Hypersexuality from resection of left occipital arteriovenous malformation
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- Cao, Y., Zhu, Z., Wang, R. et al. Neurosurg Rev (2010) 33: 107. doi:10.1007/s10143-009-0232-2
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The authors report their experience on one patient with hypersexuality from resection of left occipital arteriovenous malformation. To the best of our knowledge, this is the first case reported in the literature. A 35-year-old right-handed female farmer suffered a sudden left occipital hemorrhage with subarachnoid and subdural hemorrhages of the left hemisphere. Transient left uncal herniation occurred at the onset and was released by conservative treatment. Digital subtraction angiography showed a brain left occipital arteriovenous malformation. After microsurgical resection of the arteriovenous malformation, the patient developed hypersexual behavior. Positron emission tomography showed hypermetabolism in the left frontal region and left posterior hippocampal gyrus and hypometabolism in the left anterior hippocampal gyrus and the left occipital surgical area. Theories concerning normal pressure perfusion breakthrough and specific areas in the brain responsible for the human sexual response are discussed.
KeywordsArteriovenous malformationHypersexualityNormal pressure perfusion breakthroughPositron emission tomography
Arteriovenous malformations (AVMs) of the brain are lesions composed of a coiled batch of arteries and veins, joined by arteriovenous fistulas and partially separated by sclerotic tissue . AVMs are pulsatile vascular masses that impact on brain hemodynamics and physiology in many ways . Removal of AVMs of the brain also can produce a variety of anatomic and physiologic changes, which include patient’s neurological deficit and neuropsychological or cognitive dysfunction. The major neurological deficit after surgical removal of AVMs includes hemiparalysis, aphasia, hemianopia, and cranial nerve dysfunction . There are also a few neuropsychological or cognitive changes reported from the resection of AVM [2, 4, 10, 17]. To the best of our knowledge, there are no hypersexuality from resection of AVM reported in the literature. We reported a case of brain occipital AVM that the female patient had hypersexual behavior after microsurgical removal of the AVM. We have discussed the postoperative changes of cerebral perfusion and metabolism on perifocal and remote brain tissue in this patient.
FSFI domain scores and full-scale scores: first versus second
First individual domain scores
Second individual domain scores
3, 4, 5, 6
7, 8, 9, 10
11, 12, 13
14, 15, 16
17, 18, 19
We treated the patient with sertraline hydrochloride tablets, 50 mg/day orally, and risperidone tablets 1 mg orally at bedtime. After 30 days, the patient showed relief of hypersexual symptoms to some extent. On June 22, 2009, she completed the FSFI questionnaire once more; her FSFI score dropped to 24.0 (Table 1).
AVMs can cause widespread derangements of the cerebral circulation [3, 7]. Lower resistance and high-flow malformations may produce relative ischemia in the brain adjacent to or distant from the malformation. Clinically, this may produce episodes of transient ischemia, or chronic ischemia may result in insidious cognitive decline . Resection of AVMs may have an effect on the cerebral circulation. Abrupt removal of the AVM from the cerebral circulation by either open or endovascular surgery is complicated by a transient circulatory derangement commonly called normal pressure perfusion breakthrough (NPPB) in approximately 10% of cases . In mild forms, NPPB produces migraine-like headaches and may be associated with seizures. In more severe cases, NPPB may result in extensive brain edema, hemorrhage, or both in regions of the brain adjacent to or distant from the AVM . The decreased perfusion in areas distant from an AVM and the reperfusion of hypoperfused tissue following obliteration of AVM feeding arteries can produce a few neuropsychological or cognitive changes [2, 4, 7, 10, 17]. We reported one case of hypersexuality from resection of left occipital AVM, which was not reported in the literature to our knowledge.
Hypersexuality, which is characterized by an unbearable need for frequent genital stimulation, is often manifested as an increase in frequency or change in types of sexual behaviors, which fail to produce long-term sexual and emotional satisfaction. These behaviors include compulsive masturbation, compulsive sex with prostitutes, anonymous sex with multiple partners, multiple affairs outside a committed relationship, frequent patronizing of sexually oriented establishments, habitual exhibitionism, habitual voyeurism, inappropriate sexual touching, sexual abuse of children, and rape. In addition to these, fantasy sex, prostitution, pedophilia, masochism, fetishes, and sex with animals may also be associated behaviors. It is a combination of these behaviors along with the compulsivity that comprises hypersexuality .
As is the case with many other psychiatric disorders, the etiology of hypersexuality is complex and involves a variety of physiological and psychological mechanisms. Frontal lobe dysfunction can lead to disinhibition of sexual behavior and hypersexual behavior. Temporal lobe abnormalities, which have been associated with hypersexuality, also seem to be involved in development of various fetishes, paraphilias, and pedophilia. Head traumas, brain surgeries, and medications have been associated with hypersexuality. Onset of hypersexuality has been associated with frontal lobe lesions, frontal and temporal lesions, temporal lobe epilepsy, dementia, Klüver–Bucy syndrome, multiple lesions in multiple sclerosis, and treatment of Parkinson’s disease with dopaminergic agents [6, 8, 9, 11, 13, 14, 16, 19].
Our patient’ behaviors met the definition for hypersexuality resulting from removal of left occipital AVM. We do not think it is the surgical injury to the occipital lobe that is the cause of the hypersexuality. There are no reports that occipital lobe is associated with sexual behavior. Besides the surgical damage to the occipital lobe, there were two major events in the patient. Firstly, there was an uncal herniation before the surgery, which might have injury to the left uncus and hippocamp of temporal lobe. Secondly, it is the abrupt removal of the AVM from the cerebral circulation might lead to NPPB in regions of the brain adjacent to or distant from the AVM. Although CT perfusion imaging did not show any hemodynamic changes in the patient, the PET of the brain, which seemed to be more sensitive showed hypermetabolism in the left frontal region and left posterior hippocamp and hypometabolism in anterior hippocamp, which all areas related with human sexual response. PET seemed to provide imaging evidences to explain the patient’s disinhibition of sexual behavior and hypersexual behavior. We supposed that, in our case, the hypermetabolism in the frontal lobe may have two possibilities: One was that the reperfusion of frontal lobe after the resection of occipital AVM led to active brain function, and the other was that it was secondary to functional change in the uncus and hippocamp. The hyper- or hypo-metabolism in the hippocamp was more complicated; it might be the result of a mix of the injury of hippocamp from uncal herniation and reperfusion of hippocamp. We think that both of the events contributed to the hypersexuality of the patient. Derangements of the cerebral circulation and reperfusion of hypoperfused tissue following obliteration of AVM may facilitate development of preexisting injury of the uncus and hippocamp of the temporal lobe. NPPB perhaps triggered the symptom of hypersexuality. This could explain why hypersexuality happened 1 day after the resection of the AVM and not after the onset of the uncal herniation.
Specific areas in the brain responsible for the human sexual response are poorly understood and much of our information comes from lesion studies in animals. Animal studies are not completely applicable to humans, however, and even non-human primates have sexual behaviors that differ markedly from those of men and women . Hyposexuality is common with injury to the brain, although the lesions seen with this condition are sufficiently variable that it is difficult to identify one specific area in the brain responsible for the diminished sexual interest, whereas hypersexual behavior are much less common following brain injury . Study of hypersexuality in humans after focal brain injury will give valuable clues about areas of the brain involved in the normal sexual response. Case studies and the literature provide evidence that different brain systems may play a role in this disorder. Frontal lesions may be accompanied by disinhibition, including impulsive hypersexual response to external cues . Our patient’s presentation feature is only the development of increasing libido with fairly normal cognitive function; meanwhile, the brain PET presented local hot spot in the frontal lobe that did not directly surgically damaged, which will contribute to the understanding of this evolving and complex field.