Facial Soft Tissues

From a clinical perspective, the soft-tissue deficiencies associated with HFM can be considered to affect four anatomic regions of the head and neck: 1) the external ear; 2) the eyelid–adnexal structures; 3) the preauricular–cheek–lip soft tissues; and 4) the temporal fossa (Figs. 28-2 and 28-3).^{19,51,85,98,101,106,130,133,129,228,251} The soft tissues within each region that may be deficient and dysmorphic include the bulk of the cutaneous and subcutaneous tissue, the volume of fat, the muscles of mastication and facial expression, the cranial nerves, and the parotid and submandibular glands.

According to the research of Kane and colleagues, in patients with HFM, the extent of hypoplasia of specific muscles of mastication frequently predicts the extent of dysplasia of the osseous origin and insertion of those muscles.⁹⁸ If the temporalis muscle is hypoplastic, a deficiency of the coronoid process will be present. When the masseter muscle is hypoplastic, the gonial region of the mandible will also be deficient. When the lateral pterygoid muscle is deficient, the condylar head is deficient or absent.

Preauricular–cheek region soft-tissue thickness and skin surface area are generally related to the skeletal deficiencies. Skin tags are small vestigial rests of epithelial tissue that are generally found at the cleft between the first and second arches. Skin tags are frequently associated with small cartilaginous remnants that are found within the subcutaneous tissue. Sinus tracts may form inclusion cysts or result in infection if they are obstructed. Macrostomia is a clefting or failure of fusion of the maxillary and mandibular processes and of the first branchial arch, respectively. This results in a cleft of the orbicularis oris muscle, the overlying skin, and the underlying mucosa directly through the oral commissure. Anophthalmia or microphthalmia may also occur. Colobomata of the iris or eyelids with the absence of the eyelashes is a frequent finding. Ptosis of the upper eyelid (i.e., as a result of levator palpebral muscle function) with narrowing of the vertical palpebral fissure is often seen. Deficiencies of the lateral canthi with a decreased horizontal fissure are common.

Epibulbar dermoid cysts are found in a significant percentage of patients; they appear as solid yellowish or pinkish-white ovoid masses that vary in size from that of a pinhead to 8 to 10 mm in diameter. The cysts occur most often at the inferotemporal quadrant of the limbus. The surface is usually smooth, and it frequently has fine hairs. These cysts can occur at any location on the globe or in the orbit, and they may be movable or fixed to the dermis. Unilateral epibulbar dermoid cysts are seen in a high percentage of patients, and multiple lesions may occur in each eye. Occasionally, vision is impaired by encroachment on the pupillary axis or by lipid infiltration of the cornea.

Abnormalities of the external ear are a consistent finding and range from anotia to a mildly dysmorphic ear. Farkas and James were unable to demonstrate a direct relationship between the degree of microtia and the extent of skeletal deformity.⁵¹

The External Auditory Canal, the Middle- and Inner-Ear Structures, and Audiologic Findings

A narrow external auditory canal is frequently found in patients with mild external ear deformity, whereas atretic canals are expected in more severe cases.17,18,223,280 At times, a small external ear with normal middle-ear architecture is seen. Audiometry will delineate the nature of the hearing loss, which likely includes conductive and, less frequently, sensorineural loss in 15% of patients. Hypoplasia or agenesis of the ossicles may occur. In a comprehensive study, Caldarelli and colleagues used air and bone conduction audiometry and temporal bone tomography to evaluate 57 patients with hemifacial microsomia.^17,18 The authors were unable to correlate the degree of auricular (i.e., external ear) deformity with hearing function. Focused temporal bone CT scans offer the best documentation of the middle-ear structures. Assessment of the unaffected and presumed normal ear is an essential part of the evaluation.

Maxillomandibular Region

A variable degree of hypoplasia of the skeletal structures within the first and second branchial arches will occur. As a result, the anteroposterior, transverse, and vertical dimensions of the face are diminished on the affected side, with secondary deformities on the contralateral side. This is especially true in the maxillomandibular region (see Figs. 28-2 and 28-3).¹³⁴

Cranio–Orbito–Zygomatic Region

A variable degree of hypoplasia of the zygomatic complex is a frequent finding. The consequences of zygomatic hypoplasia may be seen clinically as orbital dystopia; maxillary hypoplasia; deficiency of the glenoid fossa; and deficiency of the squamous aspect of the temporal bone. This is best visualized with a three-dimensional reconstruction of a craniofacial CT scan. As a result of the asymmetric nature of the deformity, identifying consistent reproducible landmarks is difficult in the upper face. This makes a quantitative assessment of the skeletal malformation difficult and imprecise.

1. In perspective, even if a small degree of asymmetric jaw growth occurs during childhood in the patient with HFM, significant progressive dysfunction (i.e., in the areas of chewing, breathing, speech, lip control, or swallowing) that would justify a first-stage mandibular procedure is not generally seen.

2. The effectiveness of a first-stage mandibular procedure during the mixed dentition as a definitive long-term maxillofacial reconstruction has not been documented. Not only would a successful initial lengthening and derotation of the mandible be required, but the fabrication and then the periodic modification of an effective removable acrylic splint to create and maintain an ipsilateral posterior open bite (to allow for the supra-eruption of the maxillary posterior teeth) would be needed. The necessity of constant clinician monitoring and splint modification in conjunction with ongoing orthodontic treatment and rigorous patient compliance would be essential. This requires an experienced and dedicated orthodontist, a persistent surgical coordinator, and a zealous patient and family. All of the treating clinicians and the family must be in close geographic proximity and without socioeconomic barriers to treatment. Unfortunately, this is not a combination of events that is readily achievable.

3. Unfortunately, the costochondral graft construction of the neocondyle–ascending ramus required for the treatment of HFM Type IIB and III mandibular deformities carried out during the mixed dentition will result in a significant percentage of poorly growing mandibles (i.e., overgrowth or undergrowth). At minimum, a second definitive orthognathic reconstruction will be required (see the section to follow in this chapter about costochondral grafting).

4. Researchers have documented that, when a distraction osteogenesis (DO) approach is used as a first-stage mandibular procedure for HFM during the mixed dentition, symmetric lower jaw growth does not occur. The resulting residual mandibular asymmetry and deformity will, at minimum, require later definitive orthognathic reconstruction. In addition, the completion of a mandibular ramus–body osteotomy and DO during the mixed dentition is documented to result in the destruction of ipsilateral developing permanent molars in a significant percentage of children who have undergone such an operation.

Zygomatic and Orbital Reconstruction

The benefits of thoughtfully timed and meticulously executed orbito–zygomatic reconstruction for the patient with HFM are often underappreciated.209–213 When upper face (i.e., orbito–zygomatic) reconstruction is needed, it is carried out through a coronal scalp incision. Correction of the upper face skeletal deformity may require the intracranial disassembly and reconstruction of the dysmorphic ipsilateral anterior cranial vault; the squamous portion of the temporal bone; and the lateral orbit regions with complete construction of the zygomatic complex, including the glenoid fossa. The use of full- and split-thickness cranial bone grafts is required (Fig. 28-8).

This author believes that the reconstruction of the cranial vault, malar, and orbital deficiencies before the age of 7 years is not indicated unless functional disability warrants it.²¹³ After the patient reaches this age, cranio–orbitozygomatic skeletal development is nearly mature. When indicated, an adult-sized anterior cranial vault, orbit, and zygomatic complex may be constructed and matched with the contralateral normal side with little concern about how future growth will alter the initial results achieved. Donor site skull reconstruction is also simpler after the age of 7 years than it is in younger children, because the presence of bicortical cranial bone will facilitate the splitting of the inner and outer tables for efficient reconstruction. Artificial bone material may also be used to reconstruct the graft donor site of the posterior skull (see Chapter 27).

Other surgeons have recommended variations in the methods and timing of the reconstruction of the upper face skeletal anomalies associated with HFM.³¹ Posnick and colleagues confirmed that the construction of the complete zygomatic complex with full-thickness autogenous cranial bone maintains volume and shape better than onlay grafts, which have been universally disappointing. Over time, onlay autogenous bone grafts that have been placed in the craniofacial region (e.g., supraorbital ridge, zygoma, anterior maxilla, angle of mandible, chin) from all tried donor sites (e.g., skull, hip, rib) have demonstrated significant and unpredictable resorption. Furthermore, no commensurate growth (i.e., volume expansion) of the graft with the underlying bone (e.g., the zygoma) should be expected. Some feel that, even if the onlay graft partially resorbs, at least something has been gained; however, this philosophy should be abandoned, because surface irregularities result in secondary deformities that further detract from the baseline malformation.

Maxillomandibular Reconstruction

An essential consideration when choosing the timing and techniques for maxillomandibular reconstruction in a patient with HFM is an understanding of the presenting TMJ–mandibular anatomy.2,6,7,20,25,30–32,43,46,59,96,97,180,209–212,226 The classification of the TMJ–mandibular malformation, which was described earlier in this chapter, is clinically relevant and facilitates communication.^94,157 In the patient with HFM with either a Type I or Type IIA malformation, the basic maxillomandibular skeletal asymmetry and dysmorphology that requires reconstruction include the following: 1) degrees of altered facial height (i.e., decreased posterior facial height on the ipsilateral side); 2) diminished horizontal projection (i.e., deficiency more prominent on the ipsilateral side); and 3) decreased facial width (i.e. deficiency on the ipsilateral side). These malformations frequently result in canting (i.e., roll orientation) of the pyriform apertures, the maxilla, and the gonial angles; the shifting of the maxillary, mandibular, and chin midlines off of the facial midline (i.e. yaw orientation); clockwise rotation of the occlusal plane (i.e., pitch orientation); and an asymmetric class II malocclusion often with anterior open-bite malocclusion. According to the classification described, a patient who presents with a Type IIB malformation requires the construction of a neocondyle. For the individual with a Type III malformation in addition to this, there is a need for the construction of a neo–glenoid fossa.

The most favorable long-term function (i.e., speech, swallowing, chewing, breathing) and enhanced facial aesthetics are generally achieved in patients who undergo definitive reconstructive surgery of the maxilla, mandible, and chin regions at any time after jaw maturity (i.e., at 13 to 15 years of age for the HFM patient); this should be carried out in combination with effective orthodontic treatment after the permanent teeth have erupted and before the patient graduates from high school. As with other dentofacial deformities, extractions may be required to orthodontically relieve dental root crowding and to normalize the inclination of the anterior teeth in preparation for the surgical repositioning of the jaws.

Type I and IIA mandibular malformations are best reconstructed after all of the permanent teeth have erupted and orthodontic goals have been reached (see Fig. 28-4, A, B, and C). Surgical objectives can be met by making use of sagittal split ramus osteotomies of the mandible in combination with Le Fort I osteotomy (often in segments) and osseous genioplasty. This combination represents standard techniques, and it does not require bone grafts. The success of reconstruction is dependent on approximating mirror-image symmetry and Euclidian proportions of the skeleton through orthognathic procedures.

For Type IIB mandibular malformations, costochondral graft reconstruction of the deficient condyle ascending ramus at the time of skeletal maturity remains this surgeon’s preferred approach in most cases (see Fig. 28-4, D), despite its limitations (see the section to follow concerning condylar reconstruction with the use of costochondral graft). A sagittal split ramus osteotomy is completed on the contralateral side to derotate the distal mandible. This is combined with a Le Fort I osteotomy (often in segments) and an osseous genioplasty.

A ramus osteotomy of the contralateral side of the mandible is completed first to control repositioning of the distal mandible. The distal mandible is then secured to the maxilla via intermaxillary fixation through a custom designed acrylic splint to create the preferred reorientation of the lower jaw (see Chapter 14). It may be necessary to resect the ipsilateral coronoid process before repositioning the distal mandible. The contralateral ramus osteotomy is then rigidly fixed with bicortical screws. The ipsilateral proximal mandible is reconstructed with the autogenous costochondral graft. Harvesting the rib graft from the contralateral chest wall provides the best contour for mandibular reconstruction. The fixation of the rib graft to the native distal mandible is accomplished with a titanium miniplate and 2.0-mm or 2.3-mm screws. The fixation plate extends from the graft forward along the inferior border of the body of the mandible (fig. 28-15). It is generally necessary to recontour (i.e., with a bur on a rotary drill) the outer cortex of the distal mandible before onlaying the graft. Graft placement and fixation is often carried out through an extraoral Risdon neck incision. The avoidance of intraoral incisions on the ipsilateral mandibular ramus region during this procedure may minimize the incidence of infection. The effective seating of the neocondyle in the glenoid fossa is a critical step for successful reconstruction. In some patients, the extent of associated soft-tissue and skeletal deficiency will favor use of a vascularized fibula composite flap over the use of a costochondral graft.

The Type III glenoid fossa–mandibular malformation requires the surgical construction of the congenitally missing parts (see Fig. 28-4, E). The mandibular reconstruction is generally carried out as previously described for the Type IIB deformity. When the glenoid fossa requires construction (i.e., with a Type III malformation), so will the zygomatic complex. The glenoid fossa–zygoma and orbital reconstruction are best carried out as a separate operation when the patient is at least 7 years old (see the previous section about orbito–zygomatic reconstruction) and before mandibular reconstruction. With regard to the mandibular reconstruction, the deficiency of both the condyle-ascending ramus and the associated soft tissues may benefit from a vascularized fibula composite flap rather than a costochondral graft.

Personal Perspective Concerning First-Stage Mandibular Reconstruction during Childhood

The maxillofacial malformations in patients with HFM may be severe enough that, for airway or psychosocial reasons, the clinician feels compelled to recommend first-stage mandibular reconstruction during the mixed detention (i.e., between the ages of 7 and 11 years). Reported options for first-stage mandibular reconstruction in patients with HFM generally include 1) a ramus/body osteotomy followed by DO carried out over time for Type I and Type IIA malformations or 2) costochondral grafting with the immediate construction of a neocondyle ascending ramus for Type IIB and Type III malformations. In either case, derotation of the mandible to position the chin in the facial midline with the creation of an ipsilateral posterior open bite is usually considered the objective. The reconstructive option selected (i.e., costochondral graft versus DO) should be based on the presenting malformation and then on the following: 1) the three-dimensional morphologic results believed to be achievable; 2) the anticipated ongoing growth potential after surgery; 3) the differences in perioperative morbidity among the techniques; and 4) the overall burden of treatment to the patient, the family, the clinicians, and the health care system.209–212

Interestingly, most published reports of first-stage mandibular reconstruction in patients with HFM do not fully clarify the details of the presenting dysmorphology of the native glenoid fossa and condylar components before surgery (Figure 28-9). As stated, for DO to be effective, a functional glenoid fossa and an adequate condyle must be present (i.e., in the presence of Type I or Type IIA malformation). Unfortunately, a high incidence of bony and fibrous ankylosis has been reported in conjunction with mandibular DO in HFM.⁴⁹ In addition, the consistent occurance of “undergrowth” after mandibular DO reconstruction carried out during the mixed dentition was confirmed by Meazzini and colleagues.¹⁴³ Those authors conducted a comparison study of long-term follow up until the completion of facial growth of two homogenous samples of children with HFM. The experimental group was treated with mandibular DO during the deciduous or early mixed dentition in an attempt to correct the mandibular deformity. The control group was not subjected to any treatment until adulthood. The experimental group included children (n = 14) who underwent mandibular ramus osteotomies with DO (mean age, 5.9 years) with a mean follow up of 11.2 years. With the use of quantitative measurements on serial panorex radiographs, the DO group was compared with the control group (n = 8). The study results document that facial proportions in patients with HFM are maintained throughout growth when no treatment is undertaken. Unfortunately, after ramus osteotomies with DO, the mandibular disproportions returned to their original level of asymmetry during growth. The authors concluded the following: 1) HFM does not progress with regard to the degree of facial asymmetry or deformity when the patient is left to mature naturally and 2) early intervention with mandibular ramus osteotomies and DO does not effectively reduce long-term facial asymmetry in the patient with HFM.

Ongoing problems when choosing the costochondral graft option to be carried out during the mixed dentition for the Type IIB or III malformation include overgrowth, undergrowth, ankylosis, and resorption of the mandible (Fig. 28-10). The overgrowth concerns are diminished but not eliminated when the costochondral graft procedure is carried out in a child who is in the late mixed dentition stage (i.e., 9 to 11 years old) and in whom only a minimum amount of cartilage (i.e., ≈2 mm) is left at the articulating surface of the graft (see section to follow concerning condylar reconstruction with costochondral graft).

If a first-stage mandibular procedure is carried out during the mixed dentition for the Type IIB or III malformation, then the need for definitive mandibular reconstruction in conjunction with a Le Fort I maxillary osteotomy and an osseous genioplasty at the time of skeletal maturity should be anticipated.3,16,28,27 The idea of carrying out a mixed dentition mandibular reconstruction to avoid definitive maxillomandibular orthognathic reconstruction during adulthood is generally unrealistic.

The rationale for postponing the correction of the dentofacial deformity until the time of “early” skeletal maturity is the same as that for the correction of the jaw deformity associated with a repaired cleft palate and most other routine dentofacial deformities (e.g., long face growth pattern, primary mandibular deficiency, maxillary deficiency in combination with relative mandibular excess). This rationale includes the consideration of the following: 1) avoiding injury to the developing permanent dentition and the inferior alveolar nerves 2) avoiding soft-tissue scarring 3) avoiding the loss of ramus marrow space that will complicate the success of necessary redo ramus osteotomies 4) minimizing negative psychosocial memories of earlier jaw surgery that may prevent the patient and family from later pursuing definitive reconstruction and 5) avoiding the iatrogenic three-dimensional deformation of the mandible that limits long-term success. In addition, any orthodontics carried out during the mixed dentition will not be reflective of the permanent dentition; a final course of braces would also be required.

Patients with HFM who have a significant degree of vertical, horizontal, and transverse maxillary deformity will also require Le Fort I maxillary osteotomy in segments. No lesser approach will be sufficient. The surgical objectives of the upper jaw surgery may include the following:

Safe and effective first-stage Le Fort I osteotomies carried out during the mixed dentition are not practical as a result of the location of the developing teeth and the expected inhibition of any postoperative horizontal maxillary growth.

Osteodistraction Reconstruction of the Mandible during Childhood

Since 1992, DO has been used as a technique in an attempt to correct mandibular morphology in young patients with HFM.* In 2002, a literature overview was published by Mommaerts and colleagues to address the long-term results of early DO among patients with HFM.¹⁴⁸ It was concluded that no convincing evidence supported the benefits of DO over other techniques for the treatment of HFM, and DO was not found to offer advantages with regard to the limiting of complications. In 2009, Nagy and colleagues published a critical and exhaustive literature review in search of evidence of long-term stability after early distraction osteogenesis of the mandible in patients with HFM.¹⁶³ Eighty-nine relevant articles were reviewed. Only 13 studies were found to have sufficient scientific merit to be included. The authors concluded the following:

Costochondral Graft Reconstruction of the Mandible during Childhood

There are several basic theories regarding the mechanism of how mandibular growth occurs:

When the condyle and the components of the ascending ramus are hypoplastic or absent in patients with conditions such as HFM, surgeons have selected varied donor sources to replace the missing parts.* Autogenous replacements for the condyle have included 1) the metatarsal bones 2) the proximal head of the fibula 3) the costochondral junction of the rib (CCJ/rib) and 4) the sternoclavicular joint.

In general, the CCJ/rib bone is an expendable donor site and often an ideal morphologic contour match, with adequate strength at the mandibular recipient site. For these reasons, the CCJ/rib bone has been commonly used as a substitute for the “condylar growth center” and for the replacement of components of the ascending ramus. Unfortunately, in the skeletally immature patient, clinical observation after transplantation confirms unpredictable growth of the graft as compared with what would be considered “normal” growth or growth that is equivalent to the contralateral “normal” side of the mandible.

Clinical experience indicates that the CCJ/rib graft that is placed in the child or young teenager is prone to excessive growth. Although some would argue that overgrowth of the mandible and the need for additional surgical correction is a relatively minor consequence, experienced clinicians have found the secondary distortions in the maxillomandibular complex difficult to correct in a satisfactory way as compared with what could be achieved by single-stage reconstruction at the time of early skeletal maturity (Fig. 28-11). Unless pressing functional issues occur during childhood (e.g., obstructive sleep apnea), the advantages of a single-stage approach at jaw growth maturity to achieve favorable long-term function and aesthetic enhancement are difficult to ignore (Figs. 28-12 through 28-16).

Scientists began to focus on the biodynamics and growth of the transplanted CCJ/rib during the early 1990s. Researchers have addressed questions about differences in the biologic nature of the cartilage of the CCJ/rib; the epiphyseal growth plate; the mandibular condyle; and the sternoclavicular joint. It is now clear that the cartilage associated with the CCJ/rib and the epiphyseal plate of a long bone are derived from the primary cartilaginous template of the skeleton. These cartilages are relatively resistant to extrinsic competitive growth-related factors. Alternatively, the mandibular condyle and sternoclavicular cartilages arise from their own blastemas in association with the periosteum and then form as secondary cartilages.

Peltomaki and colleagues used a rat model to analyze the dynamic histology that occurred with the growth of the CCJ/rib.183–192 The observed histologic findings are dependent on the amount of cartilage that remains on the autogenous CCJ at the time of transplant. One of the initial findings of these authors was that “costochondral grafts do not adapt to the new environment, but rather preserve their inherent growth potential.” In subsequent studies, Peltomaki and colleagues continued to explore the idea of whether the intrinsic growth of the CCJ/rib may also be influenced by the interaction of hormonal factors and masticatory movements.^183–192 It is now clear that variations in mandibular growth after the use of a CCJ/rib graft in both humans and experimental animal model simulations likely vary in accordance with the amount of cartilage used in the graft.

Peltomaki notes that, in the CCJ/rib the distance from the bone cartilage junction to the germinative zone is approximately 0.4 mm to 0.5 mm. It is evident that the germinative cells will invariably be included in a CCJ/rib graft. It is also evident that, in clinical practice, considerable variation in the amount of germinative cells maintained will occur. The reported clinical differences in mandibular growth after the replacement of the condylar process with a CCJ/rib graft is likely the result of variations in the amount of cartilage included in each graft. Peltomaki believes that the number of germinative cells has a direct impact on rib elongation (i.e., overgrowth). Clinicians should be cognitive of the fact that the amount of cartilage included in the graft is of significance with respect to the future growth of the reconstructed condyle–ramus unit. It is currently impossible to estimate the optimal amount of cartilage to be preserved in each individual case. Nevertheless, clinical and experimental studies support the notion that, by including only a “small amount” of cartilage in the CCJ/rib graft, less postoperative growth (i.e., overgrowth) can be expected.

Peltomaki examined the growth of the mandible after CCJ/rib graft condyle replacement in marmoset monkeys. Interestingly, in adult animals, the amount of cartilage did not make any difference with regard to the postoperative mandibular measurements. In the growing animal, a postoperative gradual deviation of the midline to the unoperated side occurred. The greater the amount of cartilage left in the graft at the time of transplantation, the greater the amount of midline deviation observed over time.

For a series of human patients, Peltomaki reviewed the histology of surgically removed overgrown CCJ/rib grafts that were initially placed during childhood. On the basis of the data, he believes that the unpredictable growth of the constructed condyle–ascending ramus is a common occurrence. Clinical and histologic examination revealed that the clinical type of overgrowth (i.e., linear versus exuberant) was not related to any specific microarchitecture, which in itself showed considerable variation. This suggested that other local factors (e.g., mandibular movement, loading of the reconstructed condyle) may also have an effect on the subsequent and eventual growth and overgrowth.

In general, for a first-stage mandibular procedure that is carried out during childhood to be considered successful in a patient with HFM, the growth of the reconstructed side of the mandible should equal or at least be close to that of the contralateral normal mandible. Unfortunately, despite decades of surgical experience with the use of the CCJ/rib graft as a reconstructive option for the missing condyle–ascending ramus in the growing patient with HFM, this option has not passed the test. Overgrowth is a frequent and serious consequence. The mechanism that causes this postsurgical growth disturbance is now clear. Predictable methods of overcoming this complication while still allowing the CCJ/rib graft to be effective have not been realized.

Osteodistraction of the Residual Mandibular Deformity after Costochondral Graft Placement during Childhood

Wan and colleagues reported their findings for the reconstruction of Type IIB and III mandibular malformations in patients with HFM.²⁷⁹ Their basic approach was to reconstruct the ipsilateral mandible with a CCJ/rib graft (N = 30 grafts, 27 patients) during the mixed dentition (mean age, 9.9 years ± 4.1 years). As a result of a high failure rate, this was followed by DO treatment of the residual deficient mandible. CCJ/rib graft failure that required regrafting occurred in 7 out of 30 cases (23%). Undergrowth that required DO during the mixed dentition occurred in 17 cases (57%). Overgrowth with severe distortion that required surgical correction occurred in 3 of the 30 cases (10%). For the majority of study patients who later required CCJ/rib graft DO, the complication rate was exceedingly high. In addition, all study patients (N = 27) required definitive orthognathic surgery at growth maturity, despite earlier procedures having been carried out.

Corcoran and colleagues presented a series of 8 patients with HFM who underwent mixed dentition costochondral reconstruction and who then submitted to DO.³³ They found a 62.5% complication rate. Stelnicki and colleagues similarly reported on 9 patients who underwent mixed dentition CCJ/rib graft reconstruction and then required distraction.^252A They reported a 33% rate of fibrous nonunion as well as other significant complications and limited ultimate success. On the basis of current published studies, it must be concluded that first-stage mandibular reconstruction with the use of CCJ/rib grafts during the mixed dentition for patients with Type IIB and Type III HFM deformities is fraught with complications and achieves limited success. The theory that a mandible reconstructed in this way can be reliably “distracted’ later if it does not grow normally has proved to be false.

Bone-Anchored Hearing Aid Option

A bone-anchored hearing aid (BAHA) consists of a permanent titanium fixture that is surgically implanted into the skull bone behind the ear and a small detachable sound processor that clips onto the fixture. BAHAs are suitable for people with conductive or mixed hearing loss who cannot adequately benefit from or who prefer not to use conventional hearing aids.

Colquitt and colleagues completed a systematic review and economic evaluation of BAHAs for people who are bilaterally deaf. ²⁹ Previous prospective studies of adults or children with bilateral hearing loss were eligible for review. Twelve studies were included; there were seven cohort pre-post studies and five cross-sectional audiologic comparison studies. Comparisons were made between BAHAs and the following: 1) conventional hearing aids, which are also known as air-conduction hearing aids (ACHAs) 2) bone-conduction hearing aids (BCHAs) and 3) unaided hearing and ear surgery. The study also looked at 4) unilateral versus bilateral BAHAs. Outcomes reviewed included hearing measures, validated measures of quality of life, adverse events, and measures of cost-effectiveness.

There appeared to be some audiologic benefits of BAHAs for this group of patients including 1) improvements in speech as compared with BCHAs and 2) understanding and noise as compared with ACHAs (however, ACHAs may produce better audiologic results for other outcomes). In addition, 3) hearing was found to be improved with the use of BAHAs as compared with unaided hearing and 4) studies that compared unilateral with bilateral BAHAs suggested the benefits of bilateral BAHAs in many situations.

However, prospective case series of BAHAs reported rates of 6.1% to 19.4% for the loss of the implants over time. In addition, the financial analysis suggests that BAHAs may not be a cost-effective option in terms of hearing gain and probability of needing alternative aids. Interestingly, the greater the benefit to the individual from aided hearing and the greater the difference in the proportion of people using the hearing aid for more than 8 hours per day, the more likely the BAHAs were a cost-effective option.

Janssen and colleagues completed a systematic review of bilateral BAHAs for bilateral permanent conductive hearing loss.⁸⁸ This was a review of the literature from between 1977 and 2011, and it included studies in which subjects of any age had permanent conductive hearing loss and bilateral implanted BAHAs. Eleven studies met the criteria for data analysis. Bilateral BAHAs were found to provide audiologic benefit as compared with unilateral BAHAs (i.e., improved thresholds for tones, speech in quiet and in noise, improved localization/lateralization, and patient’s perceived subjective benefit). Disadvantages of bilateral BAHAs included 1) listening in noise in some condition 2) presumed additional cost and 3) presumed increasing adverse event risk. The authors concluded that bilateral BAHAs provided additional objective and subjective benefit as compared with unilateral BAHAs.

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