Identification of Putative Bacterial Pathogens for Orofacial Granulomatosis Based on 16S rRNA Metagenomic Analysis.

Orofacial granulomatosis (OFG) is a chronic inflammatory disease characterized by nontender swelling of the orofacial tissues, the underlying cause of which remains unknown. Our previous study demonstrated that tooth apical periodontitis (AP) is involved in the development of OFG. To characterize the AP bacterial signatures of OFG patients and identify possible pathogenic bacteria that cause OFG, the compositions of the AP microbiotas in OFG patients and controls were compared using 16S rRNA gene sequencing. Pure cultures of putative bacterial pathogens were established by growing bacteria as colonies followed by purification, identification, and enrichment and then were injected into animal models to determine the causative bacteria contributing to OFG. A specific AP microbiota signature in the OFG patients was shown, characterized by the predominance of phyla Firmicutes and Proteobacteria, notably members of the genera Streptococcus, Lactobacillus, and Neisseria, were found. Streptococcus spp., Lactobacillus casei, Neisseria subflava, Veillonella parvula, and Actinomyces spp. from OFG patients were isolated and successfully cultured in vitro and then injected into mice. Ultimately, footpad injection with N. subflava elicited granulomatous inflammation. IMPORTANCE Infectious agents have long been considered to play a role in the initiation of OFG; however, a direct causal relationship between microbes and OFG has not yet been established. In this study, a unique AP microbiota signature was identified in OFG patients. Moreover, we successfully isolated candidate bacteria from AP lesions of OFG patients and assessed their pathogenicity in laboratory mice. Findings from this study may help provide in-depth insights into the role of microbes in OFG development, providing the basis for targeted therapeutic approaches for OFG.

Is orofacial granulomatosis a distinct clinical disorder?

Orofacial granulomatosis is a rare disorder that is heterogeneously defined in the published literature. Herein, we describe a patient with orofacial granulomatosis with clinical and histologic evidence, discuss differential diagnoses, and offer clinical pearls for diagnosing and assessing this disorder. Our case provides support that orofacial granulomatosis is a distinct disorder as opposed to a sequela of other systemic granulomatous diseases. This information will aid dermatologists in decision making and diagnosing the disorder.

Orofacial granulomatosis associated with gingival enlargement: a case report.

Orofacial granulomatosis (OFG) is an uncommon condition with varying clinical presentation. Gingival enlargement in children could be due to a varied etiology. The present case report is of an adolescent female with initial presentation of generalized gingival enlargement, lip swelling and perioral discoloration without any known etiopathological factors or systemic involvement. Conservative excision of the enlargement was performed and histopathological examination revealed a non caseating granulomatous lesion. Diagnosis of orofacial granulomatosis in context to sarcoidosis was arrived after excluding other granulomatous diseases. Follow up after 18 months showed no recurrence and regression of lip swelling and perioral discoloration. Gingival enlargement can be considered as one of the presenting features of sarcoidosis.

Orofacial Granulomatosis and Crohn Disease: Coincidence or Pattern? A Systematic Review.

BACKGROUND: To systematically review all cases of orofacial granulomatosis (OFG) and evaluate the association between OFG and Crohn disease (CD). SUMMARY: This review was conducted according to PRISMA guidelines and a search of the PubMed, MEDLINE, and Embase databases, and the Cochrane Library in March 2020, using keywords and MeSH terms associated with "orofacial granulomatosis," "Crohn disease," and their variants, with no language restrictions and across all age groups. All relevant articles were accessed in full text. Single case reports and articles on sarcoidosis, allergy, ulcerative colitis, and infectious diseases were excluded from the analysis. RESULTS: We retrieved 507 reports on OFG. The mean age at onset was 23.3 years (range 2-89 years). A total of 240 (47.3%) females and 267 (52.6%) males were included. CD was present in 93 children aged <16 years (68.3%) and in 43 adults (31.9%). In most cases, the OFG appeared before the CD. The most common clinical manifestations were intraoral mucosa abnormalities (n = 251; 49.5%), lower-lip swelling (n = 249; 49.1%), upper-lip swelling (n = 227; 44.7%), and gingivae (n = 193; 38.7%). Patients with concurrent CD were more likely to experience involvement of the buccal sulcus. Key Messages: OFG presents primarily as a solo entity. The OFG that was associated with CD was present in 93 children aged under 16 years (68.3%) and in 43 adults (31.9%). Childhood onset of OFG carries with it a higher risk of developing CD.

Granulomatous Periorificial Dermatitis in an Adult: A case report with review of literature.

Granulomatous periorificial dermatitis (GPD) is a benign, self-limiting eruption that is considered a clinical variant of periorificial dermatitis, also known as perioral dermatitis. It presents primarily in prepubertal children as monomorphic scaly papules over perioral, paranasal and periorbital areas of the face with rare occurrence in adults. We report a 36-year-old Omani male patient who presented to the Dermatology Clinic at Bahla Polyclinic, Bahla, Oman, in 2018 with a papular eruption over his face for the previous six months. Based on clinical and histopathological findings the patient was diagnosed with GPD with sarcoid-like histology. He was treated effectively with oral doxycycline and topical metronidazole. This report provides a review of the literature on GPD and summarises all reported cases in adults to date.

Oral pulse granuloma and oral pulse granuloma associated with odontogenic keratocyst: two clinical cases and a review of the literature / Granuloma oral pulse y granuloma oral pulse asociado a un queratoquiste odontogénico: 2 casos clínicos y revisión de la literatura

Two cases of oral pulse granuloma (OPG) or vegetable granuloma (VG) are presented, one of which was concomitant with an odontogenic keratocyst (OKC), which is an unusual finding. OKC is characterized by the presence of hyaline rings which include vessels, giant cells, other inflammatory cells and collagen fibres. There are two hypotheses as to its histogenesis: firstly, as a reaction to vegetable matter, such as legumes (thus the nomenclature "pulse" or edible seed) and secondly as a degenerative change in the vessel walls as a result of localized vasculitis. Due to the deceptive appearance of OPG, diagnosis can be challenging

En este artículo breve se presentan 2 casos de granuloma oral pulse (GOP) o granuloma vegetal, uno de ellos asociado a un queratoquiste odontogénico. Esta entidad está caracterizada por la presencia de estructuras hialinas en anillo que incluyen vasos, células gigantes, otras células inflamatorias y haces de fibras de colágeno. Sobre su origen todavía se barajan 2 hipótesis: una en la que se sospecha que se producen como reacción a estructuras vegetales como legumbres (de donde toma el nombre de «pulse» o semilla comestible de una leguminosa), y otra en la que se trataría de un cambio degenerativo de las paredes vasculares, resultado de una vasculitis localizada. Debido a la apariencia engañosa del GOP es fácil que a los patólogos les suponga un esfuerzo su diagnóstico. Se describe a continuación un hallazgo inusual de un GOP relacionado con un queratoquiste odontogénico

Non-infectious Granulomatous Lesions of the Orofacial Region.

Granulomatous lesions of the orofacial region are a heterogeneous group of disorders characterized by a granulomatous reaction to a variety of stimuli. Infectious agents, foreign material, systemic inflammation and metabolic disorders can all be associated with granulomatous inflammation. In the orofacial region primary causes of granulomatosis include foreign body reaction, delayed hypersensitivity to topical agents and idiopathic orofacial granulomatosis. Secondary causes of granulomas include infectious agents, sarcoid, and Crohn disease. For this review, infectious causes of orofacial granulomatosis (OFG) including bacteria, parasites and fungi will not be discussed.

Orofacial granulomatosis and diet therapy: a review of the literature and two clinical cases.

Orofacial granulomatosis is a nonspecific term that contains a wide variety of granulomatous entities, which share a clinical and histopathological presentation. It manifests as persistent or recurrent orofacial swelling, amongst other findings. Idiopathic orofacial granulomatosis, characterized by an absence of systemic granulomatous disease, is a diagnosis of exclusion. The main differential diagnosis is Crohn's disease. Its pathogenesis is unknown, however, it seems to be immune-mediated. Patch-test sensitivity to multiple allergens is well documented. Currently, therapeutic options consider restrictive diets, topical, intralesional, and systemic agents. First-line therapy is currently a matter of debate. We present a review of the value of diet therapy in this syndrome, along with two illustrative cases.

Orofacial granulomatosis and diet therapy: a review of the literature and two clinical cases

Abstract: Orofacial granulomatosis is a nonspecific term that contains a wide variety of granulomatous entities, which share a clinical and histopathological presentation. It manifests as persistent or recurrent orofacial swelling, amongst other findings. Idiopathic orofacial granulomatosis, characterized by an absence of systemic granulomatous disease, is a diagnosis of exclusion. The main differential diagnosis is Crohn's disease. Its pathogenesis is unknown, however, it seems to be immune-mediated. Patch-test sensitivity to multiple allergens is well documented. Currently, therapeutic options consider restrictive diets, topical, intralesional, and systemic agents. First-line therapy is currently a matter of debate. We present a review of the value of diet therapy in this syndrome, along with two illustrative cases.

Granuloma vegetal oral: a propósito de un caso / Oral pulse granuloma: a case report

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[Manifestation of Crohn's disease in the oral cavity: clinical case]. / Debiut bolezni Krona v polosti rta. Klinicheskii sluchai.

The article contains the review of possible oral manifestations of Crohn's disease and clinical case presentation of purulent alveolitis resistant to conventional local treatment, geographical glossitis and xerostomia in 42 y.o. female with the history of periodical digestive disorders, diagnosed with Crohn's disease. The diagnosis was based on abovementioned oral manifestations.

Orofacial granulomatosis in a 12-year-old girl successfully treated with intravenous pulse corticosteroid therapy and chloroquine.

Orofacial granulomatosis, a rare disease in childhood, is characterized by orofacial swelling in the absence of systemic disease. We report the case of a 12-year-old girl with asymptomatic erythematous infiltration of her upper lip, cheeks, and chin that had persisted for more than 2 years; biopsy confirmed granuloma formation. Because a large area was affected, intralesional corticosteroids were inappropriate and six cycles of 3-day intravenous pulse corticosteroid therapy (dexamethasone 1.5mg/kg), repeated once after 4 weeks, was given. Our patient also received oral chloroquine and topical emollients. At the end of the sixth pulse cycle, the infiltration had completely resolved, leaving slight residual erythema.

Effectiveness of surgical treatment of severe macrocheilia in a patient with orofacial granulomatosis.

Orofacial granulomatosis (OFG) is the term given to a group of diseases characterized by the presence of non-necrotizing granulomatous inflammation affecting the soft tissues of the orofacial region. Treatment of OFG is often challenging and unsatisfactory. We report on a 32-year-old man with a 2-year history of oedema and swelling of the upper lip without systemic symptoms. The history, clinical features and histopathological findings led to the diagnosis of cheilitis granulomatosa (CG), a disease included in the spectrum of OFG. The patient was treated with oral diaminodiphenyl sulfone (DDS) and clofazimine without success. Oral doxycycline led to a slight improvement of the disease. Because the volume of the upper lip was twice normal size, surgical reduction was performed, followed by administration of oral doxycycline for 3 months. This therapeutic approach led to complete remission, with no recurrence after 3 years.

Orofacial granulomatosis: A disease or a concealed warning.??

Granulomatosis is any condition characterized by the formation of multiple nodules or granulomas in the soft tissues. Differential diagnosis for orofacial region includes a wide spectrum of diseases, but most of these lesions present histopathologically as noncaseating granulomas, giving a nonspecific depiction and leading to a diagnostic impasse. In the absence of any diagnosable entity, the disease is labelled as "orofacial granulomatosis". A nine-year-old girl child reported with recurrent gingival enlargement and persistent macrochelia which histopathologically presented as noncaseating granulomas. The disease was progressive raising the suspicion of being oral manifestation of a systemic disorder such as Sarcoidosis or Crohn's disease. This paper throws some light on this rare entity and reports rarer features of this disease, like eye involvement and staphylococcal mucositis in the case report.

Histopathological Features of Orofacial Granulomatosis.

Orofacial granulomatosis (OFG) is characterized by granulomatous inflammation in the orofacial region. Although several series have been reported, biopsy has not been performed in all cases and the histopathological features have not been extensively evaluated. Our purpose was to analyze the histopathological features of our patients with OFG. Twenty-two patients diagnosed with OFG at Bellvitge Hospital (Barcelona, Spain) from 1985 to 2010 were included in the study. All of our patients (14 men and 8 women; mean age 46.77 years, SD 13.61) presented with labial swelling, involving the upper lip in 13 cases and the lower lip in 9 at presentation. Fissured tongue was observed in 9 cases. Three patients suffered facial palsy. Granulomas were observed in 16 patients. All patients showed a perivascular, lymphohistiocytic inflammatory infiltrate with prominent plasma cells in 21 cases. In 5 cases, mast cells were easily observed. In 13 patients, an interstitial infiltrate was also present. All cases showed dilated lymphatic channels, and 19 edema of the lamina propria or the dermis. Perilymphatic granulomas were observed in 12 cases, intralymphatic granulomas in 2, and intralymphatic histiocytes in 2. In conclusion, loose epithelioid cells or lymphonodular granulomas were observed in 73% of our patients. Although none of our patients developed Crohn disease, OFG is histopathologically similar to cutaneous lesions of Crohn disease. The perilymphatic disposition of granulomas and the presence in some cases of intralymphatic histiocytes or intralymphatic granulomas may explain the dilatation of lymphatic vessels and the development of edema.