OpenMed/multicare-articles huggingface dataset:
Compassionate Use of the ROCK Inhibitor Fasudil in Three Patients With Amyotrophic Lateral Sclerosis
Authors: Jan C Koch, Josua Kuttler, Fabian Maass, Teresa Lengenfeld, Eirini Zielke, Mathias Bähr, Paul Lingor
Journal: Front Neurol (2020)
Keywords: fasudil, rock inhibition, rho kinase, sod1, amyotrophic lateral sclerosis, compassionate use, therapy
MeSH terms: Case Reports

The Rho kinase (ROCK) inhibitor Fasudil is a promising drug for a disease-modifying therapy of amyotrophic lateral sclerosis (ALS). In preclinical models, Fasudil was shown to increase motor neuron survival, inhibit axonal degeneration, enhance axonal regeneration and modulate microglial function in vitro and in vivo. It prolonged survival and improved motor function of SOD1-G93A-mice. Recently, a phase IIa clinical trial has been commenced to investigate the safety, tolerability, and efficacy of Fasudil in ALS patients at an early stage of disease (ROCK-ALS trial, NCT03792490, Eudra-CT-Nr.: 2017-003676-31). Although Fasudil has been approved in Japan for many years for the treatment of vasospasms following subarachnoid hemorrhage and is known to have a favorable side effect profile in these patients, there is no data on its use in human patients with ALS or any other neurodegenerative conditions. Here, we report the first three cases of compassionate use of Fasudil in patients with ALS. Between May 2017 and February 2019, one male (66 years old) and two female (62 and 68 years old) subjects with probable or definite ALS according to the El Escorial criteria (one of the females having a pathogenic SOD1 mutation) were administered Fasudil 30 mg intravenously twice daily over 45 min on 20 consecutive working days. Blood pressure, heart rate and routine laboratory tests were constantly controlled. All three subjects tolerated the Fasudil infusions well without any obvious side effects. Interestingly, the slow vital capacity showed a significant increase in one of the patients. Taken together, we report here the first compassionate use of the ROCK inhibitor Fasudil in three ALS patients, which was well-tolerated.
A Case of Levamisole-Induced Agranulocytosis
Authors: Thamer Kassim, Lakshmi Chintalacheruvu, Osman Bhatty, Mohammad Selim, Osama Diab, Ali Nayfeh, Jayadev Manikkam Umakanthan, Maryam Gbadamosi-Akindele
Journal: Case Rep Hematol (2018)
MeSH terms: Case Reports

A sixty-eight-year-old male with a past medical history of recurrent cocaine use presented to the emergency department with recurrent diarrhea and was found to have a white blood cell (WBC) count of 1.9 × 109/L with agranulocytosis (absolute neutrophil count (ANC) of 95 cell/mm3). At admission, the patient disclosed that he used cocaine earlier during the day, and a urine drug screen tested positive for this. On hospital day one, the patient was found to have a fever with a maximum temperature of 313.6 K. After ruling out other causes and noting the quick turnaround of his neutropenia after four days of cocaine abstinence, the patient's neutropenia was attributed to levamisole-adulterated cocaine.
Hepatotoxicity Due to Azole Antimycotic Agents in a HLA B*35:02-Positive Patient
Authors: Tim Bühler, Michael Medinger, Jamal Bouitbir, Stephan Krähenbühl, Anne Leuppi-Taegtmeyer
Journal: Front Pharmacol (2019)
Keywords: hla b*35
MeSH terms: Case Reports

We will present a 42-year-old woman with acute myeloid leukemia and pulmonary aspergillosis. She was treated with several antifungal agents, including three triazoles. Voriconazole, posaconazole, and isavuconazole all led to hepatocellular liver injury. Voriconazole administration led to a peak alanine aminotransferase (ALT) value of 1,793 U/L (normal range, 9-59 U/L). After posaconazole and isavuconazole treatment, ALT rose over 500 U/L. The typical course of events, exclusion of differential diagnoses, and normalization of the liver function tests (LFTs) after stopping the triazoles were highly suspicious for a drug-induced liver injury (DILI). Interestingly, our patient carries a rare HLA B allele (HLA B*35:02), which occurs in less than 1% of the population and is known to be associated with minocycline-induced liver injury. Over the course of 4 months, the patient received two induction chemotherapies and afterward underwent a successful allogenic hematopoietic stem cell transplantation. Her liver function recovered rapidly and favorable clinical findings concerning the aspergillosis led to a de-escalation of the antifungal treatment to prophylactic dose fluconazole. Delayed hepatotoxicity suggested a dose dependency and a cumulative effect. The question of a common pathophysiology and a cross-toxicity was raised. At the present time, only a few case reports describe cross-toxicity or its absence after rechallenge with different azoles. The pathophysiology is not well understood. Ketoconazole was found to impair rat mitochondrial function in vitro. Further investigations showed cell membrane toxicity and ATP depletion in isolated human liver cancer cells. Our case report suggests a cross-toxicity, dose-dependency, and a possible genetic predisposition of triazole-induced liver injury.
Collision Tumor in the Pituitary, Concurrent Pituitary Adenoma, and Craniopharyngioma
Authors: Zaid Shareef, Connor Kerndt, Trevor Nessel, Devin Mistry, Bryan Figueroa
Journal: Case Rep Otolaryngol (2020)
MeSH terms: Case Reports

Collision tumors are two independent, distinct tumors occupying the same anatomical space. This case presents a pituitary adenoma-craniopharyngioma collision tumor presenting with hemianopsia. A 60-year-old with a past history of a nonsecretory pituitary adenoma presented with progressive headaches, bitemporal hemianopsia, and nausea. Previously, in 2008, his adenoma was effectively treated with nasal septal flap and transsphenoidal pituitary resection. A magnetic resonance imaging (MRI) was ordered for concern of recurrence, given his history and neurologic complaints. The MRI revealed a suprasellar mass extending into the third ventricle with displacement of the hypothalamus and optic chiasm. Laboratory testing revealed no indicators of endocrinopathy. The neurosurgical and otolaryngologic teams were elected to perform tumor resection given the ongoing symptoms. An image-guided transsphenoidal tumor resection with abdominal fat graft harvest and septal mucosal flap CSF leak repair was performed. Histopathological examination revealed two tumor components within the resection including an adamantinomatous craniopharyngioma and recurrent pituitary adenoma.
Relapsed ocular squamous surface neoplasia treated with topical interferon alfa-2b
Authors: Carlos Rocha-de-Lossada, Carmen Alba-Linero, Davide Borroni, Rahul Rachwani-Anil, Francisco Zamorano-Martín, Marina Rodríguez-Calvo-de-Mora
Journal: Rom J Ophthalmol (2020)
Keywords: interferon α-2β, conjunctival intraepithelial neoplasia, epidermoid carcinoma, impression cytology, limbal neoplasia, ocular squamous surface neoplasia
MeSH terms: Antineoplastic Agents / therapeutic use, Carcinoma in Situ / drug therapy, Conjunctival Neoplasms / drug therapy, Corneal Diseases / drug therapy, Interferon alpha-2 / therapeutic use, Neoplasm Recurrence, Local / drug therapy, Case Reports, Administration, Ophthalmic, Carcinoma in Situ / pathology, Conjunctival Neoplasms / pathology, Corneal Diseases / pathology, Humans, Male, Middle Aged, Neoplasm Recurrence, Local / pathology, Ophthalmic Solutions, Antineoplastic Agents, Interferon alpha-2, Ophthalmic Solutions

A 54-year-old patient presented with a large, fleshy conjunctival lesion in the right eye that had been previously surgically removed in another centre. Diagnosis of Conjunctival intraepithelial neoplasia (CIN) was confirmed by conjunctival impression cytology. The tumor was treated with topical IFN-α-2β (1,000,000 IU/ (ml) adjuvant to surgery for chemoreduction purposes. One month after treatment, the lesion showed almost complete involution, therefore the treatment was continued up to 6 months, and the lesion showed full regression. The patient is currently under periodical follow-up by conjunctival impression cytology. We consider that topical IFN-α-2β can be used in CIN for chemoreduction and/ or curative purposes. We believe that conjunctival impression cytology is a useful and non-invasive method for diagnosis and follow-up in this condition.
Case Report: Case report: Mixed infection of Plasmodium vivax and Plasmodium falciparum in a tertiary hospital
Authors: Abeer M Al-Subaie
Journal: F1000Res (2021)
Keywords: falciparum malaria, malaria, plasmodium falciparum, plasmodium vivax, case report
MeSH terms: Coinfection / drug therapy, Malaria, Vivax / diagnosis, Malaria, Vivax / drug therapy, Case Reports, Adult, Female, Humans, Plasmodium falciparum, Plasmodium vivax, Tertiary Care Centers

Mixed infections with two or more species of Plasmodium are frequently reported due to vector factors, parasite factors (formation of hypnozoites) and host factors (residing in endemic areas, travel to endemic areas, inadequately treated previous infection, lack of compliance to therapy). Here we report a case of a 33-year-old Saudi female who had a significant travel history, and a peripheral blood smear (PBS) revealed mixed infection with P. falciparum and P. vivax. The case was successfully treated with a combination therapy of artemisinin and primaquine with follow up testing at three, seven, 14, and 28 days. Mixed malaria infections are especially reported in travelers to endemic areas. Hence, adequate diagnosis and appropriate treatment of the cases contributes majorly to preventing relapse and controlling the disease. Travel consultations should be given to all travelers before their trips to endemic countries.
Acute Kidney Failure in a Young African American Male
Authors: Thuan V Nguyen, Nada M Bader, Harpreet Sidhu, Caprice Cadacio, Susana M Mendoza, Phuong Thu T Pham, Phuong Chi T Pham
Journal: Case Rep Nephrol (2019)
MeSH terms: Case Reports

Retroperitoneal fibrosis (RPF) is a condition characterized by chronic inflammatory and fibrotic changes in the retroperitoneum that can lead to serious complications including kidney failure, mesenteric and limb ischemia, and deep venous thrombosis among others. Affected individuals may present with nonspecific symptomology that would require a high clinical index of suspicion for prompt diagnosis. We herein discuss a case of a young African-American man with recurrent deep venous thrombosis who presents with a 4-week history of constant aching pain of abdomen and back and kidney failure. Initial noncontrast computed tomogram (CT) only revealed mild bilateral hydroureteronephrosis with inflammatory changes but without obvious mass or lymphadenopathy. At the insistence of the renal consulting team to rule out RPF, a CT-urogram was performed which revealed an infiltrative mass encasing the aorta, inferior vena cava, and common iliac vessels. Laparoscopic biopsy revealed dense fibroadipose tissue, lymphocytic aggregates, focal scattered IgG4-positive plasma cells, and fibrin deposition. Patient underwent bilateral nephrostomy placement and empirical corticosteroid therapy with resolution of kidney failure. Our case illustrates a classic presentation of RPF with relatively benign findings on noncontrast CT that could have been missed if clinicians did not keep a high index of suspicion for the condition.
Multimodal Diagnostic Workup of Primary Pericardial Mesothelioma: A Case Report
Authors: Jiahui Liu, Zhi Wang, Ying Yang, Yan Xiong, Wei Wang, Jianxing Qiu, Kai Zhao, Bo Zheng
Journal: Front Cardiovasc Med (2021)
Keywords: case report, computed tomography, contrast-enhanced ultrasonography, echocardiography, magnetic resonance imaging, primary pericardial mesothelioma
MeSH terms: Case Reports

Background: Primary pericardial mesothelioma is an extremely rare tumor, and early identification and accurate diagnosis may improve its clinical outcome. Case presentation: In this study, we reported a case of a 70-year-old woman who presented with dyspnea. Conventional transthoracic echocardiography showed massive pericardial effusion. Contrast-enhanced ultrasonography revealed a hyper-enhancing mass in the pericardium. Further imaging methods, including cardiac MRI and positron emission tomography/computed tomography, showed invasion of the pericardial mass into the adjacent tissues and distant metastases. Pathologic examination of a puncture biopsy specimen finally confirmed the diagnosis of PPM. Conclusion: Pericardial masses are difficult to detect when a large amount of pericardial effusion is present and the mass is small. The combination of multiple modalities plays a meaningful role in identifying PPM.
Neurocysticercosis in Oregon, 1995-2000
Authors: John M Townes, Christopher J Hoffmann, Melvin A Kohn
Journal: Emerg Infect Dis (2004)
MeSH terms: Neurocysticercosis / epidemiology, Case Reports, Research Support, Non-U.S. Gov't, Adolescent, Adult, Aged, Child, Child, Preschool, Fatal Outcome, Female, Hospital Records, Humans, Incidence, Male, Middle Aged, Neurocysticercosis / pathology, Neurocysticercosis / physiopathology, Oregon / epidemiology

The unexpected death of a teenager from neurocysticercosis prompted an investigation of this disease in Oregon. We found 89 hospitalizations, 43 newly diagnosed cases, and 6 deaths from 1995 to 2000. At least five cases occurred in persons who had not traveled or lived outside the United States. Enhanced surveillance for neurocysticercosis is warranted.
Cases of common carotid artery pseudoaneurysm treated by stent graft
Authors: Hee Ok Kim, Yong Bae Ji, Seung Hwan Lee, Cheolkyu Jung, Kyung Tae
Journal: Case Rep Otolaryngol (2012)
MeSH terms: Case Reports

Common carotid artery (CCA) pseudoaneurysms are rare and potentially lethal, and adequate treatment is warranted in order to prevent rupture or neurologic sequelae. The causes of CCA pseudoaneurysm include blunt or penetrating trauma, infection, and vasculitis, as well as iatrogenic and unknown causes. Previously, surgery was the standard treatment for pseudoaneurysm. However, endovascular surgical approaches such as stent graft or coiling have become effective alternatives with minimal morbidity and high success rates. Here, we report two cases of CCA pseudoaneurysms that were successfully treated by stent graft and review the current literature.
Clinicoradiographic Features and Histopathologic Variations of Intraosseous Lipoma: Report of a Case and Review of the Literature
Authors: Saede Atarbashi-Moghadam, Ali Lotfi, Mohammad Mehdizadeh, Fazele Atarbashi-Moghadam
Journal: Case Rep Dent (2021)
MeSH terms: Case Reports

Intraosseous lipoma is a very rare lesion, and the jaw is its least common bone location. Fibrolipoma is a microscopic variant of lipoma which is characterized by a significant fibrous component intermixed with lobules of fat cells. Intraosseous fibrolipoma of the jaw is a rare lesion, and to the best of our knowledge, only two cases have been reported from 1948 in English literature. This paper presents a 39-year-old man with a chief complaint of tooth displacement in the anterior region of the mandible. Radiographic evaluation revealed a unilocular radiolucent lesion with sclerotic borders located between the left lateral incisor and canine. Histopathologic evaluation after an excisional biopsy confirmed the diagnosis of intraosseous fibrolipoma. We also reviewed the literature on this rare lesion.
Case report: Revascularization failure in NF1-related moyamoya syndrome after selumetinib: A possible pathophysiological correlation?
Authors: Cristina Chelleri, Marcello Scala, Patrizia De Marco, Monica Traverso, Marzia Ognibene, Irene Bruno, Gianluca Piccolo, Pasquale Striano, Mariasavina Severino, Federico Zara, Maria Cristina Diana, Marco Pavanello
Journal: Front Pediatr (2023)
Keywords: vegf, brain mri, moyamoya vasculopathy, neurofibromatosis type, selumetinib
MeSH terms: Case Reports

Neurofibromatosis type 1 (NF1) is a neurocutaneous syndrome caused by pathogenic variants in the NF1 gene, encoding a multidomain inhibitor of Ras activity. Thus, NF1 is considered a RASopathy and drugs targeting the RAS/mitogen-activated protein kinase (MAPK) pathway, such as the MAP kinase (MEK) 1/2 inhibitor Selumetinib, are promising therapeutic options to treat NF1-associated tumors, especially plexiform neurofibromas and optic way gliomas. However, surgical treatment is often required for NF1-related cerebrovascular manifestations, such as moyamoya syndrome (MMS). We report a case of an 8-year-old patient receiving Selumetinib at the dose of 25 mg/m2 orally 2 times a day as a treatment for many plexiform neurofibromas. He suffered from two close strokes and brain MRI revealed a severe cerebral vasculopathy consistent with MMS, with marked stenosis of both the internal carotid arteries. A two-step surgical revascularization procedure was performed, consisting of a direct by-pass with an encephalo-mio-synangiosis (EMS) followed by encephalo-duro-arterio-synangiosis (EDAS). Surprisingly, despite the surgical technical success, follow-up MRI revealed lack of the expected revascularization. Selumetinib is a powerful therapeutic option in the treatment of severe NF1-related tumors. However, our findings suggest that this drug may interfere with cerebral neovascularization in patients with MMS requiring surgical revascularization. This is supported by the crucial role of the Vascular-Endothelial Growth Factor (VEGF), whose signaling pathway involve MAPK, as promoter of the neovascularization. Our observations suggest to adopt an imaging surveillance strategy to prevent unfavorable surgical outcome in patients with NF1-associated MMS receiving Selumetinib, and that priority should be given to surgical revascularization.
Legg-Calve-Perthes disease in an 8-year old girl with Acrodysostosis type 1 on growth hormone therapy: case report
Authors: Whei Ying Lim, Emily L Germain-Lee, Nancy S Dunbar
Journal: Int J Pediatr Endocrinol (2020)
Keywords: acrodysostosis, growth hormone, legg-calve-perthes disease, pseudohypoparathyroidism
MeSH terms: Case Reports

Background:
Acrodyostosis type 1 (ACRDYS1) is a rare skeletal dysplasia, and sometimes it can be misdiagnosed as pseudohypoparathyroidism type 1A (PHP1A), a subtype of Albright hereditary osteodystrophy (AHO), due to overlapping features. Growth hormone releasing hormone (GHRH) resistance with severe short stature is common in both ACRDYS1 and PHP1A (Emily L. Germain-Lee, et al. J Clin Endocrinol Metab, 88:4059-4069, 2003). Whereas growth hormone (GH) treatment has been studied in patients with PHP1a, the same is not true for the rarer ACRDYS1. Here in we report an adverse orthopedic outcome in a patient with ACRDYS1 with severe short stature treated with growth hormone. Our experience could have implications for the treatment of other patients with this disorder.
Case presentation:
We report a case of Legg-Calve-Perthes Disease (LCPD) in an 8-year old female with ACRDYS1 treated with GH. She initially presented with marked short stature (height Z-score - 3.46) with a low normal insulin like growth factor-1 (IGF1) level, and had biochemical evidence of thyrotropin and parathyroid hormone resistance. GH therapy was initiated at 0.35 mg/kg/week leading to increased growth velocity. After 7 months on GH, she developed right knee pain. Radiographic images revealed flattening of her right femoral head consistent with LCPD. GH was discontinued. Six weeks later, radiographs revealed further collapse of the entire femoral head. Her lesion stabilized after 8 months with conservative management and she never resumed GH. Her final adult height is 4'2″ (128 cm).
Conclusion:
Patients with ACRDYS1 on GH therapy may be at increased risk of LCPD. This has not been reported in patients with PHP1A treated with GH. Clinicians and families need to be aware of this potential complication when counseling about GH treatment.
Bilateral circumscribed posterior keratoconus: visualization by ultrasound biomicroscopy and slit-scanning topography analysis
Authors: Robert Rejdak, Katarzyna Nowomiejska, Dariusz Haszcz, Anselm G M Jünemann
Journal: J Ophthalmol (2012)
MeSH terms: Case Reports

This paper documents a rare nonprogressive developmental disorder-bilateral circumscribed posterior keratoconus-in a 60-year-old man referred for a cataract surgery. For the first time ultrasound biomicroscopy was used to visualise the local anterior bulging of the posterior corneal surface with concomitant thinning of the stroma. The amount of localized posterior depression, corneal thickness and the refractive power of both the posterior and anterior corneal curvature were measured using slit-scanning topography analysis (Orbscan).
An unusual complication of Meckel’s diverticulum: Littre’s hernia
Authors: Tariq Bouhout, Badr Serji, Ebo Usman Egyir, Benyounes El Amri, Imad Bouhout, Mehdi Soufi, Mohammed Bouziane, Tijani El Harroudi
Journal: Pan Afr Med J (2018)
MeSH terms: Hernia, Abdominal / etiology, Intestinal Obstruction / etiology, Meckel Diverticulum / complications, Case Reports, Adult, Female, Hernia, Abdominal / diagnosis, Humans, Intestinal Obstruction / diagnosis

Meckel diverticulum is the most common congenital anomaly of the gastrointestinal tract. Any hernia sacs containing Meckel’s diverticulum is called Littre’s hernia. It was described for the first time in 1700 by Alexis Littre. The diagnosis is unlikely to be made preoperatively and surgery is the treatment of choice. We report a rare case of Littre’s hernia who presented with clinical signs of intestinal obstruction.
A Female With Synchronous Multiple Primary Malignant Tumors in the Esophagogastric Junction, Duodenum and Pancreas: Case Report and Review of the Literature
Authors: Yongxing Du, Yunjie Duan, Lipeng Zhang, Zongting Gu, Xiaohao Zheng, Zongze Li, Chengfeng Wang
Journal: Front Oncol (2022)
Keywords: comprehensive treatment, literature review, multiple primary tumors, pancreas, prognostic analysis
MeSH terms: Case Reports

The incidence of multiple primary carcinomas (MPCs), which are defined as two or more malignancies detected in an individual person, is gradually increasing around the world. According to the timing of diagnosis for each constituent tumor, MPCs are classified into 2 categories: synchronous MPCs if constituent tumors emerge simultaneously or within 6 months or metachronous MPCs otherwise. In this report, we describe our recent observation and treatment of a female patient with synchronous primary esophagogastric junction adenocarcinoma, duodenal adenocarcinoma and pancreatic ductal adenocarcinoma (PDAC). To the best of our knowledge, this combination has not yet been reported in the literature. A crucial aspect is the decision regarding which tumor to treat initially and how to schedule further treatments according to individual tumor hazards. Our multidisciplinary team devised an individualized treatment regimen for this patient. The patient ultimately achieved an overall survival time of 18 months, which was much longer than the median survival time (6~11 months) of patients with locally advanced pancreatic cancer. Moreover, treating this rare combination raised a series of diagnostic, etiological and therapeutic questions, motivating us to carry out a critical review of the literature. In summary, an individualized treatment strategy with input from a dedicated multidisciplinary team and consideration of all options at different points along the disease trajectory is essential to optimize outcomes for patients with MPC.