Serena Cocca

A rare case of osteonecrosis of the jaw related to imatinib

Osteonecrosis of the jaw (ONJ) is commonly described as an adverse effect of the use of bisphosphonates. A few cases of ONJ associated with tyrosine kinase inhibitors (sunitinib, imatinib) have been reported in the literature and usually they occurred in patients simultaneously treated with bisphosphonates. We report an atypical case of ONJ related only to imatinib. A 72-year-old male patient was treated with imatinib for metastases from gastrointestinal stromal tumors (GISTs). The patient developed ONJ after 22 months of imatinib only therapy. During his whole life, the patient had never been treated with bisphosphonates or radiotherapy. Microscope examination of the tissues confirmed the clinical diagnosis of diffuse osteonecrosis and showed absence of neoplastic cells. Thus, secondary localisations from GISTs were ruled out. Osteonecrosis of the lower jaw appeared 22 months after initial and exclusive therapy with imatinib. Therefore, imatinib monotherapy can induce ONJ in patients that have never been treated with bisphosphonates or radiotherapy.

A case of bisphosphonate-related osteonecrosis of the jaw with a particularly unfavourable course: a case report

Bisphosphonates are drugs used to treat osteoclast-mediated bone resorption, including osteoporosis, Paget disease, multiple myeloma, cancer-related osteolysis, and malignant hypercalcemia. The use of these drugs has increased in recent years as have their complications, especially bisphosphonate-related osteonecrosis of the jaw (BRONJ), which more frequently affects the mandible. Here we report a case of BRONJ with a particularly unfavorable course due to cervical inflammation that developed into necrotizing fasciitis, followed by multiorgan involvement leading to septic shock and death.

Rhinosinusal Inflammation and High Mobility Group Box 1 Protein: A New Target for Therapy.

Background/Aims: High mobility group box 1 (HMGB1) is a protein belonging to the class of damage-associated molecular pattern molecules, which activates innate immunity and powerful inflammatory factors. The aim of this review is to show the importance of HMGB1 in the pathogenesis of nasal inflammatory diseases and to suggest that inhibition of HMGB1 may be an innovative therapeutic target. Methods: We used immunohistochemistry to study whether HMGB1 increases in chronic rhinosinusitis with nasal polyps and whether its expression is associated with eosinophils and inflammatory cytokines. Using primary cultures of human nasal epithelial cells, we localised lipopolysaccharide-induced active translocation and release of HMGB1 by immunofluorescence assay and Western blot. Results: Patients with severe symptoms have the highest HMGB1 serum levels. Glycyrrhetic acid inhibits the chemotactic and mitogenic function of HMGB1, binding to the hydrophobic residues that delimit the pockets in box A and B. Conclusions: Chronic inflammatory diseases of the nose and paranasal sinuses are increasingly prevalent and are a financial burden for society. HMGB1 has been shown to play a role in several inflammatory diseases of otolaryngological interest. The inhibition of HMGB1 may be an innovative therapeutic target for patients with chronic upper airway inflammatory diseases having nasal obstruction as a major symptom.

Gingival Leiomyosarcoma in a Young Woman: Case report and literature review

Leiomyosarcoma (LMS) is a rare mesenchymal malignancy, of which 3-10% of cases occur in the head and neck region. We report a 22-year-old woman who was referred to the University Hospital of Siena, Italy, in 2016 with an ostensibly benign asymptomatic lump on the mandibular gingiva. The lesion grew rapidly, causing otalgia in the right ear. An excisional biopsy was performed and primary LMS was diagnosed histologically. Subsequently, the patient underwent radical re-excision of the perilesional mucosa, a partial bone resection and the extraction of four teeth. No recurrences or metastases were detectable at a 20-month follow-up. This report discusses the differential diagnosis of LMS with regards to other benign and malignant lesions and reviews the recent literature on primary and secondary oral LMS. Due to its innocuous clinical features-including its asymptomatic nature and presentation at a young age-this aggressive malignancy can go undetected; therefore, an early histopathological diagnosis is crucial.

HMGB1 in the Pathogenesis of Nasal Inflammatory Diseases and its Inhibition as New Therapeutic Approach: A Review from the Literature

Introduction  This study is a systematic review on recent developments about the importance of HMGB1 protein in the pathogenesis of rhino-sinusal inflammatory diseases. We also report data on the use of 18-β-glycyrrhetic acid (GA), which has been shown able to inhibit the pro-inflammatory activities of HMGB1, in young patients affected by allergic rhinitis and complaining of nasal obstruction as main symptom. Objectives  The objective of this study was to review the literature to demonstrate the importance of HMGB1 in the pathogenesis of nasal inflammatory disorders and understand whether the inhibition of this protein may be an efficacious and innovative therapeutic strategy for patients with rhino-sinusal inflammation. Data Synthesis  Authors searched for pertinent articles indexed in PubMed, Scopus, and other health journals between 2004 and 2015. In total, the authors gathered 258 articles: 219 articles through Pubmed and 39 articles from other search engines. The search terms used were as follows: HMGB1 AND “respiratory epithelium,” “airway inflammation,” “rhinitis,” “allergic rhinitis,” “rhinosinusitis,” “nasal polyposis,” “glycyrrhetic acid,” “children.” Conclusions  Patients with severe symptoms have the highest serum levels and the highest extracellular expression of HMGB1. GA inhibits HMGB1 chemotactic and mitogenic function by a scavenger mechanism on extracellular HMGB1 accumulation stimulated by lipopolysaccharides in vitro. Treatment of allergic rhinitis with GA is not associated with local or systemic side effects in children and adults.

Multicentric study on the efficacy and tolerability of Streptococcus salivarius 24SMB and Streptococcus oralis 89a in respiratory tract infections

Abstract BACKGROUND. Bacteriocins are peptides with antimicrobial efficacy produced by certain bacterial species. Probiotics indeed seem a promising method in the prevention of upper respiratory infections and our study would like to contribute to the results available in the literature, in order to underlie their true therapeutic potential role. MATERIAL AND METHODS. Our multicenter pilot prospective study investigates 366 patients from September 2015 to February 2016. All the patients were treated with a topical device made up of a suspension of two specific bacterial strains: Streptococcus salivarius 24SMB and Streptococcus oralis 89a to be administered as nasal spray. The nasal spray was administered twice daily for 7 days per month for three consecutive months. A questionnaire about the subjective efficacy of the therapy correlated to an improvement of symptoms was also collected from patients. RESULTS. After one year from the enrolment, a 70.07% reduction in the number of events compared with the number of expected episodes was observed. CONCLUSION. The aim of our data is to propose a new therapeutic approach to treat the recurrence of upper airway infection and to support an adequate therapy in all cases where the traditional antibiotic therapeutic protocol did not obtain completely efficient results in terms of recurrence.

Multiple brain abscess after professional tooth cleaning: case report and literature review

Brain abscess (BA) is an encapsulated infection and can be the consequence of head and cranio-maxillo-facial surgery or trauma, or may be secondary to cavernous thrombophlebitis, but is most frequently due to hematogenous septic dissemination from an adjacent site of infection, such as the paranasal sinuses, middle ear or oral cavity. We report a rare and unfortunate case of multiple BA caused by dental procedures in a young man with undiagnosed patent foramen ovale (PFO). Simple routine dental procedures, such as tooth brushing and professional oral hygiene, can predispose to life-threatening conditions. This case report and literature review highlights that multiple BA after professional tooth cleaning is extremely rare, but cardiac defects (in first place PFO which is a potential source of paradoxical embolism) promote BA formation.

Blastic plasmacytoid dendritic cell neoplasm: a rare case of gingival lesion with leukaemic presentation

Blastic plasmacytoid dendritic cell neoplasm (BPDCN) is a rare haematological malignancy with an aggressive clinical course. It has been recognised as a distinct entity in the WHO 2008 classification of haematolymphoid neoplasm. This disease usually presents with cutaneous involvement as the first manifestation, with subsequent or simultaneous spread to bone marrow and peripheral blood with leukaemic dissemination. Gingival lesion as the first manifestation, in the absence of a cutaneous lesion, is an uncommon presentation of this rare disease. We report a case of an elderly woman, aged 84 years, who presented with a lump on the mandibular gingiva and ipsilateral otalgia without any cutaneous lesion and associated with an highly aggressive and rapid leukaemia. This case is an addition to the handful of those cases of BPDCN which presents without cutaneous involvement, but it is exceedingly rare for clinical features that make it a unique case in the literature.

Stevens-Johnson syndrome and abuse of anabolic steroids

Stevens-Johnson syndrome (SJS) is characterized by mucocutaneous tenderness and typical hemorrhagic erosions, erythema and epidermal detachment presenting as blisters and areas of denuded skin. SJS is often observed after drug use as well as after bacterial or viral infections. Several drugs are at high risk of inducing SJS, but there are no cases in the English literature regarding anabolic steroid use triggering SJS. In our paper, we describe a case in which use of anabolic androgenic steroids (AAS) was associated with SJS. The patient participated in competitive body-building and regularly took variable doses of AAS. Initial symptoms (headache, weakness, pharyngodynia, and fever) were ignored. After a week he presented to the Emergency Department with a burning sensation on the mouth, lips, and eyes. Painful, erythematous, maculopapular, and vesicular lesions appeared all over the body, including on the genitals. During hospitalization, he also developed a cardiac complication. The patient had not taken any drugs except AAS.

Unusual complications caused by lipoma of the tongue

Lipoma is the most common, benign, soft tissue, mesenchymal tumour and is composed of mature adipose tissue. It is infrequent in the oral cavity (approximately 0.3% of all tongue neoplasias). We describe the case of a 68-year-old man with a swelling at the tongue edge and tongue dysesthesia. Medical history, clinical assessment, radiographic images, and cytological analysis enabled specialists to classify this neoplasia as a lipoma. The patient recovered fully after surgical excision of the affected area, and the neuralgic symptoms regressed. Surgical excision is an elective treatment; however, accurate differential diagnosis, histological examination, and follow-up are required.