Papillon-lefèvre syndrome: clinical case of oral rehabilitation with osseointegrated implants

Papillon-Lefèvre syndrome (PLS) is a rare benign, autosomal recessive condition caused by a mutation in the cathepsin C gene. This alteration results in palmar-plantar hyperkeratosis, or thickening of the soles of the feet and palms of the hands, as well as aggressive periodontal disease resulting in the premature loss of deciduous and permanent teeth. The etiology is multifactorial, and is influenced by immunological, genetic or microbial factors. This case report presents a successful prosthetics oral rehabilitation with osseointegrated implants in a 48-year-old female patient with PLS. Although few reports of dental implants in patients with PLS have been published, it seems to be a possible approach in selected patients. (AU)

Medication-related osteonecrosis in torus palatinus: Report of a case and literature review.

BACKGROUND: Medication-related osteonecrosis of the jaw bones have been frequently reported. However, its occurrence in torus palatinus is very rare with only 10 cases published in the English-language literature. CASE REPORT: We describe an additional case in a 79-year-old woman, who was referred for evaluation of a painful swelling with areas of suppuration on the hard palate. CONCLUSION: Conservative treatment was performed and after spontaneous sequestrectomy, total healing was achieved.

The importance of early treatment of oral squamous cell carcinoma: Case report.

BACKGROUND: Oral squamous cell carcinoma (OSCC) is frequently diagnosed at an advanced stage, with the five-year survival rate varying around 50%, however, ifs the sooner OSCC in early-stage is treated, the lower the chances of recurrence and death. In this study, we described an OSCC diagnosed early, highlighting how the early surgical excision prevents the morbidity. CASE REPORT: A 74-year-old man presented a whitish and symptomatic plaque and a discreet ulcerated area on the right lateral border of the tongue. Due to close contact with the second inferior molar, a clinical diagnosis of a frictional keratosis was suspected, however as the lesion persisted after two weeks of follow-up, an incisional biopsy was performed and the microscopic features revealed an OSCC. Early surgical resection was performed, preventing the morbidity and improve benefits to patient. CONCLUSION: OSCC can manifest of several clinical forms, including indolent appearances, which can delay the diagnosis, and consequently the prognosis. Further, it is important to recognize and not neglect any intraoral lesions, in order to avoid the far more invasive surgery that a more advance disease would imply.

Clinicians' perception of oral potentially malignant disorders: a pitfall for image annotation in supervised learning.

OBJECTIVE: The present study aims to quantify clinicians' perceptions of oral potentially malignant disorders (OPMDs) when evaluating, classifying, and manually annotating clinical images, as well as to understand the source of inter-observer variability when assessing these lesions. The hypothesis was that different interpretations could affect the quality of the annotations used to train a Supervised Learning model. STUDY DESIGN: Forty-six clinical images from 37 patients were reviewed, classified, and manually annotated at the pixel level by 3 labelers. We compared the inter-examiner assessment based on clinical criteria through the κ statistics (Fleiss's kappa). The segmentations were also compared using the mean pixel-wise intersection over union (IoU). RESULTS: The inter-observer agreement for homogeneous/non-homogeneous criteria was substantial (κ = 63, 95% CI: 0.47-0.80). For the subclassification of non-homogeneous lesions, the inter-observer agreement was moderate (κ = 43, 95% CI: 0.34-0.53) (P < .001). The mean IoU of 0.53 (±0.22) was considered low. CONCLUSION: The subjective clinical assessment (based on human visual observation, variable criteria that have suffered adjustments over the years, different educational backgrounds, and personal experience) may explain the source of inter-observer discordance for the classification and annotation of OPMD. Therefore, there is a strong probability of transferring the subjectivity of human analysis to artificial intelligence models. The use of large data sets and segmentation based on the union of all labelers' annotations holds the potential to overcome this limitation.

Prognostic outcomes of oral squamous cell carcinoma derived from proliferative verrucous leukoplakia: A systematic review.

OBJECTIVE: This study aimed to evaluate prognostic outcomes of PVL-derived oral squamous cell carcinomas (P-OSCC) based on recurrence, new primary tumour, metastasis and survival information. STUDY DESIGN: Five databases and grey literature were searched electronically with the following main keywords (proliferative verrucous leukoplakia, squamous cell carcinoma and malignant transformation) to answer the following review question: 'Are survival outcomes for P-OSCC worse?' based on the PECOS principle. The Joanna Briggs Institute Critical Appraisal tool was used to identify possible biases and assess the quality of each of the primary studies. RESULTS: A total of 21 articles met the inclusion criteria, and the results of this systematic review suggest that P-OSCC can recur and generate new primary tumours; however, metastases are rare. Thus, most patients remain alive for an average period of 5 years. CONCLUSION: Apparently, P-OSCC has better clinical prognostic characteristics than conventional OSCC. There is a lack of information on the main prognostic outcomes of P-OSCC; therefore, specific studies must be performed to achieve a better comparison between P-OSCC and conventional OSCC progression.

A Rare Case of Mandibular Aspergillus Osteomyelitis in an Immunocompetent Patient.

Aspergillosis is a fungal infection caused by Aspergillus species, which is contracted through spores that colonize the respiratory tract, causing rhinosinusitis and pulmonary infections. Oral aspergillosis is rare and, when present, may cause soft tissue and bone destruction, generally in immunodeficient patients. Mandibular Aspergillus osteomyelitis is even rarer, with few cases reported in the literature. A 57-year-old Caucasian woman was referred for the evaluation of painful recurrent swelling in the anterior mandibular alveolar ridge, with purulent drainage, previously treated with multiple surgical debridement procedures and antibiotics without success. The patient was otherwise systemically healthy. Surgical debridement was performed and histopathological examination showed osteomyelitis associated with Aspergillus species. Therapy with oral itraconazole (400 mg per day) was administered for 3 months, resulting in complete resolution. No recurrence was detected after 15 years of follow-up. The patient was rehabilitated with dental implants. In conclusion, non-bacterial microorganisms, such as Aspergillus, should be considered in cases of mandibular osteomyelitis that do not heal after surgical debridement and antibiotic therapy.

Adenocarcinoma not otherwise specified (NOS) arising in the sublingual gland: Rare case report and follow-up.

BACKGROUND: Adenocarcinoma not otherwise specified (AdCaNOS) is a rare malignant salivary gland tumor that occurs with higher frequency in the parotid gland of male patients. In this study, we described a second case of AdCaNOS arising in the sublingual gland, in a female patient with 39 years of age, highlighting the clinical, radiographic, microscopic, treatment features and follow-up. CASE REPORT: A 39-year-old female patient presented a fibroelastic nodule with a yellowish coloration, in the left region of the floor of mouth, measuring about 4.0 cm in its largest diameter. An incisional biopsy was performed and the main microscopic features revealed an infiltrative lesion with glandular differentiation organized in cystic spaces, where neoplastic cells secreting eosinophilic material were observed. The tumor showed immunopositivity for pancytokeratin (AE1/AE3), keratins 7 (CK7) and 14 (CK14), and negativity for p63. The proliferation level measured by Ki-67 marker was considered higher. The patient underwent radical surgical resection, but unfortunately, she developed local recurrence, lymph node mestastasis and died 1.5 year after diagnosis. CONCLUSION: Although rare in the sublingual gland, particularly at this age, AdCaNOS can occur and early diagnosis and early treatment are essential for a better prognosis and survival rates of the patients.

Head and neck radiotherapy leading to extensive late oral soft-tissue necrosis.

Radiotherapy is generally used as an adjuvant treatment in malignant head and neck tumors, which can cause a series of toxicities to tissues involved in the radiation field. Hard tissue necrosis surrounding the tumor is widely debated and recognized; however, oral mucosa necrosis as a late effect of head and neck radiotherapy is a toxicity that has been little explored and understood in the literature. The present report describes a 53-year-old Caucasian, diabetic man with a painful yellowish mass in the buccal mucosa, remaining oral mucositis with history of radiotherapy completed 90 days ago as adjuvant treatment for a polymorphic adenocarcinoma in the palate. Photobiomodulation was used as therapy for tissue necrosis with good therapeutic response despite discontinuation of treatment by the patient. Since there are few descriptions and illustrations of radiotherapy-related soft tissue necrosis, the current case may bring some new experience with this important topic, which directly impacts on the patient's quality of life.

Langerhans cell histiocytosis in the oral and maxillofacial region: An update.

BACKGROUND: Langerhans cell histiocytosis (LCH) is a rare disorder characterized by abnormal proliferation and infiltration of histiocytic cells. METHODS: This review focused on the main aspects associated with LCH. RESULTS: LCH can involve single or multiple organs and systems, with bone and skin being the most commonly affected sites. Regarding skeletal manifestations, the jawbones are involved in about 20%-30% of all cases. Such lesions may present as unilocular or multilocular images mainly affecting the posterior mandible. Oral soft tissue lesions may also occur, with the gingiva and hard palate being the most frequently affected sites. CONCLUSION: The diagnosis and management of LCH are challenging, requiring a multidisciplinary approach, with dentists playing a central role since oral manifestations can be the first sign of the condition.

Lingual necrosis leading to the diagnosis of giant cell arteritis.

Giant cell arteritis (GCA) is a chronic vasculitis comprising large- and medium-sized arteries branches originating from the aortic arch. GCA diagnosis might be challenging due to its several clinical characteristics. This clinical report describes a case of a 91-year-old woman who was admitted with tongue necrosis. Medical history revealed mild headache episodes and jaw numbness. Summing up clinical and laboratory findings, the diagnosis of GCA was established. Surgical debridement and corticotherapy led to complete resolution of the tongue lesion with no need for any additional intervention. In this case report, we highlight the importance of GCA early diagnosis in order to avoid more serious complications.

Candidiasis of the tongue in cannabis users: a report of 2 cases.

Cannabis, a psychoactive drug widely used for medicinal, recreational, and religious purposes, can have detrimental effects on several body systems, including the respiratory, cardiovascular, and central nervous systems. The use of cannabis in cigarette form can produce a series of oral alterations, including periodontitis, caries, xerostomia, a decreased salivary pH, and an increase in the density of Candida albicans. However, the occurrence of oral candidal lesions related to cannabis use is little reported in the literature. This article reports 2 cases of oral candidiasis associated with cannabis use. The adult male patients, both of whom were systemically healthy, had white and red spots consistent with oral candidiasis on the dorsal surface of the tongue. One of the patients also had a red lesion on an area of the hard palate that was in contact with the affected area of the tongue. Neither patient was currently undergoing antibiotic or corticosteroid treatment, and both reported frequent smoking of cannabis. One patient was initially treated with an oral suspension of nystatin without clinical improvement. Miconazole gel therapy was then prescribed, and clinical improvement was observed after 2 weeks. The patient did not stop smoking cannabis, and a recurrence of oral candidiasis was observed 6 months posttreatment. Treatment with miconazole gel was repeated, resulting in resolution of the infection. The second patient declined treatment. The reported cases demonstrate that, although it is infrequently reported, oral candidiasis may occur in cannabis smokers.

Morphological patterns of circumpulpal dentin affected by radiation-related caries.

BACKGROUND: The aim of this work was to evaluate the microscopic characteristics through polarized light microscopy, scanning electron microscopy and the mineral content of circumpulpal dentin of irradiated (IT) and non-irradiated teeth (NIT), with deep caries that reached the root canal. MATERIAL AND METHODS: A total of 25 IT were analyzed macroscopically, and radiographed. 5 NIT were used as controls. Two 100-µm-thick sections, per specimen, were evaluated in a polarized light microscopy and a scanning electron microscope. Demographics and clinical data were collected. RESULTS: The results did not demonstrate distinct morphology of the IT compared with NIT. Mineral content values by weight percentage of Ca and P were also similar, corresponding to 66.65% and 33.21% in the IT and 66.60% and 33.29% in the NIT. The Ca/P ratio did not show statistical differences between groups being respectively 2.74 and 2.72, in the IT and NIT (p> 0.05). CONCLUSIONS: Radiotherapy does not change morphology and mineral content of circumpulpal dentin in IT. Key words:Root canal, radiation-related caries, polarization microscopy, scanning electron microscopy, radiotherapy, oral neoplasms, dentin.

Orthodontic treatment in a patient with cherubism: Benefits and limitations.

AIM: Cherubism is an uncommon hereditary disease that leads to the development of giant cell lesions in the jaws, alterations in the dentition, and malocclusion. The biological behavior of bones to orthodontic forces in these patients is not described in the literature, leading dentists to avoid this management. The aim of this article was to describe a case report of management with orthodontics. We present details regarding clinicoradiographic features, diagnosis, treatment, and follow-up. CASE REPORT: A 12-year-old male patient diagnosed with cherubism presented to our service with complaints about his esthetic facial and dental appearance. Management was interdisciplinary, including careful and controlled orthodontic treatment. The results were satisfactory; alignment, dental leveling, and correction of the malocclusion were achieved. CONCLUSION: Patients with cherubism may benefit from orthodontics, improving oral function, and esthetic and psychosocial well-being. The orthodontic treatment might be performed according to the severity of clinical manifestation, expectations of the patients, and limitations of each case.

Oral carcinoma development after 23 years of renal transplantation

Renal transplant patients are treated with immunosuppressive drugs that decrease the effectiveness of the immune system, making them more prone to developing cancer. Skin and lip carcinomas are common malignancies encountered after transplantation, whereas oral carcinomas are rare. We report the case of a 51-year-old female Caucasian patient, with no history of smoking, who presented white lesions on the tongue and an ulcerated lesion on the lower lip beginning 4 months prior. Diagnosis of squamous cell carcinoma for both lesions was made following incisional biopsies. Interestingly, the patient reported a renal transplantation 23 years prior, and was maintained on a combination of cyclosporine, mycophenolate sodium and prednisone. The patient also presented a history of several basal and squamous cell carcinomas on sun-exposed areas of the skin. Both lesions were surgically excised. No sign of recurrence or new lesions in the oral cavity have been observed; however, new skin lesions are frequently diagnosed. This case report highlights that oral cancers may occur in transplant patients in the absence of classical risk factors. Thus, clinicians must be aware of the importance of thorough oral examination in transplant patients in routine follow-up.

Oral carcinoma development after 23 years of renal transplantation.

Renal transplant patients are treated with immunosuppressive drugs that decrease the effectiveness of the immune system, making them more prone to developing cancer. Skin and lip carcinomas are common malignancies encountered after transplantation, whereas oral carcinomas are rare. We report the case of a 51-year-old female Caucasian patient, with no history of smoking, who presented white lesions on the tongue and an ulcerated lesion on the lower lip beginning 4 months prior. Diagnosis of squamous cell carcinoma for both lesions was made following incisional biopsies. Interestingly, the patient reported a renal transplantation 23 years prior, and was maintained on a combination of cyclosporine, mycophenolate sodium and prednisone. The patient also presented a history of several basal and squamous cell carcinomas on sun-exposed areas of the skin. Both lesions were surgically excised. No sign of recurrence or new lesions in the oral cavity have been observed; however, new skin lesions are frequently diagnosed. This case report highlights that oral cancers may occur in transplant patients in the absence of classical risk factors. Thus, clinicians must be aware of the importance of thorough oral examination in transplant patients in routine follow-up.