SAGE Open Medical Case Reports最新文献

Dipeptidyl peptidase-4 inhibitors are oral antihyperglycemic medications often used as adjuncts to other antidiabetic agents to treat type 2 diabetes mellitus. Henoch-Schönlein purpura, also known as ImmunoglobulinA (IgA) vasculitis, is a form of IgA-mediated leukocytoclastic vasculitis, which is rarely reported in adults. Herein, we report the case of a 52-year-old male with type 2 diabetes mellitus who presented with petechiae, ecchymoses, and palpable purpura below the knees bilaterally after starting sitagliptin, a dipeptidyl peptidase-4 inhibitor. A punch biopsy for direct immunofluorescence showed granular Immunoglobulin A, M, and complement 3 in vessel walls, suggestive of Henoch-Schönlein purpura. Sitagliptin was discontinued, and the patient was treated with celecoxib, colchicine, and cefadroxil for wound infection. Three months after initial presentation, brown hyperpigmentation was appreciated, suggestive of post-inflammatory changes and resolution of the lesions. However, urinalysis revealed new 0.3 g/L protein. Overall, this case highlights a new potential association of dipeptidyl peptidase-4 inhibitors with Henoch-Schönlein purpura, which may have systemic consequences.

An 88-year-old male presented with sepsis and multi-organ dysfunction due to Neisseria meningitidis serogroup B (MenB) infection, a severe and often atypical presentation in the elderly. His condition, characterized by subtle symptoms, rapidly improved with early broad-spectrum antibiotics, susceptibility-guided therapy, and multidisciplinary care. The patient fully recovered and was discharged on day 8, highlighting the importance of prompt diagnosis and aggressive support in this vulnerable population.

Fahr's disease is a rare neurodegenerative disorder that involves bilateral calcifications within the brain, most notably affecting areas such as the basal ganglia, thalami, and cerebellum. It is typically identified through its association with movement abnormalities, cognitive decline, or psychiatric symptoms; however, asymptomatic cases may be underrecognized, with studies suggesting a substantial proportion of patients remain without symptoms.³ In this report, a 45-year-old male came to the emergency department after experiencing a fall, complaining of rib pain and dizziness. During his trauma workup, a noncontrast computed tomography scan of the head incidentally revealed widespread bilateral calcifications in the basal ganglia, thalami, subcortical white matter, and cerebellar dentate nuclei. Subsequent magnetic resonance imaging supported the computed tomography findings, offering a clearer view of the extent of the calcifications. Laboratory tests, including assessments of calcium, phosphate, parathyroid hormone, magnesium, vitamin D, and thyroid function, returned normal results. Furthermore, comprehensive neurological and psychiatric evaluations found no abnormalities. Despite the extensive radiological evidence of brain calcification, the patient had no clinical symptoms. There was also no relevant family history of Fahr's disease. These findings point to an idiopathic, asymptomatic form of the disease. This case emphasizes the importance of recognizing radiographic findings in the absence of clinical symptoms. Although identification and follow-up of such cases is often suggested, there is currently no evidence that this improves patient outcomes. Moreover, documenting asymptomatic cases helps expand understanding of the disease's variability, natural course, and prognostic implications.

Erosive lichen planus is a rare, chronic, immune-mediated inflammatory disease that can lead to significant morbidity. Erosive lichen planus is often resistant to therapies, and spontaneous remission is uncommon. We report the case of a 62-year-old woman with severe and refractory erosive lichen planus involving the oral, genital, and esophageal mucosa, who failed multiple systemic treatments. Initiation of abrocitinib, a selective Janus Kinase 1 inhibitor, led to complete and sustained clinical remission.

Erosive pustular dermatosis is an uncommon chronic inflammatory condition of the scalp characterized by crusted, erosive plaques and subsequent cicatricial alopecia. Management of erosive pustular dermatosis remains challenging, and various treatments have been described in the literature with inconsistent results. Herein, we report three cases of erosive pustular dermatosis with a pityriasis amiantacea-like presentation in the context of poor scalp hygiene. All patients presented with thick adherent crusts on the scalp that were refractory to multiple therapies. Significant clinical improvement was achieved by addressing scalp hygiene through regular cleansing and the use of dilute sodium hypochlorite compresses, in combination with topical corticosteroids as well as systemic and topical antibiotics. Our findings highlight the importance of addressing scalp hygiene to ameliorate treatment outcomes and minimize the risk of recurrence in patients with erosive pustular dermatosis, especially those with a pityriasis amiantacea-like presentation.

A 15-year-old boy presented with pink proliferative plaque over both lips. The child was born to a non-consanguineous couple, at full term, and uneventful antenatal, perinatal history. Examination revealed a cobblestone appearance over tongue with papilliferous projections in both labial mucosa with slate-grey pigmentation. The child had multiple lipomatosis and thyroid swelling. Father had thyroid swelling with multiple epidermoid cysts. A final diagnosis of Cowden syndrome. Genetic analysis could not be performed due to financial constraints. Oral mucosal cobblestone appearance is seen in Darier disease, Cowden's syndrome, lipoid proteinosis, granulomatous cheilitis, mucosal neuroma, Crohn's disease, and mucous membrane plasmacytosis. Diffuse oral mucosal melanosis is described in Peutz-Jeghers syndrome, Laugier-Hunziker-Baran syndrome, Cowden syndrome, and Addison's disease. We report this rare genodermatosis, which presents as oral papillomatosis. Early recognition of Cowden syndrome is crucial, as timely diagnosis enables appropriate cancer surveillance, genetic counseling, and preventive interventions, thereby reducing morbidity and improving long-term outcomes.

Tardive dyskinesia is a hyperkinetic movement disorder most often induced by chronic antipsychotic use. It can be challenging to treat, particularly when symptoms persist despite standard therapies such as vesicular monoamine transporter 2 inhibitors. We describe a 37-year-old male with schizoaffective disorder and persistent tardive dyskinesia involving oro-buccal-lingual and axial movements. Despite withdrawal of antipsychotics, a trial of deutetrabenazine, and high-dose tetrabenazine (150 mg/day for several weeks), symptoms persisted and significantly impaired critical care management. On hospital day 46, phenobarbital was initiated (dose and route here), resulting in rapid improvement of dyskinetic movements within 3 days. By hospital day 54, complete resolution of tardive dyskinesia was observed, and notably, symptoms did not recur after phenobarbital discontinuation. The patient maintained clinical improvement during long-term follow-up, with Abnormal Involuntary Movement Scale score improving to 2 at 18 months. This case illustrates a potential novel role for phenobarbital in refractory tardive dyskinesia, particularly when conventional therapies fail. The sustained response beyond the drug's pharmacokinetic profile raises the possibility of a disease-modifying or prolonged therapeutic effect. Given phenobarbital's activity on Gamma Aminobutyric Acid B receptors, its mechanism may involve modulation of GABAergic pathways implicated in the pathophysiology of tardive dyskinesia. Further investigation in clinical studies is warranted.

The following case report details the case of a 40-year-old Caucasian patient who presented with dyspnea following a serologically confirmed mild-to-severe pulmonary infection with SARS-CoV-2. Chest computer tomography revealed a solitary ground-glass pulmonary nodule in the lower right lobe, measuring 2.1 cm in diameter. Video-assisted thoracoscopic surgery wedge resection revealed well-circumscribed lymphoid aggregates adjacent to the round, smaller airways, bronchioles, and blood vessels. IgKappa B exhibited a monoclonal polyclonal pattern, in contrast to the behavior exhibited by IgKappa A and IgLambda. In the following discussion, the lymphoid lesion was considered in the context of lymphoid hyperplasia, accompanied by an early infiltration of low-grade extranodal B cell lymphoma of the bronchus-associated lymphoid tissue (BALToma).

Acute cerebral infarction, or stroke, is one of the leading causes of death and disability among adults worldwide, and the key to its treatment is achieving early recanalization of occluded blood vessels, methods for which include intravenous thrombolysis and mechanical thrombectomy. This article reports a case of a 43-year-old female who suddenly suffered acute cerebral infarction and was treated with tissue plasminogen activator intravenous thrombolysis. During the thrombolysis process, transcranial Doppler ultrasound was used to evaluate acute occlusion of the left middle cerebral artery, which was confirmed via an emergency head magnetic resonance angiography and digital subtraction angiography. Before mechanical thrombectomy, repeat angiography showed that the occluded left middle cerebral artery had regained blood flow, and after postoperative follow-up transcranial Doppler ultrasound, the blood flow of the left middle cerebral artery and anterior cerebral artery was found to have fully recovered. The patient's symptoms were completely relieved, and they were discharged after 7 days of treatment, with follow-up at 2 weeks and 3 months showing no abnormal symptoms or intracranial blood flow. This case not only provides evidence that tissue plasminogen activator intravenous thrombolysis can salvage the ischemic penumbra, dissolve thrombi, and encourage reperfusion in occluded intracranial large arteries, but it also emphasizes the critical role of transcranial Doppler ultrasound in the rapid assessment of acute large vessel occlusions. As such, our report highlights the importance of transcranial Doppler ultrasound in clinical interventions to optimize outcomes in acute stroke management.

Providencia rettgeri is a Gram-negative bacterium, rarely reported as a cause of diabetic wound infections. Its frequent resistance to β-lactams and carbapenems limits treatment options, especially in immunocompromised patients. We present the case of a 59-year-old unhoused male living with diabetes, peripheral artery disease, and prior bilateral transmetatarsal amputations who presented with a University of Texas 3B diabetic foot ulcer. Magnetic resonance imaging showed osteomyelitis of metatarsals 2-4, and bone biopsy grew Providencia rettgeri and Proteus mirabilis. The patient declined further surgery and was treated with 6 weeks of cefepime and metronidazole, with outpatient wound care, achieving wound closure in 3 months. We performed a systematic scoping literature review on Providencia rettgeri wound infections in humans, detailed below, which identified only eight studies that met our inclusion and exclusion criteria. Most cases involved snake bites or critically ill patients, and none documented lower extremity bone involvement. Resistance patterns varied, but co-infection was common. Given its rarity and potential for multidrug resistance, Providencia rettgeri should remain on the differential for non-healing diabetic wounds. This case expands the limited literature and highlights the importance of culture-driven antibiotic therapy in complex infections. Recognition of Providencia rettgeri as a rare but clinically significant pathogen in diabetic foot osteomyelitis has implications for empiric antibiotic selection and clinical outcomes.