Apotemnophilia, body integrity identity disorder or xenomelia? Psychiatric and neurologic etiologies face each other

Figures & data

Table 1 Studies favoring a psychological/psychiatric etiology for the desire to amputate a healthy limb

Author(s)	Year	Sample size	Clinical features	Limb	Psychological/psychiatric comorbidities/diagnosis	Sexual difficulties	Method	Results
Money^Citation7	1977	n=2	Unilateral	Leg	No	Yes	Interview (on the phone in one case)	Paraphilia (apotemnophilia)
Wakefield et al^Citation44	1977	n=1	Unilateral	Finger, toe	No	Yes	Case description	Apotemnophilia
Beresford^Citation45	1980	n=1	Unilateral	Leg	Yes	No	Consultation	Schizoid personality disorder/BIID
Everaerd^Citation10	1983	n=1	Unilateral	Leg	No	Yes	Case description	Bodily identity disturbance
Bruno^Citation17	1997	n=2	Unilateral/bilateral	Arms, legs	Yes (1 individual)	Yes/not known	Case description	Factitious disability disorders
Bensler and Paauw^Citation11,^*	2003	n=1	Bilateral	Legs	No	Yes	Case description	Apotemnophilia
Berger et al^Citation13	2005	n=1	Bilateral/unilateral	Legs, arm	Yes	Yes	Case description	Apotemnophilia
Braam et al^Citation12	2006	n=1	Unilateral	Leg	No	No	Case description	Apotemnophilia
Clervoy et al^Citation16	2009	n=1	Bilateral	Legs	No	No	Case description	BIID
Kasten and Stirn^Citation46,#	2009	n=1	Unilateral	Leg	No	Not known	Case description	Apotemnophilia
Sorene et al^Citation15	2006	n=1	Unilateral	Hand/leg	Not known	Yes	Case description	BIID
First^Citation2	2005	n=52	Mixed	Leg, arm, toe, fingers	Yes (11 individuals)	Yes (15 individuals)	Structured telephone interview	BIID
Baubet et al^Citation47	2007	n=1	Bilateral	Legs	Yes	No	Case description	Cluster B personality disorder, eating disorder, BIID
Wise and Kalyanam^Citation18,^*	2000	n=1	–	Penis	Yes	Yes	Case description	Major depressive disorder
Storm and Weiss^Citation48,^*	2003	n=1	Bilateral	Legs	Not known	Yes	Case description and neurological examination	Apotemnophilia

Notes: Clinical features of the described individuals and the methodology adopted to study them are reported. The final column indicates which diagnosis has been chosen by the authors of each report. Studies recognized as unclear apotemnophilia/BIID following analysis⁵ are reported with an asterisk.

# Data for this publication are derived from the study of Bou Khalil and Richa,¹⁹ as the original work is not available in Medline.

Abbreviation: BIID, body integrity identity disorder.

Table 2 Experimental studies supporting a neurological etiology of the condition

Author(s)	Year	Sample size	Clinical features	Limb	Psychological/psychiatric diagnosis	Sexual difficulties	Technique	Task	Results
									Area	Side
Brang et al^Citation26	2008	n=2	Mixed (1 bilateral)	Legs	No	No	SCR	Pain perception	Increased SCR response for the to-be-amputated limb	–
McGeoch et al^Citation3	2011	n=4	Mixed (1 bilateral)	Legs	No	No	MEG	Tactile stimulation	SPL (reduction of activity) Intact visual and somatosensory cortices	R
Aoyama et al^Citation28	2012	n=5	Unilateral	Legs	No (psychiatric, neurological and neuropsychological examination)	No	Psychophysics (just noticeable difference + Points of subjective simultaneity)	Temporal order judgment of tactile stimuli	Altered integration of tactile stimuli on the to- be-amputated leg	–
Hilti et al^Citation30	2013	n=13	Mixed (3 bilateral)	Legs	No (psychiatric, neurological and neuropsychological examination)	No	Surface-based morphometry	No task	SPL IPL Subcentral cortex (SII) Paracentral lobule (SI) Anterior insular cortex	R
									IPL SII	L
van Dijk et al^Citation31	2013	n=5	Unilateral	Leg + 1 individual (finger)	Yes (1 individual) (DSM-IV axis I and II screening)	No	fMRI	Tactile stimulation + motor execution task	Frontoparietal and occipital network (heightened sensitivity) PM (hypofunction)	R, Bilateral
Lenggenhager et al^Citation32	2014	n=13	Mixed (3 bilateral)	Leg	No	No	CVS	Rating of estrangement + body temperature	No diminishing of desire	No lateralization effects
Bottini et al^Citation33	2014	n=7	Mixed (1 bilateral)	Leg/arm	Yes (1 individual) (semi-structured psychiatric interview)	No	Single subject analysis (Crawford t-test)	Facial emotion recognition task + disgust rating task + disgust scale	Altered disgust rating only in unilateral individuals	–

Abbreviations: DSM, Diagnostic and Statistical Manual of Mental Disorders; SCR, skin conductance responses; MEG, magnetoencephalography; fMRI, functional magnetic resonance; CVS, caloric vestibular stimulation; PM, premotor cortex; SPL, superior parietal lobe; IPL, inferior parietal lobe; SII, secondary somatosensory cortex; SI, primary somatosensory cortex; R, right hemisphere; L, left hemisphere.

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