BACKGROUND: Adult-onset asthma and periocular xanthogranuloma is an uncommon and recently described disease. Little is known about the condition because only a few case reports and series are available. OBJECTIVE/ METHODS: To describe the clinical manifestations, lung physiology, and response to systemic treatment of three patients with adult-onset asthma and periocular xanthogranuloma, followed by a review of the literature. RESULTS: Three men, with an age at diagnosis ranging from 48 to 51 years, presented with right periorbital swelling, asthma and chronic rhinosinusitis. The patients' lung physiology was consistent with airway obstruction. Diagnosis was established by periorbital biopsy. All patients received oral corticosteroids for their periorbital swelling, without significant clinical response. Two patients received oral methotrexate, with nearly complete resolution of periorbital swelling. A third patient received oral azathioprine, without clinical response. The three patients had improvement of their asthma with inhaled steroids/long-acting bronchodilator, and immunosuppressive medication. CONCLUSION: A triad consisting of periorbital swelling, asthma and chronic rhinosinusitis should raise the suspicion of adult-onset asthma and periocular xanthogranuloma. Oral methotrexate should be considered as an alternative to corticosteroids in the treatment of this disorder.
BACKGROUND: Adult-onset asthma and periocular xanthogranuloma is an uncommon and recently described disease. Little is known about the condition because only a few case reports and series are available. OBJECTIVE/ METHODS: To describe the clinical manifestations, lung physiology, and response to systemic treatment of three patients with adult-onset asthma and periocular xanthogranuloma, followed by a review of the literature. RESULTS: Three men, with an age at diagnosis ranging from 48 to 51 years, presented with right periorbital swelling, asthma and chronic rhinosinusitis. The patients' lung physiology was consistent with airway obstruction. Diagnosis was established by periorbital biopsy. All patients received oral corticosteroids for their periorbital swelling, without significant clinical response. Two patients received oral methotrexate, with nearly complete resolution of periorbital swelling. A third patient received oral azathioprine, without clinical response. The three patients had improvement of their asthma with inhaled steroids/long-acting bronchodilator, and immunosuppressive medication. CONCLUSION: A triad consisting of periorbital swelling, asthma and chronic rhinosinusitis should raise the suspicion of adult-onset asthma and periocular xanthogranuloma. Oral methotrexate should be considered as an alternative to corticosteroids in the treatment of this disorder.
Authors: J A Sivak-Callcott; J Rootman; S L Rasmussen; R A Nugent; V A White; D Paridaens; Z Currie; G Rose; B Clark; A A McNab; F V Buffam; J M Neigel; M Kazim Journal: Br J Ophthalmol Date: 2006-05 Impact factor: 4.638
Authors: Christopher I Zoumalan; Melanie H Erb; Narsing A Rao; Robert See; Michael A Bernstine; Samir B Shah; Timothy J McCulley Journal: Br J Ophthalmol Date: 2007-08 Impact factor: 4.638
Authors: Ioannis Asproudis; Maria Kanari; Ioannis Ntountas; Vasileios Ragos; Anna Goussia; Anna Batistatou; Paraskevi Vasileios Voulgari Journal: Rheumatol Int Date: 2019-08-07 Impact factor: 2.631
Authors: Jonathan London; Antoine Martin; Michael Soussan; Isabelle Badelon; Thomas Gille; Yurdagul Uzunhan; Bénédicte Giroux-Leprieur; Ursula Warzocha; Alexis Régent; Olivier Galatoire; Robin Dhote; Sébastien Abad Journal: Medicine (Baltimore) Date: 2015-10 Impact factor: 1.817