations by broadening the definition of health through various models. Recognizing and addressing the unique needs of those patients can lead to a future where they can live with more dignity, respect, and optimal well‐being.

AUTHOR CONTRIBUTIONS

Nadia Nadeau: Conceptualization; data curation; formal analysis; funding acquisition; investigation; methodology; project administration; resources; software; supervision; validation; visualization; writing – original draft; writing – review and editing.

FUNDING INFORMATION

No grand support was obtained for this paper.

CONFLICT OF INTEREST STATEMENT

Dre Nadia Nadeau reports no financial relationship with commercial interests.

ETHICS STATEMENT

This case report adheres to ethical principles outlined in the Declaration of Helsinki. All procedures performed involving the patient were conducted in accordance with ethical standards and with the patient's informed consent.

CONSENT

Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.

COPYRIGHT TRANSFERT

I hereby transfer and assign all copyright ownership of the case report, including all rights, title, and interest therein, to the Journal. I understand that this transfer enables the journal to handle the publication process efficiently and to protect the intellectual property rights of the authors and the journal.

PATIENT CONSCENT FOR PUBLICATION

I, Nadia Nadeau, hereby confirm that I have obtained written informed consent from the patient involved in this case report for the publication of their clinical information in this manuscript. The patient has been thoroughly briefed on the purpose, nature, and potential risks of publication. Additionally, the patient was informed that efforts would be made to ensure anonymity. However, due to the distinctive nature of the case, there is a possibility that individuals familiar with the patient could identify him. To safeguard privacy, identifiable details have been appropriately anonymized. The consent form is available upon request.

Supporting information

Data S1.

Nadeau N. Successful treatment of body integrity dysphoria with amputation: A case report. Clin Case Rep. 2024;12:e8720. doi: 10.1002/ccr3.8720

DATA AVAILABILITY STATEMENT

Data sharing not applicable to this article as no datasets were generated or analysed during the current study.