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Article history: Purpose: Unilateral Condylar Hyperplasia (UCH) is an acquired deformity of the mandible, which can
Paper received 16 March 2018 highly influence the symmetry of the face due to its progressive nature. It is caused by growth resembling
Accepted 5 June 2018 pathology in one of the mandibular condyles. Definition as well as classification is subject to discussion.
Available online 25 June 2018
The aim of this study is to evaluate a large cohort of alleged UCH patients, and to describe the clinical
characteristics, demographic features, classification and follow up. Secondly an algorithm is presented, in
Keywords:
order to achieve uniformity in diagnosis and treatment.
unilateral condylar hyperplasia
Patients and methods: From 1994 to 2014 a database of consecutive patients from 3 maxillofacial de-
Unilateral condylar hyperactivity
Mandibular asymmetry
partments (Academic Medical Center, Amsterdam; VU Medical Center, Amsterdam and Spaarne Gasthuis,
Hemimandibular hyperplasia Haarlem) with suspected UCH was set up. Patients were referred by orthodontists, dentists, general prac-
Hemimandibular elongation titioners or maxillofacial surgeons. Demographic features, bonescan outcomes, laterality, classification and
Demographic follow-up were noted. Secondarily, all patients were retrospectively diagnosed by one surgeon (JWN), using
available documentation. Missing data and follow-up were additionally retrieved from orthodontic offices.
Results: 394 asymmetric patients were evaluated. In 309 (78%) patients, the diagnosis UCH was justified and
SPECT data were available. The mean age at presentation was 20.3 years (SD ± 7.7, range 9.0e54.5 years). In
48% of the patients, the bonescan was positive. 80% of these patients received surgical treatment, of which
62% were treated with a condylectomy only, 33% were treated with condylectomy plus additive corrective
surgery, and 5% underwent corrective surgery only. Of the patient group without positive bonescan 42% of
the patients received surgical treatment: 34% condylectomy only, 15% condylectomy plus additive corrective
surgery, and 51% corrective surgery only. In total (N ¼ 309) 96 (31%) patients underwent condylectomy as
only surgical treatment and 124 (40%) patients received no surgical treatment at all. Treatment could be
finalized with orthodontic treatment without further surgery in 64% and 41% respectively. 96 patients were
subject to comparison of the classification as noted by the clinician and the author (JWN). In only 72% of the
cases, the secondary screening was in agreement with the initial classification.
Conclusion: Based on this study not all (active) UCH patients require corrective (orthognathic) surgery. A
(transoral) partial condylectomy for active patients is recommended, with a postoperative remodeling
period of 6 months with or without orthodontic treatment. Second stage correcting surgery may be
necessary upon evaluation, using general orthognathic diagnostic and planning procedures. It appears
difficult to classify patients reliably using the available clinical and radiological documentation. Objec-
tivity and quantification in the diagnostic process is necessary: uniformity in documentation and pa-
rameters. The attached documentation form and UCH treatment algorithm is recommended.
© 2018 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights
reserved.
*
Manuscript was presented at the following meetings ICOMS Hong Kong 2017.
* Corresponding author. Department of Oral and Maxillofacial Surgery, Academic Medical Centre, Meibergdreef 9, 1105 AZ, Amsterdam, The Netherlands. Fax: þ31 20
5669032.
E-mail address: j.w.nolte@amc.nl (J.W. Nolte).
https://doi.org/10.1016/j.jcms.2018.06.007
1010-5182/© 2018 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved.
J.W. Nolte et al. / Journal of Cranio-Maxillo-Facial Surgery 46 (2018) 1484e1492 1485
Table 1
Different strategies for correction in UCH.
Type of corrective Corrective surgery Corrective surgery No need for corrective Same stage condylectomy
surgery surgery? and orthognatic surgery
Stage END STAGE DELAYED STAGE NO STAGE ONE STAGE
Considerations Severe asymmetry Surgery twice Unpredictable Overtreatment?
Long-term stigmata Chance for observation Pre-existing deformity?
1486 J.W. Nolte et al. / Journal of Cranio-Maxillo-Facial Surgery 46 (2018) 1484e1492
The aim of this study was to evaluate a large cohort of alleged Fig. 2: The mean age at presentation was 20.3 years (SD ± 7.7,
UCH patients, and to describe the clinical characteristics, de- range 9.0e54.5 years), with a male to female ratio of 135: 174 (44%:
mographic features, classification and follow up. An algorithm is 56%). The right side was predominantly affected (right: 168 left: 140
created in order to achieve uniformity in diagnosis, documentation (55%: 45%). In 1 patient, the laterality was not noted in the patient
and treatment. chart nor in the SPECT-application form, and thus scored as un-
known laterality: 1 (0%).
2. Materials and methods Fig. 3: 148 (48%) patients were diagnosed as “active” UCH, based
on the progressive clinical information in combination with a
From 1994 to 2014 a database of consecutive patients from 3 positive bonescan. The remaining 161 (52%) patients showed no
maxillofacial departments (Academic Medical Center, Amsterdam; significant left to right difference on the bonescan, or the results
VU Medical Center, Amsterdam and Spaarne Gasthuis, Haarlem) were controversial to the clinical observation (eg. right side activity
with suspected UCH was maintained. All patients were clinically on bonescan with left side clinically suspected). Of the 148 patients
supervised by 3 surgeons (DBT, AGB, JWN). All patients were with active UCH, 118 (80%) received surgical treatment; 30 patients
referred with suspected progressive asymmetry of the mandible by did not receive surgical treatment at all (20%). Of the surgically
an orthodontist, dentist, general practitioner or maxillofacial treated patients, 73 (62%) underwent a growth-stopping con-
surgeon. dylectomy as the only surgical treatment. 39 patients (33%) un-
Patient charts were reviewed and the following parameters derwent condylectomy plus additive corrective surgery. Corrective
were noted: surgery included orthognathic surgery and/or transpalatal
Gender, date of birth, date of first visit, date, number and distraction osteogenesis (TPD), genioplasty and gonial angle
outcome of bone scintigraphies, the presence of orthodontic gear correction. 6 patients received corrective surgery only (5%).
during treatment, date and laterality of performed condylectomy 94 of the 161 patients (58%) without a positive bonescan
and date and specification of orthognathic or corrective surgery. received no surgical treatment. Surgical treatment in 67 patients
Patient charts were screened for laterality and classification (HE/ without a positive bonescan was subdivided as follows: 23 (34%)
HH/HY/not noted). One surgeon (JN) separately diagnosed every condylectomy only, 10 (15%) condylectomy and corrective surgery,
patient in laterality and classification (HE/HH/HY), using available 34 (51%) corrective surgery only.
photographs, X-rays and/or CBCT, cast models. Missing data and In summary, within the population of 309 patients, 124 (40%)
follow up were additionally retrieved from orthodontic offices. patients did not receive any surgical treatment. 96 (31%) received a
When insufficient information was available, it was noted as “not condylectomy only, 49 (16%) underwent a condylectomy and
possible to diagnose”. additional corrective surgery and 40 (13%) patients underwent
Descriptive statistics were used to evaluate the retrieved data. corrective surgery only. In 3 (1%) patients, a revision of the con-
This research was approved by the Ethical Committee (VUMC, dylectomy was necessary due to ongoing asymmetric deviation of
ref. nr 2012/156). the mandible.
A specification of the 89 corrective treatments is provided in
3. Results Table 2.
Fig. 4: In the condylectomy only patient group (N ¼ 96), treat-
3.1. Demographics, clinical characteristics and follow up ment could be finalized orthodontically in 61 (64%) patients; or-
thodontic treatment was started pre-condylectomy in 49 of these
In the period of 20 years, 394 asymmetric patients were eval- 61 patients (80%). In the group of UCH patients that received no
uated. In Figs. 1e4, the following outcomes are shown. surgery at all (N ¼ 124), treatment was finalized with orthodontics
Fig. 1: 15 (4%) patients were excluded because of known facial in 51 (41%) patients. In the corrective surgery group (N ¼ 89), 7 (8%)
trauma, bilateral condylar hyperplasia and other diagnosis such early cases received surgery without orthodontics.
as hemifacial microsomia, cleft, etc. In 379 (94%) patients, the
clinical and radiological diagnosis of UCH was justified. For 3.2. Classification
further evaluation, another 70 (18%) patients were excluded
because of missing SPECT data. The remaining 309 (78%) patients Table 3: The available documentation of 309 patients was
were analysed. reviewed (JWN) to score again the classification in HE, HH or HY,
Fig. 2. Gender, mean age at presentation and laterality in UCH patients (N ¼ 309).
without insight in the primary diagnosis. In 63 (20%) of the the SPECT-scan and degree of hyperactivity, and surgical
screened cases, there was insufficient documentation to be able to possibilities.
classify the type of UCH. The classification of the remaining 246
patients by the author was as follows: HE 183 (74%), HH 47 (19%)
and HY 16 (7%). 4.1. Classification
On initial diagnosis by the clinician, a specification of the sub-
type was provided in 96 patients (31% of 309). The classification According to these results it proves to be difficult to classify
was as follows: 54 HE (56%), 35 HH (37%), HY 7 (7%). patients reliably. Although the classification parameters of
Thus, 96 patients were subject to comparison of the classifica- Obwegeser are quite clearly described (Obwegeser and Makek,
tion as noted by the clinician and the author (JWN). In only 72%, the 1986), the subjective nature of interpreting the clinical obser-
secondary screening was in agreement with the initial classification vation and available documentation appears to be an important
and laterality. In the HE group 9 (9%) patients were scored differ- factor. Only in 72% of the patient group the secondary screening
ently, in HH 13 (14%) patients, and in HY 5 (5%) patients. matched the initial classification. In many cases, it is difficult to
decide which side is the “affected” side. A one-sided hemi-
mandibular hyperplasia may resemble a hemimandibular elon-
gation on the other side (Fig. 5) and it is sometimes even
4. Discussion difficult to predict which side is expected to be more active on
bone scanning. Classifying a patient who has obvious canting of
Diagnosis and treatment of UCH is subject to different ap- the occlusal plane with a slight midline shift and chin deviation
proaches. It varies per surgeon or maxillofacial center and is but no signs of hyperplasia or open bite may be confusing. In
dependent on the patient's wishes and expectations. Different fact, it can be considered “HE with canting” but that does not fit
parameters are responsible for decision-making in the man- the classic types of Obwegeser. Nitzan observed that clinical
agement of this condition, such as type of UCH, interpretation of appearance may be different from the radiological observation
Fig. 3. Distribution of characteristics in UCH patients; C ¼ condylectomy only; C þ CS ¼ condylectomy þ corrective surgery; CS ¼ corrective surgery only.
1488 J.W. Nolte et al. / Journal of Cranio-Maxillo-Facial Surgery 46 (2018) 1484e1492
and points out that the direction of asymmetry (ie vertical or asymmetries, and the term hemimandibular hyperplasia only
horizontal) should be used instead of the Obwegeser classifica- when actual deformity develops (Chen et al., 1996). Khorsandian
tion (Nitzan et al., 2008). With modern three-dimensional et al., and Xu et al., define subtypes of HH with a typical form
quantitative measurements, Nolte et al. also could not objec- including enlarged condyle, and an atypical form with an increase
tively confirm Obwegeser's classification (Nolte et al., 2015, in volume of ramus and body but no enlargement of the condyle
2016). (Khorsandian et al., 2001; Xu et al., 2014). All of these different
In 2014, Wolford proposed a new classification system for all classification proposals point out that true classification is very
condylar hyperplasia related asymmetries in 4 categories based difficult and that there is large heterogeneity in mandibular
on type of pathology and occurrence rates. Type 1A and 1B show asymmetries. Misdiagnosis of UCH can also occur due to other
respectively bilateral and unilateral elongated accelerated pitfalls or similar conditions. A small transverse maxillary
growth, mainly in pubertal patients. Type 2 includes unilateral dimension, or the presence of sliding in a forced bite situation,
vertical condylar enlargement that is supposed not to be self- may mimic a progressive asymmetry. Although at first sight this
limiting, and expresses either non-exophytic growth of the seems obvious or easy, a small transverse maxillary dimension
condyle (2A) or enlargement of condyle with exophytic growth of with (unilateral) collapse leading to crossbite, can paradoxically
the head (2B), based on osteochondroma. Type 3 and type 4 can appear as hemimandibular ‘elongation’ of the mandible. It can be
cause unilateral facial enlargement at any age, including also subtly asymmetric and may mislead the surgeon or orthodontist
known causes such as osteoma and other benign tumours (type to think there is a mandibular asymmetry. Mouallem et al.
3), or malignant tumors (type 4) (Wolford et al., 2014). Chen et al. describe that mandibular laterognathia in maxillary transverse
recommend using the term condylar hyperplasia in case of discrepancies results in contralateral cross-bite and may cause
Table 2
Specification of corrective surgeries.
TPD 1 3 4*
SS unilateral 1 1
SS bilateral 7 5
SS þ genioplasty 1 1
SS þ angle contouring 1
SS þ genioplasty þ angle contouring 1
Le Fort 3 1
Le Fort þ SS unilateral 7
Le Fort þ SS unilateral þ genioplasty 1
Le Fort þ zygoma þ genioplasty 1
Bimax 11 12
Bimax þ genioplasty 11 y 3
Bimax þ genioplasty þ angle contouring 1
Bimax þ zygoma 1
Bimax þ zygoma þ genioplasty 3 1
Genioplasty 3 1
Genioplasty þ angle contouring 1
Unspecified 1 1
TOTAL 42 7 40
*1 + TMD. y out of 11 bimax+genioplasty, 3 genioplasties were performed simultaneously with the first condylectomy.
J.W. Nolte et al. / Journal of Cranio-Maxillo-Facial Surgery 46 (2018) 1484e1492 1489
Table 3 is the most important factor. Although the mean age of UCH is
Classification of UCH patients. around 21 years of age, there is a substantial number of patients
Classification Clinician Author Difference presenting in childhood or early adolescence (up to 18 years). In
(N¼96) (N¼246) (N¼96) the Netherlands, orthodontic therapy is easily available for chil-
HE 54 (56%) 183 (74%) 9 (9%) dren under 18 years of age; asymmetries, and in particular pro-
HH 35 (37%) 47 (19%) 13 (14%) gressive conditions such as UCH, may therefore be identified
HY 7 (7%) 16 (7%) 5 (5%) earlier and in milder cases. Early diagnosis of asymmetry should
TOTAL 27 (28%)
however not lead to overdiagnosis and (surgical) overtreatment
of UCH. In this study, 40% of the UCH patients received no surgery
at all, and 31% were treated with a condylectomy only. Treatment
could be finalized with orthodontics without further surgery in
adaptive condylar elongation without condylar increase in vol- 64% and 41% respectively. This reinforces the authors’ opinion
ume or occlusal plane tilting (Mouallem et al., 2017). Even uni- that a delayed stage (method 2) or non-stage (method 3) strategy
lateral masseteric hypertrophia can mimic hemimandibular can be recommended to prevent further invasive treatments. The
hyperplasia in clinical appearance. In general, asymmetric dental morbidity of a condylectomy is low (Saridin et al., 2010), and
and skeletal features may be subsequent to compensational di- currently the trans-oral approach for condylectomy becomes
common practice (Herna ndez-Alfaro et al., 2016). The absence of
mensions because of UCH, or just the result of structural asym-
metries in other parts of the face such as the maxilla or soft a visible scar, the relatively small intraoral incision and the
tissues. Proper standard documentation, preferably with ortho- minimal postoperative morbidity plead for this approach instead
dontic documentation, is advisable for accurate diagnosis, and as of a wait-and-see policy until ceasing of activity to try to avoid the
Olate et al. stated: “the study of TMJ and condylar hyperplasia condylectomy. Di Blasio et al. concluded in young patients up to
must be incorporated into all treatment protocols for facial 18 years, that after condylectomy, the healthy side continues to
asymmetry as a starting point for any treatment” (Olate et al., grow in a normal way, and the affected side first ceases growth
2013). after condylectomy, but then recovers to normal growth again (Di
Blasio et al., 2015). Xavier et al. described a two-stage (delayed
stage) treatment, but noticed that after the first stage (con-
4.2. Treatment options dylectomy) noticeable improvements in occlusion were already
visible (Xavier et al., 2014). Farina et al. studied the possibility of
As described in the introduction, there are roughly 4 methods condylectomy as a sole treatment (non-stage) and found satis-
for treatment management in UCH: end-stage, delayed stage, fying results, concluding that a proportional condylectomy re-
non-stage and one-stage surgery. Abuzinada et al. described a duces the need for secondary orthognathic procedures (Farin ~a
great diversity in presentation of condylar hyperplasia. They et al., 2015, 2016).
stated that assessment of condylar growth activity is the most A relevant question would be if we can predict which of the 4
important factor in treatment management and concluded that described methods would be the best treatment in an early stage.
after that, each case has its own diverse treatment plan to reach Objective determination of progression is an important key to
satisfactory facial symmetry (Abuzinada and Alyamani, 2012). The this problem and therefore, uniformity in SPECT-registration and
authors of this study also suppose that different approaches may SPECT-interpretation is highly relevant. At present, there is a
lead to the same satisfying results, but prevention of overtreatment variety of ways to present the SPECT-data from nuclear physician
to the clinician (Saridin et al., 2009). Especially in growing chil- vertical distortion, even without clear signs of hyperactivity
dren it is important to consider that the mandible can grow (Elbaz et al., 2014).
intermittently on each side. Thus, the combination of patient Based on the findings in this study, the authors propose the
history (hetero-) anamnesis, clinical observations and documen- attached diagnostic standard form for documentation (appendix
tation are obligatory in addition to the SPECT-scan before A). This will enable comparison of series of UCH patients, which
deciding on any surgical treatment. In fact, a SPECT-scan is not will lead to better understanding of this rare disorder. In
justified if there is no circumstantial evidence for progression of appendix B, the proposed treatment algorithm is presented. If
mandibular asymmetry with UCH characteristics. In this study the patient presents with mandibular asymmetry and suspected
group, the majority (52%) of the patients turned out to have a UCH on first visit, the standard documentation is performed,
negative or controversial bone-scan outcome, but 33 (20%) of paying special attention to the clinical and radiological charac-
these patients showed clinical progression leading to con- teristics such as dental compensation, maxillary or occlusal
dylectomy anyway. plane canting. The type of asymmetry is noted, and if there are
Secondary procedures sometimes may be indicated to correct clear signs for progression of the asymmetry, a SPECT-CT is
the remaining asymmetry. An observation period after con- made. According to the outcome of the bone-scan, a high con-
dylectomy is advisable before indicating further therapy (delayed dylectomy is performed, or in case of a negative or controversial
stage, method 2). Elbaz et al. concluded that after early con- outcome, a wait and see period is conducted with repeat of
dylectomy an interval of at least 6 months should be respected documentation after 6 months. If progression becomes visible, a
before indicating second stage surgeries (Elbaz et al., 2014). The high condylectomy is performed. After a remodeling period of 6
one-stage approach (combining the condylectomy with orthog- months, further corrective surgery can be done, dependent on
nathic surgery, method 4) may look like an efficient treatment. the level of remaining asymmetry and malocclusion. In this
But, after a partial condylectomy, remodeling may lead to phase, the normal orthognathic treatment schedules and con-
acceptable occlusion and facial features without further correc- cepts are used.
tions. The one-stage approach may be considered as over-
treatment in an unknown amount of cases. Naturally, this decision
5. Conclusion
is also related to the age of the patient and the amount of defor-
mity that might need surgical correction in the first place,
Based on the findings of this study it becomes clear that not
regardless of ongoing growth. In this study, corrective surgery was
all (active) UCH patients require corrective (orthognathic) surgery
performed in addition to the condylectomy in 49 (16%) patients,
for the correction of the asymmetry. A (transoral) partial con-
and 40 (13%) patients received corrective surgery without pre-
dylectomy is recommended, with a postoperative remodeling
ceding condylectomy. The condylectomy was combined with
period of 6 months with or without orthodontic treatment. After
simultaneous other surgery in 7 out of 49 cases (14%), this con-
this, second stage corrective surgery may be necessary upon
cerned simultaneous transpalatal distraction or simultaneous
evaluation. The type of orthognathic surgery is dependent on
genioplasty.
individual cases according to severity and nature of the remain-
ing asymmetry. The use of an algorithm may be helpful, but
general orthognathic diagnostic and planning procedures are
4.3. Relation between classification and treatment options recommended.
An objective and standard diagnostic process is mandatory,
Although classifying UCH patients proves to be difficult, an preferably with uniformity in parameters to compare series from
interesting question would be whether the type of UCH (HE, HH, different centers. This includes standardized clinical assessment,
HY) or vector of asymmetry matters in the treatment choice and 2D and 3D radiologic imaging, and dental casts with accurate
stage. One can imagine that the HE patient group can very well wax-bites. SPECT data should be interpreted in a standardized
benefit from a partial condylectomy in the early hyperactive protocol. The authors recommend the attached protocol and
phase of the disease. The patients in the HH patient group usu- algorithm.
ally demonstrate more complex asymmetry. This kind of asym-
metry can be more difficult to correct (ie lower mandibular
Sources of support
border correction). Further research should be directed to the
None.
growth patterns after condylectomy, and ideally reveal if an early
partial condylectomy in HH causes “natural correction” of the
asymmetric deformity of the mandible in the same way as in HE. Financial interest in this study
Xu et al. proposed a surgical management algorithm on the basis None.
of the severity of the deformity in HH, distinguishing a typical
type and an atypical type (Xu et al., 2014). However, none of the
patients in their group had undergone a condylectomy, so it can Conflicts of interest
be considered more as an orthognathic asymmetry algorithm None.
than as a UCH algorithm. Elbaz et al. introduced a treatment
algorithm that distinguishes functional laterognathia and
condylar hyperplasia, in which the latter was diagnosed with Acknowledgments
SPECT outcome and the degree of vertical deformity. It was
suggested to perform a condylectomy in cases with severe Sophie van de Vijfeijken for her contribution in collecting data.
J.W. Nolte et al. / Journal of Cranio-Maxillo-Facial Surgery 46 (2018) 1484e1492 1491
Appendix A
CLINICAL DOCUMENTATION
FACIAL MEASUREMENTS
Orbital cant : yes no
Nose deviaon : median right le
Lip cant : median right lower le lower
Chin deviaon : median right le
Mandibular angles : equal right lower le lower
Maxillary cant to pupil line : parallel right lower le lower
Occlusal plane cant to pupil line : parallel right lower le lower
ADDITIONAL DOCUMENTATION
Previous orthodoncs : yes no specificaon:
Orthodoncs on first visit : yes no specificaon:
Cast models + wax bite : check
CLASSIFICATION
Obwegeser : HE HH HY NA
Wolford : 1A 1B 2A 2B 3 4 NA
Nitzan : horizontal vercal NA
SPECT-CT
Outcome : mean L: R:
max L: R:
1492 J.W. Nolte et al. / Journal of Cranio-Maxillo-Facial Surgery 46 (2018) 1484e1492