OpenMed/multicare-articles huggingface dataset:
Successful Elective Thoracoscopic Resection of Complicate Extralobar Bronchopulmonary Sequestration after Intrafoetal Vascular Laser Ablation: The Paediatric Surgeon's Point of View
Authors: Giulia Fusi, Agnès Sartor, Marion Groussolles, Solene Joseph, Julie Vial, Lea Roditis, Christophe Vayssière, Olivier Abbo
Journal: Case Rep Pediatr (2023)
MeSH terms: Case Reports

Few reports of laser coagulation for foetal bronchopulmonary sequestration (BPS), a rare congenital malformation characterised by the absence of tracheobronchial connection and the presence of a systemic feeding artery, have been published. Additionally, very few of them focus also on the postnatal management, with results limited and controversial. Postnatal treatment of residual malformation remains debated, hence the need to share our experience of a combined pre- and postnatal approach to complicated extra-lobar BPS. We report the case of a female foetus with the diagnosis of a pulmonary lesion. Due to mediastinal shift, unilateral compressive hydrothorax, ascites, and hydrops, a foetal treatment with ultrasound-guided laser coagulation of the anomalous vessel was performed. At birth, due to the persistence of the malformation, an elective delayed thoracoscopical surgery was performed. Prenatal laser ablation for complicated BPS is a life-saving procedure not always resulting in lesion disappearance. Thoracoscopical surgical exploration in case of persistent lesions at birth offers the possibility of a minimally invasive sequestrectomy feasible and safe.
Urethral Caruncle in Pediatrics: A Northern Tanzania Experience
Authors: Anteneh Tadesse Kifle, Janeth Mpelumb, Frank Bright, Orgeness Jasper Mbwambo, Tizazu Abebayehu Tsega
Journal: Case Rep Urol (2024)
MeSH terms: Case Reports

Urethral caruncles are the most frequent benign tumors of the female urethra. Most of them are found in postmenopausal women, and they are rare in childhood. Only a few pediatric cases have been published in the literature. In this report, we present a case series of three pediatric patients with a urethral caruncle.
Sternal Dislocation and Associated Lung Lobe Hernia in a Cat
Authors: Martin Hamon, Philippe Haudiquet, Aurelie Bruwier
Journal: Case Rep Vet Med (2024)
MeSH terms: Case Reports

Sternal luxation/dislocation is a rare condition and is most often the result of trauma. Medical and surgical management have been described, with scarce information regarding the best treatment option for these cases. A 1-year-old domestic shorthair cat was presented for severe sternal dislocation and a left humeral fracture. Given the displacement observed at the level of the sternum and pain associated, surgical stabilization was elected. A partial lung lobe hernia was identified during the open stabilization of the sternum. Management of the hernia and sternal luxation stabilization were performed with the aid of video-assisted thoracoscopy. The cat recovered uneventfully, and no postoperative complications were reported.
Efficiency trends of essential public health services and possible influencing factors since the new round health reform in China: a case study from Hainan Province
Authors: Ye Tian, Jia Peng, Yumei Liu, Jiayan Huang
Journal: Front Public Health (2023)
Keywords: malmquist productivity index, tobit regression, data envelopment analysis, efficiency, public health service
MeSH terms: Efficiency, Organizational, Health Care Reform, Research Support, Non-U.S. Gov't, Aged, China, Ethnicity, Health Services, Humans, Minority Groups

Objective:
This article aimed to evaluate the efficiency trends and influencing factors of essential public health services in Hainan Province after the healthcare reform launched in 2009 in China.
Methods:
The efficiency of essential public health services (EPHS) at primary health institutions was assessed using data envelopment analysis (DEA), and the efficiency change was analyzed by employing the Malmquist productivity index (MPI). We used Tobit regression to identify the influence of environmental factors on the efficiency of public health services. The bootstrap method was adopted to reduce the impact of random errors on the result.
Results:
The bootstrapping bias-corrected efficiency revealed that the average values of technical efficiency, pure technical efficiency, and scale efficiency were 0.7582, 0.8439, and 0.8997, respectively, which meant that the EPHS in Hainan Province were not at the most effective state. The average bias-corrected MPI was 1.0407 between 2010 and 2011 and 1.7404 between 2011 and 2012. MPIs were less than 1.0000 during other periods investigated, ranging from 0.8948 to 0.9714, indicating that the efficiency of EPHS has been decreasing since 2013. The Tobit regression showed that the regression coefficients of per capita GDP, population density, the proportion of older people aged over 65, and the proportion of ethnic minority population were 0.0286, -0.0003, -0.0316, and - 0.0041 respectively, which were statistically significant (p < 0.05).
Conclusion:
There was a short-term improvement in the efficiency of EPHS in Hainan after the launch of the new round of health reform. However, this trend has not been sustained after 2013. In particular, equalized financial investment in essential public health could not fulfill the needs of poor counties. This has resulted in the inability to improve scale efficiency in some counties, which in turn has affected the improvement of overall EPHS efficiency. Therefore, to promote EPHS efficiency sustainably, it is suggested that under this model of provincial control of counties, the equity of resource allocation should be effectively improved while further advancing the technology of service delivery.
Utility of Pulmonary Angiography by 128-Slice Computed Tomographic Scanner in Diagnosis of Tetralogy of Fallot Cases
Authors: Abhishek Dwivedi, Ankur Sharma, Rachit Sharma, Prateek Awasthi, Satveer Singh Choudhary
Journal: Case Rep Radiol (2024)
MeSH terms: Case Reports

Tetralogy of Fallot (TOF) is a significant cause of cyanotic congenital heart disease (CHD) encountered in childhood with few cases manifesting in adulthood. It has four classical features (ventricular septal defect, overriding of aorta, hypertrophy of right ventricular hypertrophy, and right ventricular outflow tract obstruction), but the clinical presentation and course can be variable. Due to various anatomical variations and complex anatomy, presurgical planning and postoperative follow-up by pulmonary computed tomographic angiography (CTA) have a very important role. With continued technological advances and the availability of 128-slice computed tomographic (CT) scans, they now play an important role in TOF preoperative evaluation and workup, assisting by minimizing routine invasive digital subtraction catheter angiography. The fast scan of a 128-slice CTA with very sensitive detectors is a very useful modality for studying the complex anatomy and variations as well as its utilization for postoperative management. In this article, we report four cases of TOF where we used a 128-slice scan for performing pulmonary angiography (Optima 660, GE 128, 2180 Premier Row, Orlando, FL 32809, U.S.A.) for preoperative diagnosis and management of three cases and work up for revision surgery for an already operated case with a nonfunctional modified Blalock-Taussig shunt with additional lung parenchymal findings simultaneously. This study will explain the advantageous role of the 128-slice CT scanner over the lesser-slice CT scanners with the ability of pulmonary CTA to facilitate accurate diagnosis and postoperative management.
Late Airway Compromise Secondary to Dural Tear and Cerebrospinal Fluid Leak
Authors: Larry R Hutson Jr, Russell K McAllister
Journal: Ochsner J (2023)
Keywords: airway management, airway obstruction, cerebrospinal fluid leak, cervical vertebrae, neck
MeSH terms: Case Reports

Background: While dysphagia after anterior cervical spine surgery is common, a dural tear is a rare complication. Airway compromise resulting from cerebrospinal fluid collection is an even rarer complication that has only been described to occur in the first few days postoperatively. Case Report: A 55-year-old male presented with progressive dysphagia and respiratory compromise 3 weeks after anterior cervical discectomy and fusion surgery at C3-C6. Imaging demonstrated extensive fluid collection in the retropharyngeal space and lateral neck, resulting in displacement of the cricoid cartilage rightward and anteriorly while also narrowing the pharyngeal space. After the patient's airway was secured by awake fiberoptic intubation, the fluid was determined to be cerebrospinal fluid (CSF) from a cervical dural tear. The tear was identified and repaired. The patient was extubated the next day, and a lumbar drain was placed to reduce the strain on the repair. After 11 days in the hospital, the patient made a full recovery. Conclusion: Dural tears following cervical disc surgery are rare and almost always identified in the immediate postoperative period; however, a dural tear should still be considered when a patient presents with a fluid collection at a later date. While techniques for securing the airway would not be different based on the type of fluid, knowing that the fluid collection is CSF could prompt the anesthesia team to place a lumbar drain.
Case report: Successful treatment of a rare HER2-positive advanced breast squamous cell carcinoma
Authors: Gui Wang, Chenghui Yang, Donglin Zeng, Jihao Wang, Huaxin Mao, Yu Xu, Chao Jiang, Zhen Wang
Journal: Front Pharmacol (2024)
Keywords: her2, antibody drug conjugate, breast squamous cell carcinoma, metaplastic breast cancer, targeted therapy
MeSH terms: Case Reports

Background: Breast squamous cell carcinoma (SCC) is an uncommon and highly aggressive variant of metaplastic breast cancer. Despite its rarity, there is currently no consensus on treatment guidelines for this specific subtype. Previous studies have demonstrated that chemotherapy alone has limited efficacy in treating breast SCC. However, the potential for targeted therapy in combination with chemotherapy holds promise for future treatment options. Case presentation: In this case report, we present a patient with advanced HER2-positive breast SCC, exhibiting a prominent breast mass, localized ulcers, and metastases in the lungs and brain. Our treatment approach involved the administration of HER2-targeted drugs in conjunction with paclitaxel, resulting in a sustained control of tumor growth. Conclusion: This case represents a rare occurrence of HER2-positive breast SCC, with limited available data on the efficacy of previous HER2-targeted drugs in treating such patients. Our study presents the first application of HER2-targeted drugs in this particular case, offering novel therapeutic insights for future considerations. Additionally, it is imperative to conduct further investigations to assess the feasibility of treatment options in a larger cohort of patients.
Exploring the Role of the Pulvinar Nucleus of the Thalamus in Occipital Lobe Epilepsy: A Case Report
Authors: Hael Abdulrazeq, Anna R Kimata, Andrew Blum, Athar N Malik, Wael F Asaad
Journal: Cureus (2024)
Keywords: epilepsy surgery, intracranial electroencephalography, neuromodulation, pulvinar nucleus, thalamus
MeSH terms: Case Reports

Understanding the role of the pulvinar nucleus may be critical for guiding circuit-targeted neurosurgical intervention in some patients. In this report, a 33-year-old female presented with focal onset occipital epilepsy with secondary generalization and with a previously radiated arteriovenous malformation within the right primary visual cortex. Phase II monitoring demonstrated the pulvinar nucleus was not involved in subclinical seizures restricted to the primary visual cortex, but it did become involved in clinical events with more extensive seizure spread into higher visual cortical regions. She underwent responsive neurostimulation (RNS) with implantation of leads within the primary visual cortex. This case demonstrates the late propagation of epileptic activity from the visual cortex to the pulvinar nucleus and illustrates the pulvinar nucleus' connections with higher-order visual areas.
Case Report: Mast cell anergy: absence of symptoms after accidental re-exposure to amoxicillin/clavulanic acid 3 days after anaphylaxis
Authors: Loris Guyénard, Marie Tauber, Sophie Debord-Peguet, Frédéric Berard, Audrey Nosbaum, Florence Hacard, Mariana Castells, Jean-François Nicolas
Journal: Front Allergy (2024)
Keywords: beta-lactams, case report, drug allergy, skin test, systemic reaction
MeSH terms: Case Reports

Empty mast cell syndrome, also named post anaphylaxis mast cell anergy (PAMA), is a temporary state of loss of mast cell responsiveness after a severe immediate hypersensitivity reaction. In this study, we describe a case of PAMA after accidental re-exposure to amoxicillin in a patient who developed severe anaphylaxis to this drug three days earlier in the operating room. To our knowledge, this report is the second to document this phenomenon.
Primary malignant melanoma of the parotid gland, a rare case arising from a duct: a case report and literature review
Authors: Moe Thu Zar Aung, Jo-Eun Kim, Hye-Jung Yoon, Kyoung-Hoe Huh, Won-Jin Yi, Min-Suk Heo, Sam-Sun Lee
Journal: Front Oncol (2024)
Keywords: computed tomography, diagnosis, magnetic resonance imaging, malignant melanoma, parotid gland
MeSH terms: Case Reports

Malignant melanoma of the parotid gland is an unusual tumor in the head and neck region, and most parotid melanoma is reported as a metastatic lesion of cutaneous malignant melanoma. We report a case of primary malignant melanoma arising in the parotid gland duct with diagnostic challenge. The patient was a 68-year-old man who complained of repeated right facial swelling that presented 3 months prior. Swelling was detected along the Stensen's duct of the cheek, and brown-colored saliva-like fluid was aspirated. On MR and CT images, a fluid-filled duct with small nodule and heterogeneously enhancing mass in the parotid parenchyma was detected. The nodular mass on the ductal wall grew rapidly, and the hyperintense T1 signal became significant on follow-up images. The final diagnosis via histopathologic examination using biopsy and parotidectomy specimen revealed the lesion as malignant melanoma of the duct and pleomorphic adenoma of the parenchyma. Even if the incidence of primary malignant melanoma is very low among tumors occurring in the parotid gland, efforts supporting an early diagnosis using imaging characteristics are important.
Vaginal Cuff Dehiscence Following Remote Hysterectomy: An Unusual Case of Bowel Evisceration
Authors: Harsh Desai, Alexander Canales
Journal: Case Rep Surg (2024)
MeSH terms: Case Reports

Hysterectomy is one of the most common gynecologic procedures performed worldwide. Vaginal cuff dehiscence with resultant bowel evisceration, while rare, is one of the most serious complications of the procedure. We present the case of a 79-year-old female with vaginal cuff dehiscence several decades after hysterectomy. The patient had no significant antecedent symptoms until she experienced bowel evisceration during Valsalva. She underwent a successful reduction of bowel contents with limited resection and transvaginal cuff repair. This case highlights the risk of vaginal cuff dehiscence even decades following the procedure.
Case report: Late-onset MELAS syndrome with mtDNA 5783G>A mutation diagnosed by urinary sediment genetic testing
Authors: Hao Cai, Li-Min Li, Miao Zhang, Yuying Zhou, Pan Li
Journal: Front Genet (2024)
Keywords: melas, mt-tc, late-onset, m5783g>a, urinary sediment
MeSH terms: Case Reports

Background:
Patients with mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes (MELAS) usually present with multisystemic dysfunction with a wide range of clinical manifestations. When the tests for common mitochondrial DNA (mtDNA) point mutations are negative and the mtDNA defects hypothesis remains, urine epithelial cells can be used to screen the mitochondrial genome for unknown mutations to confirm the diagnosis.
Case presentation:
A 66-year-old Chinese woman presented with symptoms of MELAS and was initially misdiagnosed with acute encephalitis at another institution. Although genetic analysis of blood lymphocyte DNA was negative, brain imaging, including magnetic resonance imaging, magnetic resonance spectroscopy, and clinical and laboratory findings, were all suggestive of MELAS. Finally, the patient was eventually diagnosed with MELAS with the mtDNA 5783G>A mutation in the MT-TC gene with a urinary sediment genetic test.
Conclusion:
This case report expands the genetic repertoire associated with MELAS syndrome and highlights the importance that full mtDNA sequencing should be warranted beside the analysis of classical variants when a mitochondrial disorder is highly suspected. Furthermore, urine sediment genetic testing has played a crucial role in the diagnosis of MELAS.
Case report: Is bilateral renal dioctophymosis and severe uremia in a dog synonymous of euthanasia? Not today
Authors: Pâmela Caye, Jean Carlos Gasparotto, Anna Vitória Hörbe, Letícia Rodrigues Leitao Andrade, Rainer da Silva Reinstein, Luiza Tonietto Mangini, Daniel Curvello de Mendonça Müller, Maurício Veloso Brun
Journal: Front Vet Sci (2024)
Keywords: dioctophyme renale, dioctophymosis, giant kidney worm, hyperbaric oxygen therapy, nephroscopy
MeSH terms: Case Reports

A dog with bilateral renal dioctophymosis presented with stage 5 acute kidney injury, weight loss, vomiting, apathy, and hematuria. Laboratory tests showed creatinine of 17.2 mg/dL and Dioctophyme renale eggs in the urine. It underwent a 30-min session of hyperbaric oxygen preconditioning at a pressure of 2 ATA. Subsequently, bilateral nephroscopy was performed, without warm ischemia, using Amplatz-type renal dilators. Five parasites were removed, three females from the right kidney, one female from the left kidney, and one male from the abdominal cavity. After surgery, the patient continued doing daily hyperbaric oxygen therapy (HBOT) sessions and clinical therapy. Postoperative care consisted of analgesics, antimicrobials, antioxidants, gastric protector and fluid therapy. Ultrasound monitoring showed a reduction in the area of renal dilation and the hematological and biochemical tests showed rapid recovery from acute kidney injury. There was no bacterial growth in the urine sample collected directly from the kidneys. The patient had an excellent clinical progression and was discharged from hospital 7 days postoperatively, with creatinine values of 2.9 mg/dL. This is the first report of the use of nephroscopy in the treatment of dioctophymosis and indicates excellent chances of cure even in severe cases of bilateral parasitosis. HBOT was shown to be an ally in the clinical therapy of patients with D. renale by helping with stabilization and postoperative recovery.
A novel PHKA2 variant in a Chinese boy with glycogen storage diseases type IXa
Authors: Hongdan Zhu, Tao Zhang, Hua Yuan, Yan Chen, Jinlong Ding, Haigang Ding, Xiaoliang Shi, Dalei Gu, Yingying Ma
Journal: Front Endocrinol (Lausanne) (2023)
Keywords: 3d structure, phka2 gene, x-chromosome recessive genetic diseases, glycogen storage disease, whole exome sequencing
MeSH terms: Amino Acids, Glycogen Storage Disease / diagnosis, Glycogen Storage Disease / genetics, Phosphorylase Kinase / genetics, Research Support, Non-U.S. Gov't, Child, East Asian People, Humans, Male, Amino Acids, PHKA2 protein, human, Phosphorylase Kinase

Background:
Glycogen storage diseases (GSDs) are a group of heterogeneous inherited metabolic disorders with an incidence of 4%-5%. There are 19 types of GSDs, making diagnosis one of the greatest challenges.
Methods:
The proband and his parents were referred to our hospital for genetic diagnosis. Ultrasound screening suggested hepatomegaly. A novel insertion variant NM_000292 c.1155_1156insT (p. 386N>*) in PHKA2 gene was identified using trio whole exome sequencing (Trio-WES), which resulted in the codon of amino acid 386 from asparagine to termination (p. 386N>*). The 3D mutant protein structure was predicted using AlphaFold, and the results showed that the truncated PHKA2 protein contained 385 of the 1,235 amino acids of the mature protein.
Conclusion:
We describe a previously unreported case of a GSDs IXa type Chinese boy caused by a novel PHKA2 variant. This clinical case contributes to the understanding of the characteristics of GSDs type IXa and expands the variants spectrum of genes related to GSDs type IXa. Our findings demonstrated the significance of genetic testing in the diagnosis of GSDs.
Application of eculizumab, a terminal complement inhibitor, in the management of atypical hemolytic uremic syndrome in a 14-month-old Chinese pediatric patient: a case report
Authors: Xin Wei, Xinzhu Liu, Yingying Yu, Wei Xie, Wentao Luo, Ye Tu, Shuhong Bu, Guimei Guo
Journal: Front Pediatr (2024)
Keywords: ahus, complement, eculizumab, pediatric, thrombotic microangiopathy
MeSH terms: Case Reports

Eculizumab, a recombined humanized monoclonal antibody which possesses high affinity for the complement protein C5, is a therapeutic agent utilized in the treatment of atypical hemolytic uremic syndrome (aHUS) by inhibiting the terminal complement complex C5b-9. In a pediatric patient with aHUS of 14 months, the administration of eculizumab therapy was accompanied by the inclusion of meningococcal vaccine as part of the national immunization program. Notably, no other antibiotics were administered prior to or during the course of eculizumab treatment. Moreover, there were no occurrences of infusion reactions or meningococcal infections observed throughout the course of treatment. Due to the presence of anti-factor H antibodies and insufficient recovery, glucocorticoids and eculizumab were administered at week 0 and week 1, followed by the initiation of mycophenolate mofetil (MMF) at a dosage of 250 mg (approximately 548 mg/m2) per day starting from Day 10. Due to the recovered of complement antibody after 8 doses of eculizumab, the therapeutic interval was extended from once every 3 weeks to once a month since 9th administration. We experienced and successfully treated a rare case of aHUS with eculizumab in a 14-month-old Chinese pediatric patient.
Drug-induced lung injury in a patient treated with prior atezolizumab and subsequent sotorasib: A case report
Authors: Yuta Takahashi, Hiroaki Tachi, Ryo Watanabe, Kei Shimizu, Yusuke Yamamoto
Journal: Respirol Case Rep (2024)
Keywords: clinical diagnosis, drug‐induced lung injury, immune checkpoint inhibitor, response to prednisolone, sotorasib
MeSH terms: Case Reports

We report a case of drug-induced lung injury treated with prior atezolizumab and subsequent sotorasib. The patient was a 62-year-old woman with lung adenocarcinoma harbouring a KRAS G12C mutation that was resistant to chemotherapy, including immune checkpoint inhibitors. Cough and dyspnea appeared on day 80 after sotorasib was administered as second-line therapy, and chest computed tomography revealed ground glass opacities in all lung lobes. Bronchoalveolar lavage fluid showed an increased total cell count with lymphocyte predominance. The patient was considered to have lung injury caused by prior atezolizumab or sotorasib administration. Withdrawal of sotorasib did not improve symptoms and shadows in both lungs. We administered moderate-dose prednisolone and the lung disorder quickly resolved. Prednisolone tapering was completed in 2 months, followed by several months without relapse. Definitive identification of the responsible drug for the drug-induced lung injury proved challenging in the setting of exposure to multiple potential inciting agents. There is a need for high levels of clinical suspicion for timely evaluation and management.