Background: Body integrity identity disorder (BIID) is a rare and unusual psychiatric condition characterized by a persistent desire to acquire a physical disability (e.g., amputation, paraplegia) since childhood that to date has not been formally described in the psychiatric nosology. Most BIID sufferers experience a chronic and dysphoric sense of inappropriateness regarding their being able-bodied, and many have been driven to actualize their desired disability through surreptitious surgical or other more dangerous methods. This review aims to characterize the history and phenomenology of this condition, to present its differential diagnosis, and to consider possible etiologies, treatment options, and ethical considerations. Sampling and Method: Review of the psychiatric and neurological literature. Results: A growing body of data suggests the existence of a discrete entity with onset by early adolescence and a negative impact on functioning. Parallel neurological conditions and preliminary experimental investigations suggest a possible neurobiological component in at least a portion of cases. While attempts at treatment have been described, no systematic evidence for efficacy has emerged. Discussion: BIID is a unique nosological entity with significant consequences for its sufferers and as such may warrant inclusion in some form in the forthcoming DSM-5 and ICD-11.

1.
First MB: Desire for amputation of a limb: paraphilia, psychosis, or a new type of identity disorder. Psychol Med 2005;35:919–928.
2.
Veale D: A compelling desire for deafness. J Deaf Stud Deaf Educ 2006;11:369–372.
3.
Johnson TW, Wassersug RJ, Roberts LF, utherland MB, First MB: Desire for castration is not a body integrity identity disorder (BIID): a response. J Sex Med 2010;7:853–855.
4.
Money J, Jobaris R, Furth G: Apotemnophilia: two cases of self-demand amputation as paraphilia. J Sex Res 1977;13:115–125.
5.
Money J, Simcoe KW: Acrotomophilia, sex, and disability. Sex Disability 1984;7:285–293.
6.
Wakefield PL, Frank A, Meyers RW: The hobbyist: a euphemism for self-mutilation and fetishism. Bull Meninger Clin 1977;41:539–552.
7.
Dixon D: An erotic attraction to amputees. Sex Disabil 1983;6:3–19.
8.
Johnson TW, Brett MA, Roberts LF, Wassersug RJ: Eunuchs in contemporary society: characterizing men who are voluntarily castrated. Part I. J Sex Med 2007;4:930–945.
9.
Bruno RL: Devotees, pretender, and wannabes: two cases of factitious disability disorder. Sex Disabil 1997;15:243–260.
10.
Brenner I: Upper-extremity self-amputation in a case of dissociative identity disorder. J Clin Psychiatry 1999;60:705–706.
11.
Everaerd W: A case of apotemnophilia: a handicap as sexual preference. Am J Psychother 1983;37:285–293.
12.
Furth G, Smith R: Apotemnophia: Information, Questions, Answers, and Recommendations about Self-Demand Amputation. Bloomington, 1st Books Library, 2000.
13.
Fisher K, Smith R: More work is needed to explain why patients ask for amputation of healthy limbs. BMJ 2000;320:1147.
14.
Elliott C: A new way to be mad. Atlantic Monthly 2000;286.
15.
Dotinga R: Out on a limb (August 29, 2000). salon.com 2000.
16.
First MB: The origin and evolution of the concept of body integrity identity disorder; in Stirn A, Thiel A, Oddo S (eds): Body Integrity Identity Disorder: Psychological, Neurobiological, Ethical and Legal Aspects. Lengerich, Pabst Science Publishers, 2009, pp 49–57.
17.
Berger BD, Lehrmann G, Larson L, Alverno L, Tsao CL: Nonpsychotic, nonparaphilic self-amputation and the internet. Compr Psychiatry 2005;46:380–383.
18.
Braam AW, Visser S, Cath DC, Hoogendijk WJ: Investigation of the syndrome of apotemnophilia and course of cognitive-behavioral therapy. Psychopathology 2006;39:32–37.
19.
Braam A, de Boer-Kreeft N: Case report – the ultimate relief; resolution of the apotemnophilia syndrome; in Stirn A, Thiel A, Oddo S (eds): Body Integrity Identity Disorder: Psychological, Neurobiological, Ethical, and Legal Aspects. Lengerich, Pabst Science Publishers, 2009.
20.
Clervoy P, Vautier V, Naudin J: Trouble de l’identité et de l’intégrité corporelle: interrogations pathogéniques et enjeux éthiques autour d’une pathologie émergente. Ann Medico-Psychol 2009:167.
21.
Kasten E: Body Integrity Identity Disorder (BIID): Befragung von Betroffenen und Erklärungsansätze. Fortschr Neurol Psychiatr 2009;77:16–24.
22.
Sorene ED, Heras-Palou C, Burke FD: Self-amputation of a healthy hand: a case of body integrity identity disorder. J Hand Surg Br 2006;31:593–595.
23.
Schlozman SC: Upper extremity self-amputation and replantation: 2 case reports and a review of the literature. J Clin Psychiatry 1998;59:681–686.
24.
Stewart DE, Lowrey MR: Replantation surgery following self-inflicted amputation. Can J Psychiatry 1980;25:143–150.
25.
Hunter GA, Kennard AB: Mania operativa: an uncommon unrecognized cause of limb amputation. Can J Surg 1982;25:92–93.
26.
Parsons JA, Brown WA, Sirota AB: Inappropriate amputation requests. Psychosomatics 1981;22:822–823.
27.
Bensler JM, Paauw DS: Apotemnophilia masquerading as medical morbidity. South Med J 2003;96:674–676.
28.
Kolla NJ, Zucker KJ: Desire for non-mutilative disability in a nonhomosexual, male-to-female transsexual. Arch Sex Behav 2009;38:1057-1063.
29.
Storm S, Weiss MD: Self-inflicted tourniquet paralysis mimicking acute demyelinating polyneuropathy. Muscle Nerve 2003;27:339–344.
30.
Wise TN, Kalyanam RC: Amputee fetishism and genital mutilation: case report and literature review. J Sex Marital Ther 2000;26:339–344.
31.
Stirn A, Thiel A, Oddo S: Body Integrity Identity Disorder: Psychological, Neurobiological, Ethical, and Legal Aspects. Lengerich, Pabst Science Publishers, 2009.
32.
Lawrence AA: Clinical and theoretical parallels between desire for limb amputation and gender identity disorder. Arch Sex Behav 2006;35:263–278.
33.
De Guypere G, Van Hemelrijck M, Michel A, Carael B, Heylens G, Rubens R: Prevalence and demography of transsexualism in Belgium. Eur Psychiatry 2007;22:137–141.
34.
Wilson P, Sharp C, Carr ST: The prevalence of gender dysphoric in Scotland: a primary care study. Br J Gen Pract 1999;49:991–992.
35.
Jaspers KI: General Psychopathology. Chicago, University of Chicago Press, 1968.
36.
American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders, ed 4, text revision. Washington, American Psychiatric Association, 2000.
37.
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;42:322–324.
38.
Strain JJ: Care of the psychotic self-amputee undergoing replantation. Ann Surg 1983;197:210–214.
39.
Spitzer M: On defining delusions. Compr Psychiatry 1990;31:377–397.
40.
McKenna PJ: Disorders with overvalued ideas. Br J Psychiatry 1984;145:579–585.
41.
Borras L, Huguelet P, Eytan A: Delusional ‘pseudotranssexualism’ in schizophrenia. Psychiatry 2007;70:175–179.
42.
Phillips KA, Menard W, Fay C, Weisberg R: Demographic characteristics, phenomenology, comorbidity, and family history in 200 individuals with body dysmorphic disorder. Psychosomatics 2005;46:317–325.
43.
Phillips KA, Kaye W: The relationship of body dysmorphic disorder and eating disorders to obsessive-compulsive disorder. CNS Spectrums 2007;12:347–358.
44.
Phillips KA, Diaz SF: Gender differences in body dysmorphic disorder. J Nerv Ment Dis 1997;185:570–577.
45.
Smith YL, van Goozen SH, Kuiper AJ, Cohen-Kettenis PT: Transsexual subtypes: clinical and theoretical significance. Psychiatry Res 2005;137:151–160.
46.
Blanchard R: The concept of autogynephilia and the typology of male gender dysphoria. J Nerv Ment Dis 1989;1989:616–623.
47.
Walworth JR: Sex-reassignment surgery in male-to-female transsexuals: client satisfaction in relation to selection criteria; in Bullough B, Bullough VL (eds): Gender Blending. Amherst, Prometheus Books, 1997.
48.
Blanchard R, Clemmensen LH, Steiner BW: Social desirability response set and systematic distortion in the self-report of adult male gender patients. Arch Sex Behavior 1985;14:505–516.
49.
Sean: A quick recap of my life to date. Available at transabled.org <http://transabled.org/thoughts/sean-thoughts/a-quick-recap-of-my-life-up-to-date.htm> (accessed September 21, 2010). 2010.
50.
Barbet T, Gal B, Dendoncker-Viry S, Masquelet AC, Gatt MT, Moro MR: Apotemnophilia as a contemporary frame for psychological suffering. Encephale 2007;33:609–615.
51.
Mitchell SW, Morehouse GR, Keen WW: Reflex Paralysis Circular, No 6. Surgeon’s General Office, 1864. Reprinted by Yale University School of Medicine, 1941.
52.
Babinski J, Froment J: Hysteria or Pithiatism and Reflex Nervous Disorders in the Neurology of War. London, University of London Press, 1918.
53.
Leontev AN, Zaporozhets AV: Rehabilitation of Hand Function. New York, Pergamon Press, 1960.
54.
Saadah ES, Melzack R: Phantom limb experiences in congenital limb-deficient adults. Cortex 1994;30:479–485.
55.
Vallar G, Ronchi R: Somatoparaphrtenia: a body delusion – a review of the neuropsychological literature. Exp Brain Res 2009;192:533–551.
56.
Vuilleumier P, Reverdin A, Landis T: Four legs: illusory reduplication of the lower limbs after bilateral partietal lobe damage. Arch Neurol 1997;62:301–306.
57.
Assal F, Schwartz S, Vuilleumier V: Moving with or without will: functional neural correlates of alien hand syndrome. Ann Neurol 2007;54:1543–1547.
58.
Cappa S, Sterzi R, Vallar G, Bisiach E: Remission of hemineglect and anosognosia during vestibular stimulation. Neuropsychologia 1987;25:775–782.
59.
Ramachandran VS, McGeoch PD: Can vestibular caloric stimulation be used to treat apotemnophilia? Med Hypotheses 2007;69:250–252.
60.
Brang D, McGeoch PD, Ramachandran VS: Apotemnophilia: a neurological disorder. Neuroreport 2008;19:1305–1306.
61.
McGeoch PD, Brang D, Song T, Lee R, Huang M, Ramachandran VS: Apotemnophilia – the neurological basis of a ‘psychological’ disorder. Nature Precedings 2009;posted March 17, 2009.
62.
Ramachandran VS, Brang D, McGeoch PD, Rosar W: Sexual and food preference in apotemnophilia and anorexia: interactions between ‘beliefs’ and ‘needs’ regulated by two-way connections between body image and limbic structures. Perception 2009;38:775–777.
63.
Blanke O, Morgenthaler FD, Brugger P, Overney LS: Preliminary evidence for a fronto-partietal dysfunction in able-bodied participants with a desire for limb amputation. J Neuropsychol 2009;3:181–200.
64.
Poldrack RA: Can cognitive processes be inferred from neuroimaging data? Trends Cogn Sci 2006;10:59–63.
65.
Bayne T, Levy N: Amputees by choice: body integrity identity disorder and the ethics of amputation. J Appl Philos 2005;22:75–86.
66.
Muller S: Body integrity identity disorder (BIID) – is the amputation of healthy limbs ethically justified? Am J Bioethics 2009;9:36–43.
67.
Ofgang K: C.A. upholds doctor’s conviction in botched Mexican surgery. Metropolitan News-Enterprise, August 6, 2001. http://www.metnews.com/articles/brow0806.htm.
68.
Ryan CJ: Out on a limb. Neuroethics 2009;2:21–33.
69.
Bridy A: Confounding extremities: surgery at the medico-ethical limits of self-modification. J Law Med Ethics 2004;32:148–158.
70.
Meyer-Bahlburg HF: From mental disorder to iatrogenic hypogonadism: dilemmas in conceptualizing gender identity variants as psychiatric conditions. Arch Sex Behav 2009;39:461–476.
Copyright / Drug Dosage / Disclaimer
Copyright: All rights reserved. No part of this publication may be translated into other languages, reproduced or utilized in any form or by any means, electronic or mechanical, including photocopying, recording, microcopying, or by any information storage and retrieval system, without permission in writing from the publisher.
Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug.
Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.
You do not currently have access to this content.