Tessier Number 30 Facial Cleft: A Rare Maxillofacial Anomaly.
Number 30 facial cleft was first reported as a caudal extension of number 14 and number 0 facial clefts by Tessier in 1976.  The deformities vary in their presentation, ranging from minor clefting of the lower lip  to complete clefts of the mandible with the absence of the hyoid bone, thyroid cartilage, and manibrium.  In this article, we present a case of midline lower lip cleft that was repaired in late adolescent period and a review of the literature.
A 20-year-old male patient was referred to our clinic with a midline cleft and ulceration of the lower lip without any other involvement [Figure 1]. Several treatment methods such as antibiotic ointments and antihistaminic drugs had been tried for the ulcer by dermatologists; however, the pathology was not resolved.
In physical examination, the upper lip, tongue, nose, and frontal region were found normal. The mandible and the soft tissue of the neck were not involved. Radiographic confirmation was done. Written consent was obtained from the patient, and the surgical approach was planned.
Under local anesthesia, an elliptic incision was performed, ulcer was debrided, and the orbicularis oris muscle was identified. Free edges of the muscle were dissected and sutured together in the midline. Mucosa and the vermillion line were connected meticulously. No complications were observed in the early postoperative period. At 10-month follow-up, the patient was satisfied with the result [Figure 2].
The incidence of craniofacial clefts is estimated between 1.4 and 4.9/100,000 live births.  Although two genders are being affected by craniofacial clefts, girls are more commonly affected. The pathology can develop due to large doses of radiation, infection, vitamin deficiencies, metabolic syndromes, and Vitamin A toxicity. [5-7]
In Tessier Classification, clefts 0-14 radiate around the orbital bone rims [Figure 3 and Table 1]. Tessier number 0 and number 3 are seen to be the most commonly reported cleft types in both genders in the literature. [8,9] Tessier number 30 cleft of the face is a rare congenital anomaly. The deformity occurs as insufficient fusion of first branchial arc or the failure of mesodermal penetration in midline.  Anomaly can range from a lower vermillion notching to the involvement of the teeth, tongue, mandible, and neck. [11-14] Tafreshi et al. reported a Tessier number 30 cleft case with congenital heart defects.  One patient was reported by Adetayo and Martin  with a Tessier number 30 cleft concomitant with levocardia, ventricular septal defect, a patent foramen ovale, double outlet right ventricle, intestinal malrotation, and bilateral undescended testicles. Vendramini-Pittoli et al. published two Tessier number 30 cleft cases. One of them was accompanied by a Tessier number 7 cleft, and the other one had developed together with Tessier number 4, 7, and 9 clefts.  Moreover, nasal anomalies and cleft lip and palate can also be seen with Tessier number 30 facial clefts. [17-19]
Midline cleft of the lower lip and mandible can also be a sign of a syndrome, such as the Robinow syndrome which presents itself with extremity, genitalia, costa and vertebra anomalies,  or the Richieri-Costa syndrome which involves limb defects. 
The prenatal diagnosis of the Tessier number 30 cleft can be made through ultrasonography at 15-18 weeks of the pregnancy. 
Reports about this pathology are rare in the literature.  Less than 80 cases have been reported since the first article by Couronne. [10,23] There are a few cases of craniofacial clefts reported from Turkey; [24-27] moreover, only two papers reporting cases from Turkey that are accompanied by a simple lower lip defect. [24,28] Due to this rarity, it can be said that there is no consensus on the management of this deformity. Therefore, we decided to prepare an algorithm for the treatment of the pathology.
Simple notching of the lower lip can be treated with wedge excision or Z-plasty.  Lip deformity and bifid tongue should be corrected as early as possible due to functional deficits. Duplication of the tongue can be corrected with wedge excision or Z-plasty. In addition, the tongue should be released from the floor of the mouth. 
Clefts of the mandible can be treated with one- or two-stage procedure depending on the range of the defect.  Most surgeons begin the procedure when the child is about 10-year-old to avoid tooth bud damage and use interosseous wires or plates.  On the other hand, Oostrom et al. report that a carefully planned osteosynthesis of the base of the mandible does not harm the buds and may provide better occlusion.  If the mandibular cleft is too wide, the bones should be stabilized at an early age to avoid obstructing eating and breathing.  A bone graft harvested from the costa, calvarium, or iliac crest can be inserted around the age of 10 in the second stage. [28,35,36]
One of the largest case series was reported by Freitas et al. with 17 patients aged from 1 month to 30 years.  Early surgery was performed in two patients with sleep apnea and impaired nutrition using distraction osteogenesis to achieve better tongue movement. They used Z-plasty for mucosal bands, muscle repair for the contraction, and straight closure of the skin in a few patients. Early bone grafting was recommended in this report to establish mandibular continuity.
The strap muscles of the neck are often replaced with dense scar tissue and cause flexion contracture bands and can be repaired with multiple Z-plasties.  Facial anomalies can extend to the widened interclavicular space, bifid or absent manubrium, presternal skin tags, cleft lip and palate, hemifacial microsomia, dermoid cyst of the face, extremity anomalies, and cardiac deformities.  Treatment choices for all these anomalies should be determined according to the type of the deformity.
The lower lip notching was corrected with wedge resection and three-layer closure in our patient. No complications occurred. Functional and esthetic results were good in the postoperative 10-month follow-up period. Tessier number 30 cleft should be kept in mind in lower lip anomalies.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
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Department of Plastic, Reconstructive and Aesthetic Surgery, Faculty of Medicine, Kirikkale University, Kirikkale, Turkey
Address for correspondence: Dr. Elif Sari, Department of Plastic, Reconstructive and Aesthetic Surgery, Kirikkale, Turkey.
Table 1: Clinical features of the Tessier craniofacial clefts Tessier classification Synonyms Tessier number 0 Median craniofacial dysraphia Centrofacial microsomia Frontonasal dysplasia Median cleft face syndrome Holoprosencephaly Tessier number 1 Type 3 nasoschisis nasal dysplasia Tessier number 2 - Tessier number 3 Oro-naso-ocular cleft Tessier number 4 Meloschisis Oro-ocular cleft Orofacial cleft Medial maxillary dysplasia Tessier number 5 Oculofacial cleft II Morian III cleft Lateral maxillary dysplasia Oro-ocular type 2 cleft Tessier number 6 Zygomatic-maxillary cleft Maxillozygomatic dysplasia Tessier number 7 Temporozygomatic facial cleft Craniofacial microsomia Hemifacial microsomia Otomandibular dysostosis First and second branchial arch syndrome Auriculo- branchiogenic dysplasia Hemignathia and microtia syndrome Oro-aural cleft Group B1 lateral otocephalic branchiogenic deformity Zygotemporal dysplasia Tessier number 8 Frontozygomatic cleft The equator of the Tessier craniofacial time zones Tessier number 9 Frontosphenoid dysplasia Tessier number - 10 Tessier number - 11 Tessier number - 12 Tessier number - 13 Tessier number Median craniofacial 14 dysraphia Tessier number Mandibular process cleft 30 Midline branchiogenic syndrome Intermandibular dysplasia Tessier classification Major clinical features of the pathology Tessier number 0 Deficiency of midline structures Developmental arrest range from hypoplasia of the nasomaxillary region and hypotelorism to a severe cyclopia, ethmocephaly, or cebocephaly False median cleft lip and absence of philtral columns Narrowed columella or totally absence of columella Lack of septal support Skeletal deficiencies range from separation between the upper central canines to absence of the premaxilla and cleft of the secondary palate Partial or total absence of nasal bones and septal cartilages Hypotelorism or cyclopia Encephalocele Excess of midline tissue True median cleft lip with broad philtral columns Duplication of the labial frenulum Bifid nose with a broad columella Laterally displaced alae and upper lateral cartilages Skeletal excess can be seen as a diastema between the upper central incisors Duplicate nasal spine Keel-shaped maxillary alveolus Anterior open bite Shortened central midface height Cartilaginous and bony nasal septum is thickened or duplicated Nasal bones and nasal process of the maxilla are broad, flattened, and displaced laterally Ethmoid and sphenoid sinuses may be enlarged, contributing to symmetric widening of the anterior cranial fossa and hypertelorism Displacement of the pterygoid plates away from the midline Tessier number 1 Soft tissue involvement It is similar to the common cleft lip Notching on soft triangle of the nose Short and broad columella The nasal tip and nasal septum deviate away from the cleft Malpositioned medial canthus, and telecanthus Skeletal involvement A keel-shaped maxilla and anterior open bite An alveolar cleft that passes between the central and lateral incisors The cleft may extend posteriorly as a complete cleft of the hard and soft palate The nasal bones are displaced and flattened Hypertelorism Tessier number 2 Soft tissue involvement Ala nasi is hypoplastic, lateral aspect of nose is flattened and dorsum is broad Cleft passes medially to the palpebral fissure and eyelid is not involved Lacrimal duct is usually intact Skeletal involvement Cleft begins between lateral incisor and canine, extends into apertura piriformis Hard or soft palate cleft can be accompanied with the pathology Cleft passes between nasal bone and frontal process of maxilla Orbital hypertelorism can be occurred Asymmetry of the sphenoid wings and anterior cranial base Tessier number 3 Soft tissue involvement Begins like number 1 and number 2 clefts Nose is shortened at the effected side Cleft extends between medial canthus and lacrimal punctum, lacrimal system is affected Recurrent ductal infection is common Medial canthus is displaced inferiorly, colobomas of lower eyelid are medial to inferior punctum Microphthalmia may occur, eye is malpositioned inferiorly and laterally Skeletal involvement This is an oro-naso-ocular cleft characterized by communication of these 3 cavities Cleft begins between lateral incisor and canine Anterior maxillary arch is flattened If the cleft is bilateral, skeletal disruption is significant Ethmoid and sphenoid sinuses are narrowed and anterior cranial base is displaced inferiorly Tessier number 4 Soft tissue involvement Begins lateral to the Cupid's bow, between tubercle and oral commissure Orbicularis aris muscle is located laterally to cleft and makes a mass-like appearance Cleft passes lateral to the ala Although nose is intact, alar base can be displaced superiorly Cleft extends to lower eyelid lateral to inferior lacrimal punctum Medial canthus and lacrimal system are intact Globe is normal, however microphthalmia or anophthalmia can be seen Skeletal involvement Alveolar cleft begins between lateral incisor and canine Extends lateral to piriform aperture and involves maxillary sinus Medial wall of maxillary sinus is intact Cleft passes through inferior orbital rim medial to infraorbital foramen Globe can prolapse inferiorly due to absence of medial aspect of inferior orbital wall Sphenoid body is asymmetric and pterygoid plates are displaced Anterior cranial base is unaffected Tessier number 5 Soft tissue involvement Begins just medial to oral commissure, courses along cheek lateral to alar rim Terminates in lateral half of lower eyelid Globe is normal but microphthalmia can be seen Skeletal involvement Alveolar cleft begins lateral to canines Extends lateral to infraorbital foramen Cleft terminates in lateral orbital rim and floor Cleft does not affect the inferior orbital fissure Maxillary sinus can be hypoplastic Vertical orbital dystopia can be seen Lateral orbital rim and wall may be thickened; greater sphenoid wing is affected but cranial base is normal Tessier number 6 Soft tissue involvement Can be seen as a hypoplastic soft tissue that begins from orbital commissure and extends to lateral lower eyelid Lateral palpebral fissure is pulled down, lateral canthus is displaced inferiorly Pathologies can cause severe ectropion of lower eyelid and an antimongoloid appearance Colobomas are located on lateral lower eyelid and mark the cephalic end of the cleft Skeletal involvement Cleft separates zygoma and maxilla via affecting the zygomaticomaxillary suture No alveolar cleft but shortened posterior maxilla can cause occlusal tilt Choanal atresia is common Cleft end in lateral third of the orbital rim and floor Connects to inferior orbital fissure Zygoma is hypoplastic but zygomatic arc is intact Anterior cranial fossa is narrow but sphenoid is normal Tessier number 7 Soft tissue involvement Cleft begins from oral commissure and extends to preauricular hairline Pathology can range from a mild broadening of oral commissure with a preauricular skin tag to a complete fissure extending toward the microtic ear Cleft does not extend beyond anterior border of masseter muscle Ipsilateral tongue, soft palate and muscles and mastication can be affected Parotid gland and duct can be absent Facial nerve weakness may occur External ear absence or microtia or only preauricular skin tag can be seen depending on the level of pathology Middle ear can also be affected Preauricular hair is usually absent in craniofacial microsomia patients Hairy skin between oral commissure and temporal region can be seen in Teacher Collins Syndrome The ipsilateral soft palate and tongue are often hypoplastic Skeletal involvement Skeletal cleft passes through pterygomaxillary junction Cleft is centered in the region of temporozygomatic suture according to Tessier Posterior maxilla and mandibular ramus are vertically hypoplastic and cause abnormal occlusal plane Coronoid process and condyle are often hypoplastic, contributing to a posterior open bite Zygomatic body is hypoplastic and displaced In severe pathology, zygomatic arch represents as a small stump In severe form, true orbital dystopia can occur Zygomatic process of temporal bone is normal Cranial base is asymmetric and glenoid fossa is malpositioned There can be a rudimentary medial and a lateral pterygoid plate Sphenoid bone is usually abnormal Tessier number 8 Soft tissue involvement Cleft extends between lateral canthus and temporal region Hair markers can be seen along a line between temporal area and lateral canthus True lateral commissure coloboma with absence of lateral canthus is seen Epibulbar dermoids are also presented Skeletal involvement Cleft is located at frontozygomatic suture The only support of lateral palpebral fissure is the greater wing of the sphenoid bone There is soft-tissue continuity of the orbit and temporal fossa Tessier number 9 Soft-tissue involvement Lateral third of the upper eyelid and eyebrow are abnormal Lateral canthus is distorted In severe pathology, microphthalmia is presented Superolateral bone defect causes lateral displacement of globe Cleft extends to temporoparietal hair-bearing scalp Temporal hairline is anteriorly displaced, and a temporal hair projection is often seen Nervus facialis palsy in forehead and upper eyelid is common Skeletal involvement Bone defect extends through superolateral aspect of the orbit, involving supraorbital rim and roof Upper part of greater wing of sphenoid, squamous portion of temporal bone, and surrounding parietal bones are distorted Posterolateral rotation of lateral orbital wall can be seen Pterygoid plates are hypoplastic There may be a reduction in anteroposterior dimension of anterior cranial fossa Tessier number Soft tissue involvement 10 Begins at the middle third of upper eyelid and eyebrow Lateral eyebrow may angulate temporally Palpebral fissure may be elongated with an amblyopic eye displaced inferolaterally In severe pathology, entire upper eyelid can be absent Colobomas can be seen Frontal hair projection may connect temporoparietal region to lateral brow Skeletal involvement Begins in the middle of upper orbital rim just lateral to supraorbital foramen A prominent mass can be seen in forehead due to encephalocele Orbit rotated lateroinferiorly In severe disease, hypertelorism can be seen Anterior cranial base is affected Tessier number Soft tissue involvement 11 Medial third of upper eyelid is affected and upper eyebrow is disrupted Pathology can involve a tongue-like projection of the frontal hairline Skeletal involvement A notch can be seen in the medial third of the supraorbital rim If cleft passes through ethmoid air cells, orbital hypertelorism can be seen Cranial base and sphenoid anatomy are normal Tessier number - Soft tissue involvement 12 Cleft lies medial to medial canthus There is a lateral displacement of the canthus that causes telecanthus There is no eyelid clefting Aplasia of medial eyebrow can occur Downward projection of paramedian frontal hairline possible, but forehead skin is normal Skeletal involvement Passes through frontal process of maxilla Transverse dimension of ethmoid cells are increased and orbital hypertelorism occurs Frontal and sphenoid sinuses are usually enlarged Cleft is lateral to olfactory groove, so cribriform plate is normal Encephaloceles have not been observed in this cleft Anterior and middle cranial fossae are widened Tessier number - Soft tissue involvement 13 There is typically a paramedian encephalocele located between nasal bone and frontal process of maxilla Upper eyelid and eyebrow are intact Medial end of eyebrow can be displaced inferiorly A V-shaped frontal hair line can be seen Skeletal involvement Anomaly in cribriform plate is characteristic in this cleft There is widening in olfactory groove Cribriform plate and ethmoid sinus are enlarged, thus hypertelorism can be seen A paramedian encephalocele can relocate the cribriform plate inferiorly leading to orbital dystopia Tessier number Soft tissue and skeletal tissue deficiency 14 Hypotelorism can be seen A spectrum of holoprosencephalic disorders such as cyclopia, ethmocephaly and cebocephaly may also be seen Cranium is typically microcephalic Cranial base components are absent which causes orbital fusion Soft tissue excess Hypertelorism can also be seen with number 14 cleft Frontonasal encephalocele or a midline frontal encephalocele can cause lateral displacement of the orbits Forebrain remains in a low position during embryologic development Glabella is flattened Periorbita, eyelids and eyebrow are normal Long midline projection of frontal hairline can be seen Skeletal tissue excess Caudal frontal bone is flattened While frontal sinus is nonpneumatized, sphenoid sinus is over-pneumatized Bifid crista galli and perpendicular plate can be seen Crista galli and ethmoid bone are widened and caudally displaced Cribriform plate is displaced inferiorly Relative shortening of middle cranial fossa is caused by rotation of sphenoid wings Harlequin eye deformity can be seen on X-ray Tessier number Soft tissue involvement 30 Pathology can range from a notch in the lower lip to an entire lower lip and chin cleft Anterior tongue can be bifid and attached to split mandible Ankyloglossia and total absence of tongue have also been reported Skeletal involvement Cleft is located between central incisors and extends to mandibular symphysis Anomaly is thought to be caused by failure of fusion of first branchial arch If lower branchial arches are affected, neck anomalies can be included in the pathology Hyoid bone is absent in most cases and thyroid cartilages are underformed Flexion contracture of neck due to atrophia of neck strap muscles
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|Title Annotation:||Original Article|
|Publication:||Turkish Journal of Plastic Surgery|
|Date:||Jan 1, 2018|
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