The patient, a 65-year-old woman with a prior history of pulmonary and ocular sarcoidosis treated 10 years previously, had been hospitalized for headaches. Since 2 years, she reported chronic sinusitis with a nasal enlargement and more recently headaches. Clinical and ear nose throat examinations revealed arthralgia of the ankles, morning facial edema, suborbital facial pain, bilateral nasal obstruction, crusting, epistaxis, an ocular and nasal dry syndrome. Biological work-up revealed elevated angiotensin-converting enzyme (93 U/l (20–70)) and C-reactive protein level (20 mg/l). Imaging was as follows: rhinosinusitis, pulmonary fibrosis, multiple lymphadenopathy and splenomegaly on computed tomography (CT)-scan, frontal pachymeningitis on magnetic resonance imaging and remodeling of the craniofacial bone mostly of the sinonasal cavities and mandibular on 99mtechnetium bone scintigraphy (Figure 1A). Apart from the pulmonary fibrosis, all the radiological findings reported previously had fluorine-18-fluorodeoxyglucose uptakes on positron emission tomography /CT, mostly ethmoidal bone and disseminated lymph nodes. The patient underwent sinusal biopsy, as well as salivary biopsy, which confirmed mucosal, salivary and osteomyelitis noncaseating granulomas (Figure 1B) and ruled out fungal or mycobacterial infection with negative acid-fast and periodic acid-Schiff stains, confirming the diagnosis of systemic sarcoidosis. Thus, infliximab and methotrexate were started.

Sinonasal and mandibular aeras uptake of 99mtechnetium methylene diphosphonate bone scintigraphy.
Figure 1.

Sinonasal and mandibular aeras uptake of 99mtechnetium methylene diphosphonate bone scintigraphy.

Sinonasal sarcoidosis remains atypical and varied from 1% to 30% in cases series.1,2 Atrophic, hypertrophic, destructive and associated with nasal enlargement are the four subgroups reported, the latter being found in our patient. In our patient with a prior history of sarcoidosis, differential diagnosis of nasal enlargement included lupus pernio as an erythematoviolaceous plaques infiltrating the nose, an angiolupoid sarcoidosis and a metastatic disease.3

Conflict of interest. None declared.

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