Characteristics of patients with orofacial granulomatosis.

OBJECTIVES Orofacial granulomatosis has mostly been described in reports of very small numbers of cases. Few large case groups have been described. The aim of this study was to describe the demographics, symptoms, clinical features and laboratory findings in a large cohort of cases. SUBJECTS AND METHODS Clinical and laboratory data for 119 cases of orofacial granulomatosis who attended oral medicine clinics in Dublin, Ireland, were examined for demographic characteristics at the time of first presentation. The male/female ratio was approximately 1:1, with a median age (and range) of 28 (5-84) years. RESULTS Symptoms had been present for a median duration of 12 weeks. A food association was suspected by 30% of patients. The predominant complaint was lip swelling (77%) with only 15% reporting facial swelling, while 8% complained of both. Almost all patients had clinical evidence of lip or facial swelling (95%). Other common extra-oral manifestations were lip fissuring (30%), angular cheilitis (28%) and perioral erythema (28%). Common intra-oral manifestations were cobblestoning of the buccal mucosa (63%), ulcers (36%), granulomatous gingivitis (33%), mucosal tags (29%) and fissured tongue (17%). Over half of the biopsies (56%) performed were reported as typical of orofacial granulomatosis. CONCLUSION This is one of the largest cohorts of orofacial granulomatosis patients to have been described in detail.

[1]  J. Sanderson,et al.  Distinguishing orofacial granulomatosis from crohn's disease: Two separate disease entities?1 , 2011, Inflammatory bowel diseases.

[2]  C. Healy,et al.  Patch testing for food-associated allergies in orofacial granulomatosis. , 2011, Journal of oral pathology & medicine : official publication of the International Association of Oral Pathologists and the American Academy of Oral Pathology.

[3]  J. Sanderson,et al.  Subepithelial dendritic B cells in orofacial granulomatosis , 2010, Inflammatory bowel diseases.

[4]  S. Porter,et al.  Orofacial granulomatosis: clinical features and long-term outcome of therapy. , 2010, Journal of the American Academy of Dermatology.

[5]  S. Lourenço,et al.  Oral manifestations of inflammatory bowel disease: a review based on the observation of six cases , 2010, Journal of the European Academy of Dermatology and Venereology : JEADV.

[6]  S. Porter,et al.  Onset and progression of clinical manifestations of orofacial granulomatosis. , 2009, Oral diseases.

[7]  M. Mccullough,et al.  Orofacial granulomatosis--a 20-year review. , 2009, Oral diseases.

[8]  F. Fortune,et al.  Orofacial granulomatosis: review on aetiology and pathogenesis. , 2007, Journal of oral pathology & medicine : official publication of the International Association of Oral Pathologists and the American Academy of Oral Pathology.

[9]  H. Endo,et al.  Cinnamon products as a possible etiologic factor in orofacial granulomatosis. , 2007, Medicina oral, patologia oral y cirugia bucal.

[10]  J. Sanderson,et al.  Improvement in orofacial granulomatosis on a cinnamon‐ and benzoate‐free diet , 2006, Inflammatory bowel diseases.

[11]  J. Sanderson,et al.  Oro‐Facial Granulomatosis: Crohn's Disease or a New Inflammatory Bowel Disease? , 2005, Inflammatory bowel diseases.

[12]  C. Scully,et al.  Review article: orofacial granulomatosis , 2004, Alimentary pharmacology & therapeutics.

[13]  M. El-Hakim,et al.  Orofacial granulomatosis presenting as persistent lip swelling: review of 6 new cases. , 2004, Journal of oral and maxillofacial surgery : official journal of the American Association of Oral and Maxillofacial Surgeons.

[14]  D. Bayramgürler,et al.  Possible role of Mycobacterium tuberculosis complex in Melkersson–Rosenthal syndrome demonstrated with Gen‐Probe amplified Mycobacterium tuberculosis direct test , 2004, The Australasian journal of dermatology.

[15]  S. Taibjee,et al.  Orofacial granulomatosis worsened by chocolate: results of patch testing to ingredients of Cadbury's chocolate , 2004, The British journal of dermatology.

[16]  A. Lazarov,et al.  Contact orofacial granulomatosis caused by delayed hypersensitivity to gold and mercury. , 2003, Journal of the American Academy of Dermatology.

[17]  J. Sciubba,et al.  Orofacial granulomatosis: presentation, pathology and management of 13 cases. , 2003, Journal of oral pathology & medicine : official publication of the International Association of Oral Pathologists and the American Academy of Oral Pathology.

[18]  B. Kumar,et al.  Chronic macrocheilia: a clinico‐pathological study of 28 patients , 2003, Clinical and experimental dermatology.

[19]  R. Bergman,et al.  Resolution of orofacial granulomatosis with amalgam removal , 2003, Journal of the European Academy of Dermatology and Venereology : JEADV.

[20]  S. Fedele,et al.  The multiform and variable patterns of onset of orofacial granulomatosis. , 2003, Journal of oral pathology & medicine : official publication of the International Association of Oral Pathologists and the American Academy of Oral Pathology.

[21]  E. H. van der Meij,et al.  Cheilitis granulomatosa: overview of 13 patients with long‐term follow‐up – results of management , 2002, International journal of dermatology.

[22]  P. Fleming,et al.  The oral cavity in Crohn's disease. , 2001, The Journal of pediatrics.

[23]  D. Wray,et al.  The role of allergy in oral mucosal diseases. , 2000, QJM : monthly journal of the Association of Physicians.

[24]  H. Vahaboğlu,et al.  Detection of Mycobacterium tuberculosis DNA in a patient with Melkersson–Rosenthal syndrome using polymerase chain reaction , 2000, The British journal of dermatology.

[25]  J. Holton,et al.  Is the detection of Mycobacterium tuberculosis , 2000 .

[26]  S. Challacombe.  Oro-facial granulomatosis and oral Crohns disease: are they specific diseases and do they predict systemic Crohns disease? , 2008, Oral diseases.

[27]  P. Lamey,et al.  Contact hypersensitivity in patients with orofacial granulomatosis. , 1997, American journal of contact dermatitis : official journal of the American Contact Dermatitis Society.

[28]  F. Kiesewetter,et al.  [Glossitis granulomatosa symptom of oligosymptomatic Melkersson-Rosenthal syndrome]. , 1996, HNO.

[29]  F. Kiesewetter,et al.  Glossitis granulomatosa-Sympton eines oligosymptomatischen Melkersson-Rosenthal-Syndroms , 1996 .

[30]  A. Bircher,et al.  Delayed-type hypersensitivity to cow's milk protein in Melkersson-Rosenthal syndrome: coincidence or pathogenetic role? , 1996, Dermatology.

[31]  M. Walsh,et al.  The Melkersson‐Rosenthal syndrome and food additive hypersensitivity , 1994, The British journal of dermatology.

[32]  M. Grossin,et al.  Granulomatous cheilitis in two children with sarcoidosis. , 1993, Journal of the American Academy of Dermatology.

[33]  M. Bilous,et al.  Orofacial sensitivity reactions and the role of dietary components. Case reports. , 1993, Australian dental journal.

[34]  R. Rogers,et al.  Orofacial manifestations of Melkersson-Rosenthal syndrome. A study of 42 patients and review of 220 cases from the literature. , 1992, Oral surgery, oral medicine, and oral pathology.

[35]  A. J. Oliver,et al.  Monosodium glutamate-related orofacial granulomatosis. Review and case report. , 1991, Oral surgery, oral medicine, and oral pathology.

[36]  C. Camisa,et al.  Cheilitis granulomatosa: report of six cases and review of the literature. , 1990, Journal of the American Academy of Dermatology.

[37]  C. King,et al.  Orofacial granulomatosis associated with delayed hypersensitivity to cobalt , 1990, Clinical and experimental dermatology.

[38]  R. Rogers,et al.  Melkersson-Rosenthal syndrome: a review of 36 patients. , 1989, Journal of the American Academy of Dermatology.

[39]  R. Corrocher,et al.  Is the Melkersson-Rosenthal syndrome related to the exposure to food additives? A case report. , 1989, Oral surgery, oral medicine, and oral pathology.

[40]  O. Nahlieli,et al.  [Melkersson-Rosenthal syndrome]. , 1989, Harefuah.

[41]  D. M. Walker,et al.  Orofacial granulomatosis in childhood , 1987, British Dental Journal.

[42]  O. Hornstein.  Klassifikation und klinische Variatrionsbreite des Melkersson-Rosenthal-Syndroms (MRS) , 1987 .

[43]  MacFadyen Ee,et al.  Orofacial granulomatosis--a 10 year review. , 1986 .

[44]  J. Warner,et al.  Oro‐facial granulomatosis. Response to elemental diet and provocation by food additives , 1986, Clinical allergy.

[45]  M. Ferguson,et al.  Food intolerance in patients with oro-facial granulomatosis. , 1986, Human nutrition. Applied nutrition.

[46]  M. Ferguson,et al.  Orofacial granulomatosis--a 10 year review. , 1986, Annals of the Academy of Medicine, Singapore.

[47]  B. Speculand,et al.  Amyloidosis with oral involvement. , 1985, The British journal of oral & maxillofacial surgery.

[48]  D. Patton,et al.  Oro-facial granulomatosis: a possible allergic basis. , 1985, The British journal of oral & maxillofacial surgery.

[49]  D N Mitchell,et al.  Oro-facial granulomatosis--a clinical and pathological analysis. , 1985, The Quarterly journal of medicine.

[50]  A. Eklund,et al.  Kveim test reactivity in Melkersson-Rosenthal syndrome (cheilitis granulomatosa). , 1985, Acta dermato-venereologica.

[51]  M. Schiødt,et al.  Melkersson-Rosenthal syndrome and cheilitis granulomatosa. A clinicopathological study of thirty-three patients with special reference to their oral lesions. , 1982, Oral surgery, oral medicine, and oral pathology.

[52]  R. Veldhuizen,et al.  Oral involvement in sarcoidosis. , 1982, International journal of oral surgery.

[53]  S. Bondesen,et al.  Melkersson-Rosenthal syndrome and Crohn's disease. , 1980, The British journal of oral surgery.

[54]  W. Tyldesley.  Oral Crohn's disease and related conditions. , 1979, The British journal of oral surgery.

[55]  T. Axéll.  A prevalence study of oral mucosal lesions in an adult Swedish population. , 1976, Odontologisk revy. Supplement.

[56]  Hornstein Op.  Melkersson-Rosenthal syndrome. A neuro-muco-cutaneous disease of complex origin. , 1973 .

[57]  M. Issa.  Crohn's disease of the mouth. A case report , 1971, British Dental Journal.

[58]  Dudeney Tp,et al.  Crohn's Disease of the Mouth , 1969 .

[59]  W. Hauser.  [Melkersson-Rosenthal syndrome and cheilitis granulomatosa]. , 1953, Deutsche zahnarztliche Zeitschrift.

[60]  G. Miescher.  I. Vortrge Communications ber essentielle granulomatse Makrocheilie (Cheilitis granulomatosa) , 1945 .