OpenMed/multicare-articles huggingface dataset:
Recurrence of Drug-Induced Lupus Secondary to Vedolizumab Use in a Patient With Crohn's Disease
Authors: Vanessa I Rodriguez, Akshay Mathavan, Akash Mathavan, Diana N Rodriguez, Catalina Sanchez Alvarez, Angela Pham
Journal: ACG Case Rep J (2024)
Keywords: crohn's disease, ibd, drug-induced lupus, infliximab, vedolizumab
MeSH terms: Case Reports

Drug-induced lupus is an autoimmune phenomenon characterized by the development of systemic lupus erythematosus-like clinical features after drug exposure. The entity is a clinical diagnosis. Evaluation consists of recognizing systemic lupus erythematosus-like features, identifying an appropriate causative agent, observing elevations of characteristic autoantibodies, and obtaining positive response with drug discontinuation. Vedolizumab is an anti-α4β7 antibody used in the treatment of ulcerative colitis and Crohn's disease. We report a novel case of drug-induced lupus recurrence secondary to vedolizumab use in a patient with Crohn's disease, emphasizing diagnostic evaluation, and provide a brief review of the published literature.
Conservative Management With a Multimodal Approach of a 12-Week Cervical Ectopic Pregnancy With Fetal Megacystis
Authors: Daniela Nuti Oprescu, Ana Elena Martiniuc, Monica Mihaela Cirstoiu, Elena Theodora Giubegeanu, Oana Daniela Toader
Journal: Cureus (2024)
Keywords: cervical pregnancy, ectopic pregnancy, fertility preservation, megacystitis, uterine artery embolization
MeSH terms: Case Reports

Cervical ectopic pregnancy is the rarest kind of ectopic pregnancy, and it is known as the implantation of an embryo into the cervical mucosa. It is commonly associated with complications such as hemorrhage from the cervix and can lead to severe consequences if it is not treated early. For this reason, the treatment for a cervical pregnancy often requires an abdominal hysterectomy. To avoid such radical management, several conservative methods of termination have been used. In this paper, we report a complex management of one of our ectopic cervical cases, which includes embolization of the uterine arteries, treatment with methotrexate and mifepristone, evacuation of the pregnancy followed by local hemostatic sutures and application of a balloon in the cervix. The post-operative period was uneventful. After a three-day postoperative stay, the patient was discharged. The management options employed in the presented case achieved the goal of preserving fertility for our patient. There are no specific guidelines for the treatment of cervical pregnancies in advanced gestational age.
Case Report: From epilepsy and uterus didelphys to Turner syndrome-associated dysgerminoma
Authors: Jinghua Li, Haipeng Zhu, Xuelian Ma, Jia Li, Jing Xue, Limin Feng
Journal: Front Genet (2024)
Keywords: kit mutation, turner syndrome, dysgerminoma, epilepsy, uterus didelphys
MeSH terms: Case Reports

Dysgerminoma is a rare occurrence in Turner syndrome patients without Y chromosome mosaicism or hormone therapy during puberty. We present a unique case of a 33-year-old nulliparous Chinese woman with intermittent epilepsy and Mullerian anomalies carrying a double uterus, cervix, and vagina. The patient is also characterized as having Turner syndrome accompanied by 46,X, del(Xp22.33-11.23) and del(2)(q11.1-11.2). MRI exhibited a 17.0 cm × 20.0 cm × 10.5 cm solid ovarian lesion. Radical surgery and pathology revealed dysgerminoma at stage IIIc with lymphatic metastases and a KIT gene mutation identified in exon 13. Furthermore, the tumor microenvironment (TME) displayed robust expression of CD4+ T lymphocytes and PD-1, whereas the distribution of CD8+ T lymphocytes and PDL-1 was sporadic. Despite the administration of enoxaparin to prevent thromboembolism, the patient experienced multiple cerebral infarctions during chemotherapy. Subsequently, the patient chose to decline further treatment and was discharged. This exceptional case imparts several noteworthy lessons. First, the coexistence of Mullerian anomalies, although rare, is not incompatible with Turner syndrome. Second, screening for KIT mutations is imperative to reduce the risk of dysgerminoma in Turner syndrome, especially for patients with Y mosaicism who are recommended for hormone replacement therapy. Lastly, comprehensive anticoagulation therapy is crucial for Turner syndrome patients undergoing cisplatin-based chemotherapy.
Case report: Febrile infection-related epilepsy syndrome in a 14-year-old girl with multiple organ failure and lethal outcome
Authors: Lars Ruttkowski, Ines Wallot, Marie Korell, Elke Daur, Peter Seipelt, Andreas Leonhardt, Stefanie Weber, Nadine Mand
Journal: Front Neurosci (2024)
Keywords: fires, norse, multiple organ failure, pediatric neurology, pediatric status epilepticus
MeSH terms: Case Reports

We report a case of an otherwise healthy 14-year-old girl with febrile infection-related epilepsy syndrome (FIRES), multiple organ failure (MOF), and ultimately a lethal outcome. This is a rare case of FIRES with MOF and consecutive death. Only a few cases have been described in the literature. The adolescent girl was initially admitted to our pediatric emergency department with a first episode of generalized tonic-clonic seizures after a short history of fever a week before admission. Seizures progressed rapidly into refractory status epilepticus without any evidence of the underlying cause, and treatment subsequently had to be escalated to thiopental anesthesia. Since the initial diagnostics showed no promising leads, the rare syndrome of FIRES was suspected, representing a catastrophic epileptic encephalopathy linked to a prior benign febrile infection. Methylprednisolone, intravenous immunoglobulins, and a ketogenic diet were initiated. Respiratory, circulatory, kidney, and liver failure developed during treatment, requiring increasing intensive care. Multiple attempts to deescalate antiepileptic treatment resulted in recurrent status epilepticus. A cranial MRI on the 10th day of treatment revealed diffuse brain edema and no cerebral perfusion. The patient was declared dead on the 11th day of treatment. FIRES should be taken into account in previously healthy children with a new onset of difficult-to-treat seizures after a short febrile infection when no other cause is apparent. First-line treatment, besides seizure control, is the early initiation of immunomodulatory therapy and the start of a ketogenic diet. As treatment is difficult and MOF may develop, patients should be transferred to a specialized children's hospital providing full intensive care.
Ventilator-associated pneumonia due to Aeromonas hydrophila: A rare case report
Authors: Amit Srivastava, Sarvodaya Tripathy, Shreyas Gutte, Chinmoy Sahu, Mohan Gurjar, Sangram Singh Patel
Journal: Access Microbiol (2023)
Keywords: aeromonas hydrophila, critically ill, intensive care unit, ventilator-associated pneumonia
MeSH terms: Case Reports

Introduction:

Aeromonas hydrophila is an opportunistic pathogen that can cause various infections, including pneumonia, in immunocompromised individuals. This case report presents a rare occurrence of ventilator-associated pneumonia (VAP) caused by Aeromonas hydrophila in an apparently non-immunocompromised patient.
Case presentation:
The patient exhibited signs and symptoms of VAP and was successfully treated with intravenous ciprofloxacin. The discussion highlights the characteristics of Aeromonas species, its virulence factors, risk factors for infection, and antibiotic profile.
Conclusion:
It emphasizes the need for awareness and suspicion of Aeromonas as a potential cause of VAP in ICU settings, as well as the importance of early detection and appropriate treatment for improved outcomes.
Staged Tendon Repair to Improve Range of Motion in Tamai Zone 4 Replantation: Two Case Reports
Authors: Takeo Matsusue
Journal: Arch Plast Surg (2024)
Keywords: articular, finger, range of motion, rehabilitation, replantation, tendon

Tamai zone 4 replantation, defined as the replantation at a level proximal to the flexor digitorum superficialis' insertion and distal to where the common digital artery branches into the proper digital artery, has poor functional results because making orthosis and rehabilitation protocols that protect the bone and the flexor and extensor tendons simultaneously difficult. Two cases of Tamai zone 4 replantation are presented: one case of an index finger replantation at the proximal phalanx and a case of ring finger replantation at the proximal interphalangeal joint. The author did not repair the flexor tendon intentionally in the primary replantation and performed two-stage flexor tendon reconstruction later. The total active motions (TAMs) at the last follow-up were 215 and 180 degrees, respectively, with the latter distal interphalangeal joint being an arthrodesis. Both cases had no extension lag in the proximal interphalangeal joint. These results were much better than those in previous reports, in which the mean TAM was 133 degrees or less. The good results appeared to be mainly due to the reasonable and clear postoperative rehabilitation protocols made by the proposed procedure. This procedure may be useful for obtaining reproducible functional results even in Tamai zone 4 replantation.
Case report: Systemic multi-organ involvement in an adult-onset immunodeficiency patient infected with Talaromyces marneffei
Authors: Kun Li, Yuping Zhang, Dan Zhang, Qing Chen, Xueling Fang
Journal: Front Immunol (2024)
Keywords: talaromyces marneffei, adult-onset immunodeficiency, immunodeficiency, interferon-γ, respiratory failure
MeSH terms: Mycoses / diagnosis, Mycoses / drug therapy, Mycoses / immunology, Mycoses / microbiology, Talaromyces, Case Reports, Antifungal Agents / therapeutic use, Autoantibodies / blood, Autoantibodies / immunology, Female, High-Throughput Nucleotide Sequencing, Humans, Middle Aged, Antifungal Agents, Autoantibodies, Talaromyces marneffei

Adult-onset immunodeficiency (AOID) mediated by anti-interferon-γ autoantibodies (AIGA) is a rare condition, particularly prevalent in Southeast Asia and southern China. We present a case study of a 62-year-old female with AOID who developed a severe pulmonary infection caused by Talaromyces marneffei (TM), leading to acute respiratory failure, generalized rash, multiple lymphadenopathies, bone destruction, and a mediastinal mass. Treatment included mechanical ventilation, antifungal medication, and corticosteroids, resulting in complete recovery and discharge. This case underscores the challenges of managing complex infections in AOID patients and highlights the importance of early diagnosis through metagenomic next-generation sequencing (mNGS) and appropriate intervention to improve clinical outcomes.
Pregnancy in Morrison Pouch: a case report
Authors: Frank Martin Sudai, Lusajo Mwagobele, Lameck Mdengo, Rabson Bagoka, Stanley Zakaria Binagi, Joseph Bee, Jesca Paul Lebba
Journal: Pan Afr Med J (2024)
Keywords: abdominal pregnancy, case report, ectopic pregnancy, maternal hemorrhage
MeSH terms: Abdominal Pain / etiology, Laparotomy / methods, Pregnancy, Abdominal / diagnosis, Pregnancy, Abdominal / surgery, Case Reports, Anemia / diagnosis, Anemia / etiology, Blood Transfusion, Fatal Outcome, Female, Hemorrhage / etiology, Humans, Pregnancy, Shock, Hemorrhagic / etiology, Young Adult

Abdominal pregnancy is a rare form of ectopic pregnancy (accounting for 1% of all ectopic pregnancies). Depending on gestational age and its location various symptoms and signs may be exhibited. This study aimed to report a case of abdominal pregnancy occurring in the Morrison Pouch with a primary presentation of right upper quadrant pain and to highlight complications that may arise in the management of abdominal pregnancy located in the Morrison Pouch. A 22-year pregnant woman at gestation of 22 weeks presented with a right upper quadrant mass and pain. Ultrasound examination revealed a live extrauterine singleton at Morrison Pouch, full blood count showed severe anemia. The patient received a blood transfusion in seven days and underwent emergency laparotomy after experiencing sudden acute internal hemorrhage but died a few hours post laparotomy due to hemorrhagic shock. Abdominal pregnancy carries a high risk of maternal hemorrhage as described in this case.
Case report: Stereotactic MR-guided adaptive radiotherapy for inoperable urothelial carcinoma at the renal pelvis
Authors: Wajana Thaweerat, Pittaya Dankulchai
Journal: Front Oncol (2024)
Keywords: mr-guided radiotherapy, adaptive radiotherapy, renal pelvis tumor, stereotactic body radiotherapy, transitional cell carcinoma, urothelial carcinoma
MeSH terms: Case Reports

We report the case of an 87-year-old woman with upper tract urothelial carcinoma at the left renal pelvis. She received stereotactic body radiotherapy of 35 Gy in five fractions for palliative treatment of hematuria that was delivered by a 1.5-T magnetic resonance (MR) imaging-guided linear accelerator. Her symptom was relieved after treatment, and posttreatment imaging revealed a complete response of the primary tumor. Thus, this case showed that stereotactic MR-guided radiotherapy could be an appealing option for inoperable patients although radiotherapy is infrequently mentioned in the current treatment guideline of upper tract urothelial carcinoma. Daily adaptive planning from MR images obtained before treatment could improve the target dose and minimize the organ at risk dose. This may lead to a decrease in radiation adverse effects including worsening renal function due to the renal pelvis tumor's proximity to the kidney.
Malignant lung PEComa (clear cell tumor): rare case report and literature review
Authors: Marcos Adriano Garcia Campos, Lucas Fernandes Vasques, Rafael Goulart de Medeiros, Érico Murilo Monteiro Cutrim, Ana Júlia Favarin, Sarah Rebecca Machado Silva, Gyl Eanes Barros Silva, Marcelo Padovani de Toledo Moraes, Mariana Lopes Zanatta, Diego Aparecido Rios Queiróz
Journal: Front Oncol (2023)
Keywords: hmb-45, pecoma, cathepsin k, pulmonary cancer, sugar tumor
MeSH terms: Case Reports

Clear cell tumors of the lung (CCTL), or "sugar tumors" of lung, are very uncommon lesions and are mostly benign perivascular epithelioid cell (PEC) tumors with no specific morphologic features. Fewer than 100 cases have been reported; the aggressive nature demonstrated in sporadic reports has rarely been described in the literature. Although the course is generally described as benign, eight reported cases showed malignant behavior. We report a case of a PEC with a malignant presentation in a young man, correlating the main characteristics of the tumor with other cases reported in the literature to better elucidate this rare presentation. We also performed a literature review of reports on benign and malignant CCTL cases, with a focus on clinical, imaging, and immunohistochemical differentiation. CCTLs are rare tumors that require histopathological and immunohistochemical confirmation; to date, criteria that can predict malignant evolution are lacking.
Neoadjuvant Chemotherapy Approach to Pineal Germinoma: A Case Report
Authors: Nagham Bazzi, Wajih A Saad, Hala Bazzi, Mohammad Ali Almokdad, Abdo Mghames
Journal: Cureus (2024)
Keywords: brain tumor, germinoma, headache, neoplasms, pineal gland tumor
MeSH terms: Case Reports

Intracranial germ cell tumors (GCTs) are rare malignant tumors with a peak incidence around puberty. The pineal region is the most commonly involved area of all intracranial GCTs. Due to the heterogeneous tumor origin, subtypes, and presentation, diagnosis and management are challenging. Complicated pineal germinomas are rarely reported in the literature. Here, we report a rare case of pineal germinoma with hydrocephalus and discuss the potential treatment approach. A 20-year-old boy presented to the hospital with vomiting and a decreased level of consciousness. The brain magnetic resonance imaging (MRI) revealed a pineal tumor. A ventriculoperitoneal shunt was placed to relieve the increased intracranial pressure. The patient underwent a suboccipital craniotomy with excisional biopsy of the pineal region tumor due to its critical location, as imaging studies alone may not be sufficient to establish a definitive diagnosis. Although there has been a rise in reported cases of germinoma tumors, there is currently no standardized therapeutic approach for treating them. Therefore, more randomized controlled cohort studies are necessary to evaluate potential treatments and develop a therapeutic approach.
A Novel Ornithine Aminotransferase Splice Site Mutation Causes Vitamin B6-Responsive Gyrate Atrophy
Authors: Samira Molaei Ramshe, Safoura Zardadi, Elham Alehabib, Ramin Nourinia, Javad Jamshidi, Mohsen Soosanabadi, Hossein Darvish
Journal: J Ophthalmic Vis Res (2024)
Keywords: mutation, ornithine aminotransferase, vitamin b6, gyrate atrophy
MeSH terms: Case Reports

Purpose:
Gyrate atrophy of the choroid and retina (GACR) is a rare congenital disorder and mutations in the ornithine aminotransferase (OAT) gene has been specified as the underlying cause. Patients show a high level of ornithine in body fluids which may be controlled by low protein diets. Pyridoxine (vitamin B6) supplementation may also be effective, however, most patients appear to be nonresponsive to this modality of treatment.
Case report:
Here, we report a characterized case of a vitamin B6-responsive GACR who had a splicing mutation in the OAT gene. The GACR diagnosis was confirmed through the clinical features, imaging, biochemical findings, and whole-exome sequencing (WES) results. WES data revealed the splicing mutation in intron 4 of the OAT gene (NM_001322967: c.425-1G>A).
Conclusion:
Our knowledge about the diagnosis and treatment of GACR can be improved by identifying novel mutations in the OAT gene and accurate follow-up of the patients to determine how they respond to treatment.
Three Cases of Arteritic Anterior Optic Neuropathy Several Months after COVID-19 Vaccination
Authors: Yuichi Yamamoto, Ken Ogino, Satoshi Yasuhara, Yu Kawashima, Toshiya Miki
Journal: Case Rep Ophthalmol Med (2023)
MeSH terms: Case Reports

Background:
Vaccines have been approved worldwide to control the coronavirus disease-19 (COVID-19). However, the postvaccination side effects remain controversial. Here, we describe three Japanese cases of arteritic anterior ischaemic optic neuropathy (AAION) following COVID-19 vaccination. Case presentation. The first case involved an 87-year-old woman who presented with vision loss in the right eye 2 months after her second COVID-19 vaccine and in the left eye 2 days later. The second case involved an 88-year-old woman who presented with vision loss in both eyes 3 months after receiving a second vaccine. The third case involved an 80-year-old man who presented with vision loss in the right eye 5 months after receiving a second vaccine. The C-reactive protein level and erythrocyte sedimentation rate were elevated in all patients. Biopsy of the temporal artery or auricular cartilage showed arteritic occlusion in case 2 and polychondritis in case 3. These patients were referred to a local Japanese hospital in 2021 over a period of no longer than 3 months.
Conclusion:
We observed three cases of AAION after the affected individuals received their second COVID-19 vaccine. Further long-term investigations of ophthalmological events after COVID-19 vaccination are warranted.
Fibromyalgia in patients infected with HTLV-1 and HTLV-2
Authors: Bianca Lumi Inomata Silva, Francisco Erivan da Cunha Rodrigues, Márcio Yutaka Tsukimata, Bruno José Sarmento Botelho, Luciana Cristina Coelho Santos, Gabriel Dos Santos Pereira Neto, Aline Cecy Rocha Lima, Natália Pinheiro André, Sarah Marques Galdino, Danniele Chagas Monteiro, Gilberto Toshimitsu Yoshikawa, Leonardo Teixeira Mendonça, Juliana Lasmar Ayres do Amaral, Rosana de Britto Pereira Cruz, Débora Oliveira Onuma, Antonio Carlos Rosário Vallinoto, Bárbara Nascimento de Carvalho Klemz, Izaura Maria Vieira Cayres Vallinoto
Journal: Front Med (Lausanne) (2024)
Keywords: htlv-1, htlv-2, fibromyalgia, infection, pain, virus

Background:
Reports on the association between HTLV-2 infection and the development of diseases in the human host are rare, which has led the scientific community to believe that HTLV-2 is not an important etiological agent of lymphoproliferative or neurodegenerative disorders, which is the case for HTLV-1. In the present study, we demonstrated cases of fibromyalgia in HTLV-1 carriers and, in an unprecedented finding, in two patients with confirmed HTLV-2 infection.
Methods:
A total of 957 individuals visited the Virology Laboratory at the Federal University of Pará for screening and confirmation tests for HTLV-1/2 infection. Individuals with confirmed HTLV-1 and HTLV-2 infection were clinically evaluated for signs and symptoms associated with infection.
Results:
Sixty-nine individuals (7.2%) were identified as positive for HTLV infection, with 56 confirmed cases of HTLV-1 infection (5.9%), 12 cases of HTLV-2 infection (1.2%) and one case classified as undetermined (0.1%). Sixteen (23.2%) of these patients presented with rheumatological signs and complained of diffuse pain throughout the body; 12 of whom were infected by HTLV-1 (75%) and 4 were infected by HTLV-2 (25%). After anamnesis and careful evaluation, four patients were diagnosed with fibromyalgia, two of whom were infected by HTLV-1 (16.7%; 2/12) and two by HTLV-2 (50%; 2/4). The clinical follow-up and laboratory analysis results are reported in detail in this paper.
Conclusion:
Considering the clinical cases presented herein as the first reports of patients with HTLV-2 infection with clinical symptoms of fibromyalgia, the importance of further studies on the pathogenicity of HTLV-2, similar to what have already been performed for HTLV-1, is highlighted. Our results also confirm previous evidence of an association between HTLV-1 infection and fibromyalgia.
Cytomegalovirus-Induced Hemophagocytic Lymphohistiocytosis in an Immunocompromised Patient with Inflammatory Bowel Disease
Authors: Alessandro Pedicelli, René P Michel, Nick Krassakopoulos
Journal: Case Rep Hematol (2024)
MeSH terms: Case Reports

Hemophagocytic lymphohistiocytosis (HLH) is a rare and often fatal syndrome of immune hyperactivation, cytokine dysregulation, and severe inflammation. This severe syndrome is commonly triggered by infection, malignancy, autoimmunity, or immunosuppression. We present herein the case of a 56-year-old-female diagnosed with HLH triggered by an acute cytomegalovirus (CMV) infection with viremia in the context of immunosuppression for inflammatory bowel disease. This case highlights the importance of utilizing multiple diagnostic tools, prompt initiation of anti-hemophagocytic treatment, and management of the underlying etiology, to prevent significant morbidity and mortality.