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46 rev port estomatol med dent cir maxilofac. 2021;62(1):42-49

The patient used a Kinesio tape (elastic bandage) around the with no need for the father to be present in the clinic or phys-
lips to increase the lip muscles’ motor function, improving lip ical restraint to carry out the treatment proposed. In this ap-
sealing, mouth opening, and speech. pointment, the cooperative behavior was reinforced again.
Treatment planning included a multidisciplinary approach Two weeks after hygiene and dietary guidance, improve-
and involved the extraction of the permanent first molar and ment in biofilm formation and gingival tissue was observed.
restoration of the deciduous tooth’s occlusal surface using The patient is under follow-up, with quarterly returns sched-
light-curing glass-ionomer cement (GIC). An orthodontic eval- uled to improve her and her family’s quality of life. A follow-up
uation was conducted in the preservation consultation, but individualized preventive program was established involving
due to the patient’s cognitive delay (difficulty to mechanically parents’ education to ensure appropriate daily oral hygiene
control biofilm), orthodontic therapy was not indicated. and diet and accompany the left mandibular second perma-
The patient cried a lot and manifested non-collaborative nent molar eruption. The timeline of interventions and out-
behavior. Because this was an emergency appointment due to comes is presented in Figure 8.
the child’s pain, who had been having difficulty eating and
sleeping for days, the permanent tooth extraction was per-
formed at the first visit. Protective stabilization with active Discussion and Conclusions
immobilization, involving restraint by her parents and dental
auxiliary, was performed to minimize the psychological stress CSS is a rare multiple congenital anomaly associated with
and/or decrease the risk of physical injury to the patient, the mutations in BAF complex genes. 2,7,8 These mutations are
parents, and the staff. Alternative behavioral approaches to linked to a broad range of diseases that, according to the gene
reduce movement and resistance and increase cooperation affected, range from non-syndromic or syndromic neurode-
when providing dental care for special-needs patients, such as velopmental disorders to low-grade tumors and malignan-
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distraction, shaping, modeling, and reinforcement, were used cies. Therefore, this case report’s patient is undergoing ge-
prior to implementing protective stabilization. netic monitoring to investigate the genotype and diagnose
Intraoral antisepsis was performed using 0.12% chlorhex- possible molecular mutation.
idine digluconate (Periogard™, São Paulo, SP, Brazil) for one The diagnosis of this condition is based on the presence of
minute, followed by extraoral antisepsis with 2% chlorhexidine the most frequent clinical characteristics, and the role of ge-
4
digluconate (Riohex Gard™, Rioquímica, São José do Rio Preto, netic evaluation is to complement diagnosis. Due to the ab-
SP, Brazil). After topical anesthesia, the lower alveolar and buc- sence of molecular tests, it was hypothesized that the patient
6
cal nerve was blocked using 2% lidocaine + epinephrine had CSS type A based on Schrier et al.’s clinical criteria. These
1:100.00 (Alphacaine™, Nova DFL, Taquara, RJ, Brazil), followed criteria require the presence of three predominant clinical
by syndesmotomy. findings and at least one of each of the three categories of the
Subsequently, odontosection was performed in the tooth’s syndrome’s less frequent clinical characteristics. These char-
buccal-lingual direction using a tapered drill (KG Sorensen, acteristics were found in our clinical case, similar to another
Cotia, SP, Brazil). The roots were removed using #151 dental study conducted in India. Due to the reported prevalence of
4
forceps (Quinelato, Rio Claro, SP, Brazil). The alveolar cavity was various findings, ‘classic’ clinical characteristics will not al-
irrigated with 0.9% saline (Tayuyna Laboratory, Nova Odessa, ways be present to support such a diagnostic strategy; thus,
SP, Brazil) and suture was performed using 4.0 silk thread next-generation molecular sequencing techniques are re-
(Sertix™, Shalon Fios Cirúrgicos, São Luís de Montes Belos, GO, quired to confirm the diagnosis of CSS.
Brazil). After the surgery, analgesics and antibiotics were pre- Fifth-digit hypoplasia/aplasia, a characteristic that gives
scribed, and the mother was instructed on feeding, hygiene, the syndrome its name, may not be evident in some cases of
and supervision to prevent the child from biting her lips invol- CSS. Although the presence of ‘classic’ characteristics can alert
untarily. the clinician to suspect the presence of CSS, the wide pheno-
At the second appointment, the suture was removed, and type variability suggestive of the absence of these character-
further instructions on oral hygiene and dietary advice were istics does not exclude the possibility of CSS. 2
provided. The patient was still withdrawn and non-coopera- The management and dental treatment of syndromic pa-
tive. Behavior guidance techniques, such as modeling and pos- tients vary according to the needs of each individual affected,
itive reinforcement, were used to alleviate anxiety and nurture and it is important to promptly detect the disease to enable
a positive dental attitude. Dental prophylaxis was performed early treatment and promote good development of the child’s
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using Clinpro™ toothpaste (3M, São Paulo, SP, Brazil) to condi- general and oral health. Our oral findings were not consis-
tion behavior for the next dental appointment for primary tent with some previously described findings, such as delayed
tooth restoration. Four applications of fluoride varnish (Du- eruption, macroglossia, micrognathia, and the occurrence of
raphat TM , Colgate-Palmolive Ind. and Com. Ltda, São Paulo, SP, cleft lip and palate and conoid teeth. However, high palate
1,6
2
Brazil), one per week, were planned for enhancing remineral- and diastema were observed, which may denote the genetic
ization of the permanent incisors. heterogeneity of the disorder since the phenotype is geno-
The patient returned seven days later to restore her decid- type-dependent. 5
uous mandibular second molar’s occlusal surface with encap- Abnormal growth of gingival tissue has been observed in
sulated resin-modified GIC using relative isolation (Riva Light patients with several syndromes and, when associated with
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Cure™, SDI Brasil Indústria e Comércio Ltda., São Paulo, SP, hypertrichosis, may denote the presence of CSS. Although
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Brazil). An improvement in the child’s behavior was observed, gingival growth’s pathogenesis is still unknown, it has been