OpenMed/multicare-articles huggingface dataset:
Vagus Nerve Stimulation (VNS) in Super Refractory New Onset Refractory Status Epilepticus (NORSE)
Authors: Mohankumar Kurukumbi, James Leiphart, Anam Asif, Jing Wang
Journal: Case Rep Neurol Med (2019)
MeSH terms: Case Reports

The treatment protocol of status epilepticus has many associated toxicities so there is interest in alternate nonmedicinal therapies for managing New Onset Refractory Status Epilepticus (NORSE) patients. Vagus nerve stimulation (VNS) is an FDA-approved therapy for refractory epilepsy that has been shown to decrease the frequency and severity of seizures. We present the case of a patient with new-onset refractory status epilepticus (NORSE) whose seizures were successfully treated with vagus nerve stimulation. A 25-year-old male with no history of epilepsy or other neurological disorders presented with altered mental status and generalized tonic-clonic seizures following a two-week history of an upper respiratory tract infection. Lumbar puncture showed neutrophilic pleocytosis, and he was treated for bacterial and viral meningoencephalitis. In spite of treatment, his seizures began increasing in frequency. On day three, the patient entered status epilepticus (SE) refractory to intensive pharmacotherapy with maximal doses of valproate, levetiracetam, and propofol. On day four, SE remained refractory, so pentobarbital was introduced with targeted burst suppression pattern on electroencephalography (EEG). Patient continued to be refractory to these measures, so a vagus nerve stimulator (VNS) was implanted (day eight). Following VNS implantation, EEG demonstrated significant reduction of seizure activity and subsequent magnet swiping continued aborting electrographic seizures. No SE or electrographic seizures were reported for seventy-two hours, but few occasional discharges were reported. Seizures eventually recurred on day fourteen and the patient succumbed to his multiple comorbidities on day seventeen. Due to the efficacy of VNS in refractory epilepsy, there was interest in using it in refractory status epilepticus. Multiple case reports have described a benefit from implantation of VNS in the treatment of SE. The successful use of VNS to acutely terminate status epilepticus for seventy-two hours in this critically ill patient adds to current evidence that there is utility in using VNS for refractory status epilepticus.
Dentigerous Cyst associated with Impacted Maxillary Premolar: A Rare Site Occurrence and a Rare Coincidence
Authors: Nitul Jain, Gaurav Gaur, Vivek Chaturvedy, Ajay Verma
Journal: Int J Clin Pediatr Dent (2018)
Keywords: dentigerous cyst, impacted premolars, missing teeth, nonsyndromic patient
MeSH terms: Case Reports

A dentigerous cyst is a developmental odontogenic cyst occurring most commonly in the third molar region of mandible and maxilla and maxillary canine region followed by mandibular premolar areas. However, dentigerous cyst occurring in association with impacted maxillary premolars is a very rare presentation accounting for less than 0.5% of total dentigerous cyst cases. In the following case report, a dentigerous cyst was reported in maxillary premolar region in a nonsyndromic patient having one missing premolar and another malformed one associated with the dentigerous cyst. How to cite this article: Jain N, Gaur G, Chaturvedy V, Verma A. Dentigerous Cyst associated with Impacted Maxillary Premolar: A Rare Site Occurrence and a Rare Coincidence. Int J Clin Pediatr Dent 2018;11(1):50-52.
Case Report: Long-Term Survival With Anlotinib in a Patient With Advanced Undifferentiated Large-Cell Lung Cancer and Rare Tonsillar Metastasis
Authors: Tianwei Xu, Chenchen Wei, Xiaoteng Zou, Binbin Lu, Zhaoxia Wang
Journal: Front Oncol (2021)
Keywords: anlotinib, antiangiogenic therapy, platelet-derived growth factor receptors (pdgfr), tonsillar metastasis, undifferentiated large-cell lung cancer
MeSH terms: Case Reports

Undifferentiated large-cell lung cancer is a rare type of non-small cell lung cancer (NSCLC) with a poor prognosis. It is insensitive to chemotherapy and easily develops drug resistance. Analysis of the Surveillance, Epidemiology, and End Results (SEER) database showed that patients with stage IV undifferentiated large-cell lung cancer had a median overall survival (OS) of only 4 months and that those who received chemotherapy had a median OS of only 5 months longer than those who did not. For the first time, we report a case of advanced large-cell undifferentiated lung cancer with rare tonsil metastasis. The patient developed resistance after 3 months of platinum-based systemic chemotherapy and local treatment. Antiangiogenic therapy has been continuously progressing and has shown certain efficacy in treating many malignant tumors, such as lung cancer. However, there are no relevant studies or case reports on antiangiogenic therapy in the treatment of undifferentiated large-cell lung cancer. Anlotinib, an orally delivered small-molecule antiangiogenic tyrosine kinase inhibitor (TKI), was administered to this patient after chemotherapy resistance occurred, and the outcome was assessed as continued stable disease (SD). As of the last follow-up evaluation, the progression-free survival (PFS) of the patient was 21.5 months, and the OS was 27.5 months. Retrospective immunohistochemical analysis showed that the patient was positive for one of the targets of anlotinib (PDGFR). In general, the findings in this case suggest that anlotinib may be an option with good efficacy for patients with large-cell undifferentiated lung cancer after chemotherapy resistance that may have good efficacy and also suggest that PDGFR may be the target underlying this effect.
Adult Periodic Alternating Nystagmus Masked by Involuntary Head Movements
Authors: Diego Kaski, Salman Haider, Amanda Male, Alex Radunovich, Fan Liu, Carla Cordivari, Kailash P Bhatia, Adolfo M Bronstein
Journal: Front Neurol (2018)
Keywords: head tremor, oscillopsia, periodic alternating nystagmus, psychogenic, rhombencephalitis, vestibulo-ocular reflex
MeSH terms: Case Reports

Acquired periodic alternating nystagmus (PAN) describes a horizontal jerk nystagmus that reverses its direction with a predictable cycle, and is thought to arise from lesions involving the brainstem and cerebellum. We report a 20-year-old patient with PAN who presented with an acute vertiginous episode and developed an involuntary head movement that initially masked the PAN. The involuntary head movements were abolished with a subtherapeutic dose of botulinum toxin to the neck muscles. We propose that the head movements initially developed as a compensatory movement to the nystagmus, to maintain visual fixation in the presence of the underlying nystagmus, and became an entrained involuntary behavior. This case highlights the importance of disambiguating psychogenic from organic pathology as this may have clinical therapeutic implications, in this case resolution of the most disabling symptom which was her head oscillations, leading to improved day-to-day function despite PAN.
Apolipoprotein-A-I for severe COVID-19-induced hyperinflammatory states: A prospective case study
Authors: Stanislas Faguer, Arnaud Del Bello, Chloé Danet, Yves Renaudineau, Jacques Izopet, Nassim Kamar
Journal: Front Pharmacol (2022)
Keywords: apoa-i, covid-19, hdl, apolipoprotein-a-i, cytokine storm, endothelium, inflammation

Viral infections can promote cytokine storm and multiorgan failure in individuals with an underlying immunosuppression or specific genetic background. Hyperinflammatory states, including critical forms of COVID-19, are characterized by a remodeling of the lipid profile including a dramatic decrease of the serum levels of apolipoprotein-A-I (ApoA-I), a protein known for its capacity to reduce systemic and lung inflammation, modulate innate and adaptive immunity, and prevent endothelial dysfunction and blood coagulation. In this study, four immunocompromised patients with severe COVID-19 cytokine storm that progressed despite standard-of-care therapy [Omicron (n = 3) and Delta (n = 1) variants] received 2- 4 infusions (10 mg/kg) of CER-001, an ApoA-I-containing HDL mimetic. Injections were well-tolerated with no serious adverse events. Three patients treated while not on mechanical ventilation had early clinical and biological improvement (oxygen withdrawal and correction of hematological and inflammatory parameters, including serum levels of interleukin-8) and were discharged from the hospital 3-4 days after CER-001 infusions. In the fourth patient who received CER-001 after orotracheal intubation for acute respiratory distress syndrome, infusions were followed by transient respiratory improvement before secondary worsening related to ventilation-associated pneumonia. This pilot uncontrolled exploratory compassionate study provides initial safety and proof-of-concept data from patients with a COVID-19 cytokine storm receiving ApoA-I. Further randomized controlled trial evaluation is now required to ascertain whether ApoA-I has any beneficial effects on patients with a COVID-19 cytokine storm.
A case of welder's pneumoconiosis treated with corticosteroid followed by nintedanib
Authors: Hideyuki Kaida, Takuhide Utsunomiya, Yohei Koide, Yusuke Ueda, Kenji Wada, Yuji Yoshida, Yoshiaki Kinoshita, Hisako Kushima, Hiroshi Ishii
Journal: Respir Med Case Rep (2022)
Keywords: alveolar hemorrhage, ct, computed tomography, ipf, idiopathic pulmonary fibrosis, nintedanib, pf-ild, progressive fibrosing interstitial lung disease, progressive fibrosing interstitial lung disease, welder's pneumoconiosis
MeSH terms: Case Reports

A 32-year-old man who had worked as a welder for 13 years was hospitalized for a fever and hemosputum with dyspnea. He was diagnosed with welding fume-associated lung disease with alveolar hemorrhaging and acute respiratory failure. Despite surviving the acute phase with corticosteroid therapy, hypoxemia persisted after a month and a half, requiring home oxygen therapy. As a result of the introduction of nintedanib, his clinical findings gradually improved, and the patient was weaned from oxygen therapy after six months. Inhalation of a large amount of welding fumes in a short period can cause alveolar hemorrhaging and prolonged pulmonary dysfunction.
Development of Extragenital Lichen Sclerosus in Malignant Melanoma Patients Treated With Ipilimumab in Combination With Nivolumab
Authors: S Morteza Seyed Jafari, Laurence Feldmeyer, Robert E Hunger
Journal: Front Oncol (2020)
Keywords: checkpoint inhibitors, extragenital lichen sclerosus et atrophicus, ipilimumab, malignant melanoma, metastatic, nivolumab
MeSH terms: Case Reports

The immune checkpoint inhibitors opened a new era for the treatment of melanoma. Nowadays, combined immune checkpoint inhibitors are administered to provide additive or synergistic effects on anti-melanoma immunity. The use of these drugs comes with serious adverse events related to excessive immune activation. Here, we present development of extragenital lichen sclerosus in a patient with metastatic malignant melanoma, during the combined therapy with checkpoint inhibitors.
An Unusual Presentation of Kawasaki Disease: Gallbladder Hydrops and Acute Cholestatic Hepatitis
Authors: B O Kılıç, Ş Baysun, T C Gökşen, I Akınbingöl, Z Arslan
Journal: Case Rep Med (2018)
MeSH terms: Case Reports

Kawasaki disease is the most common vasculitis of childhood. In its classical form, at least four of five diagnostic criteria including cervical lymphadenopathy (1.5 cm or more), nonsuppurative conjunctivitis, intraoral mucosal changes, edema in hands and feet, and maculopapular rash are required with prolonged fever over 5 days. Atypical cases which are different from the classical type or incomplete cases which does not include all the diagnostic criteria can be seen. The typical Kawasaki disease is a self-limiting disease with fever lasting for an average of two weeks. In such patients who have not been diagnosed and whose treatment has been delayed, coronary artery aneurysm, myocardial depression, arrhythmia, and vascular complications may increase morbidity and mortality. We would like to present a rare case of an atypical patient with gallbladder hydrops and acute cholestatic hepatitis.
Long-Term Outcomes of Radial Keratotomy, Laser In Situ Keratomileusis, and Astigmatic Keratotomy Performed Consecutively over a Period of 21 Years
Authors: Priyanka Chhadva, Florence Cabot, Anat Galor, Sonia H Yoo
Journal: Case Rep Ophthalmol Med (2015)

Purpose. To describe a case of 3 refractive procedures performed in one eye over 2 decades. Methods.
Case report:
Results. A 41-year-old patient presented for refractive surgery evaluation. His ocular history includes bilateral radial keratotomy performed 21 years ago for moderate myopia: spherical equivalence of -4.25 D bilaterally. Postoperative uncorrected visual acuity (UCVA) was 20/30; however, over time he developed a hyperopic shift and UCVA decreased to 20/40 in the right eye. Thus, laser-assisted in situ keratomileusis (LASIK) was performed at an outside institution 6.5 years later, and the patient had initial improvement of UCVA to 20/25. Due to a change in refractive error, the patient underwent uneventful astigmatic keratotomy 13 years after LASIK in the right eye, and 1.5 years after surgery best-corrected visual acuity was 20/25 with manifest refraction of -1.00 + 5.50 × 134°. Conclusion. We report the outcomes and natural refractive progression in a patient who underwent three corneal refractive procedures over two decades. This case underlines the difficulties of long-term management of post-RK hyperopia and astigmatism.
Case report: A variant of wall-eyed bilateral internuclear ophthalmoplegia from unilateral pons infarction
Authors: Tingting Wang, Duanhua Cao, Jingzhe Han
Journal: Front Neurosci (2022)
Keywords: case, infarction, magnetic resonance imaging, pons, wall-eyed internuclear ophthalmoplegia acute infarction
MeSH terms: Case Reports

Wall-eyed bilateral internuclear ophthalmoplegia (WEBINO) is an uncommon ocular motility disorder that encompasses the following clinical signs: bilateral adduction deficits, bilateral abducting nystagmus, convergence lost, and a large angle exotropia in primary gaze. Here we report a case of a 55-year-old man presenting with atypical WEBINO syndrome with unilateral exotropia. The coverage test was used to record the patient's alternating exotropia. The patient experienced diplopia and ophthalmoplegia and was admitted to our hospital 3 days after the onset of the double vision. Neurologic examination showed left eye exotropia and bilateral internuclear ophthalmoplegia with impaired convergence. Vertical saccades of the left eye were also limited. Consequently, an MRI scan suggested an acute infarction in the left of the pontine tegmentum. The patient was finally diagnosed with pons infarction and was treated with anticoagulation and anti-platelet aggregation therapy.
Mullerian adenosarcoma of uterus with sarcomatous overgrowth and heterologous component associated with stromal deposit in omentum: a case report and review of the literature
Authors: Anuradha Sinha, Jyoti Prakash Phukan, Sanjay Sengupta, Paulami Guha
Journal: Case Rep Med (2012)

Background. Mullerian adenosarcoma with sarcomatous overgrowth (MASO) is a very rare variant of uterine sarcomas first described by Clement et al. as early as 1974. The presence of heterologous sarcomatous components is associated with aggressive biological behavior. Case Presentation. This is a case report of a 62-year female (P(2 + 0)) presenting with postmenopausal vaginal bleeding. Her preoperative USG revealed subserosal fibroid with adherent omentum. She underwent abdominal hysterectomy with bilateral oophorectomy. Histopathological diagnosis of resected specimen was Mullerian adenosarcoma with sarcomatous overgrowth and presence of heterologous elements involving body of the uterus. The whole thickness of the myometrium was involved along with the presence of serosal nodules and omental deposits of sarcomatous component. Conclusion. MA is considered as a low-grade malignant tumor, but MASO is a high-grade tumor frequently associated with invasion and metastasis with poor treatment outcome. Because of its rarity, correct identification of these tumors and distinction from other uterine sarcomas are a challenging job and hence its morphological features merits attention.
Recovery from intracranial hemorrhage due to leptospirosis
Authors: Farhang Babamahmoodi, Abdolreza Babamhmoodi
Journal: Case Rep Med (2011)
MeSH terms: Case Reports

Intracranial hemorrhage is a rare and fatal presentation of leptospirosis. In this paper we present the case of a 51-year-old male farmer who lives in northern Iran. He came to our hospital with a severe headache. A paraclinical evaluation showed clear signs of thrombocytopenia, and a brain MRI revealed left temporoparietal hemorrhage. Our preliminary diagnosis was Leptospirosis, and after 26 days of hospital care the patient was discharged in good condition. This paper will educationally help physicians in better diagnosis and treatment of leptospirosis.
Primary colorectal adenocarcinoma metastatic to the breast: case report and review of nineteen cases
Authors: Rodney E Shackelford, Pushpa Allam-Nandyala, Marilyn M Bui, John V Kiluk, Nicole Nicosia Esposito
Journal: Case Rep Med (2011)
MeSH terms: Case Reports

Metastases to the breast from extramammary primaries are uncommon and account for 0.5-6% of all breast malignancies (Georgiannos et al., 2001, and Vizcaíno et al., 2001). Malignant melanoma, lymphoma, and lung and gastric carcinomas are the most frequently encountered nonmammary metastases to the breast in adults (Georgiannos et al., 2001, and Chaignaud et al., 1994). Primary colorectal adenocarcinoma (CRC) metastatic to the breast is extremely rare, with the medical literature having only 19 recorded cases. Typically CRC metastatic to the breast is indicative of widely disseminated disease and a poor prognosis. Here we present a case of poorly differentiated colon cancer metastatic to the breast and review the current literature on this rare event.
Identifying and Screening At-Risk Patients for Hepatitis Delta Virus: A Case Report
Authors: Bhavana Tetali, Brianna Kuperus, Nikhilesh Mazumder
Journal: Cureus (2023)
Keywords: hbv, hdv, hepatitis, hepatitis delta virus, screening, viral
MeSH terms: Case Reports

Hepatitis Delta Virus (HDV) is associated with one of the most severe forms of viral hepatitis, with rapid progression to cirrhosis and hepatocellular carcinoma. While HDV was thought to be uncommon in the United States, recent data shows that its prevalence may be significantly higher than formerly acknowledged. Early identification of HDV is critical since approved therapeutic options are only available for compensated patients. All patients with a reactive hepatitis B virus surface antigen should undergo HDV screening, especially those with additional risk factors, including migration from an endemic area, immunosuppressed patients, hemodialysis patients, gay and bisexual men, persons who inject drugs, and persons employed in healthcare or public safety professions.
Unusual Complication of Multidrug Resistant Tuberculosis
Authors: Prerna Sharma, Ravindra Nath Sahay
Journal: Case Rep Nephrol (2017)
MeSH terms: Case Reports

Introduction:
Capreomycin is a second-line drug often used for multidrug-resistant tuberculosis which can result in nephrotoxic effects similar to other aminoglycosides. We describe a case of capreomycin induced Bartter-like syndrome with hypocalcemic tetany.
Case report:
23-year-old female patient presented with carpopedal spasms and tingling sensations in hands. Patient was being treated with capreomycin for two months for tuberculosis. On further investigation, hypocalcemia, hyponatremia, hypomagnesemia, hypokalemia, and hypochloremic metabolic alkalosis were noted. Vitamin D and serum PTH levels were within normal limits. Hypercalciuria was confirmed by urine calcium/creatinine ratio. Calcium, potassium, and magnesium supplementation was given and capreomycin was discontinued. Electrolytes normalized in two days after cessation of capreomycin with no further abnormalities on repeat investigations.
Discussion:
Aminoglycosides can result in renal tubular dysfunction leading to Fanconi syndrome, Bartter syndrome, and distal tubular acidosis. Impaired mitochondrial function in the tubular cells has been hypothesized as the possible cause of these tubulopathies. Acquired Bartter-like syndrome phenotypically resembles autosomal dominant type 5 Bartter syndrome. Treatment consists of correction of electrolyte abnormalities, indomethacin, and potassium-sparing diuretics. Prompt diagnosis and treatment of severe dyselectrolytemia are warranted in patients on aminoglycoside therapy.
Giant mesenteric cyst: Successful management in low-resource setting
Authors: Mario Antunes, Damiano Pizzol, Marcella Schiavone, Anna Claudia Colangelo
Journal: Int J Surg Case Rep (2020)
Keywords: giant mesenteric cyst, low-income setting, lymphatic cyst, mesothelial cyst
MeSH terms: Case Reports

Introduction:
Mesenteric cysts are rare, generally benign intra-abdominal lesions with a wide range of presentation in terms of size, clinical presentation, etiology, radiological features, and pathological characteristics.
Presentation of case:
We reported a case of giant mesenteric cyst in a 16-month-old girl successfully managed in a low-resource setting.
Discussion:
This case is particularly important not only due to the rarity of the presented case, but also for the highlighted aspects from a public health point of view. We faced of the problem of a late stage disease and the lack of preoperative diagnosis due to cultural and economic reasons and the weaknesses of healthcare systems, as in the majority of low- and middle-income countries.
Conclusion:
Despite all these limitation, this case illustrates that complex, rare diseases can also be managed successfully in a low-resource setting. It is mandatory to strengthen and improve the health system both in terms of equipment both in terms of public health policies in order to offer a better and more effective quality of care to patients also in low-income countries.
Diagnostic challenges of tuberculous lymphadenitis using polymerase chain reaction analysis: a case study
Authors: Hirokazu Taniguchi, Masahiko Nakamura, Kazuki Shimokawa, Fumi Kamiseki, Shin Ishizawa, Hitoshi Abo, Hideaki Furuse, Takeshi Tsuda, Yasuaki Masaki, Kensuke Suzuki
Journal: Case Rep Infect Dis (2015)

This report presents a case of tuberculous lymphadenitis that was difficult to diagnose using polymerase chain reaction analysis. An 80-year-old Japanese female was hospitalized due to swollen cervical lymph nodes. Her lymph node tests revealed paradoxical polymerase chain reaction results. Polymerase chain reaction analysis of two biopsy tissues using the Cobas TaqMan revealed a positive result for Mycobacterium avium and a negative result for Mycobacterium tuberculosis. However, polymerase chain reaction analysis of a cultured colony of acid-fast bacteria from biopsy tissue using the Cobas TaqMan and an alternative polymerase chain reaction analysis of biopsy tissue yielded discordant results. The patient was diagnosed as having tuberculous lymphadenitis. She was treated with antitubercular drugs and subsequently had a reduction in cervical lymph node swelling. Polymerase chain reaction analysis is not 100% accurate; hence, its use as a diagnostic tool for mycobacterial infection requires increased attention.