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Case report/Kazuistyka

Pyomyositis in the course of acute lymphoblastic leukemia

Ropne zapalenie mięśni szkieletowych w przebiegu ostrej białaczki limfoblastycznej

Grazyna Sobol-Milejska

1,

*, Agnieszka Mizia-Malarz

1

, Katarzyna Musiol

1

, Tomasz Koszutski

2

, Halina Wos

1

1OddziałOnkologii,HematologiiiChemioterapii,KatedraiKlinikaPediatriiSUM,GórnośląskieCentrumZdrowiaDziecka,Kierownik:prof.dr hab.n.med.HalinaWoś,Katowice,Poland

2KlinikaChirurgiiDziecięcejSUM,GórnośląskieCentrumZdrowiaDziecka,Kierownik:prof.drhab.n.med.JanuszBohosiewicz,Katowice, Poland

Introduction

Pyomyositis is a term used to asses pyogenic infection of the skeletal muscle. Pyomyositis develops as the result of bacteriemia and occurs most commonly in patients with various immunosuppressive diseases. The usual causative

organismisStaphylococcusaureus.Otherpathogensthatmay causepyomyositisarecoagulase-negativeStaphylococcusspe- cies, various streptococcal species, Gram-negative bacteria and fungi.Pyomyositis should beconsidered in the differ- entialdiagnosisinpatientscomplainingofintensivemuscle pain and fever. Routine laboratory investigations are non- specific,andthediagnosisrestsonimagingmodalities[1–6]. actahaematologicapolonica 44(2013) 405–408

article info

Articlehistory:

Received:05.03.2013 Accepted:10.05.2013 Availableonline:23.05.2013

Keywords:

 Pyomyositis

 Acutelymphoblasticleukemia

 Childhood

Słowakluczowe:

 ropnezapaleniemięśniszkieleto- wych

 ostrabiałaczkalimfoblastyczna

 dzieci

abstract

Pyomyositisisatermusedtoassespyogenicinfectionoftheskeletalmuscleanddeve- lopsas theresultof bacteriemiaand occursmostcommonly inpatients withvarious immunosuppressivediseases.Pyomyositisshouldbeconsideredinthedifferentialdiag- nosisinpatientscomplainingof intensivemusclepain andfever.The usualcausative organismisStaphylococcusaureus.Wepresenta3-year-oldboywithacutelymphoblastic leukemia(ALL) and pyomyositiscaused by Pseudomonasaeruginosa diagnosed in the courseofinductiontherapy.Thediagnosisofpyomyositisinthecruralmusclesofboth legs was given based on imaging examinations: ultrasonography and magnetic reso- nance.

©2013PolskieTowarzystwoHematologówiTransfuzjologów,InstytutHematologiii Transfuzjologii.PublishedbyElsevierUrban&PartnerSp.zo.o.Allrightsreserved.

*Corresponding author at: Oddział Onkologii, Hematologiii Chemioterapii, Katedrai KlinikaPediatrii SUM, Górnośląskie Centrum ZdrowiaDziecka,ul.Medyków16,40-752Katowice.Tel.:+48322071745;fax:+48322071745.

E-mailaddress:gsobol@o2.pl(G.Sobol-Milejska).

ContentslistsavailableatSciVerseScienceDirect

Acta Haematologica Polonica

journalhomepage:www.elsevier.com/locate/achaem

0001-5814/$seefrontmatter©2013PolskieTowarzystwoHematologówiTransfuzjologów,InstytutHematologiiiTransfuzjologii.PublishedbyElsevierUrban&PartnerSp.zo.o.Allrightsreserved.

http://dx.doi.org/10.1016/j.achaem.2013.05.001

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Magneticresonancescanisthemostusefulinvestigationin thediagnosis andcanpick upearlychangesinthe muscle [7, 8]. Treatment involves appropriate antibiotic therapy withorwithoutdrainage,accordingtolocalconditions[1–6]. We present 3 years old boy with acute lymphoblastic leukemiaandpyomyositiscausedbyPseudomonasaeruginosa diagnosedinthecourseofinductiontherapy.

Case report

A 3-year-old boy diagnosed with acute lymphoblastic leu- kaemia(ALL),preB type,SRG (standardriskgroup)treated inaccordancewithALLICBFM2002Protocol(AcuteLympho- blastic Leukemia Inter-Continental Berlin-Frankfurt-Mun- ster). We observed a good response to the treatment.

Becauseoftheobservedfeveranddiarrheainthecourseof neutropenia,thechemotherapywashaltedonthe18thday ofinductiontherapy.Theculturesof bloodandstoolswere negative.Theantibiotic cefepimwasadministeredandwas continued until the 10th day. On the 3rd day after the completionoftheantibiotictreatmentthechildcomplained ofintensepaininthelowerlimbs,especiallyinthecrura.In thephysicalexaminationtherewasonlyapaininthecrura during palpation.Laboratory testsrevealeda high valueof ESR(erythrocytessedimentationrate;180mm/h),C-reactive protein (CRP; 177mg/l) and creatine kinase (CK; 788.0U/l).

Tests for the presence of myoglobin in urine came back negative whichexcluded rhabdomyolysis.Typical bacterio- logicaltests,includingbloodculturecamebacknegative.On the6thdayafterthepatient'snotificationofpainanedema and redness of the calves appeared. Due to the pain the child was not able to walk despite the administration of analgesics.Theultrasonography(USG)examinationrevealed significant extension of contoursand intensifiedechogeni- city of the subcutaneous tissue and crural muscles which suggestedaninflammatoryprocess.Abroad-spectrumanti- biotic therapy (imipenem, teicoplanin) and an antifungal therapy(amphotericinB)wereintroduced.

After a10-daytherapy the edema,rednessof cruraand painonpalpationreduced. Parametersoftheinflammatory status improved significantly (ESR 54mm/h, CRP10.1mg/l) and the controlled level of CK was normal (72U/l). Itwas decided toresume theinduction chemotherapy while con- tinuingtheantibiotic therapy.3weeksaftertheoccurrence of theailments the paininthe cruraintensifiedagainand aphysical examination revealedanincreased warmthand edema. Another USG examination showed a two-sided elevatedechogenicityandfluidbetweenthecruralmuscles.

A magnetic resonance (MRI) revealed two excessive fluid areas of 2.2  4.3cm and 2.4  5.7cm in the area of the left crural muscles. Similar lesions of 4.1  2.3cm and 4.3 1.4cmwereobservedin therightcrus(Fig.1, Fig.2).

The lesions observed in the crura were consistent with pyomyositis. The fluid areas were drained and pus was extruded. The culture revealed P. aeruginosa. The crural pains retreated completelyupon the incision and drainage of the lesions. A 14-day antibiotic therapy compliant with the antibiogram (tazobactam/piperacyllin) was introduced andthe drainageof the cruralsoft tissues wasmaintained

for7days.Theclinicalconditionallowedforacontinuation ofchemotherapy.Thefollow-upUSGofthecruraperformed on the 7th day after the surgery did not show fluid reservoirs. On the 3rd day after the completion of the antibiotictherapyamassivepneumoniawasdiagnosed.The child rapidly developed respiratory insufficiency and requiredtreatmentat theIntensive CareUnit.After 4days the childwasadmitted toourdepartmentinanimproving condition. The antibiotic therapy continued until the 14th day. The patient underwent a rehabilitation of the lower extremities. The clinical condition of the child improved completelywhichenabledthecompletionofthechemother- apy. Theboyfeelswell andthe motoractivityof hislower extremitiesisnormal.

Fig.1–TheMRIimages–pyomyositisareasinthecrural muscles,cross-section

Ryc.1–ObrazMRI–ropnezapaleniemięśnipodudzi,przekrój

Fig.2–TheMRIImages–pyomyositisareasinthecrural muscles

Ryc.2–ObrazMRI–ropnezapaleniemięśnipodudzi acta haematologicapolonica 44 (2013) 405–408

406

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Discussion

Pyomyositisisan endemicdisease commonlydiagnosed in tropical regions; hence the name ‘‘tropical pyomyositis’’.

Reports of cases from temperate climates are seldom and concentrate on immunocompromised patients or patients withunderlyingchronicdisease[1–6,9–11].Pyomyositishas three stages, from diffuse inflammation to focal abscess formationandtoasepticstate.Itbeginswith theinsidious onsetof progressivepain with lowgrade fever and muscle ache. In second stage muscle abscess formationsare orga- nizedandinthenextstagethemanifestationsofsepsismay be observed [1–3]. Due tonon-specific symptoms the diag- nosis is rarelygiven inthe early stage of the disease. The differential diagnosis should include above others venous thrombosis, osteomyelitis, intramuscular haematoma and rhabdomyolysis and in the case of a situation when the processinlocalisedintheiliopsoasmuscle,alsoappendicitis [1–6]. In the case of patients with leukaemia, treated with cytostaticdrugs, aswith our patient, initialsymptoms sug- gested drug-induced toxicity above all after steroids and vincristine[9–11].Thelaboratorytestsresultsarenon-specific forthecourseofpyomyositis,whichmayindicatemoderate leucocytosiswithaleftshift,elevatedESRandnormalmuscle enzymelevels[1–6].USGexaminationsmayrevealpyomyosi- tis, but if the USG picture is inconclusive MRI is the most usefulimagingtechniquefordiagnosis.Becauseofitsavail- ability,aUSG examinationremainsveryhelpfulinmonitor- ingthosepatients[7,8].Thetreatmentdependsonthestage ofthe diseaseinwhichitwasdiagnosed. Anearlystageof pyomyositis,thesocalleddiffuseinflammatoryinfiltrationof themuscle istreated exclusivelywith antibiotics,involving antibiotics recommended for the treatment of infections caused by S.aureus, such asvancomycin or teicoplanin. In thecaseofanabscess,apartfromantibiotictherapy,itisalso importanttoinciseanddrainit.Accordingtotheauthorsof publicationonthispyomyositis,therapeuticsuccessiscondi- tionalonearlydiagnosis,theuseofarelevantantibioticand surgical intervention[1–6,9–11]. What probably could have contributedtoamorecomplicatedcourseofpyomyositis in thepresentedcase,washisunderlyingdisease–leukaemia.

Asaresultofboththediseaseandtheappliedchemotherapy, significantimmunologicaldeficitsareobserved,whichinthe relevantliterature isconsidered as a significant risk factor.

Another important risk factor for a severe course of the diseasewasisaninfectioncausedbytheaggressivePseudo- monas aeruginosapathogen[9–11].Asimilar clinical course ofpyomyositis ina5year-oldboybutof atypicalS.aureus etiologywaspresentedbyTaksandeetal.[1].

Pyomyositisissporadicallydiagnosedinchildrenwhichis confirmedbyonlyafewpublicationsonthismatter.Publica- tionsonpyomyositisconcerningoncologicalchildren,includ- ingthosewithleukaemia aresparse. Aconsiderablygreater numberofpublicationsconcernadultpatients.Falagasetal.

in their study presenting a case of an adult woman with pyomyositis of the iliopsoas muscle in the course of the Hodgkin'slymphomareviewedthe literature onthesubject oftheoccurrenceofthisdiseaseinpatientswithhaematolo- gical malignancies and analysed 44 cases including only

4children[12].Blatetal.presentedthecasesof2boyswith acute lymphoblasticleukaemia,whointhecourseofremis- sion-inducingtherapy,duringneutropenia,developedmulti- focal pyomyositis, similarly to our patient [9]. Also Corden etal.describedintheirpublicationcasesof2boyswithALL and pyomyositis diagnosed during the induction of the remission[10].Kaoetal.reportedonacaseofa10-year-old girl treated for ALL in whom pyomyositis localised in the thoracicmusclesandthighwasdiagnosedduringtheremis- sion-inducing therapy. The antibiotic therapy (vancomycin) corresponding with the results of the culture of the pus content(S.aureus)andsurgicalinterventionwerecompletely effective [10]. According to the available literature, so far 5casesofchildrenwith acutelymphoblasticleukaemiaand pyomyositis during the induction therapy have been pub- lished. Our case report seems to be the 6th one following asimilarclinicalsuit[9–11].Inallthepatientsthecourseof the disease in its initial stage was non-characteristic and suggesteddrug-inducedtoxicity.Inthecasesofchildrenwith allpublishedtodate,S.aureuswasineachcasethecauseof pyomyositis.Thepathogenresponsibleforthediseaseinour case was P. aeruginosa, an etiological factor so far not published in the group of cases of children with ALL and pyomyositis.Thesebacteriamaybethereasonforthemore complicatedcourseofpyomyositisinourpatient.

To summarize, in children with acute lymphoblastic leukaemia,local musclepainsduring theinductiontherapy require in the differential diagnosis the consideration of pyomyositis and instantimaging examination (MRI) of the affected area. Theuse of abroad-spectrum antibiotic ther- apy withpossible surgicalintervention is also of a crucial importance.

Authors' contributions/Wkład autorów

GS-M – study design, data collection and interpretation, manuscript preparation. TK, HW – study design. AM-M – data collection and interpretation, manuscript preparation, literaturesearch.KM–literaturesearch.

Financial support/Finansowanie

Nonedeclared.

Conflict of interest/Konflikt interesu

Nonedeclared.

Ethics/Etyka

Thework describedinthisarticlehavebeen carriedoutin accordance with The Code of Ethics of the World Medical Association (Declaration of Helsinki) for experiments involving humans; EU Directive 2010/63/EU for animal experiments; Uniform Requirements for manuscripts sub- mittedtoBiomedicaljournals.

acta haematologicapolonica 44 (2013)405–408

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references/pi smiennictwo

[1] TaskandeA,VilhekarK,GuptaS.Primarypyomyositisina child.IntJInfectDis2009;13:149–151.

[2] MitsionisGI,ManoudisGN,LykissasMG,etal.Pyomyositis inchildren:earlydiagnosisandtreatment.JPediatrSurgery 2009;44:2173–2178.

[3] GubbayAJ,IsaacsD.Pyomyositisinchildren.PediatrInfect DisJ2000;19:1009–1019.

[4] WeinbergJ,FriedmanS,SoodS,CriderRJ.Tropicalmyositis (pyomyositis)inchildrenintemperateclimates:areportof 3casesonLongIslands,NewYork,andreviewofthe literature.AmJOrthop2007;36:E71–E75.

[5] DrososG.Pyomyositis–aliteraturereview.ActaOrthop Belg2005;71:9–16.

[6] SmallLN,RossJJ.Tropicalandtemperatepyomyositis.

InfectDisClinNorthAm2005;19:981–989.

[7] KarmazynB,KleimanMB,BuckwalterK,etal.Acute pyomyositisofpelvis:thespectrumofclinicalpresentations andMRfindings.PediatrRadiolog2006;36:338–343.

[8] YuCW,HsiaoJK,HsuCY,ShihTT.Bacterialpyomyositis:

MRIandclinicalcorrelation.MagnResonImaging 2004;22:1233–1241.

[9] BlattJ,ReamanG,PizzoP.Pyomyositisinacute lymphpblasticleukemiaheraldedbycutaneosvasculitis.

MedPediatrOncol1979;7:237–239.

[10] CordenTE,MorganER.Pyomyositisduringinduction chemotherapyforacutelymphocyticleukemia.JPediatr HematolOncol1996;18:323–326.

[11] KaoKL,HungGY,HwangB.Pyomyositisduringinduction therapyforacutelymphoblasticleukemia.JChinMed Assoc2006;69:184–188.

[12] FalgasME,RafailidisPi,KapaskelisA,PeppasG.

Pyomyositisassociatedwithhematologicalmalignancy:

casereportareviewoftheliterature.IntJInfectDis 2008;12:120–125.

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