Facial necrotizing fasciitis from an odontogenic infection

Necrotizing fasciitis (NF) is defined as rapidly progressive necrosis of subcutaneous fat and fascia. It is a rare but life-threatening infection characterized by a progressive, usually rapid, necrotizing process of the subcutaneous tissues and fascial planes. The condition is commonly described in the extremities, abdominal wall, and perineum but rarely seen in the head and neck. The diagnosis of NF depends mainly on clinical features, which are not always observable, so that the disease is often diagnosed late in its course, resulting in high mortality. Broad-spectrum antibiotics, aggressive surgical treatment and supportive therapy are the most widely accepted modalities of successful treatment. We describe a case of necrotizing fasciitis of the head and neck, arising from odontogenic origin.     

Ascending necrotizing fasciitis of the face following odontogenic infection

Necrotizing fasciitis (NF) of the face is a rare but extremely dangerous complication of dental infection associated with a nearly 30% mortality rate. This infection spreads rapidly along the superficial fascial planes of the head and neck and can lead to severe disfigurement. Reports in the literature of cases of NF of the face caused by dental infection are few. We report such a case in a 36-year-old woman and review the current standards of diagnosis and management. The patient initially presented with pain and severe swelling in the left side of her face subsequent to a dental infection. The symptoms had progressed quickly and had not improved with administration of oral antibiotics in the outpatient setting. The patient had no palpable crepitus despite its classic association with NF. The infection also took a rare, ascending route of spread with involvement of the temporalis muscle. Cultures taken during debridement grew Streptococcus anginosus and Bacteroides. Biopsies of involved muscle showed histologic evidence of necrosis. Through early surgical intervention including aggressive debridement, and the adjunctive use of appropriate antibiotics, the patient recovered with minimal loss of facial mass and no skin loss. Although NF of the face is rare, the surgeon must maintain a high index of suspicion with any patient presenting after a dental infection with rapid progression of swelling and a disproportionate amount of pain that is unresponsive to antibiotics.     

Clinico-pathologic correlations of myofibroblastic tumors of the oral cavity: 1. nodular fasciitis

BACKGROUND: Nodular fasciitis (NF), a soft tissue lesion mainly composed of myofibroblastic cells, is well documented in various body locations however, in the oral cavity it is rare. The NF has non-specific histologic characteristics that might result in misdiagnosis and mistreatment. The aim of the study was to analyze clinico-pathologic correlations of NF occurring in the oral cavity. METHODS: A total of 36 cases of oral NF were analyzed including review of the English language literature and five new cases from our files. RESULTS: Oral mucosa NF was found to peak in the fourth and fifth decades, which is a decade later than NF occurring in other sites of the body. The most common locations were the buccal mucosa (52.8%) and the lips (16.7%). Duration of lesions ranged from 3 days to 2 years, with approximately 61% being present for more than a month, which is longer than the duration of NF from other body locations. Histologically, oral NF showed varying degrees of cellularity and frequently contained myxomatous areas, and often demonstrated local infiltration into adjacent tissues. However, the myofibroblastic, spindle-shaped lesional cells were uniform and lacked any major signs of atypia. Mitotic figures, characteristically abundant in NF lesions throughout the body, ranged from absent to moderately high in oral NF cases. Treatment modality of choice was complete surgical excision. Recurrence was reported for only one case. Extensive, mutilating surgical procedures for oral mucosa NF are unnecessary, since lesions resolve even when surgical margins are partly involved. CONCLUSIONS: The NF should be included in the clinical differential diagnosis of superficial and deep soft tissue masses of the oral cavity, especially of the buccal mucosa. Histopathologically, NF should be differentiated from other spindle cell lesions, mainly myofibroma, neurofibroma, fibrosarcoma, solitary fibrous tumor, fibromatosis and fibrous histiocytoma.     

Acute cervicofacial necrotizing fasciitis: three clinical cases and a review of the current literature

Cervicofacial necrotizing fasciitis (NF) is a rare condition that can quickly become life-threatening if appropriate treatment is delayed. This condition is observed as a rapidly progressive infection that causes extensive necrosis of the superficial fascia. This report presents a case of cervicofacial NF with microbiological isolation of methicillin-resistant Staphylococcus aureus in a patient with a history of uncontrolled diabetes mellitus following a minor scalp trauma. The article also presents two cases of NF secondary to odontogenic infection. The radiographic finding of the presence of gas locules in the facial planes on the CT scan helped to confirm the diagnosis. Patients were treated with broad-spectrum antibiotic therapy, extensive surgical drainage, debridement, and supportive care. Awareness in the dental community of the signs of NF will facilitate optimal patient management.     

口腔颌面部结节性筋膜炎6例报道

目的:总结口腔颌面部结节性筋膜炎的临床病理特点及鉴别诊断要点。方法:对1995-2003年收治的6例口腔颌面部结节性筋膜炎患者的临床资料进行回顾分析,探讨其临床特征、组织病理学特点、鉴别诊断、治疗及预后等。结果:6例结节性筋膜炎均为病程短、生长快的肿瘤中年女性多见。肿块呈结节形,直径为1.5～3.5cm。组织学特点为纤维母细胞增生活跃,核分裂像易见“,S”形结构、组织裂隙、外渗红细胞、黏液背景;免疫组化染色显示Vim( )、SMA( )、Des(-)。手术切除后,随访1～9a,无1例复发。结论:结节性筋膜炎的本质是纤维母细胞和肌纤维母细胞增生,属良性病变,切除后不易复发,预后良好。     

Bisphosphonate-associated osteonecrosis of the jaws. 3. Two case reports

The cases presented show that bisphosphonate-induced osteonecrosis of the jaw is difficult to treat. Recently, a classification of bisphosphonate-induced osteonecrosis of the jaw based on clinical appearance was published. On the basis of this classification the seriousness of the osteonecrosis can be evaluated and a method of treatment determined. The common opinion is that treatment should be as conservative as possible. This means that treatment should have as its objective the prevention of the spread of the disease by means of antibiotics and disinfectant mouthwash. Sharp bony edges may be trimmed. Extensive surgical treatment should be reserved for those rare cases in which the osteonecrosis is progressive.     

Solitary neurofibroma of the temporal bone

Neurofibroma is a benign peripheral nerve sheath tumor that can be occasionally found in the head and neck region as multiple lesions associated with neurofibromatosis type 1 (NF-1) or as a solitary tumor. The real frequency of isolated neurofibromas not associated with NF is uncertain, and lesions in the temporal region are extremely rare. The aim of the current article was to report an unusual case of solitary neurofibroma localized in the temporal and infratemporal regions with 10 years of evolution in a female patient without any other manifestation or familiar history of NF-1. The patient underwent surgical treatment for complete excision of the lesion, and the 2-year follow-up revealed no signs of recurrence.     

Parapharyngeal space tumors: surgical approaches in a series of 13 cases

Tumors originating in the parapharyngeal space are rare; they comprise approximately 0.5% of head and neck tumors. Most (70–80%) are benign and the most frequent origins are salivary and neurogenic. The aim of this study is to present the surgical procedures used for the treatment of 13 patients with parapharyngeal space tumors; 11 of them were suffering from benign tumors (the most frequent being pleomorphic adenoma; 8 cases) and 2 from malignant lesions. The following surgical approaches were used: intraoral (2 cases), transcervical (4 cases) and transmandibular (7 cases) with different types of mandible osteotomies. The type of surgical approach was dictated by the type of the lesion (malignant or benign), the exact location, the size, the vascularity and the relation of the tumor to the neck neurovascular bundle. In all cases the selected surgical approach allowed the complete resection of the tumor, obtaining clear margins in cases of malignancy, without adding to the patient's preoperative morbidity. It was concluded that the surgical approach to the parapharyngeal space tumors must be adjusted to the tumor characteristics and be as wide is necessary to achieve its complete removal with safety.     

Necrotizing fasciitis of the maxillofacial region caused by dental infection. A case report and review

Necrotizing fasciitis (NF) is a destructive and potentially fatal soft tissue infection characterized by extensive necrosis and gas formation in subcutaneous tissues and fascia, with serious involvement of muscles, vessels, nerves, and fat. In the maxillofacial region, NF is less common. The process can represent the evolution of a dental infection supported by aerobic and anaerobic bacteria that are resistant to antibiotic therapy (multidrug resistance) in immunocompromised patients or the natural evolution of untreated infection. Because of the rarity of the disease, diagnosis and treatment are often delayed, which may result in a fatal outcome due to respiratory problems or systemic complications. The success of the treatment is surgical debridement and high doses of antibiotic therapy. The AA described a case of NF in a female, 59 years old, who developed NF in the maxillofacial and neck region following dental infection and after consulting our Institute for remarkable swelling of the right cheek, palpebral and parotid regions, submaxillary region, and neck; this swelling is associated with hyperpyrexia, trismus, poor systemic conditions, and serious respiratory difficulty. Through prompt clinical diagnosis, early surgical treatment, appropriate antibiotic therapy (culture analysis revealed sensitivity to Imipenem and Levofloxacina), and local control of the lesion through surgical medications twice daily, we were able to not only avoid serious and fatal evolution of the process, but also to limit tissue involvement, preventing further extension of the necrosis to other anatomical structures of the region. A satisfactory clinical result was thus obtained.     

Ameloblastoma and Intracranial Involvement: The Current Challenge of the Radical Surgical Treatment. Comprehensive Review of the Literature and Institution experience

BackgroundAmeloblastoma (AMBL) is an odontogenic tumor, considered to be benign, but aggressive, whose principal risk is a recurrence. The growth can be enormous, and it can extend into the intracranial compartment with serious consequences. PurposeThe intracranial involvement of AMBL is rare, and it may require an extensive surgery. Although it is a rare condition for the neurosurgeon to treat, knowing this condition can lead to a significant increase in survival for these patients. MethodsA case of a 56-year-old woman presented with a history of recurrent left maxilla AMBL with intracranial extension and dural involvement of the anterior and medial cranial fossa is reported, followed by a systematic review of the literature with the aim to identify the best surgical treatment. ResultsA total of 32 cases were included in the qualitative analysis. Management is varied and often not described, resulting in an almost complete lack of information and indications for treatment. Radical surgery tends to yield the best outcomes, and it is recommended to have adequate surgical margins when possible.ConclusionsIntracranial involvement from AMBL compartment is an uncommon manifestation of this rare pathology, but which deserves to be treated in a multidisciplinary way in order to ensure maximum surgical radicality. Recurrence reflects failure of the primary surgical resection. If recurrence is the major consideration, surgeons are encouraged to select radical surgery. Whenever a follicular-type maxillary AMBL is diagnosed, it is advisable to check for intracranial spreading and distant metastases during follow-up.     

Rapidly growing intraparotid mass in a young child

Nodular fasciitis (NF) is a pseudosarcomatous lesion typically involving the subcutaneous tissue and fascia; rarely, it has been observed within a parotid gland. We report a case of NF in an 8-year-old boy who presented with a rapidly growing nodule in the right parotid gland. A preoperative fine-needle aspiration suggested a diagnosis of nodular (pseudosarcomatous) fasciitis. After fine-needle aspiration, the surgical excision of nodule with a conservative approach was made. Intraparotid NF was diagnosed by histopathologic examination confirming the former cytologic diagnosis. This case demonstrates that NF, although rarely may occur in unusual site as parotid gland, in cases of rapidly growing tumor masses in the parotid, a correct cytologic diagnosis in the fine-needle aspirate might spare unnecessary aggressive surgical treatment.     

Lethal invasive mucormycosis: case report and recommendations for treatment

A case of lethal invasive mucormycosis (IM), a rare fungal infection which predominantly affects immunocompromised patients, is reported in a 73-year-old female patient who presented with a cervical abscess. The patient had asthma treated with steroids and had previously undiagnosed diabetes mellitus. Despite surgical treatment and parenteral antibiotic therapy, there was fatal progression of the condition. The pathogenesis, histological appearances and treatment of mucormycosis are discussed, particularly the importance of urgent histological examination of debrided tissue to distinguish this condition from necrotizing fasciitis (NF) earlier than microbiological culture alone would allow, thus permitting the early introduction of appropriate antifungal therapy.     

Branchial cleft or cervical lymphoepithelial cysts: etiology and management

BACKGROUND: The cervical lymphoepithelial or branchial cleft cyst is a developmental cyst that has a disputed pathogenesis. The objective of this article is to provide a brief review of the literature and to define diagnostic terms related to this anomaly, as well as to describe its etiology, clinical presentation and treatment. CASE DESCRIPTION: The cervical lymphoepithelial or branchial cleft cyst usually presents as a unilateral, soft-tissue fluctuant swelling that typically appears in the lateral aspect of the neck, anterior to the sternocleidomastoid muscle, and becomes clinically evident late in childhood or in early adulthood. Clinicians can diagnose the cyst with appropriate imaging to assess the extent of the lesion before definitive surgical treatment. The authors describe a patient who underwent excision of a well-encapsulated cystic structure that was diagnosed as a branchial cleft cyst. CLINICAL IMPLICATIONS: The cervical lymphoepithelial or branchial cleft cyst can be easily misdiagnosed as a parotid swelling or odontogenic infection. It is imperative that clinicians make an accurate diagnosis so that appropriate treatment (that is, surgical excision) can be performed. If the cysts are treated properly, recurrences are rare.     

双侧冠突过长导致开口受限2例

冠突过长（CPH）是一种罕见的下颌骨发育过程中冠突形态和长度的异常疾病,本文报道2例双侧CPH引起下颌运动及开口受限的病例资料,通过报道病例的临床表现、X线检查、治疗及预后,为该疾病的诊疗提供参考。这2例CPH病例经外科切除伸长的冠突后,即刻开口度均增加到近三指,其中1例治疗6个月后,随访开口度正常,而另外1例术后2年CPH复发。CPH的病因暂不明确,可能与多种因素相关。CPH导致开口受限的情况在临床上极其少见,易与其他疾病混淆导致误诊。因此,应综合运用临床及影像学加以鉴别诊断。外科手术切除伸长的冠突是目前公认的治疗方式,而术后长期有效的开口训练对治疗效果的维持至关重要。     

Prosthetic management of a patient with Treacher Collins syndrome.

Treacher Collins syndrome encompasses a group of closely related defects of the head and neck. It is a rare syndrome characterized by bilaterally symmetrical abnormalities derived from the first and second brachial arches and the nasal placode. It is an autosomal dominant disorder and its occurence ranges from 1 in 25,000 to 1 in 50,000 live births. The facial appearance of these patients can be improved by either surgical or prosthetic rehabilitation. In this case report we are presenting the features of a 13-year-old boy with Treacher Collins syndrome. A multidisplinary approach was followed in managing the situation. The various treatment options and the steps involved in making an auricular prosthesis are also discussed.     

Subtotal and total resection of superficial plexiform neurofibromas of face and neck: four case reports.

INTRODUCTION: Plexiform neurofibromas are benign tumours of the peripheral nerves and connective tissue. They develop most often in patients with neurofibromatosis type 1 (NF1) and often grow continuously. Removal of plexiform neurofibromas is usually unsatisfactory because the network-like growth of these tumours often involves multiple nerve fascicles and other adjacent tissues. It has been previously shown that magnetic resonance tomography can distinguish the growth patterns of plexiform neurofibromas into three different categories: superficial, displacing and invasive. PATIENTS AND METHODS: Three cases are described with successful subtotal resections of superficial plexiform neurofibromas, and one case with total resection following the diagnosis of tumour subtype using magnetic resonance imaging (MRI). RESULTS: There was a significant, lasting improvement in appearance which demonstrates that surgical intervention in the case of superficial plexiform neurofibroma is valuable. CONCLUSION: Careful classification of plexiform neurofibroma by means of MRI provides valuable information for the surgical management of patients. It enables the distinction to be drawn between this subtype and the other two subtypes of plexiform neurofibromas.     

Congenital infantile fibromatosis of the cheek: report of a rare case and differential diagnosis

Infantile fibromatosis is a benign tumour that consists of dense masses of fibroblasts and myofibroblasts with marked collagen production. Although it is a nonmetastasising tumour, it has significant potential for local invasion and recurrence and may be fatal because of its size and location. The authors report an unusual case of infantile fibromatosis of the cheek in an 8-month-old boy, present since birth. The patient underwent surgical treatment with tumour-free margins and the 6-month follow-up revealed no signs of recurrence. Infantile fibromatosis of the cheek is a very rare condition at this location, especially congenital cases. It should be considered in the differential diagnosis of congenital lesions in childhood.     

Neurofibromatosis type I with periodontal manifestation. A case report and literature review

The term neurofibromatosis (NF) is used for a group of genetic disorders that primarily affect the cell growth of neural tissues. Neurofibromatosis type 1 (NF1), also known as von Recklinghausen's disease, is the most common type of NF and accounts for about 90% of all cases. It is one of the most frequent human genetic diseases, with a prevalence of one case in 3,000 births. The expressivity of NF1 is extremely variable, with manifestations ranging from mild lesions to several complications and functional impairment. Oral manifestations can be found in almost 72% of NF1 patients. A case of a NF1 patient with a gingival neurofibroma in the attached gingiva of the lingual aspect of the lower central incisors is presented. The lesion was nodular, with sessile base, non-ulcerated, non-painful, with normal colour and measured 1 cm in diameter. An excisional biopsy of the oral lesion was performed. Histopathological and immunohistochemical analysis confirmed the clinical hypothesis of neurofibroma. Because NF1 is one of the most common genetic diseases and oral manifestations are very common, dentists should be aware of the characteristics of this disease.     

Primary oral malignant melanoma

Malignant melanoma of the oral cavity is a rare neoplasm. It is well known for his poor prognosis and the need for an evidence-based treatment. Therapy is commonly based on surgical excision of the primary tumour, supplemented by adjuvant therapy. In this paper two new cases of primary oral melanoma are reported with a review of the literature.     

面颈部血管滤泡性淋巴结增生临床病理研究

目的:结合文献复习,研究面颈部血管滤泡性淋巴结增生(AFH)的临床表现、病理形态特征。方法:报道了3例该病变,并进行临床、病理及免疫组化等回顾性分析。结果:临床分型主要有局灶型及多中心型2类,组织学分型主要有透明血管型、浆细胞型2型。本文3例均为局限性肿块、透明血管型,手术切除预后较好。文献指出,AFH是一种交界性的淋巴组织增生性病变,局灶型肿块为良性,而多中心型常为恶性结局。结论:手术切除肿大淋巴结是治疗本病的主要方法。单中心型多可通过手术达到治愈的目的,多中心型手术切除后有复发可能,需结合放疗化疗等治疗方法。对于多中心型AFH其生物学行为及治疗预后,还有待于进一步研究探讨。对于淋巴组织增生性病变,鉴别诊断免疫组化尤为重要。