Painful Limbs/Moving Extremities
Report of Two Cases
Tsuyoshi Miyakawa MD, Mitsunori Yoshimoto MD,
Tsuneo Takebayashi MD, Toshihiko Yamashita MD
Received: 25 January 2010/Accepted: 7 June 2010/Published online: 29 June 2010
? The Association of Bone and Joint Surgeons1 2010
tively rare condition characterized by aching pain in one
limb and involuntary movement in the affected fingers or
toes. Its pathomechanism is unknown. We report two
patients with painful limbs/moving extremities. In one
patient with a painful arm and moving fingers, the symp-
toms were resolved after surgery.
Patient 1 was a 36-year-old man with
a painful arm and moving fingers. Treatment with admin-
istration of analgesics was not effective. Postmyelographic
CT showed stenosis of the right C5/C6 foramen attribut-
able to cervical spondylosis and a defect of the contrast
material at the foramen. He was treated with cervical for-
aminotomy. Patient 2 was a 26-year-old woman with a
painful leg and moving toes. The pain and involuntary
movement appeared 2 weeks after discectomy at L5/S1.
Lumbar MRI and myelography showed no indications of
Painful limbs/moving extremities is a rela-
nerve root compression. She was treated with a lumbar
nerve root block. The pain and involuntary movement
completely disappeared in both patients after treatment.
Numerous studies report treatments for
painful limbs/moving extremities, but few report successful
treatment. Recently, botulinum toxin A injection and epi-
dural spinal cord stimulation have been used and are
thought to benefit this condition. Successful surgical
treatment previously was reported for only one patient.
Purposes and Clinical Relevance
compression of nerve tissue, we believe surgical decom-
pression should be considered for patients with painful
limbs/moving extremities who do not respond to nonop-
If imaging indicates
Painful arm and moving fingers (PAMF), which is char-
acterized by aching pain in the upper extremity and
spontaneous involuntary movements of the fingers, was
first reported by Verhagen et al.  in 1985. A similar and
more common syndrome in the lower extremity is called
painful legs and moving toes (PLMT), first coined by
Spillane et al.  in 1971. Papapetropoulos and Argyriou
 described these disorders as painful limbs/moving
extremities (PLME). These were divided into several
subtypes according to localization of movement and pain
and laterality of symptoms. PLME is a relatively rare
disorder and its pathomechanism is unknown.
We treated two patients with PLME. The first was a
patient with PAMF in whom cervical nerve root com-
pression potentially led to the symptoms, as indicated by
improvement after cervical foraminotomy. The second
was a patient with PLMT after surgery for lumbar disc
Each author certifies that he or she has no commercial associations
(eg, consultancies, stock ownership, equity interest, patent/licensing
arrangements, etc) that might pose a conflict of interest in connection
with the submitted article.
Each author certifies that his or her institution approved the reporting
of this case report, that all investigations were conducted in
conformity with ethical principles of research, and that informed
consent for participation in the study was obtained.
Electronic supplementary material
article (doi:10.1007/s11999-010-1437-y) contains supplementary
material, which is available to authorized users.
The online version of this
T. Miyakawa (&), M. Yoshimoto, T. Takebayashi,
Department of Orthopaedic Surgery, Sapporo Medical
University School of Medicine, S1 W16, Sapporo,
Hokkaido 060-8543, Japan
Clin Orthop Relat Res (2010) 468:3419–3425
herniation. We found only one previous report of patients
with PAMF whose symptoms were resolved after surgery
. We herein report these two patients and review the
A 36-year-old man was referred to our hospital after a
followed by aching or burning pain in his right thumb, index
finger, and upper arm and involuntary movements of the
right thumb. Treatment with administration of analgesics
was not effective. He and his family had no remarkable
contributory history including psychogenic disease.
On examination, there was aching pain at rest in his right
thumb and index finger and involuntary flexion-extension
movement of the thumb at the carpometacarpal joint (Video
1, Supplemental Website Material; supplemental materials
are available with the online version of CORR).
Movement of the thumb increased in proportion to the
degree of pain and could not be stopped by conscious
effort. Moreover, the patient could not voluntarily repro-
duce the movement on the unaffected side when asked to
do so. It disappeared during sleep but otherwise, basically,
was a continuous movement. Neurologic examinations
showed Grade 4 of 5 in manual muscle testing of the right
biceps muscle and wrist extensor muscles, sensory distur-
bance in the right C6 distribution, weakness in the right
brachioradialis tendon reflex, and radiating pain to the
thumb and index finger reproduced by neck extension and
side flexion (positive test using the criteria of Spurling and
Scoville ) on the right side, indicating right C6 radic-
ulopathy. MRI showed no compression of the spinal cord
but minimal stenosis at the right C5/C6 foramen. Also,
postmyelographic CT showed stenosis of the right C5/C6
foramen attributable to cervical spondylosis and a defect of
the contrast material at the foramen (Fig. 1). A right C6
nerve root block was performed and resulted in temporary
disappearance of the pain. The pain recurred the next
morning and the involuntary movement did not stop while
the nerve block was in effect.
Right C6 cervical spondylotic radiculopathy was diag-
nosed on the basis of this patient’s pain. We consulted a
neurologist and planned to observe the patient closely and
obtain additional studies including electromyography
(EMG) and brain MRI. However, because the pain was
severe and resistant to oral medication or nerve root block,
we proceeded with surgery for the right C6 cervical
spondylotic radiculopathy. Two months later a C5 lamin-
oplasty and right C5/C6 foraminotomy were performed.
After the surgery, the pain and involuntary movements had
completely disappeared. At 2 years followup, the patient
had no recurrence of pain or involuntary movements.
A 26-year-old woman was referred to our hospital after a
6-month history of left sciatica unsuccessfully treated by
nonsurgical therapies. Lumbar disc herniation at L5/S1 was
diagnosed and she underwent discectomy. Two weeks
later, she felt pain in her left leg and involuntary movement
of her left toes appeared. The movement was a combina-
tion of flexion-extension and adduction-abduction, and
each toe motion was independent (Video 2, Supplemental
Website Materials; supplemental materials are available
with the online version of CORR). Moreover, the move-
ment could not be stopped by conscious effort and the
patient could not voluntarily reproduce the movement on
the unaffected side when asked to do so. However, the
movement did disappear during sleep. Lumbar MRI and
myelography showed no indications of nerve root com-
pression. The neurologist to whom we referred the patient
ruled out any psychogenic disorder and other neurologic
movement disorders by MRI of the brain. The pain was not
relieved and the involuntary movement was not resolved
by a caudal block and administration of clonazepam,
diazepam, haloperidol, and adenosine triphosphate diso-
dium. A left S1 nerve root block had a temporary effect,
resulting in disappearance of the pain in her leg and the
involuntary movement of the toes for a few days. There-
fore, we performed the same nerve root block seven times,
and 4 months after administration of the first nerve root
block, the pain and involuntary movement had completely
disappeared. Three months after disappearance of the pain
and involuntary movement, the patient was lost to followup
because she changed her residence.
Fig. 1 A postmyelographic CT scan shows stenosis of the right C5/
C6 foramen (arrow) attributable to cervical spondylosis and a defect
of the contrast material at the foramen.
3420 Miyakawa et al. Clinical Orthopaedics and Related Research1
PLME is defined as the association of aching or burning
pain in at least one limb and involuntary movement of at
least one finger or toe. The involuntary movement is
spontaneous and continuous and characterized by flexion-
extension and/or abduction-adduction movement of affec-
ted fingers or toes. Typically, the movement increases in
proportion to the degree of pain and cannot be reproduced
on the unaffected side. Some of the patients can stop the
movement for a short time by conscious effort and the
movement disappears during sleep. There are some reports
of patients with PLME without pain, called painless limbs/
moving extremities [2–5, 21, 31, 41].
We identified 12 patients with PLME of the upper
extremity (PAMF), and of these patients, only one was
successfully treated surgically  (Table 1). There are
more reported cases of PLME in the lower extremity
(PLMT) than in the upper extremities (PAMF). However,
there have been no reports of PLMT being successfully
resolved surgically, and only two reports of PLMT being
partially or temporary improved surgically [14, 42]
PLME is a relatively rare disorder; therefore, the path-
ophysiology is not fully understood. Based on the
effectiveness of the lumbar sympathetic ganglion block,
Spillane et al.  speculated that abnormal impulses
from afferent fibers in the sympathetic nervous system
activated ventral horn cells in the spinal cord, resulting in
the involuntary movement. Nathan  proposed that
abnormal stimulation to the nerve root or peripheral nerve
caused pain in the limbs and evoked involuntary movement
by stimulating ventral horn cells via the spinal interneuron.
Meanwhile, as involuntary movement does not appear
during sleep, Schott suggested it was associated with the
reticular activating system , which is controlled by
In our patients, the pain and involuntary movement
disappeared after decompression or block of the spinal
nerve root, supporting Nathan’s theory regarding the
pathomechanism of PLME (Fig. 2). He theorized that
ectopic impulses of the nerve root caused by irritation are
conducted to the spinal dorsal horn neurons through the
afferent pathway, leading to pain perception. At the same
time, the impulses from the nerve root would excite the
local spinal interneuron and lead to stimulation of spinal
ventral horn neurons, resulting in involuntary movement of
Nonsurgical treatments, including administration of
benzodiazepine or c-aminobutyric acid and sympathetic
ganglion block, are commonly performed [6–8, 13, 15, 18,
19, 22, 24–29, 32, 35–37, 40, 42] for patients with PLME
because the central nervous system or sympathetic nervous
system also is considered to be the cause of PLME, as
mentioned above. However, symptoms of PAMF (Table 1)
and PLMT (Table 2) usually are resistant to such
Dressler et al.  reported that sympathetic ganglion
block was effective in approximately 50% of patients with
PLME but the effects were transient. Guieu et al. 
Table 1. Review of the literature (painful arms and moving fingers)
Verhagen et al. 54 FBrachial plexus
Funakawa et al.  52 M Peripheral nerve (trauma)Oral medicationNo changeNo change
Ebersbach et al. 64 MOral medicationNo changeNo change
Jabbari et al. 35 FPeripheral nerve (trauma)Oral medicationNo changeNo change
Botulinum toxin A injectionNo changeTransient relief
Supiot et al. 68 MBrachial plexusTransportion of the ulnar nerveNo changeNo change
Oral medication (calcitonin)Transient reliefDecreased
25 FNerve root (CSR)Oral medication (calcitonin)Transient reliefTransient relief
49 MPeripheral nerve (trauma)Oral medication (amantadine)DecreasedDecreased
66 FCNS (stroke) Oral medicationNo changeNo change
Sudo et al. 56 FSpinal cord (CSM)Cervical laminoplasty DisappearedDisappeared
Singer and Papapetropoulos  20 FPainless Botulinum toxin A injectionNoneDisappeared
Schwingenschuh and Bhatia  62 F Hypothyroidism Oral medicationNo changeNo change
Current study36 MCSRCervical foraminotomy DisappearedDisappeared
F = female; M = male; CNS = central nervous system; CSR = cervical spondylotic radiculopathy; CSM = cervical spondylotic myelopathy.
Volume 468, Number 12, December 2010Painful Limbs/Moving Extremities 3421
Table 2. Review of the literature (painful legs and moving toes)
Spillane et al. 48 MSNSBlock of SNSTransient
SympathectomyNo changeNo change
54 MSNSBlock of SNS DisappearedDisappeared
67 F, 54 MSNS Block of SNSTransient
68 F, 53 M Oral medicationNo change No change
Okamoto et al. 62 FOral medicationNo change
Block of SNSDisappeared
Nathan  46 F Nerve root (herpes zoster)
30 F Nerve root (trauma)
57 MNerve root (cyst) Nerve root
No change No change
62 F Peripheral nerve (trauma)
Schott  69 F TraumaBlock of SNSTransient
56 F, 77 FTraumaBlock of SNS No changeNo change
66 M, 57 FOral medication No changeNo change
Wulff 56 M, 64 MOral medicationNCNC
41 FHerniotomyHerniotomy Disappeared Transient
Montagna et al. 60 MPeripheral nerve
74 F, 76 FPeripheral nerve
Oral medicationNo changeNo change
Schoenen et al. 71 F, 80 F, 49 M,
69 F, 68 F,
Oral medicationNo changeNo change
Sahashi et al. 62 F, 75 MBlock of SNSNo changeNo change
Oral medicationNo changeNo change
Sandyk 51 FNeurolepticOral medication (baclofen)DecreasedDecreased
Mitsumoto et al. 40 FPeripheral nerve
Guieu et al. 29 MCNS, peripheral nerve
Clonazepam injection Transient
Uchihara et al. 56 MHypophysectomy Epidural blockTransient
Block of SNSDisappearedDisappeared
46 M Parkinson’s diseaseEpidural block Transient
Block of SNSDisappearedDisappeared
Walters et al. 25 MPeripheral nerve
Guieu et al. 
30 MPeripheral nerve (trauma)ATP injection Disappeared
72 F ATP injectionDisappeared
Dressler et al. 36 M Spinal cord (trauma)Block of SNSNo changeNo change
3422Miyakawa et al.Clinical Orthopaedics and Related Research1
Table 2. continued
76 FNerve root (herpes zoster) Block of SNSTransient
49 FNerve rootBlock of SNS No changeNo change
69 FNerve root
28 MNerve root (hemangioma)
66 F, 73 M Nerve root
28 FPeripheral nerve (trauma) Block of SNSDecreased Decreased
68 F Peripheral nerve (trauma)
67 FPeripheral nerve (trauma) Block of SNSTransient
66 F, 74 FPeripheral nerve (trauma) Block of SNSNo changeNo change
61 F, 72 F Peripheral nerve
69 F, 54 MPeripheral nerve
67 F, 72 F
65 FBlock of SNS No change No change
28 M, 36 M PainlessNone
45 M PainlessNone
Mosek et al. 57 FNerve root LaminectomyNo changeNo change
Dorsal root block Transient
Pla et al.  35 MPeripheral nerve
Block of plantar nerveTransient
Ebersbach et al. 64 MOral medication No changeNo change
Touge et al.  37 MPeripheral nerve
Oral medicationNo changeNo change
Block of SNSDecreasedDisappeared
Okuda et al. 36 FEpidural blockDisappearedDisappeared
56 MEpidural blockTransient
Block of SNS No changeNo change
50 FOral medication No changeNo change
Shime and Sugimoto 63 FBlock of SNSTransient
Pitagoras de Mattos
et al. 
38 M, 51 MPeripheral nerve
Oral medicationNo changeNo change
Sanders et al. 76 FOral medication No changeNo change
Takahashi et al. 51 M Epidural block, TENSNo changeNo change
Block of SNS Transient
Dziewas et al.  38 F, 70 FPainless None
Ikeda et al. 75 FSpinal cord (herpes zoster) Block of SNS No changeNo change
Epidural block, ESCS No changeNo change
Volume 468, Number 12, December 2010 Painful Limbs/Moving Extremities3423
treated two patients with PLMT by injection of adenosine
triphosphate. The pain disappeared but the effect on
movement was not mentioned. Okuda et al.  suggested
several advantages of epidural block over sympathetic
ganglion block, and Takahashi et al.  reported the
benefit of epidural spinal cord stimulation for PLMT.
Recently, treatment with botulinum toxin A (BTA) injec-
tion was reported by Singer and Papapetropoulos  and
Eisa et al. . The injection resulted in substantial pain
relief and reduction of involuntary movement owing to
reduction of the muscle spindle leading to decreased
activity of the gamma loop and central sensitization.
Sudo et al.  reported the only case of a patient with
PAMF in whom pain and involuntary movement were
resolved by surgery. In their patient, cervical radiculopathy
or segmental myelopathy was thought to be the cause of the
symptoms, and bilateral open-door laminoplasty was per-
formed. Similarly, the symptoms in our first patient were
resolved by decompression of the nerve root. Therefore, we
believe surgical treatment should be considered for patients
in whom compression of nerve tissue is recognized by MRI
and/or CT but in whom no response is obtained by non-
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Papapetropoulos and Singer
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