Idiopathic Orofacial Granulomatosis with Varied Clinical Presentation

Ravindran, Rathy; Karunakaran, Anila

doi:10.1155/2013/701749

Cited by 8 publications

(17 citation statements)

References 8 publications

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“…Andrew Zbar et al in their recent review stated that a frank labial enlargement, exfoliative angular stomatitis and oral ulcerations clinically distinguish OFG from CD. These authors have advocated a complete evaluation for underlying systemic conditions in those OFG cases showing gingival enlargement alone [6]. In our case, suspected leukemia induced gingival hyperplasia was refuted by a normal bone marrow aspirate.…”

Section: Discussionmentioning

confidence: 68%

“…An increase incidence of orofacial granulomatosis is seen globally, particularly in the pediatric age group [6]. However, despite advanced diagnostic techniques, it is considered to be a diagnosis of exclusion [3,5,8,9].…”

Section: Discussionmentioning

confidence: 99%

“…The most common clinical symptom is persistent labial enlargement which is non-pitting, and non-tender, involving upper and/ or lower lips [6]. The other oral and facial manifestations include oral ulcers, fissured tongue, mucosal tags, gingival enlargement, facial nerve palsy, facial swelling and cervical lymphadenopathy [2,8].…”

Section: Discussionmentioning

confidence: 99%

“…There are a number of theories, both genetic and immunological, which attempt to explain the course and clinical outcome of this disease [6]. Current research in OFG supports an immunologic or allergen triggering factor as the chief aetiological basis [10].…”

Section: Discussionmentioning

confidence: 99%

“…Orofacial granulomatosis (OFG) is a term used to describe granulomatous lesions affecting the orofacial tissues in the absence of provable systemic granulomatous conditions such as sarcoidosis or Crohn's disease [2]. Till date, some authors consider OFG to be a non-specific disease because of several overlapping clinical and histopathological features with other granulomatous conditions, in particular with Crohn's disease [3][4][5][6]. The unknown etiology of both orofacial granulomatosis and Crohn's disease further challenges efforts in categorizing the disease entities [2,7].…”

Section: Introductionmentioning

confidence: 99%

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Idiopathic orofacial granulomatosis: A case report

Dhupar¹,

M.²,

Spadigam³

et al. 2016

IJCRI

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International Journal of Case Reports and Images (IJCRI) is an international, peer reviewed, monthly, open access, online journal, publishing high-quality, articles in all areas of basic medical sciences and clinical specialties.Aim of IJCRI is to encourage the publication of new information by providing a platform for reporting of unique, unusual and rare cases which enhance understanding of disease process, its diagnosis, management and clinico-pathologic correlations. Orofacial granulomatosis (OFG) is a lesser known disease entity, which presents a confounding spectrum of oral manifestations. It is a non-specific granulomatous inflammation caused by an elusive etiopathogenesis. It usually manifests as persistent and/or recurrent upper and/or lower labial enlargement. The clinical features, both extraoral and intra-oral, are highly variable and at times, insidious. This disease can be both cosmetically and functionally debilitating. Case Report: This is a report of an unusual case of orofacial granulomatosis which presented as a persistent severe generalized gingival enlargement in a nine-year-old child. There was no evidence of an underlying allergic or systemic cause. Surgical intervention (i.e., gingivectomy) showed no alleviation of symptoms. Conclusion: Making a prompt and precise diagnosis of orofacial granulomatosis is often challenging, however complete remission is possible with targeted therapy.

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Section: Discussionmentioning

confidence: 68%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

Idiopathic orofacial granulomatosis: A case report

Dhupar¹,

M.²,

Spadigam³

et al. 2016

IJCRI

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Orofacial Granulomatosis

Nemade¹,

Shinde²

2021

Granulomatous Diseases in Otorhinolaryngology, Head and Neck

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Orofacial granulomatosis: Clinical and therapeutic features in an Italian cohort and review of the literature

Galdiero

Maio

Arcoleo

et al. 2021

Allergy

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Background Orofacial granulomatosis (OFG) is characterized by granulomatous inflammation of the soft tissues of maxillofacial region. We explored OFG patients from 10 different Italian centers and summarized the most recent literature data. Methods A review of patients with OFG was carried out. An extensive online literature search was performed to identify studies reporting diagnosis and management of OFG. Results Thirty‐nine patients were recruited between January 2018 and February 2020. Most of them (97.4%) displayed involvement of the lips, and 28.2% suffered from Melkersson‐Rosenthal syndrome. Two patients received diagnosis of CD and one patient of sarcoidosis, suggesting secondary OFG. Oral aphthosis and cervical lymphadenopathy were also described. The mean diagnostic delay was 3.4 years. Histological evaluation was performed in 34/39 patients (87.2%); non‐caseating granulomas were found in 73.5% of them. Neurological symptoms (28.2%), gastrointestinal symptoms in absence of overt inflammatory bowel disease (IBD) (20.5%), and atopy (35.9%) were also identified. Therapeutic approaches varied among the centers. Steroids (51.3%) were used with good or partial results. Anti‐TNF‐α and anti‐IgE monoclonal antibodies were used in 6 (15.4%) and 1 (2.6%) patients, respectively, with variable results. Surgery was the choice for 2 patients with good response. Conclusions OFG is a rare and neglected disease showing multiple clinical phenotypes. While early diagnosis is crucial, management is difficult and highly dependent on the expertise of clinicians due to the lack of international guidelines. There is a need to establish registry databases and address challenges of long‐term management.

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Idiopathic Orofacial Granulomatosis with Varied Clinical Presentation

Cited by 8 publications

References 8 publications

Idiopathic orofacial granulomatosis: A case report

Idiopathic orofacial granulomatosis: A case report

Orofacial Granulomatosis

Orofacial granulomatosis: Clinical and therapeutic features in an Italian cohort and review of the literature

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