Complex Regional Pain Syndrome Type 1 Produced by Hematoma Formation after Blood Donation: A Case Report

Abstract

The occurrence of hematoma and bruise formation, accounting for the majority of donation-related complications in the arm, rarely results in complex regional pain syndrome (CRPS). We report a 24-year-old man who presented with CRPS on his right upper limb two months later due to hematoma and bruising formation just after a blood donation following with immediate performance of strenuous exercise in the upper limbs. Triple phase bone scan, one of the bone scintigraphic studies, revealed positive findings and was compatible with the symptoms of CRPS, e.g. hyperalgesia, swelling and discoloration. The potentially disabling condition, however, ended up with a thankfully benign outcome because of our early finding and proper treatment that included three-day oral prednisolone and two-week physiotherapy and occupational rehabilitation. To our knowledge, CRPS produced by donation-related complications with subsequent hematoma and bruise due to vigorous exercise is rare. CRPS should be taken into consideration in a blood donor who demonstrated allodynia because of performing heavy exercise immediately after blood donation.

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Citation: Chang CC, Chang ST (2021) Complex Regional Pain Syndrome Type 1 Produced by Hematoma Formation after Blood Donation: A Case Report. Open J
Orthop Rheumatol 6(1): 012-015. DOI: https://dx.doi.org/10.17352/ojor.000032
https://dx.doi.org/10.17352/ojor
DOI:
2641-3116
ISSN:
CLINICAL GROUP
Abstract
The occurrence of hematoma and bruise formation, accounting for the majority of donation-related complications in the arm, rarely results in Complex Regional
Pain Syndrome (CRPS). We report a 24-year-old man who presented with CRPS on his right upper limb two months later due to hematoma and bruising formation just
after a blood donation following with immediate performance of strenuous exercise in the upper limbs. Triple phase bone scan, one of the bone scintigraphic studies,
revealed positive fi ndings and was compatible with the symptoms of CRPS, e.g. hyperalgesia, swelling and discoloration. The potentially disabling condition, however,
ended up with a thankfully benign outcome because of our early fi nding and proper treatment that included three-day oral prednisolone and two-week physiotherapy and
occupational rehabilitation. To our knowledge, CRPS produced by donation-related complications with subsequent hematoma and bruise due to vigorous exercise is rare.
CRPS should be taken into consideration in a blood donor who demonstrated allodynia because of performing heavy exercise immediately after blood donation.
Case Report
Complex Regional Pain
Syndrome Type 1 Produced by
Hematoma Formation after
Blood Donation: A Case Report
Cheng-Chiang Chang1 and Shin-Tsu Chang1,2*
1Department of Physical Medicine and Rehabilitation, Tri-Service General Hospital, School of Medicine,
National Defense Medical Center, Taipei, Taiwan
2Department of Physical Medicine and Rehabilitation, Taichung Veterans General Hospital, Taichung,
Taiwan
Received: 11 January, 2021
Accepted: 01 February, 2021
Published: 02 February, 2021
*Corresponding author: Dr. Shin-Tsu Chang, Depart-
ment of Physical Medicine and Rehabilitation, Tri-
Service General Hospital, School of Medicine, National
Defense Medical Center, No.161, Sec. 6, Minquan East
Road, Neihu District, Taipei 11490, Taiwan, Tel: 886-
935-605578; E-mail:
Keywords: Hematoma; Complex regional pain syn-
drome; Blood donation; Triple phase bone scan
https://www.peertechz.com
Introduction
Approximately one third of whole-blood donors have an
adverse physical event during or after whole-blood donation.
Most of the common adverse effects associated with arm
fi ndings after blood donation are bruise (22.7%), soreness
(10.0%), and hematoma (1.7%) [1]. The reasons of the upper
limbs adverse events due to blood donation are multifactorial,
including genetic factors, infl ammatory process, peripheral/
central dyssensitization, sympathetic malregulation,
somatosensory cortex reorganization, and psychophysiologic
interactions, and all symptoms are likely a result of different
combinations following with time elapse [2]. Complex Regional
Pain Syndrome (CRPS) induced by hematoma might be another
severe complication of blood donation.
Anatomically speaking, sensory branches of the
musculocutaneous nerve locate below the antecubital veins,
as is classically taught, although they are also above the
antecubital veins or intertwine with them. Local nerve injuries
are unavoidable after phlebotomy because nerve branches
are situated so close to the vessels and are impalpable. The
frequency of nerve irritation is relatively high. Newman
reported an occurrence of 40% of the nerve injuries after a
straightforward phlebotomy [3]. Another study based on a
donor interview reported that sensory changes in the forearm
and hand occur in approximately 1% of whole-blood donors [1].
The nerve distribution in the donor’s arm might play
an important role on the possibility of occurrence of CRPS,
which has been shown to be developed due to abnormalities
in the central and peripheral nervous systems after a
nociceptive painful event. The characteristic of CRPS develops
unproportionately with the painful event and is not limited to
a single nerve course. Pathophysiological aspects including
neurogenic infl ammation, impairment of sympathetic
function, and coupling between sympathetic efferents and
nociceptive afferents should all be taken into consideration

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Citation: Chang CC, Chang ST (2021) Complex Regional Pain Syndrome Type 1 Produced by Hematoma Formation after Blood Donation: A Case Report. Open J
Orthop Rheumatol 6(1): 012-015. DOI: https://dx.doi.org/10.17352/ojor.000032
[4,5]. The diagnosis is ordinarily made on a clinical basis.
There is no pathognomonic laboratory fi nding for CRPS. An
alternative way in bone scintigraphy, the Triple Phase Bone
Scan (TPBS), can show increased uptake in the involved limb
earlier in the process. Multidisciplinary treatment combining
Transcutaneous Electrical Nerve Stimulation (TENS),
physical therapy, psychotherapy using behavior modifi cation
techniques, and oral medications are sometimes helpful [6].
CRPS induced by formation of hematoma followed blood
donation has never been described in the literature before.
We present a 24-year-old patient, which developed CRPS two
months after blood donation. It was considered that hematoma
with bruise on the antecubital fossa or antecubital cutaneous
nerve injury could probably have led to the development of
CRPS.
Case report
A 24-year-old man experienced progressive painful
swelling of the right upper extremity two months after blood
donation. He had an unremarkable past medical history, and is
enthusiastic about donating blood. Voluntarily, he had already
donated blood two times during his service in the army. Two
months prior to this admission, he donated blood for the third
time via the right antecubital vein as usual in the morning. An
amount of 250 ml whole blood was collected from the vein with
a 16-gauge needle using aseptic technique. Eight hours later
in the early evening, he was ordered to join routine military
training that consisted of chin-ups, push-ups and running and
he practiced them vigorously. Unknowingly, he found bruising
and hematoma appearing on his forearm, which spread from
the site of venipuncture with slight pain during the resting
period. From that day on, he began to suffer intermittent
painful swelling and discoloration in the right upper extremity
after every training course, which became more severe when he
performed grenade-throwing. Two days before the admission,
he developed a burning pain on the right hand with obvious
cyanosis, swelling, weakness and limited active Range of
Motion (ROM) after the strenuous exercise. According to his
statement these problems were not related to any trauma.
On admission, the patient had a pain Visual Analogue
Scales (VAS) of 8/10, and the pain in the forearm progressively
worsened and was unrelieved by rest, with a hematoma on
the antecubital fossa combined with a large area of bruise
by 15x7 cm2. Together with were extensive swelling, mild
hyperpigmentation, allodynia, coldness, cyanosis and limited
ROM of the right upper limb, especially on the right wrist.
The hyperalgesia on the affected muscle-guarding limb was
so intense that to undergo even a slight touch could induce a
burning and tingling pain out of proportion to the injury and
extend beyond the confi nes of dermatomal distribution. All
of the following studies were within normal limits, including
C-reactive protein, hemoglobin, white blood cell count, blood
chemistry, rheumatoid factor and nerve conduction studies. The
TPBS showed the increased uptakes in the right forearm, wrist
and hand, and is consistent with the fi nding of CRPS type 1. He
was undergone physiotherapy with whirl-pool hydrotherapy
and silver spike point stimulation, as well as occupational
therapy such as daily living training, hand function training,
and motor-sensory training. A signifi cant improvement was
noted followed by three-day prednisolone administration with
15 mg four times a day on the fi rst day of admission, and was
weaned by 20 mg a day over three-day period.
All symptoms gradually disappeared in two weeks. The
patient had a VAS of 1/10 before being discharged from our ward.
Three months since outpatient follow-up, he has made fairly
good recovery with regards to his overall general condition.
Discussion
CRPS type 1 or 2 are both clinical syndromes with symptoms
including burning pain, hyperalgesia, allodynia, edema,
sudomotor or vasomotor changes in the distal extremity. CRPS
type 1 most often occurs following soft-tissue trauma to a limb,
while CRPS type 2 occurs after injury to a peripheral nerve trunk.
The mechanisms underlying these two disorders are poorly
understood, but may be related to changes in the sympathetic
nervous system and/or neurogenic infl ammation [6]. Sasano,
et al. reported a 61-year-old woman who presented with CRPS
type 2 in the hand after transradial coronary intervention
for coronary catheterization [7]. Papadimos and Hofmann
reported a rare case of CRPS type 1 following transradial
coronary intervention, which etiology was that the radial
artery was occluded for a distance of 12 cm proximal to the
puncture site, possibly due to the longer period of hemostatic
compression [8]. A fi ve-year Japanese study revealed 133 cases
of resultant persistent pain and 19 cases of neuropathic pain
after performing venipunctures [4].
It has also been shown that venipunctures may cause
CRPS type 2 [9]. Horowitz reported 11 patients with injury to
upper extremity cutaneous nerves after routine venipuncture
developed causalgia (CRPS type 2), and proposed that nerve
injury appeared secondary to direct trauma via “inappropriate”
needle or bolused material entry into the plane of the nerves
beneath the veins, or nerves overlying the veins [10]. Unek et al
reported a patient with end-stage renal disease presenting with
refl ex sympathetic dystrophy syndrome (CRPS type 1) on the
patient’s left hand 1 month after Arteriovenous Fistula (AVF)
surgery. Magnetic resonance angiography confi rmed a steal
syndrome at the AVF level and the bone scintigraphy confi rmed
RSDS in early stage [11]. Genc, et al. also reported an 11-year-old
young girl with CRPS type 1 precipitated by rubella vaccination
[12]. Therefore, intramuscular injections, percutaneous venous
catheter insertion, or percutaneous arterial catheter insertion
might induce CRPS on the affected limbs, which may or may
not be related to the nerve injury adjacent to these punctured
vessels. However, blood donation with subsequent vigorous
exercise induced extensive hematoma and bruise triggering
CRPS has never been reported in the literature. The symptom
of allodynia in our patient was compatible with the image of
TPBS (Figure 1) and was worse on the right hand and wrist
instead of the forearm, the exact site of punctured injury by the
insertion of a 16-gauge needle to the antecubital vein.
Because subjective cutaneous hypoesthesia and
hyperalgesia in the vicinity of the antecubital fossa mainly on

014
https://www.peertechz.com/journals/open-journal-of-environmental-biology
Citation: Chang CC, Chang ST (2021) Complex Regional Pain Syndrome Type 1 Produced by Hematoma Formation after Blood Donation: A Case Report. Open J
Orthop Rheumatol 6(1): 012-015. DOI: https://dx.doi.org/10.17352/ojor.000032
the wrist and hand instead of the punctured site or the path of
the nerve trunk, we thought that the regional reaction did not
result from punctured trauma but from hematoma stasis which
could be an initiating noxious event and lead to CRPS type 1.
Whereas, a negative result of nerve conduction study could not
exclude the contribution of a nerve injury, because the nerve
conduction study only detects dysfunction in larger peripheral
nerves, and also cannot address the issue of whether there are
possible differences of signs or symptoms between patients
with and without dysfunction in smaller nerve fi bers.
The scintigraphic imaging is valuable in the evaluation
and early detection of CRPS. Adult patients with CRPS
characteristically have a TPBS pattern consisting of diffusely
increased tracer uptake with juxtaarticular accentuation of
tracer uptake on images. However, Hod and Horne, presented
an uncommon adult case of CRPS, whose bone scintigraphy
demonstrated decreased activity on early and late phase
images of in the affected limb [13]. The imaging of TPBS in our
case demonstrated increased uptake in the right forearm, wrist
and hand that was consistent with the fi nding of CRPS type 1.
The early fi nding of CRPS can also be seen in brain image as
increased uptake in the contralateral thalamus [14,15], but our
case did not have the perfusion scan at the time.
Diagnosing a CRPS at an early stage is important because
treatment in the early stage may lead to a good outcome.
The best treatment for CRPS is not known yet. For the relief
of symptoms, many kinds of treatment, such as TENS,
corticosteroids, adrenergic blocking agents, calcium channel
blockers, anti-depressants, hyperbaric oxygen therapy and so
on, have been used with varying effects. Two small randomized
controlled trials have reported that a 4- to 12-week course of
high-dose glucocorticoids administered orally 3 or 4 times
daily, can alleviate pain, edema and hyperalgesia in CRPS
patients [16,17]. A latest review article described the various
medical treatment and disease management [18]. Our case
was given analgesics and corticosteroids orally (prednisolone)
60 mg on the fi rst day after admission, 40 mg on the second
day, and 20mg on the third day. He presented a good response
to the prednisolone administration in combination with
rehabilitation therapy, such as physiotherapy with applying
hydrotherapy and TENS and occupational therapy with hand
function training, fi ne motor and sensory motor therapy on the
affected right upper limb.
In the present patient, insuffi cient rest after blood donation
resulted in the hematoma on the antecubital fossa combined
with a large area of bruising. Furthermore, the ensuing fi erce
military training including push-ups, chin-ups and a long-
distance running made the ecchymosis even more serious. Two
painful episodes on the forearm following vigorous training
within two months between the time of blood donation and
the fi rst attack of swelling with allodynia might already denote
CRPS. Moreover, two days prior to his admission, he kept
running at least 2000 meters long together with his right upper
limb immobilized in a guarded position, which may exacerbate
the vasomotor changes in the affected forearm, wrist and
hand because of pain induced by the previous hematoma. This
reminds us that longer and more adequate resting period after
blood donation before facing intense exercise, the possibility of
occurrence of CRPS on account of the formation of hematoma
is reduced.
In summary, we present a case of CRPS type 1 due to
blood donation complications which was exacerbated with
hematoma and bruising. Early intervention plays an important
role in reducing the long-term sequelae. Whenever performing
a blood collection, donors must be informed to be aware of the
possibility of the formation of hematoma if inadequate resting
period, which may also be noxious for the emergence of CRPS.
In order to shorten the clinical course, to reduce the possibility
of disability, and to prevent long-term morbidity medical
personnel should make an early fi nding and provide proper
treatment as soon as possible.
Concise Paragraph
1. CRPS type 1 or 2 are both clinical syndromes with
symptoms including burning pain, hyperalgesia,
allodynia, edema, sudomotor or vasomotor changes in
the distal extremity. CRPS type 1 caused by hematoma
per se has rare been reported before, while CRPS type 2
can occur after venipuncture due to nerve injury.
2. Blood donation with subsequent vigorous exercise
induced extensive hematoma and bruise triggering
CRPS type 1 has never been reported in the literature.
3. Whenever performing a blood collection, donors must be
Figure 1: The delayed images of bone scintigraphy demonstrate increased tracer
uptake throughout the symptomatic right forearm, right radius, right ulna, right carpal
and right proximal interphalangeal joints as compared with the left side. There is a
marked hot spot on the left leg of the patient due to stasis of tracer during injection.

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Citation: Chang CC, Chang ST (2021) Complex Regional Pain Syndrome Type 1 Produced by Hematoma Formation after Blood Donation: A Case Report. Open J
Orthop Rheumatol 6(1): 012-015. DOI: https://dx.doi.org/10.17352/ojor.000032
informed to be aware of the possibility of the formation
of hematoma if inadequate resting period, which might
also be noxious for the emergence of CRPS.
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Copyright: © 2021 Chang CC, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use,
distribution, and reproduction in any medium, provided the original author and source are credited.