Journal of Medical Case Reports最新文献

Introduction: Malignant glaucoma is an uncommon but serious complication of intraocular surgery, including filtration surgery and phacoemulsification, with an incidence of 2-4%. It arises from an obstruction in aqueous humor flow, leading to elevated posterior pressure and subsequent complications. Delayed or insufficient treatment can result in irreversible vision loss. This case highlights the importance of early surgical intervention and anti-inflammatory management for preventing complications and achieving improved outcomes.

Case presentation: A 56-year-old Chinese woman presented with bilateral malignant glaucoma. The initial treatment of the right eye involved medications for 3 months, followed by yttrium aluminium garnet laser posterior capsulotomy, which offered limited success. Irido-zonulo-hyaloid-vitrectomy was eventually performed, successfully normalizing her intraocular pressure but resulting in macular cystoid edema 6 months later. Her left eye, diagnosed with malignant glaucoma 2 months after the right eye's presentation, received early intervention with cataract extraction, intraocular lens implantation, and irido-zonulo-hyaloid-vitrectomy. This approach yielded excellent outcomes, with optimal intraocular pressure control and preserved vision without complications.

Conclusion: This case underscores the importance of early surgical intervention, particularly irido-zonulo-hyaloid-vitrectomy, in managing malignant glaucoma. Early intervention can prevent postoperative complications such as macular edema and ensure superior visual outcomes. Tailored anti-inflammatory approaches further mitigate associated risks and enhance long-term prognosis in cases of bilateral involvement.

Background: Olfactory reference syndrome is a psychiatric disorder characterized by a persistent and distressing preoccupation with emitting a foul body odor despite the absence of objective evidence. It is classified under "other specified obsessive-compulsive and related disorders" in the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition, Text Revision. Although olfactory reference syndrome has been described in several regions globally, there are no published case reports from East Africa.

Case presentation: A 50-year-old Ethiopian man was referred from the otorhinolaryngology department to psychiatry with 3 years history of preoccupation that his nose emits a foul odor, described as "like a dead mouse." Despite repeated reassurance and medical evaluations, he remained convinced of the odor, engaging in excessive nasal cleansing and social avoidance. His symptoms led to marked functional impairment, marital separation, and social withdrawal.

Management and outcome: Treatment included fluoxetine titrated to 60 mg/day, risperidone 2 mg nightly, and supportive psychotherapy incorporating cognitive-behavioral techniques. Over the course of 1 year, the patient showed marked improvement in odor-related preoccupation, depressive symptoms, and social engagement, with no significant side effects reported.

Discussion: This case highlights the debilitating impact of olfactory reference syndrome and the diagnostic challenges it poses, particularly in differentiating from delusional disorder, primary psychotic disorders, and mood disorders with psychotic features.

Conclusion: Olfactory reference syndrome is an underrecognized condition with significant psychosocial consequences. Reporting this case from Ethiopia aims to raise awareness among clinicians in low-resource settings, encourage timely diagnosis, and promote multidisciplinary management.

Background: Fibrodysplasia ossificans progressiva is a rare genetic disorder characterized by the progressive heterotopic ossification of soft connective tissues, ultimately leading to cumulative disability and ankylosis. Airway management during general anesthesia in patients with fibrodysplasia ossificans progressiva is particularly challenging because of anatomical abnormalities and the risk of triggering flare-ups due to minor trauma. However, reports on the anesthetic management of pediatric patients remain limited.

Case presentation: We present the case of a 10-year-old Japanese girl with genetically confirmed fibrodysplasia ossificans progressiva who underwent an uneventful elective inguinal hernia repair under general anesthesia. She had hallux valgus at birth and was diagnosed with fibrodysplasia ossificans progressiva at the age of 2 years. Preoperative imaging revealed temporomandibular joint ankylosis with limited mouth opening (interincisal distance ~2 cm), cervical spine fusion, and thoracic heterotopic ossification. A high-flow nasal cannula was used for preoxygenation and apneic oxygenation. Mask ventilation was confirmed to be adequate after induction with remifentanil and propofol. Gentle mandibular elevation and nasally guided fiberoptic intubation were successfully performed while maintaining peripheral oxygen saturation levels above 99%. Anesthesia was maintained using remifentanil and sevoflurane. Perioperative corticosteroids and nonsteroidal anti-inflammatory drugs were administered to prevent flare-ups. The surgery lasted 82 minutes, and the patient was discharged uneventfully the following day without any signs of flare-up or airway complications.

Conclusion: General anesthesia in pediatric patients with fibrodysplasia ossificans progressiva requires meticulous planning to address the dual challenges of difficult airway management and the prevention of flare-ups. This case demonstrates that nasal fiberoptic intubation under high-flow nasal cannula oxygenation, combined with pharmacological prophylaxis and atraumatic handling, can contribute to safe anesthetic care. This patient case contributes to the limited body of literature on pediatric fibrodysplasia ossificans progressiva anesthesia, underscoring the practical strategies for managing the airway and minimizing the risk of flare-ups.

Background: Cerebral venous air embolism is a rare but potentially fatal complication of central venous catheter insertion and removal. Although most venous air emboli remain clinically silent or cause only transient symptoms, they may exceptionally lead to extensive arterial cerebral ischemia, particularly in patients with impaired pulmonary filtration or pre-existing lung disease. We report a distinctive sequence of computed-tomography-demonstrated cortical venous air followed by extensive right middle and posterior cerebral artery infarction on magnetic resonance imaging despite a negative saline-contrast echocardiography for intracardiac shunt, occurring after elective internal jugular central venous catheter removal in a frail patient with idiopathic pulmonary fibrosis.

Case presentation: We report the case of a 68-year-old Lebanese, right-side dominant, cachectic man with multiple comorbidities, including coronary artery disease, heart failure with reduced ejection fraction, pulmonary fibrosis on long-term oxygen therapy, chronic kidney disease, and a history of nephrectomy for metastatic renal carcinoma. He was admitted for weight loss and hypotension, diagnosed with cardiogenic shock, and managed with vasopressor support in the intensive care unit. After stabilization and transfer to the ward, removal of a right internal jugular central venous catheter led to the sudden onset of acute paraplegia, altered consciousness, and left-sided neurological deficits. A non-contrast brain computed tomography scan revealed multiple cortical venous air emboli, and magnetic resonance imaging confirmed extensive ischemic lesions in the right-middle and posterior cerebral artery territories. Despite immediate supportive measures and antiseizure treatment, the patient developed refractory status epilepticus. Given his advanced comorbidities, extensive cerebral injury, and poor pre-morbid status, a shared decision was made with the family to limit further aggressive treatment, and the patient died a few hours later.

Conclusion: This case suggests that venous cortical air embolism can be a hidden precursor of extensive arterial ischemia even without an intracardiac right-to-left shunt, particularly when pulmonary filtration reserve is limited. Clinicians should maintain a high index of suspicion when new neurological deficits appear after central venous catheter manipulation, ensure meticulous preventive measures and close observation after removal, and rapidly initiate appropriate supportive treatment if venous air embolism is suspected.

Background: Recurrent in-stent thrombosis is a rare but life-threatening complication following percutaneous coronary intervention, even when newer-generation stents and standard dual antiplatelet therapy are used. Mechanical issues such as stent malapposition and underexpansion, often undetectable by angiography, play a critical role in early stent failure. Additionally, systemic risk factors such as diabetes and dyslipidemia contribute significantly to thrombotic events. This case highlights the importance of high-resolution intravascular imaging in identifying hidden mechanical causes of stent failure and underscores the need for a comprehensive, individualized treatment approach.

Case presentation: A 43-year-old South Asian man with newly diagnosed type 2 diabetes and elevated cholesterol presented with acute chest pain and was diagnosed with an anterior ST-elevation myocardial infarction. He underwent primary percutaneous coronary intervention with drug-eluting stent placement in the proximal segment of the left anterior descending artery. Despite a successful procedure and appropriate medical therapy, the patient experienced two additional episodes of acute in-stent thrombosis within 17 days. These were managed with repeat balloon angioplasty, a second stent, and administration of intravenous antiplatelet agents. On his third presentation, intravascular imaging using optical coherence tomography revealed significant stent underexpansion and malapposition that were not appreciated during prior angiographic assessments. High-pressure balloon dilatation was performed to achieve full expansion and proper apposition of the stents. Following this, intensive secondary prevention strategies were implemented, including strict glucose control, high-dose cholesterol-lowering therapy, and continued antiplatelet treatment. The patient's left ventricular ejection fraction improved from ~30% at day 16 to 40-45% by month 6, with stability thereafter.

Conclusion: This case illustrates how recurrent in-stent thrombosis may occur despite adherence to current procedural and pharmacological standards. Mechanical causes, particularly those not visible on angiography, should be actively investigated using intravascular imaging in patients with unexplained or repeated stent thrombosis. Early identification and correction of these issues, combined with aggressive control of metabolic risk factors, are essential for preventing further events and improving clinical outcomes.

Background: Undifferentiated endometrial carcinoma is a solid tumor that originates from endometrial epithelial cells, accounting for approximately 9% of endometrial cancers. Undifferentiated endometrial carcinoma exhibits high invasiveness, is often diagnosed at an advanced stage, and demonstrates poor sensitivity to chemotherapy. Clinical manifestations lack distinct specificity, with the most common symptoms being abnormal uterine bleeding or discharge and lower abdominal pain.

Case description: We report the case of a 50-year-old Chinese female individual who was diagnosed with undifferentiated endometrial carcinoma. Surgery was successfully performed; however, on postoperative day 20, she developed fever, dyspnea, pulmonary infection, and renal insufficiency. Computed tomography scan revealed worsening lung metastases and abdominal tumor recurrence. The patient succumbed to respiratory failure on postoperative day 26.

Conclusion: The etiology of undifferentiated endometrial carcinoma remains unclear, and its treatment principles align with those for endometrial carcinoma. Patients with early stage disease should undergo staging surgery, while those with advanced-stage disease should follow the surgical approach for ovarian cancer, aiming to remove as much visible tumor tissue as possible. Advanced-stage patients experience rapid disease progression and typically face short-term mortality, with no highly effective treatment options currently available.

Background: Skin areas chronically exposed to solar radiation, such as the face, neck, scalp, and upper limbs, are at significantly increased risk of developing basal cell carcinoma. The objective of this case report is to describe the treatment of a basal cell carcinoma located on the chin using electrochemotherapy in a patient with advanced heart disease, a condition that contraindicated invasive surgical intervention.

Case presentation: An 83-year-old Caucasian female patient presented with a persistent nodular lesion on the chin, present for approximately 6 months. The diagnosis of cutaneous basal cell carcinoma was confirmed through histopathological analysis of tissue obtained via punch biopsy of the lesion. A single electrochemotherapy application was sufficient. Monitoring was discontinued at 24 months due to the patient's death from cardiac causes. Her clinical condition did not allow for further diagnostic procedures such as biopsy or positron emission tomography-computed tomography.

Conclusion: In the present study, the patient successfully underwent a single session of electrochemotherapy, with a favorable outcome and no complications. The therapeutic approach allowed us to avoid general anesthesia while ensuring effective treatment of the neoplastic lesion with excellent aesthetic outcomes.

Background: The association of right ventricular dysfunction, atrial tachyarrhythmias, and cardiac conduction system disease is rarely reported in literature. Previous case reports include right ventricular dysfunction associated with sudden cardiac death and ventricular arrhythmia. Other case reports with similar clinical presentation triad differ by either left ventricular involvement or having an abnormal presentation of systemic diseases.

Case presentation: We report a case series of right ventricular dysfunction, atrial tachyarrhythmias, and cardiac conduction system disease in a single family. The proband in this case series is a 70-year-old Egyptian male who presented to our care for dual-chamber pacemaker generator replacement. He had the pacemaker for 10 years because of complete heart block. Device interrogation showed recorded paroxysmal atrial fibrillation episodes. Transthoracic echocardiography showed normal left ventricle internal dimensions and ejection fraction, dilated right ventricle with preserved systolic function, severe tricuspid regurgitation, and systolic pulmonary artery pressure of 40 mmHg. On reviewing his old records, there was no right ventricular affection 10 years ago; progressive right ventricular dysfunction occurred over the last decade. Despite guideline-directed medical therapy, the patient's clinical status deteriorated, and surgical tricuspid valve replacement was performed with a tissue valve. Since the surgical intervention, his clinical status and functional capacity improved, and repeated echocardiography showed no tricuspid regurgitation with persistence of the right ventricular dilatation but with normal right ventricular function. On reviewing his family history, the triad of right ventricular dysfunction, atrial tachyarrhythmias, and cardiac conduction system disease occurred in affected family members with various presentations. There was no left ventricular involvement, no history of ventricular arrhythmias in any of the affected members, and there was no history of sudden cardiac death across all three generations.

Conclusion: Familial clustering of right ventricular dysfunction, atrial tachyarrhythmias, and cardiac conduction system disease do exist. This constellation of findings is not well described in literature. Further reporting of similar cases and investigating of genetic bases are required.

Background: Sacituzumab-govitecan-related pulmonary toxicity is rare. Herein, we describe a case of sacituzumab-govitecan-induced interstitial pneumonitis that was successfully rechallenged with a reduced drug dose.

Case presentation: A 62-year-old Taiwanese female patient with metastatic triple-negative breast cancer was refractory to the previous four lines of chemotherapy. She was subsequently given therapy with sacituzumab govitecan (10 mg/kg). After the second cycle, she developed progressive dyspnea, dry cough, and diarrhea. Chest computed tomography revealed diffuse ground grass opacity of the bilateral lungs with multiple foci of black area (reverse hall sign) and thickening of the interlobular septa. In the absence of other potential causes, sacituzumab-govitecan-induced grade 3 interstitial pneumonitis was strongly suspected, with a Naranjo score of 7. Sacituzumab govitecan discontinuation and corticosteroid therapy (prednisolone 2 mg/kg/day) were initiated. Her clinical symptoms and radiographic pulmonary infiltrations were significant improvement within 1 week. Sacituzumab govitecan was rechallenged in half dose due to lack of alternative treatment for the metastatic triple-negative breast cancer. She completed four additional cycles without recurrent pneumonitis before disease progression. The progression-free survival was 5.37 months.

Conclusion: Sacituzumab govitecan is a new generation of antibody-drug conjugates. Early detection of drug-induced interstitial lung disease, drug discontinuation ,and immediate steroid therapy are important. Permanent antibody-drug conjugates discontinuation is advised for patients with grade ≥ 2 drug-induced interstitial lung disease. In this case, due to the limited treatment for metastatic triple-negative breast cancer, the patient was rechallenged with half dose of sacituzumab govitecan without recurrent pneumonitis.

Background: Coronavirus disease 2019, is an infectious disease caused by the severe acute respiratory syndrome corona virus type-2 virus. Children affected are usually asymptomatic and those that have symptoms tend to be milder and infants are most at risk for severe disease. We describe the first two cases of coronavirus disease 2019 deaths among infants with complication of severe acute malnutrition in Sierra Leone. In the first half of June 2020, Kenema district reported first two coronavirus disease 2019 deaths of infants in Sierra Leone. We conducted a death review to understand the underlying medical conditions and predisposing factors of the deaths given the rarity of death among children with coronavirus disease 2019 disease. We describe the clinical presentation of the cases and their possible underlying medical conditions.

Case presentation: The two infant decedents were 8-month-old male and 6-month-old female Sierra Leoneans who presented with fever, vomiting, and flu-like symptoms and had complications of severe acute malnutrition and were coinfected with coronavirus disease 2019. Both patients developed severe respiratory distress which were not resolved with antibiotics and oxygen therapy. In addition, the diagnosis of coronavirus disease 2019 infection in both cases were also delayed due to low index of suspicion, and lack of treatment for coronavirus disease 2019 exacerbated their likelihood of death.

Conclusion: The severe acute malnutrition made the infants prone to severe coronavirus disease 2019 disease and thus led to their death. We recommend community sensitization on good nutrition for infants to increase the chance of survival of children with coronavirus disease 2019 infection.