coronoid hyperplasia

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YIJOM-2398; No of Pages 7

Please cite this article in press as:MulderCH, et al.Coronoid process hyperplasia: a systematic review of the literature from 1995, Int J

Oral Maxillofac Surg (2012), http://dx.doi.org/10.1016/j.ijom.2012.03.029

Systematic Review Paper

TMJ Disorders

Coronoid process hyperplasia:a systematic review of theliterature from 1995C. H. Mulder, S.I. Kalaykova,R.A. Th. Gortzak: Coronoidprocess hyperplasia: asystematic review of the literaturefrom 1995. Int.J. OralMaxillofac. Surg. 2012;xxx:xxxxxx. # 2012 International Association of Oral and Maxillofacial Surgeons.Published by Elsevier Ltd. All rights reserved.

C. H. Mulder, S. I. Kalaykova,R. A. Th. Gortzak

Department of Oral and Maxillofacial Surgery,Leiden University Medical Center, TheNetherlands

Abstract. The objective of this study was to review the literature and comparedifferent surgical methods for the management of coronoidprocess hyperplasia. Aliterature search was performed for publications since 1995. Case characteristicswere extracted (age, sex, duration of symptoms, form, maximal mouth opening andtreatment) and entered into a database for analysis. The data were split into twogroups (coronoidectomy and coronoidotomy). Maximal mouth openingmeasurementsbefore and after surgery were analyzed with several statistical tests.61 cases were entered into the database. The mean age was 23 years and meanduration of symptoms 7 years. The bilateral form occurred 4.1 times morefrequently than the unilateral form. The malefemale ratio was 3.3 to 1. In 94% ofthe cases the approach was intra-oral. 84% of the cases received a coronoidectomy.Statistical analysis showed that the preoperative and postoperative differencesbetween the groupswere significant.The results were not significantwhen correctedfor the preoperative difference. Postoperative therapy was not comparable due toheterogeneity. Cases that received a coronoidotomy had slightly betterpostoperative results.

Keywords: Coronoid process hyperplasia; Cor-onoidectomy; Coronoidotomy; Limited mouthopening.

Accepted for publication 20 March 2012

Mandibular

coronoid

process

hyperplasia(CPH) is a rare condition causing a slow,progressive reduction of mouth opening.1

CPH is defined as an abnormal elongationof the mandibular coronoid process con-sisting of histologically normal bone.1

This leads to impingement of the coronoidprocess on the body or arch of the zygo-matic bone on opening of the mouth.2,3

To date, mainly single case reports ofCPH have been published. In the mostrecent complete review published in1995 by Mcloughlin et al.,1 31 new cases

of

coronoid

hyperplasia

were

reportedtogether with a meta-analysis of previousdata. They emphasized the normal histol-ogy of the resected coronoid process todistinguish it from otherpathology. It wasfound that the condition most oftenaffected adolescent men. Surgery wasthe treatment of choice, although the out-come was generally disappointing, possi-bly due to the formation of a haematomaor intra-oral fibrosis.The authors hypothe-sized that the extra-oral approach mightcause less fibrosis, but too few extra-oral

surgeries

were

performed

to

compare

theirpostoperative measures with those ofintra-oral surgeries. Postoperative phy-siotherapy (stretching exercises) wereconsidered to be essential for the preser-vation of the increased mouth opening.1

In this article, a systematic review ofcases published since the review ofMcloughlin et al.,1 ispresented. The mainobjective is to compare the results ofdifferent surgical methods (e.g. intra-oralvs extra-oral, coronoidectomy vs. coronoi-dotomy).

Int. J. Oral Maxillofac. Surg. 2012; xxx: xxxxxxhttp://dx.doi.org/10.1016/j.ijom.2012.03.029, available online at http://www.sciencedirect.com

0901-5027/000001+07 $36.00/0 # 2012 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
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Materials and methods

A systematic search in the Pubmed data-basewas conducted to find related articles.In the search, the following Medical Sub-jects Headings (MeSH) terms were used:coronoid, hyperplasia, and mandible.The following free-text terms were

entered as synonyms. For the term cor-onoid, synonyms coronoid process,processus coronoideus and processusmuscularis were entered. For the termhyperplasia, synonyms elongation,impingement and enlargement wereentered, as well as the MeSH term hyper-trophy. For mandible, mandib* andlower jaw were entered as synonyms.Boolean operatorORwas appliedbetweensynonyms. The operator AND was usedbetween the three search terms.The search was limited to articles in

English describing cases, published in, or

after,

1995.

Titles

and

abstracts

wereassessed to select relevant articles, andthen the full-text articles were retrieved.The reference lists of the selected articleswere manually checked to trace additionalcases. Throughout the search, cases wereexcluded if no hyperplasia with impinge-ment was present, and/or histology andmorphology of the coronoid process wascharacteristic for an osteochondroma.From the included articles, specific

case-characteristics were extracted andentered into a database as numerical orcategorical data. Numerical data included

age at

diagnosis,

duration

of

symptoms,maximum mouth opening (MMO)before,during and after the operation and lengthof follow-up.Categorical data consisted ofuni- orbilateral CPH form, sex, diagnosticmethod, surgical method, and whetheradditional physiotherapy was performed.Two additional variables were calculated:age at onset and MMO improvement.To evaluate if there was a statistically

significant association between sex anduni- or bilateral type of CPH, a x2-testwas performed. The authors carried outseveralstatistical tests on theoutcome data.They split the surgery types into twogroups: coronoidectomy and coronoidot-omy groups. Cases were filtered out thathad an extra-oral approach or underwentadditional masseter stripping to make thecoronoidectomy group more homogenous.After testing normality of the distributionwith a residuals histogram, an independent ttest was done to compare the means of thepreoperative MMO, final MMO and MMOdifference. A univariate analysis of var-iance (UNIANCOVA) was performed onthefinalMMOwith thepreoperativeMMOas covariate. Differences were considered

to be significant if p


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cases, hypoplasia of the mandible andmasticatory muscles was present whichalso contradicts the temporalis hyperactiv-ity theory.Two reports suggest the influence of

mandibular hypomobility. Zhong et al.reported a case where an osteochondromawas found on one side and CPH on the

other.33 In this case the hyperplasia couldhave developed secondarily to the hypo-mobility caused by the osteochondroma.Wenghoefer et al. reported two patientswho had ankylosis of the TMJ and twoothers with arthritis beginning destruc-tion.28 These findings support the mandib-ular hypomobility theory.Some authors suggested trauma was

associated with this condition.33 In thepresent authors database, only one case(2%) reported trauma,20 so they did notfind evidence to support this theory.A new hypothesis on aetiology was

mentioned

by

Wenghoefer

et

al.

Theyinvestigated the occurrence of ankylosingspondylitis (AS) in CPH and found it tobepresent in 4 of 16 patients.28 The mainfeature ofAS is sacroiliitis and subsequentossification. They suggest that a similarmechanism might occur in the temporalistendon, although they could not confirmthis with histopathology. It is known thatin patients with AS the TMJ can also beaffected, which was the case in those fourpatients.No conclusive evidence was found to

support or discard the abovementioned

theories,

and

the

true

aetiology

of

CPHremains unclear.

Diagnostic tools

Orthopantomography (OPT) was carriedout in 87% of the cases in the database as adiagnostic imaging method to recognize acoronoid abnormality.Coronoid hyperpla-sia is suspected on OPT when its heightexceeds that of the condyle9 Levandoskipanographic analysis can be conducted tocalculate the ratio between the length ofthe coronoid and condylarprocess.Kubotaet al. found this ratio in 3 cases with CPHtobe significantly higher than the ratio in acontrol group9 It was concluded that if theratio exceeded 1.1, additional imaging toconfirm CPH was needed.Computed tomography (CT) is the pre-

ferred method to visualize CPH, because aCT can accurately visualize the relationbetween osseous structures, such as thecoronoid process and zygoma.2,3 In parti-cular, a scan with an opened mouth canprove and depict the exact location ofimpingement.32 In 51 cases (84%), CTwas carried out to confirm a diagnosis

of CPH. In 24 patients (39%), a three-dimensional (3D) reconstruction was car-ried out. 3DCT canbe used to evaluate themorphology in more detail.3,13 In 30% ofthe reports, zygomatic exostoses weredescribed at the location of impingement,on the medial surface of the zygomaticarch or dorsal surface of the zygomatic

body.2 In CPH, the coronoid process iselongated but relatively normal in shape.CT imaging is useful for differentiating

between CPH and other coronoid abnorm-alities. An osteochondroma has a charac-teristic stalked appearance also describedas a mushroom or condyle shaped. Thediagnosis can be confirmed with histo-pathology of the resected process, whichshows a bonymass coveredwith a cartilagecap and endochondral ossification at thedeep aspect. In 74% of the cases from thedatabase the removed specimens were sentfor histopathology and all were stated to

consist of

normal

bone

tissue.

In

3

cases,

apreoperativeMRI scanwas takenbecause aTMJ abnormality was suspected.4,15

Treatment

The condition is treated by surgery,because the restriction is principallycaused by a mechanical obstruction. Bothintra-oral and extra-oral approaches havebeen described. Two types of surgery areperformed: coronoidectomy and coronoi-dotomy.

The

intra-oral

approach

usually

pro-vides enough exposure to remove thehyperplastic process and leaves no visiblescar. The biggest disadvantage is the riskof a postoperative haematoma and fibro-sis. Several extra-oral approaches havebeen described, such as submandibular,pre-auricular, (bi)temporal3,4 or endosco-pically assisted.10 Supposed advantagesare less fibrosis and/or haematoma forma-tion, no intra-oral scarring and betterexposure to resect the coronoid processand release the temporalis muscle. The

risk of facial nerve damage and a visiblescar are the main disadvantages.During a coronoidectomy, the tempor-

alis muscle fibres are stripped from theprocess after which it is entirely resected.Advantages are that the mechanical causeof the impingement is removed and his-tology of the specimen can be undertaken

to confirm or revise the diagnosis. On theother hand, the release of the temporalisinsertion can be a difficult and traumaticprocedure. In a coronoidotomy theprocessis sectioned at the base and left in situ.Supposedly this method leads to lesstrauma, less postoperative morbidity andbetter results. Disadvantages are the riskof recurrence caused by reattachment ofthe process and the inability to performhistology. In some cases additional mass-eter muscle stripping was performed toincrease mouth opening because musclesmay undergo fibrotic changes after a sig-

nificant period

of

disuse.7,25

Postoperative physiotherapy is consid-ered to play an important role in main-taining and increasing the MMO. Activeand passive stretching exercises with orwithout the use of abiteblock,20 spatulas,7

a mouth screw,2,7 a wedge,32 dynamicdevices20 and a TheraBite123,24,27,28werereported.In 9 of the reported cases (15%) nothing

was stated about therapy, or the patientsrefused surgery. The intra-oral approachwas used most frequently; in 47 cases(94%). In 3 cases (6%) the approach

was extra-oral.

The

coronoidectomy

wasthe preferred method in 42 cases (84%);the remaining 8 cases had a coronoidot-omy. In 6 cases (12%) the surgeonsdecided to perform additional massetermuscle stripping. In 5 cases (10%) theMMO data were incomplete, so thosecould not be included for MMO analysis.For the remaining 45 cases the results aresummarized in Table 2.With an independent t test, the authors

calculated that the coronoidotomy grouphad a significantly larger preoperative

4 Mulder et al.




Table 2. Outcome measures for different surgery types.

Surgery type

CoronoidectomyCoronoido-tomy Total

Intra-oral Extra-oral Total

N 34 3 37 8 45Percentage 75.5 6.6 82.2 17.8 100Mean final MMO (mm) 34.0 33.3 34.0 40.8 35.2Range (mm) 2246 2045 2046 3548 2048MMO 30 mm (%) 79.4 66.6 78.4 100 82.2MMO 35 mm (%) 52.9 66.6 54.1 100 62.2Mean DMMO (mm) 19.5 22.7 19.7 22.1 20.2

N, number of cases; Range, minimum and maximum.
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(p = 0.003) and final MMO (p = 0.016)compared to the coronoidectomy group.In the UNIANCOVA the preoperativeMMO was included as a covariate in thecalculation. This p-value (0.069) was notsignificant. With this test the outcome wascorrected for the significant difference in

preoperative

MMO

(Fig.

1).

An

indepen-dent t test was also performed on theMMO difference, which confirmed thedifference between the groups was notsignificant (p = 0.376).Some form ofphysiotherapywas applied

in 45 of 50 cases that received surgery(90%). In the case reports, different timesof initiating physiotherapy and exercisesand different duration were noted makingoutcome comparison impossible.Follow-up data in the articles analyzed

was diverse. In most cases, several post-operative measurements at different timepoints were reported. The length of fol-low-up was specified in 49 cases (80%),ranging from 4 to 60 months with a meanof 14 months.Regeneration of the coronoid process

after coronoidectomy was described in 2cases in the database.21,31 Both cases werediscovered by imaging, not due to theclinical features.

Discussion

This review provides an update on theliterature that hasbeenpublished regarding

CPH. The authors attempted to summarizeand meta-analyze the findings from 61cases. A comparison of results after coro-noidectomy or coronoidotomy has notbeenmade inprevious literature. Other findingsaresimilar to those reportedbyMcloughlinet al.1

The

authors

extracted

more

data

thanprevious reviews for the database. This isdue to more extensive documentation,new diagnostic tools and new exerciseappliances. The authors chose to limitthe search to articles that were publishedafter the review by Mcloughlin et al.,1

because they provided a quite completereview and the present authors wanted tocompare their results with those ofMcloughlin et al. This caused a selectionbased on publication date, but also agreater chance of having a more completedatabase for comparing data. The searchwas limited to English literature, becauseof accessibility and language.After careful consideration some cases

were excluded, because the authors ques-tioned whether the correct diagnosis hadbeen made. Izumi et al. and Murakamiet al. diagnosed their patients with CPH,while they emphasized no impingementwas present12,18. The present authorsview is that the MMO restriction is prin-cipally caused by the coronoid processimpinging on the zygoma. Leonardiet al. conducted the Levandoski pano-graphic analysis in 10patientswith nevoid

basal cell carcinoma syndrome17 and found4 to haveCPH,but the ratios they calculatedwere well below the lowest ratio Kubotaetal. found in theirpatients.9Based on thesefacts the present authors did not includethose cases. In the latter of the excludedcases, the diagnosis of osteochondroma washighly suspected, because of characteristic

morphology and/or histology.6,15,30,37CPH appears to be a rare condition but

little is known about its true incidence orprevalence. Two studies have been pub-lished that tried to objectify this. In 1987Isberg et al.published a prospectivestudy inwhich they investigated patients with amouth opening restriction and found therestriction was caused by CPH in 5%.36

This number does not represent the trueprevalence because selection had takenplace based on symptoms. It does indicatethat CPH should not be overlooked as acause for limited MMO. The second was a

retrospective

study

of

2000

randomOPTs.38

They found unilateral hyperplasia in 1 case,so a prevalenceof 0.05%.What is debatableis that this patient did not have restrictedmouth opening, the main feature of CPH.The authors found the average age at

diagnosis was 23 years, near the previousaverage of 25 years. The average length ofhistory is also in accordance withpreviousdata.1 The age distribution for age at diag-nosis shows a peak in cases in a youngerage group than that which contains themean age. The authors think this givesmore valuable information than the mean

agealone

which

is

influenced

by

the

range.With the age at onset calculation theauthors observed the highest case countis in an even younger group. This findingcan lead to more clinical awareness of theonset of this condition in a youngerpatientgroup. It is hoped this will lead to adecrease in misdiagnosis and duration ofsymptoms. The ratio for uni- or bilateralform is supported by previous literature.1

The authors also found the conditionaffects men more often than women (aratio of 3.3 to 1), although the previouslystated ratio was 5 to 1.Several theories havebeen suggested to

explain the aetiology of CPH. The influ-ence of the temporalis muscle has beensuggested by numerous authors. Isberget al. found that coronoid process elonga-tion could be induced by mandibularhypomobility and temporalis hyperactiv-ity from a study in monkeys.35 Otherfindings supporting these theories are theshortened muscle tendon units in trismuspseudocamptodactyly syndrome,25 fibrousand hypertrophied masticatory muscletendons3,7,24 and hypertrophic or hyper-tonic temporalis muscles.28 These features

Coronoid process hyperplasia: a systematic review of the literature from 1995 5




Fig. 1. Scatterplot ofpreoperative and final MMO measurements, illustrating the difference inMMO distribution for the two surgery types. Circles and squares represent separate cases.
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are not present in every CPH patient andseveral EMG studies revealed no abnorm-alities. Nevertheless more findings werereported supporting this theory thanopposing it.All 3 of the authors patients had pro-

minent mandibular angles as was seen onthe OPT. One of them had apparent mass-

eter hypertrophy. The other two had par-afunctional habits. Isberg et al. observedbone deposition occurring in the area ofthe insertion of the masseter muscle due tohyperactivity,35 which lead to the appear-ance of a square-shaped mandible (SQM).Other cases of the association betweenSQM and CPH have been reported inliterature. Yoshida et al. published a casewith hyperplasia of the coronoid pro-cesses, masseter muscles and mandibularangles.29 Murakami et al. reported 12cases, but in their patients no coronoidelongation or impingement waspresent.12

In

fact

only

a

few

cases

were

reported

thatmentioned this association. The aetiologyof hyperplastic mandibular angles is notclearly stated. Masseter muscle hyperac-tivity was suggested to have an influ-ence.35 This indirectly supports thetemporalis hyperactivity theory for CPHbecause the masticatory muscles are clo-sely related. No conclusive evidence wasfound affirming the aetiology of either ofthese conditions.For the MMO analysis, the authors used

the last stated MMO after surgery for thefinal MMO. This was not measured at the

same

point

in

time

in

all

cases.

This

mighthave an effect on the analysis, although theauthors expect it to be limited becauseMMO stabilizes after some time. In theirreview Mcloughlin et al. wanted to com-pare the intra-oral approach with the extra-oral one.1 Since their review, only 3 extra-oral approaches havebeen reported, so thepresent authors could not compare theresults of the two approaches either.Wenghoefer et al. reported 14 cases butdid not specify which approachwas used.28

The authors assumed they used an intra-oralincision for inclusion in the MMO analysis.An interesting observation was made

when the outcome data were split intotwo groups (coronoidectomy and coronoi-dotomy). The authors found a significantdifference inpreoperative and final MMO,but not in the MMO improvement. Thepower of these findings is limited by thefact that the group sizes were not equal (30vs 8) and the postoperative therapy mea-sures could not be taken into account.A coronoidotomy is supposed to be an

easierprocedure,which results in a shorterduration of surgery. It was thought thatthe obstruction would reoccur when the

process reattached to the ramus, requiring are-operation,39 but in 5 cases the coronoidhad repositioned and reattached in a poster-ior fashion not causing restriction at longterm follow-up.5 This means the supposedmain disadvantage did not occur. Anotherdisadvantage is that no histopathology canbe performed. This investigation was par-

ticularly useful in the differentiation froman osteochondroma. Nowadays with helpof a 3DCT an osteochondroma can also berecognized based on the morphology, sohistopathology is less crucial to the diag-nosis. Thus, because of the slightly betterresults and an easier procedure it seemsuseful to perform more coronoidotomiesto evaluate if those outcome measures sup-port the observation.It was difficult to compare the post-

operative therapy data due to heterogene-ity and limited reporting.The authors usedthe TheraBite1 appliance for passive

stretching

which

led

to

satisfactory

resultsin those cases. There are no guidelines onfrequency of exercises or duration. Ideallya randomized controlled trial should beperformed to evaluate differentpostopera-tive therapy options,but this seems impos-sible because the abnormality occurs soinfrequently.In the cases analyzed, follow-up data

were diverse. In the authors cases a con-siderable dip waspresent in thepostopera-tive course. At some point in the earlypostoperative course the MMO was evensmaller than the preoperative measure-

ment,

despite

adequate

exercising.

Withintensification and continuation of rehabi-litation measures the MMO graduallyincreased to satisfying levels. In otherreports a similar course has beendescribed,20,24,32 so clinicians should notbe discouraged by this observation. Theauthors advise regular follow-up, espe-cially for the first 3 months, so patientcompliance and therapy canbe improved.In the literature it was stated that the

results after surgery were generally dis-appointing. The question arises what out-come should be regarded as beingdisappointing. According to the AAOMSimpairment guidelines a MMO of 35 mmor more was considered to be an accep-table interincisal distance.40 Others con-sidered a mouth opening of 30 mm ormore to be successful, so the authors alsocalculated that percentage.28 The overallsuccess rate if the first criterion is usedwasfairly disappointing (62%). The other cri-terion results in an 82% success rate. So itis of great influence which value is chosenfor evaluation of success.In conclusion, theauthors foundthat there

seems to be slightly better postoperative

mouth opening in patients who received acoronoidotomy. This procedure is alsoassumed tobe faster and easier.More casestreated in this way are necessary to confirmor discard this finding.9

Funding

None.

Competing interests

None declared.

Ethical approval

Not required.

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Leiden

University

Medical

CenterP.O. Box 96002300 RC LeidenThe NetherlandsTel.: +31 71 5262371fax: +31 71 5266766E-mail: [email protected]

Coronoid process hyperplasia: a systematic review of the literature from 1995 7


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protected]://dx.doi.org/10.1016/j.ijom.2012.03.029mailto:[email protected]://www.aaoms.org/docs/practice_mgmt/impairment_guidelines.pdfhttp://www.aaoms.org/docs/practice_mgmt/impairment_guidelines.pdfhttp://www.aaoms.org/docs/practice_mgmt/impairment_guidelines.pdfhttp://dx.doi.org/10.1016/S0901-5027(89)80143-2http://dx.doi.org/10.1016/S0901-5027(89)80143-2http://dx.doi.org/10.1016/S0901-5027(89)80143-2http://dx.doi.org/10.1016/S0278-2391(97)90189-0http://dx.doi.org/10.1016/S0278-2391(97)90189-0http://dx.doi.org/10.1016/S0278-2391(97)90189-0http://dx.doi.org/10.1097/SCS.0b013e3181c46a30http://dx.doi.org/10.1097/SCS.0b013e3181c46a30http://dx.doi.org/10.1097/SCS.0b013e3181c46a30http://dx.doi.org/10.1016/j.tripleo.2008.10.017http://dx.doi.org/10.1016/j.tripleo.2008.10.017http://dx.doi.org/10.1016/j.tripleo.2008.10.017http://dx.doi.org/10.1016/j.joms.2009.04.022http://dx.doi.org/10.1016/j.joms.2009.04.022http://dx.doi.org/10.1016/j.joms.2009.04.022http://dx.doi.org/10.1111/j.1600-9657.2009.00830.xhttp://dx.doi.org/10.1111/j.1600-9657.2009.00830.xhttp://dx.doi.org/10.1111/j.1600-9657.2009.00830.xhttp://dx.doi.org/10.1016/j.ijom.2007.07.021http://dx.doi.org/10.1016/j.ijom.2007.07.021http://dx.doi.org/10.1016/j.ijom.2007.07.021http://dx.doi.org/10.1097/SCS.0b013e318176ac3bhttp://dx.doi.org/10.1097/SCS.0b013e318176ac3bhttp://dx.doi.org/10.1097/SCS.0b013e318176ac3bhttp://dx.doi.org/10.1097/scs.0b013e3180a772bahttp://dx.doi.org/10.1097/scs.0b013e3180a772bahttp://dx.doi.org/10.1097/scs.0b013e3180a772bahttp://dx.doi.org/10.1016/j.bjoms.2006.05.005http://dx.doi.org/10.1016/j.bjoms.2006.05.005http://dx.doi.org/10.1016/j.bjoms.2006.05.005http://dx.doi.org/10.1016/j.joms.2005.10.032http://dx.doi.org/10.1016/j.joms.2005.10.032http://dx.doi.org/10.1016/j.joms.2005.10.032http://dx.doi.org/10.1007/s00508-006-0663-5http://dx.doi.org/10.1007/s00508-006-0663-5http://dx.doi.org/10.1007/s00508-006-0663-5http://dx.doi.org/10.1016/j.joms.2004.07.018http://dx.doi.org/10.1016/j.joms.2004.07.018http://dx.doi.org/10.1016/j.joms.2004.07.018http://dx.doi.org/10.1038/sj.dmfr.4600672http://dx.doi.org/10.1038/sj.dmfr.4600672http://dx.doi.org/10.1038/sj.dmfr.4600672http://dx.doi.org/10.1038/sj/dmfr/4600646http://dx.doi.org/10.1038/sj/dmfr/4600646http://dx.doi.org/10.1038/sj/dmfr/4600646http://dx.doi.org/10.1054/bjom.1999.0159

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