We report a case of hyperplastic callus formation that occurred in both femurs in a patient with type V osteogenesis imperfecta (OI), with 4-year follow-up and resolution. The clinical, histological and imaging aspects of this condition are discussed. Recognition of the hyperplastic callus formation in this particular type of OI is important in order to avoid misdiagnosis.

BackgroundMinor salivary gland biopsy (MSGB) is a simple procedure that can be used for the diagnosis of Sjögren's syndrome (SS) and other infiltrative diseases, such as amyloidosis, hemochromatosis, and sarcoidosis. The revised version of the European criteria from the American-European Consensus Group (AECG) in 2002 defined that MSGB with a focus score ≥1 (defined as a number of inflammatory infiltrates of at least 50 cells in 4 mm2 of gland surface unit), is a major criteria for SS classification. MSGB is also included in American College Rheumathology Classification criteria for SS.ObjectivesTo describe the diagnostic usefulness and safety of MSGB performed in our centre in the evaluation of suspected SS.MethodsIn this study, the authors retrospectively analyzed the clinical and laboratory findings of all patients seen at our centre who underwent a MSGB for suspected SS from January 2013 to February 2015. The data collected included demographic, clinical, serologic and histological features. Patients were classified according to the AECG criteria.ResultsA total of 93 MSGB were performed for suspected SS – 11 patients for secondary SS (6 previously diagnosed with rheumatoid arthritis, 2 with systemic upus erythematosus, 1 with rhupus, 1 with systemic sclerosis and 1 with autoimmune hepatitis) and 82 with suspected primary SS. Of the 93 MSGB, 91 (97.8%) samples provided adequate material for histologic analysis, however 3 other patients were excluded because of lack of clinical and laboratorial information. Eighty-two patients were women, with a mean age of 50,8±12 years old. Eighty-six patients had sicca symptoms: 90,7% presented xerostomy and 80,2% xerophtalmia. Twenty-seven patients (30,7%) were positive for antibodies anti-SSa or anti-SSb. Twenty-six patients (29,5%) had a focus score ≥1, and 20 (94.5%) of these 26 satisfied the AECG criteria. Classification of SS was possible for 32 (36.3%) of 88 patients. In 12 (37,5%) of the 32 patients, MSGB was essential to reach the number of criteria needed for classification. There were no immediate or late complications with the procedure.ConclusionsDespite the limitations of this study, which is retrospective and has a small sample size, it can be concluded that MSGB is a simple, safe, and useful tool in the evaluation of suspected of SS. It should be performed whenever there is a suspicion of SS but there are not sufficient criteria met to make the diagnosis.Disclosure of InterestNone declared

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