Priapism as an initial manifestation in an adult patient with chronic myeloid leukemia in Japan: case report and review of the literature.

We report the first documented Japanese case in the English literature of chronic myeloid leukemia (CML) in which priapism was the presenting symptom. Priapism, a rare manifestation in CML patients, is particularly uncommon in Japan. This can be attributed to the high quality of medical services and proactive health strategies implemented by the Japanese government. These strategies include recommending regular blood tests for company employees aged 35 and above, thereby facilitating early detection of CML. Hence, it is crucial to consider CML when examining any patient presenting with priapism, particularly among those who have not undergone regular medical check-ups.

ANCA-associated vasculitis with scleritis, corneal melt, and perforation rescued by rituximab: Case report and literature review.

Key Clinical Message: Patients with antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis, specifically with myeloperoxidase (MPO)-ANCA, would have a risk for developing corneal melt and perforation abruptly in a short period. It is desirable to have a team of collaboration of rheumatologists and other specialties. Abstract: An 80-year old man who had been diagnosed 5.5 years previously as ANCA-associated vasculitis by temporal artery biopsy developed corneal melt and perforation with scleritis in both eyes. He underwent successful cataract surgery and retained ambulatory vision with the aid of intravenous rituximab. Two additional patients with similar manifestations were found in the literature.

Isolation of OXA-48-like carbapenemase-producing Escherichia coli susceptible to piperacillin/tazobactam in a Japanese patient without a history of travel abroad.

Oxacillinase (OXA)-48-like ß-lactamases are the most common carbapenemases in Enterobacterales in certain regions of the world and are being introduced on a regular basis into regions of non-endemicity. Japan has been characterized by low rates of carbapenemase-producing Enterobacterales, and among them, OXA-48-like carbapenemase-producing isolates are extremely rare. Here we describe a Japanese medical worker, without a history of travel abroad, who was diagnosed as having a community-acquired urinary tract infection, and whose urine sample was found to be positive for OXA-48-like carbapenemase-producing Escherichia coli. None of her close contacts had a history of foreign travel, and the same drug-resistant organism was not observed in other patients who had been hospitalized and undergone environmental culture tests in the same medical institution. This isolate was resistant to penicillins, narrow-spectrum cephalosporins, fluoroquinolones, and cefmetazole, but was susceptible to broad-spectrum cephalosporins, piperacillin/tazobactam, and meropenem and displayed reduced susceptibility to imipenem. The modified carbapenem inactivation test supported carbapenemase production, but inhibitor-based synergistic tests yielded negative results of carbapenemase production. Multiplex polymerase chain reaction revealed the presence of the carbapenemase gene (blaOXA-48) blaTEM and AmpC ß-lactamase gene (blaDHA). Singleplex polymerase chain reaction targeting the blaOXA-48 region amplified a product sequencing to nearly the full length (722 bp) and matching 100% with OXA-48. The present case highlights a new concern regarding OXA-48-like carbapenemase-producing Enterobacterales, which remain challenging to detect for clinical laboratories in regions of non-endemicity, and may already be latent in Japan.

Presumptive complicating Clostridium paraputrificum bacteremia as a presenting manifestation in a patient with undiagnosed ulcerative colitis followed by acute colonic pseudo-obstruction.

Clostridium paraputrificum is a member of the commensal flora of the gastrointestinal tract and skin. Despite being linked with cases of severe invasive infection, this organism is an uncommon pathogen in humans. Here, we report a case of undiagnosed ulcerative colitis in which the presentation was one of presumptive complicating C. paraputrificum bacteremia and, later, acute colonic pseudo-obstruction. The patient was an elderly male with prostate cancer who was admitted in a state of shock secondary to suspected septicemia from an abdominal source. Only one of two sets of anaerobic blood cultures were positive for C. paraputrificum. Endoscopic and pathological investigations revealed proctitis consistent with ulcerative colitis. The patient's abdominal manifestations worsened, and abdominal imaging demonstrated de novo massive colonic dilatation without any apparent mechanical obstruction. We speculated that C. paraputrificum bacteremia caused by undiagnosed ulcerative colitis had created ideal conditions for acute colonic pseudo-obstruction. This case demonstrates that C. paraputrificum bacteremia can be associated with latent severe gastrointestinal pathologies, indicating the need to investigate any abdominal source of infection, even if only a single blood culture is positive.

The Dangers of Immediately Performing a Sensitive and Specific Investigation versus the Dangers of Delaying It: A Cautionary Case of Hodgkin's Lymphoma.

A 60-year-old man was admitted with a 1-month history of fever and weight loss. Multiple lymphadenopathies and haemophagocytic lymphohistiocytosis were noted from the beginning, suggesting lymphoma. However, lymph node biopsy was deferred because lymph node biopsy was regarded as being invasive and requires general anaesthesia, and because other possible differential diagnoses including gastrointestinal malignancies and TAFRO syndrome were being considered. Instead, investigations including gastrointestinal endoscopy and bone marrow biopsy were prioritized. The patient was eventually diagnosed with Hodgkin's lymphoma based on lymph node biopsy but died during chemotherapy. Physicians should prioritize the tests that are most directly related to the diagnostic outcome, even if they are invasive. LEARNING POINTS: Lymph node biopsy should be planned early in the course of investigation in patients with enlarged supraclavicular lymph nodes for the diagnosis of malignant lymphoma.Hodgkin's lymphoma can be associated with hemophagocytic lymphohistiocytosis and myelofibrosis.Analytical diagnostic reasoning based on the evidence is vital for timely and appropriate diagnosis in patients with uncommon presentations.

Mycophenolate mofetil treatment in a patient with recurrent lymphocytic hypophysitis.

Lymphocytic hypophysitis (LHP) is a relatively rare disease characterised by lymphocytic infiltration of the pituitary gland, resulting in pituitary dysfunction. LHP is generally responsive to corticosteroid therapy, but cases with recurrence require clinicians to select second-line therapy. We report here the case of a 58-year-old patient with LHP who developed panhypopituitarism and bitemporal hemianopia. He responded to prednisolone 40 mg/day but relapsed during tapering. The prednisolone dose was increased again and mycophenolate mofetil (MMF) was added. Thereafter, over the course of 1 year, prednisolone was tapered to 8 mg/day without relapse. Because of the rarity of LHP, there are no standard treatment protocols that support the choice of a specific immunosuppressive drug. MMF was effective for recurrent LHP in our case. Further accumulation of cases is needed to establish the standard treatment for this disease.