CASE REPORT

Combined orthodontic-orthognathic
management of a patient with
generalized short root anomaly and
anterior open bite
Charlotte Marechal, Laurent A. M. Thierens, and Guy A. M. De Pauw
Ghent, Belgium

Short root anomaly is a rare dental disorder affecting tooth root development. It is characterized by reduced root-
to-crown ratios (1:1 or less) and rounded apices. The short roots introduce a potential complication during ortho-
dontic treatment. This case report describes managing a girl with generalized short root anomaly, an open bite,
impacted maxillary canines, and a bilateral crossbite. In the first phase of treatment, the maxillary canines were
extracted, and the transverse discrepancy was corrected with a bone-borne transpalatal distractor. In the second
phase of treatment, a mandibular lateral incisor was removed, fixed appliances were placed in the mandibular
arch, and bimaxillary orthognathic surgery was performed. A satisfactory result was obtained without further root
shortening, adequate smile esthetics, and 2.5-year posttreatment stability. (Am J Orthod Dentofacial Orthop
2023;164:131-42)

S
hort root anomaly (SRA) is an uncommon condi- resorption is frequently seen secondary to orthodontic
tion characterized by short and blunt dental roots. tooth movement and affects the maxillary incisors
It mainly affects the central incisors and second most frequently. In most of these patients, the reduction
premolars bilaterally but may also appear generalized. in root length is not clinically significant. SRA must also
Short roots can be observed in teeth with a reduced ratio be distinguished from dentin dysplasia type 1, character-
of root-to-crown, measuring 1:1 or less.1 In most pa- ized by pulp obliteration and bone radiolucencies sur-
tients, the teeth are asymptomatic, and diagnosis is rounding the roots.5
made on the basis of an accidental radiographic finding. Little is known about the prevalence of SRA. For
An association between tooth agenesis and ectopic ca- maxillary incisors, a prevalence of 2.4%-2.7% in Cauca-
nines has been reported.2 SRA is often misdiagnosed sians and 10% in Mongolian populations has been
as external apical root resorption, defined as permanent reported. This anomaly is also more common in
loss of dental root structure mediated by odontoclasts females.1,6,7 A U.S. cohort study observed a higher prev-
and cementoclasts in response to a mechanical, inflam- alence among Latinos. In this study, 27 SRA subjects
matory, autoimmune, or infectious stimulus.3 In teeth were analyzed clinically and radiographically. The disor-
with this kind of resorption, the apical portion of the der typically appears in several members of one family.
tooth is typically rounded and irregular.4 Apical root Only 2 of the studied patients were diagnosed with
generalized SRA. In these patients, teeth were more
severely affected, having higher crown-root ratios than
From the Oral Health Sciences, Department of Orthodontics, Ghent University,
Ghent, Belgium. localized forms. Moreover, teeth were always affected
All authors have completed and submitted the ICMJE Form for Disclosure of Po- bilaterally, with molars and mandibular canines least
tential Conflicts of Interest, and none were reported.
commonly affected (10%) and maxillary central inci-
Informed consent was obtained from the patient and permission to publish this
report was granted by the Ethics Committee of Ghent University Hospital (refer- sors most commonly affected (63%).8 SRA is usually
ence EC/028-2020). asymptomatic and frequently misdiagnosed so the prev-
Address correspondence to: Laurent A.M. Thierens, Oral Health Sciences, Depart-
alence might be underestimated.
ment of Orthodontics, Ghent University, Corneel Heymanslaan 10, 9000 Ghent,
Belgium; e-mail, [email protected]. An unfavorable root-crown ratio is a contraindica-
Submitted, October 2020; revised and accepted, June 2021. tion for orthodontic treatment. However, no studies
0889-5406/$36.00
have reported an increased risk for root resorption in
Ó 2023 by the American Association of Orthodontists. All rights reserved.
https://doi.org/10.1016/j.ajodo.2023.05.004 SRA patients. A retrospective case-control study using

131
132 Marechal, Thierens, and De Pauw

Fig 1. Panoramic radiograph with impacted maxillary canines.

cone-beam computed tomography did not find a signif- transverse discrepancy between the maxilla and
icant difference in the change in root length after ortho- mandible with a bilateral crossbite. Functional examina-
dontic treatment in SRA patients compared with a tion revealed a tongue thrust with an anterior resting
control group. Nevertheless, more intensive monitoring tongue posture (Fig 2).
is indicated (eg, periapical radiographs of affected teeth Radiographic examination revealed permanent inci-
every 6 months), and a long-lasting treatment with high sors and first and second premolars with a reduced
forces should be avoided.9 Treating patients with SRA is crown-to-root ratio and short, blunted roots. The first
challenging, especially in patients with open bite. In this and second permanent molars had normal morphology.
case report, we would like to present the orthodontic- These findings correspond with the characteristics of
orthognathic treatment of a patient with a generalized SRA. However, the patient reported no familial occur-
SRA and an anterior open bite. rence. No periodontal or periapical problems were
observed (Fig 3, A).
Cephalometric analysis revealed a Class I relationship
DIAGNOSIS AND ETIOLOGY
with a retrusive maxilla (SNA, 77.7 ) and mandible (SNB,
A healthy 10-year-old girl presented for the first time 75.1 ) and a horizontal growth pattern (SN-GoGn,
in 2007 at the Department of Orthodontics of Ghent 26.4 ). The vertical maxillary height was deficient (Pr
University Hospital. Her chief complaint was the lack t ANS-PNS, 7.2 mm; ANS-N, 48.6 mm), and the lower
of occlusal contacts and deficient maxillary incisor expo- anterior facial height was short (ANS-Me/N-Me, 52.5%).
sure on smiling. The patient’s medical history was The maxillary and mandibular anterior teeth were pro-
noncontributory. Radiographic examination revealed 2 clined (U1-SN, 118.7 ; IMPA, 106.7 ) with an interin-
palatally impacted canines in the maxillary arch (Fig 1). cisal angle of 108.2 (Figs 3, B and C; Table).
At that moment, the decision was made to remove these
teeth surgically. TREATMENT OBJECTIVES
In 2011 new records were taken. The patient clini-
The main goals of the treatment were to (1) correct
cally presented with a concave profile with a pronounced
the open bite, (2) improve the amount of maxillary
chin projection at that time. The frontal view showed a
incisor exposure, (3) correct the transversal discrepancy,
slight deviation of the nose tip to the right. The smile es-
(4) relieve the crowding in the mandibular arch, (5)
thetics were poor because the incisors were not exposed
establish a stable Class I canine relationship and Class
on smiling. A bilateral full Class II molar occlusion with
II molar relationship, and (6) improve dental and facial
an overjet of 3 mm and a 8 mm anterior open bite
esthetics without further root shortening.
was present. The patient only had occlusal contacts in
the molar region. An arch length-tooth size discrepancy
TREATMENT ALTERNATIVES
was observed in the maxilla and mandible. This discrep-
ancy was characterized by crowding in the mandibular It is not possible to define an equivalent treatment
anterior region and by the position of the maxillary first alternative because of the specific combination of clin-
premolars next to the lateral incisors. There was a ical, radiographic, and functional characteristics in this

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Marechal, Thierens, and De Pauw 133

Fig 2. Pretreatment facial and intraoral photographs.

patient. Canine impaction has multiple treatment mo- create adequate space for alignment, but this approach
dalities. The canines can be orthodontically aligned after would result in prolonged treatment with potentially
surgical exposure if orthodontic treatment is recommen- harmful side effects on the neighboring teeth.
ded. Transalveolar transplantation of the impacted Adequate correction of the open bite also implicated
canine is possible if the forced eruption is not feasible. correction of the transverse discrepancy. Expansion with
Extraction of an impacted canine is not an ideal treat- a bone-borne transpalatal distractor was preferred over a
ment plan because the canine has a long root, good tooth-borne expander to minimize unnecessary force
bony support, and an important function in canine application on the teeth.11 Because of the skeletal origin
guidance. In addition, it is an excellent abutment for of the open bite (vertical maxillary deficiency), correction
fixed and removable prostheses.10 In this patient, no of this would require bimaxillary orthognathic surgery.
attempt was made to force the eruption of the impacted This approach would also improve maxillary incisor
canines because of lack of space and potential high exposure to smiling, which was a major concern of the
forces exerted on the adjacent teeth. Despite the absence patient.
of the canines, the maxillary arch was well aligned Functional habits are important factors in the etiol-
without spacing. By extracting the canines, it was ogy of an open bite. The patient showed a tongue thrust;
possible to avoid orthodontic treatment in the maxillary therefore, a removable retainer with a tongue guard was
arch and to decrease the risk of further root shortening. provided after the transpalatal distraction. The patient
A treatment alternative would have been to extract the was asked to perform myofunctional exercises during
maxillary lateral incisors (which presented with very this retention phase to strengthen her tongue function
short roots), followed by a forced eruption of the and coordinate swallowing movements. This would
impacted maxillary canines. However, this would have improve long-term stability. Several sessions were
required orthodontic treatment in the maxillary arch. planned with the speech therapist. In each session, the
Extraction of 1 mandibular incisor allowed alignment therapist focused on a specific goal and explained a
quickly and without needing fixed appliances in the few exercises which needed to be repeated at home daily.
maxillary arch or intermaxillary elastic traction. Extrac- The goals were (1) to raise awareness of the tongue tip
tion of mandibular first or second premolars would also and the palatal rugae and to stimulate the contact

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134 Marechal, Thierens, and De Pauw

Fig 3. A, Pretreatment panoramic radiograph; B, Pretreatment lateral cephalogram; C, Pretreatment

cephalometric tracing.

between these structures, (2) to obtain a correct tongue TREATMENT PROGRESS

and lip posture at rest, (3) to reinforce the anterior and After the surgical removal of the 2 maxillary canines
posterior part of the tongue, (4) to automate the physi- in September 2007, the patient was followed up regu-
ological swallowing pattern, (5) lip force training, and (6) larly. The first phase of treatment started in April
maintenance of new habits. 2012. A bone-borne transpalatal distractor was placed
In conclusion, the multidisciplinary plan can be sum- supported with corticotomies to release the areas of
marized as follows: bony resistance.
1. Surgically assisted rapid maxillary expansion with a The patient started the distraction 1 week after the
bone-borne transpalatal distractor followed by a placement of the appliance. The distractor was activated
removable retainer with a tongue guard and myo- twice daily, resulting in a transverse expansion of 0.5
functional exercises. mm/d. Regular follow-up during the active expansion
2. Extraction of 1 mandibular left lateral incisor and was performed by the orthodontist. The correct trans-
fixed appliances in the mandibular arch to resolve verse width was obtained after 21 days, and the distrac-
the crowding. tor was immobilized subsequently (Fig 4). The maxilla
3. Bimaxillary orthognathic surgery: bilateral sagittal was expanded by 10.5 mm. The distractor was kept in
split osteotomy and LeFort I with clockwise rotation place for 8 months until it was removed in January
of the occlusal plane. 2013 by the maxillofacial surgeon (Fig 5). Subsequently,

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Marechal, Thierens, and De Pauw 135

Table. Cephalometric summary

Measurement Norm Pretreatment Posttreatment 2.5 y Posttreatment
SNA ( ) 82.0 77.7 78.7 78.9
SNB ( ) 80.0 75.1 75.0 74.9
ANB ( ) 2.0 2.6 3.7 4.0
Wits (mm) 1.0 5.3 3.5 3.9
SN-GoGn ( ) 32.0 26.4 31.3 30.2
FH-GoGn ( ) 24.0 20.0 24.9 24.2
SN-ANS-PNS ( ) 8.0 12.0 22.2 22.0
Pr t ANS-PNS (mm) 17.8 7.2 11.0 11.0
ANS-N (mm) 55.7 48.6 53.1 52.9
LAFH (ANS-Me/N-Me) (%) 55.0 52.5 49.4 49.5
U1-ANS-PNS ( ) 110.0 130.7 123.9 123.8
U1-SN ( ) 104.0 118.7 101.7 101.8
U1-NA ( ) 22.0 41.0 23.0 22.9
IMPA ( ) 90.0 106.7 100.3 100.9
L1-NB ( ) 25.0 28.2 26.6 26.0
U1-L1 ( ) 131.0 108.2 126.7 127.1
Upper lip (mm) 4.0 7.1 5.6 6.3
Lower lip (mm) 2.0 6.9 5.1 5.9
IMPA, incisor mandibular plane angle; LAFH, lower anterior facial height; Pr t ANS-PNS, prosthion perpendicular to palatal plane.

Fig 4. Intraoral photographs at the time of distractor immobilization.

a removable retainer with a tongue guard was placed on surgery. Elastic traction on the maxillary molars implied
maintaining the maxillary transverse dimension. The no risk because these teeth showed a normal root-to-
patient also consulted a speech therapist and performed crown ratio.
myofunctional exercises at home. At the time of In July 2016, the orthognathic surgery consisted of a
distractor removal, the anterior open bite had already LeFort I osteotomy and a bilateral sagittal split osteot-
reduced by 3 mm. In the subsequent months, the omy. The impacted maxillary third molar on the left
overbite further decreased by 1 mm and remained stable side was removed during the osteotomy. Four screws
at 4 mm. were placed, 2 paranasally in the maxilla and 2 in the
In January 2014, the mandibular left lateral incisor mandible between the lateral incisors and canines.
and the mandibular third molars were extracted, and Immediately after surgery, the patient was instructed
fixed appliances (Ormco, Orange, Calif) were placed in to wear intermaxillary elastics (in the posterior region us-
the mandible. After leveling with a 0.014-inch nickel- ing the molar tubes and in the anterior region using the
titanium wire, space closure was started with intraarch fixation screws) and to wear a wafer 24 h/d. The wafer
mechanics on a 0.016-inch Australian wire (Figs 6 and was gradually ground in the posterior region to allow
7). In July 2015, the patient was ready for surgery. How- settling. The fixed appliances in the mandible were
ever, for personal reasons, she wanted orthognathic sur- removed when proper occlusal contact was obtained. A
gery in July 2016. Between July 2015 and July 2016, the fixed retainer was provided in the mandibular arch,
patient was seen every 3 months and no active forces and a removable retainer with a tongue crib was pro-
were applied. Tubes were placed on the first and second vided to maintain the transverse dimensions of the
maxillary molars during the final appointment before maxillary arch.

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136 Marechal, Thierens, and De Pauw

Fig 5. Facial and intraoral photographs 3 months after transpalatal distraction.

TREATMENT RESULTS rotation and anterior movement of the maxilla.12 The

A stable Class II molar relationship was obtained with maxillary height increased (Pr t ANS-PNS, 11.0 mm;
evenly distributed occlusal contacts. A positive overbite ANS-N, 53.1 mm). The angulation of the palatal plane
of 2 mm and an acceptable overjet of 3 mm were relative to the cranial base showed an increase of 10 .
achieved. The mandibular arch was well aligned and lev- Consequently, the angulation of the maxillary incisors
eled, with complete closure of the extraction site. The relative to the cranial base also improved. The incisor
alignment in the maxillary arch remained unchanged extraction reduced the inclination of the mandibular in-
because no fixed appliances were used there. The trans- cisors relative to the mandibular corpus. There was a
verse relationship on the molars was improved compared small increase in the length of the mandibular corpus.
with the initial situation; however, still without a perfect This was not noticeable as a change in the SNB angle
interdigitation. The posttreatment facial photographs because of the clockwise rotation of the mandible. The
showed adequate maxillary incisor exposure and a skeletal jaw relationship remained stable (Fig 10; Table).
more balanced, mild concave facial profile. The lip rela- Retention records taken 2.5 years after the end of or-
tionship and nose tip projection in the sagittal plane thodontic treatment presented an excellent stability of
showed improvement, but the slight asymmetry of the the achieved result with a solid bilateral Class II molar
nose in the frontal plane was still present (Fig 8). occlusion and a stable overbite and overjet (Fig 11).
A panoramic radiograph was taken a few months Comparison of the pretreatment panoramic radiograph
before debonding to evaluate root parallelism and heal- and 2.5 years posttreatment radiograph proved that
ing after surgery. Because the screws were still in place, the overall root length remained stable throughout
detecting orthodontic-induced apical root resorption treatment (Fig 12). Cephalometric analysis revealed no
was difficult. However, no further root shortening was skeletal or minor dental changes (Fig 13; Table).
suspected. The third molar in the first quadrant was still
DISCUSSION
present and required further follow-up (Fig 9). Accord-
ing to the American Board of Orthodontics guidelines, SRA is not a common finding and is often misdiag-
cephalometric superimposition demonstrated a slight nosed. There is a lack of scientific evidence about the dis-
anterior movement of point A because of the clockwise order. The occurrence among family members, the female

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Marechal, Thierens, and De Pauw 137

Fig 6. Facial and intraoral photographs before orthognathic surgery.

Fig 7. Panoramic radiograph and cephalometric radiograph before orthognathic surgery.

preference, and the variable prevalence with race suggest analysis study has investigated the effects of root
it is a genetic disorder rather than a developmental distur- morphology on stress distribution at the root apex. Teeth
bance.2,9 However, the etiology of SRA remains unknown. with short roots experience greater loading and stress
A recent study stresses the importance of Wnt/b-catenin than teeth with normal root configurations.14 In addition,
signaling in pathologic mechanisms. Suppression or over- orthodontic-induced root resorption can further aggra-
activation of this signaling is known to influence normal vate the root-crown ratio. The patient described in this
odontogenesis. Efforts are made to find potential treat- report already had consulted several dental specialists un-
ments for SRA by interfering in these molecular pathways willing to perform any treatment. This is not so remark-
of cell differentiation and tooth formation.13 able because only a few case reports have described
A reduced root-to-crown ratio can complicate the orthodontic treatment in patients with SRA. Marques
success of orthodontic treatment. A finite element et al15 treated a patient with short roots but minimized

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138 Marechal, Thierens, and De Pauw

Fig 8. Posttreatment facial and intraoral photographs.

Fig 9. Panoramic radiograph before debonding.

orthodontic tooth movement using an activator and a all first premolars but used a targeted mechanics
headgear. Lamani et al16 presented a nonextraction treat- approach. In the first phase, the canines were retracted
ment with proclination of incisors and broadening of the using mini-implants without the involvement of posterior
arches in a patient with generalized short roots. Extrac- teeth. In a later stage, appliances were bonded on the
tion treatment was avoided because of the higher risk other teeth for final alignment and space closure. Using
of root resorption. However, after treatment, the pano- this technique, the premolars and incisors—mostly
ramic radiograph still showed some root resorption (\2 affected by short root anomaly—only receive orthodontic
mm). Dutra et al17 chose a treatment with extraction of forces for a short period. Vishwanath et al4 presented the

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Marechal, Thierens, and De Pauw 139

Fig 10. A, Posttreatment cephalometric radiograph; B, Posttreatment cephalometric tracing; C, Super-

imposition of pretreatment (black) and posttreatment (red) cephalometric tracings.

Fig 11. Facial and intraoral photographs 2.5 years posttreatment.

orthodontic management of a patient with SRA associ- damage. Fortunately, the molar roots were unaffected.
ated with multiple impacted teeth, ectopically erupted These teeth could be used for intermaxillary elastic trac-
teeth, and peg-shaped lateral incisors. They aligned the tion in the posterior region to obtain proper occlusal
impacted teeth successfully by using light forces, taking contacts after surgery. In the anterior region, fixation
longer intervals between activations, and performing pe- screws successfully stabilized the jaws after surgery. In
riodic monitoring with radiographs. the mandible, extracting 1 incisor made it possible to
The roots of the maxillary incisors and premolars align the teeth in a few months without excessive force.
were severely affected in our patient. Therefore, no Radiographic examination showed no difference in root
brackets were placed on these teeth to avoid further length before and after treatment. This corresponds with

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140 Marechal, Thierens, and De Pauw

Fig 12. Panoramic radiograph 2.5 years posttreatment.

Fig 13. A, Cephalometric radiograph 2.5 years posttreatment; B, Cephalometric tracing 2.5 years
posttreatment; C, Superimposition of posttreatment (red) and 2.5 years posttreatment (green)
cephalometric tracings.

the findings of Cutrera et al9 that there is no increased myofunctional treatment in patients aged 6-10 years.
risk for root resorption in patients with SRA. After 6 months of active treatment and another 6
This patient was challenging because of the SRA, the months of follow-up, the results confirmed a significant
impacted canines, and the open bite. However, even improvement in tongue posture and anterior occlusal
treating an open bite can be challenging because it is relationship. Koletsi et al20 reviewed all published ran-
not always easy to identify an unambiguous cause. An domized controlled trials and controlled clinical trials
anterior open bite is often associated with an infantile comparing interventions to manage anterior open bite
swallowing pattern. In these patients, myofunctional and other muscle functions such as swallowing pattern
therapy can be indicated to harmonize the orofacial and tongue resting position. They concluded that the
functions. Our patient consulted a speech therapist quality of the existing evidence was questionable.
and did myofunctional exercises at home. She was also Further, there seems to be no evidence that using tongue
given a removable retainer with a tongue guard. A recent spurs is effective. In this patient, it was not possible to
pilot study showed a possible beneficial influence of my- correct the open bite completely with the removable
ofunctional treatment on tongue behavior, but further retainer and myofunctional exercises. However, surgical
research is recommended to confirm the success of this bite closure was the preferential treatment because of
type of therapy as an adjunct to orthodontic treat- deficient vertical maxillary height and dental show. A
ment.18 J
onsson19 evaluated the success of orofacial meta-analysis indicated the stability of 82% for surgical

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Marechal, Thierens, and De Pauw 141

Fig 14. Changes in smile esthetics (left, pretreatment; middle, posttreatment; right, 2.5 years
posttreatment).

open bite treatment, measured by a positive vertical and Drs Bart Knockaert, Pieter-Jan Van Bever, and Peter
overbite at $12 months after the treatment interven- Bouvry (orthognathic surgery).
tion.21 Another systematic review could not present clear
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(orthodontics), Dr Gerry Orye (transpalatal distraction), record-preparation/superimpositions/. Accessed August 16 2020.

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