INTRODUCTION
Craniomaxillofacial clefts are rare malformations but with disfiguring potential.
The incidence is estimated at 1.4-4.9% for every 100,000 live births. Tessier developed
a classification system based on facial lines, starting from 0 to 141.
Cleft number 0 has unique characteristics due to the possibility of having normal
tissue, in excess or even deficient, in addition to the possibility of association
with other clefts, such as cranial number 14, generating a wide spectrum of phenotypes.
The variability of presentations is expressed from a simple lip notch or a slight
bifid nose to a complete division of the structures of the midline of the face2.
When there is the involvement of the nasal tip, it is important that there is an improvement
in support, gaining projection and, when necessary, also greater rotation. For this
purpose, the tongue-in-groove technique is an arsenal to create a connection between
the septum and medial crura, which can be very useful in certain presentations of
median clefts3.
OBJECTIVE
This article aims to present a report of a bifid nose approach in a patient with 0-14
Tessier cleft.
CASE REPORT
AJPO, 15 years old, female, being followed up since she was 8 years old at the Plastic
Surgery outpatient clinic, complaining of an enlarged nose associated with episodes
of bullying at school. She had no other complaints, such as hyperteleorbitism. She
had no other comorbidities.
Surgical planning involved radiological evaluation with computed tomography in thin
sections and 3D reconstruction, and the ideal time was planned for after the patient
had completed the phases of puberty and bone development.
She underwent a one-time procedure on December 9, 2019, lasting 4 hours, under general
anesthesia at the Hospital das Clínicas of the Faculty of Medicine of Botucatu - UNESP
(HC FMB UNESP).
Patient in the horizontal supine position, under orotracheal intubation. A 4 cm subcostal
incision was made in the right inframammary fold with bright green infiltration with
a solution containing 2% lidocaine, 0.9% saline solution and 1:200,000 adrenaline.
This site was incised, followed by dissection until the identification of the sixth
costal arch and opening of the perichondrium to collect cartilage 5 cm in length.
The closure was done by planes and with an occlusive dressing.
Nasal access was made with a transcolumellar “W” and infracartilaginous incision after
previous local infiltration, with 1:100,000 epinephrine and 0.75% ropivacaine added
to 0.9% saline. The covering flap was lifted after careful dissection with exposure
of the osteocartilaginous framework, where it was noted: nasal bone with double contour,
in the shape of a gull’s wing; absence of conventional cartilaginous septum; distant
triangular cartilages; and distant alar cartilages with thick and unusually curved
ligaments.
The medial crus were separated, and the bone cartilages of the nasal dorsum were separated,
allowing three osteotomies on each side, the first being paramedian close to the center;
the second, oblique from lateral to medial, which removed a triangular slice that
contained bilateral bone defect; and the third, low-high completed by digital pressure.
After hemostasis, spreader grafts measuring 40 mm x 4 mm on each side were implanted
using 5-0 polypropylene thread.
Closure made in 5 layers: septum (osteofibrous remnant), two grafts and two triangular
with three stitches. Afterward, the medial crura of the wings were sutured with a
columellar cartilage strut measuring 25 mm x 3 mm and fixed with 5-0 polypropylene
thread and the strut with a tongue-in-groove spreader. The cephalic margins of the
alar cartilages were then transectioned, maintaining 6 mm in length, followed by transdomal
and interdomal sutures with 5-0 polypropylene thread, maintaining a diamond shape
in the nasal neodome.
Subsequently, a shield-type cartilage graft was performed for projection and camouflage
of the nasal tip and a graft of crushed cartilage on the back, covered by perichondrium.
Finally, the transcolumellar and intracartilaginous incisions were synthesized with
6-0 mononylon and 4-0 catgut, respectively, with a bilateral nasal splint implant
fixed by a 3-0 mononylon thread. The dressing was made with micropore, aquaplast and
nasal packing.
She was discharged 24 hours after the procedure, removing the nasal pack, prescription
of non-steroidal anti-inflammatory for 5 days, amoxicillin with clavulanate for 7
days and nasal solution with a saline solution daily. On the 8th postoperative day, the nasal splint and aquaplast and the external non-absorbable
sutures were removed.
The outpatient return took place on the 4th, 8th, 30th, 6th and 15th postoperative month. She was asymptomatic and satisfied with the aesthetic and functional
results, with nasal projection gain and absence of the bifid appearance (Figure 1). The radiological and pre, intra and postoperative images are shown below (Figures 2 to 5).
Figure 1 - Right to left: preoperative, 6th month post-operative and 15th month post-operative photos in anterior view.
Figure 1 - Right to left: preoperative, 6th month post-operative and 15th month post-operative photos in anterior view.
Figure 2 - Right to left: preoperative photo, 6th month postoperatively and 15th postoperative month in left profile view.
Figure 2 - Right to left: preoperative photo, 6th month postoperatively and 15th postoperative month in left profile view.
Figure 3 - Right to left: preoperative photo and computed tomography and 6th postoperative month.
Figure 3 - Right to left: preoperative photo and computed tomography and 6th postoperative month.
Figure 4 - Intraoperative cartilage grafts (right). Aspect of the incision in the right inframammary
region after 6 months (left).
Figure 4 - Intraoperative cartilage grafts (right). Aspect of the incision in the right inframammary
region after 6 months (left).
Figure 5 - Intraoperative aspect of the nasal dorsum (right). Strut appearance with tongue-in-groove
spreader (center). “Shield” cartilage for nasal tip camouflage (left).
Figure 5 - Intraoperative aspect of the nasal dorsum (right). Strut appearance with tongue-in-groove
spreader (center). “Shield” cartilage for nasal tip camouflage (left).
DISCUSSION
The mechanism of Tessier’s cleft 0 is not fully understood, but its onset in the third
week of gestation is recognized4. It results in failure of fusion of the two medial nasal processes, and when this
occurs, the presence of a bifid nose is frequent. Eventually, this error in embryogenesis
can cause agenesis or even duplication of these structures in the midline. Meanwhile,
cleft 14 is characterized by cranial alteration, with marked hyper or hypoteleorbitism5.
In our report, the patient presents, in addition to a bifid nose, hypertelorbitism.
However, the patient did not present this complaint during the follow-up, and we opted
for an exclusive approach to the nose6.
The treatment of bifid nose has been sought for a long time7, and in previous articles, there is a report of an open approach, resecting the tissues
that were in excess and with associated cartilage grafts4. Due to the rare incidence, studies are scarce, even though the craniofacial cleft
is the most common. The great phenotypic variation of the face in this syndrome is
also present in the nose: short and wide columella, duplicated or absent nasal septum,
flat dorsum, also variable nasal tip. The case series shows that this diversity also
alters the standardization of treatment, which must be individualized8.
The technique chosen for rhinoplasty was the open structure, with nasal tip reconstruction
being the most challenging step. Based on previous good results, the tongue-in-groove
technique was chosen for predictable and permanent projection and tip rotation, based
on previous good results9.
The treatment still proves successful when performed as a single procedure in most
series, obviously varying depending on the number of associated procedures to correct
common defects, especially labral defects10.
CONCLUSION
Open structure rhinoplasty using the tongue-in-groove technique can provide a satisfactory
esthetic result in a patient with a bifid nose secondary to a 0-14 Tessier cleft.
REFERENCES
1. Tessier P. Anatomical classification facial, cranio-facial and latero-facial clefts.
J Maxillofac Surg. 1976;4(2):69-92.
2. da Silva Freitas R, Alonso N, Shin JH, Busato L, Ono MC, Cruz GA. Surgical correction
of Tessier number 0 cleft. J Craniofac Surg. 2008;19(5):1348-52.
3. Antunes MB, Quatela VC. Effects of the Tongue-in-Groove Maneuver on Nasal Tip Rotation.
Aesthet Surg J. 2018;38(10): 1065-73.
4. Mazzola RF, Mazzola IC. Facial clefts and facial dysplasia: revisiting the classification.
J Craniofac Surg. 2014;25(1):26-34.
5. Nam SM, Kim YB. The Tessier number 14 facial cleft: a 20 years follow-up. J Craniomaxillofac
Surg. 2014;42(7):1397-401.
6. Pidgeon TE, Flapper WJ, David DJ, Anderson PJ. From birth to maturity: midline tessier
0-14 craniofacial cleft patients who have completed protocol management at a single
craniofacial unit. Cleft Palate Craniofac J. 2014;51(4):e70-9.
7. Miller PJ, Grinberg D, Wang TD. Midline cleft. Treatment of the bifid nose. Arch Facial
Plast Surg. 1999;1(3):200-3.
8. Spataro EA, Most SP. Tongue-in-Groove Technique for Rhinoplasty: Technical Refinements
and Considerations. Facial Plast Surg. 2018;34(5):529-38.
9. Tawfik A, El-Sisi HE, Abd El-Fattah AM. Surgical correction of bifid nose. Int J Pediatr
Otorhinolaryngol. 2016;86:72-6.
10. Kolker AR, Sailon AM, Meara JG, Holmes AD. Midline Cleft Lip and Bifid Nose Deformity:
Description, Classification, and Treatment. J Craniofac Surg. 2015;26(8):2304-8.
1. Hospital das Clínicas, Faculty of Medicine of Botucatu, Plastic Surgery, Botucatu,
SP, Brazil.
Corresponding author: Balduino Ferreira de Menezes Neto, Avenida Professor Mário Rubens Guimarães Montenegro, S/N - Jardim Sao Jose, Botucatu
- SP, Brazil, Zip Code 18618-970, E-mail: balduinofmneto@gmail.com
Article received: December 02, 2020.
Article accepted: May 18, 2021.
Conflicts of interest: none.