Nasal oral fistula revealing Langerhans´ cell histiocytosis in adult patient: case report.

Langerhans cell histiocytosis (LCH) is a rare systemic disease caused by proliferation of mature histiocytes; its association to histiocyto fibroma is rarely reported. It rarely affects adults. We report a case of systemic LCH, in an adult patient with osteolytic lesion causing a fistula between the left nasal cavity and hard palate, involving the bone, lung, lymph node and associated to multiple histiocyto fibroma. The patient was operating for a fistula, and he was treated by chemotherapy and corticosteroids. Langerhans´ cell histiocytosis is a rare case, especially in adult patient. The diagnosis was based on histological and immunohistochemical analyses. This patient was treated by steroids and chemotherapy.

Speech and Surgical Outcomes in Children With Veau Types III and IV Cleft Palate: A Comparison of Internationally Adopted and Nonadopted Children.

OBJECTIVE: This study compares speech and surgical outcomes in internationally adopted and nonadopted patients undergoing cleft palate repair, and examines the influence of age at initial palatoplasty. DESIGN: Retrospective cohort study setting: Tertiary Care Children's Hospital. PATIENTS: 70 international adoptees and 211 nonadoptees with Veau type III and IV clefts (without associated syndrome) repaired at our institution. OUTCOME MEASURES: Outcomes included VPI, compensatory misarticulations, intelligibility, nasal air emission, oronasal fistula, and secondary speech surgery. Speech evaluations completed near 5 years of age were gathered from a prospectively collected database. RESULTS: Adoptees underwent palatoplasty 5.2 months after arrival, a mean of 10.4 months later than nonadoptees. Adoptees were significantly more likely to develop moderate/severe VPI and trended toward more frequent need for secondary speech surgery. Oronasal fistula occurred at similar rates. Increased age at initial palatoplasty was a significant predictor of moderate to severe VPI, and need for secondary speech surgery. CONCLUSIONS: International adoptees undergo palatoplasty 10.4 months later than nonadoptees and are significantly more likely to develop moderate/severe VPI, with a trend toward increased secondary speech surgery. An association between treatment delay and moderate/severe VPI and secondary speech surgery has been demonstrated. While a causal relationship between delayed repair and inferior outcomes in international adoptees has not been proven, this data suggests that surgical intervention upon unrepaired cleft palates soon after adoption may be beneficial. The opportunity for a change in practice exists, as half of the 10.4-month relative delay in palate repair occurs postadoption.

Cleft Palate Repair: Description of an Approach, Its Evolution, and Analysis of Postoperative Fistulas.

BACKGROUND: Fistulas following cleft palate repair impair speech, health, and hygiene and occur in up to 35 percent of cases. The authors detail the evolution of a surgical approach to palatoplasty; assess the rates, causes, and predictive factors of fistulas; and examine the temporal association of modifications to fistula rates. METHODS: Consecutive patients (n = 146) undergoing palatoplasty during the first 6 years of practice were included. The technique of repair was based on cleft type, and a common surgical approach was used for all repairs. RESULTS: The fistula rate was 2.4 percent (n = 125) after primary repair and 0 percent (n = 21) after secondary repair. All complications occurred in patients with type III or IV clefts. Cleft width and cleft-to-total palatal width ratio were associated with fistulas, whereas syndromes, age, and adoption were not. Most complications could also be attributed to technical factors. During the first 2 years, modifications were made around specific anatomical features, including periarticular bony hillocks, maxillopalatine suture, velopalatine pits, and tensor insertion. The fistula rate declined by one-half in subsequent years. CONCLUSIONS: The authors describe a surgical approach to cleft palate repair, its evolution, and surgically relevant anatomy. Fistulas were associated with increasing cleft severity but could also be attributed to technical factors. A reduction in frequency and severity of fistulas was consistent with a learning curve and may in part be associated with modifications to the surgical approach. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

Case for diagnosis. Palate perforation due to cocaine use

Abstract: We report a 42-year-old cocaine addicted female patient referred for evaluation of hard palate ulceration resulting in oro-sinus communication with difficulties in swallowing and phonation, an rhino-sinusitis. Acrylic and removable silicone prosthesis was prescribed to relieve severe functional disorders. It is essential that the patient permanently abandons cocaine use to perform surgical reconstruction.

Total Palatal Mobilization and Multilamellar Suturing Technique Improves Outcome for Palatal Fistula Repair.

BACKGROUNDS: The success rate of the surgical repair of palatal fistula after palatoplasty is often unsatisfactory. This study is a review of 15 years of single surgeon's experience with the evolution of a reliable surgical technique with high success rate. METHODS: This is a retrospective chart review of consecutive cleft cases undergoing repair of palatal fistula from 2000 to 2015. The study included 37 consecutive fistula repair cases with wide elevation and mobilization of the palatal tissues and nasal and oral layer repair. Group 1 (n = 20) were treated earlier in the study using either midline, von Langenbeck, or 2-flap palatoplasty with 3-layer suturing. Group 2 (n = 17) were treated through a Dorrance-type incision and additional repair of the oral periosteum for a total of 4-layer suturing. RESULTS: The overall fistula closure rate was 94.6% (90% in group 1 and 100% in group 2). The difference in outcome between the 2 groups was statistically insignificant (P > 0.05). Most patients (83.8%) had concomitant velar muscle retropositioning for treatment of velopharyngeal incompetence. CONCLUSIONS: Fistula repair using wide mobilization of the entire palate through previous repair incisions and multilamellar suturing technique has a very low fistula recurrence rate. Addition of the fourth layer of suturing and the use of a Dorrance-type incision further improves the outcome. This approach provides wide tissue release and access to tissue layers for better repair and tension-free closure. Combining intravelar veloplasty with fistula repair is safe and allows management of the fistula and its possible consequences on palatal function in a single procedure.

Oronasal fistula post-septoplasty.

Nasal septal surgery is one of the most common otolaryngology procedures performed, with rare occurrences of major postoperative complications. Oronasal fistula is a rare complication of septal surgery, with only few cases being reported in the literature. It is commonly caused by a congenital cleft palate, cleft palate surgery, traumatic injuries, infections, or neoplasms; however, it does not usually occur post-septal surgery. Here, we report the case of a 55-year-old male who presented with a symptomatic oronasal fistula post-septoplasty. Unlike other reported cases, there were no features of underlying palatal pathologies, and no previous history of other nasal or oral procedures. We described a multilayer surgical closure technique that was utilized in this case, with no recurrence seen during a 6-month follow-up period. Additionally, we reviewed the other 3 reported cases of this complication, and discussed the possible etiology in this case.

Use of a purified collagen membrane to aid closure of palatal fistulae.

INTRODUCTION: Fistula formation following closure of palatal clefts remains a difficult clinical complication. A significant recurrence rate has also been reported following attempted closure. We present our results of fistula closure augmented with Bio-Gide(®), a purified porcine collagen membrane designed to promote guided tissue regeneration. METHODS: We reviewed the records of 263 patients operated between 1993 and 2011 for closure of palatal fistula. The patients selected comprised 61 who underwent fistula closure augmented with Bio-Gide and 202 with other techniques in the absence of Bio-Gide. We reviewed the age at surgery, sex, location of fistula, cleft type and outcome. Operation success was defined as an asymptomatic patient along with visible confirmation of closure of the fistula. RESULTS: The overall fistula closure rate was 75% in the Bio-Gide group and 63% in the non-Bio-Gide group (p = 0.070) and 86% versus 61% in the unilateral cleft palate patients (p = 0.027). DISCUSSION: Bio-Gide has improved the success rate in fistula closure in this study. Using this technique, fistula closure can be performed as a day case procedure and does not need to transgress any original suture lines; an additional advantage is that this procedure does not require harvesting of any autologous tissue to augment the repair.

Progressive midfacial bone erosion and necrosis: case report and differential diagnosis.

A 31-year-old woman presented with a large oro-nasal communication (ONC), loss of vomer and significant nasal cartilage and nose deformity. Physical examination of the patient revealed a typical midline destructive lesion (MDL) with nasal septum and hard/soft palate perforation with a friable granular surface and a large amount of necrotic tissues. Medical history was unremarkable and the patient denied previous local trauma, including surgical procedures or drug assumption. Pathological examination revealed the presence of necrosis and chronic inflammation. MDLs have numerous etiologies. Signs and symptoms of MDLs can be similar and an accurate diagnosis may be elusive. We hereby present detailed clinicopathological findings.

Submandibular hydatid cyst fistulized into the oral cavity.

UNLABELLED: Hydatid cysts of the head and neck are rare, even in regions where echinococcal infestation is endemic. Although complications, like cyst rupture and infection, may occur, an external fistula is extremely rare. This study examined a case of hydatid cyst that had fistulized in the right submandibular region of the oral cavity, which eroded the mandible. CASE REPORT: A 45 year-old female patient visited our clinic with complaints of a discharge that left a bad taste in her mouth that persisted for 1 month and a painless swelling in the right submandibular region that had slowly expanded over the past 5 months. A physical examination revealed a cystic mass located in the right submandibular region that had fistulized in the oral cavity. The results were consistent with a hydatid cyst. We performed submandibular gland exeresis and a marginal mandibulectomy to excise the hydatid cyst together with the fistulous tract.

Repair of cocaine-related oronasal fistula with forearm radial free flap.

BACKGROUND: Cocaine snorting may cause significant local ischemic necrosis and the destruction of nasal and midfacial bones and soft tissues, leading to the development of a syndrome called cocaine-induced midline destructive lesion. A review of the English-language literature reveals only a few articles describing the treatment of hard and/or soft palatal perforation related to cocaine inhalation. Described here are 4 patients with a history of cocaine abuse showing palatal lesions. MATERIALS AND METHODS: From 2010 to 2013, a total of 4 patients affected by cocaine-related midline destructive lesions were referred to our department. They all presented signs of a cocaine-induced midline destructive lesion. They showed wide midfacial destruction involving the nasal septum as well as the hard and soft palates causing an ample oronasal communication. RESULTS: In 3 patients, oronasal communication has been treated successfully using a personal technique based on a partially de-epithelialized forearm free flap. The fourth patient had been treated only with local debridement because, when she came to our attention, her abusive habits were still unsolved. DISCUSSION: Different surgical options have been reported such as local, regional, and free flaps for hard and soft palate reconstruction. However, because of an unpredictable vascularization of the palatal tissues and owing to the scarceness of the local soft tissues, local flaps are at high risk for partial and complete failure. The transfer of free vascularized tissue, however, seems to be the most reliable and logical solution for medium- to large-sized fistulas. Among the various free flaps, we choose the radial forearm type because of the pedicle length and the flap thickness.

Oral cutaneous sinus tract, vertical root fracture, and bisphosphonate-related osteonecrosis: a case report.

INTRODUCTION: Oral cutaneous sinus tracts (OCSTs) of dental origin are often initially misdiagnosed and inappropriately treated. Accurate diagnosis is especially important in cases of bisphosphonate (BP) therapy because extraction may lead to a risk of osteonecrosis. A case report of misdiagnosis related to a tooth with a vertical root fracture in an oncologic patient treated with BPs is reported here. METHODS: In 2011, a 75-year-old woman was examined at the oral medicine clinic because of pain and swelling of the left submandibular area. The patient's medical history included oral and intravenous BP therapy because she was diagnosed with metastatic breast cancer and left maxillary stage 1 antiresorptive agent-induced osteonecrosis of the jaw. The lower left odontogenic region showed no signs or symptoms, and no apical pathosis was observed on imaging. Although antibiotics were applied, clinical symptoms worsened and an OCST appeared. Intravenous antibiotic treatment was pursued. Biopsy and direct smear from fistula were not conclusive. A diagnosis of a nonexposed variant of stage 3 antiresorptive agent-induced osteonecrosis of the jaw was established. RESULTS: Symptoms resolved after 2 weeks of antibiotic treatment and reappeared a month later. Endodontic examination revealed that the origin of the OCST was tooth no. 18 caused by a vertical root fracture, and the tooth was extracted. The patient was scheduled for routine checkups because of the fact that osteonecrosis may occur in intravenous BP-treated patients. CONCLUSIONS: Early correct diagnosis can prevent unnecessary and ineffective antibiotic therapy and surgical intervention, which is not recommended in intravenous BP cases.

Parotid fistula--an extra-orally draining infected dentigerous cyst associated with a supernumerary fourth molar in ascending ramus.

UNLABELLED: An infected cyst, associated with a supernumerary fourth molar in the ascending ramus of the mandible, presented with parotid swelling, trismus and pain. It was managed as a parotid infection but recurred and a draining pre-auricular sinus developed, which was thought to be a parotid fistula. This was managed by cautery, followed by excision of the fistula, both of which were unsuccessful. Following this, further investigations revealed that the cause of the facial sinus was in fact a cyst associated with a mandibular fourth molar in the posterior ascending ramus. The tooth was extracted, via a pre-auricular extra-oral approach, under general anaesthetic. CLINICAL RELEVANCE: This case shows how a dental panoramic radiograph can be extremely helpful at ruling out certain pathology. It also demonstrates how the symptoms of an infection arising from a tooth in the ramus may be misdiagnosed as salivary gland pathology.

Ectopic mandibular third molar leading to osteomyelitis of condyle: a case report with literature review.

BACKGROUND: A tooth is said to be ectopic if it is malpositioned either due to congenital factors or displaced due to pathological lesions. The incidence of osteomyelitis of condyle has a rare occurrence, very few cases have been reported, either their etiology is unknown or is due to hematogenous spread or a tuberculous focus. This particular case may be a rare of its kind, as recent literature did not reveal any case where an ectopic mandibular third molar led to osteomyelitis of the condyle. CASE REPORT: This report presents a unique case where an ectopically placed mandibular third molar led to extraoral sinus and scar formation below the ear lobule with osteomyelitis of the mandibular condyle and proposes various indications for its removal along with literature review. DISCUSSION: Ectopic eruption of a tooth into the dental environment is common, whereas ectopic eruption of tooth in other sites is rare. The exact etiology of ectopic eruption of mandibular third molar in condyle is a rare occurrence and to the best of our knowledge, only 14 cases have been reported in the literature. Management of such cases should be meticulously planned after ruling out various local as well as systemic factors as an underlying cause for osteomyelitis and on the basis of the position and type of ectopic tooth and related potential trauma which could be caused by surgical intervention with less morbidity. CONCLUSION: This particular case may be the unique of its kind, as recent literature did not reveal any case where an ectopic mandibular third molar led to osteomyelitis of the condyle. Diagnosis and management in such cases deserve very special attention to rule out various local and systemic causes which can cause osteomyelitis to affect very unusual site like mandibular condyle. The aim of surgical intervention should be to cause minimum morbidity without affecting the functional efficiency of the mandibular condyle. In this report, we also have proposed the indications for surgical intervention to remove ectopically erupted teeth.

Asymptomatic patent nasopalatine ducts after rapid maxillary expansion.

This article describes the clinical case of a patient, a 13-year-old boy, in whom the appearance of patent nasopalatine ducts was noted after having a rapid maxillary expansion performed. To confirm this finding, inspection and radiographic examination were performed in which gutta-percha cones were used as contrast. The patient related no symptoms or discomfort due to the ducts. The patient was followed up for a period of 6 months, during which he was instructed as regards correct cleaning to prevent the accumulation of food and infection.

[Florid cemento-osseous dysplasia of the jaws]. / Dysplasie cémento-osseuse floride des maxillaires.

INTRODUCTION: Florid cemento-osseous dysplasia is a benign and rare tumor of the jaws. It is more commonly seen in middle-aged black women. Most cases are asymptomatic and are found during routine radiographic examination. OBSERVATIONS: We report two complicated cases of florid cemento-osseous dysplasia, one with facial deformity and the other with chronic osteitis. DISCUSSION: The diagnosis of florid cemento-osseous dysplasia is based on clinical and radiological features. The lesions are commonly bilateral and symmetrical.

[Maxillary sinus osteoma associated with a mucocele]. / Ostéome du sinus maxillaire associé à une mucocèle.

INTRODUCTION: Maxillary sinus osteomas are rare benign tumours with a poorly documented clinical evolution. Their craniofacial localization may be part of a syndrome. We report a case of maxillary sinus osteoma associated to a mucocele and a cyst probably of dental origin, with no sign of associated Gardner syndrome. CASE REPORT: A 52-year-old woman consulted for chronic maxillary sinusitis and an oral vestibular fistula. The CT-scan suggested a mucocele with reactive bone formation. Macroscopically, a pediculated bone tumour was found next to a mucocele, and to a cyst probably of dental origin. The anatomopathological examination led to a diagnosis of cancellous osteoma. DISCUSSION: Several hypotheses have been made on the etiology of sinus osteomas. When identified, screening for Gardner's syndrome should be implemented because of the associated risk for colic malignancy.

A rare complication after septoplasty procedure in a misdiagnosed submucous cleft palate case: palatal fistula.

Nasal septoplasty is a common and low-risk procedure performed very frequently in plastic surgery as well as in otorhinolaryngology. The development of a palatal perforation following a nasal septoplasty procedure is a very rare event with only a few cases reported in the literature. A patient with palatal fistula formation after septoplasty procedure is presented here, who was later on diagnosed with submucous cleft palate during the repair of the palatal fistula.

Defect in the nasal septum and maxillopalatine bone with an oronasal fistula: traumatic stimulation caused by a peculiar habit?

We report the case of a 25-year-old man with defects in the nasal septum and maxillopalatine bone and an oronasal fistula.