Median mandibular cleft in SAMS syndrome - A rare case report.

Rationale: SAMS syndrome is a rare genetic disorder characterized by midline facial clefting, skeletal anomalies, and other defects. Salient Features: Among the craniofacial manifestations of SAMS syndrome is the presence of a median mandibular cleft (MMC). MMC is a rare occurrence and in this syndrome, it poses a complex challenge for both functional and aesthetic reasons. Patient. Findings: This rare case report describes the successful correction of an MMC in an 18-month-old child diagnosed with SAMS syndrome. Treatment: This report describes the presentation, diagnosis and treatment. The surgical intervention involved a meticulous, single stage, osseous reconstruction. The mechanism of MMC in SAMS syndrome is discussed. Outcomes: Early intervention for MMC in SAMS syndrome patients can offer promising outcomes.

'Frozen eye' - Post blowout fracture surgical correction - A case report.

The Rationale: "Frozen Eye" is a very uncommon sequel after treatment of orbital blowout fractures requiring implant placement. Patient Concerns: The implant may faultily impinge on the ocular and extra-ocular muscle(s), causing the abnormality in the movement of the eye. Diagnosis: We present a 56-year-old male whose ocular implant impinged on muscle, causing "frozen eye" and had an infected implant. Treatment Outcomes: The same was removed and surgically corrected. The manuscript describes the details and discusses the possible mechanism that led to the "Frozen Eye".

Modified Placement of C-flap in Unilateral Cleft Lip Repair to Avoid Alar Base Scar - A Retrospective Study.

Introduction: In spite of several techniques, Millard's approach for the correction of cleft lip still is widely used. Although versatile, it has certain drawbacks including scar contraction, short lip and notching. A modification of the 'C' arm helps to address this drawback. The purpose of this study was to describe the qualitative and quantitative outcomes of the author's modification of Millard's C flap. Materials and Methods: Archival photographic records of cleft lip repairs and post-operative photos of cases treated by author were screened. Anthropometric measurements of the height and width of the lip, the height of the vermilion and the width of the alar base were considered. The outcome was quantified by comparing the ratio of each feature between the cleft and normal sides. The quality of cleft lip repair was assessed using the Steffensen criteria. Appropriate statistical tests were performed. Results: In all, 233 cases with a mean age of 4.46 ± 0.91 years and 106 (45.5%) males formed the study group. The mean lip height ratio was 0.936 ± 0.04, the mean lip width ratio was 0.938 ± 0.037, the mean vermillion height ratio was 0.9433 ± 0.35 while the mean alar base width ratio was 0.932 ± 0.35. The physical parameter ratios' mean difference between the normal side and the cleft side was below 0.06 mm. Discussion: The mechanism behind the drawback of the classical Millard's technique is discussed and compared with present modification. The authors' modification shows that modified Millard's technique produces better aesthetic outcomes.

Rare case of granular cell tumour at 16 years - A case report.

Rationale: Granular cell tumours (GCTs) of the tongue are a rare, soft tissue pathological entity at young ages. This case report aims to present one such case. Patient Concerns: A 16-year-old female patient sought treatment for a small, slow-growing, painless nodule in the dorsum of the tongue since six months. She underwent successful orthodontic treatment in recent past and is in the retention phase. Diagnosis: Excisional biopsy revealed the lesion to be a GCT under histopathology. Treatment: The patient was treated for an abnormal, small, slow-growing, painless nodule in the dorsum of the tongue. Outcomes: The patient had satisfactory esthetics and early diagnosis. The active intervention dispelled the confusion about the role of trauma and orthodontics appliance in the etiology of GCT in this particular case to the patient. Take-away Lessons: GCT can occur at any age. Early diagnosis and corrective surgery would help to avoid late complications.

Rigid versus Resorbable Plate Fixation in Fronto-Orbital Advancement in Unicoronal Stenosis - A Retrospective Study.

Introduction: Rigid plating fixation (RPF) and resorbable plating systems (RPS) advanced the field of reconstruction in craniomaxillofacial region. However, their performance in patients, particularly the effect on bone remodeling at site of hardware placement is not much documented. This manuscript aims to compare the performance of RPF and RPS in a cohort using a retrospective audit of case records. Methods: Archival records were searched for patients who had undergone cranial metal-RPF or RPS or combination for the correction of craniofacial deformities following inclusion-exclusion criteria. From records, data of the quality and quantity of bone formed along the site of plate fixation as compared with the adjacent site, accommodating or facilitating brain growth, and persistence of bone deformity at the site of hardware placement were collected at the end of the follow-up period. A total of 128 sites from 18 individuals (6 with exclusive metal-RPF and 12 with RPS) mean age of 7.45 ± 7.28 (Median 4; IQR of 8.88;2.6-11.5) who underwent cranial bone remodeling surgery formed the study group. Results: There was a statistically significant difference between the RPF and PRS system at the fronto-orbital suture (P = 0.002) and coronal suture (P = 0.036) with bone quality and quantity. Discussion: The RPF system was rigid but had a set of issues, while RPS has advantages and limitations. The qualitative difference in between the two systems is different. Due to inherent dissimilarity, the two systems cannot be interchanged and due diligence has to be exercised while deciding on the system. More prospective studies are needed to validate the findings.

Unusual Number of Phleboliths in a Lip Vascular Malformation - A Case Report.

Rationale: Vascular malformation (VM) associated with jaws may cause jaw size discrepancy. Multiple phleboliths in VM are relatively rare. This case report aims to present one such case. Patient Concerns: A 33-year-old female patient sought surgical correction of her abnormally sized jaw and on examination, she was identified with VM. Diagnosis: Subsequent imaging tests revealed the presence of several phleboliths. Treatment: The patient was treated for an abnormal-sized mandible as well as partial removal of the superficial part of VM. Outcomes: The patient had satisfactory esthetics and there was less bleeding than anticipated. Take-away Lessons: VMs could cause jaw size discrepancy and the extent of the malformation could cause blood flow abnormalities leading to multiple phlebolith formation. Proper surgical planning and education of the patient are essential for successful treatment.

Accessory Maxilla in a Tessier 07 Cleft - A Case Report.

The Rationale: Accessory maxilla is a rare condition often associated with Tessier type-7 clefts with fewer than 25 cases recorded in the literature. This manuscript reports a unilateral accessory maxilla with six supernumerary teeth. Patient Concerns: A 5-1/2-year-old boy, a treated macrostomia case, on follow-up visit showed evidence of accessory maxilla with teeth on radiological examination. The structure was interfering with growth, and hence, surgical removal was planned. Diagnosis: Based on clinical history, diagnosis and imaging, accessory maxilla with supernumerary teeth was diagnosed. Treatment and Outcomes: The accessory structures and teeth were removed surgically via an intraoral approach. Healing was uneventful. The growth deviation was arrested. Take-Away Lessons: Intraoral approach is a good option to remove an accessory maxilla. Tessier type-7 cleft may be accompanied by type-5 clefts and such accessory structures when impinging on vital structures such as temporomandibular joint or facial nerve should be immediately removed to facilitate proper form and function.

Idiopathic bilateral mandibular condylar resorption causing anterior open bite - A case report.

Introduction: Mandibular condylar resorption (MCR) is a rare pathological entity, often affecting young females. Patient Concerns: It is accompanied by pain, malocclusion and compromised quality of life including aesthetic perception. Due to this multiplicity of features, the diagnosis, treatment and management of MCR are always a challenge. Diagnosis: This article reports a 25-year-old female suffering from progressive temporomandibular joint pain and compromised aesthetics. This article describes the clinical and radiological findings of this case. Treatment: The possible aetiopathogenesis and treatment are described.

Hemifacial hypertrophy - Report of 2 cases.

Hemifacial hyperplasia (HH) is a rare congenital condition involving enlargement of one or more tissues of the face. The treatment is surgically challenging and requires expertise. This manuscript aims to report two similar appearing HH but warranting different surgical treatment. A 19-year-old female and a 14-year-old boy presented with right facial asymmetry since birth and sought correction of the same. Surgical treatment was planned. Based on clinical history, diagnosis and imaging, HH was diagnosed. The first case was entirely a soft tissue abnormality that was treated with debulking while the second case had involvement of facial bones, necessitating surgical recontouring. The facial asymmetry was addressed. Healing was uneventful. Though the aesthetical concern and appearance of the two cases of HH were same, the treatment vastly differed. This was based on the source of asymmetry. Proper diagnosis and informed decision are a key for successful surgical outcome.

A rare case report of perioral sarcoidosis.

THE RATIONALE: Sarcoidosis is a multisystemic granulomatous disease of unknown aetiology that usually presents with systemic lesions including the skin. Isolated cutaneous lesions are very rare. PATIENT DETAILS: We present a middle-aged female patient, a known systemic lupus erythematosus patient, in remission developing an isolated, single sarcoidosis lesion in the perioral labial mucosa. The lesion was a rapidly growing nodular lesion with a papillary surface near the philtrum of the upper lip. DIAGNOSIS: A biopsy revealed that to be a granulomatous lesion with large multinucleated giant cells. Using a panel of tests, by excluding tuberculosis and establishing the immunoprofile, the diagnosis was established. Although infrequent, perioral and oral lesions may constitute the first signs of systemic sarcoidosis. OUTCOMES AND TAKE-AWAY LESSONS: For this reason, the oral health care provider needs to be watchful and when there is a suspicion of sarcoidosis, systemic sarcoidosis must be included in the differential diagnosis of oral and perioral granulomatous lesions.

Management of Large Dural Defect with CSF Leak in Hypertelorism Correction.

THE RATIONALE: Dural tear is a serious complication during hypertelorism corrective surgeries. Identifying the tear and managing requires considerable expertise. Managing large dural tears correctly is necessary to prevent cerebrospinal fluid (CSF)-related complications in craniofacial surgery. PATIENT CONCERNS: The patient presented with hypertelorism as a part of the Tessier Cleft 0 and sought to correct the widely placed eyes. DIAGNOSIS: Large critical-sized dural tear during modified box osteotomy surgery. TREATMENT: Besides successful modified box osteotomy surgery, the critical-sized dural tear was managed with fascia lata and fibrin glue. OUTCOMES: There was no CSF leak or related complication postsurgically indicating successful sealing and healing of the dural tear. TAKE-AWAY LESSONS: The synergistic mechanism by which fascia lata graft and fibrin glue help to hermetically seal the critical-sized defect, especially when there are variable amounts of hydrostatic-hydrodynamic forces of CSF exerting pressure on the patched area, is discussed.

Large odontome compressing inferior alveolar nerve - A case report.

A young female sought treatment for a painful swelling in the right mandibular posterior region for the past 1 month with severe halitosis and paresthesia of right lower lip. Past history revealed that #46 was impacted due to an odontogenic tumor and an unsuccessful attempt was made to remove the impacted teeth and the tumor. A warm, tender, mild extra-oral diffuse swelling with right side cervical lymphadenopathy present. Intra-orally, there was a missing crown of 46 with a diffuse, tender buccal cortical expansion with a draining sinus. Imaging studies revealed a radiopaque lesion with a well-defined and rounded radiolucent halo. A provisional diagnosis of acute suppurative osteomyelitis was made and the remaining roots along with the radio-opaque material removed in toto via an open approach method and the entire area reconstructed with autograft harvested from rib. Histopathologically lesion was diagnosed to be a complex composite odontome. The challenges of diagnosis and the implication are discussed.

Recurrent ossifying fibroma of the orbit - A case report.

RATIONALE: Ossifying fibromas are mainly found in the jaws and are rare in other parts of the cranium. Orbital involvement is relatively rare. PATIENT DETAILS: A case of an otherwise healthy 38-year-old overseas patient with gradual onset of a large growth in the right ocular area involving adjacent bone and operated twice over the last eight years is being presented. The lesion is involving the entire medial wall. TREATMENT: Considering the complex loco-regional anatomy as well as the restriction of overseas residence, preservation of a thin margin of bone was performed. TAKE-AWAY LESSONS: The need for repeat radiological surveillance was stressed.

Isolated Medial Orbital Wall Fracture Correction with Revision of Levator Plication - A Case Report.

The Rationale: Treatment options for isolated medial orbital wall fractures (IMOWF) vary. In young and favourable situations, most of the times, the fracture is left to heal spontaneously. If not properly monitored, it may improperly fuse causing poor form and function. Patient Concerns: A 24-year-old male presented for correction of his sunken left eye. The patient had a blunt trauma 10 years back and had left upper eyelid as well as ptosis correction elsewhere. Since that surgery, the patient noticed that the eye had been slowly and progressively "sinking" with time. Diagnosis: An IMOWF with periorbital fat entrapment and detachment of levator palpebrae superioris was identified. Treatment and Outcome: The fracture site was reached via a transcaruncular approach, the fracture corrected and orbital defect corrected with a mesh. One week later, in a second-stage surgery, under local anaesthesia, the muscle was repositioned. Recovery was uneventful and the desired aesthetic-functional results were achieved. Take-Away Lessons: An untreated IMOWF in young adolescents could persist and subsequent growth could amplify the enophthalmos. A careful diagnosis and treatment is warranted.

Complete ankylosis of temporomandibular joint and cervical spine - A case report.

Ankylosing spondylitis (AS) is a chronic, multi-systemic inflammatory disorder. It rarely involves peripheral joints such as Temporomandibular joint (TMJ). Here, we present a case of a 44-year-old male, known case of AS for eight years who had TMJ-related complaints for two years. On examination, bony ankylosis of TMJ possibly due to pre-existing AS was made. A bilateral standard gap arthroplasty procedure was done. The challenges faced during the surgical procedure are discussed.

Cicatricial ectropion correction for a case of amniotic band syndrome.

BACKGROUND: Amniotic band syndrome (ABS) or amniotic deformity, adhesions and mutilations (" ADAM" ) is a spectrum of rare congenital malformations related to an early phase of organogenesis. In the craniofacial region, ABS could manifest in several forms. A rare form of ABS with Tessier Cleft-9 associated with atypical Cleft-2 presenting with a cicatricial ectropion is reported and management of the condition is discussed. KEY POINTS: ABS causing oro-facial deformities is increasingly being reported. Surgical management requires staged correction and is often challenging. As growth is dynamic, evolving problems should be anticipated. Frequent follow-up and early intervention could prevent unfavourable complications. MAIN LESSONS LEARNT: ABS has craniofacial components and it requires an adequate understanding of principles of growth and healing to plan corrective surgeries for such complicated and rare conditions.

Square Face Correction by Gonial Angle and Masseter Reduction.

INTRODUCTION: The association of mandibular gonial angle, facial height, and jaw relationship in masseteric hypertrophy (MH) has not been adequately described for the typical Indian population. The aim of this study was to report the gonial angle relationship with facial height parameters in cases diagnosed with bilateral MH and its possible influence on the treatment plan. MATERIALS AND METHODS: This is a retrospective study based on archival records of bilateral MH cases surgically treated over a 10-year period at the author's center. Patients' records fulfilling inclusion and exclusion criteria were considered for the study. Age, gender, upper anterior facial height (UAFH), lower anterior facial height (LAFH), upper posterior facial height (UPFH), ramus height (also a reflection of the lower posterior facial height), and gonial angle were collected along with the type of bite (open/normal/deep), surgical procedure (debulking with/without bone removal), and concomitant jaw bone corrections (yes/no). These were subjected to statistical analysis using SPSS, and P ≤ 0.05 was taken as statistically significant. RESULTS: Overall, 21 patients formed the study group comprising 9 females and 12 males. Gender influenced the UAFH, LAFH, UPFH, ramus height, and gonial angle significantly. Males had higher values than females. Normal bite had an obtuse gonial angle than the deep bite, and the difference was statistically significant (P = 0.036). When the gonial angle was acute or square faced, the need for other surgical procedures was high and the difference was statistically significant (P = 00.048). DISCUSSION AND CONCLUSION: The results are discussed in the light of Indian skeletal anthropometry. The relationship of the gonial angle with facial height parameters in bilateral MH cases in this part of the world is presented.

Comparative evaluation of direct sinus lift with bone graft and zygoma implant for atrophic maxilla.

INTRODUCTION: Implant rehabilitation of atrophic maxilla is challenging. The aim of this manuscript is to compare the clinical outcome of rehabilitation of atrophic maxilla (long span) by zygomatic implants (ZI) versus conventional sinus lift with augmentation procedures dental implants (SLAP-DI). MATERIALS AND METHODS: A retrospective record analysis of a cohort of 25 patients fulfilling the inclusion and exclusion criteria, over a period of 6 years were performed. The patients had been either treated with ZI or SLAP-DI. The outcome measures were: prosthesis, implant and augmentation failures, any complications, patients' number of days with total or partial impaired activity, time to function and number of dental visits, assessed by independent assessors from records. The data was analyzed using SPSS with P ≤ 0.05. RESULTS: There were 16 (64%) men and 11 (44%) had ZI while 14 (56%) underwent SLAP-DI. There were no prosthesis or implant failures in this cohort. Between ZI and SLAP-DI, there was no gender difference (P = 0.648), border line significance in minor complication (P = 0.075), with less complication encountered in SLAP-DI than the ZI. None of the cases had major complications that required hospitalization or severe medical therapy. Of the 14 cases that underwent SLAP, 11 (78.6%) had no augmentation failure (resorption <10%). ZI had older age (P = 0.000), with greater number of teeth missing (P = 0.002), with lesser time for functional loading (P = 0.000), required less number of visit to dentist (P = 0.000), and lesser bone loss at insertion site (P = 0.005). DISCUSSION & CONCLUSION: The results are discussed in light of previous pertinent literature. This short term (~ 1 year follow-up) suggest that SLAP-DI and ZI have certain similar features. There is no statistical difference in outcome barring the time needed to functional loading (1.3 days vs. 444.3 days). Relatively more complications were reported for ZI, but had no statistical significance. Long-term and multi-center data are needed to confirm the results.

Impacted wisdom tooth in the floor of the orbit.

Identification and management of ectopic supplemental tooth in anatomically complex areas such as the floor of orbit are challenging. This arises from the rarity and lack of consensus over management. The situation gets complex when there is an evidence of follicular pathology such as dentigerous cyst. In this report, a case of maxillary third molar associated with maxillary sinus and a distomolar in association with the floor of orbit medially to the inferior-orbital canal is presented. The surgical management of the condition is presented.

Reconstruction of moustache and ala of the nose with a single forehead flap.

Reconstruction of critical-sized defects of composite facial units is a challenging one. This is amplified when facial hair-bearing areas are included. A case of reconstruction of multiple midfacial defects is presented. Initially, defective jaws were addressed. Later, appropriate modification of existing extended paramedical forehead flap to sequentially reconstruct defective lip, moustache, and parts of alae are described. The flap design was such that hair-bearing areas were used to reconstruct moustache while the nasal alar defect was reconstructed using the non-hair-beating forehead skin. Finally, dental implants were also placed. The technique was reliable, giving minimal donor site deformity but with maximum result. However, it is a multi-stage procedure. The paper also describes the challenges of midfacial unit reconstruction as handled in this case.