CASE REPORT

Orthodontic space closure in a young

female patient with solitary median
maxillary central incisor syndrome
Ute E. M. Schneider and Lorenz Moser
Ferrara, Italy

A solitary median maxillary incisor can occur as a rare single dental anomaly or a symptom of the early-
intrauterine developmental brain disorder of holoprosencephaly. The few published case reports about ortho-
dontic treatment for this disorder have only described space opening for prosthodontic replacement of a central
incisor. In contrast, the present patient was treated with extraction of the solitary median maxillary central incisor
and orthodontic space closure with subsequent minimally invasive restorations in order to avoid looming esthetic
or periodontal sequelae associated with any type of fixed bridgework or implant-borne crowns in the sensitive
maxillary anterior area—which is especially indicated in young girls with a hyperdivergent growth pattern.
(Am J Orthod Dentofacial Orthop 2021;160:132-46)

T
he solitary median maxillary central incisor
(SMMCI) syndrome is a rare early-intrauterine
(from 35th to 38th day intrauterine) develop-
mental disorder of unknown etiology, with a prevalence
of 1 in 50,000 live births, first described by Scott as an
isolated defect in 1958.1 Associations with defects, for
example, deletions on chromosomes 7 and 18, and mu-
tations on the sonic hedgehog gene have been re-
ported.2-4
In its least expression, the SMMCI appears only as an
isolated trait of the complex holoprosencephaly spec-
trum. In the worst case, patients present incomplete
cleavage of the embryonal forebrain leading to mental
retardation, growth impairment, malformations of the
sella turcica, choanal atresia, and midnasal pyriform
aperture stenosis, and abnormal formation of the mid-
face soft tissues and the maxillary alveolar bone.5-7
The perfectly symmetrical central incisor that
erupts exactly in the maxillary midline is attributed
Private practice, Bolzano and University of Ferrara, Ferrara, Italy.
Ute E.M. Schneider and Lorenz Moser are joint first authors and contributed
equally to this work.
All authors have completed and submitted the ICMJE Form for Disclosure of
Potential Conflicts of Interest, and none were reported.
Address correspondence to: Ute E.M. Schneider, City Center, Via Alto Adige 40,
Bolzano 39100, Italy; e-mail, info@perfect-smile.it.
Submitted, February 2020; revised and accepted, May 2020.
0889-5406/$36.00
Ó 2021 by the American Association of Orthodontists. All rights reserved.
https://doi.org/10.1016/j.ajodo.2020.05.018
132
to the fusion of the mesial halves of 2 maxillary central
incisors.1,8
The 5 typical features that are phenotypical for
SMMCI children are an indistinct philtrum with an
arc-shaped upper lip, a single symmetrical maxillary
central incisor, the absence of both the upper labial
frenulum and the papilla between the maxillary central
incisors, fusion of the anterior part of the palatal su-
ture, and a bulging bony midpalatal ridge.9,10 In a
study by Kjaer and Balsev-Olesen,11 the condition has
been described for both the deciduous and the perma-
nent dentition.
Although a substantial number of articles illus-
trating the clinical findings in subjects affected by
SMMCI syndrome is available in the literature, only 3
case reports of comprehensive orthodontic treatment
have been published up to date. In all 3 patients, the
treatment goal was to maintain and to distalize the
SMMCI and to open space for a second maxillary cen-
tral incisor. Bolan et al12 reported tentative rapid maxil-
lary expansion in a 6-year-old boy, but the cone-beam
computed tomography evidenced that correction of the
posterior crossbite had merely occurred by dentoalveo-
lar tipping without any opening of the midpalatal su-
ture.12 Similarly, Lygidakis et al13 described a 2-phase
treatment of a male SMMCI patient during which, after
2 failing attempts of early maxillary expansion when
aged 4 and 7 years, an anterior space was finally created
by maxillary molar distalization with fixed appliances
for insertion of a double-winged Maryland bridge
Schneider and Moser
Fig 1. Pretreatment facial and intraoral photographs.
when aged 16 years. In 2014, Pseiner14 published a case
report of a female patient with an SMMCI with space
opening and subsequent autotransplantation of a
mandibular second premolar. After 4 years of active
treatment, the transplanted premolar was restored
with a ceramic crown.
To our knowledge, no SMMCI patient has ever been
treated orthodontically with the extraction of the fused
cyclops tooth followed by orthodontic space closure,
although this approach offers several advantages
compared with maintenance of the SMMCI and substi-
tution of a maxillary central incisor. Therefore, we
consider this approach worthwhile presenting in the
following case report.
DIAGNOSIS AND ETIOLOGY
A 9-year-old girl was referred to our office with the
request for a second opinion by a befriended orthodon-
tist. The preliminary diagnosis was Class II Division 1
malocclusion with a congenitally missing maxillary cen-
tral incisor.
American Journal of Orthodontics and Dentofacial Orthopedics
133
The parents and the patient were disturbed and
worried about the altered smile esthetics and reported
episodes of bullying at school because of the unusual
appearance of the solitary maxillary median cyclops
tooth.
The lateral view exhibited a balanced profile. Facial
frontal inspection revealed a slightly asymmetrical
slender face with a mild chin deviation to the patient's
right side. An atypical arch-shaped outline of the upper
cupid's bow with an indistinct philtrum, albeit good lip
competence, was evident. The patient presented a mild
gummy smile with an altered smile arc and smile line
display because of the overerupted single central incisor.
Intraorally, the patient presented an early mixed denti-
tion with only 1 single, totally symmetrical, large central
incisor with equal crown height and length (9 3 9 mm),
positioned exactly in the maxillary midline. Closer in-
spection revealed the absence of the upper labial frenum
but the presence of the incisive papilla and a thick me-
dian palatal raphe because of an underlying prominent
bony ridge.
July 2021  Vol 160  Issue 1
134 Schneider and Moser

Fig 2. Pretreatment dental casts.

Both maxillary lateral incisors, especially the left,

were reduced in width (7 mm and 5.5 mm), which
contributed further to the existing anterior spacing. On
model analysis, an anterior Bolton ratio between the
maxillary and mandibular incisors of 112% was calcu-
lated. Bilaterally, the deciduous canines and the first mo-
lars were in full Class II occlusion, and a 4 mm overjet
could be assessed between the SMMCI and the mandib-
ular incisors. The overbite was mildly increased (5 mm)
because of a steepened lower curve of Spee. The lower
midline was mildly deviated to the right side by 1 mm
(Figs 1 and 2).
The panoramic radiograph evidenced an SMMCI
exactly in the maxillary midline and an age-typical
development of all other permanent teeth. The cephalo-
metric analysis revealed a Class I hyperdivergent skeletal
pattern (A-N-Pg, 2.0 ; SN/Go-Gn, 36.7 ) and a normal
inclination of the maxillary and mandibular incisors
(U1/SN, 105 ; L1/Go-Gn, 89.4 ) (Fig 3).
The parents reported that they had already noted the
condition around 2 years of age when a solitary median
deciduous maxillary incisor had erupted. Subsequent pe-
diatric and neurologic consultations had not revealed
any other health issues and the girl performed normally
at school.
As no history of dental trauma with avulsion of a
central incisor was reported, and all 5 typical extraoral
and intraoral traits of the SMMCI phenotype were pre-
sent, the final diagnosis of SMMCI syndrome was Fig 3. Initial panoramic radiograph and lateral cephalo-
made. metric radiograph.

July 2021  Vol 160  Issue 1 American Journal of Orthodontics and Dentofacial Orthopedics
Schneider and Moser
TREATMENT OBJECTIVES AND ALTERNATIVES
The main treatment goal was to create pleasing and
symmetrical smile esthetics without the need for a future
invasive restorative treatment and to establish incisor
guidance while maintaining a solid posterior intercuspa-
tion with full Class II relationships. As the patient did not
exhibit major skeletal discrepancies and the posterior
teeth were already in full Class II occlusion before treat-
ment, extraction of the SMMCI with space closure
deemed the most appropriate approach, requiring the
least biomechanics and tooth movement. This space
closure treatment option would lead to a symmetrical
array of the anterior dentition in the very sensitive
esthetic zone and provide a healthy periodontal outcome
with natural-looking transition areas in the long term.
These treatment objectives were explained to the par-
ents, who emphasized their preference for the least inva-
sive postorthodontic restorative treatment.
The parents were thoroughly informed about all
possible alternative treatment strategies.
The first strategy was orthodontic space opening by
distalization with either headgear or palatal temporary
anchorage devices (TADs).
Maxillary molar distalization with headgear would
require excellent patient compliance during phase I, a
comprehensive retention protocol to maintain the mo-
lars in Class I relationship during the eruption of the per-
manent dentition, followed by a second phase of fixed
appliance treatment for anterior space opening. Maxil-
lary molar distalization with noncompliance palatal
TADs would need to be either delayed until after the
eruption of the permanent dentition or performed in 2
phases. Although the presence of the maxillary third
molar buds could not be assessed at the time of the first
consultation with the patient's parents, any distalization
approach would have necessitated early surgical removal
of subsequently developing third molars and additional
burden for the patient.
Moreover, this treatment approach would require en-
ameloplasty of the SMMCI to normalize its shape in
conjunction with subsequent prosthodontic substitution
of one central incisor. Because of the fusion etiology, the
panoramic x-ray evidenced a relatively large root of the
SMMCI, so that slenderizing of the crown might have
caused an unnatural crown-root transition area with
potentially looming periodontal sequelae (Fig 3). It was
explained to the parents that the least invasive restor-
ative option after orthodontic space opening would be
a bonded single-winged cantilever resin-bonded bridge
without any tooth preparation. Admittedly, this prostho-
dontic approach would have been the least invasive
restorative approach after space opening and would
achieve a good esthetic result.15,16
American Journal of Orthodontics and Dentofacial Orthopedics
135
However, without the bony support of an incisor root
in the area of the pontic, vestibular alveolar bone atro-
phy would very likely occur in the long term, making
connective tissue grafting necessary for restoring
acceptable dental esthetics.17
The idea of a central incisor implant-borne crown
was discarded because of the invasiveness, the poor
long-term predictability, and the problem of infrao-
clusion over time because of the patient's young
age.18,19
The second strategy was unilateral space closure sup-
ported by temporary palatal skeletal anchorage with the
future restoration of the mesialized lateral incisor,
canine, and first premolar, and the small opposite lateral
incisor.20,21
In this instance, orthodontic treatment would have
been delayed until after the eruption of the entire per-
manent dentition and carried out as a 1-phase treatment
with the advantage that the overall treatment time
would have been much shorter.
Both the required biomechanics for unilateral space
closure and several interdisciplinary treatment results
of former patients affected by maxillary incisor agenesis
were shown and explained to the patient and her par-
ents. Because of the necessity of palatal TADs and the
difficulty to achieve a truly symmetrical display of the
maxillary anterior dentition despite intense restorative
therapy, this treatment alternative was rejected by the
patient and her parents.
As the patient was bullied at school because of
the uncommon esthetic aspect of her large single
central incisor in the facial midline during smiling,
she and her parents requested immediate onset of
treatment, concentrating only on the maxillary ante-
rior area with the aim to improve smile esthetics
with a short first interceptive approach. After the
eruption of the permanent dentition, a second or-
thodontic phase with fixed appliances followed by
minimally invasive restorative treatment mainly per-
formed with additive composite techniques would
then provide the patient with an esthetically pleasing
and functionally acceptable result during her teenage
years. Only after the end of the growth period,
definitive restorative therapy with ceramic veneers/
crowns would optimize the achieved teenage result
even further. Given the existing full Class II canine
and molar occlusion, extraction of the SMMCI and
orthodontic mesialization of the maxillary canines
and lateral incisors seemed the most efficient and
appropriate option, although the 6 anterior teeth
would require restorative treatment to correct the
significant Bolton discrepancy and to achieve satis-
factory esthetics.
July 2021  Vol 160  Issue 1
136 Schneider and Moser

Fig 4. Phase I treatment progress.

Fig 5. Situation after 9 months of phase I treatment.

TREATMENT PROGRESS
Orthodontic treatment started when the patient was
aged 10 years 2 months and was performed in 2 phases:
After extraction of the SMMCI, two 0.022-in lateral
incisor brackets (MBT Prescription Victory Series; 3M
Unitek, St Paul, Minn) for a sectional arch (0.016-in
nickel-titanium followed by a 0.019 3 0.025-in stainless
steel archwire) were bonded on the maxillary lateral in-
cisors with overangulation (negative tip) to avoid mesial
crown tipping during space closure. During the first
2 months, a denture tooth with a central incisor bracket
was inserted to cover the large extraction space for
improved esthetics (Fig 4). Two different kinds of Fig 6. Panoramic radiograph after phase I treatment.

July 2021  Vol 160  Issue 1 American Journal of Orthodontics and Dentofacial Orthopedics
Schneider and Moser
Fig 7. Facial and intraoral photographs before phase II treatment.
elastomeric chains (Power chain generation II with wide
space and closed space; Ormco Corporation, Glendora,
Calif) were applied according to the amount of residual
space in order not to exert too much force on the lateral
incisors. It was not intended to completely close the
SMMCI space but to merely narrow the large extraction
site, as future restorations of the maxillary anterior teeth
were regarded as inevitable. After 4 appointments at 8-
weeks intervals, space closure was finished simply
changing the power chain every 2 months. On the day
of bracket removal, provisional in-office composite res-
torations of the small mesialized lateral incisors were
performed to further reduce the remaining diastema
(Figs 5 and 6).
Because of the long journey to our office, arising
family and professional issues, and the necessity for a
second phase of orthodontic treatment, the joint deci-
sion was made not to apply any type of retainer and to
take the risk of partial space opening of the SMMCI
extraction site. The patient was not seen until the com-
plete eruption of the permanent dentition (Figs 7-9). As
expected, the SMMCI space had partially reopened, but
American Journal of Orthodontics and Dentofacial Orthopedics
137
the posterior occlusion had remained unchanged.
Phase II deemed to be straightforward, aiming merely
at orthodontic leveling of the lower curve of Spee and
at strategic positioning of the 6 maxillary anterior
teeth, including the first premolars, to avoid invasive
tooth preparation by the restorative dentist and to
improve the final interdisciplinary treatment outcome.
The interdisciplinary treatment plan was established
with the prosthodontist who was present during the
consultation with the patient and her parents before
the onset of phase II.
Orthodontic phase II treatment was initiated
with.022-in full fixed appliances (MBT prescription;
3M) at age 12 years 8 months. After complete leveling
and aligning of the dentition with 0.016-in, 0.014
3 0.025-in, and 0.019 3 0.025-in nickel-titanium arch-
wires, strategic space distribution in the maxillary arch
was achieved with sliding mechanics on a 0.019
3 0.025-in stainless steel posted archwire to provide
the restorative dentist with a manageable situation for
providing minimally invasive preliminary restorative ther-
apy (Fig 10). The patient was seen every 8 weeks.
July 2021  Vol 160  Issue 1
138 Schneider and Moser

Fig 8. Dental casts before phase II.

ORTHODONTIC TREATMENT RESULTS

After 13 months of phase II treatment, the fixed ap-
pliances were removed, a lower 3-3 lingual retainer was
bonded, and an upper Essix retainer was delivered to the
patient. She was immediately referred to her dentist for
the planned long-term provisional restorative treatment.
After debonding, dental arches are completely lev-
eled and aligned with adequate spaces for the planned
provisional restorations. As planned, a solid full Class
II canine and molar occlusion has been achieved by
mesialization of the maxillary dentition (Figs 11 and
12). Because of an atypical crown form of the
mandibular left second molar, a small mesial marginal
ridge discrepancy is evident. The orthopantomogram
shows the absence of root resorptions and good over-
all root parallelism. The developing maxillary third
molars have sufficient space for eruption, whereas
the mandibular right wisdom tooth is planned to be
removed. The condyles appear asymmetrical in shape,
which is reflected in a double contour of the lower
border of the mandible on the cephalometric radio-
graph. The cephalometric analysis evidences that
mandibular growth (A-N-Pg, 2.0 vs 0.5 ) in combi-
nation with mandibular autorotation (SN/Go-Gn,
36.7 vs 34.4 ), has occurred during orthodontic
treatment. The torque of the maxillary incisors has
been well-maintained despite substantial space closure
after extraction of the SMMCI (U1/SN, 104.7 vs
103.8 ). A 2 mm flattening of the upper and lower Fig 9. Panoramic and lateral radiograph before phase II
lip profile has occurred, which can be mainly treatment.

July 2021  Vol 160  Issue 1 American Journal of Orthodontics and Dentofacial Orthopedics
Schneider and Moser 139

Fig 10. Strategic space distribution with phase II treatment.

Fig 11. Posttreatment facial and intraoral photographs.

American Journal of Orthodontics and Dentofacial Orthopedics July 2021  Vol 160  Issue 1
140 Schneider and Moser

Fig 12. Posttreatment dental casts.

Fig 13. Posttreatment panoramic and cephalometric radiograph, pre-and posttreatment superimposi-
tion on cranial base.

July 2021  Vol 160  Issue 1 American Journal of Orthodontics and Dentofacial Orthopedics
Schneider and Moser 141

Table. Cephalometric summary

Variable Mean 6 SD Preorthodontcs Postorthodontics Postrestorative
SNA ( ) 82.0 6 3.5 80.4 79.4 79.7
SNB ( ) 80.0 6 3.5 77.4 78.4 78.6
A-N-Pg ( ) 2.0 6 2.5 2.0 0.5 1.0
SN/ANS-PNS ( ) 8.0 6 3.0 2.0 4.5 4.0
SN/Go-Gn ( ) 33.0 6 2.5 36.7 34.4 34.0
ANS-PNS/Go-Gn 25.0 6 6.0 34.9 30.6 30.0
U1/SN ( ) 102.0 6 6.0 104.7 103.8 104.6
L1/Go-Gn ( ) 94.0 6 7.0 89.4 92.2 91.4
L1/A-Pg (mm) 2.0 6 2.0 1.4 0.2 0.5
Overjet (mm) 3.5 6 2.5 4.0 3.0 3.5
Overbite (mm) 2.0 6 2.5 5.1 2.1 3.6
Interincisal Angle ( ) 132.0 6 6.0 129.0 130.0 128.0
LL/E-plane (mm) 2.0 6 0.0 0.5 3.7 3.2
UL/E-plane (mm) 4.0 6 2.0 2.0 5.5 4.7

Fig 14. Facial and intraoral photographs after restorative therapy.

attributed to the growth of the nose and chin (Fig 13, Chatsworth, Calif) 11.9 Edit feature was performed in
Table). the presence of the patient and her parents, who
An ad-hoc digital smile design using the Dolphin Im- watched how the smile could be improved by the
aging (Dolphin Imaging and Management Solutions, planned restoration of all 6 maxillary anterior teeth. In

American Journal of Orthodontics and Dentofacial Orthopedics July 2021  Vol 160  Issue 1
142
Fig 15. Facial and intraoral photographs 12 months in retention.
the meantime, the restorative dentist had created an
analog mock-up of the restoration, which the patient
could try on to show her how the final restorations
would improve her smile esthetics. One week later, the
patient received the fabricated restorations.
RESTORATIVE TREATMENT RESULTS
At debonding, the patient was 13 years 9 months
old and still growing (onset of menstruation 6 months
ago); hence, minimally invasive restorations were the
only restorative treatment option. The mesial and
distal contours of the mesialized small lateral incisors
were slightly parallelized for insertion of 2 resin
crowns with the form and size of 2 central incisors,
whereas the first premolars and canines received com-
posite restorations. The patient exhibits a harmonious
profile with good lip competence and lip projection
and a consonant smile arc with a mild increase of
gingival display (restorative dentist; Dr Giuseppe Pel-
litteri, Bolzano, Italy) (Fig 14). Only after the end of
the patient's growth period, definitive restorations
with ceramic crowns and laminate veneers would be
July 2021  Vol 160  Issue 1 American Journal of Orthodontics and Dentofacial Orthopedics
Schneider and Moser
indicated for further improvement of smile esthetics
and optimum periodontal health. In the meantime,
the patient was instructed to adopt a strict oral hy-
giene protocol with regular professional cleaning.
The patient wears an upper Hawley-retainer during
nighttime.
Twelve months into retention, when aged 15 years
4 months, the occlusion has remained stable, and the
gingival tissues around the 6 provisional restorations
have matured, although the patients's oral hygiene
could be improved (Fig 15). The dental casts show main-
tenance of solid bilateral interdigitation with full Class II
molar and canine dental relationships, normal overjet
and overbite, and the persistence of the mild 1 mm lower
dental midline deviation because of asymmetrical
mandibular growth. The crown form of the mandibular
left second molar is atypical, and because of its slight
mesial inclination, the occlusal contact with the antago-
nist is suboptimal. (Fig 16). The panoramic radiograph
evidences a smaller right condyle which may account
for the very mild lower midline deviation to the right
side. The cephalometric analysis reveals ongoing facial
Schneider and Moser
Fig 16. Dental casts 12 months in retention.
growth expressing a normodivergent very mild pseudo-
Class III pattern (SN/Go-Gn, 34.0 ; A-N-Pg, 1.0 mm),
and overall good maintenance of the interincisal angle
(129.0 vs 128.0 ). Maxillary molar mesialization has
occurred over the entire 2-phase treatment because of
spontaneous mesial migration with the use of the leeway
space and space closure after extraction of the SMMCI.
No further flattening of the profile has occurred during
the 1-year retention period (Fig 17, Table).
The patients and her parents are very satisfied with
the overall treatment result.
DISCUSSION
A SMMCI is a very rare dental anomaly which only a
few orthodontists may encounter in their professional
life, occuring either as a single dental anomaly or as a
symptom of the holoprosencephaly spectrum.
In these patients, both orthodontic space opening
and space closure are challenging procedures that
require comprehensive management of the malocclu-
sion and close interdisciplinary collaboration with the
involved dental specialists. However, a prosthodontic
replacement for a maxillary central incisor with either a
resin-bonded bridge or an implant-borne crown after
orthodontic space opening is a more aggressive proced-
ure than the only minimally invasive restoration of the
anterior dentition after orthodontic space closure or
spatial distribution.
Although many clinical papers about the SMMCI syn-
drome have been published, to our knowledge, up to
date, no single case report with the extraction of the
American Journal of Orthodontics and Dentofacial Orthopedics
143
SMMCI and orthodontic space closure is available in
the peer-reviewed literature.12-14,22-27
Space closure in association with minimally invasive
restorative treatment bears several advantages over the
space opening alternative, especially that the major
part of the entire treatment is already finished during
the patient's teenage years, and that no removable or
fixed interim substitution of the missing tooth with
the inherent risk of root approximation during the pre-
implant retention time, as reported by Olsen and Ko-
kich,28 is necessary.
Some critics might argue that 6 maxillary anterior
composite restorations can only be regarded as long-
term provisionals and will require future substitution
by ceramic crowns and laminate veneers, but the great
advantage of avoiding either a cantilever bridge or an
implant-borne crown cannot be overestimated enough.
The present patient has a dolichofacial pattern and ex-
poses a significant amount of the maxillary gingival
height during smiling. During the next years, passive
eruption and further maturation of the gingival soft tis-
sue will take place, leading to a lengthening of the clin-
ical crowns.29,30 Therefore, postponing any expensive
and invasive restorative treatment in the esthetic zone
is a wise decision. The literature offers overwhelming ev-
idence to prove that, especially in young female patients
with a high-smile line, an anterior dentition without
implants or bridgework is esthetically more pleasing
and periodontally healthier in the long run.30-33
The looming sequela of infraocclusion is unpredictable
and can never be completely excluded. Therefore,
July 2021  Vol 160  Issue 1
144 Schneider and Moser

Fig 17. Panoramic and cephalometric radiograph with superimpositions after restorative treatment.

implant-borne crowns in the esthetic zone should either
be avoided or delayed as long as possible.19,31-33
A second advantage of the space closure approach for
the present patient is that she will finish her treatment
with 28 natural teeth as her maxillary third molars
have enough space to spontaneously erupt. Had space
opening with molar distalization been performed,
surgical removal of these teeth would have been neces-
sary in the future.
An interesting alternative treatment strategy to or-
thodontic space closure in the esthetic zone in young
patients for the avoidance of long-term pink or white
esthetic issues is autotransplantation of a mandibular
premolar. In general, this approach has a very favorable

July 2021  Vol 160  Issue 1 American Journal of Orthodontics and Dentofacial Orthopedics
Schneider and Moser
long-term prognosis if the optimum time frame for au-
totransplantation is respected, and the combined
orthodontic-surgical-restorative treatment is performed
by a skilled and experienced interdisciplinary team.34-39
In these patients, prosthodontic treatment can be
limited to only the transplanted tooth. Despite the
encouraging long-term success rate for autotrans-
planted premolars into the anterior maxilla (.91%),
not all transplanted teeth survive long-term, so that
space closure is still the more predictable approach.
For the present patient, autotransplantation of a
mandibular premolar was not the best treatment option,
as no mandibular extractions were indicated.
If the patient and her parents had not requested an
immediate onset of treatment during the mixed denti-
tion to normalize the appearance of the smile, a single
phase of treatment after the complete eruption of the
entire dentition would have achieved a similar result in
less time. Considering that a significant amount of
relapse of the closed extraction site occurred between
the end of phase I and the beginning of phase II, main-
taining the achieved interim result with a lingual fixed
retainer or a removable device would have been indi-
cated and might have shortened the second phase of
treatment.
CONCLUSION
Orthodontic space closure after extraction of an
SMMCI with subsequent minimally invasive restorative
treatment should be considered a viable treatment op-
tion to finish treatment already at a young age,
providing the affected teenagers with a natural dentition
that can adapt to the continuous changes over the entire
life span, and achieving pleasing pink and white es-
thetics together with good function immediately after
the end of orthodontic therapy.
ACKNOWLEDGMENTS
The authors thank Dr. Karin Becktor, Hellerup,
Denmark, for her precious contribution to the present
case report.
REFERENCES
1. Scott DC. Absence of upper central incisor. Br Dent J 1958;104:
247-8.
2. Dolan LM, Willson K, Wilson WG. 18p-syndrome with a single cen-
tral maxillary incisor. J Med Genet 1981;18:396-8.
3. Nanni L, Ming JE, Du Y, Hall RK, Aldred M, Bankier A, et al. SHH
mutation is associated with solitary median maxillary central
incisor: a study of 13 patients and review of the literature. Am J
Med Genet 2001;102:1-10.
4. El-Jaick KB, Fonseca RF, Moreira MA, Ribeiro MG, Bolognese AM,
Dias SO, et al. Single median maxillary central incisor: new data
American Journal of Orthodontics and Dentofacial Orthopedics
145
and mutation review. Birth Defects Res A Clin Mol Teratol 2007;
79:573-80.
5. Cohen MM Jr. An update on the holoprosencephalic disorders. J
Pediatr 1982;101:865-9.
6. Hall RK, Bankier A, Aldred MJ, Kan K, Lucas JO, Perks AG. Solitary
median maxillary central incisor, short stature, choanal atresia/
midnasal stenosis (SMMCI) syndrome. Oral Surg Oral Med Oral
Pathol Oral Radiol Endod 1997;84:651-62.
7. DiBiase AT, Cobourne MT. Beware the solitary maxillary median
central incisor. J Orthod 2008;35:16-9.
8. Kjaer I. Etiology Based Dental and Craniofacial Diagnostics. United
Kingdom: John Wiley & Sons Ltd; 2017. p. 183.
9. Kjaer I, Becktor KB, Lisson J, Gormsen C, Russell BG. Face, palate,
and craniofacial morphology in patients with a solitary median
maxillary central incisor. Eur J Orthod 2001;23:63-73.
10. Becktor KB, Sverrild L, Pallisgaard C, Burhøj J, Kjaer I. Eruption of
the central incisor, the intermaxillary suture, and maxillary growth
in patients with a single median maxillary central incisor. Acta
Odontol Scand 2001;59:361-6.
11. Kjaer I, Balslev-Olesen M. The primary maxillary central incisor in
the solitary median maxillary central incisor syndrome. Eur J Pae-
diatr Dent 2012;13:73-5.
12. Bolan M, Derech CD, C^ orrea M, Ribeiro GLU, Almeida ICS. Palatal
expansion in a patient with solitary median maxillary central incisor
syndrome. Am J Orthod Dentofacial Orthop 2010;138:493-7.
13. Lygidakis NN, Chatzidimitriou K, Petrou N, Lygidakis NA. Solitary
median maxillary central incisor syndrome (SMMCI) with congen-
ital nasal puriform aperture stenosis: literature review and case
report with comprehensive dental treatment and 14 years
follow-up. Eur Arch Paediatr Dent 2013;14:417-23.
14. Pseiner BC. Premolar transplantation in a patient with solitary me-
dian maxillary central incisor syndrome. Am J Orthod Dentofacial
Orthop 2014;146:786-94.
15. Sailer I, Bonani T, Brodbeck U, H€ammerle CH. Retrospective clin-
ical study of single-retainer cantilever anterior and posterior
glass-ceramic resin-bonded fixed dental prostheses at a mean
follow-up of 6 years. Int J Prosthodont 2013;26:443-50.
16. Kern M, Passia N, Sasse M, Yazigi C. Ten-year outcome of zirconia
ceramic cantilever resin-bonded fixed dental prostheses and the
influence of the reasons for missing incisors. J Dent 2017;65:51-5.
17. Marzadori M, Stefanini M, Mazzotti C, Ganz S, Sharma P,
Zucchelli G. Soft-tissue augmentation procedures in edentulous
esthetic areas. Periodontol 2000 2018;77:111-22.
18. Thilander B, Odman J, Lekholm U. Orthodontic aspects of the use
of oral implants in adolescents: a 10-year follow-up study. Eur J
Orthod 2001;23:715-31.
19. Schwartz-Arad D, Bichacho N. Effect of age on single implant sub-
mersion rate in the central maxillary IncisorRegion: A long-term
retrospective study. Clin Implant Dent Relat Res 2015;17:509-14.
20. Cocconi R, Rapa S. Unilateral agenesis of the maxillary lateral
incisor: space closure versus space preservation in growing pa-
tients. Semin Orthod 2020;26:24-32.
21. Rosa M. Missing teeth in the smile area: space closure in all mal-
occlusions looking for long term health, esthetics and function.
Semin Orthod 2020;26:52-60.
22. Harrison M, Calvert ML, Longhurst P. Solitary maxillary central
incisor as a new finding in CHARGE association: a report of two
cases. Int J Paediatr Dent 1997;7:185-9.
23. Hall RK. Solitary median maxillary central incisor (SMMCI) syn-
drome. Orphanet J Rare Dis 2006;1:12.
24. Cho SY, Drummond BK. Solitary median maxillary central incisor
and normal stature: a report of three cases. Int J Paediatr Dent
2006;16:128-34.
July 2021  Vol 160  Issue 1
146
25. Machado E, Machado P, Grehs B, Grehs RA. Solitary median maxil-
lary central incisor syndrome: case report. Dent Press J Orthod
2010;1584:55-61.
26. Blackmore K, Wynne DM. A case of solitary median maxillary central
incisor (SMMCI) syndrome with bilateral pyriform aperture stenosis
and choanal atresia. Int J Pediatr Otorhinolaryngol 2010;74:967-9.
27. Holl I, Bartzela T, Jost-Brinkmann PG. Solitary Median Maxillary
Central Incisor (SMMCI)-Syndrom—mehr als eine Nichtanlage. Ein
Patientenbericht Oralprophylaxe Kinderzahnheilkd 2019;41:80-4.
28. Olsen TM, Kokich VG Sr. Postorthodontic root approximation after
opening space for maxillary lateral incisor implants. Am J Orthod
Dentofacial Orthop 2010;137:158.e1: discussion 158.
29. Morrow LA, Robbins JW, Jones DL, Wilson NHF. Clinical crown
length changes from age 12-19 years: A longitudinal study. J
Dent 2000;28:469-73.
30. Theytaz GA, Kiliaridis S. Gingival and dentofacial changes in ado-
lescents and adults 2 to 10 years after orthodontic treatment. J Clin
Periodontol 2008;35:825-30.
31. Jemt T, Ahlberg G, Henriksson K, Bondevik O. Tooth movements
adjacent to single-implant restorations after more than 15 years
of follow-up. Int J Prosthodont 2007;20:626-32.
32. Dierens M, De Bruecker E, Vandeweghe S, Kisch J, de Bruyn H,
Cosyn J. Alterations in soft tissue levels and aesthetics over a
16-22-year period following single implant treatment in peri-
odontally- healthy patients: a retrospective case series. J Clin Pe-
riodontol 2013;40:311-8.
July 2021  Vol 160  Issue 1 American Journal of Orthodontics and Dentofacial Orthopedics
Schneider and Moser
33. Op Heij DG, Opdebeeck H, van Steenberghe D, Kokich VG,
Belser U, Quirynen M. Facial development, continuous tooth
eruption, and mesial drift as compromising factors for
implant placement. Int J Oral Maxillofac Implants 2006;21:
867-78.
34. Kristerson L, Lagerstr€ om L. Autotransplantation of teeth in cases
with agenesis or traumatic loss of maxillary incisors. Eur J Orthod
1991;13:486-92.
35. Czochrowska EM, Stenvik A, Zachrisson BU. The esthetic outcome
of autotransplanted premolars replacing maxillary incisors. Dent
Traumatol 2002;18:237-45.
36. Stange KM, Lindsten R, Bjerklin K. Autotransplantation of premo-
lars to the maxillary incisor region: a long-term follow-up of 12-22
years. Eur J Orthod 2016;38:508-15.
37. Plakwicz P, Fudalej P, Czochrowska EM. Transplant vs implant
in a patient with agenesis of both maxillary lateral incisors: A
9-year follow-up. Am J Orthod Dentofacial Orthop 2016;149:
751-6.
38. Andreasen JO, Andersson L, Tsukiboshi M, Czochrowska EM. Au-
totransplantation of teeth to the anterior region. In: Andreasen JO,
Andreasen FM, Andersson L, editors. Textbook and color atlas of
traumatic injuries to the teeth. 5th ed. Wiley & Sons: Oxford,
UK; 2018. p. 853-75.
39. Czochrowska EM, Plakwicz P. Guidelines for autotransplantation
of developing premolars to the anterior maxilla. Semin Orthod
2020;26:61-72.