OpenMed/multicare-articles huggingface dataset:
Temporary Parenteral Tacrolimus Requirement due to Unexplained Low Through Levels in a Liver Transplant Patient with Short Bowel Syndrome
Authors: V Ince, F Ozdemir, B Isik, M Yilmaz, C Kayaalp, S Yilmaz
Journal: Int J Organ Transplant Med (2014)
Keywords: liver transplantation, living donor, short bowel syndrome, tacrolimus, immunosuppressive agent
MeSH terms: Case Reports

An adequate level of tacrolimus in serum should be obtained to prevent acute rejection following liver transplantation. Because of good gastrointestinal absorption of oral tacrolimus, adequate trough levels can be achieved even in patients with short bowel syndrome. Rarely, adequate through levels cannot be obtained by oral administration of the drug for several reasons such as inadequate absorption, having a discordant patient, laboratory error, and/or interactions with other drugs and foods. Here, we described a 16-year-old patient who had undergone massive intestinal resection due to mesenteric torsion 5 years previously and required liver transplantation for cryptogenic cirrhosis. Her remnant small bowel length was 90 cm. After a successful living donor liver transplantation, oral tacrolimus administration resulted in inadequate through levels in some parts of the postoperative period. We checked up all the potential reasons but could not identify any cause. An intravenous tacrolimus including immunosuppressive regimen was temporarily required. She maintained adequate blood levels of tacrolimus by parenteral route for a while; thereafter, oral administration resulted in enough blood drug levels. She was discharged with oral tacrolimus therapy. We concluded that very rarely, adequate blood levels of tacrolimus cannot be achieved by oral administration for unexplained reasons. In such cases, temporary administration of parenteral tacrolimus can be life-saving.
Hemodynamics of Prefrontal Cortex in Ornithine Transcarbamylase Deficiency: A Twin Case Study
Authors: Afrouz A Anderson, Andrea Gropman, Cynthia Le Mons, Constantine A Stratakis, Amir H Gandjbakhche
Journal: Front Neurol (2020)
Keywords: ornithine transcarbamylase deficiency, cerebral autoregulation, fnirs, functional connectivity, functional near infrared spectroscopy, urea cycle disorders, wavelet coherence

Ornithine transcarbamylase deficiency (OTCD) is the most common form of urea cycle disorder characterized by the presence of hyperammonemia (HA). In patients with OTCD, HA is known to cause impairments in domains of executive function and working memory. Monitoring OTCD progression and investigating neurocognitive biomarkers can, therefore, become critical in understanding the underlying brain function in a population with OTCD. We used functional near infrared spectroscopy (fNIRS) to examine the hemodynamics of prefrontal cortex (PFC) in a fraternal twin with and without OTCD. fNIRS is a non-invasive and wearable optical technology that can be used to assess cortical hemodynamics in a realistic clinical setting. We quantified the hemodynamic variations in total-hemoglobin as assessed by fNIRS while subjects performed the N-back working memory (WM) task. Our preliminary results showed that the sibling with OTCD had higher variation in a very low frequency band (<0.03 Hz, related to mechanism of cerebral autoregulation) compared to the control sibling. The difference between these variations was not as prominent in the higher frequency band, indicating the possible role of impaired autoregulation and cognitive function due to presence of HA. We further examined the functional connectivity in PFC, where the OTCD sibling showed lower interhemispheric functional connectivity as the task load increased. Our pilot results are the first to show the utility of fNIRS in monitoring OTCD cortical hemodynamics, indicating the possibility of inefficient neurocognitive function. This study provides a novel insight into the monitoring of OTCD focusing on the contribution of physiological process and neurocognitive function in this population.
A Custom-Made Surgical Guide for Accurate Enucleation of Nasopalatine Duct Cysts: A Technical Note and Case Report
Authors: Masako Fujioka-Kobayashi, Naoki Miyasaka, Ayako Miyasaka, Masateru Koyanagi, Ryo Inada, Takahiro Miyasaka, Takafumi Satomi
Journal: Case Rep Dent (2023)
MeSH terms: Case Reports

Nasopalatine cysts are common nonodontogenic cysts that occur in the maxilla. During the nucleation of large cysts extending to the floor of the nasal cavity, care must be taken to avoid damage to the nasal mucosa. In the present report, an innovative custom-made surgical guide made by a Three-dimensional printer is introduced for accurate enucleation surgery. The patient's cone-beam computerized tomography and dental model scan data were obtained, and a tooth-supported type of surgical guide was designed containing a circular plate structure showing the size of the cystic region, an indicator that showed the position of the bottom of the cyst, and a sliding stopper that was used to accurately indicate the position of the deepest cyst wall. The surgical tool enabled us to indicate the accurate size, location of the cysts, and approach direction. Although effective and accurate navigation systems have become increasingly available, the cost-effective and accurate computer-aided design/computer-aided manufacturing surgical guide system introduced in the present report could support the safe enucleation of large nasopalatine duct cysts.
Extraskeletal Myxoid Chondrosarcoma: Long-Term Survival in the Setting of Metastatic Disease
Authors: Karim Masrouha, Iqbal Multani, Om Bhatt, Michelle Ghert
Journal: Case Rep Orthop (2020)
MeSH terms: Case Reports

Three cases of extraskeletal myxoid chondrosarcoma (EMC) in patients who presented with pulmonary metastases and were managed with long-term close observation without systemic intervention are presented. Follow-up imaging showed slow progression of their disease over several years, and the patients remained asymptomatic from their pulmonary metastases. This clinical experience provides insight into the natural history of the disease and suggests that some patients may experience long-term survival and remain asymptomatic even without systemic intervention, thereby improving their quality of life by avoiding potentially debilitating treatments.
The BEHAVE application as a tool to monitor inclusive interventions for subjects with neurodevelopmental disorders
Authors: Gianluca Merlo, Antonella Chifari, Giuseppe Chiazzese, Paola Denaro, Noemi Firrera, Nicola Lo Savio, Simona Patti, Luisa Palmegiano, Davide Taibi, Luciano Seta
Journal: Front Psychol (2023)
Keywords: applied behavior analysis, communication impairments, inclusive intervention, neurodevelopmental disorders, technologies for monitoring

In the last few years, many educational and therapeutic interventions for young people with neurodevelopmental disorders are based on systematic monitoring of the outcomes. These interventions are typically conducted using single-case experimental designs, (SCEDs) a set of methods aimed at testing the effect of an intervention on a single subject or a small number of subjects. In SCEDs, an effective process of decision-making needs accurate, precise, and reliable data but also that caregivers and health professionals can gather information with minimal effort. The use of Information Communication Technologies in SCEDs can support the process of data collection and analysis, facilitating the collection of accurate and reliable data, providing reports accessible also by non-experts, and promoting interactions and sharing among clinicians, educators, and caregivers. The present paper introduces the BEHAVE application, a web-based highly customizable application, designed to implement SCEDs, supporting both data collection and automatic analysis of the datasets. Moreover, the paper will describe two case studies of kindergarten children with neurodevelopmental disorders, highlighting how the BEHAVE application supported the entire process, from data collection in multiple contexts to decision-making based on the analysis provided by the system. In particular, the paper describes the case studies of Carlo and Dario, two children with severe language and communication impairments, and the inclusive education interventions carried out to maximize their participation in a typical home and school setting increasing their mand repertoire. Results revealed an increase in the mand repertoire in both children who become able to generalize the outcomes to multiple life contexts. The active participation of the caregivers played a crucial role in the ability of children to use the learned skills in settings different from the ones they were learned in.
Case Report: Imaging immune checkpoint inhibitor-induced yin-yang effects in the brain
Authors: K F Bol, E Peeters, C M L van Herpen, H Westdorp, E H J G Aarntzen
Journal: Front Immunol (2023)
Keywords: cd8+ t-cells, pet/ct imaging, immune checkpoint inhibitors, melanoma, toxicity
MeSH terms: Immune Checkpoint Inhibitors / adverse effects, Melanoma, Case Reports, Adult, Brain / diagnostic imaging, Humans, Positron Emission Tomography Computed Tomography / methods, Radioisotopes, Yin-Yang, Zirconium, Immune Checkpoint Inhibitors, Zirconium-89, Radioisotopes, Zirconium

Background:
Treatment with immune checkpoint inhibitors (ICI) can induce durable responses in cancer patients, but it is commonly associated with serious immune-related side effects. Both effects are suggested to be mediated by CD8+ T-cell infiltration. Whole body CD8+ T-cell distribution can be visualized by PET imaging of a 89Zr-labeled anti-humanCD8a minibody, currently investigated in a phase 2b trial.
Main body:
An adult patient diagnosed with metastatic melanoma developed ICI-related hypophysitis after two courses of combined immunotherapy (ipilimumab (3 mg/kg) and nivolumab (1 mg/kg) at 3 weeks interval). On a [89Zr]Zr-crefmirlimab berdoxam PET/CT scan, made 8 days before clinical symptoms occurred, increased CD8+ T-cell infiltration in the pituitary gland was detected. Simultaneously, tracer uptake in a cerebral metastasis was increased, indicating ICI-induced tumor infiltration by CD8+ T-cells.
Conclusions:
The observations in this case report underscore the role of CD8+ T-cell in non-tumor tissues in ICI-related toxicity. In addition, it illustrates a potential role for molecular imaging by PET/CT for investigation and monitoring of ICI-induced effects.
Case Report: Efficacy of Pyrotinib in ERBB2 Amplification Pulmonary Adenoid Cystic Carcinoma
Authors: Zhongben Tang, Feng Lin, Jiarong Xiao, Xiaojun Du, Jian Zhang, Sini Li, Gongshun Tang, Chen Chen, Jian Li
Journal: Front Oncol (2021)
Keywords: erbb2, efficacy, pulmonary adenoid cystic carcinoma, pyrotinib, stable disease, targeted therapy
MeSH terms: Case Reports

Primary pulmonary adenoid cystic carcinomas are salivary tumors that are low-grade malignant and prone to recurrence and metastasis. Surgery is currently the main treatment, but there is no standard with regard to postoperative adjuvant therapy. Adenoid cystic carcinoma is more sensitive to radiotherapy and patients benefit less from chemotherapy, but few studies have focused on targeted therapy, and their conclusions are inconsistent. With respect to primary pulmonary adenoid cystic carcinoma, large-scale studies cannot be conducted due to its low incidence, and studies on the targeted therapy of it are very scarce. A few case reports indicate that targeted therapy can be effective however, suggesting that it may be a good option. The current report is the first on the occurrence of human epidermal growth factor receptor 2 amplification in pulmonary adenoid cystic carcinoma. The patient was treated with pyrotinib for 6 months and achieved stable disease.
Candida Empyema as a Red Flag for Esophageal Rupture: A Case Report and Literature Review
Authors: Naureen Ali, Getahun Abate
Journal: Case Rep Infect Dis (2020)
MeSH terms: Case Reports

Background. Candida empyema is a rare entity with an extremely high mortality rate.We present a case of multi species Candida empyema in an immunocompetent female patient with Boerhaave syndrome secondary to retching and vomiting after heroin withdrawal. This case highlights an unusual presentation of a common infection but stresses on the fact that a high index of suspicion is necessary for early identification, prompt initiation of antifungal therapy, and drainage with surgical repair to improve overall survival and outcomes.
Serous endometrial intraepithelial carcinoma: a case report
Authors: Younesse Najioui, Nassia Karich, Anas Haloui, Achraf Miry, Amal Bennani
Journal: Pan Afr Med J (2023)
Keywords: serous endometrial intraepithelial carcinoma, case report, endometrium, serous carcinoma
MeSH terms: Carcinoma in Situ / diagnosis, Carcinoma in Situ / therapy, Cystadenocarcinoma, Serous / diagnosis, Cystadenocarcinoma, Serous / etiology, Cystadenocarcinoma, Serous / therapy, Endometrial Neoplasms / diagnosis, Endometrial Neoplasms / therapy, Uterine Neoplasms / diagnosis, Case Reports, Female, Humans, Middle Aged, Uterus / pathology

Serous endometrial intraepithelial carcinoma (SEIC) is a rare but highly aggressive form of uterine endometrial cancer. We report two cases of post-menopausal, 58-year-old patients with abundant vaginal bleeding and pelvic pain. The first patient had a history of surgical hysteroscopy in 2019 for an endocervical polyp. The second patient had a history of breast resection, axillary lymph node dissection, chemotherapy, radiation therapy, and tamoxifen therapy for breast carcinoma 6 years ago. An abdominal hysterectomy was performed in both patients. The pathological assessment showed serous endometrial intraepithelial carcinoma. Diagnosis of a serous proliferation of the uterus implies the exploration of other genital tract organs as well as distant locations in search of metastatic disease.
Pleomorphic Adenoma Presenting as an Atypical Nasal Mass in a 26-Year-Old Female
Authors: Racheal Hapunda, Chibamba Mumba, Owen Ngalamika
Journal: Case Rep Surg (2020)
MeSH terms: Case Reports

Pleomorphic adenoma (PA) is a salivary gland tumor that may rarely occur in the nasal cavity. It can be a clinical diagnostic dilemma in many instances due to many possible differential diagnoses. We report the case of a 26-year-old female who presented with a 3-year history of a right nasal growth associated with ipsilateral nasal blockage, nasal pain, and rhinorrhea. Radiological image showed a mild enhancing lesion in the right nasal cavity. The patient underwent a lateral rhinotomy with wide excision of the mass. Histopathological exam was consistent with PA. Nasal PA is a rare entity and should be suspected as a diagnosis for intranasal tumors.
Case Report: Long-Term Outcome Following Maximal Partial Resection in a Primary Pericardial Lymphangiohemangioma
Authors: Long Song, Chukwuemeka Daniel Iroegbu, Chengming Fan
Journal: Front Cardiovasc Med (2021)
Keywords: case report, lymphangiohemangioma, management, rare tumor, surgery
MeSH terms: Case Reports

Introduction: Cardiac tumors are significantly rare, with pericardial lymphangiohemangioma amongst the rarest cardiac tumor types, with very few reported cases in the literature. Clinically, lymphangiohemangiomas are generally deemed unresectable due to their proximity to the myocardium and the uncertainty of the outcomes following subtotal resection. Case Report: Herein, we report a case of a 40-year-old man diagnosed with a pericardial mass, dull thoracodorsal pain, and over a 10-years history of palpitation. Notably, the pericardial mass in the present case was found extended within the myocardium. Thus, a maximal safe resection was deemed preferable to a total resection. The surgically resected specimen showed pathological characteristics of a lymphangiohemangioma. After surgical resection of the lymphangiohemangioma, the patient was free of any tumor-related symptoms. Also, there was no evident tumor progression after a 4-year post-operative follow-up. Conclusion: To the best of our knowledge, the present case study is the first in the literature to report on a long-term post-operative outcome following subtotal resection of a pericardial lymphangiohemangioma.
A Worm's Tale or Why to Avoid the Raccoon Latrine: A Case of Baylisascaris procyonis Meningoencephalitis
Authors: Adam E Goldman-Yassen, Anna Derman, Rebecca Pellett Madan, Alireza Radmanesh
Journal: Case Rep Radiol (2022)
MeSH terms: Case Reports

The raccoon roundworm Baylisascaris procyonis (B. procyonis) may infect humans to cause severe or fatal meningoencephalitis, as well as ocular and visceral larva migrans. Young children are at greater risk for cerebral larva migrans with severe meningoencephalitis, and early empiric therapy may improve outcomes. Familiarity with characteristic brain imaging findings may prompt earlier diagnosis, particularly in the setting of CSF eosinophilia. We report a case of a 19-month-old boy who presented with truncal ataxia and was found to have peripheral and CSF eosinophilia. MRI demonstrated symmetric, confluent T2 hyperintense signal in the cerebral and cerebellar deep white mater, which helped differentiate B. procyonis meningoencephalitis from other infectious and non-infectious causes of eosinophilic meningoencephalitis. Early recognition and treatment of B. procyonis meningoencephalitis are important for improved outcomes, and careful review of neuroimaging can play a critical role in suggesting the diagnosis.
Fatal SARS in X-Linked Lymphoproliferative Disease Type 1: A Case Report
Authors: Ming Hin Chung, Gilbert T Chua, Daniel Leung, Koon Wing Chan, John Nicholls, Yu Lung Lau
Journal: Front Pediatr (2022)
Keywords: covid-19, x-linked lymphoproliferative disease type 1 (xlp1), agammaglobulinemia, inborn error of immunity, severe acute respiratory syndrome (sars)
MeSH terms: Case Reports

X-linked lymphoproliferative disease (XLP1) is an inborn error of immunity (IEI) with severe immune dysregulation caused by a mutation in the SH2D1A gene resulting in the absence or dysfunction of signaling lymphocytic activation molecule (SLAM)-associated protein (SAP). The severe acute respiratory syndrome (SARS) caused by SARS-coronavirus (CoV), a highly pathogenic CoV, has been shown to only cause mild diseases in Asian children. We report on a 5-year-old Nepalese boy with agammaglobulinemia and probable SARS who died of diffuse alveolar damage 22 days after admission amid the SARS outbreak. The index patient and his younger brother were genetically confirmed to have XLP1. In the current coronavirus disease 2019 (COVID-19) pandemic, most children also had mild disease only. Children with severe COVID-19 would warrant investigations for underlying IEI, particularly along the pathways leading to immune dysregulation.
Management of Pediatric Mandibular Fracture With Acrylic Cap Splint
Authors: Himaja Swayampakula, Shreya Colvenkar, Bhuvaneshwari Kalmath, Jayasri Vanapalli, Mohammed Ali Zaheer
Journal: Cureus (2023)
Keywords: circummandibular wiring, fracture, mandible, pediatric, trauma
MeSH terms: Case Reports

Trauma during childhood can have an extreme mental jolt on the minds of growing children. The fundamentals of treatment of jaw fractures vary among children and adults. In children, minimal manipulation of facial skeleton is necessary to rehabilitate the supporting bony framework to pre-trauma condition. The procedure should not only be non-intrusive but also cause minimum malfunction and aesthetic disability. The case report presents the successful management of a seven-year-old boy with mandibular symphysis fracture using an acrylic cap splint retained with circum-mandibular wiring.
Atypical Myocardial Infarction with Apical Thrombus and Systemic Embolism: A Rare Presentation of Likely JAK2 V617F-Positive Myeloproliferative Neoplasm
Authors: Muhammed Atere, Rana Al-Zakhari, Jennifer Collins, Francesco Rotatori, Lloyd Muzangwa
Journal: Case Rep Oncol Med (2020)
MeSH terms: Case Reports

A few types of myeloproliferative neoplasms may be significant for Janus-associated kinase 2 mutation, JAK2 V617F, including polycythemia vera, essential thrombocythemia, and primary myelofibrosis. The prevalence of JAK2 mutation is low in the general population but higher in patients with myeloproliferative neoplasms. Some patients with JAK2 V617F-positive essential thrombocythemia are asymptomatic, but others may develop hemorrhagic or thromboembolic complications. Thromboembolism may occur in vessels of high flow organs like the heart and, thereby, present as myocardial infarction. Nonetheless, these patients are usually symptomatic with complaints of chest pain, for example. Atypical (asymptomatic) myocardial infarction with mild thrombocytosis may be the first clue for possible essential thrombocythemia with JAK2 V617F. In this report, we discuss a case of atypical (asymptomatic) myocardial infarction with secondary thromboembolism in a patient positive for JAK2 V617F with a likely myeloproliferative neoplasm.
Mucocutaneous leishmaniasis in a cocaine user: diagnostic and therapeutic knowledge
Authors: Lissiê Lunardi Sbroglio, Viviane Maria Maiolini, Irene Machado Moraes Alvarenga Rabelo, Gabriela Almeida Giraldelli, Luciana Patrícia Tuccori, Rodrigo Guimarães Cunha
Journal: Rev Soc Bras Med Trop (2020)
MeSH terms: Antiprotozoal Agents / administration & dosage, Cocaine-Related Disorders / complications, Leishmaniasis, Mucocutaneous / diagnosis, Meglumine Antimoniate / administration & dosage, Case Reports, Adult, Humans, Leishmaniasis, Mucocutaneous / complications, Leishmaniasis, Mucocutaneous / drug therapy, Male, Antiprotozoal Agents, Meglumine Antimoniate

Mucocutaneous leishmaniasis (MCL) is a chronic infection that can affect the skin and mucous membranes. We report a case of oral, nasopharyngeal, and penile lesions in a 35-year-old cocaine user. The patient presented with ulcerated lesions in 2014. Histopathologic analysis revealed amastigotes, and serological test results were positive for leishmaniasis. Systemic therapy with meglumine antimoniate was administered; however, the patient failed to present for follow-up. In 2018, he returned with nasal collapse, and another histopathologic test confirmed MCL. This case illustrates the importance of careful differential diagnosis of skin and mucous ulcers to identify the particular pathology.
Case Report: A Chlamydia psittaci pulmonary infection presenting with migratory infiltrates
Authors: Jundi Wang, Yurou Zhu, Qiongya Mo, Yanfei Yang
Journal: Front Public Health (2022)
Keywords: ct, chlamydia psittaci, mngs, migratory infiltrates, pneumonia
MeSH terms: Chlamydophila psittaci, Pneumonia, Psittacosis / diagnosis, Case Reports, High-Throughput Nucleotide Sequencing, Humans, Lung, Male, Middle Aged

Community-acquired pneumonia is a public health problem in all countries in the world, with a broad range of causative agents and Chlamydia psittaci infection tends to be overlooked. Pulmonary migratory infiltrates are commonly seen in eosinophilic pneumonia, cryptogenic organizing pneumonia, etc. However, the association of Chlamydia psittaci and pulmonary migratory infiltrates has been seldom described in literatures before. We reviewed a 64-year-old man referred to our hospital for treatment against Chlamydia psittaci pneumonia which was diagnosed by metagenomics next generation sequencing (mNGS). During the treatment period, chest imaging showed migratory infiltrates, which has been rarely described before.