nthis dentist is likely to have complex regional pain syn- of the clinical symptoms is not always seen immediately

burning, pricking, or shooting in character, and it feelslocalized deep in somatic tissue.10

ORIGINAL CONTRIBUTIONSCopyright 2015 American Dental Association. All rights reserved.shoulder-hand syndrome, algodystrophy, peripheraltrophoneurosis, sympathetically maintained pain, and

stimuli evoked, and it can be continuous, paroxysmal,or episodic.1 The pain may be described as aching,drome (CRPS), which is a chronic painful disorder that isprimarily a disease of the limbs.1 It has been calledcausalgia, reex sympathetic dystrophy, Sudeck atrophy,

after initiating events, and it may delay for months.3

In the early stages, the affected area is warm, painful,red, swollen, and sweaty.20 The pain is spontaneous orEXPLANATIONGiven this symptom history and these clinical ndings,cation in her right carpal bones. Thermography revealedPAIN UPDATE

Complex regional paiNeslihan Tinastepe, DDS, PhD; Koray Oral, DDS, PhD

CLINICAL PROBLEM

A 45-year-old female dentist fell on a slipperyoor at work and immediately felt pain in herright wrist. Her physician diagnosed asprained wrist without bony fracture orrupture of the ligaments. The physician wrapped abandage around the wrist, and anti-inammatory anal-gesics were prescribed to alleviate the pain. Rest, iceapplication, and elevation of the hand higher than theelbowwere recommended, and after 3 or 4 days, everythingseemed ne. However, in the following week, she felt aburning pain in her hand and forearm that became steadilyworse. She also noted swelling over the dorsum of her righthand. In subsequent months, the dentist was having dif-culties in performing her job owing to muscle weaknessand severe pain with movement of her arm. She alsonoticed sweating in the region of the painful area, as well asincreased growth of her ngernails and of the hair on thedorsum of her right hand.

Three months after her injury, the dentist visiteda university pain clinic. On physical examination, herorthopedist observed erythematous, edematous, andshiny skin on the swollen distal part of her forearm andthe dorsal side of her hand. Passive and active range ofmotion of her wrist was restricted. Physical systemicexamination and blood test results revealed no abnor-mality. On neurologic examination, hyperalgesia andallodynia were noted in her right forearm, wrist, andhand. Results of nerve conduction studies were normal.Grip strength was 7 kilograms and 24 kg in the right andleft wrists, respectively. Radiographs showed decalci-

a temperature 1.0 C higher than on the unaffected side.200 JADA 146(3) http://jada.ada.org March 2015syndromeposttraumatic pain syndrome.2 In 1993, the InternationalAssociation for the Study of Pain agreed on the descrip-tive term CRPS to avoid the question of etiology ormechanism.3 CRPS type I replaced the term reex sym-pathetic dystrophy, and CRPS type II replaced the termcausalgia.2,4-6 The previously used term sympatheticallymaintained pain now is used only for dening a symptomand might be a feature of several painful diseases; it maybe found in association with CRPS as well, but it is not anessential requirement for diagnosing CRPS.7,8

CRPS types I and II typically arise as a result ofminor tosevere trauma that leads to sensory, autonomic, motor,and trophic changes.9-11 Trauma severity and CRPSsymptoms are not proportionate. CRPS type I is distin-guished fromCRPS type II by the absence of obviousnervelesions, and CRPS type II is classied under neuropathicpain.7 Researchers have reported that CRPS may occur inboth sexes but predominantly in women,4 and it is mostcommon in people aged 55 to 75 years.12 The incidence ofCRPS type I is higher than that of CRPS type II.10

The internationally accepted Budapest criteria arehelpful for clinical diagnosis of CRPS. The box13 showsthe 2013 version of the clinical criteria; there are alsoresearch diagnostic criteria presented in the samearticle.13 There is no specic diagnostic test for CRPS,but some tests, including thermography, radiographs,and sympathetic blocks, may be useful.14 Ruling outserious neurologic disease may require imaging of thebrain and spinal cord.

Although researchers have proposed various theoriesto explain the pathophysiology of CRPS, it is not wellunderstood as yet. Neurogenic inammation,15,16 auto-nomic dysfunction,17 and neuroplastic changes6,18,19 arepossible mechanisms to account for CRPS development.Evidence suggests that both peripheral and centralmechanisms play a role in the syndromes pathogen-esis.7 A genetic predisposition and psychologicaldysfunction may play a role as well.8

Clinical symptoms change throughout the course ofCRPS in accordance with its pathophysiology.10 Onset

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ORIGINAL CONTRIBUTIONSIncreased hair and nail growth are usually presentwithin a few weeks after the initiating event,20 and 50% ofcases have these trophic changes.21Muscle weakness occursduring the acute stage as indicative of motor abnormality,which is seen in approximately 50% of patients withCRPS.15 Initially, the symptoms are conned to the injuredtissue but not necessarily within the same nerve root.20

Allodynia and hyperalgesia with thermal and me-chanical stimuli are frequently present in CRPS.6,19,22-24

Conversely, sensory loss to touch, heat, and pressuremay develop.11 Thus, CRPS may be characterized byboth positive and negative sensory disturbances.25 Thesesensory decits are seen in almost all patients withCRPS, with glove- or stocking-like distal distribution.10

In the chronic stage, hair and nail growth decreases,and atrophy of the skin with ulcerations may be present.

BOX

Budapest criteria for complex regionalTO MAKE THE CLINICAL DIAGNOSIS, THE FOLLOWING CRITERIA MU

1. Continuing Pain, Which Is Disproportionate to Any Inciting Even

2. Must Report at Least 1 Symptom in 3 of the 4 Following Categor

Sensory: Reports of hyperesthesia and/or allodynia

Vasomotor: Reports of temperature asymmetry and/or skin color chang

Sudomotor/edema: Reports of edema and/or sweating changes and/o

Motor/trophic: Reports of decreased range of motion and/or motor dysf

3. Must Display at Least 1 Sign at Time of Evaluation in 2 or More

Sensory: Evidence of hyperalgesia (to pinprick) and/or allodynia (to lighjoint movement)

Vasomotor: Evidence of temperature asymmetry (> 1C) and/or skin co

Sudomotor/edema: Evidence of edema and/or sweating changes and/

Motor/trophic: Evidence of decreased range of motion and/or motor dskin)

4. There Is No Other Diagnosis That Better Explains the Signs and

* Reproduced with permission of the publisher from Harden and colleagTremor, dystonia, exaggerated tendon reexes, andmyoclonic jerks are the other features of this stage.10

The skin color appears rather pale or livid with chro-nicity,6 and the skin temperature becomes colder thanthat on the unaffected side. This nding is contrary tondings during the acute stage, during which the skintemperature of the affected side is approximately 1Cwarmer than the temperature of the unaffected side.10

Over time, symptoms may begin to spread to otherparts of the body contralaterally, ipsilaterally, or diag-onally.11 Patients with CRPS may report that theirinvolved limb feels foreign to them (cognitive neglect)or that they need to pay mental and visual attention tomove their limb (motor neglect).26

The management of CRPS requires an interdisci-plinary approach because the occurrence and themaintenance of CRPS are not explained by only 1pathologic pathway. A stepwise time-contingent func-tional restoration algorithm, involving occupationaltherapy, recreational therapy, physiotherapy, andvocational rehabilitation, is the main strategy fortreating CRPS. The clinical team should consider usingpsychotherapy, as well as pharmacologic and interven-tional therapies, if needed, at any step.13

CONCLUSIONSTrauma to the upper extremities can compromise adentists work ability considerably. Even minor traumacan lead to CRPS in vulnerable people. This condition ispoorly understood, although it appears that both pe-ripheral and central mechanisms play a role in itspathogenesis. Early diagnosis is crucial for preventingCRPS from becoming chronic; however, the signs andsymptoms of CRPS mimic those of some other com-mon diseases, which may delay diagnosis. Dentalpractitioners encounter many patients with orofacial

ain syndrome.*BE MET:

:

and/or skin color asymmetry

eating asymmetry

tion (weakness, tremor, dystonia) and/or trophic changes (hair, nail, skin)

the Following Categories:

uch and/or temperature sensation and/or deep somatic pressure and/or

changes and/or asymmetry

sweating asymmetry

nction (weakness, tremor, dystonia) and/or trophic changes (hair, nail,

ptoms.

s.13pain in their daily practice, but they also should be awareof clinical features of painful conditions in other parts ofthe body. As this case history shows, a dentist also can beaffected personally by a clinically signicant pain dis-order. Therefore, dental practitioners should recognizeand refer any patient suspected of having CRPS(including themselves) immediately to a neurologist,rheumatologist, or pain specialist for conrming thediagnosis of CRPS and for initiation of appropriatetreatment. n

http://dx.doi.org/10.1016/j.adaj.2013.11.020

Dr. Tinastepe is a prosthodontist and is in private practice, Atasehirbulvar, Manolya 2/13, 7/15, Atasehir/Istanbul, Turkey, e-mailneslihantinastepe@hotmail.com. Address correspondence to Dr. Tinastepe.Dr. Oral is a professor, Department of Prosthodontics, School of

Dentistry, Yeditepe University, Istanbul, Turkey.

Disclosure. Drs. Tinastepe and Oral did not report any disclosures.

Pain Update is published in collaboration with the Neuroscience Group ofthe International Association for Dental Research.

JADA 146(3) http://jada.ada.org March 2015 201

1. Andreas G. Complex regional pain syndrome in adults. Rheumatology.2011;50(10):1739-1750.2. Colton AM, Fallat LM. Complex regional pain syndrome. J Foot Ankle

Surg. 1996;35(4):284-296.3. McBride A, Atkins R. Complex regional pain syndrome. Curr Orthop.

2005;19(2):155-165.4. Stanton-Hicks M, Burton AW, Bruehl SP, et al. An updated inter-

disciplinary clinical pathway for CRPS: report of an expert panel. PainPract. 2002;2(1):1-16.5. Merskey H, Bogduk N. Classication of Chronic Pain: Descriptions of

Chronic Pain Syndromes and Denitions of Pain Terms. 2nd ed. Seattle,WA: IASP Press; 1994.6. Maihfner C, Seifert F, Markovic K. Complex regional pain syn-

dromes: new pathophysiological concepts and therapies. Eur Neurol. 2010;17(5):649-660.7. Jnig W, Baron R. Complex regional pain syndrome: mystery

explained? Lancet Neurol. 2003;2(11):687-697.8. Racz GB, Heavner JE, Noe CE. Complex regional pain syndrome.

Semin Anesth. 1996;15(1):70-87.9. Drummond PD. Mechanism of complex regional pain syndrome:

no longer excessive sympathetic outow? Lancet. 2001;358(9277):168-169.10. Birklein F, Handwerker HO. Complex regional pain syndrome: how

to resolve the complexity? Pain. 2001;94(1):1-6.11. Birklein F, Riedl B, Sieweke N, Weber M, Neundorfer B. Neurological

ndings in complex regional pain syndromes: analysis of 145 cases. ActaNeurol Scand. 2000;101(4):262-269.12. de Mos M, De Bruijn AG, Huygen FJ, Dieleman JP, Stricker BH,

Sturkenboom MC. The incidence of complex regional pain syndrome: apopulation-based study. Pain. 2007;129(1-2):12-20.13. Harden RN, Oaklander AL, Burton AW, et al. Complex regional pain

syndrome: practical diagnostic and treatment guidelines, 4th edition. PainMed. 2013;14(2):180-229.14. Harris J, Fallat L, Schwartz S. Characteristic trends of lower-extremity

complex regional pain syndrome. J Foot Ankle Surg. 2004;43(5):296-301.

15. Ludwig J, Baron R. Complex regional pain syndrome type: aninammatory pain condition?DrugDiscov TodayDisMech. 2004;1(4):449-455.16. Schinkel C, Gaertner A, Zaspel J, Zedler S, Faist E, Schuermann M.

Inammatory mediators are altered in the acute phase of posttraumaticcomplex regional pain syndrome. Clin J Pain. 2006;22(3):235-239.17. Birklein F, Riedl B, Claus D, Neundorfer B. Pattern of autonomic

dysfunction in time course of complex regional pain syndrome. Clin AutonRes. 1998;8(2):79-85.18. Juottonen K, Silen T, Hurri H, Hari R, Forss N. Altered central

sensorimotor processing in patients with complex regional pain syndrome.Pain. 2002;98(3):315-323.19. Maihfner C, Handwerker HO, Neundrfer B, Birklein F. Patterns of

cortical reorganization in complex regional pain syndrome. Neurology.2003;61(12):1707-1715.20. Marinus J, Moseley GL, Birklein F, et al. Clinical features and

pathophysiology of complex regional pain syndrome. Lancet Neurol. 2011;10(7):637-648.21. Birklein F. Complex regional pain syndrome. J Neurol. 2005;252(2):

131-138.22. Maihofner C, Forster C, Birklein F, Neundorfer B, Handwerker HO.

Brain processing during mechanical hyperalgesia in complex regionalpain syndrome: a functional MRI study. Pain. 2005;114(1-2):93-103.23. Sieweke N, Birklein F, Riedl B, Neundorfer B, Handwerker HO.

Patterns of hyperalgesia in complex regional pain syndrome. Pain. 1999;80(1-2):171-177.24. Maier C, Baron R, Tolle TR, et al. Quantitative sensory testing in the

German Research Network on Neuropathic Pain (DFNS): somatosensoryabnormalities in 1236 patients with different neuropathic pain syndromes.Pain. 2010;150(3):439-450.25. Drummond PD. Sensory disturbances in complex regional pain

syndrome: clinical observations, autonomic interactions, and possiblemechanisms. Pain Med. 2010;11(8):1257-1266.26. Galer BS, Jensen M. Neglect-like symptoms in complex regional pain

syndrome: results of a self-administered survey. J Pain Symptom Manage.1999;18(3):213-217.

ORIGINAL CONTRIBUTIONS202 JADA 146(3) http://jada.ada.org March 2015

Complex regional pain syndromeClinical ProblemExplanationConclusions