A giant multi lobed osteochondroma of the phalanx in an adult A case report A c M a b a A R A A K O A H 1 a r m m r e a d s o s c s o R h 2 c CASE REPORT – OPEN ACCESS International Journal of Surgery[.]
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jo u r n al ho me p a g e :w w w c a s e r e p o r t s c o m
Mohammad M Al-Qattana,b,∗, Felwa Al-Marshada, Jumana Al-Shammaria, Atif Rafiqueb
a The Division of Plastic Surgery at King Saud University, Riyadh, Saudi Arabia
b The Division of Plastic Surgery at King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia
a r t i c l e i n f o
Article history:
Received 24 October 2016
Accepted 25 December 2016
Available online 27 December 2016
Keywords:
Osteochondroma
Adult
Hand
a b s t r a c t
literature
hand
1 Introduction
Solitaryosteochondroma ofthehandisa rareentity.Almost
allpreviouslyreportedcasesinthehandweresmalltumorsbut
requiredexcisionbecauseofconcurrentsymptoms
Inchildren,mostsolitarytumorsarisefromthenon-epiphyseal
metaphysisorthemetaphysisontheepiphysealplatesideofthe
middle or proximalphalanges of the fingers [1] These tumors
restrictmotionandcauseprogressivefingerdeformityandhence
earlysurgicalexcisionisrecommended[1]
Solitary osteochondromas of the hand which develop in
adulthoodare extremely rare and have different presentations
dependingonthesiteoforiginofthesetumors.Forunknown
rea-sons,mostadultsolitarytumorsariseeitherfromthedistalphalanx
orinthecarpalbones.Distalphalangealtumorsalmostalwaysarise
subunguallyandcausenaildeformity[2].Tumorsarisingfromthe
carpalbonesmaycauseextensortendonrupture[3],carpaltunnel
syndrome[4],orpain[5]
夽 This project was funded by the College of Medicine Research Center, Deanship
of Scientific Research, King Saud University, Riyadh, Saudi Arabia.
∗ Corresponding author at: The Division of Plastic surgery, King Saud University,
Riyadh, Saudi Arabia.
E-mail address: moqattan@hotmail.com (M.M Al-Qattan).
Inthispaper,wepresentararecaseofagiantmulti-lobed osteo-chondromaofthephalanxdevelopinginanadult.Nosimilarcases werefoundintheliterature.Theworkhasbeenreportedinline withtheSCAREcriteria[6]
1.1 Presentationofcase
A25-yearoldmalepresentedwithaone-yearhistoryofaslowly growingmassoftherightmiddlefinger.Therewasnohistoryof trauma,painorotherswellingsinthebody.Examinationshowed
amulti-lobedbonymasslocatedonthevolaranddorsalaspects
ofthemiddlephalanx(Fig.1).Therewerenosensorydeficits.The proximalinterphalangealjoint(PIPJ)hada25◦offlexion contrac-tureandtherangeofmotioninthearcofflexionofthePIPJwasalso restrictedto90◦becauseofthemasseffect.Plainx-raysshowed
amulti-lobedosteochondromaarisingfromthebaseofthe mid-dlephalanx(Fig.2).MRI(Fig.3)showedthatthetumormeasured 2.3×2.5cmandalsoshowedthemedialdisplacementoftheflexor tendonsbythetumormass.Totalexcisionwithbonegraftand pos-siblefusionofthePIPJwasplannedbutthepatientrefusedany compromiseoffunction.Thepatientagreedtoundergomarginal excisionknowingtheriskofrecurrence.Resectionwasdone pre-servingtheneurovascular bundles,theflexor/extensor tendons, andthePIPJ.Inordernottocompromisethebloodsupplyofthe overlyingskin,thevolarlobeofthetumorwasresectedfirstviaa volarlazy“S”incision;andonemonthlaterthedorsallobeofthe
http://dx.doi.org/10.1016/j.ijscr.2016.12.015
2210-2612/© 2016 The Author(s) Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd This is an open access article under the CC BY-NC-ND license ( http://
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Fig 1.(A–C) Clinical presentation of a multi-lobulated osteochondroma of the finger in an adult.
tumorwasresectedviaadorsallongitudinalskinincision
Histo-logicalexaminationwasconsistentwithbenignosteochondroma
showingregularbonytrabeculaearrangedat90◦totheoverlying
cartilaginouscap(Fig.4).Thepostoperativecourseswere
unevent-ful.Atfinalfollow-up8 monthslater, therewasnoevidenceof
recurrence.Sensoryexaminationofthefingerdidnotrevealany
deficits.TheflexioncontractureatthePIPJimprovedfrom25◦to
15◦andtherangeofmotioninthearcofflexionofthePIPJimproved
from90◦to110◦(Fig.5).X-raysshowedtheresidualtumoratthe
baseofthemiddlephalanx(Fig.6)andthepatientwasadvised
forlongtermfollow-up.However,hedidnotcomebackafterthe
8-monthvisit
2 Discussion
Osteochondromasarecommonbenignbonytumorswhichhave
acartilaginouscap.Themajorityareseenarisingfromthelong
bones,particularlyaroundthekneejointandintheupperhumerus
Osteochondromasofthehandarerareandareusuallyseenin
chil-drenaspartofthemultipleexostosessyndromessuchashereditary
multipleexostoses[7]andMuenkeSyndrome[8].Asmentionedin
theintroduction,solitaryosteochondromasofthehandarerare
inchildrenandextremelyrareinadults.Ourcasewasinanadult
andhadseveraluniquefeatures:theoriginbeingfromthemiddle
phalanx,thelargesizeandbeingmulti-lobed.Themulti-lobulation
requiredstagedresectiontoprotectthebloodsupplyofthe
over-lyingskin.Ourliteraturereviewdidnotrevealanysimilarcasein
theadulthand.Onecaseofalargemulti-lobedosteochondroma
ofthehamateinachildwasreportedandresectionwasalsodone
throughseparatevolaranddorsalincisionspreservingtheulnar
nerve[9]
Asymptomatic solitary osteochondromas of the long bones
maybetreatedconservativelywithregularfollow-up.Complete
resectionisthetreatmentofchoiceforsymptomaticsolitary
osteo-chondromas in adults In our case, complete resection meant
compromiseofPIPJmotionandthepatientrefusedourinitialplan
ofmanagementofcompleteexcision.Incomplete(marginal)
resec-tionshouldtakeintoconsiderationtheriskofrecurrenceandalso
theriskofmalignanttransformation.Theprevalenceofmalignant
Fig 2. Plain X-ray showing the tumor arising from the base of the middle phalanx.
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Fig 3.MRI of the tumor.
Fig 4.Histology showing the regular bony trabeculae covered by the cartilage cap
(H & E × 100).
transformationofbenignosteochondromasvariesfromlessthan
1%forsolitarytumorsand4%forhereditarymultipleexostoses[10]
Inrecurrenttumors,acartilaginouscapthicknessofgreaterthan
2cm(asassessedbyMRIorCTScan)stronglyindicatesasecondary
chondrosarcoma[10]
Large osteochondromas involving the phalanges of adults
shouldbedifferentiatedfrombizarreparosteal
osteochondroma-Fig 5.(A&B) Range of motion 8 months after surgery.
tousproliferation(BPOP)whichisalsocalledNora’slesion[11]
Itisusuallyseeninadultsandalmostalwaysarisefromthe pha-langes,metacarpalsormetatarsals.Radiologically,Nora’slesions resembletheappearanceof solitary osteochondromasalthough matrix calcificationis moreirregular in Nora’slesions.The dif-ferentiation betweenBPOP and osteochondroma is made from histologicalexamination.BPOPiscomposedofarandommixture
offibroustissue,irregularbonytrabeculaeandcartilagecells.In contrast,osteochondromasarecomposedofregularbony trabecu-laearrangedat90◦totheoverlyingcartilagecapasseeninFig.4 Finally,therecurrenceratefollowingexcisionismuchhigherin patientswithBPOPcompared tothose withsolitary osteochon-dromas[11].Infact,spontaneousresolutionofapediatricsolitary osteochondromahasbeenreported[12]
3 Conclusion
Ararecaseoflargemulti-lobedosteochondromaofthephalanx
inanadultispresentedandthemanagementisdiscussed
Conflict of interest
None
Funding
TheworkwassupportedbytheCollegeofMedicineResearch Center, Deanship of Scientific Research, King Saud University, Riyadh,SaudiArabia
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Fig 6.X-ray 8 months after surgery showing the residual tumor.
Ethical approval
ThestudywasapprovedbytheResearchcommittee ofKing
FaisalSpecialistHospitalandResearchCenter,Riyadh,SaudiArabia
Consent
Writteninformedconsentwasobtainedfromthepatientfor
publicationofthiscasereportandaccompanyingimages.Acopy
ofthewrittenconsentisavailableforreviewbyEditor-In-Chiefof
thisjournal
Allauthorscontributedsignificantlyandinagreementwiththe contentofthemanuscript.Allauthorsparticipatedindata collec-tionandinwritingofthemanuscript
Guarantor
MMAl-Qattan
References
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