Geertzen, J.H.B.

Leeman, J.F.W.J.

Meek, M.F.

Robinson, P.H.

van den Dool, H.J.W.

English

ARTS repository - University of Groningen

   University of GroningenAn uncommon cause of unilateral hand and forearm oedemaMeek, M.F.; van den Dool, H.J.W.; Robinson, P.H.; Leeman, J.F.W.J.; Geertzen, J.H.B.Published in:European Journal of Plastic SurgeryIMPORTANT NOTE: You are advised to consult the publisher's version (publisher's PDF) if you wish to cite fromit. Please check the document version below.Document VersionPublisher's PDF, also known as Version of recordPublication date:2007Link to publication in University of Groningen/UMCG research databaseCitation for published version (APA):Meek, M. F., van den Dool, H. J. W., Robinson, P. H., Leeman, J. F. W. J., & Geertzen, J. H. B. (2007). Anuncommon cause of unilateral hand and forearm oedema. European Journal of Plastic Surgery, 29, 245-247.CopyrightOther than for strictly personal use, it is not permitted to download or to forward/distribute the text or part of it without the consent of theauthor(s) and/or copyright holder(s), unless the work is under an open content license (like Creative Commons).The publication may also be distributed here under the terms of Article 25fa of the Dutch Copyright Act, indicated by the “Taverne” license.More information can be found on the University of Groningen website: https://www.rug.nl/library/open-access/self-archiving-pure/taverne-amendment.Take-down policyIf you believe that this document breaches copyright please contact us providing details, and we will remove access to the work immediatelyand investigate your claim.Downloaded from the University of Groningen/UMCG research database (Pure): http://www.rug.nl/research/portal. For technical reasons thenumber of authors shown on this cover page is limited to 10 maximum.Download date: 06-06-2022CASE REPORTAn uncommon cause of unilateral hand and forearm oedemaMarcel F. Meek & Hans J. W. van den Dool &Peter H. Robinson & Jaap F. W. J. Leeman &Jan H. B. GeertzenReceived: 20 April 2006 /Accepted: 19 August 2006 / Published online: 4 November 2006# Springer-Verlag 2006Abstract A case with an uncommon cause of unilateralhand and forearm oedema in a 37-year-old man ispresented. Complex regional pain syndrome type I wasdiagnosed and treated with mannitol infusion, dimethylsulfoxide, vitamin C, ketanserin and carnitine. Also,physiotherapy and occupational therapy were started. Noneof these treatment modalities resulted in reduction orprevention of recurrence of the oedema. All diagnosticprocedures did not reveal a cause for this oedema.Keywords Hand . Forearm . Oedema . UncommonIntroductionThe term “oedema” is used in the case of excess volume offluid in the extracellular–extravascular compartment (theinterstitial space). Most commonly, the fluid accumulates inthe dependent parts of the body due to the effects of gravity.Oedema can be generalised or localised, and this classifica-tion can be a helpful guide when investigating its cause [3].Oedema of the hand and forearm can have differentcauses. Oedema after contusion or a fracture is well known.During an infection or after burn injury, it can also beexpected. Mostly, it is a gradual process and will resolveduring different therapies.Swelling—pitting or non-pitting oedema—is usuallypresent when complex regional pain syndrome type I(CRPS I) is diagnosed and localised to the painful region.One of the clinical features in these cases is a more diffuseswelling (generalised oedema). Uniform diagnostic criteri-on for CRPS I is (consensus conference IASP 1995) thepresence of four out of the following five symptoms:unexplained diffuse pain, color changes relative to thecontralateral extremity, temperature changes relative to thecontralateral extremity, diffuse oedema or limitations inrange of motion [6]. When CRPS I is diagnosed, symptomsoccur or are aggravated during or after exercise. Also,symptoms are present over a larger area and distal to theprimary site of injury or trauma.Localised oedema is limited to a particular organ orvascular bed. Unilateral extremity oedema is usually due tovenous or lymphatic obstruction, such as deep venousthrombosis, tumour obstruction or post-coronary bypasswith a harvested leg vein [5]. Stasis oedema of a paralysedlower or upper extremity may also occur. A more balloon-like aspect of oedema is seen after, for example, aconstricting bandage (localised oedema) [3].Oedema delays healing and causes pain and stiffness andthereby compromises functional healing of an extremity. Itis a constant challenge for hand surgeons and handtherapists to prevent oedema. During treatment of oedema,the principles of elevation and active motion are thereforeimportant. Each cause of oedema has its own specificEur J Plast Surg (2007) 29:245–247DOI 10.1007/s00238-006-0094-6M. F. Meek :H. J. W. van den Dool : P. H. RobinsonDepartment of Plastic and Reconstructive Surgery,University Medical Center Groningen,University of Groningen,P.O. Box 30.001, 9700 RB Groningen, The NetherlandsJ. F. W. J. LeemanDepartment of Dermatology,University Medical Center Groningen,University of Groningen,P.O. Box 30.001, 9700 RB Groningen, The NetherlandsJ. H. B. Geertzen (*)Center for Rehabilitation and Northern Centerfor Health Care Research,University Medical Center Groningen, University of Groningen,P.O. Box 30.001, 9700 RB Groningen, The Netherlandse-mail: j.h.b.geertzen@rev.umcg.nltreatment modality. We present a case with an unknowncause of unilateral hand and forearm oedema in a 37-year-old male patient.Case reportA 37-year-old man sustained a fall down the stairs causinga slight contusion to his right non-dominant forearm andhand. There were no fractures, and no acute oedema waspresent. There was no previous history of trauma to hisright arm. The patient was advised to elevate and rest hishand for some days.Several weeks later, the patient returned to the hospitalwith a hand and forearm oedema without an obvious cause.CRPS I was diagnosed. Treatment was started withmannitol infusion [1], dimethyl sulfoxide [2], vitamin C[10], ketanserin and carnitine [7] and physical andoccupational therapy.The swelling decreased during hospital stay, and thepatient returned home. A few days later, he returned to thehospital because of progression of the swelling of his handand forearm. Magnetic resonance imaging (MRI) andultrasound did not show a cause for the diffuse swelling.Also, a ganglion block did not decrease the swelling.The patient was sent to the department of rehabilitationin our hospital for a second opinion. A clinical evaluationshowed unilateral swelling of his right hand and forearm(Figs. 1 and 2). Initially, the patient refused inspection ofhis right upper arm because he had no complaints in thatregion. Because of the progression of the swelling and hisrefusal to allow inspection of his arm, automutilation wasconsidered as a possible cause of the swelling. Also,strangulation signs were seen on his upper arm (Figs. 1and 2). Doppler studies and new laboratory tests did notexclude the possibility of automutilation. A lymphoscintig-raphy did not indicate a late onset lymphoedema. Thepatient, however, denied any form of automutilation.Psychological help was arranged. Because the patientconsistently denied any form of automutilation, it wasdifficult for the psychologist to treat him. During thehospital stay, the swelling decreased amazingly quicklywithin 10 days, with the only treatment modalities beingFig. 1 Unilateral swelling of the patient_s right hand and forearmFig. 2 Strangulation signs seen on the patient_s upper arm246 Eur J Plast Surg (2007) 29:245–247observation and immobilization with a plaster of parissplint. In the meantime, fixed contractures of all his fingerswere observed. The patient had an end stage of anafunctional hand and forearm. During follow-up consulta-tions, oedema recurred to a minor degree, but the patient is,after 24 months, still unable to work and has been onsickness benefit since the original injury. The request foramputation by the original treatment centre and the patientwas not followed up.DiscussionThe classification of oedema into generalised or localisedcan be a helpful guide in investigating its cause. General-ised oedema is soft tissue swelling of most or all regions ofthe body. Localised oedema is limited to a particular organor vascular bed. Oedema delays healing and causes painand stiffness and thereby compromises functional healingof an extremity.In the first instance, the frequent occurrence of swellingafter a trauma was suspected, but because of the steadyprogression of the swelling, other possible causes wereconsidered. No fractures were seen on X-ray. MRI orultrasound showed no obvious cause such as thrombosis ortumour. Blood tests did not reveal an infection or rheumaticdisorder. Other causes such as lymphogenic pathology wereexcluded because of a normal lymph scintigraphy.CRPS I was a possible diagnosis, but during clinicalevaluation, the possibility of automutilation was discussed.Our patient had no psychiatric history. Because of the veryfast progression of oedema in our case and also the balloon-like appearance, we considered automutilation as a mostlikely cause. Localisation on one side, fast progression ofoedema out of hospital and fast regression during hospitalstay are factors that make automutilation a distinct possibil-ity. Signs of strangulation on his upper arm confirmed oursuspicion. Most frequently, automutilation occurs (in aboutone third of all cases) in patients with psychiatric disorders.Potential patients are difficult to recognize [4].Thirty years ago, Smith [8] published a large series ofpatients with factitious lymphoedema of the hand, some-times caused by a tourniquet and proximally ending in acircumferential discoloured constriction ring.The findings of strangulation of the affected upperextremity oriented to the diagnosis of a factitious disorderor Munchausen’s syndrome. In this psychiatric condition,the patient simulates a clinical condition in a veryconvincing way, mimicking to perfection a disease.Usually, the patients are either crying for attention orcausing some sort of automutilation as a means ofpunishment by unconscious guilt. Mostly, the sole purposeis assuming the sick role. It is important to always keep inmind that patients with unexplained and unspecific signs,such as oedema present in this case, may be indications ofMunchausen’s syndrome. After identification of this syn-drome, the patient should be referred to a psychiatrist.Of course, in our patient, automutilation in combinationwith CRPS I could be a possibility. The features in ourpatient also resemble the clenched fist syndrome [9]. Thedevelopment of a swollen clenched fist is thought to be “asymbolic expression of forbidden wishes”. It “representsfeelings of anger that are suppressed by paralyzing thehand”. Swift reported that resistance to treatment is afeature of this syndrome. Although our patient did notrefuse therapy, further psychological intervention wascancelled, and he was made aware of his own responsibilityin preventing a recurrence by the psychologist involved.In conclusion, oedema is a very simple clinical sign butcan reveal serious diseases. Our case report represents anunusual cause of unilateral oedema of the hand andforearm. Localisation on one side, fast progression out ofhospital and fast regression during hospital admissions arefactors that make automutilation highly likely. The com-plexity of these problems requires long-term multidisci-plinary therapy.References1. van der Gaag A, van Kleef M, Patijn J (2001) Mannitol: zin enonzin. Ned Tijdschr Pijn Pijnbestrijd (Dutch) 21:5–102. Geertzen JHB, de Bruijn H, de Bruijn-Kofman AT, Arendzen JH(1994) Reflex sympathetic dystrophy: early treatment and psy-chological aspects. Arch Phys Med Rehabil 75:442–4463. Hunter JM, Mackin EJ, Callahan AD (1995) Rehabilitation of theHand: Surgery and Therapy, 4th edn., vol I, Chapter 6. Mosby, StLouis, Missouri4. Ignjatovic I, Potic B, Paunkovic L, Ravangard Y (2002)Automutilation of the penis performed by the kitchen’s knife.Int Urol Nephrol 34:113–1155. Kontoyianni A, Maragou M, Alvanou E, Kappou I, Dantis P(1999) Unilateral distal extremity swelling with pitting oedema ingiant cell arteritis. Clin Rheumatol 18:82–846. Merskey H, Bogduk N (eds) (1994) Classification of chronic pain:descriptions of chronic pain syndromes and definition of terms,2nd edn. IASP Press, Seattle7. Moesker A (2000) Complex regional pain syndrome, treatmentwith ketanserin and carnitine. Thesis, Erasmus University,Rotterdam, The Netherlands8. Smith RJ (1975) Factitious lymphedema of the hand. J Bone JtSurg Am 57:899. Swift DW, Walker SE (1995) The clenched fist syndrome.Arthritis Rheum 38:57–6010. Zollinger PE, Tuinebreijer WE, Kreis RW, Breede RS (1999)Effect of vitamin C on frequency of reflex dystrophy in wristfractures: a randomised trial. Lancet 11:2025–2028Eur J Plast Surg (2007) 29:245–247 247

An uncommon cause of unilateral hand and forearm oedema

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An uncommon cause of unilateral hand and forearm oedema

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