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Indian Pediatr 2019;56: 8 80-881 |
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A Rare Cause of Swelling and Pain in Extremities in Children:
Complex Regional Pain Syndrome
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Husniye Yucel, Meltem Akcaboy* and Saliha Senel
Department of Pediatrics, Dr. Sami Ulus Maternity and
Children’s Health and Diseases Training and Research Hospital, Altindag,
Ankara, Turkey.
Email:
[email protected]
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Complex regional pain syndrome is a
condition of uncertain etiology characterized by spontaneous or
stimulus-induced pain that is out of proportion to the inciting event.
We report a 14-year-7-month-old girl with swelling of the left hand and
wrist, was diagnosed as complex regional pain syndrome. The patient was
treated successfully with physical therapy and non-steroidal
anti-inflammatory drugs. This condition should be kept in mind in the
differential diagnosis of musculoskeletal non-inflammatory and
inflammatory pains.
Key words: Diagnosis, Management, Recurrent
pain.
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M usculoskeletal pain and swelling are common
reasons of admission for pediatricians. The causes include a variety of
inflammatory and non-inflammatory diseases such as arthritis, myositis,
fibromyalgia, hypermotility, growing pains [1]. Complex regional pain
syndrome (CRPS), previously known as reflex sympathetic dystrophy, is a
chronic pain condition usually affecting distal extremities
characterized by spontaneous or stimuli-induced pain [2]. Herein we
report an adolescent patient diagnosed with CRPS and treated
successfully.
A 14-year-7-month-old girl was admitted to our
hospital with swelling of the left hand and wrist, pain and redness on
the whole arm, and difficulty in moving the arm for 2 days. There was a
feeling of numbness and tingling on the left hand and in the left upper
extremity with an itching and burning sensation in her left arm
described as hyperalgesia. The pain had flammable character and was
exacerbated with movement or light touch suggesting allodynia. The
decrease of the pain was reliable when the contact was stopped. The
patient reported no preceding trauma prior to admission. No prior
infection or fever was reported. Her past medical and family history was
unremarkable.
On physical examination, the patient was afebrile.
Hyperemia and swelling was prominent on the distal part of the left
upper extremity and on the palms of the left hand, plantar skin and the
fingers. There was edema of the entire left upper limb. She had
difficulty in moving the left and the fingers. Hyperalgesia,
hyperesthesia and allodynia were prominent in the left hand and upper
extremity. Pain sensitivity was prominent. In the laboratory evaluation,
all of the results were in normal ranges for the complete blood count,
erythrocyte sedimentation rate, C- reactive protein, coagulation tests,
vitamin B 12,
antistreptolysin O (ASO), and routine blood chemistry. Serologic
evaluation for infections and rheumatologic evaluation were negative.
Superficial tissue and Doppler ultrasonography, Echocardiography, direct
radiographs were all normal. The magnetic resonance imaging of spine was
did not show any abnormality. These findings, in conjunction with the
history, were suggestive of CRPS type I.
The patient received physiotherapy that was performed
intermittently in the therapy pool; as well as using passive therapy as
the massage of the left arm. Beside the physical therapy, non-steroid
anti-inflammatory drug treatment with naproxen sodium was initiated with
a dose of 10 mg/kg/day. On the third day of therapy, the pain of the
patient improved. Medical treatment lasted for 15 days, but the course
of the disease lasted for approximately one month. She has been followed
out-patiently by physical therapy for has 3 months without any symptoms.
CRPS is a term to describe conditions predominantly
characterized by spontaneous or stimulus-induced pain that is
inconsistent with the provocative event [3]. CRPS diagnosis entirely
depends on observable signs and reported symptoms, which have been put
together into various diagnostic criteria sets for adults [4]. The
diagnosis of CRPS in children may be delayed as long as four months
because of the low incidence and considerably different clinical
presentation compared with adults [5]. The clinical manifestations of
CRPS may imitate rheumatologic diseases in children.
CRPS has been suggested to be a multifactorial
condition that is related to an unusual host response to certain tissue
damage. The disease often includes a wide diversity of autonomic and
motor disturbances like hyperalgesia, allodynia, and sensory loss [4,5].
The patient can exhibit particularly painful, red, warm, and swollen
extremities, mimicking trauma. Other probable accompanying features are
changes in sweating, reduced hair and nail growth, allodynia and
hyperalgesia, and also muscle weakness [2]. However, apparent trauma or
initiating factor is absent in most of the pediatric patients. Exclusion
of other possible causes is necessary and investigations are needed to
exclude infections of skin, connective tissues, muscles, bone and
joints. Further investigations may be needed if inflammatory diseases,
fractures, neoplasms and deep venous thrombosis are suspected [6]. In
our patient, the possible conditions were excluded by further clinical
and laboratory evaluation.
The optimal management approach should consist a
multidisciplinary treatment of noninvasive interventions including
physiotherapy, occupational therapy, anal-gesics and psychotherapy
[5,6]. Sensory rehabilitation is sometimes added in order to gradually
improve the allodynia [6]. The standard medications include drugs
non-steroidal anti-inflammatory drugs, antidepressants, anticonvulsants,
topical analgesic patches. In contrast to adults, the response to
treatment, particularly exercise therapy with behavioral management will
achieve almost 97% remission in children [3], as in the reported child.
We report a pediatric CRPS Type I patient, treated
successfully by conservative methods, in order to attract attention to
this rare benign condition in children.
Contributors: HY: contributed to diagnosis and
management of patient; MA and SS: contributed to manuscript writing and
critical inputs into case management.
Funding: None; Competing Interests: None
stated.
References
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pain syndrome: A review. Pediatr Rheumatol Online J. 2016; 29:29.
2. Gierthmuhlen J, Baron R, Blankenburg M, Zernikow
B, Zernikow B, Maier C. Spontaneous recurrent episodes of wrist pain in
a 16-year-old girl: A case of complex regional pain syndrome. Pain Rep.
2016; 30:e578.
3. Rodriguez MJ, Fernandez-Baena M, Barroso A, Yanez
JA. Invasive management for pediatric complex regional pain syndrome:
Literature review of evidence. Pain Physician. 2015;18:621-30.
4. Harden RN, Bruehl S, Stanton-Hicks M, Wilson PR.
Proposed new diagnostic criteria for complex regional pain syndrome.
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5. Lascombes P, Mamie C. Complex regional pain
syndrome type I in children: What is new? Orthop Traumatol Surg Res.
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6. Abu-Arafeh H, Abu-Arafeh I. Complex regional pain
syndrome in children: A systematic review of clinical features and
movement disorders. Pain Manag. 2017;7: 133-40.
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