Case Report
The Cleft Palate-Craniofacial Journal
Amniotic Band Syndrome: A Multidisciplinary
Care Approach to the Treatment of a
Rare Case
Padma M. Mukherjee, BDS, DMD, PhD1,
Marianella Natera, DMD, MDS2, Howard Drew, DMD3,
and Adriana Creanga, BDS, MS, DABOMR4
Abstract
Amniotic band syndrome (ABS) is a rare developmental disorder associated with defects such as syndactyly, facial and/or palatal
clefts, and dental anomalies like malformed or impacted teeth. In this report, a patient with ABS was successfully treated with
orthodontic, endodontic, and periodontal therapies. Cone beam computed tomography revealed a unique eruptive path of the
impacted central incisor through the incisive canal and cleft area. The tooth was occlusally and functionally stable after 1 year of
treatment. Multidisciplinary care was critical to the success of this case. Available literature on ABS is also briefly reviewed.
Keywords
amniotic band syndrome, cleft palate, orthodontics, CBCT
Introduction
Amniotic band syndrome (ABS) is a rare congenital disorder
caused by early amnion rupture resulting in entrapment
of body parts in utero (Torpin, 1965). It is found in 1 in
1200 to 15 000 live births. There is no gender or ethnic pre-
disposition (Temtamy and McKusick, 1978). Amniotic band
syndrome can result in a number of developmental abnorm-
alities in ring-like finger constrictions, syndactyly, facial
clefts, cleft lip, and/or cleft palate associated with dental
anomalies (Coady et al., 1998). Amniotic band syndrome is
associated with the ADAM (amniotic deformity, adhesions
and mutilations) complex (Keller et al., 1978). Amniotic band
syndrome is treated by managing specific symptoms that may
include in utero surgery, surgery to manage congenital ampu-
tations, 3-D printed prosthetics, manoplasty, and so on. Oro-
facial management may involve cleft repair surgeries, caries
control, orthodontic treatment to help develop the arches, and
improve alignment, function, and prosthetic treatment (Coady
et al., 1998; Hotwani and Sharma, 2015).
Case Report
A 12-year-old girl presented to the department of Orthodontics
at the Rutgers Dental School of Medicine. The patient had a
previously known diagnosis of ABS and was surgically treated
1-5
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for her facial and palatal clefts as a child. Figure 1 depicts her
pretreatment records. Extraorally, the patient had a facial
asymmetry and postsurgical scarring at the lip and ocular areas.
She had asymmetry in the vertical heights of her eyes and ears.
Upon smiling, the patient had limited movement of her lip on
the left side. Intraorally, the patient had maxillary arch con-
striction and presented with a unilateral posterior crossbite on
the left side. The patient was in mixed dentition with a partially
impacted maxillary left central incisor (tooth #9). The ortho-
dontic treatment plan was to perform comprehensive fixed
appliance therapy with preadjusted bracket system, expand the
maxilla with rapid palatal expansion (RPE), facilitate the erup-
tion of the impacted maxillary left central incisor (tooth #9),
1
Department of Orthodontics, Rutgers School of Dental Medicine, Newark,
NJ, USA
2
Department of Endodontics, Rutgers School of Dental Medicine, Newark, NJ,
USA
3
Department of Periodontics, Rutgers School of Dental Medicine, Newark, NJ,
USA
4
Department of Diagnostic Sciences, Rutgers School of Dental Medicine,
Newark, NJ, USA
Corresponding Author:
Padma M. Mukherjee, Department of Orthodontics, Rutgers School of Dental
Medicine, 110 Bergen Street, Newark, NJ 07103, USA.
Email: pmmukherjee@yahoo.com
2 The Cleft Palate-Craniofacial Journal XX(X)

Figure 1. Initial photographs: 8-year-old female patient with amniotic band syndrome. Extraorally: facial and ocular asymmetry is present with
postsurgical scarring. Intraorally: maxillary constriction, unilateral crossbite, and partially impacted tooth #9 are noted.

Figure 2. Periapical (PA) radiographic images of tooth #9. A, Periapical radiograph showing mesio-vertical radiolucency with apical root
resorption. B, Gutta percha point demonstrating endo-perio lesion. C, After completion of root canal therapy and (D) at 1-year follow-up.

and improve the patient’s functional occlusion and esthetics.
Bands were fitted on the permanent first molars and the maxilla
was expanded with a Hyrax type of expander with a RPE pro-
tocol of 2 turns per day until the posterior crossbite was cor-
rected (6 mm). A preadjusted edgewise bracket system (3M
Unitek, Monrovia, California) with 0.022  0.028-inch slot
was utilized, and maxillary left central incisor was brought into
the arch using overlay wire (0.014 NiTi) on a 0.017  0.025-
inch stainless steel base arch wire. Patient had poor oral
hygiene that led to localized gingivitis around tooth #9. During
treatment, the mobility of tooth #9 increased and a periapical
radiograph was taken (Figure 2A). Probing depths around tooth
#9 revealed a pocket of 8 mm with purulent exudate and a
negative response to cold and electronic pulp test and the endo-
dontic diagnosis was pulpal necrosis with chronic apical
abscess with respect to tooth #9. A periapical radiograph with
a gutta percha point inserted into the periodontal pocket of
tooth #9 revealed a mesio-vertical bone loss extending up to
Mukherjee et al
Figure 3. CBCT images of maxillary left central incisor. Presence of communication of the maxillary left central incisor with the incisive canal
and path of traction during the orthodontic treatment. CBCT indicates cone beam computed tomography.
the apex (Figure 2B). Upon consultation with the endodontics
and periodontics departments, it was decided to orthodontically
stabilize the tooth #9 and then perform root canal therapy
(RCT; Figure 2C), followed by supragingival debridement and
occlusal adjustment. Follow-up appointments at 3 and 6
months showed improvement in mobility and the tooth #9 was
stable clinically. After a year, a periapical radiograph
appeared to depict some “bone fill” on the mesial aspect of
the tooth (Figure 2D). Although probing depths >4 mm were
still present mesially and buccally with respect to the max-
illary left central incisor, no purulent exudate was present.
The patient was referred for a small volume cone beam com-
puted tomography (CBCT) scan to obtain a 3-dimensional
view of the periodontal status of the maxillary let central
incisor (Figure 3). The bone “loss” noted around the tooth
was most likely due to the tooth movement traction path inter-
section with the incisive canal/foramen and the area of the
cleft. The tooth was stable, so orthodontic appliances were
removed. A 0.0195-inch dead-soft coaxial bonded wire retai-
ner (Respond; Ormco Corp, Orange, California) was placed
on the lingual surfaces of the maxillary incisors to prevent
relapse of the teeth in the cleft area. In addition to the bonded
retainer, the patient was provided with removable Essix Ace®
(Raintree Essix Inc, Sarasota, Florida) retainers to facilitate
ease in oral care maintenance at home.
Discussion
Amniotic Band Syndrome cases are rare and unique. While the
precise etiology is not known, Torpin et al., in 1965, proposed
that ABS is likely caused by the rupture of the amnion during
early embryonic development. The amnion splits into several
strands that could adhere to the body parts of the developing
fetus. This could lead to amputations and/or constriction rings
causing fusion of digits or parts that get entangled (Levy,
1998). While the etiology of facial clefts is multifactorial,
amniotic bands are considered the least probable cause (Losee
3
and Kirschner, 2008). With advance in imaging technology in
the past 5 years, there are newer investigational and in utero
surgeries that are now being carried out at select craniofacial
centers throughout the country (Javadian et al., 2013). Early
intervention has the potential to minimize the structural defor-
mities in the developing fetus. However, the patient described
in this report received several surgical corrections for her facial
and limb deformities, but only after birth. An increase in aware-
ness about ABS is therefore required to help families that have
been diagnosed with ABS in their developing fetuses to seek
treatment early when applicable.
In addition to craniofacial and limb deformities, patients
with ABS may have several dental anomalies such as missing,
malformed, impacted and/or supernumerary teeth. However,
due to the preexisting medical needs of such patients, dental
care at home is often compromised. There is limited literature
regarding dental and/or orthodontic management of patients
with ABS. The case discussed here helps outline possible
orthodontic treatment options and potential risks of that treat-
ment for patients with ABS. Due to the presence of dental
malformations such as impactions, dilacerations, and missing
teeth, dental treatment for such cases involves a need-based
multidisciplinary care approach along with patient and parental
motivation (Coyle et al., 2008). Poor oral hygiene alone can
become an issue if patients require orthodontic treatment. Pla-
cement of fixed orthodontic appliances may increase plaque
retention and lack of oral hygiene maintenance by the patient
can aggravate gingivitis and may lead to periodontal complica-
tions. Close monitoring of the patient is needed especially
throughout active orthodontic treatment. In addition to regular
6 month visits to the dental office, these patients may require
frequent visits such as every 3 months with their dentist. Here,
we highlight the significance of multidisciplinary dental care of
a patient with ABS. In the case presented, timely referral was
promoted when periodontal involvement was suspected which
ensured better care for the patient. During orthodontic treat-
ment, patients tend to develop gingivitis due to lack of proper
4 The Cleft Palate-Craniofacial Journal XX(X)

Figure 4. Posttreatment photographs: records taken after orthodontic appliance removal and at 1-year retention.

dental home care. After removal of the orthodontic appliances,
gingival inflammation was noted (Figure 4). Oral hygiene
instruction was reinforced, (toothbrush, floss, and floss threa-
ders), and full mouth scaling and root planning was performed.
The gingival inflammation reduced considerably, although
marginal inflammation remained due to inadequate oral
hygiene. Better brushing techniques and patient counselling
is required to motivate the patient to improve dental care. In
the case presented here, the patient responded well to ortho-
dontic forces, and we were able to successfully bring the par-
tially impacted tooth #9 into the arch. Rapid palatal expansion
was utilized to widen the maxilla and eliminate the unilateral
posterior crossbite of the patient. With the underlying cleft
history of the patient, special attention was given to maintain
the transverse correction and the expander was kept in the
mouth for 6 months during the leveling and aligning phase of
orthodontic treatment. With frequent periodontal maintenance,
orthodontic treatment could continue. However, due to the
endodontic complications with tooth #9, RCT was performed
mid treatment. Retrospectively, temporary cessation from
orthodontic treatment and removal of anterior appliances could
have been done until the oral hygiene of the patient improved.
Mukherjee et al
However, the risk of losing the palatal expansion obtained and
dental relapse due to early removal of appliances would have
been a possible outcome. Also, during treatment, only periapi-
cal radiographs were taken, and they did not reveal the approx-
imation of the root of tooth #9 with the incisive canal. A CBCT
image mid treatment would have been helpful. The periapical
radiograph in addition to the clinical examination suggested the
need for RCT for tooth #9. The local infection was addressed
with the RCT; the pain subsided and the tooth was occlusally
stable. One-year follow-up CBCT revealed the unique path of
extrusion of the maxillary left central incisor with orthodontic
treatment. It is possible that during treatment, the infection was
impinging against the soft tissues of the canal and spread rap-
idly at that time. The local periodontal and periapical infection
was resolved after a year. Due to the presence of the cortica-
tions around the incisive canal, bone fill was not completely
possible despite the periodontal and endodontic therapies. In
the long term, if the treated incisor can be maintained until
growth is complete, the tooth can be reassessed for additional
therapy and/or extraction and guided bone regeneration.
Furthermore, the severity of the root resorption of maxillary
left central incisor could be associated with the large amount of
orthodontic movement and the contact with the incisive canal
(Chung et al., 2015). For better initial stability, after removal of
the orthodontic appliances, a bonded palatal retainer 0.0195-
inch dead-soft coaxial bonded wire retainer (Respond; Ormco
Corp) was placed to act as a splint (Figure 4).
The initial presentation of tooth #9 was high in the vestibule
and partially impacted. With respect to orthodontic treatment,
tooth #9 had a favorable outcome. However, due to the period-
ontal and endodontic complications during treatment, the long-
term prognosis of tooth #9 remains questionable.
It is perceivable that periodontal laser therapy with an
Nd:YAG PerioLase laser (Millennium Dental Technologies
Inc, Cerritos, California) could reduce pocket depths, gain
probing attachment levels, and histologically gain new cemen-
tum and new connective tissue attachment (Yukna et al., 2007).
Although the soft tissue present from the incisive canal could
harm regeneration, the clot formed after bone modification
could prevent soft tissue migration and allow for regeneration.
In addition, proximal cells from the periodontal ligament and
alveolus can aid in regeneration. Recent human histological
studies using the PerioLase have shown that there is a potential
for a degree of regeneration with new cementum, periodontal
ligament, and alveolar bone is possible (Nevins et al., 2012). In
this ABS case, postorthodontic treatment, the tooth stabilized a
year after successful endodontic treatment and comprehensive
periodontal care, possibly due to the removal of local infection.
5
We conclude that orthodontic treatment can be successfully
performed on patients with ABS using a team approach.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to
the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research, author-
ship, and/or publication of this article.
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