The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014 Brief Clinical Studies

11. Toscano D, Baciliero U, Gracco A, et al. Long-term stability of alveolar LLC aplasia. We used en bloc nasal dorsal hump material for recon-
bone grafts in cleft palate patients. Am J Orthod Dentofacial Orthop struction in this case.
2012;142:289–299
12. Meazzini MC, Capasso E, Morabito A, et al. Comparison of growth
results in patients with unilateral cleft lip and palate after early CLINICAL REPORT
secondary gingivoalveoloplasty and secondary bone grafting: 20 years
follow up. Scand J Plast Reconstr Surg Hand Surg 2008;42:290–295
A 29-year-old woman presented with airway obstruction caused
13. Thuaksuban N, Nuntanaranont T, Pripatnanont P. A comparison of
by external nasal valve deficiency, a concavity at the right alar
autogenous bone graft combined with deproteinized bovine bone and rim, nasal tip asymmetry, underdeveloped dome, as well as defi-
autogenous bone graft alone for treatment of alveolar cleft. cient nasal tip projection and definition. She had a hump mainly
Int J Oral Maxillofac Surg 2010;39:1175–1180 in the cartilaginous dorsal vault. A basal view demonstrated a big-
ger nostril on the right side than that on the left side (Fig. 1).
She was operated on under general anesthesia after an adminis-
tration of a 1:100,000 mixture of 1-mg adrenaline with 1% lidocaine
solution. Nasal dorsal and cartilaginous exposure was achieved via
Congenital Isolated Aplasia of the Goodman classic inverted V-shaped transcolumellar incision
and an infracartilaginous incision performed as an open rhino-
Lower Lateral Cartilage and plasty procedure. During exploration of the cartilaginous structures,
the absence of the upper halves of the medial, intermediate, and lat-
Reconstruction Using Dorsal eral crus of the right LLC was determined (Fig. 2). A fibrotic band
Hump Material was present in place of absent structures. Because the patient did
not undergo surgical procedures or had any infection previously,
Gökhan Temiz, MD, Nebil Yeşiloğlu, MD, Murat Sarici, MD, this deformity was considered congenital. Cephalic resection from
Gaye Taylan Filinte, MD the left LLC, leaving 7 mm of a caudal segment, was then per-
formed. This was followed by en bloc resection of the bony and car-
Abstract: Congenital nasal abnormalities are very rare in the litera- tilaginous nasal hump. After harvesting a 2
3-cm septal cartilage
ture. There are 3 reported cases of isolated partial lower lateral car- graft, a columellar strut graft was prepared and adapted to the col-
tilage defects. In this article, we report the case of a patient with umellar pocket. The caudal end of the dorsal hump material was
then sutured to the remnant of the right medial crus with 5-0
congenital complete absence of the lower lateral cartilage. The pa-
polydioxanon suture material (Fig. 3). Using the same material,
tient had severe external nasal valve dysfunction and a concavity interdomal sutures to both left and reconstructed right sides were
of the alar vault, even in the resting position. The defect was recon- placed to achieve symmetry and to create a natural dome. The cra-
structed using resected and reshaped nasal dorsal hump material. nial end of the hump material was sutured to the remnants of the
At the end of a 12-month follow-up period, the patient was satisfied lateral crus at the right side, and cephalic resection was performed
with the functional and aesthetic results of the operation. No exter- to produce a symmetric dome (Fig. 3). After placing septocolu-
nal or internal nasal valve collapse occurred during inspiration. mellar sutures and septal diced cartilage for radix augmentation,
Such developmental abnormalities may be syndromic and require mucosa and skin closure was performed. A nasal splint was used
special attention for reconstruction. for 1 week. At the end of a 12-month follow-up period, the patient
was satisfied with the functional and aesthetic results of the opera-
tion (Fig. 4).
Key Words: Lower lateral cartilage, nasal dorsal hump
DISCUSSION
Nasal structures develop at the third gestational week from
frontonasal prominences by the formation of nasal placodes.4,5
C ongenital nasal anomalies have an incidence of 1:20 000 to
1:40 000 in newborns.1 There are various reports describing
non–syndromic aplasias of nasal cartilaginous structures in the liter-
These placodes grow as convex thickenings of the surface ectoderm
at the end of the fourth week.5
ature.1,2 We found 3 reported cases of isolated non–syndromic Then, primitive nasal pits are reshaped by the collapse of the
lower lateral cartilage (LLC) aplasia.3 Alar cartilage defects cause central portions of the placodes. Medial and lateral nasal processes
nasal tip deformities, and septal or conchal cartilages are usually appear around the nasal pits through mesenchymal proliferation.1
used for reconstruction. Our patient had congenital isolated nasal The caudal halves of the nasal septum and the medial crura are de-
veloped from the nasal process. Nasal bones, upper lateral car-
From the Department of Plastic Reconstructive and Aesthetic Surgery, tilages, and the lateral crura of LLC develop from the lateral nasal
Dr Lütfi Kırdar Kartal Training and Research Hospital, Kartal, Istanbul, process.5 The nasal apex and dorsum derive from the frontonasal
Turkey.
Received January 18, 2014.
Accepted for publication February 10, 2014.
Address correspondence and reprint requests to Gökhan Temiz, MD,
Department of Plastic Reconstructive and Aesthetic Surgery, Kartal
Lütfi Kirdar Training and Research Hospital, Dr. Lütfi Kirdar Eğitim
ve Araştirma Hastanesi Plastik Cerrahi Kliniği, Şemsi Denizer Cad.
E-5 Karayolu Cevizli Mevkii Kartal, Istanbul 34890, Turkey;
E-mail: [email protected]
The authors report no conflicts of interest.
Copyright © 2014 by Mutaz B. Habal, MD
ISSN: 1049-2275 FIGURE 1. Preoperative lateral, oblique, and basal views of the patient showing
DOI: 10.1097/SCS.0000000000000909 the nasal tip deformity.

© 2014 Mutaz B. Habal, MD e411

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Brief Clinical Studies The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014

FIGURE 2. View of the right lower lateral cartilage defect and a fibrotic band FIGURE 4. Lateral, oblique, and basal views 1 year after the surgery.
(arrow).
a long and strong graft material.14,22 According to Menick, a graft
prominence.1 Developmental defects in the lateral nasal processes for anatomic reconstruction must be longer than 35 mm. Moreover,
result in anomalies of the alar region.4 neither septum nor conchal cartilage alone is ideal for alar cartilage.
Because of these complicated stages of nasal development, iso- Although septum is thin, it cannot be bent as easily as alar cartilage,
lated deformities are extremely rare.3 Potter et al6 published the first and scoring may break the septal cartilage. On the other hand, con-
report on congenital anomalies of the nasal cartilage. Lewin7 re- chal cartilage is soft and elastic, but it is very thick and its vertical
ported columellar agenesis with a normally developed septum. support capacity is limited.14,23 En bloc–resected dorsal humps
Cases with the absence of columella that have an affected septum may contain variable amounts of bony blocks, which are sufficient
or medial crura of LLC8 and isolated nasal bone agenesis have also for nasal support.
been reported.9 There are many articles about isolated vomerine In our case, we performed 3-mm dorsal reduction with a 3-mm–
agenesis.10 A total of 7 congenital isolated alar defects have been long bony fragment on it. This bony block had a maximal width of
reported.4 Only 3 cases of LLC defects have been reported.2,11,12 1 cm and a maximal length of 4 cm, and it morphologically resem-
Craniofacial clefts, including nasal defects, were defined and bled LLC. After trimming approximately 1 mm, it was considered
classified by Tessier.13 This classification did not refer to the iso- elastic and strong enough for reshaping and supporting the nose.
lated congenital anomalies of the nose. Although our case can be It was successfully used for reconstructing the medial, intermedi-
compared with the Tessier No. 2 facial cleft, the pathway of the fa- ate, and lateral crura of LLC. The external nasal valve collapse
cial cleft overlaps with the cartilage defect, except that the bony was also corrected after the procedure with the improvement of in-
entirety of the case differs from the facial cleft. Thus, our case spiration. The contour was also improved by providing a strong
may be classified in the type 1 category according to the system structural support. The successful result in this rare deformity
of Losee et al1 (hypoplasia, atrophy). was achieved through open approach rhinoplasty combined with
The absence of the alar cartilage on one side and the different the novel use of dorsal hump material as a graft.
sizes of alar cartilages cause problems in nasal tip support, contour,
and symmetry.14 Alar cartilage defects and discrepancies have begun
to be treated using closed approaches.14 Sheen used crushed shield REFERENCES
and rim grafts to repair the defects and correct the asymmetries of 1. Losee JE, Kirschner RE, Whitaker LA, et al. Congenital nasal anomalies:
the nasal cartilage.15,16 Peck used columellar strut and umbrella a classification scheme. Plast Reconstr Surg 2004;113:676–689
grafts for tip projection and lateral alar support.17,18 Constantian19 2. Diver AJ, Hill C. Congenital nasal cartilage deformities. Ann Plast Surg
used an endonasal tip grafting method for the augmentation of the an- 2011;67:665–667
atomic subunits of the nasal tip. 3. Barutca SA, Öreroğlu AR, Usçetin I, et al. Isolated congenital partial
Sheen’s work is an important precedent for the effects of cor- absence of the left lower lateral nasal cartilage: case report. Ann Plast
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because of the difficult anatomy of cartilage defects and the need 4. Mutaf M, Günal E. A new technique for reconstruction of a congenital
for blind graft insertions into small pockets.14,17,18 Therefore, open isolated alar defect. J Craniofac Surg 2010;21:503–505
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geons.14,19,20 This technique is simpler than the closed approach, 2006;17:602–604
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by Burget and Menick,21 may cause scars and deformations of the 1988;25:58–63
nasal tip anatomy. Another problem of this method is that it needs 8. Jacobs KF. Congenital aplasia of the nasal columella and cartilaginous
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left lower lateral cartilage. Otolaryngol Head Neck Surg
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Plast Reconstr Surg 2012;130:495e–497e
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FIGURE 3. Intraoperative view. Cartilaginous end of the nasal hump excision
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material was sutured to the medial crural remnant (left), whereas the bony end 14. Neu BR. A problem-oriented and segmental open approach to alar
was sutured to the posterior remnant of the lateral crus (right) and the tip cartilage losses and alar length discrepancies. Plast Reconstr Surg
complex was formed through interdomal suturation. 2002;109:768–779

e412 © 2014 Mutaz B. Habal, MD

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014 Brief Clinical Studies

15. Sheen JH, Sheen AP. Aesthetic Rhinoplasty. 2nd ed. St Louis: Results: Ninety-one subjects were studied. Radiographic features
Mosby, 1987 and sizes were different between the 2 groups. The frequency of re-
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17. Peck GC. Techniques in Aesthetic Rhinoplasty. Philadelphia: mandible. Cox regression demonstrated a positive effect of follow-up
Lippincott, 1990 time on the risk of recurrence. In a 1-year increase in the follow-up
18. Mavili ME, Safak T. Use of umbrella graft for nasal tip projection. time, the recurrence rate increased 1.07 times. An analysis of the data
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19. Constantian MB. Elaboration of an alternative, segmental, rence rate. A 1-cm increase in the lesion's size increased the recurrence
cartilage-sparing tip graft technique: experience in 405 cases. Plast
Reconstr Surg 1999;103:237 rate 1.67 times. A receiver operating characteristic test demonstrated
20. Gruber RP. Open rhinoplasty. Clin Plast Surg 1988;15:95 5.25 cm as a cutoff point in radiographic size; the radiographic size
21. Burget GC, Menick FJ. Aesthetic Reconstruction of the Nose. sensitivity was 88.5% (69.8%–97.4%), and specificity was 93.8%
St Louis: Mosby, 1992 (85%–98.3%) for recurrence lesions. Lesions that were 5.25 cm or
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Conclusions: The radiographic size may be a predictive factor for
the frequency of recurrence lesions in OKCs. Primary multilocular le-
sions had a greater probability for recurrence than unilocular lesions.

Correlation of Radiographic Key Words: Odontogenic keratocysts, mandible, enucleation,

Features and Treatments With marsupialization, cyst

the Frequency of Recurrence

in Odontogenic Keratocysts of O dontogenic keratocysts (OKCs) are benign neoplasms defined
by the World Health Organization as a benign unicystic or
multicystic, intraosseous tumor of odontogenic origin with a char-
the Mandible acteristic lining of parakeratinized stratified squamous epithelium
and a potential for aggressive, infiltrative behavior.1
Reza Tabrizi, DMD,* Mahsa Omidi, DMD,† The OKC has occurred in approximately 11% of odontogenic
Mehrnoosh Dehbozorgi, DMD,† Mina Hekmat‡ cysts.2 The peak occurrence of OKC is in the second and third decades
of patients’ lives. The peak seems to be roughly a decade younger for
Purpose: Odontogenic keratocysts (OKCs) have high recurrence women.3 Nevertheless, data have shown that OKCs are found much
potential. The aim of the study is to evaluate the correlation of ra- more frequently in men than in women. This sex difference seems to
diographic features and treatments with the frequency of recurrence be even more pronounced in the black community.4
in OKCs in the mandible. This lesion is commonly found in the maxilla and mandible and
Materials and Methods: This is a cohort retrospective study. The can become quite large because of its potential for significant ex-
size of the lesions was measured on orthopantomography. Radio- pansion, extension into adjacent tissues, and rapid growth.5,6
graphic features (unilocular or multilocular), location, treatment type Recurrence rates ranging anywhere from less than 10% to more
than 60% have been reported.7,8 Despite high recurrence of OKCs,
(enucleation or marsupialization), follow-up time, age, and sex were
few studies focused on the correlation of radiographic and clinical
variable factors. Subjects were studied in 2 groups: a recurrence group features of them with recurrence rate.9
and a nonrecurrence group. The radiographic size was considered to be The aim of the current study was to address the following
a predictive factor, and the frequency of recurrence was an outcome of questions: Is there any correlation between the radiographic size
the study. An independent t-test and chi-squared test were used to com- of OKCs and the frequency of the recurrence of OKCs? Do the
pare variables between recurrence and nonrecurrence groups. Cox multilocular lesions have higher recurrence rate than unilocular le-
regression was applied to the time effect evaluation of the lesion’s size sions? Which treatments are associated with higher recurrence rate?
and recurrence. A receiver operating characteristic test was used to find
a cutoff point for recurrence in the size of radiographic views. MATERIALS AND METHODS
The authors designed and implemented a cohort retrospective study.
The study sample was derived from the population of patients who
From the *Department of Oral and Maxillofacial Surgery, Shahidbeheshti had scheduled appointments at the Department of Oral and Maxillofa-
University of Medical Sciences, Tehran, Iran; and Departments of †Oral cial Surgery between September 1, 2005, and October 31, 2011. Ap-
and Maxillofacial Radiology and ‡Dentistry, Shiraz University of Medi- proval for the study was given by the research committee of the
cal Sciences, Shiraz, Iran. medical ethics group of Shiraz Medical Science University. Approved
Received December 23, 2013. subjects eligible to be included in the study had a pathologic-proved
Accepted for publication February 10, 2014. OKC and underwent enucleation (without Carnoy solution) or
Address correspondence and reprint requests to Reza Tabrizi, DMD,
marsupialization. Subjects were excluded from the study enrollment if
Department of Oral and Maxillofacial Surgery, Shahidbeheshti Dental
School, Velenjak, Tehran, Iran; E-mail: [email protected] they had nevoid basal cell carcinoma syndrome (Gorlin syndrome)
The authors report no conflicts of interest. and had undergone resection of the mandible. Each subject’s age, sex,
Copyright © 2014 by Mutaz B. Habal, MD and follow-up time were documented. Sites of lesions were categorized
ISSN: 1049-2275 into 4 sites: ramus, angle body, and symphysis. An orthopan-
DOI: 10.1097/SCS.0000000000000910 tomography (OPG) was taken immediately before the first operation,

© 2014 Mutaz B. Habal, MD e413

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.