Accepted Manuscript

Ten Years of Observations and Demographics of Hemimandibular Hyperplasia and

Elongation

Kamil H. Nelke, DMD, PhD, Wojciech Pawlak, DMD, PhD, Monika Morawska-
Kochman, MD, PhD, Klaudiusz Łuczak, DMD, PhD

PII: S1010-5182(18)30103-3
DOI: 10.1016/j.jcms.2018.04.003
Reference: YJCMS 2945

To appear in: Journal of Cranio-Maxillo-Facial Surgery

Received Date: 20 August 2017

Revised Date: 23 March 2018
Accepted Date: 4 April 2018

Please cite this article as: Nelke KH, Pawlak W, Morawska-Kochman M, Łuczak K, Ten Years of
Observations and Demographics of Hemimandibular Hyperplasia and Elongation, Journal of Cranio-
Maxillofacial Surgery (2018), doi: 10.1016/j.jcms.2018.04.003.

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 ACCEPTED MANUSCRIPT
Ten Years of Observations and Demographics of Hemimandibular Hyperplasia and

Elongation. ab

Kamil H Nelke DMD, PhD ab*, Wojciech Pawlak DMD, PhD b, Monika Morawska-Kochman MD,

PhD c, Klaudiusz Łuczak DMD, PhD b

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a
Department of Dental Anatomy, Head Prof Wiesław Kurlej, ul. T. Chałubińskiego 6a, 50-368

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Wrocław, Medical University, Poland, phone: +717841337, fax: +717840079, e-mail:

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wieslaw.kurlej@umed.wroc.pl
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b
Department of Otolaryngolgoy, Head and Neck Surgery, Head Prof Marek Bochnia, ul.

Borowska 213, 50-556 Wrocław, Silesian Piast’s Medical University, Poland, phone
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+717343700, fax: +717343709

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b
Department of Maxillo-Facial Surgery, Head AJ Komorski, PhD, ul. Borowska 213, 50-556
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Wrocław, Silesian Piast’s Medical University, Poland, phone +717343600, fax: +717343609

e-mail: kamil.nelke@gmail.com
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*Corresponding author: Kamil Nelke, DMD,PhD, Department of Maxillo-Facial Surgery,

Borowska 213, 50-556 Wrocław, Wroclaw Medical University; tel/fax (+48)717343600

e-mail address: kamil.nelke@gmail.com (K.H.Nelke)

 ACCEPTED MANUSCRIPT

Abstract

Introduction

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The full epidemiology and etiology of hemimandibular hyperplasia (HH) has not yet been

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clarified. In most cases it starts before puberty and results in various forms of dento-alveolar and

skeletal discrepancies. This study is the first attempt at evaluating and describing some of the

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authors’ key experiences, clinical philosophical approach, and gathered demographic data on

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hemimandibular hyperplasia and hemimandibular elongation (HE) among the Polish population.
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Material and method

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A total of 45 patients (M=8; F=37; p<0.05) with HE (n=16; 35.6%; p<0.05), HH (n=28; 62.2%;

p<0.05), or HH+HE (n=1; 2.2%; p>0.05) had been diagnosed and treated. Epidemiological,
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geographical, and clinical data concerning the occurrence and treatment protocols in these
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mandibular malformations were measured in the Polish study groups.

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Results

Women more often suffered from these mandibular malformations (82–87%). The occurrence of

the first symptoms was highest at the age of 13–15 years and was statistically significant for both

sides (p<0.05). The disorders were found earlier in young girls, therefore an early compensatory

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orthodontic treatment in some cases had been used with a limited degree of success (p>0.05). All

values of bone scintigraphy were significant (p<0.001).

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Conclusions

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A very fast growth with visible major asymmetry and enlarged condylar head should be an

indication for condylectomy. Women’s expectations from surgery and treatment are more

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demanding than men’s, a fact that is connected with the predominance of females in the study

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group. Almost all possible treatment alternatives are not only related with the degree of skeletal
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deformity, but also with the patient’s willingness to undergo any necessary treatment protocols,

which in most cases involve more than one stage. Skeletal scintigraphy tests are an important
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factor in estimating bone growth and possible surgical approaches in these disorders.
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1. Introduction
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Hemimandibular hyperplasia (HH) and hemimandibular elongation (HE) were first presented

and described by Obwegeser and Makek (Obwegeser and Makek, 1986). In time, other proposals
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for the classification of condylar enlargement (condylar hyperplasia [CH]) were also presented.
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The terms used to distinguish those conditions might be confusing. Some of them were focused

on the condylar head’s increased growth potential, while others described benign or malignant

temporomandibular joint (TMJ) condyle conditions (Wolford et al., 2014a). One classification

presented (by, e.g., Wolford et al., 2014a) underlines four different CH forms. According to this

classification, CH Type 1 describes the increased prolonged condylar growth during puberty and
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can be bilateral (CH Type 1A) or unilateral (CH Type 2A). Primary or secondary TMJ tumors

will not be discussed in this paper.

Nowadays, both of the above-mentioned classifications describe deformities of the condyle and

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mandible, depending on their clinical severity and existing features. On the other hand,

hemimandibular hyperplasia (HH) is caused by many potential factors that might influence

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mandible condylar enlargement. So far, this disorder has been described as self-limiting,

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abnormal condylar growth, resulting in pathological, one-sided bone overgrowth in the mandible.

Over time, during its continuous growth, it might result in various intense forms of skeletal

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asymmetry and jaw discrepancies. In CH, this condition doesn’t only cause visible symptoms in
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the condylar head of the mandible, but it also affects the neck, ramus, and body of the mandible

and results in facial skeletal asymmetry, malocclusion, masticatory, and speech problems
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(Rodrigues and Castro, 2015). In most cases, it starts before puberty. The mechanism still

remains uncertain, though factors such as vascular anomalies, Meckel cartridge remnants,
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trauma, and endocrine and other factors are taken into consideration (Chia et al., 2008; Nitzan et
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al., 2008; Raijmakers et al., 2012).

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When the first symptoms of facial asymmetry start to occur, it is quite important to determine the

type, etiology, and degree of its progression. In order to determine the growth activity, single-
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photon emission computed tomography (SPECT) or bone scintigraphy should be used

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(Obwegeser and Makek, 1986; Wen et al., 2014; Wolford et al., 2014). The usage of other

diagnostic tools, such as dental plaster models, routine radiographs, and facial photographs are

also very valuable. These diagnostic methods should be repeated over time to evaluate the degree

of asymmetry and its progression.

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So far, however, most papers have described cases, treatment outcomes, and proposals, or have

reviewed minor aspects of this issue. Papers describing first-hand observations are limited. This

paper is focused on the authors’ own experience in the field of hemimandibular hyperplasia and

elongation. Furthermore, to avoid any misunderstandings in the nomenclature of the presented

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conditions, only the two above-mentioned forms will be presented and investigated. Some

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patient data regarding epidemiology, demographics, onset of symptoms, and their relation with

treatment planning based on scintigraphy will be presented and discussed. Despite the fact that

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these pathologies have been known for a relatively long time and are controversial, the aim of

this paper is to focus on the authors’ own personal views and treatment philosophies on those

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pathologies, considering some clinically important and statistically relevant data.
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2. Material and methods

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2.1. Material
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This paper includes the data of patients consulted, diagnosed, and treated at the Department of
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Maxillo-Facial Surgery from 2006–2017 with various forms of HH and HE according to

Obwegeser and Makek’s classification. A total of 45 patients (M=8; F=37; p<0.05) diagnosed
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with only HH, only HE, or hybrid HH+HE malformations were included in the demographical
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study. Almost all of these patients (n=81%; p<0.05) underwent a surgical procedure and had

been diagnosed and operated on according to the authors’ philosophy. In order to avoid any

diagnostic pitfalls, all of the patients’ radiographic and photographic studies, from various time

frames, were taken in the same place by the same technicians on the same equipment, and were

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standardized according to the available data. In a practical sense, the paper underlines a different

view of the problems of HH and HE. The main aim of this paper is to present the authors’

personal clinical experience and point of view, which may be seen as a different approach, yet is

controversial in some stages other than the approach that is known and presented in the available

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literature.

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The Local Ethics Committee and Hospital granted approval for the study: 616/2016 RVB.

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2.2 Methods AN
All of the patients’ clinical charts, along with clinical and radiological data during their

treatment, were archived in MeshLab (version:64bit_v1.3.3–private license), and then analyzed

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and gathered in SPSS-17 Statistical software database using ANOVA and McNemara testing.
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Although the mentioned time-frame is ten years of observation and collected data on mandibular
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deformities, it describes only the authors’ point of view, which is a retrospective study on the

authors’ own past and present perspective.

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Based on the Obwegeser and Makek classification, a total of 45 HH/HE patients were diagnosed

and treated by the authors (p<0.05) (Obwegeser and Makek, 1986). The Study Authors’ own
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clinical observations and treatment results will be discussed and presented.

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There were 7 criteria for inclusion in the study: 1) confirmation of HH/HE based on routine

panoramic and cephalometric radiographs; 2) confirmation in CT/3D and bone

scintigraphy/SPECT; 3) a detailed clinical examination focused on the most typical facial, bone,

and soft tissue symptoms of HH/HE; 4) early or late orthodontic treatment of tilted occlusion
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and/or asymmetry correction; 5) patients scheduled for condylectomy, marginectomy,

transpalatal distraction (TPD), orthognathic surgery, and/or corrective genioplasty; 6) at least 1

year of clinical pre- or postoperative surgical follow-up; and 7) facial photographic evaluation

performed every 3 months.

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The exclusion criteria were mostly related with the presence of TMJ tumors/pseudotumors,

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ankylosis, inflammations, bony dysplasia, cherubism, genetic conditions (ex. hemifacial

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microsomia), and other joint or joint-related conditions, especially cases of severe skeletal

malocclusion in patients without any condyle symptoms.

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Some limitations of the study were also noted. The majority of those restrictions are related with
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surgical techniques in each case, since most patient’s dento-alveolar and asymmetrical

discrepancies were related with individual factors; however, all of them were related with
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various forms of skeletal and dental asymmetries. A discussion of the surgical results and some
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of the surgical techniques used would be beyond the scope of this paper, and because of the
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severity and difficulties in presenting them they will be omitted.

All patients (n=45, 100%; p>0.05) were divided into three study groups (S1, S2, and S3). Group
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S1 was composed of patients diagnosed with HH malformation; Group S2 patients were

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diagnosed with HE, and Group S3 with a combination of HH+HE. All groups were divided into
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subgroups according to the diagnostic procedures performed, the clinical data, and diagnostics

made as preparation for surgery. Furthermore, the group of patients who had finished their

surgical treatment and were currently under routine clinical observation and scheduled for

follow-ups was also studied.

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All study groups were evaluated based on retrospective data relating to demographics,

epidemiology, occurrence (age, gender, side), and radiographic and clinical manifestation (first

diagnostics, first symptoms), as well as minor presentation on some performed surgical

procedures (condylectomy with or without arthroplasty, marginectomy, Lefort I, BSSO,

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genioplasty procedures, camouflage bone shifting, and bone chiseling/modeling techniques) in

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relation to the clinical indications for the treatment used.

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3. Results

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3.1. Age, gender, and site of syndromes

Data from a total of 45 patients (M=8; F=37; p<0.05) (SD=22.17; Mean=23.52) were evaluated
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(Table 1). The essence of the epidemiological data is that the gender-to-age ratio favors young
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women around 22 years of age. Patient age and gender are random (p>0.05), however, a slight
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predominance of women in all age groups was noted (p<0.05). Male patients did seek treatment

and/or diagnostics on average 3 years later than females. The youngest patient was 12 years old,
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while the oldest was 37. The first symptoms were the same, were related with facial asymmetry,

and were most common at the age of 13–15 (n=19; 42.3%). In about 23% of all patients, their
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condylar growth activity presented later than 25 years of age. The occurrence of HH on the right
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vs. the left side of the condyle is almost the same (50% and 46.4%, respectively); however, more

female patients suffer from HH (n=23; p<0.05). HE was less common and found in only 14

women and 2 men (n=16; p>0.05) and was mostly found on the right side of the mandible, which

was longer than the left one (p>0.05). Bilateral forms of HE were the most common ones (87.5%

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of HE cases), and bilateral forms of HH were the rarest (0%). The lower number of male patients

diagnosed might be related to their lack of awareness or to their reduced need to maintain a

symmetrical facial profile.

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3.2. Clinical pre-surgical evaluation and radiological data

Each patient (n=45; 100%) was scheduled for clinical, photographic, and radiological evaluation

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before any surgical intervention. If a patient suffered from both skeletal and facial asymmetry, a

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radiographic evaluation consisting of panoramic and lateral cephalometric radiography was first
AN
performed. It seems that low-dose computed-tomography (LDCT) with bone scintigraphy and

pantomographic radiography were the most common diagnostic tools (p<0.05). In 25 out of 45
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cases, orthodontic treatment (p=0.04) was scheduled; however, only 3 patients had their

removable braces applied before the age of 13, while by the age of 16 about half of the patients
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had their appliances (p=0.038). There was no correlation between the degree of skeletal
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asymmetry and the time of orthodontic treatment (p>0.05). It seems that this situation was

mostly related with the degree of asymmetry and the individual characteristics of the dento-
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alveolar discrepancies, where most of them involved not only a deviated mandible (p=0.035), but
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also a tilted occlusal cant (p=0.671) in more than 60% of the cases. It seems that too many
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variables were responsible for discrepancies in the dento-alveolar segments, which indicates

many possible orthodontic treatment methods. This situation might be related with the time of

the planned surgical approach, which is estimated to be at about 22–24 years of age (n=18;

p=0.197) (Table 2).

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3.3. Computed tomography and scintigraphy data

Facial skeletal scintigraphy indicates that the values of mandibular condyle growth activity vary

greatly, especially in HH and HE cases. There was no correlation between gender, age, or

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skeletal malocclusion and the value of scintigraphic evaluation (p>0.05). In all cases, orthodontic

treatment was used first in all types of skeletal asymmetries and malocclusion (p=0.038; 22%)

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(Table 3). Cases of asymmetrical overgrowth of the mandible were treated later than the HE

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symptoms, due to the prolonged time of accurate radiological diagnosis. In most cases, the

condyle head growth potential was estimated at an average ratio of 0.67–1.85 (n=34), which

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indicates moderate hyperactivity. In some rare cases, the growth ratio was higher than 2.00 (n=6;
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p>0.05). Both tomographic and scintigraphic data revealed a deviation of the mandible, occlusal

cant, and the maxillary horizontal plane in most cases, though their occurrence and degree were
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individual (p>0.05). Increased bone activity was estimated in bone scintigraphy testing (SPECT

and/or PLANAR). All patients, whether they had HH or HE mandibular malformation, were first
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scheduled for a routine bone scintigraphy. It seems that condyle bone activity in HE was in each
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case very low and was estimated to be 0.80 or less (p>0.05), which was not considered active

growth requiring a condylectomy. In those cases, standard orthognathic surgery was

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implemented, but no further bone scintigraphy was scheduled. Perhaps, scintigraphy should also
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be performed in a long-term study in all cases of mandibular malformations, both HH and HE. In
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the presented material, a complete skeletal scintigraphy of the entire skeleton was performed

each time, especially to confirm or rule out active growth in the other areas in the body. Despite

a controversial issue regarding bone scintigraphy performed in growing children, when the value

of scintigraphy could be a false positive, this diagnostic approach is a valuable procedure for

determining the differences in growth activity between both condyles, especially when the first
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symptoms of mandibular progressive asymmetry start to arise. A comparison and standardization

of bone activity in each case was made, but the analysis revealed no statistical significance

(p>0.05). Each case should be carefully examined because a false positive result might be related

with TMJ loading, joint dysfunction, increased mastication on one side, or other factors.

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3.4. Type of surgery depending on scintigraphy finding and the value of asymmetry

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It seems that cases of active and hypertrophic condyles are first scheduled for condylectomy with

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or without arthroplasty (Table 4). The study group in each case of HH had a condylectomy with
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arthroplasty, which consisted of reattaching the lateral pterygoid muscle to the newly shaped

condyle head and repositioning the joint capsule in order to maintain mandibular movements
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(n=13; p=0.03). Procedures such as Lefort I osteotomy (n=11) and BSSO (n=11) remain the

treatments of choice for most cases (p<0.05). In some cases, patients decided on surgical
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camouflage (p=0.624). This method either consisted of a bone-shift (n=4; p=0.264) or, rarely,
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just condylectomy with mandibular bone modeling using burs and chisels. The methods and time

of treatment vary for each case, so there is no gold standard for treatment; however, the authors
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indicate that excessive condyle growth followed by a deviation of occlusal cant in both jaws
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should be a mandatory indication first for condylectomy and then for an orthognathic procedure.
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Individual decision-making was also based on the degree of deviated occlusal cant and maxillo-

mandibular discrepancies. A lack of deviation or a low or moderate degree of asymmetry was

mostly acceptable to the patients. Therefore, those patients were first scheduled for a low

condylectomy (to avoid any potential relapse and to slightly reposition the mandible) and for a

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mandibular marginectomy with or without bone chiseling. These procedures were performed

within a 6-month timeframe or were all combined into one surgery in order to improve facial

balance, especially in those patients who wanted to achieve all outcomes in one procedure. Major

asymmetries and increased vertical and horizontal dimensions of unilateral mandibular

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enlargements were scheduled for a total condylectomy with arthroplasty procedures, which

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resulted in mandibular auto-rotation towards the excised condyle side.

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Severe asymmetry with a deviated bite plane required direct compensatory orthodontic treatment

consisting of Lefort I maxillary osteotomy and BSSO mandibular osteotomy. Also, in a few

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cases only a single condylectomy with mandibular auto-rotation was used with a great overall
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success rate in terms of sufficient restoration of bite and symmetry in the patients’ view. Small

dento-alveolar discrepancies were acceptable to some patients, while others who were unsatisfied
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by the post-condylectomy asymmetry were scheduled for additional surgeries.

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The authors would like to explain that their qualification for surgery was based on condylar head
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activity (scintigraphy) and bone enlargement (CT/LDCT studies) resulting in mandibular

asymmetry. The criteria of choice for treatment should firstly describe the degree of asymmetry
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and the presence or degree of condylar activity. The removal of bone growth should be used as

the first treatment of choice. As an additional tool for qualifying condylectomy, the ratio between
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condylar head shape/size vs. the value of increased growth activity compared to the healthy
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condyle should be used. In the classic definition, condylectomy refers to a complete removal of

the condyle and subsequent joint reconstruction. In the literature, it is divided into high, low, and

complete condylectomy with various indications and techniques defined by some authors. In this

paper, The authors decided that a total condylectomy including reshaping of the new condylar

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head and repositioning of the pterygoid muscles is used when at least one dimension of condylar

head diameter (length, width, or height) is longer than 25 mm along its longest axis in addition to

an active growth value higher than 1.5 (estimated in bone scintigraphy). On the other hand, when

all diameters are 20 mm or less and any value of bone growth is still present on scintigraphy,

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then a low condylectomy procedure with condyle reshaping was performed. The main goal of

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this approach is to focus on avoiding any potential relapses.

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4. Discussion

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The data on epidemiology and mandibular and skeletal asymmetry was linked retrospectively

with the authors’ data. Some points of view might be considered controversial and yet in some
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cases demanding, but these pathologies of the mandible require individual treatment planning in

each case. All of the presented cases of HH and HE have some similarities, but most of them are
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related with individual patient factors, such as growth activity, the type of dento-alveolar
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discrepancies, and the time of treatment (Angiero et al., 2009; Fisch et al., 2011; Pripatnanont et

al., 2005). A combination of two forms, commonly known as hybrid forms, are the most
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challenging ones (Nitzan et al., 2008; Raijmakers et al., 2012). The ratio of males to females
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varies among studies (Obwegeser and Makek, 1986; Angiero et al., 2009; Rodrigues and Castro,
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2015). It seems that a slightly higher predominance of hemimandibular hyperplasia in females is

more common in the available literature and that some authors correlate this finding with a

higher degree of estrogen receptor accumulation in the temporo-mandibular joint area (Nitzan et

al., 2008; Alyamani and Abuzinada, 2012; Pripatnanont et al., 2005). This study indicates a

higher occurrence of these mandibular malformations in females regardless of their age and time
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of puberty onset (82.1–87.5%; p<0.05). The predominance of women with unilateral condylar

hyperplasia seems to also be confirmed by other authors, though population and racial factors

should also be taken into consideration (Angiero et al., 2009; Alyamani and Abuzinada, 2012;

Raijmakers et al., 2012). The deformities start to present themselves earlier in young girls than in

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boys, at the onset of puberty; the peak occurrence of deformity might vary, and is estimated by

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various authors to be about 14–30 years of age (Nitzan et al., 2008), and before 25 years of age.

The authors underline that although young patients in most cases seek help because of

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asymmetry, it is easier to meet the requirements from treatment in patients at a younger age than

in patients older than 27 years, who rarely seek more than just corrected bite, symmetry, and

occlusion.
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In most cases, an inappropriate facial appearance and the patient’s willingness and need to
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undertake any necessary treatment underlines first basis and the need for treatment (Hassani et

al., 2013; Olate et al., 2013; Portelli et al., 2015). In the Study Authors’ opinion, a degree of
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deviated occlusal cant, visible one-sided asymmetrical enlargement of the mandible, and bone
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growth estimated at higher than 1.0 (p=0.006) should be an indication for further diagnostics and

a condylectomy, if necessary (Table 3). Similar indications for condylectomy have been
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addressed by Wolford et al. (Wolford et al., 2014b). One of the issues addressed by the authors is
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having routine scintigraphic measurements carried out at least once a year in growing patients
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only if visible signs of mandibular asymmetry and deformity are present. This still-controversial

step could be helpful in measuring and estimating the bone growth and could indicate the best

time for a surgical approach (Fisch et al., 2011; Mutoh et al., 1991; Nitzan et al., 2008; Olate et

al., 2013; Wen et al., 2014).

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The time of surgery is still a subject of discussion (Obwegeser and Makek, 1986; Olate et al.,

2013; Wolford et al., 2002). The authors conclude that progressive facial asymmetry should not

be considered a contraindication for early surgery. An age of over 18 years, regardless of gender,

is considered the gold standard for condylectomy. In each case of active growth resulting in

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skeletal asymmetry, a condylectomy should be performed (p=0.003) (Table 4). Because of

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patients of different ages seeking consultation for jaw asymmetry, each condylectomy was

performed after gathering two scintigraphic measurements of bone activity. This is similar to

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reports in the literature, though the main difference concerns whether condylectomy should be

performed as the only procedure, or whether it should be performed together with

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marginectomy/bone modeling or perhaps orthognathic surgery (Angiero et al., 2009; Wolford et
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al., 2014; Villanueva-Alcojol et al., 2011; Jones and Tier, 2012; Wolford et al., 2014b). Small

asymmetries with a stable occlusion and less dento-alveolar discrepancies could, in the authors’
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opinion, be an indication for combined condylectomy with osteomodeling and/or an orthognathic

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surgery (p<0.05). On the other hand, in extensive asymmetries—resulting in overall bone

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overgrowth in all three dimensions with increased bone potential—surgeries should be divided

into stages, as suggested by some authors (Jones and Tier, 2012; Olate et al., 2013; Portelli et al.,
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2015) (Table 4). Those steps could more effectively improve bite, aesthetics, and occlusion and

could decrease the risk of relapses. Increased growth potential—regardless of age and gender—
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which visibly increase jaw disproportions and increase maxillo-mandibular plane deviation
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should be considered one of the indicators for total condylectomy; however, this approach is still

controversial and requires close follow-ups and patient monitoring.

It seems that the time of onset of dental, skeletal, and/or facial asymmetry is an important factor

in predicting the growth intensity during puberty (Nitzan et al., 2008; Raijmakers et al., 2012;
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Villanueva-Alcojol et al., 2011; Hassani et al., 2013). The Authors’ study found that among the

Polish study group, a lack of facial balance and symmetry were very worrisome for more women

than men (p=0.085). Most authors indicate that parents or close relatives are the first to notice the

growing asymmetry (Fisch et al., 2011; Lippold et al., 2007; Portelli et al., 2015; Wolford et al.,

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2002). In most cases, as in this presented study, the time when the first symptoms of asymmetry

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present is at the peak of growth, estimated to be at 12–14 years of age (43%; p<0.05) (Wolford et

al., 2014; Fisch et al., 2011; Villanueva-Alcojol et al., 2011). On the other hand, there are no

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accurate data on treatment in children, only in adults (Obwegeser and Makek, 1986; Alyamani

and Abuzinada, 2012; Hodder et al., 2000; Olate et al., 2013; Wolford et al., 2002). However, the

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time when patients seek a surgical consultation is most commonly ≥18 years of age (n=24;
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p<0.05). This finding is also related to the limited success rate with standard orthodontic

treatment in very young and growing patients who underwent early treatment with removable
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braces (Jones and Tier, 2012; Olate et al., 2013; Portelli et al., 2015). This situation correlates
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with the type of skeletal and dental discrepancies followed by inappropriate bone growth
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potential (Alyamani and Abuzinada, 2012; Hassani et al., 2013; Olate et al., 2013; Wolford et al.,

2014b).
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In time, when the patient is mature, but condyle growth activity is still present, numerous types
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of surgical procedures could be performed to halt its continued growth and improve skeletal and
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facial balance (Angiero et al., 2009; Hassani et al., 2013; Rodrigues and Castro, 2015).

Scintigraphy is a very useful diagnostic tool in any condylar head hyperplasia pathology, a fact

which seems to be confirmed by various other authors as well (Nitzan et al., 2008; Raijmakers et

al., 2012; Rodrigues and Castro, 2015; Wen et al., 2014). At the moment, it is clinically relevant

that scintigraphy alone is not sufficient enough to determine the form of hyperplasia and its
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activity (Chia et al., 2008; Gray et al., 1990; Hodder et al., 2000; Lo et al., 2012). Also, the

present study indicates that scintigraphy with computed tomography and additional radiography

should be closely evaluated (p=0.031).

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On the other hand, the time elapsed from the onset of the first symptoms of asymmetry and

properly implemented diagnostics are very important (Chia et al., 2008; Olate et al., 2013;

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Portelli et al., 2015). So far, it seems that a routine panoramic radiograph with lateral

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cephalograms are the first diagnostic procedures (p=0.039) (Olate et al., 2013; Portelli et al.,

2015; Wolford et al., 2002). Performing the classic radiological tests might uncover variances in

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condylar head shape and size, along with variations in the height/length of the ramus and body of
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the mandible (Hassani et al., 2013; Wolford et al., 2014; Gray et al., 1990). The appearance of an

atypical condyle in radiography is not always a sign of HH, since typical mandibles might be
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also responsible for HH, and their shape and size might vary (Obwegeser and Makek, 1986;

Wolford et al., 2014; Wolford et al., 2014b). This finding indicates that differences in condyle
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head shape and size might result in troublesome diagnostic situations (Mutoh et al., 1991;
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Wolford et al., 2014b). Despite the authors’ follow-up estimated to be up to more than one year

in some cases, the long-term values of scintigraphy are still inconclusive and require further
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future studies (p>0.05). Despite this fact (and the lack of pathological growth in the condyle), in
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all cases monitored for less than two years, all surgical assumptions such as stable occlusion, no
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relapse, no occurrence of re-asymmetry, proper mandibular movements, and preservation of

facial midline were achieved (n=15; p=0.445).

The time of orthodontic treatment with braces or removable appliances varies greatly (Chia et al.,

2008; Angiero et al., 2009; Hassani et al., 2013; Lippold et al., 2007; Olate et al., 2013; Wolford

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et al., 2002). In this study, it was only scheduled when condyle head condylectomy was

performed and the patient was scheduled for orthognathic surgery. This took place between the

ages of 19–24 years, which was considered an individual factor related to the amount of

extensive growth of the condyle which had been excised (p=0.038<0.05). Routine CT/LDCT

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performed one and two years after the condylectomy revealed no increased bone volume or

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visible changes in the shape and size of any excised hypertrophic condyle (p<0.05). In the

present study, orthodontic treatment lasted no more than one year post-surgery in most cases;

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however, the Study Authors must point out that any minor or major tooth discrepancies (if still

present) did not influence any surgical results.

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In this study, the amount and degree of surgical approaches performed were mostly related with

problems with chewing, bite, and occlusion, while other patients had an inappropriate jaw
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movement rate or lacked facial aesthetics. The treatment protocols presented by various authors

are largely the same, despite a small amount of differences (Obwegeser and Makek, 1986; Nitzan
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et al., 2008; Wolford et al., 2014; Lo et al., 2012; Wolford et al., 2014b). In most cases, a two-
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stage surgical approach is recommended, though a few authors decided to undergo an “all-in-one

approach” (Obwegeser and Makek, 1986; Chia et al., 2008; Raijmakers et al., 2012; Olate et al.,
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2013). In the Study Authors’ point of view it is quite reasonable, but some intermediate stages,
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such as trans-palatal distraction or necessary orthodontic treatment, might influence treatment

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planning. The Study Authors cannot report on any outcomes from the all-in-one approach

because of the limited number of patients treated in this approach. It seems that the patient’s age,

the degree of asymmetry with various forms of dento-alveolar discrepancies, and increased

preserved condyle growth could influence the treatment alternatives (Rodrigues and Castro,

2015; Olate et al., 2013; Portelli et al., 2015). In other cases, some camouflage surgery (n=5;
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17.9% HH) with bone modeling or mandibular marginectomy might be performed

simultaneously with condylectomy as one procedure. This situation is called for in cases of small

and perhaps moderate asymmetry of both the mandible and/or the maxillary bone, and when the

patient does not want or need any additional orthodontic treatment (Lo et al., 2012; Obwegeser

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and Makek, 1986; Olate et al., 2013). The Study Authors conclude that the patient’s age and

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degree of bone asymmetry should determine whether early condylectomy or condylectomy after

monitoring the value of growth and level of progressing asymmetry is used (Angiero et al., 2009;

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Hodder et al., 2000; Lippold et al., 2007). In most cases, each patient requires an individual

treatment plan, consisting of the most well-known and most often used methods (Obwegeser and

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Makek, 1986; Wolford et al., 2014; Gray et al., 1990; Olate et al., 2013; Wolford et al., 2014b).
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This study indicates that both early combined orthodontic and surgical approaches statistically

influence early outcomes, such as the restoration of symmetry and decreased further dento-
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alveolar discrepancies (p<0.05) (Table 5).

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The orthodontic approach is limited (Angiero et al., 2009; Jones and Tier, 2012; Pripatnanont et
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al., 2005; Rodrigues and Castro, 2015; Wolford et al., 2014; Wolford et al., 2002). In the study

material, orthodontic treatment was initiated statistically earlier than surgery, which influenced
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the later stages of treatment (p=0.038; Table 2). Furthermore, the degree of tilted occlusal cant
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(p=0.671) varies in all study participants, which was mostly related with the degree and intensity
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of bone growth. So far, there have been no adequate studies which measure the time from the

first diagnosis, the orthodontic treatment performed, and the final surgical procedure (Obwegeser

and Makek, 1986; Angiero et al., 2009; Alyamani and Abuzinada, 2012; Nitzan et al., 2008;

Gray et al., 1990; Hodder et al., 2000). The answer to this question remains unsolved because of

the majority of those presented conditions (Portelli et al., 2015; Pripatnanont et al., 2005).
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The Study Authors’ own observations on the above-mentioned mandibular malformations

indicate that both possible treatment plans, which consist of surgical camouflage with

condylectomy, condylectomy as the sole procedure, and condylectomy with orthognathic

surgery, lead to a great overall success rate (p<0.05; Table 4). So far, in all presented Authors’

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study cases treated with simultaneous condylectomy with mandibular camouflage marginectomy

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as a sole procedure, a good improvement of facial balance, symmetry, and mandibular

movements have been achieved (Table 4). Although the patients have reported good overall

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aesthetics and satisfaction from surgery, no accurate measuring tools were used to estimate the

patients’ well-being and satisfaction from surgeries. Perhaps further studies on quality of life in

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patients suffering from mandibular malformations should be scheduled.
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Wise diagnostics are recommended (Obwegeser and Makek, 1986; Rodrigues and Castro, 2015;
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Nitzan et al., 2008). Some authors indicate that two scintigraphic studies of bone growth and

activity performed within a 6-month timeframe still remains the mandatory approach (Jones and
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Tier, 2012; Olate et al., 2013). Some discrepancies in the estimation of bone growth activity
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measured during SPECT and classic bone scintigraphy may appear (Lo et al., 2012; Wolford et

al., 2002). In this study, values of bone scintigraphy higher than 1.5 were more commonly found,
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which corresponds with increased condylar head growth activity (p<0.05) (Table 3) comparing
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to the healthy condyle on the other side. Namely, it was higher than 15% and as Hodder et al.
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suggested, a 10% or more uptake difference was regarded as an indication of condylar head

hyperplasia (Hodder et al., 2000; Lippold et al., 2007). The authors, on the other hand, suggest

that in cases of a very fast-growing mandible with progressive asymmetry, only one scintigraphy

with CT-3D examination is enough to perform a condylectomy and then to schedule orthodontic

treatment as preparation for orthognathic surgery. Then again, a fast, progressive asymmetry
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should be a case exception for total condylectomy (Obwegeser and Makek, 1986; Wolford et al.,

2002; Wolford et al., 2014b). This point of view is also supported by a study by Pripatnanont et

al. on bone scans, SPECT, and scintigraphy (Chia et al., 2008; Fisch et al., 2011; Pripatnanont et

al., 2005). The authors indicate that at least two standard measurements within a 6-month

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timeframe should be performed in any other cases of slow progressive asymmetry (Pripatnanont

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et al., 2005). Some authors indicate that condylectomy in adult patients is better than in young

growing adults, because of more stable outcomes and a lower risk of relapse; however,

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progressive or rapid facial asymmetry with visible increased growth should be set as exceptions

to this rule (Lippold et al., 2007; Nitzan et al., 2008; Wolford et al., 2002; Wolford et al., 2014;

Wolford et al., 2014b).

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5. Conclusion
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The epidemiology and etiology are still not fully understood, though the onset of puberty is
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considered to be the starting time for HH and HE. The degree and value of growth factor might

vary in each case, so its influence on the therapeutic protocol is therefore crucial. Slow growth
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might be an indication for a more conservative surgery, while a rapid or progressive growth in
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most cases is the most demanding one. Almost all of the possible treatments are not only related
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with the degree of skeletal deformity, but also with the patient’s willingness to undergo any

necessary treatment, which in most cases includes more than one stage. This study is probably

one of only a few attempts to investigate the presented malformations and their clinical

relevance, and is probably the first study to present a Polish population treated and diagnosed

with HH/HE. The Study Authors’ point of view underlines three major take-away messages.
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Firstly, in any case of progressive asymmetry with visible enlargement in three diameters of the

condyle head on the affected side, along with the mandibular body and ramus on that side when

growth potential is still active, the clinician should consider an early low or total condylectomy

as the first surgical step. Secondly, when a tilted occlusion and a lack of facial balance is present,

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an orthodontic surgery should be scheduled right after a condylectomy to prepare the patient for

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any degree of orthognathic surgery. Finally, every researcher should remember that the amount

of excised condyle is not only meant to remove the growth’s etiological factor, but is also

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responsible for the mandible’s auto-rotation toward the excised side, and could therefore

influence the later mandibular position, which in turn greatly influences any future surgeries or

orthodontic treatment.
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Financial support – none

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Conflict of interest – none

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IRB, GCP – granted

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ALL AUTHORS VIEWED AND AGREED FOR PUBLICATION OF THIS SUBMISSION.

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condylar hyperplasia: clinical, histopathological, and treatment considerations. Cranio 27(1): 24–

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Chia MSY, Naini FB, Gill DS: The aetiology, diagnosis and management of mandibular

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Gray RJ, Sloan P, Quayle AA, Carter DH: Histopathological and scintigraphic features of
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condylar hyperplasia: diagnosis, clinical aspects and operative treatment. A case report. Eur J
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Raijmakers PG, Karssemakers LH, Tuizing db. Female Predominance and effect of gender on

unilateral condylar hyperplasia: A review and meta-analysis. J Oral Maxillofac Surg 70: 72-6,

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Table legends.

Table 1. Patient’s basics characteristics – age, gender, syndrome(mandibular malformation),

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occurrence of asymmetry.

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Table 2. Orthodontic and surgical treatment vs gathered clinical records.

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Table 3. Computed tomography and scintigraphy data.

Table 4. Type of surgery depending on scintigraphy finding and the value of asymmetry.

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Table 5. Statistical correlation according to Spearman analysis (rho) between patient’s age when

first asymmetry symptoms occurred, and time of orthodontic and surgical approach.
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Table 1. Patient’s basics data characteristics – age, gender, mandibular malformation, occurrence of
asymmetry.
Mandibular malformations.
Test
HH HE HH - HE
Variable result.
N = 28 N = 16 N=1
p
n % n % n %
Gender
Female 23 82,1% 14 87,5% 0 0,0% 0,085
Male 5 17,9% 2 12,5% 1 100,0%
Age.

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<15 years of age 3 10,7% 0 0,0% 0 0,0%
16 do 18 r. ż. 4 14,3% 2 12,5% 0 0,0%
19 do 21 r. ż. 3 10,7% 2 12,5% 0 0,0% 0,915
22 do 24 r. ż. 7 25,0% 6 37,5% 1 100,0%

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25 do 28 r. ż. 5 17,9% 3 18,8% 0 0,0%
29> and more 6 21,4% 3 18,8% 0 0,0%
Occurence side.

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R 14 50,0% 1 6,3% 0 0,0%
<0,001
L 13 46,4% 1 6,3% 0 0,0%
RL 1 3,6% 14 87,5% 1 100,0%
First asymmetry symptoms visible.

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From 10 - 12 years of age 2 7,1% 0 0,0% 0 0,0%
13 - 15 11 39,3% 8 50,0% 0 0,0%
0,843
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16 - 18 9 32,1% 6 37,5% 1 100,0%
19 - 21 5 17,9% 2 12,5% 0 0,0%
22 - 24 1 3,6% 0 0,0% 0 0,0%
*abbreviations: HH-hemimandibular hyperplasia; HE-hemimandibular elongation; HH-HE-mixed form; N/n-
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number/value; R-right; L-left, RL-right and left; p-statistical value.

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Table 2. Orthodontic and surgical treatment vs gathered clinical records.
Occurence side.
Test
Both sides Left Right
Variable. result.
N = 16 N = 14 N = 15
p
n % n % n %
Implemented orthodontic treatment. 14 87,5% 4 28,6% 7 46,7% 0,004
Time of implemented orthodontic
treatment.
None 2 12,5% 7 50,0% 8 53,3%
10-12 years of age 1 6,3% 1 7,1% 1 6,7%

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13-15 years of age 2 12,5% 3 21,4% 3 20,0% 0,038
16-18 years of age 7 43,8% 0 0,0% 1 6,7%
19-21 years of age 3 18,8% 0 0,0% 0 0,0%
22-24 years of age 1 6,3% 1 7,1% 1 6,7%

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25 years of age and more> 0 0,0% 2 14,3% 1 6,7%
Time of implemented surgical
treatment.

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None 5 31,3% 5 35,7% 11 73,3%
10-12 years of age 1 6,3% 0 0,0% 0 0,0%
13-15 years of age 1 6,3% 0 0,0% 0 0,0% 0,197
16-18 years of age 0 0,0% 1 7,1% 0 0,0%

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19-21 years of age 3 18,8% 1 7,1% 2 13,3%
22-24 years of age 5 31,3% 4 28,6% 0 0,0%
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25 years of age and more> 1 6,3% 3 21,4% 2 13,3%
Tilted occlusal cant. 9 56,3% 10 71,4% 10 66,7% 0,671
Deviated mandible towards.
No deviation 6 37,5% 5 35,7% 3 20,0%
0,035
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Right side 6 37,5% 8 57,1% 3 20,0%

Left side 4 25,0% 1 7,1% 9 60,0%
Compensatory maxillary rotation. 9 56,3% 10 71,4% 10 66,7% 0,008
Maxillary rotation.
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None - symmetrical 6 37,5% 4 28,6% 5 33,3%

0,144
Right side 5 31,3% 6 42,9% 10 66,7%
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Left side 5 31,3% 4 28,6% 0 0,0%

Pre-operative clinical data.
RTG-pantomographic 14 87,5% 13 92,9% 15 100,0% 0,377
RTG-cephalometric 14 87,5% 13 92,9% 15 100,0% 0,377
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CBCT 8 50,0% 7 50,0% 6 40,0% 0,818

LDCT 11 68,8% 8 57,1% 10 66,7% 0,784
MRI 1 6,3% 0 0,0% 0 0,0% 0,396
Bone scintigraphy 8 50,0% 12 85,7% 13 86,7% 0,031
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RTG-TMJ 5 31,3% 3 21,4% 1 6,7% 0,229

No documentation 1 6,3% 1 7,1% 0 0,0% 0,588
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Post-operative clinical data.

RTG-pantomographic 10 68,8% 9 64,3% 4 26,7% 0,039
RTG-cephalometric 10 68,8% 9 64,3% 4 26,7% 0,039
CBCT 5 37,5% 6 35,7% 3 20,0% 0,520
LDCT 9 56,3% 6 42,9% 2 13,3% 0,043
MRI 0 0,0% 0 0,0% 0 0,0% 1,000
Bone scintigraphy 4 25,0% 8 57,1% 4 26,7% 0,126
RTG-TMJ 6 37,5% 4 28,6% 1 6,7% 0,124
No documentation 6 37,5% 5 35,7% 11 73,3% 0,067
*abbreviations: HH-hemimandibular hyperplasia; HE-hemimandibular elongation; HH-HE-mixed form; N/n-
number/value; p-statistical value; RTG-radiograph; CBCT-cone beam computed tomography; LDCT-low dose
computed tomography; MRI-magnetic resonance imaging; RTG-TMJ-radiograph of the temporomandibular
joint.
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Table 3. Computed tomography and scintigraphy data.
Mandibular malformations.
Test
Variable. HH HE HH - HE
result.
N = 28 N = 16 N=1
p
n % n % n %
Tilted occlusal cant. 19 67,9% 9 56,3% 1 100,0% 0,559
Deviated mandible towards.
No deviation 8 28,6% 5 31,3% 1 100,0%
0,681
Right side 11 39,3% 6 37,5% 0 0,0%
Left side 9 32,1% 5 31,3% 0 0,0%

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Compensatory maxillary rotation. 20 71,4% 8 50,0% 1 100,0% 0,272
Maxillary rotation.
None - symmetrical 8 28,6% 7 43,8% 0 0,0%
0,232
Right side 16 57,1% 4 25,0% 1 100,0%

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Left side 4 14,3% 5 31,3% 0 0,0%
Orthodontic camouflage. 0 0,0% 2 12,5% 0 0,0% 0,150
Surgical camouflage. 5 17,9% 0 0,0% 0 0,0% 0,181

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Value of mandibular condyle overgrowth
potential after scintigraphic evaluation.
0 1 3,6% 3 18,8% 0 0,0%
Till 0,5 4 14,3% 10 62,5% 0 0,0%

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0,6 - 1,0 3 10,7% 2 12,5% 0 0,0% 0,006
1,1 - 1,5 4 14,3% 1 6,3% 0 0,0%
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1,6 - 2,0 11 39,3% 0 0,0% 0 0,0%
2,1 - 2,5 4 14,3% 0 0,0% 1 100,0%
2,6 and more> 1 3,6% 0 0,0% 0 0,0%
*abbreviations: HH-hemimandibular hyperplasia; HE-hemimandibular elongation; HH-HE-mixed form; N/n-
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number/value; p-statistical value.

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Table 4. Type of surgery depending on scintigraphy finding and the value of asymmetry.
Mandibular malformations.
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Test
HH HE HH - HE
Variable. result.
N = 28 N = 16 N=1
p
n % n % n %
Frequency of routine check-ups.
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Every month. 0 0,0% 1 6,3% 0 0,0%

Twice a year. 12 42,9% 10 62,5% 1 100,0% 0,445
Once a year. 15 53,6% 4 25,0% 0 0,0%
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After 1,5 years. 0 0,0% 1 6,3% 0 0,0%

Condylectomy procedure –YES. 12 42,9% 0 0,0% 1 100,0% 0,003
Type of condylectomy:
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0,003
High. 12 42,9% 0 0,0% 1 100,0%
Procedure: TMJ artroplasty. 12 42,9% 0 0,0% 1 100,0% 0,003
Genioplasty. 0 0,0% 4 25,0% 1 100,0% <0,001
Mandibular bone „shift”. 4 14,3% 0 0,0% 0 0,0% 0,264
Lefort I osteotomy. 0 0,0% 10 62,5% 1 100,0% <0,001
BSSO mandible osteotomy. 0 0,0% 10 62,5% 1 100,0% <0,001
BSSO osteotomy modification
-
according to Ferguson technique. 0 0,0% 0 0,0% 1 100,0%
Ortohognathic procedure. 0 0,0% 10 62,5% 1 100,0% <0,001
*abbreviations: HH-hemimandibular hyperplasia; HE-hemimandibular elongation; HH-HE-mixed form; N/n-
number/value; p-statistical value.; BSSO-bilateral sagittal split osteotomy of the mandible.

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Table 5. Statistical correlation according to Spearman analysis (rho) between patient’s age when first
asymmetry symptoms occurred, and time of orthodontic and surgical approach.
Age of patient, at: {1} {2} {3}
rho = 0,054 rho = 0,389
{1} Mandibular malformation occurence. ×
p > 0,05 p < 0,01
rho = 0,054 rho = 0,359
{2} Time of orthodontic treatment. ×
p > 0,05 p < 0,05
rho = 0,389 rho = 0,359
{3} Time of surgical treatment. ×
p < 0,01 p < 0,05

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