Tessier Number 30 Median Mandibular Cleft With Congenital Heart Anomalies in Qena,Egypt

Abstract

Median cleft deformities of the lower lip and mandible are very rare congenital anomalies. Our patient had median cleft of the lower lip, mandible, and the chin with tongue duplication, ankyloglossia, and cleft strap muscles with 2 neck contracture bands. This anomaly was associated with congenital heart disease transposition of great vessels, large ventricular septal defect, and severe pulmonary stenosis. Early repair was done at 6 months to improve feeding.

Keywords

Tessier no. 30 tongue duplication median mandibular cleft heart anomalies

Introduction

Median cleft of the lower lip and mandible is a very rare craniofacial anomaly. Couronné (1819) was the first author to report this abnormality. In 1976, Paul Tessier introduced the number system for craniofacial clefts, wherein the mandible midline cleft was notified as no. 30. They are considered as caudal extension of no. 14 (cranial) and no. 0 (facial cleft; Tessier, 1976). Oostrom et al., (1996) reviewed the literature and found a total of 65 reported cases, including 3 cases of their own. Few cases have been published since that time.

Principle features of median cleft deformities of the lower lip and mandible are as follows: (1) complete median cleft of the lower lip with extension of cleft may vary from a simple notch in the vermilion to a complete cleft involving the tongue, lower lip, chin, and extending up to the cervical region for varying distance; (2) notching of the mandible to complete cleft of the mandible allowing independent movements of the mandibular segments; (3) anomaly of the tongue including bifid tongue and tongue tie; and (4) associated features such as lack of hyoid bone, thyroid cartilage, and even manubrium sterni. Different malformations in other organs such as heart, limbs, and internal organs are also reported concomitant with Tessier 30 (Surendran and Varghese, 1991; Ladani et al., 2013). A review of the literature about some reported cases is presented in Tables 1 and 2.

Table 1.

Clinical Findings of Previously Reported Cases.

No	Author	Patient	Spectrum of Anomaly	Associated Features	Other Anomalies
1	Rao (2015)	11-month-old female	Complete midline cleft of lower lip, mandible, tongue, floor of mouth, ankyloglossia	Flexion neck contracture, hypoplastic hyoid bone	No
2	Tapadar and Tiwari (2015)	12-year-old female	Midline cleft of mandible and lower lip, complete duplication of the tongue	No	No
3	Vendramini-Pittoli Siulan et al., 2015	Report on 2 cases	Complete lower lip and mandibular cleft	No	Goldenhar syndrome and other facial clefts 4, 7, 9
4	Tafreshi et al., (2015)	12-hour-old male	Complete median cleft of mandible, lower lip, and tongue with ankyloglossia	No	Heart anomalies: large VSD, mild ASD, and small PDA
5	Leela et al., (2014)	28-year-old male	Median mandibular cleft (when presented)	No	Residual right palatal cleft
6	Ladani et al. (2013)	1-week-old male	Midline complete lower lip and extending up to hyoid region with a complete cleft of mandible (1.5 cm apart) and ankyloglossia	No	No
7	Adetayo et al., (2013)	1-day male	Mandibular cleft, bifid tongue, supernumerary teeth	No	Bilateral cleft lip and palate with associated cardiac, gastrointestinal, and genitourinal findings
8	Utpal, (2012)	13-year-old female	Median cleft of the lower lip, mandibular symphysis, severe ankyloglossia	Midline cervical web with neck contracture	No
9	Bhattacharyya et al., (2012)	5-month-old male	Midline cleft of the lower lip and mandible, complete duplication of the tongue	No	No
10	Mutaz, (2012)	8-year-old, female	Paramedian mandibular cleft	No	Goldenhar syndrome, Tessier 2/12
11	Halli et al., (2011)	6-month-old female	Midline incomplete cleft of the lower lip, complete median mandibular cleft, fissured tongue, and ankyloglossia	No	No
12	Freitas et al., (2010)	Survey of 17 cases	Different varieties	Different varieties	Different varieties
13	Steven et al., (2007)	1-month-old male	Paramedian cleft of the lower lip	No	Cleft palate, hemifacial microsomia left Tessier 7, dermoid cyst at the angle of left eye
14	Senan et al, (2007)	13-year-old male	Median cleft of the lower lip and the mandible, ankyloglossia	No	Midline neck swelling (foregut duplication cyst)
15	Roshani et al., (2004)	6-month-old male	Median cleft of the lower lip, ankyloglossia, and notching of the mandible		Ectopic salivary gland on the dorsum of the tongue (biopsy)
16	Ishii et al., (2002)	2-week-old male	Complete cleft of the lower lip and the mandible, bifid tongue, and ankyloglossia	Flexion contracture of the neck	No
17	Seyhan and Kýlýnr, (2002)	Two cases	1st case: only a small notch in the vermilion; 2nd case: a midline incomplete cleft of the lower lip, a sublingual abnormal frenulum, a complete cleft in the mandible	2nd case: a bifid sternum, presternal skin tag	2nd case: a heart anomaly (ventricular septal defect)
18	Almeida et al., (2002)	10-year-old female	Median mandibular cleft only, ankyloglossia	Agenesia of the lower incisors	Hand anomalies: large spaces between fingers
19	Son et al., (2001)	2-day-old-male	Median cleft of the lower lip and mandible associated with bifid tongue and ankyloglossia	No	Heart anomalies: double inlet single ventricle
20	Aygit et al., (1999)	13-day-old female	Median mandibular cleft (by CT scan), normal soft tissue outside	No	Tessier 7 cleft
21	Armstrong and Waterhouse, (1996)	1-week-old female	Incomplete median cleft of lower lip associated with a furrow extending down the chin. Large rounded mass over the chin, with a smaller area of red granulation tissue at the lower pole and ankyloglossia. Midline mandibular cleft	Bifid uvula	No
22	Chidzonga et al., (1996)	3-month male	Complete median cleft of the lower lip: complete median cleft of the mandible (1.5 cm apart), and bifid tongue with ankyloglossia	No	No
23	Oostrom et al., (1996)	Report of 3 cases	Case 1: notch in the vermilion. Case 2: complete cleft of lower lip and mandible with ankyloglossia. Case 3: complete cleft of the lower lip and mandible with ankyloglossia. With neck contracture	Case 2: sever heart anomalies and agenesis of corpus callosum	No
24	Arshad, 1995	3 month female	Incomplete midline cleft of lower lip	No	No
25	Park and Takushima 1993	4 hours old male	Complete cleft of the lower lip and the mandible which extended to the manubrium, ankyloglossia	No	Heart anomalies: single atrium, double-outlet right ventricle
26	Surendran and Varghese, (1991)	21 days, female	Midline cleft of lower lip with cleft of the mandible	No	Midline dermoid in the neck
27	Millard et al., (1979)	8 years male	Median mandibular cleft, previous repaired lower lip and chin cleft	No	No
28	Millard et al., (1971)	22 days male	Midline cleft of the lower lip and the chin, ankyloglossia, and median mandibular cleft	No	No
29	Fujino et al., (1970)	17 days female	Midline cleft of lower lip mandible and bifid tongue with ankyloglossia	No	No

Abbreviations: ASD, atrial septal defect; CT, computed tomography; PDA, patent ductus arteriosus; VSD, ventricular septal defect.

Table 2.

Surgical Treatment and Results of Previous Reported Cases.

No	Author	Surgical Repair and Result
1	Rao (2015)	At 11 months, multi-Z-plasty for the tongue. Stainless steel wires for the mandibular cleft. Zigzag release for neck contracture At 3 years, scar revision of neck and lower lip. Good healing, with normal dentition and speech
2	Tapadar and Tiwari, (2015)	Staged repair. 1st stage: lower lip repair in the midline, then mandibular growth assessment finally, secondary repair for lip for cosmetic appearance, and repair of duplicated tongue
3	Vendramini-Pittoli Siulan et al., (2015)	Different techniques in the survey
4	Tafreshi et al., (2015)	Not operated because of the instability of the general condition
5	Leela et al., (2014)	Earlier repair to lower lip cleft at 1 month and 16 years (not by the author), then scar revision at 28 years. Mandibular cleft repair at 28 years with iliac bone graft fixed with titanium plates (by the author)
6	Ladani et al. (2013)	Early repair to soft and hard tissue simultaneously: at 6-month old, mandibular cleft with 1-0 Vicryl sutures, lower lip, and chin with Z-plasty, release of ankyloglossia
7	Adetayo et al., (2013)	Lip and tongue repair at 3 months and palate repair later (10 months)
8	Utpal, (2012)	Single-stage repair, release of ankyloglossia, fixation of the mandibular cleft with titanium miniplate, repair of lower lip, correction of neck contracture with multiple Z-plasties
9	Bhattacharyya et al., (2012)	First-stage repair of lower lip defect by multiple Z-plasty and repair of mandibular defect by bone graft (from a small piece of mandible present at site of the cleft). Second stage: lip revision and tongue repair
10	Mutaz, (2012)	Staged repair with rib costochondral graft and mandibular distraction
11	Halli et al., (2011)	Lip repaired by modified V-plasty, release of ankyloglossia by Z-plasty, mandibular cleft at later date (bone graft)
12	Freitas et al., (2010)
13	Steven et al., (2007)	At 6 months, repair of cleft palate with a Sommerlad style, cleft lip repaired in a straight line with Noordhoff style vermillion flaps. Excision of the dermoid cyst and cartilaginous skin tags. Repair of macrostomia (Z-plasty) at a later stage, observation of mandibular anomaly
14	Senan et al, (2007)	Release of ankyloglossia at 6 months and lip and chin repair at 6 years, then bone graft for mandibular cleft at 9 years
15	Roshani et al., (2004)	Excision of mass on dorsum of tongue, release of ankyloglossia and lip from the alveolus followed by lip repair was done. No bony work was done since the mandible was only notched
16	Ishii et al., (2002)	At 5 months of age, lower lip repair (Z-plasty), release of tongue, repair of floor of mouth, and vestibuloplasty. At 6 years of age, mandibular fixation by plates followed by definitive bone graft from iliac crest 1 year later. At 9 years, all plates were removed. At 13 years, augmentation of the mid-mandibular region by onlay iliac bone graft was done. At 16 years, the contracture in submental region was excised using a full thickness skin graft
17	Seyhan and Kýlýnr, (2002)	1st case: by Z-plasty. 2nd case: Z-plasty for release of the frenulum. Lower lip cleft by simple V-excision. Mandibular cleft by interosseous stainless steel wire
18	Almeida et al., (2002)	Z-plasty for tongue, a frenectomy of the lingual frenulum, an autogenous iliac crest bone graft, and fixed by plates and screws
19	Son et al., (2001)	At 3 months, 1-stage correction of the clefts of the lip, tongue, and mandible (repair of lower lip cleft using W-plasty. mandibular cleft by manual reduction and internal fixation with plates)
20	Aygit et al., (1999)	Repair of Tessier 7 (W-plasty) and mandibular cleft resolved at 1-year old
21	Armstrong and Waterhouse (1996)	At 5 weeks repair of lower lip and tongue tie by Z-plasty. At 4 years, repair of mandibular cleft by iliac crest bone graft and fixed by titanium miniplate (removed after 6 months). At 7 years, osseointegrated implant for missed teeth
22	Chidzonga et al., (1996)	At 1 year, repair of cleft lip (V-plasty) and tongue and release of ankyloglossia. At 1.7 years, mandibular repair (rib graft fixed with stainless steel wires and acrylic splint with circummandibular wiring [cmw]). A diastema is still present between the deciduous incisors which tipped mesially but the situation is functionally and aesthetically satisfactory
23	Oostrom et al., (1996)	Case 1: Z-plasty for vermilion notch, Case 2: died from heart failure: Case 3: z plasty for cleft lower lip, release of ankyloglossia, and neck contracture. Mandibular cleft by absorbable sutures
24	Arshad 1995	Repair by wedge excision and anatomical repair
25	Park and Takushima (1993)	No. Because he died of heart failure at the age of 15 days
26	Surendran and Varghese (1991)	1st stage: repair of midline swelling (dermoid); 2nd stage: repair of cleft lower lip and soft tissue with tongue release by Z-plasty. Later planning for mandibular defect repair at 12 years
27	Millard et al., (1979)	Autogenous rib graft fixed by wires and vitallium splint fixed by circummandibular wires
28	Millard et al., (1971)	Z-plasty for lower lip, chin, and tongue release, then bone graft for mandibular cleft
29	Fujino et al., (1970)	Tongue release, then lower lip repair by Z-plasty, then bone grafting for mandibular defect later

Case Report

The patient was referred to our department from pediatrics department when he was 5 days old. His mother complaints of facial deformity, difficult feeding (inability to suckle), and sometimes choking. He was the first baby. There was no admission to neonatal intensive care unit. There was no exposure of the mother to radiation and teratogens during pregnancy. The baby was born by normal vaginal delivery at full term. There was no consanguinity between the parents. The pediatrician had examined the baby before and he advised the parents to do echocardiography to reveal any congenital heart anomalies. The echocardiography revealed transposition of great vessels (TGAs), large ventricular septal defect (VSD), and severe pulmonary stenosis (PS).

On clinical examination, there were median cleft of the lower lip, full thickness up to the chin with duplication of the tongue that was stuck to the cleft margins of the lower lip and the mandible, and cleft strap muscles with 2 neck contracture bands (Figure 1). Otolaryngologist consultation showed patent airway, with no stridor or airway compromise, but with only cleft strap muscles. Endoscopic examination shows normal larynx and trachea (no hypoplasia or cleft) with right upper tracheal diverticulum and small anterior laryngocele were detected by fiber-optic endoscope examination. Radiologically, a noncontrast multislice computed tomography (NCMS-CT) showed complete median cleft of the lower jaw (mandible) and complete cleft of the tongue (duplication), associated with cleft of the skin and soft tissues of straps muscles seen at the upper part of the neck, normal hyoid bone. It also showed right upper tracheal diverticulum, small anterior laryngocele, normal thyroid cartilage, and normal upper facial bones.

The patient was 6 months old and weighed 6.5 kg when he had been referred to our department to correct the facial deformity. General anesthesia was carried out with nasoendotracheal intubation (Figure 2). Firstly, the duplicated tongue was released from the lower lip, floor of the mouth, and margins of the cleft, then a longitudinal excision of the midline cleft was made full thickness up to the floor of the mouth by 15-gauge blade, and suturing with 3-0 Vicryl to approximate the 2 halves of the tongue (Figure 3). For repairing the mandibular cleft, an incision was made along the margins of the cleft on each side full thickness including skin, muscle, and mucosa. There was continuity of skin up to the neck with no definite chin in the midline, so an incision was made to separate the lower lip from the neck, then dissection was done to separate the skin, muscle, and mucosa; through this incision, the 2 mandibular segmented were exposed. Mucoperiostium was elevated from the mandibular segment. The 2 ends of the cleft were identified on both sides. Both mandibular segments were fixed at the upper border, and the inferior border of the mandible was sutured with 2-0 Vicryl on both sides. I used strong suture instead of wire or plate because it may extend over the mandibular stump and damage tooth bud (Figure 4). Repair of the cleft lower lip was done, first we started with the mucosa inside and continuing suturing the mucoperiostium to cover the mandible with 4-0 Vicryl sutures, then repair of the muscle with 4-0 Vicryl sutures, and lastly skin repair with 5-0 Vicryl sutures. Finally, repair of the clefted strap muscles was made by release of the 2 contracted neck band, then muscle approximation with 4-0 Vicryl sutures at the midline, and skin closure by Z-plasty reconstruction to avoid neck contractures (Figure 5). The baby was admitted by the anesthethiologist to the intensive care unit postoperatively, as oxygen saturation was below 80, and then the baby was intubated with oxygen flowmeter. Follow-up showed good healing clinically (Figure 6). Good bony healing was revealed by CT.

Discussion

Typical craniofacial clefts (cleft lip and palate) are common, but atypical clefts are rare, the incidence being estimated at 1.4 to 4.9 per 100 000 live births (Resnick and Kawamoto, 1990; Sesenna et al., 2012). Median cleft of the lower lip and mandible is a rare congenital anomaly (Hou et al., 2011). Tessier (1979) described an anatomical classification system, in which the number is assigned to each craniofacial cleft on the basis of its position relative to the sagittal midline and the orbit. The first report of midline mandibular cleft was reported by Couronné (1819) (Armstrong and Waterhouse, 1996; Oostrom et al., 1996). Since then, 66 cases have been reported worldwide till 2007 (Senan et al., 2007).

Cleft of the lower lip and mandible is a developmental anomaly of the first branchial arches. Several hypotheses concerning its pathogenesis have been proposed in the literature. Most authors consider it to be due to a failure of fusion of the first pair of branchial arches or a failure of mesodermal penetration into the midline of mandibular part of the first branchial arch (Resnick and Kawamoto, 1990). Morton and Jordan (1935) feel that the second theory may explain the absence of hyoid, thyroid cartilage, strap muscles, and manubrium in severe varieties. In 1996, Oostrom et al proposed that there is only 1 branchial arch during early embryonic periods (7th week) into which 2 mandibular processes grow with a groove in midline. Hypoplasia of the mandibular processes during early embryonic period will lead to the severest cleft of the mandible extending to the neck. During the late embryonic period, less severe median clefts will develop (Ishii et al., 2002).

Thus, the deformity can range from minor to severe variety in various combinations. In minor forms, only the lower lip is clefted. Frequently, the cleft extends to the mandibular symphysis. The tongue may be bifid and attached to the cleft alveolar margin, at times to the cleft uvula and ankyloglossia (Tessier, 1976). In severe forms, epiglottis or hyoid may be absent; thyroid cartilage may be underdeveloped (Kawamoto, 1990). There may be a midline cervical cord. Strap muscles may be atrophic and represented by fibrous tissue causing bulging of the neck during straining (Ishii et al., 2002). Monroe (1966) and Ishii et al (2002) reported flexion contracture of the neck and that there may be a midline dermoid in the neck (Monroe, 1966 and Ishii et al., 2002). The clavicles may be widely spaced and manubrium sterni may be bifid or absent (Morton and Jordan, 1935). There may be presternal tags (Son et al., 2001). Seyhan and Kylyur (2001) reported a case of bifid sternum associated with subcutaneous mass and ventriculoseptal defect.

There may be other associated facial anomalies such as cleft of upper lip, cleft palate, Pierre-Robin anomaly, mucus pits of lower lip, hemifacial microsomia, and dermoid cyst of nose or chin, eye, and ear deformities (Richieri-Costa and Pereira, 1993). Congenital anomalies of heart (single ventricle, TGAs), hand (syndactyly, brachydactyly), and foot (clubfoot, ectrodactyly; Senan et al., 2007 and Sesenna et al., 2012) have also been reported. Although no genetic abnormality has been described. Subramani et al., (2000) have reported a mother having cleft tongue with ankyloglossia, linear band in the midline of palate, and hyperpigmented patch over dorsum of tongue and her daughter with cleft tongue, ankyloglossia, cleft of soft palate, notched vermilion of upper lip, and ectodermal dysplasia of face.

In our case, there was median cleft of the lower lip and chin with duplication of the tongue, ankyloglossia, and cleft strap muscles with 2 contracture neck bands associated with congenital heart disease: TGA, large VSD, and severe PS.

Similar cases with heart anomalies were reported. In 2001, Son et al. (2001) also presented a case with median cleft of the lower lip and mandible concomitant with double-inlet single ventricle. In 2013, Adetayo and Martin (2013) reported a 37-week-old infant with a Tessier 30 cleft concomitant with levocardia, perimembranous moderate-to-large VSD, patent foramen ovale, double-outlet right ventricle, intestinal malrotation, and bilateral undescended testicles. In 2015, Tafreshi et al. (2015) reported the first Iranian case of Tessier 30 of 12-hour-old boy with large VSD, a mild atrial septal defect, and a small patent ductus arteriosus. In 2015, Tapadar and Tiwari (2015) reported a case of Tessier 30 with complete tongue duplication up to the posterior part.

The rarity and variation in severity of this condition are responsible for the lack of consensus on the mode of management and timing of surgical procedures (Rantar, 1984). Millard et al., (1979) reported initial correction of a midline cleft of the lower lip at 6 months followed by closure of the mandibular cleft at 8 years of age because early closure of the mandibular defect could cause damage to the tooth buds. Sherman and Goulian (1980) treated median mandibular bone defects at 20 months of age successfully .On the other hand, Oostrom et al. (1996) said that a careful osteosynthesis of the base of the mandible does not harm these buds and may cause a better maxillomandibular relation in occlusion. They also stated that this timing should depend on the severity of the cleft.

Armstrong and Waterhouse (1996) treated lower lip cleft deformity at the age of 5 months and mandibular deformity with bone graft to correct occlusal cross bite at the age to 4 years. Chidzonga et al., (1996) followed the same protocol and treated soft tissue deformity at 1 year and mandibular segment fixed with bone graft to correct occlusal cross bite at the age of 19 months. Oostrom et al., (1996) had approximated the 2 ends of mandible with absorbable sutures in order to facilitate closure of skin at 3 month of age and planned to close the mandibular gap when the child was about 10 years old in order to preserve the growth of mandible and teeth. Ibrahim et al. (2002) treated a case of median mandibular cleft with large bony defect, with 2 hamartomatous masses on both sides of clefts. A pedicled cartilaginous mass from hamartoma was used to bridge a bone defect in primary repair at the age of 3 months.

In our case, the patient was 5 months old and weighed 5.5 kg when he was operated. The duplicated tongue was repaired firstly by release from the lower lip, floor of the mouth, and margins of the cleft, then a longitudinal excision of the midline cleft up to the floor of the mouth, and then suturing with 3-0 Vicryl for approximation. The lower lip was repaired along the margins of the cleft on each side including skin, muscle, and mucosa repair with absorbable Vicryl sutures. Then, both mandibular segments were fixed after mucoperiostium elevation at the upper border and the inferior border of the mandible with 2-0 Vicryl sutures on both sides. I used strong suture instead of wire or plate because it may extend over the mandibular stump and damage tooth bud. Finally, the repair of the clefted strap muscles was made by release of the contracted bands and muscle approximation with 4-0 Vicryl sutures at the midline, the skin was closed by Z-plasty reconstruction to avoid neck contractures. This procedure was same as previously described by Ladnai et al. (2013) for early repair of soft and hard tissues. Follow-up showed good healing clinically and good bony union by CT scan.

Conclusion

Median mandibular cleft are very rare congenital anomalies. Our patient had median cleft of the lower lip and the chin with bifid tongue, ankyloglossia, and cleft strap muscles associated with congenital heart disease. Early repair was recommended.

Footnotes

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, authorship, and/or publication of this article.

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