pt. 1A 25-Year-Old Woman With Hallucinations, Hypersexuality, Nightmares, and a
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Date: 06/18/04
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Date: 18 Jun 2004 03:55:48 GMT
Subj: pt. 1A 25-Year-Old Woman With Hallucinations, Hypersexuality,
Nightmares, and a
American Journal of Psychiatry 153:4, April 1996
American Journal of Psychiatry
Copyright © 1996 American Psychiatric Association.
------------------------------------------------------------------------
Volume 153(4) April 1996 pp 545-551
------------------------------------------------------------------------
A 25-Year-Old Woman With Hallucinations, Hypersexuality, Nightmares,
and a Rash
[Clinical Case Conference]
Stein, Sara L. MD; Solvason, Hugh B. MD, PhD; Biggart, Elizabeth PhD;
Spiegel, David MD
Received July 15, 1995; revision received Jan. 16, 1996; accepted Jan.
29, 1996. From the Department of Psychiatry and Behavioral Sciences,
Stanford University School of Medicine. Address reprint requests to Dr.
Spiegel, Department of Psychiatry and Behavioral Sciences, Stanford
University School of Medicine, 401 Quarry Rd., Stanford, CA 94305-5544.
Dr. Stein is supported by NIMH Institutional Training Grant MH-19908.
The authors thank Brian Fallon, M.D., and Andrew C. McNeil, M.D., for
their assistance with this case.
Outline
CASE HISTORY
History of the Present Illness
Past Psychiatric History
Social and Medical History
Neuropsychiatric and Laboratory Testing
Mental Status Examination
DISCUSSION
REFERENCES
Graphics
Figure 1
Figure 2
Figure 3
------------------------------------------------------------------------
CASE HISTORY
History of the Present Illness
A 25-year-old Asian-American single woman, diagnosed with chronic
paranoid schizophrenia since age 19, was referred from the inpatient
psychiatric unit for evaluation of atypical hallucinations. She had
been admitted 3 days earlier after an intentional overdose of 32 over-
the-counter sleeping pills. In the emergency room the patient suffered
a witnessed grand mal seizure without incontinence, which was followed
by a brief period of confusion and combativeness. A computerized
tomography (CT) scan of the brain and an EEG at the referring hospital
were reportedly normal. A repeat EEG at our hospital demonstrated
intermittent asymmetry with posterior right-hemisphere background
slowing, which was considered artifact.
The patient expressed a delusional belief that she was controlled by an
attractive, popular male former high school teacher who had put a curse
on her family before her birth and was now sexually obsessed with her.
When her eyes were closed, the patient experienced auditory
hallucinations and a single visual hallucination consisting of the
man's face in a black spot that wandered throughout her field of
vision. She constantly experienced vivid tactile hallucinations of this
man molesting her genitally but not having intercourse with her, and
she believed that he videotaped her showering and masturbating in order
to show these ``movies'' to others. When undisturbed, she spent
approximately 18 hours per day in bed, ``dreaming,'' hallucinating, and
masturbating. She suffered from nonamnestic nightmares and spontaneous
disorienting daytime sleep attacks that she blamed on her disrupted
nighttime sleep. She would awaken screaming and confused and appeared
unable to respond to external stimuli. No temporal pattern in these
outbursts was apparent. The symptoms were exacerbated in both frequency
and intensity by anxiety-provoking life events, such as changes in
residence, but never disappeared entirely under normal circumstances.
The patient was raised as a Catholic in a predominantly Buddhist
country before immigrating to the United States at the age of 8 years.
Her family history was remarkable for an older sister who had been
hospitalized for nonpsychotic major depression as an adolescent and for
a male cousin who was schizophrenic and taking clozapine.
Past Psychiatric History
The patient reported that her illness began during the summer following
high school, when her family suffered severe financial reverses and was
forced to move in with another family, which included an alcoholic
father and son. At the time, she was deeply ashamed by these
circumstances and stopped seeing her friends. According to her parents,
premorbidly the patient was a private person but not odd or a loner,
and at no time was she considered to have unusual beliefs. She had many
friends and was a hard worker both at home and at her job in a pizza
parlor, with plans to enter college that fall.
She recalled two unusual experiences from the summer of the onset of
her illness. The first incident occurred one afternoon when she was
lying on top of her bed masturbating and looked up to see the owner of
the house watching her through the open window. This was reported by
the patient with a distinctly isolated affect. She vehemently denied
any further sexual contact with either this man or his adult son, both
alcoholic, and had gone to the gynecologist recently to ``prove I am a
virgin.''
The second unusual experience occurred after an outdoor rock concert
attended by the patient and her friends, when ``a virus went inside of
me.'' Although recreational drugs were being used, the patient stated
that she took only a number of Vivarin (over-the-counter caffeine
tablets) in order to ``keep up'' with her friends. She denied any
history of illegal substance abuse and had had multiple negative urine
toxicology screens over the previous 6 years. Three days after the
concert the patient awakened with ``the flu,'' which consisted of
general malaise, fever, arthralgias in her knees bilaterally, and an
urticarial rash. The patient also noted that her ``jaw was going to the
side,'' an apparent facial hemiparesis [1]. The family did not have
health insurance and treated the patient with acetaminophen and
calamine lotion. The rash, fever, and facial droop resolved within a
week, but the patient continued to experience somnolence and malaise.
Three weeks after the onset, the patient sought medical treatment for
possible Lyme disease at a family physician's office. The medical
workup was reportedly negative, and no treatment was obtained.
She remained in a profoundly depressed, vegetative state, during which
she was unable to stop sleeping or eating. The patient attempted to go
to work but began to feel that her co-workers were talking about her
behind her back and that they wanted to fire her. This was the first
time that she heard the voice of the man who tormented her. She
underwent psychiatric evaluation and was diagnosed with chronic
paranoid schizophrenia.
For the next 6 years, despite her general compliance, her psychotic
symptoms remained refractory to numerous typical neuroleptics at both
low and high doses. Her sleep disturbance was unresponsive to
behavioral techniques, trazodone, benzodiazepines, antihistamines, and
chloral hydrate. Records indicated no trial of antidepressants, mood
stabilizers, or atypical antipsychotic medications, such as risperidone
or clozapine. Treatment with psychotropic medication had been
discontinued 1 year before this admission because of the apparent lack
of benefit; thus, the patient was receiving purely supportive
psychiatric care.
Social and Medical History
The patient was raised in a stable family environment with both parents
and several siblings; however, the family had undergone multiple
financial reverses since immigrating to the United States. She had a
childhood history of regular corporal punishment by her nanny but no
history of sexual abuse. Two traumatic events had occurred in her
childhood. The first event occurred at the age of 5, after an argument
with her 7-year-old sister over a bicycle. The patient, fearful that
she would be ``caned'' by her nanny for being disagreeable, angrily
gave up her turn on the bicycle and stated that she wished harm on her
sister, who then rode 100 yards down the street and was fatally struck
by an oncoming truck. As a result of this terrible coincidence, the
patient had lived the past 20 years of her life secretly believing that
she had ``used her powers'' to cause her sister's death.
The second event occurred at the age of 7, when the patient contracted
a severe case of dengue fever, a mosquito-borne flu-like virus that is
most often self-limited but can be fatal. She was hospitalized for 1
month and required intravenous hydration and supportive care, but she
had no noticeable long-term adverse sequelae.
Neuropsychiatric and Laboratory Testing
The patient's initial workup 6 years earlier and the workup during this
hospital admission included a CT scan of the brain, EEG, complete blood
count and chemistries, thyroid function tests, serum and urine
toxicologies, folic acid and vitamin B12 measurements, and an ECG; all
results were within normal limits. The patient was noted to have
markedly positive serum titers for Borrelia burgdorferi (Lyme disease);
titer optical density was 0.393 with a negative cutoff of 0.103. (The
negative cutoff is defined as 3 standard deviations above the mean for
10 negative controls.) Serum Western blot testing showed faint IgM
(immunoglobulin) reactivity to three antibodies (18-kDa, 41-kDa, 72-
kDa) and IgG reactivity to two antibodies (34-kDa and 70-kDa). In order
to eliminate potential antibody cross-reactivity, the following tests
were performed: HIV, rubella titers, antinuclear antibody test, and
fluorescent Treponema antibody absorption test; the results were
negative. CSF was notable for an absence of white cells and organisms,
normal protein and glucose content, and a positive CSF ELISA screen for
Lyme disease (optical density, 0.237; negative cutoff, 0.084). CSF
Western blot was negative for IgM reactivity, but mild IgG reactivity
was present (41-kDa, 45-kDa, 58-kDa, and other Centers for Disease
Control and Prevention [CDC] nonspecific bands). Polymerase chain
reaction testing of her CSF was also negative. These laboratory results
were not sufficient to meet the CDC criteria for infection with B.
burgdorferi, and no further investigation or treatment was pursued at
this point.
Neuropsychological testing was conducted, and the results indicated an
inconsistent pattern of mild right-hemisphere brain dysfunction. During
testing, the patient complained of considerable fatigue and, when
allowed a short break, immediately fell soundly asleep without changing
position. Mild impairment in nonverbal information processing and
problem solving, as demonstrated on the WAIS-R, was noted. She was
found to be in the low average range of intellectual functioning, with
normal verbal fluency and no evidence of verbal word reversal or
dyslexia. The patient showed mild constructional dyspraxia when she
initially had difficulty drawing a clock, but she was able to self-
correct and produce an intact and accurate drawing. On the Visual Form
Discrimination Test she made both minor peripheral and major rotation
errors when asked to match a series of simple geometric figures;
however, she was able to correctly draw geometric figures on the
Wechsler Memory Scale-Revised. The scale showed intact memory
abilities, as well as mental flexibility in trail making; however, she
was somewhat slow in her completion of part A, scoring in the lowest
tenth percentile. Her attention and concentration were also within
normal limits, and she performed better than would be expected given
her intelligence.
Mental Status Examination
The patient was a childlike, cheerful, animated young woman who gave a
detailed and fluent history. No facial asymmetry was apparent. The
interview was remarkable for her isolation of affect, inappropriate
smiling, and continuous self-stimulatory pelvic rocking. She
demonstrated no euphoria, irritability, pressured speech, or flight of
ideas and had no history of clear-cut manic episodes. She reported the
delusions of persecution noted already. She had no difficulty
interpreting proverbs or similarities.
The atypical and multisensory nature of her hallucinations (auditory,
visual, and tactile) and the dissociative quality of her nightly
screaming episodes were felt to be inconsistent with a diagnosis of
schizophrenia; rather, the symptoms resembled psychotic-like
dissociative spells [2,3]. Because hypnotizability is rarely seen in
schizophrenia but is strongly associated with posttraumatic stress
disorder (PTSD) and with severe dissociative states, the patient's
hypnotizability was assessed by using the Hypnotic Induction Profile
[4]. She was found to be highly hypnotizable and, while under trance,
appeared to recall the owner of the house who had molested her while
she was sleeping. She said the man lay down on top of her and began to
molest her genitally but did not have intercourse with her. She
reported that she was afraid to move because ``he has a horrible look
in his eyes and wants to hurt me.'' She stated that she was unable to
wake up even though she knew what was happening to her. The patient
said that she immediately felt ``filthy and disgusting'' and was very
angry. When asked what she would like to do at that point, she replied,
``Have a gun, shoot him in the head, shoot him in the genitals, shoot
him, and then shoot myself.''
After the hypnosis, for a brief period the patient remembered the
events as she had described them; then she became extremely agitated,
delusional, and inconsolable. She refused to be put into a trance
again. She firmly denied that any sexual molestation had occurred,
stating, ``I would know if it happened.'' Her family was unaware of any
such event, and the molestation presently remains unconfirmed. A
provisional diagnosis of PTSD with psychotic features was made, and
multimodal treatment was initiated; it consisted of broad-spectrum
pharmacotherapy to reduce symptoms [5], weekly psychotherapy directed
at uncovering and reframing traumatic events, and stricter case
management and milieu therapy.
Fluoxetine was begun at 10 mg/day, with planned maximal titration over
3 to 4 months to minimize both her sexual compulsivity and her mood
disturbance [6-8]. However, at 60 mg of fluoxetine daily, the patient
experienced a substantial diminution of sexual responsiveness. She
became extremely angry and abruptly stopped taking all of her
medications, requiring emergency hospitalization. A lower dose, 40
mg/day, provided resolution of her mood disturbance but no change in
her erotic preoccupation and continuous masturbation. In addition, a
beta blocker (propranolol, 10 mg t.i.d.) was used to block her
hyperarousal and sympathetic response. On the basis of her previous
lack of response to typical neuroleptics, treatment with an atypical
antipsychotic (risperidone, 3-6 mg/day) was initiated. At 3 mg/day of
risperidone the patient experienced extrapy-ramidal side effects and
refused to increase the dose. Within the first few months the patient
stopped taking all of her medications because ``they're not working.''
Brief trials of lithium carbonate and valproic acid [9] were conducted
in attempts to achieve mood stabilization, but the patient refused to
continue taking either drug. Because of her difficulty with compliance,
clozapine was not initiated.
Over a 10-month period the patient gradually became less anxious and
more confident and interactive. Her delusional system remained
unchanged, and although her nightmares and sleeping fits continued, she
was able to distract herself with daily activities. She made several
unsuccessful attempts to pursue work and school but complained of
daytime somnolence and difficulty concentrating on complex tasks.
Consultation was obtained for evaluation of possible narcolepsy. The
patient's daytime sleepiness was characterized by sudden onset,
hallucinations, difficulty awakening, and confusion and disorientation
with infrequent cataplexy but was not marked by automatisms, as may be
seen in temporal lobe epilepsy [10]. Overnight polysomnogram (Multiple
Sleep Latency Test) revealed a short sleep latency in a series of five
naps and REM sleep in three of the five [11]. A diagnosis of narcolepsy-
cataplexy syndrome was made, and the patient began taking clomipramine,
50 mg b.i.d. [12]. Initially she reported improved sleep at night,
including a dramatic decrease in nightmares and night terrors; however,
when under additional psychosocial stress, she experienced symptom
exacerbation.
Because of the patient's incomplete response to treatment for PTSD and
the association of narcolepsy with CNS injury (such as infection)
[12,13], the possibility of Lyme disease as an etiology was
reconsidered despite the original inconclusive antibody titers. A
magnetic resonance imaging (MRI) scan of the brain with gadolinium
contrast was obtained and showed scattered 1-2-ml punctate foci of
signal abnormality in the subcortical white matter on T2-weighted
images, predominantly in the frontal lobes Figure 1. No ventricular
enlargement or abnormal parenchymal or leptomeningeal enhancement was
noted.
[Help with image viewing]
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Figure 1. T2-Weighted MRI Image Demonstrating White Matter Lesion
(arrow) in Deep Right Frontal Lobe
------------------------------------------------------------------------
In order to determine functional cerebral blood flow, single-photon
emission computed tomography (SPECT) was performed while the patient
was actively hallucinating. This revealed lower than normal blood flow
in the frontotemporal regions bilaterally Figure 2; no ischemic areas
were noted. Because this condition may be consistent with an
obliterative process in the midsized blood vessels, neurological
consultation was obtained; no indication for cerebral vasodilators or
anticoagulants in the absence of infarction was noted. On the basis of
the combined evidence of the history of onset of illness and the serum,
CSF, and MRI results, the patient's diagnosis was revised to possible
neuroborreliosis (Lyme disease affecting the CNS).
[Help with image viewing]
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Figure 2. Horizontal (A) and Coronal (B) SPECT Images Demonstrating
Hypoperfusion of Deep Frontotemporal Regions
------------------------------------------------------------------------
A percutaneous indwelling central venous catheter was inserted, and the
patient received a 4-month course of daily intravenous ceftriaxone
administered by the home care nursing team. After 4 weeks of antibiotic
therapy, repeat Lyme serologies indicated higher than normal titers
(0.478; 0.117 negative cutoff), and CSF revealed the presence of IgM
antibodies (39-kDa, 41-kDa, and others); these findings were consistent
with acute Borrelia infection and sufficient to meet the CDC criteria
for active neuroborreliosis [14]. No change in the patient's symptoms
was evident until the fourth month of antibiotic therapy, when she
began to ``feel better.'' Her delusional system, although still fixed
and intricate, was less imposing, and she was able to work full-time
for the first time in 7 years. Unfortunately, the patient interpreted
this newly acquired organization as a ``cure'' and elected to cease
intravenous antibiotic therapy. Three weeks later she experienced a
full-blown exacerbation of symptoms, requiring hospitalization.
Colleagues who had published case reports of long-term antibiotic
therapy for refractory or recurrent neuroborreliosis were consulted,
and the decision was made to resume intravenous treatment [15-17]. The
multiaxial DSM-IV diagnosis for this patient is shown in Figure 3.
[Help with image viewing]
-----------------------------------------------------------------------
Figure 3. Multiaxial DSM-IV Diagnosis for Patient With Probable Lyme
Disease
------------------------------------------------------------------------
DISCUSSION
This patient was referred to our hospital after a serious suicide
attempt, with a 6-year history of a psychotic disorder previously
diagnosed as chronic paranoid schizophrenia. An evaluation, however,
indicated that aspects of her history and symptoms were inconsistent
with her diagnosis. Before the onset of her illness, she demonstrated
none of the typical positive or negative prodromal symptoms commonly
seen in schizophrenia, such as social isolation and withdrawal, odd or
bizarre beliefs, and loss of interest. Rather than gradual
deterioration and loss of functioning, the patient appeared to have
rapidly progressed from depression and hypersomnia to hallucinations
and delusions within a few weeks.
The patient's hallucinations were markedly atypical and reminiscent of
the ``pseudohallucinations'' often seen in dissociative or
posttraumatic stress disorders. Although vivid multisensory (auditory,
visual, and tactile) hallucinations may be found in schizophrenia,
these were present only when the patient kept her eyes closed. They
were not consistent with classic hypnagogic or hypnopompic
hallucinations, which occur as an individual is falling asleep or
awakening, since they were not sleep related. She maintained a self-
induced erotic trance state in which she remained in bed 18 hours per
day, drifting in and out of sleep and compulsively masturbating. The
quality of her single visual hallucination was particularly unusual:
when her eyes were closed, she saw the face of her tormentor in a
wandering black spot that was not present when her eyes were open and
thus could not be considered a true scotoma. In addition, a single
powerful psychologically meaningful theme dominated her delusional
system: that of being molested by a familiar man. While persistent
hypersexuality and tactile hallucinations may be infrequently found in
the setting of secondary mania due to temporal, thalamic, limbic, or
orbitofrontal lesions [18-22], this patient had no history of manic
symptoms and no evidence of structural abnormalities on a CT scan or
EEG. This combination of multisensory hallucinations, erotic
preoccupation, a singular fixed delusion with dissociative episodes at
night, and her refractoriness to medication suggested sexual trauma as
a precipitant for psychosis, rather than schizophrenia.
High hypnotizability, as seen in this patient, is not typical of
paranoid schizophrenia [23]; it is more consistent with dissociative
disorders or reactive psychoses [24,25]. Indeed, while in the trance
she gave a detailed description of a sexual assault that occurred
before the onset of her illness. This assault appeared to be repeated
in her recurrent nightmares and flashbacks, which closely resembled
reexperiencing phenomena, with accompanying autonomic hyperarousal and
disturbance of sleep and mood. Nightmares and sleep disturbance are
seen in the majority of patients with PTSD and generally contain
content and emotion similar to the original trauma [26-28]. This is
particularly true if the trauma occurred while the person was sleeping,
such as the assault described by this patient during her hypnotic
recall [29]. Traumatic reexperiencing manifested as multisensory
hallucinations has been reported in both PTSD and psychotic depression
since Freud's 1896 case of Frau P, in which a woman who had been
sexually abused as a child by her brother had vivid visual and tactile
hallucinations of being genitally fondled ([30], pp. 174-185).
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