OpenMed/multicare-articles huggingface dataset:
Pembrolizumab on pre-existing inclusion body myositis: a case report
Authors: Naohiro Uchio, Atsushi Unuma, Toshiyuki Kakumoto, Masao Osaki, Yoshitaka Zenke, Kenichi Sakuta, Akatsuki Kubota, Yoshikazu Uesaka, Tatsushi Toda, Jun Shimizu
Journal: BMC Rheumatol (2020)
Keywords: immune checkpoint, immune-related adverse events, inclusion body myositis, pembrolizumab
MeSH terms: Case Reports

Background:
Cases of exacerbation of pre-existing neuromuscular diseases induced by immune checkpoint inhibitors (ICIs) have rarely been reported because patients with autoimmune diseases have generally been excluded from ICI therapy due to the increased risk of exacerbation. We describe the first case of an elderly patient who experienced exacerbation of a previously undiagnosed sporadic inclusion body myositis (sIBM), the most common myopathy in the geriatric population, which was triggered by anti-programmed cell death-1 therapy.
Case presentation:
A 75-year-old man who was receiving pembrolizumab presented with limb weakness. Three years prior, he had noticed slowly progressive limb weakness, but he received no diagnosis. After the first infusion of pembrolizumab, his creatine kinase (CK) levels had increased. The neurological examination and muscle biopsy findings confirmed the diagnosis of sIBM and suggested exacerbation of sIBM induced by pembrolizumab. After the patient's CK levels decreased, pembrolizumab was restarted. The tumor progressed after its treatment with pembrolizumab. The patient died after 15 months of follow-up.
Conclusions:
In patients with slowly progressive limb weakness, sIBM should be explored before ICI therapy. In addition, if patients show high CK levels after ICI introduction, it is necessary to confirm whether they have sIBM in order to avoid unnecessary immunosuppressive therapies and assess whether they can tolerate ICI reintroduction.
The adverse impact of excessive internet use during the COVID-19 pandemic on adolescents' coping skills: A case study in Hanoi, Vietnam 2021
Authors: Quyen Thi Tu Bui, Chi Thi Lan Pham, Anh Ha Le, Duy Quang Pham
Journal: Front Public Health (2022)
Keywords: covid-19, adolescent, children, mental health, pandemic effects
MeSH terms: COVID-19 / epidemiology, Adaptation, Psychological, Adolescent, Humans, Internet Use, Pandemics, Vietnam / epidemiology

Introduction:
The COVID-19 pandemic has created significant stressors in Vietnamese adolescents' lives. Coping skills play important roles in helping adolescents contend with stress. This study aimed to evaluate adolescents' coping skills during the COVID-19 pandemic and examine how those skills are impacted by excessive internet use during this pandemic.
Methods:
The study used respondent-driven sampling and Google online survey forms to collect data. The study sample included 5,315 high school students aged 11- 17 years in Hanoi's rural and urban areas. The Kid Coping Scale was applied to examine adolescents' coping, and the coping score was compared among adolescents with different levels of internet use.
Results:
The average coping score measured by Kid Coping Scale was 20.40 (std = 2.13). About half of adolescents often "avoid the problem or the area where it happened" when experiencing a hard time. One-third of adolescents often stopped thinking about the problem they faced. More than one-fourth of respondents stayed online for at least 8 h per day. The online time for learning/other activities showed a reverse dose-response relationship with the coping score; the longer the internet use duration, the lower the coping score.
Conclusion:
The mean score of coping of Hanoi adolescents was moderate. Internet use has an adverse impact on their coping skills.
A Rare Presentation of Isolated CNS Posttransplantation Lymphoproliferative Disorder
Authors: Jaime Morris, Casey Smith, Andrew Streicher, Allison Magnuson, Susan Newman, Robert Bertoli
Journal: Case Rep Oncol Med (2017)
MeSH terms: Case Reports

Posttransplantation lymphoproliferative disorder (PTLD) is a recognized and extremely morbid complication of solid organ transplantation, but central nervous system involvement, particularly in isolation, is rare. There are no standardized treatment strategies for PTLD, though commonly used strategies include reduction of immunosuppression, chemotherapy, rituximab, radiation, and surgery. We present a case of an unusual morphologic variant of primary central nervous system PTLD with successful response to rituximab and cranial radiation. A 69-year-old Asian male, who underwent postrenal transplant nine years earlier, presented with a one-month history of new onset seizure activity. His evaluation revealed multiple brain lesions on magnetic resonance imaging (MRI), as well as serologic and cerebrospinal fluid studies which were positive for Epstein-Barr Virus (EBV) infection. Ultimately, he underwent craniotomy with tissue biopsy with the final pathology report showing posttransplant lymphoproliferative disorder, polymorphic type. The patient was managed with reduction in immunosuppression, rituximab therapy, and cranial radiation treatments. He had demonstrated marked improvement in his neurologic function and was ultimately discharged to inpatient rehabilitation facility.
Fatal myocardial fibrosis in an aged chimpanzee (Pan troglodytes)
Authors: Audrey Baldessari, Jessica Snyder, Joel Ahrens, Robert Murnane
Journal: Pathobiol Aging Age Relat Dis (2013)
Keywords: great apes, interstitial myocardial fibrosis, sudden death
MeSH terms: Case Reports

A 36-year-old male chimpanzee (Pan troglodytes) assigned to a life-long sign language communication project presented for sudden death. No other clinical or clinical pathological abnormalities were noted and given the signalment, death due to cardiac failure was suspected. Necropsy findings revealed moderate cardiomegaly and other chronic age-related findings including focal renal tubular cystic dilation and gingival hyperplasia. Histologic evaluation of the heart revealed interstitial fibrosing cardiomyopathy characterized by severe interstitial myocardial fibrosis replacing and separating myofibers within all chambers of the heart, especially the left ventricle, interventricular septum and subvalvular areas. This case report represents an additional case of sudden death associated with interstitial myocardial fibrosis in a chimpanzee. This process has been previously cited as the most common cause of sudden death in aged chimpanzees.
West Syndrome in an Infant With Vitamin B12 Deficiency Born to Autoantibodies Positive Mother
Authors: Pin Fee Chong, Masaru Matsukura, Kaoru Fukui, Yoriko Watanabe, Naomichi Matsumoto, Ryutaro Kira
Journal: Front Pediatr (2019)
Keywords: anti-intrinsic factor antibody, anti-parietal cell antibody, cobalamin, infantile spasm, west syndrome
MeSH terms: Case Reports

Infantile vitamin B12 deficiency, a rare nutritional disorder in developed countries, is characterized by megaloblastic anemia and non-specific symptoms, including failure to thrive, hypotonia, and seizure. Symptoms usually develop at 6 months of age. Exclusively breast-fed infants of vegan-diet mothers are particularly at risk. We report the case of a 7-month-old boy with West syndrome born to a mother with subclinical vitamin B12 deficiency due to autoantibodies. Electroencephalography revealed the characteristic hypsarrhythmia pattern, whereas cranial magnetic resonance imaging revealed cerebral atrophy and hypomyelination. Biochemical analysis revealed elevated urinary methylmalonic acid and homocysteine and reduced plasma methionine. Serum vitamin B12 levels were extremely low in both the child and his mother. The mother tested positive for both anti-intrinsic factor and anti-parietal cell antibodies. Low-dose adrenocorticotropic hormone was effective for seizure control. Contrary to previous reports, age-appropriate neurodevelopment was not achieved despite rapid normalization of metabolic profile with vitamin B12 supplementation. Further investigations failed to detect any causative mutations in the genes associated with developmental and epileptic encephalopathy as well as metabolic and other identifiable disorders known to cause West syndrome. To the best of our knowledge, this is the first reported case in which maternal anti-intrinsic factor antibody was considered to be the reason for infantile vitamin B12 deficiency with West syndrome. Differential diagnosis of West syndrome should include vitamin B12 deficiency due to its treatable nature, and early diagnosis is essential to prevent permanent neurological consequences.
Laparoscopic management of cholecystocolonic fistula: A case report and a brief literature review
Authors: G Bonventre, G Di Buono, S Buscemi, G Romano, A Agrusa
Journal: Int J Surg Case Rep (2020)
Keywords: cholecystocolonic fistula, complicated cholelithiasis, laparoscopy
MeSH terms: Case Reports

Introduction:
Cholecystoenteric fistula is a rare and late complication of cholelithiasis. The clinical presentation is mostly chronic and is not distinguishable from the dyspeptic symptoms of non-complicated cholelithiasis. For this reason, the preoperative diagnosis is difficult and uncertain, and it is often made up primarily intraoperatively and incidentally during cholecystectomy. In this article, we report a case of cholecystocolonic fistula management by laparoscopic approach.
Presentation of case:
We studied a 64 years old male patient with fever of an unknown origin for two months and abdominal pain. He underwent a contrast enhanced CT abdominal scan that showed a sclerotic gallbladder with a disorganized fluid collection. The colonoscopy identified a cholecystocolonic fistula with hepatic flexure. A laparoscopic cholecystectomy was performed to repair the colonic wall with intra-corporeal sutures.
Discussion:
Thanks to the advancements in CT scan's resolution and the application of endoscopic technology such as ERCP or colonscopy, preoperative diagnosis of cholecystoenteric fistula has been greatly improved. In addition, cholecystoenteric fistula has been successfully managed laparoscopically with laparoscopic cholecystectomy and closure of the fistula tract.
Conclusion:
The laparoscopic management of cholecystoeneteric fistula is a feasible and safe procedure but the operative strategy should be individualized on diagnosis, patient characteristics, availability of resources and experience of surgical team.
A Case of Status-Epilepticus-Associated Transient Hyperammonemia in the Emergency Department
Authors: Brittany Pelsue, Jonathan G Rogg
Journal: Case Rep Emerg Med (2017)
MeSH terms: Case Reports

This report describes a case of transient hyperammonemia following tonic-clonic status epilepticus with an initial ammonia level of 537 Umol/L. This appears to be the highest transient ammonia level reported in the literature in this clinical scenario. This is an affirmation that an initial elevated ammonia level in the absence of hepatic dysfunction should be interpreted with caution when associated with status epilepticus. Repeat levels should be drawn to identify transient hyperammonemia and determine the need for treatment if levels do not decrease.
Intestinal Adenocarcinoma Arising from a Mature Cystic Teratoma
Authors: King Man Wan, Forough Foroughi, Rajni Bansal, Martin K Oehler
Journal: Case Rep Pathol (2019)
MeSH terms: Case Reports

Mature cystic teratomas are the most common ovarian germ cell tumour and account for 10-20% of all ovarian neoplasms. Malignant transformation of mature cystic teratomas is rare and has an incidence rate of less than 1%. The most common malignancy are squamous cell carcinomas. Here we present the case of an intestinal adenocarcinoma which is an exceedingly rare malignant entity arising within a mature cystic teratoma. Clinical presentation, imaging and histopathological diagnosis are discussed and previously presented cases in the literature reviewed.
Skull metastasis as initial manifestation of pulmonary epithelial-myoepithelial carcinoma: a case report of an unusual case
Authors: Masamitsu Nishihara, Naoya Takeda, Shoutarou Tatsumi, Keiji Kidoguchi, Shigeto Hayashi, Takashi Sasayama, Eiji Kohmura, Kimio Hashimoto
Journal: Case Rep Oncol Med (2011)
MeSH terms: Case Reports

Epithelial-myoepithelial carcinoma (EMC) of the lung is rare and is considered to be low-grade malignancy. Intracranial metastasis of pulmonary EMC has not previously been reported according to our search of the literature. We report a case of skull metastasis as the initial manifestation of pulmonary EMC. An 81-year-old man complained of left leg motor weakness. Neurological examination showed left hemiparesis. Computed tomography and magnetic resonance imaging revealed an osteolytic tumor in the right frontal bone with invasion to the dura and subdural space, attached to the superior sagittal sinus. Subtotal removal of the tumor was performed, and the left hemiparesis showed improvement. Histopathological study revealed the tumor to consist of epithelial and myoepithelial cells. Pulmonary EMC was diagnosed. The MIB-1 index in primary lesion was approximately 10%. The skull and dura are possible sites for metastasis from pulmonary EMC. The MIB-1 index is a predictive marker of malignant potential.
Familial Mediterranean Fever with Neonatal Onset: Case Report
Authors: Kübra Arslan, Serdar Ümit Sarici, Gonca Kolukisa, Demet Altun
Journal: Case Rep Pediatr (2020)
MeSH terms: Case Reports

Familial Mediterranean fever (FMF) is an autosomal recessively inherited disorder characterized by recurrent fever and attacks of abdominal pain, chest pain, and joint pain. Attacks of recurrent fever and serositis are encountered clinically. Attacks may present either with only one symptom or many simultaneous symptoms. Although most of the patients are diagnosed clinically above the age of 2, those cases who are diagnosed before 2 years of age and with clinical course of isolated fever are believed to have a more serious course and tend to develop amyloidosis. In this article, a case who was admitted first on the 22nd day of life and later diagnosed to have FMF with recurrent attacks of isolated fever and no other focus is presented. We emphasize that FMF may present as early as in the first month of life, and it should be considered in cases presenting with fever of unknown origin and misdiagnosed to have late neonatal sepsis or occult bacteremia at this age group.
Acute dapsone poisoning with methemoglobinemia: a case report
Authors: Hamza El Hamzaoui, Idriss Chajai, Mohamed Chahidi El Ouazzani, Abdelkader Benhalima, Manal El Arfaoui, Mustapha Alilou
Journal: Pan Afr Med J (2022)
Keywords: poisoning, case report, dapsone, methemoglobinemia
MeSH terms: Methemoglobinemia / chemically induced, Methemoglobinemia / diagnosis, Methemoglobinemia / drug therapy, Case Reports, Adolescent, Ascorbic Acid, Charcoal / therapeutic use, Dapsone, Female, Humans, Tachypnea, Dapsone, Charcoal, Ascorbic Acid

Methemoglobinemia is a common complication of dapsone poisoning. Its´ treatment usually relies on methylene blue infusion. The aim of this study was to report a case of an acute dapsone poisoning with methemoglobinemia treated only with ascorbic acid and activated charcoal. A 16-year-old female voluntary ingested 3 grams of dapsone in an attempt of suicide and presented with desaturation and tachypnea. Lab findings were compatible with methemoglobinemia. After two days of treatment with ascorbic acid and activated charcoal, we observed the disappearance of desaturation and tachypnea. Methemoglobinemia can be treated with ascorbic acid and activated charcoal in limited resource settings.
Gastric Plexiform Fibromyxoma Arising in the Cardia in an Adolescent Male: A Rare Tumor with an Unusual Location
Authors: Awrad Nasralla, Mufeed Alwabari, Osama Alsaif, Samir S Amr
Journal: Case Rep Surg (2020)
MeSH terms: Case Reports

Plexiform fibromyxoma of the stomach, also known as plexiform angiomyxoid myofibroblastic tumor, is a rare benign gastric mesenchymal tumor, first described in 2007, which usually arises in the gastric antrum and affects adults. Few cases have been reported in children and adolescents. It can present with different clinical manifestations including abdominal pain, dyspepsia, hematemesis, and vomiting. Preoperatively, this tumor is usually diagnosed as gastrointestinal stromal tumor (GIST), and the correct diagnosis is made only after histopathological examination following surgical resection. Most cases were reported from East Asia (China, Japan, and Korea), North America, and Europe. We report herein a unique case of plexiform fibromyxoma, the first to be reported from the Middle East, arising in the cardia of the stomach in a 16-year-old adolescent male, with a brief review of the literature.
TMJ Ankylosis in Children: A Case Report and Literature Review
Authors: Abdouldaim Ukwas, Mahmoud Elshik, Mohammed Elbialy
Journal: Case Rep Dent (2023)
MeSH terms: Case Reports

Temporomandibular joint (TMJ) ankylosis is a serious disabling condition characterized by fusion of the mandibular condyle with the glenoid fossa, disc, and/or eminence, resulting in severely restricted mouth opening and significantly reduced mandibular movements. The condition often has a deteriorating effect on the patient's daily functions such as speech, chewing, breathing, and oral hygiene as well as their wellbeing and quality of life. Furthermore, childhood TMJ ankylosis frequently has a detrimental impact on the facial growth resulting in facial asymmetry, micrognathia, and/or class II malocclusion with posterior or anterior open bite. Trauma is the main cause of TMJ ankylosis, but the condition can also occur as a result of surgery, local or systemic infections, or systemic disease. Surgery is the mainstay of treatment, and several approaches have been applied, including gap arthroplasty (GA), interpositional gap arthroplasty (IGA), reconstruction arthroplasty (RA), or distraction osteogenesis (DO). The aim of this article is to present a post-traumatic TMJ ankylosis case in a 5-year-old male child who was treated with resection and simultaneous costochondral grafting and to provide a succinct update of literature.
Ultrasound-Guided Morcellation During Holmium Laser Enucleation of the Prostate
Authors: David T Tzou, Ian S Metzler, Marshall L Stoller, Thomas Chi
Journal: J Endourol Case Rep (2018)
Keywords: bph, holep, morcellation, ultrasound
MeSH terms: Case Reports

Background: Holmium laser enucleation of the prostate (HoLEP) has emerged as an accepted standard of care for the treatment of benign prostatic hyperplasia. This surgery relies on morcellation of the prostate adenoma once enucleation of the transition zone has been completed. Caution is required during this portion of the operation, as engaging bladder mucosa within the morcellator can result in bladder injury, a rare but potentially catastrophic complication of HoLEP. Morcellation of the prostatic tissue can be additionally challenging if visualization is poor from either equipment failure or increased bleeding from a highly vascularized prostate. Case Presentation: We report the case of a 66-year-old Caucasian man with an estimated 158 g prostate who underwent HoLEP at our institution. Enucleation was uneventful; however, upon placement of the nephroscope to begin morcellation, it was immediately evident that the lens of the nephroscope was damaged as there was extremely poor visualization. Without a replacement nephroscope available, this would have normally resulted in aborting the case and returning another day to complete the morcellation. Concurrent bladder ultrasonography was performed and allowed for additional visual feedback to the operator, helping guide the morcellator to safely engage the enucleated adenoma and complete the operation. Conclusion: This case report demonstrates the ability of performing the morcellation portion of HoLEP mainly with the visualization provided by concurrent bladder ultrasonography. By providing additional imaging feedback to the operator, ultrasound can be a complementary tool to assist in safely performing morcellation in situations of suboptimal cystoscopic visualization during HoLEP.
A refractory liver metastatic solid pseudopapillary neoplasm pancreas harbored CTNNB1 mutation showed good response to celecoxib: A case report
Authors: Yu Shang, Yunkun Zhang, Evenki Pan, Peng Yang, Lingling Xu, Jinghua Sun
Journal: Front Oncol (2022)
Keywords: ctnnb1 mutation, solid pseudopapillary neoplasm (spn), celecoxib, liver metastasis, pancreas
MeSH terms: Case Reports

Solid pseudopapillary neoplasm (SPN) of the pancreas is rare relatively low-grade malignant neoplasm and metastasis rarely. Surgical resection is the primary treatment option for primary and metastatic lesions of SPN, and chemotherapy is often ineffective in non-operable SPNs. SPNs are characterized by the presence of somatic CTNNB1 exon 3 mutations, leading to the activation of Wnt/β-catenin/Cox-2 signal pathway. Here, we firstly report that a refractory liver metastatic pancreatic SPN patient after the failure of multi-line chemotherapies benefited from the Cox-2 selective inhibitor (Celecoxib) based on CTNNB1 D32V mutation detected by next-generation sequencing (NGS), achieving a more than 22-month progression-free survival without any adverse events. Our case provides a potential treatment option for liver metastatic SPN patients with CTNNB1 mutations and highlights the application of NGS for the better treatment decision making.
Case Report: Splenic Irradiation for the Treatment of Chronic Active Antibody-Mediated Rejection in Kidney Allograft Recipients With De Novo Donor-Specific Antibodies
Authors: Lan Zhu, Zhiliang Guo, Rula Sa, Hui Guo, Junhua Li, Gang Chen
Journal: Front Immunol (2021)
Keywords: case report, chronic active antibody-mediated rejection (cabmr), de novo donor-specific antibody, renal transplantation, splenic irradiation
MeSH terms: Antibodies / immunology, Graft Rejection / immunology, Kidney Transplantation / methods, Spleen / immunology, Tissue Donors, Case Reports, Research Support, Non-U.S. Gov't, Adult, Allografts / immunology, Graft Rejection / prevention & control, Humans, Immunoglobulins, Intravenous / therapeutic use, Immunologic Factors / therapeutic use, Kidney / drug effects, Kidney / immunology, Male, Rituximab / therapeutic use, Spleen / radiation effects, Transplant Recipients, Antibodies, Immunoglobulins, Intravenous, Immunologic Factors, Rituximab

Chronic active antibody-mediated rejection (AMR) in renal transplantation is usually refractory to current conventional treatment with rituximab, plasmapheresis (PP), and intravenous immunoglobulins (IVIG). Splenic irradiation has been reported to be effective in the rescue of early severe acute AMR after kidney transplantation; however, its effect in chronic active AMR has not been reported to date. In order to reduce donor-specific antibody (DSA) and prevent the progression of chronic AMR, we used repetitive low-dose splenic irradiation, together with rituximab and PP/IVIG, in two living-related kidney transplant recipients with pathologically diagnosed chronic active AMR and the presence of long-term class II-de novo DSA. DSA monitoring and repeated renal biopsy revealed significantly reduced DSA levels as well as alleviated glomerulitis and peritubular capillaritis in both patients after treatment, and these therapies may have played a role in delaying the progression of chronic AMR. Although DSA levels in both patients eventually rebounded to some extent after treatment, serum creatinine increased slowly in one patient during the 16-month follow-up period and remained stable in the other during the 12-month follow-up period. Given the poor efficacy of conventional treatment at present, splenic irradiation may still be one of the treatment options for chronic active AMR.