A case of adult-onset Still's disease (AOSD)-like manifestations abruptly developing during confirmation of a diagnosis of metastatic papillary thyroid carcinoma.

A 68-year-old Japanese man was admitted to our hospital suffering from abrupt onset of high fever accompanied by arthralgia, myalgia, sore throat, macular eruption, and liver dysfunction. Six months before the onset of these manifestations, (18)F-fluoro-deoxy-glucose positron emission tomography/computed tomography ((18)F-FDG PET/CT) had detected, unexpectedly, three calcified thyroid lesions without (18)F-FDG uptake. Two months before the onset of the present manifestations, ultrasonography-guided fine-needle aspiration had led to a diagnosis of papillary thyroid carcinoma (PTC). Soon after the occurrence of the rheumatic manifestations, a subsequent (18)F-FDG PET/CT scan showed not only the three thyroid lesions, the same as those in the previous scan, but also (18)F-FDG uptake in the thyroid lesion. A diagnosis of adult-onset Still's disease (AOSD)-like manifestations associated with PTC was made, and treatment with 40 mg/day of prednisolone (PSL) resolved the symptoms promptly. PSL was gradually tapered, without recurrence of the (AOSD)-like manifestations. Five months after the initiation of treatment with PSL, total thyroidectomy, followed by (131)I thyroid ablation treatment, was performed while the patient was on a PSL dose of 18 mg/day. Seven months after the thyroidectomy, the dose of PSL was tapered to 2 mg/day, and neither the AOSD-like manifestations nor the PTC relapsed. On confirming a diagnosis of AOSD, it may be necessary to consider the presence of an associated malignancy, including solid tumors such as PTC.

Hypomagnesemia associated with a proton pump inhibitor.

Severe hypomagnesemia is a serious clinical condition. Proton pump inhibitor (PPI) induced hypomagnesemia has been recognized since 2006. In March 2011 the U.S. Food and Drug Administration advised that long-term use of PPI can induce hypomagnesemia. We report the first Japanese case of hypomagnesemia associated with chronic use of PPIs in a 64-year-old man hospitalized for nausea, bilateral ankle arthritis, and tremor of the extremities who had convulsions 3 days after admission. Blood analysis showed severe hypomagnesemia. He had been taking rabeprazole (10 mg/day) for 5 years. After stopping rabeprazole and correcting the electrolytes imbalances, his symptoms improved without recurrence.

IgG4-related tubulointerstitial nephritis and hepatic inflammatory pseudotumor without hypocomplementemia.

Immunoglobulin G4 (IgG4)-related tubulointerstitial nephritis (TIN) is often accompanied by autoimmune pancreatitis (AIP) or chronic sclerosing dacryoadenitis and sialoadenitis. However, IgG4-related TIN without AIP or lacrimal and/or salivary gland lesions has not been well recognized. Here, we report a case of IgG4-related TIN associated with hepatic inflammatory pseudotumor without AIP or lacrimal and/or salivary gland lesions. A 58-year-old Japanese man with epigastralgia underwent contrast-enhanced computed tomography (CT), which revealed multiple low-density lesions in both kidneys and a low density hepatic mass. Laboratory tests showed an extremely high level of serum IgG4. Percutaneous renal and hepatic biopsies showed diffuse infiltration of lymphocytes and IgG4-positive plasma cells with fibrosis in both tissues. Two months after administration of oral prednisolone, both lesions decreased in size on follow-up CT, and the serum creatinine level also improved. No recurrence has been detected for two years with a maintenance dose of prednisolone.