Orbital apex syndrome (OAS) is a rare complication of odontogenic maxillary sinusitis. An 87-year-old woman with gingivitis caused by a dental implant infection developed diplopia, followed by a rapid visual deterioratio...Orbital apex syndrome (OAS) is a rare complication of odontogenic maxillary sinusitis. An 87-year-old woman with gingivitis caused by a dental implant infection developed diplopia, followed by a rapid visual deterioration and limited extraocular movement in her left eye. Imaging revealed penetration of the upper left first molar into the maxillary sinus and inflammation involving the extraocular muscles and the orbital apex. Staphylococcus lugdunensis (S. lugdunensis) was cultured from the patient's sinus. Steroid pulse therapy, antimicrobial treatment, tooth extraction, and sinus irrigation partially improved her visual acuity, while extraocular movement was fully recovered. This report presents a rare case of OAS caused by S. lugdunensis.
Otsuka H, Ideguchi Y, Onizuka K
… +10 more, Amano T, Torimura D, Yamashita A, Hirayama T, Tomita Y, Shima T, Yoshimura S, Miyazaki T, Tateishi Y, Tsujino A
Acute autonomic and sensory neuropathy (AASN) is a rare disorder characterized by severe autonomic and sensory dysfunction without motor involvement, often occurring after a febrile illness. A 47-year-old woman had recur...Acute autonomic and sensory neuropathy (AASN) is a rare disorder characterized by severe autonomic and sensory dysfunction without motor involvement, often occurring after a febrile illness. A 47-year-old woman had recurrent syncope episodes four days after fever onset, followed by progressive deep sensory loss. On day 14 of hospitalization at the previous hospital, she was transferred to our institution. Magnetic resonance imaging revealed hyperintensity in the posterior columns of the cervical cord spanning three vertebral levels on T2-weighted images and diffuse enhancement of the posterior roots in the cervical and lumbar spinal cord on contrast-enhanced T1-weighted images. Sensory responses were absent in nerve conduction studies, whereas motor responses in both the upper and lower extremities were preserved. Two weeks after syncope became apparent, aggressive immunotherapy was administered; however, the outcome was poor. The early recognition of autonomic and sensory neuropathy, along with contrast-enhanced MRI of the spinal cord, may be useful for the diagnosis and management of AASN.
Yamamoto U, Oba Y, Rikihisa T
… +13 more, Taniguchi S, Mizuno H, Sekine A, Yamanouchi M, Hasegawa E, Suwabe T, Tamaoka M, Takaya H, Kono K, Ohashi K, Wada T, Sawa N, Ubara Y
A 73-year-old woman with bronchial asthma presented with a rash and peripheral neuropathy. Since 21 years old, her asthma had been treated with glucocorticoids. Benralizumab had been introduced six years ago, and glucoco...A 73-year-old woman with bronchial asthma presented with a rash and peripheral neuropathy. Since 21 years old, her asthma had been treated with glucocorticoids. Benralizumab had been introduced six years ago, and glucocorticoids were discontinued one year later. Bronchial asthma had been in remission for 5 years. On admission, urinalysis findings were mildly abnormal, myeloperoxidase-specific antineutrophil cytoplasmic antibody was positive, and eosinophilia was observed. A renal biopsy showed signs of mild necrotizing vasculitis with eosinophilia. Eosinophilic granulomatosis with polyangiitis (EGPA) was diagnosed. Benralizumab was discontinued, glucocorticoids were restarted, and mepolizumab was administered. Since then, the EGPA and asthma have not recurred.
We report an extremely rare case of recurrent Takotsubo syndrome (TTS) in an 83-year-old woman who initially experienced the mid-ventricular type and later developed the focal type. TTS is a transient cardiac condition t...We report an extremely rare case of recurrent Takotsubo syndrome (TTS) in an 83-year-old woman who initially experienced the mid-ventricular type and later developed the focal type. TTS is a transient cardiac condition that mimics acute coronary syndrome but lacks obstructive coronary lesions. While recurrence is uncommon, a change in morphological subtype is particularly rare. In this case, echocardiography was inconclusive, and the diagnosis was confirmed via left ventriculography. This case highlights the diagnostic challenges of focal-type TTS and emphasizes the importance of comprehensive imaging, especially left ventriculography, in patients with suspected recurrence and inconclusive non-invasive findings.
A 66-year-old man presented with dyspnea which occurred while running for 3 months. His resting electrocardiogram was normal, but a treadmill test revealed a 2:1 atrioventricular (AV) block when the sinus rate increased...A 66-year-old man presented with dyspnea which occurred while running for 3 months. His resting electrocardiogram was normal, but a treadmill test revealed a 2:1 atrioventricular (AV) block when the sinus rate increased to 142 beats per minute. We diagnosed exercise-induced AV block and implanted a dual-chamber pacemaker. Initially, the ventricles were paced mainly during exercise. However, the patient became dependent on ventricular pacing at rest 4 years after the initial presentation, and progression to complete AV block was thereafter confirmed. This case highlights that exercise-induced AV block is not a benign condition, and AV conduction can deteriorate further over time.
Small molecules used to treat inflammatory bowel disease (IBD) are orally administered therapies that offer a rapid onset without immunogenicity. Currently, three Janus kinase (JAK) inhibitors (tofacitinib, filgotinib, a...Small molecules used to treat inflammatory bowel disease (IBD) are orally administered therapies that offer a rapid onset without immunogenicity. Currently, three Janus kinase (JAK) inhibitors (tofacitinib, filgotinib, and upadacitinib), two Sphingosine-1-phosphate receptor (S1PR) modulators (ozanimod and etrasimod), and one alpha4-integrin antagonist (carotegrast methyl) are available in Japan. Each JAK inhibitor has its own efficacy and safety profile: Upadacitinib is the most effective, despite its higher frequency of associated adverse events, whereas filgotinib has a weaker efficiency but with fewer adverse events. Although tofacitinib has been associated with major cardiovascular events and thromboembolism in patients with rheumatoid arthritis, such findings have not been demonstrated in patients with ulcerative colitis. S1PR modulators have similar mechanisms of action, except for the titration at the beginning and the recovery time of the lymphocyte count after discontinuation. Overall, small-molecule drugs are effective and convenient options for IBD, with safety profiles that require individual patient assessment.
Vascular Ehlers-Danlos syndrome (vEDS), a rare connective tissue disorder caused by pathogenic COL3A1 variants, rarely presents with isolated pulmonary manifestations in asymptomatic cases. We herein report the case of a...Vascular Ehlers-Danlos syndrome (vEDS), a rare connective tissue disorder caused by pathogenic COL3A1 variants, rarely presents with isolated pulmonary manifestations in asymptomatic cases. We herein report the case of a 36-year-old asymptomatic man with incidentally discovered fluctuating pulmonary nodules and cavitary lesions. A histopathological examination revealed nodular fibrosis and focal pulmonary ossification, indicating a disruption of the pulmonary architecture and pulmonary hemosiderosis. Genetic testing revealed a novel COL3A1 frameshift variant, confirming that the attenuated vEDS phenotype is attributable to haploinsufficiency. This case report highlights the diagnostic significance of asymptomatic pulmonary diseases.
Despite the increasing number of patients diagnosed with postprandial reactive hypoglycemia (PRH), standardized diagnostic criteria and evaluation methods have not been established. We report three cases of PRH in young...Despite the increasing number of patients diagnosed with postprandial reactive hypoglycemia (PRH), standardized diagnostic criteria and evaluation methods have not been established. We report three cases of PRH in young adult Japanese women with a normal body weight, identified through oral glucose tolerance testing and continuous glucose monitoring. In these patients, PRH was likely driven by disproportionately increased insulin secretion, possibly related to residual insulin resistance stemming from a history of childhood obesity. As PRH can occur in individuals with various known and yet-to-be-defined predispositions, careful assessment of the underlying contributing factors is essential.
A 58-year-old man underwent left lower lobectomy for lung cancer. During the postoperative follow-up, upper-lung field pulmonary fibrosis (upper-PF) radiologically consistent with pleuroparenchymal fibroelastosis, gradua...A 58-year-old man underwent left lower lobectomy for lung cancer. During the postoperative follow-up, upper-lung field pulmonary fibrosis (upper-PF) radiologically consistent with pleuroparenchymal fibroelastosis, gradually emerged in the left upper lobe. Seventeen years after surgery, the patient presented with acute dyspnea. Chest computed tomography demonstrated bilateral ground-glass opacities, predominantly in the contralateral non-fibrotic lung. After excluding other potential causes of acute respiratory failure, an acute exacerbation of unilateral upper-PF was diagnosed and successfully treated with anti-inflammatory therapy. Chest physicians should recognize that an acute exacerbation can develop even in patients with unilateral upper-PF, although it might be extremely rare.
Chronic pulmonary aspergillosis typically affects individuals with normal or mildly compromised immune systems and it is categorized into several subtypes based on the lesion morphology and disease progression patterns....Chronic pulmonary aspergillosis typically affects individuals with normal or mildly compromised immune systems and it is categorized into several subtypes based on the lesion morphology and disease progression patterns. We present a rare case of a bacterial lung abscess developing within a pre-existing pulmonary cavity, representing a transitional phase from simple pulmonary aspergilloma to chronic cavitary pulmonary aspergillosis. The patient underwent percutaneous drainage of the purulent cavity, followed by combined antibacterial and antifungal therapy, resulting in favorable outcomes without any complications. In selected cases in which the cavitary structure remains intact, minimally invasive treatments, such as percutaneous lung abscess drainage, may offer a viable therapeutic option.
We report the case of a 53-year-old woman with type 2 diabetes and bulimia nervosa who developed prolonged paralytic ileus after self-injecting tirzepatide 7.5 mg, 10 mg, and 15 mg on three consecutive days. Tirzepatide...We report the case of a 53-year-old woman with type 2 diabetes and bulimia nervosa who developed prolonged paralytic ileus after self-injecting tirzepatide 7.5 mg, 10 mg, and 15 mg on three consecutive days. Tirzepatide had been prescribed for diabetes treatment. However, the excessive self-administration resulted in paralytic ileus. No mechanical obstruction was found, and the condition resolved after three weeks of conservative therapy, including gastrointestinal decompression and prokinetic agents. Because tirzepatide has a long half-life, an overdose may cause the persistent suppression of intestinal motility. Misunderstanding about drug effects may lead to an overdose, thus requiring careful instruction and medication management.
Objective Sternal wound infection (SWI) is a devastating complication following cardiac surgery. Although most SWI are bacterial and well described, Candida SWI remains poorly characterized. This study aimed to examine t...Objective Sternal wound infection (SWI) is a devastating complication following cardiac surgery. Although most SWI are bacterial and well described, Candida SWI remains poorly characterized. This study aimed to examine the clinical characteristics, management, and outcomes of Candida SWI. Methods We retrospectively reviewed the data of patients with Candida SWI following cardiac surgery at Baylor St. Luke's Medical Center between 2013 and 2024. Results Of the 11,108 cardiac surgeries, 55 Candida SWI were identified: superficial (10.9%), deep (21.8%), sternal osteomyelitis (40.0%), and mediastinitis (27.3%). Most occurred within 90 days postoperatively (70.9%). Candida albicans was most common (69.1%), followed by C. parapsilosis (12.7%), C. glabrata (9.1%), and C.tropicalis (7.3%). Surgical intervention was performed in 87.3% of the patients, and 60.0% received antifungal therapy for >6 weeks. The ninety-day mortality was 20.0%. In-hospital mortality was significantly associated with postoperative mechanical ventilation >48 hours [100% (in-hospital death group) vs. 63.9% (survivor group), OR: 17.38, 95% CI, 0.95-316.99; P =.008] and a lack of surgical debridement [26.4% (in-hospital death group) vs. 6.8% (survivor group), OR: 0.14, 95% CI, 0.02-0.79, P =.023]. However, it was not associated with mediastinitis [54.5% (in-hospital death group) vs. 20.5% (survivor group), OR: 4.41, 95% CI, 1.15-16.90; P =.052]. Mediastinitis was associated with a longer cardiopulmonary bypass time [213 min (mediastinitis group) vs. 142.5 min (non-mediastinitis group), P =.005] and extended postoperative hospital stays [55 days (mediastinitis group) vs. 36 days (non-mediastinitis group), P =.027]. Conclusions Candida SWI presented with variable extents and onsets. Despite aggressive surgical management and prolonged antifungal therapy, the prognosis remains challenging.
We present the case of a 74-year-old woman with a large ovarian tumor who developed cancer-associated stroke with multiple acute cerebral infarcts and markedly elevated D-dimer levels. Unfractionated heparin was initiate...We present the case of a 74-year-old woman with a large ovarian tumor who developed cancer-associated stroke with multiple acute cerebral infarcts and markedly elevated D-dimer levels. Unfractionated heparin was initiated. However, thrombocytopenia and rising D-dimer levels on Day 11 led to its discontinuation and a diagnosis of probable heparin-induced thrombocytopenia, supported by a strongly positive PF4/heparin antibody assay. Despite subsequent non-heparin anticoagulation, she developed recurrent ischemic stroke and systemic embolic events, and surgery for the ovarian tumor was no longer feasible. This case highlights the therapeutic dilemma posed by the coexistence of cancer-associated stroke and immune-mediated heparin-induced thrombocytopenia.
Acute intermittent porphyria (AIP) is a condition characterized by the neurotoxic accumulation of δ-aminolevulinic acid and porphobilinogen. However, chronic kidney disease is an under-recognized complication. A 50-year-...Acute intermittent porphyria (AIP) is a condition characterized by the neurotoxic accumulation of δ-aminolevulinic acid and porphobilinogen. However, chronic kidney disease is an under-recognized complication. A 50-year-old woman experienced recurrent abdominal pain with repeated acute kidney injury and hyponatremia, resulting in a rapid decline in renal function. Marked elevations of both metabolites, as well as her genetic mutation, confirmed AIP. The administration of hemin markedly decreased the frequency of porphyria attacks and significantly attenuated the annual estimated glomerular filtration rate decline. Although the patient eventually developed end-stage kidney disease, hemin treatment slowed the progression of renal failure.
Enteric-type thymic adenocarcinomas are rare histological variants with limited treatment options. We report the case of a 56-year-old woman with stage IVB enteric-type thymic adenocarcinoma who was treated with lenvatin...Enteric-type thymic adenocarcinomas are rare histological variants with limited treatment options. We report the case of a 56-year-old woman with stage IVB enteric-type thymic adenocarcinoma who was treated with lenvatinib. The patient underwent chemoradiotherapy and subsequently received lenvatinib for local recurrence. Despite the patient receiving a low dosage owing to side effects, the size of the recurrent lesions decreased. However, 5 months later, new brain metastases and carcinomatous meningitis developed, and the patient eventually succumbed to cancer. This case suggests that lenvatinib may be an effective treatment for enteric-type adenocarcinomas. However, at low doses, its efficacy may be limited to intracranial lesions.
Neuroleptic malignant syndrome (NMS) can be complicated by rhabdomyolysis, a known cause of acute kidney injury (AKI) that typically resolves within a short period. However, some cases of NMS-associated rhabdomyolysis re...Neuroleptic malignant syndrome (NMS) can be complicated by rhabdomyolysis, a known cause of acute kidney injury (AKI) that typically resolves within a short period. However, some cases of NMS-associated rhabdomyolysis require hemodialysis. We report a woman in her fifties who developed AKI secondary to NMS-associated rhabdomyolysis and required prolonged renal replacement therapy (RRT). She experienced sepsis and contrast exposure, and dialysis independence was achieved only after 2 months, the longest reported duration. This case highlights that NMS-associated rhabdomyolysis-induced AKI may lead to prolonged dialysis, particularly in the presence of additional risk factors for renal injury.
A 40-year-old man with acquired immunodeficiency syndrome (AIDS) presented with severe thrombocytopenia and disseminated Kaposi's sarcoma (KS). Initially diagnosed with immune thrombocytopenic purpura (ITP), he was treat...A 40-year-old man with acquired immunodeficiency syndrome (AIDS) presented with severe thrombocytopenia and disseminated Kaposi's sarcoma (KS). Initially diagnosed with immune thrombocytopenic purpura (ITP), he was treated with corticosteroids and antiretroviral therapy; however, his condition worsened. He met the criteria for Kaposi's sarcoma-associated inflammatory cytokine syndrome (KICS) and received liposomal doxorubicin, showing temporary improvement. KS progression was observed after immune recovery, suggesting KS-associated immune reconstitution inflammatory syndrome. Rituximab was administered, but the patient deteriorated and died. This case highlights the difficulty in distinguishing KICS from ITP, and the importance of an early diagnosis and multidisciplinary care in advanced AIDS.