OpenMed/multicare-articles huggingface dataset:
Congenital malaria and neonatal bacterial co-infection in twins prematurely born to a mother with sickle-cell anaemia in the Democratic Republic of the Congo
Authors: Junior E Mudji, Johannes Blum, Timothy D Rice, Frederick N Baliraine
Journal: Malariaworld J (2017)
MeSH terms: Case Reports

Background:
We report cases of gestational and congenital malaria with twin prematurity, low birth weight and bacterial co-infection. Congenital malaria is often misdiagnosed for lack of specific symptoms and a general lack of awareness of this presumably uncommon condition, and its diagnosis and prognosis become even more complex in the event of bacterial co-infections.
Case presentation:
A 35-weeks pregnant woman with sickle-cell disease and a history of spontaneous abortions was admitted at Vanga Hospital in DR Congo. She had fever (38.9°C) and microscopy-confirmed P. falciparum malaria and was put on 80/480 mg artemether-lumefantrine. She soon went into active labour, during which both twins developed acute foetal distress and were promptly delivered by C-section. The twins were underweight, and both had P. falciparum malaria at birth and were given 20 mg quinine twice daily. Both developed fever on the third day; a bacterial infection was suspected and 200 mg ceftriaxone was added to their treatment. Fever in both twins quickly resolved, and one twin totally recovered within 2 days of antibiotic treatment. The other twin developed acute respiratory distress and hypoxia and died.
Discussion:
This is a case of gestational and congenital malaria with prematurity, low birth weight and bacterial co-infection, but the patients were initially only treated for malaria based on their malaria-positive blood smears at birth.
Conclusions:
In malaria-endemic areas, babies should be screened for congenital malaria. Even with a confirmed malaria infection in the new-born, it is important consider the possibility of bacterial co-infections.
Thunderclap headache revealing dural tears with symptomatic intracranial hypotension: Report of two cases
Authors: Dana Antonescu-Ghelmez, Ioana Butnariu, Florian Antonescu, Cristina Maier, Adriana Moraru, Amanda Ioana Bucur, Daniela Nicoleta Anghel, Sorin Tuţă
Journal: Front Neurol (2023)
Keywords: csf leakage, case report, intracranial hypotension, post-traumatic dural tear, sacral fracture, sacral osseous fragment, severe headache, thunderclap headache
MeSH terms: Case Reports

Cerebrospinal fluid (CSF) leakage is considered the cause of spontaneous intracranial hypotension (SIH), an important etiology for new daily persistent headaches and a potentially life-threatening condition. Minor traumatic events rarely lead to CSF leakage, contrasting with iatrogenic interventions such as a lumbar puncture or spinal surgery, which are commonly complicated by dural tears. Most meningeal lesions are found in the cervicothoracic region, followed by the thoracic region, and rarely in the lumbar region, and extremely rarely in the sacral region. We describe two patients admitted to our hospital for severe headaches aggravated in the orthostatic position, with a recent history of minor trauma and sustained physical effort, respectively. In the first case, a bone fragment pierced an incidental congenital meningocele creating a dural fistula. An extensive extradural CSF collection, spanning the cervicothoracic region (C4-T10), was described in the second case. In both patients, the clinical evolution was favorable under conservative treatment.
Case report: Bi-atrial thrombus after occlusion of atrial septal defect with acute cerebral infarction and pulmonary embolism
Authors: Wei Xiong, Li Tang, Wei Long, Jin Liu, Haibo Song
Journal: Front Cardiovasc Med (2022)
Keywords: acute cerebral infarction, atrial septal defect, bi-atrial thrombus, pulmonary embolism, transcatheter closure
MeSH terms: Case Reports

A 49-year-old man presented to the hospital with symptoms of acute cerebral infarction and pulmonary embolism who underwent transcatheter closure of atrial septal defect a year before. Transthoracic echocardiography showed a 13 × 9 mm hypoechoic mass attached to the left-atrial side of the device, which was suspected to be a neoplasm or thrombus. The patient was indicated for surgery after multidisciplinary discussion due to ineffective medical therapy and typical stroke and pulmonary embolism symptoms. Three-dimensional transesophageal echocardiography (3D-TEE) revealed left-atrial vegetation (21 × 16 mm) and right-atrial vegetation (8 × 6 mm) attached to the device, which was confirmed as thrombus by surgical separation and laboratory examination. This case highlights the importance of 3D-TEE and a multidisciplinary team in the diagnosis and therapy of device-related thrombus.
Primary Cardiac Lymphoma Presenting with Thrombocytopenia, Right Heart Failure, and Cardiogenic Shock
Authors: Samantha Kurniawan, Gita Mathur, Yvonne Bogun, Giselle Kidson-Gerber
Journal: Case Rep Hematol (2023)
MeSH terms: Case Reports

Primary cardiac lymphoma (PCL) is a rare, potentially fatal subtype of non-Hodgkin's lymphoma. Thrombocytopenia has also infrequently been reported in association with other primary cardiac tumours and can add substantial morbidity to an already life-threatening diagnosis if present. We report a rare case of a 70-year-old man who presented with thrombocytopenia (91 × 109/L) and progressive right heart failure. Transthoracic echocardiogram revealed a large 8 × 4 cm right atrial mass with severe tricuspid obstruction, confirmed as PCL on subsequent endomyocardial biopsy and immunohistochemistry. He deteriorated into cardiogenic shock precipitated by atrial fibrillation, with worsening thrombocytopenia (18 × 109/L) in the setting of ischaemic hepatitis. The patient stabilised with initiation of high dose steroids prior to tissue diagnosis and platelet counts normalised following chemotherapy. This case demonstrates the importance of considering PCL as a diagnosis and preemptive initiation of high dose steroids to improve outcomes in PCL associated with cardiogenic shock. This case also elucidates a potential pathophysiological association between PCL and thrombocytopenia.
Asymptomatic pneumoperitoneum in pneumatosis coli: A misleading operative indication
Authors: Marta Ribolla, Luigi Conti, Edoardo Baldini, Gerardo Palmieri, Carmine Grassi, Filippo Banchini, Maria Diletta Dacco', Patrizio Capelli
Journal: Int J Surg Case Rep (2020)
Keywords: pneumatosis cystoides intestinalis, pneumoperitoneum
MeSH terms: Case Reports

Introduction:
Pneumatosis cystoides intestinalis (PCI) is a rare clinical pathology characterized by sub-mucosal and/or sub-serous cysts of free gas, forming cystic lesions usually ranging from 0.5 to 2.0 cm in size within the gastrointestinal tract. About 3% of patients with PCI develop complications such as pneumoperitoneum, intestinal volvulus, obstruction, or hemorrhage, these cases need immediate surgical intervention. Cyst rupture can produce peritoneal irritation and pneumoperitoneum.
Presentation of case:
A 65-years-old woman was admitted to the Emergency Department for epileptiform convulsions. Her medical hystory included epilepsy, diabetes, lichenoid dermatitis, hypothyroidism, severe cognitive impairment. Abdominal CT scan revealed a dilated large intestine with parietal pneumatosis from the appendix to the transverse colon associated to extensive pneumoperitoneum. The patient underwent emergency laparotomy which revealed the presence of gas within the wall of right and transverse colon and distension of great omentum. No resection was needed as normal blood supply to the bowel present.
Discussion:
Pneumatosis coli can be both asymptomatic or life-threatening condition associated to bowel infarction; this situation can mimic a bowel perforation causing pneumoperitoneum - that sometimes is a non-surgical pneumoperitoneum - and it could be a misleading indication to surgical exploration especially in the case of uncertain origin of a septic shock.
Conclusions:
We report a case of pneuomoperitoneum due to PCI. Surgical intervention was required for patient's conditions and unclear origin of the sepsis.
Gallbladder agenesis: A case report and review of the literature
Authors: Irakli Pipia, George Kenchadze, Zaza Demetrashvili, Grigol Nemsadze, Lika Jamburia, Tamari Zamtaradze, Ivane Abiatari
Journal: Int J Surg Case Rep (2018)
Keywords: gallbladder agenesis, laparoscopy, mrcp

Introduction:
Gallbladder agenesis is a rare congenital abnormality with an incidence of 10-65 per 100,000. Approximately half of these patients are surgically operated on because of the symptoms similar to biliary colic, and correct diagnosis is established intra-operatively. We present a clinical case of gallbladder agenesis from our practice.
Presentation of case:
A 49 (forty-nine) - year- old women was admitted in the Emergency Department of our clinic. Symptoms were similar to the biliary colic. Ultrasonography showed hyperechogenic acoustic shadow on the projection of the gallbladder which was considered as constricted gallbladder and cholecystolithiasis was diagnosed. Laparoscopic cholecystectomy was considered. During laparoscopy gallbladder could not be found. Surgical operation was completed without conversion. Postoperative treatment included analgesics and antispasmodics. Pre-operative symptoms disappeared. One month later magnetic resonance cholangiopancreatography (MRCP) confirmed gallbladder agenesis diagnosis. Health condition of the patient is satisfactory, without any complications after a year of surgery.
Discussion:
Gallbladder agenesis presented with symptoms similar to biliary colic can be diagnosed without surgical intervention. Conservative treatment consists of antispasmodic drugs.
Conclusion:
If the shrunken gallbladder is detected on the ultrasound, additional radiological examinations are required. MRCP is considered as a test of choice among the radiological investigations. If gallbladder agenesis is identified on laparoscopy, there is no need for further conversion. For postoperative follow up examination MRCP investigation is recommended.
Meretoja's Syndrome: Lattice Corneal Dystrophy, Gelsolin Type
Authors: I Casal, S Monteiro, C Abreu, M Neves, L Oliveira, M Beirão
Journal: Case Rep Med (2017)
MeSH terms: Case Reports

Lattice corneal dystrophy gelsolin type was first described in 1969 by Jouko Meretoja, a Finnish ophthalmologist. It is caused by an autosomal dominant mutation in gelsolin gene resulting in unstable protein fragments and amyloid deposition in various organs. The age of onset is usually after the third decade of life and typical diagnostic triad includes progressive bilateral facial paralysis, loose skin, and lattice corneal dystrophy. We report a case of a 53-year-old female patient referred to our Department of Ophthalmology by severe dry eye and incomplete eyelid closure. She had severe bilateral facial paresis, significant orbicularis, and perioral sagging as well as hypoesthesia of extremities and was diagnosed with Meretoja's syndrome at the age of 50, confirmed by the presence of gelsolin mutation. At our observation she had bilateral diminished tear film break-up time and Schirmer test, diffuse keratitis, corneal opacification, and neovascularization in the left eye. She was treated with preservative-free lubricants and topical cyclosporine, associated with nocturnal complete occlusion of both eyes, and underwent placement of lacrimal punctal plugs. Ocular symptoms are the first to appear and our role as ophthalmologists is essential for the diagnosis, treatment, and monitoring of ocular alterations in these patients.
Rituximab Followed by Belimumab Controls Severe Lupus Nephritis and Bullous Pemphigoid in Systemic Lupus Erythematosus Refractory to Several Combination Therapies
Authors: Luca Petricca, Maria Rita Gigante, Annamaria Paglionico, Stefano Costanzi, Gisella Vischini, Clara Di Mario, Valentina Varriano, Giacomo Tanti, Barbara Tolusso, Stefano Alivernini, Giuseppe Grandaliano, Gianfranco Ferraccioli, Elisa Gremese
Journal: Front Med (Lausanne) (2020)
Keywords: belimumab, bullous pemphigoid, lupus nephritis, rituximab, sequential therapy
MeSH terms: Case Reports

Systemic lupus erythematosus (SLE) and bullous pemphigoid (BP) are chronic autoimmune diseases in which B cells play an important pathogenic role in the different stages of the disease. B cell-targeted therapies have been suggested as a new rational approach for treating SLE. Rituximab (RTX), an anti-CD20 chimeric monoclonal antibody, failed to achieve primary endpoints in two clinical trials (EXPLORER and LUNAR) despite multiple observational and retrospective studies showing its beneficial effect on SLE. Moreover, RTX is recommended in cases of BP that is unresponsive to conventional treatments. Belimumab (BLM), a human immunoglobulin G1 λ monoclonal antibody that inhibits soluble B-lymphocyte stimulator (BlyS)/B-cell activating factor (BAFF), is the only biological treatment approved for standard therapy of refractory autoantibody-positive active SLE. Animal models and a few case reports have supported the efficacy of the combined use of RTX followed by BLM as maintenance therapy in severe lupus nephritis (LN), suggesting that their combined use may be more effective than their single use, without compromising safety. In this study, we describe the clinical case of a SLE patient with predominant renal involvement in overlap with BP, refractory to conventional therapy including RTX alone, achieving significant steroid sparing and clinical remission under sequential treatment of RTX-BLM. Moreover, we describe the first case of BP successfully treated with BLM. This case report may encourage further clinical research studies in B lymphocyte targeted combination therapy in patients affected by SLE with major organ involvement or with refractory disease, suggesting that RTX and BLM sequential therapy may be a valid option for the treatment of SLE manifestations, including conventional therapy and RTX-resistant LN.
Acute Posterior Multifocal Placoid Pigment Epitheliopathy Sharing Characteristic OCT Findings of Vogt-Koyanagi-Harada Disease
Authors: Yuta Kitamura, Toshiyuki Oshitari, Masayasu Kitahashi, Takayuki Baba, Shuichi Yamamoto
Journal: Case Rep Ophthalmol Med (2019)
MeSH terms: Case Reports

A 17-year-old male presented with acute bilateral paracentral scotomata and blurred vision. Funduscopic examination showed bilateral macular serous retinal detachment and yellow-white placoid lesions at the level of retinal pigment epithelium. OCT study showed typical VKH disease findings with marked choroidal thickening and macular serous retinal detachment partly with subretinal septa in both eyes. FA demonstrated hypofluorescence at the placoid lesions in the early phase and hyperfluorescence in the late phase. Laboratory investigation showed negative result for HLA-DR4 serotype and the patient's cerebrospinal fluid test values were within normal range. We made the diagnosis of APMPPE from these results. At 2-month follow-up without the use of corticosteroids, OCT reexamination showed complete amelioration of subretinal fluid in both eyes. Patchy pigmentary lesions also resolved clinically with partial chorioretinal scars. The results in this case suggested OCT findings in APMPPE patients could be similar to characteristic features usually found in acute VKH disease. We recommend comprehensive assessments such as FA, cerebral spinal fluid analysis, and HLA typing which help in leading proper diagnosis.
Long term efficacy and safety of rivaroxaban plus cilostazol in the treatment of critical ischemia of the lower limbs in a frail, elderly patient with non valvular atrial fibrillation
Authors: Antonio Trani, Pietro Benedetto, Ferdinando Di Leo, Angela Baiano, Andrea Esposito, Danilo Menna, Arianna Allegretti, Pierluigi Antonino Cappiello, Domenico Dell'Edera
Journal: J Pharm Health Care Sci (2020)
Keywords: cilostazol, critical lower limb ischemia, elderly, non valvular atrial fibrillation, rivaroxaban
MeSH terms: Case Reports

Background:
Many patients with critical lower limb ischemia are not eligible for revascularization procedures. Still, given the emerging role of both platelet and coagulation activation in the formation of arterial thrombi, they may benefit from the novel anticoagulant and antiplatelet drugs.
Case presentation:
We describe the case of a male with critical lower limb ischemia complicated by older age, frailty, polymorbidity and non valvular atrial fibrillation, who was deemed as non eligible for surgery. The patient was successfully treated with the combination of rivaroxaban and cilostazol, and the clinical benefit was maintained throughout 32 months, with no occurrence of major or minor hemorrhagic or thrombotic events.
Conclusions:
To our knowledge, this is the first report on the efficacy and safety of such a combination therapy in critical lower limb ischemia. In a clinical setting in which alternative pharmacological approaches are urgently needed, the association of rivaroxaban and cilostazol warrants further investigations.
Autoamputation of the Appendix in a Chronic Adnexal Abscess
Authors: C Michele Markey, Lauren E Vestal
Journal: Case Rep Obstet Gynecol (2018)
MeSH terms: Case Reports

Autoamputation of the appendix has rarely been described in the literature. We present a case of a pelvic mass, thought to be a dermoid cyst based on preoperative imaging. After surgical removal and pathological examination, the mass was found to be a chronic pelvic abscess containing the right adnexa as well as an autoamputated vermiform appendix. Differentiating between gynecologic and gastrointestinal disease preoperatively can be difficult and often a definitive diagnosis cannot be made until surgical exploration and pathological review. However, to our knowledge, this is the first described case of a chronic pelvic abscess containing an autoamputated vermiform appendix.
Can early-onset acquired demyelinating syndrome (ADS) hide pediatric Behcet's disease? A case report
Authors: Mattia Pozzato, Robertino Dilena, Greta Rogani, Gisella Beretta, Sofia Torreggiani, Stefano Lanni, Alessandra Tozzo, Francesca Andreetta, Paola Cavalcante, Fabio Triulzi, Filippo Martinelli Boneschi, Francesca Minoia, Giovanni Filocamo
Journal: Front Pediatr (2023)
Keywords: behcet’s disease, acquired demyelinating syndromes (adss), children, early onset, magnetic resonance imaging
MeSH terms: Case Reports

Behcet's disease (BD) is a rare vasculitis characterized by multisystemic inflammation. Central nervous system (CNS) involvement is rare and heterogeneous, particularly in the pediatric population. A diagnosis of neuro-Behcet could be highly challenging, especially if neurological manifestations precede other systemic features; however, its timely definition is crucial to prevent long-term sequelae. In this study, we describe the case of a girl who, at 13 months of age, presented with a first episode of encephalopathy compatible with acute disseminated encephalomyelitis, followed, after 6 months, by a neurological relapse characterized by ophthalmoparesis and gait ataxia, in association with new inflammatory lesions in the brain and spinal cord, suggesting a neuromyelitis optica spectrum disorder. The neurological manifestations were successfully treated with high-dose steroids and intravenous immunoglobulins. In the following months, the patient developed a multisystemic involvement suggestive of Behcet's disease, characterized by polyarthritis and uveitis, associated with HLA-B51 positivity. The challenge presented by this unique case required a multidisciplinary approach involving pediatric neurologists, neuro-radiologists, and pediatric rheumatologists, with all of these specialists creating awareness about early-onset acquired demyelinating syndromes (ADSs). Given the rarity of this presentation, we performed a review of the literature focusing on neurological manifestations in BD and differential diagnosis of patients with early-onset ADS.
An interactive data visualisation application to investigate nosocomial transmission of infections
Authors: Catherine M Smith, David J Allen, Sameena Nawaz, Zisis Kozlakidis, Eleni Nastouli, Andrew Hayward, Katherine N Ward
Journal: Wellcome Open Res (2019)
Keywords: norovirus, cross infection, infection control, outbreak, software, virus genomics

Background: Healthcare-associated infections represent a major threat to patient, staff and visitor safety. Identification of episodes that are likely to have resulted from nosocomial transmission has important implications for infection control. Routinely collected data on ward admissions and sample dates, combined with pathogen genomic information could provide useful insights. We describe a novel, open-source, application for visualising these data, and demonstrate its utility for investigating nosocomial transmission using a case study of a large outbreak of norovirus infection. Methods: We developed the application using Shiny, a web application framework for R. For the norovirus case study, cases were defined as patients who had a faecal sample collected at the hospital in a winter season that tested positive for norovirus. Patient demographics and ward admission dates were extracted from hospital systems. Detected norovirus strains were genotyped and further characterised through sequencing of the hypervariable P2 domain. The most commonly detected sub-strain was visualised using the interactive application. Results: There were 156 norovirus-positive specimens collected from 107 patients. The most commonly detected sub-strain affected 30 patients in five wards. We used the interactive application to produce three visualisations: a bar chart, a timeline, and a schematic ward plan highlighting plausible transmission links. Visualisations showed credible links between cases on the elderly care ward. Conclusions: Use of the interactive application provided insights into transmission in this large nosocomial outbreak of norovirus, highlighting where infection control practices worked well or could be improved. This is a flexible tool that could be used for investigation of any infection in any hospital by interactively changing parameters. Challenges include integration with hospital systems for extracting data. Prospective use of this application could inform better infection control in real time.
Leiomyosarcoma arising in irradiated region after breast-conserving surgery: a case report
Authors: Satoshi Hayashi, Masahiro Kitada, Yoshinari Matsuda, Kei Ishibashi, Nana Takahashi
Journal: Surg Case Rep (2015)
Keywords: breast cancer, leiomyosarcoma, post-irradiation sarcoma, radiation therapy

Background:
Radiation therapy (RT) is considered a risk factor for the development of sarcoma in patients with breast cancer. However, there are few reports regarding post-irradiation sarcoma (PIS).
Case presentation:
The patient was a 59-year-old woman who presented with a chief complaint of induration in the lower outer quadrant of the left breast. She underwent breast-conserving surgery (BCS) for breast cancer located in the left upper inner region and received endocrine therapy following RT (50 Gy/25 fractions/5 weeks) for breast conservation 6 years previously. Core needle biopsy revealed leiomyosarcoma. There was no distant metastasis. Repeat BCS including part of the pectoralis major muscle was performed. Chest wall resection was not performed because of the lack of invasion. Based on the morphological and immunohistochemical features, a diagnosis of leiomyosarcoma was made. All of the resection margins in the specimen were tumor-free. She has been disease-free for over 20 months.
Conclusions:
Post-irradiation leiomyosarcoma is an extremely rare tumor with high malignant potential, and thus, multidisciplinary therapy and close follow-up are advised.