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Case Report
DOI: 10.5455/bcp.20150502073159
Piriformis Syndrome Misdiagnosed as AntipsychoticRelated Dystonia
Osman Virit1, Lutfiye Pirbudak2, Bahadir Demir3, Ferdi Doganay4, Haluk A. Savas5
ABS­TRACT:
Piriformis syndrome misdiagnosed as antipsychotic-related dystonia
Dystonia associated with antipsychotics is common and well known. The current case presents a 20-yearold male patient during the manic phase of bipolar disorder, developing piriformis syndrome (PS) under
antipsychotic medication treatment. Initially, the anteflexion position of the patient was considered to be
dystonia secondary to antipsychotic treatment and treated accordingly with biperiden, benzodiazepine,
baclofen, tetrabenazine, electroconvulsive therapy (ECT), and botox; however, the patient did not respond
to these treatments. Subsequently, after consulting the algology department, PS was diagnosed as a distinct
entity. The patient recovered from the symptoms of PS upon the injection of bupivacaine and triamcinolone
1
Assoc. Prof., 3MD, 5Professor, Gaziantep
University, School of Medicine, Department
of Psychiatry, Gaziantep - Turkey
2
Prof., Gaziantep University, School of
Medicine, Department of Anesthesiology
and Reanimation, Algology Unit,
Gaziantep - Turkey
4
MD, Gaziantep University, School of
Medicine, Department of Anesthesiology
and Reanimation, Gaziantep - Turkey
drug-related side effects should be carefully reviewed, especially in resistant cases.
Corresponding author:
Dr. Osman Virit,
Gaziantep Üniversitesi, Tıp Fakültesi,
Psikiyatri Anabilim Dalı, Gaziantep - Türkiye
Keywords: antipsychotic, dystonia, piriformis sydrome
E-ma­il add­ress:
[email protected]
into his piriformis muscle. This case reminds us that the accuracy of the diagnosis in the treatment of possible
Klinik Psikofarmakoloji Bulteni - Bulletin of Clinical Psychopharmacology 2015;25(2):186-9
Date of submission:
January 19, 2015
Date of acceptance:
May 02, 2015
Declaration of interest:
O.V., L.P., B.D., F.D., H.A.S.: The authors
reported no conflict of interest related to
this article.
INTRODUCTION
Movement disorders are common side effects of
antipsychotics due to blockage of dopamine D2
receptors, and these are well known to clinicians.
One of them is acute dystonia, which is
characterized as “sustained abnormal postures or
muscle spasms that occur within a few days of
starting or rapidly raising the dose of the
antipsychotic, or of decreasing the medication
used to treat or prevent acute extrapyramidal
symptoms (e.g., anticholinergics). The prevalence
is quite high, varying widely, from 2% to 90%1. The
prevalence of tardive dystonia is 0.4-21.6% (mean
186
5.3%) in patients treated with antipsychotics 2.
Acute dystonia is generally improved by
anticholinergic treatment, but there may be some
difficulty in treating chronic or late dystonia 3.
Except for being a side effect of antipsychotics,
there are numerous causes of dystonia; including
idiopathic dystonia, Parkinson disease, Multiple
Sclerosis, Huntington disease, Wilson disease,
Rett syndrome, ataxia telangiectasia,
homocystinuria, Hartnup disease, tyrosinosis,
metachromatic leukodystrophy, gangliosidosis,
Lesch-Nyhan syndrome, vitamine E deficiency,
perinatal cerebral events, viral encephalitis, Reye
syndrome, cerebral tumors, arterio-venous
Klinik Psikofarmakoloji Bulteni, Cilt: 25, Sayı: 2, 2015 / Bulletin of Clinical Psychopharmacology, Vol: 25, N.: 2, 2015 - www.psikofarmakoloji.org
Virit O, Pirbudak L, Demir B, Doganay F, Savas HA
malformations, cerebral trauma, cerebral
operations, certain toxins, levodopa,
bromocriptine, metoclopramide, fenfluramine,
flecainide, ergot alkaloids, anticonvulsants, and
calcium channel blockers4.
Piriformis syndrome (PS) is a sciatic nerve
neuropathy developed through compression of
the sciatic nerve by the piriformis muscle (PM)5.
PS may be caused by inflammation, a tumor near
the PM, or anatomic variation of the PM and
sciatic nerve, micro/macro trauma to the PM,
edema, and local ischemia5,6. PS is characterized
by hip and buttock pain. This syndrome is not
often considered in clinical situations because its
presentation may be similar to that of lumbar
radiculopathy, primary sacral dysfunction, or
innominate dysfunction 5. The most common
symptoms of patients with PS is increasing pain
after sitting for longer than 15 to 20 minutes. Many
patients complain of pain above the PM. Difficulty
in walking and pain during internal rotation of the
ipsilateral leg may exist. Reported incidence rates
for PS among patients with lower back pain vary
widely, from 5% to 36%5,7.
Here, we report the management of a patient
who had initially been misdiagnosed with
antipsychotic-related dystonia and recovered after
being treated for PS.
CASE REPORT
A 20-year-old male patient hospitalized with
typical manic symptoms that lasted for one week.
He had had three episodes of hypomania
previously and recovered without treatment. Daily
doses of 6 mg risperidone, 900 mg lithium, 2 mg
biperiden, and 1 mg clonazepam treatment were
initiated. Two weeks later, risperidone was
increased to 8 mg and lithium was increased to
1200 mg. The patient recovered and was
discharged. Risperidone and clonazepam doses
were reduced at follow-up.
The patient was doing well at the three-month
follow-up and was using 4 mg risperidone, 2 mg
biperiden, and 900 mg lithium; however, he had
complaints of spasms and left leg pain, considered
to be related to the antipsychotics. Risperidone
was reduced to 2 mg, while the other medications
were continued at the same doses.
Two months later, the patient had manic
symptoms that lasted for two weeks, and was in
the anteflexion position. The patient was
hospitalized for a manic episode and acute
dystonia. Risperidone was stopped and a
treatment regimen was initiated including 6 mg
biperiden, 900 mg quetiapine, 900 mg lithium,
and 2 mg clonazepam. The manic symptoms
regressed; however, dystonia continued.
Biperiden was discontinued on the 16th day of
hospitalization; 15 mg baclofen was initiated and
increased to 30 mg; however, the dystonia did not
improve. A daily dose of 100 mg tetrabenazine
was initiated. Then the patient’s mood was
elevated again. ECT and 25 mg clozapine were
added to the ongoing treatment. Quetiapine was
terminated. Tetrabenazine was increased to 200,
then 300 mg, with no improvement in dystonia.
The patient did not respond to walking, balance,
posture, and isometric strengthening exercises
prescribed by the Physical Therapy Department.
His mania improved. The patient received 30
sessions of ECT in total. He was discharged and
prescribed 200 mg clozapine, 300 mg
tetrabenazine, and 1500 mg lithium. The
neurology department administered botox in the
outpatient clinic, but he did not recover from
dystonia. Later, he was referred to the algology
department.
The algology assessment revealed posture
disruption while walking, sitting, and going
upstairs, and posture improvement with resting.
The leg on the affected side faced outwards,
straight leg raise test was 30 degrees positive, hand
to floor distance was 30 cm, pace test and Freiberg
test were positive, and taut band (feeling of pain
by deep palpation of the muscles) was present
around the PM. A diagnostic piriformis block was
performed at the point of a “twitch response” on
the taut band, and posture distortion was
corrected. Hence the patient was diagnosed with
PS. Two sessions of piriformis block, one week
apart, were performed with a combination of 10
Klinik Psikofarmakoloji Bulteni, Cilt: 25, Sayı: 2, 2015 / Bulletin of Clinical Psychopharmacology, Vol: 25, N.: 2, 2015 - www.psikofarmakoloji.org
187
Piriformis syndrome misdiagnosed as antipsychotic-related dystonia
Figure 1: Anteflexion position in piriformis syndrome, and
enjection into piriformis muscle
mL 0.25% bupivacaine and 80 mg triamcinolone.
Posture and walking were normal at the threemonth check-up.
The patient has last been on 1500 mg lithium
and 100 mg clozapine, and he has been on the
same treatment for the last 1.5 years. He did not
experience any mood episodes in this period.
DISCUSSION
Since physicians generally concentrate only on
their own area of expertise, they may fail to
consider other etiologies, especially rare
situations. The neurology and physical therapy
departments interested in movement disorders
also failed to consider PS in our case. They
attempted to treat the patient for dystonia.
Consequently, the patient was considered to have
chronic dystonia, and he was administered almost
all antidystonic treatments: biperiden,
benzodiazepine, baclofen, tetrabenazine, ECT,
and botox; however, he remained irresponsive and
was hospitalized for 172 days.
The algologist treating the patient, when asked
what can be used to improve dystonia, explained
that the patient suffered from PS requiring an
intervention, as an anteflexion position was
observed due to his back pain. Thus the patient
was not diagnosed with dystonia, but with PS, and
his posture improved as he recovered from PS.
Actually, PS had developed in the patient,
consequently he had taken anteflexion position
while using antipsychotics. This condition was
inadvertently considered to be dystonia associated
188
with antipsychotics. The algologist explained that
the patient had fallen, resulting in a stretched PM
and compression of the sciatic nerve, thereby
causing PS.
The literature says that PS frequently goes
unrecognized or is misdiagnosed in clinical
settings. PS symptoms are similar to other
common somatic dysfunctions, such as
intervertebral discitis, lumbar radiculopathy,
primary sacral dysfunction, sacroiliitis, sciatica,
and trochanteric bursitis 5 . Further, through
compensatory or facilitative mechanisms,
piriformis syndrome may contribute to cervical,
thoracic, and lumbosacral pain, as well as to
gastrointestinal disorders and headache5,8. Our
patient was standing in the anteflexion position to
get rid of pain, while we thought that this postures
was caused by antipsychotic dystonia.
The PS diagnosis was reached by the
algologist’s special examination as mentioned in
the case presentation, and anyway anti-dystonic
treatment did not work, whereas the patient
improved with specific treatment of PS.
In the treatment of PS, hot applications,
ultrasound treatment, non-steroidal antiinflammatory drugs (NSAIs), and muscle relaxants
are used; if these are ineffective, local anesthetics,
steroids or botulinum toxin type A (BTX-A) are
injected into the PM or the perisciatic region.
Stretching of the PM and strengthening of the
abductor and adductor muscles should also be
included in patient treatment plans. If all of the
pharmacologic and other therapies fail, the final
treatment option is surgical decompression5,9.
Psychiatrists may not know PS well nor be
aware of these situations requiring algology
consultation. This case shows that advanced
diagnostic examinations should be carefully
performed in psychiatric clinical practice,
especially in therapy-resistant cases, in
conjunction with consultations from related
clinical departments. Additionally, psychiatrists
should keep up with advances in general medicine
and follow newly developed disciplines such as
algology.
Klinik Psikofarmakoloji Bulteni, Cilt: 25, Sayı: 2, 2015 / Bulletin of Clinical Psychopharmacology, Vol: 25, N.: 2, 2015 - www.psikofarmakoloji.org
Virit O, Pirbudak L, Demir B, Doganay F, Savas HA
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4. Ozdemir C, Eryılmaz M. Dystonia, Tardive dyskinesia, and
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5. Boyajian OL, McClain R. Diagnosis and management
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6. Fishman LM, Konnoth C, Rozner B. Botulinum neurotoxin
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2004;83(1):42-50. [CrossRef]
7. Foster MR. Piriformis syndrome. Orthopedics 2002;25(8):8215.
8. Steiner C, Staubs C, Ganon M, Buhlinger C. Piriformis
syndrome: Pathogenesis, diagnosis, and treatment. J Am
Osteopath Assoc 1987;87(4):318-23.
9. Kopacz DJ, Allen HW, Thompson GE. A comparison of
epidural levobupivacaine 0.75% with racemic bupivacaine
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[CrossRef]
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