OpenMed/multicare-articles huggingface dataset:
Visceral leishmaniasis with endobronchial involvement in an immunocompetent adult
Authors: Konstantinos Kotsifas, Eugenios Metaxas, Ioannis Koutsouvelis, Athanassios Skoutelis, Panayiota Kara, George Tatsis
Journal: Case Rep Med (2011)
MeSH terms: Case Reports

Visceral leishmaniasis is characterized by fever, cachexia, hepatosplenomegaly, pancytopenia, and hypergammaglobulinemia. Cough may be a presenting symptom as well. However, pulmonary involvement is considered rare and mainly described in immunocompromised patients. We describe a case of an immunocompetent adult whose clinical presentation was dominated by cough and hemoptysis. Bronchoscopy revealed a discreet polypoid mucosal endobronchial lesion whose biopsy yielded Leishmania amastigotes within histiocytes. Transbronchial needle biopsy of a right paratracheal lymph node was also positive. Leishmania amastigotes were also found on bone marrow and liver biopsies. Treatment with IV Amphotericin B was successful. In conclusion, cough should not be overlooked as a presenting symptom of visceral leishmaniasis and may be a sign of pulmonary involvement.
Tuberculosis Presenting as Isolated Wrist Swelling: A Case Report and Review of Literature
Authors: Oshan Basnayake, Ahamed Nihaj, Ranji Pitagampalage, Harsha Mendis
Journal: Case Rep Surg (2019)
MeSH terms: Case Reports

Background:
Tuberculosis is a common disease entity in South East Asian countries with a significant global burden. An extra skeletal manifestation such as monoarticular TB is common, but isolated involvement of the wrist is rare.
Case summary:
A 53-year-old Sri Lankan male with long-standing diabetes presented with an isolated hand swelling for a 7-month duration. His initial imaging and MRI showed multiple destructive lesions in the carpal bones, surrounding focal fluid collections and found to have caseous material intraoperatively. His histology and microbiological studies were positive for TB with no other concurrent evidence of TB elsewhere.
Conclusion:
Different presentations of tuberculosis should be considered when patients are presenting with atypical clinical and initial basic investigation findings in relation to monoarticular pathologies.
Splenic B-Cell Lymphomas with Diffuse Cyclin D1 Protein Expression and Increased Prolymphocytic Cells: A Previously Unrecognized Diagnostic Pitfall
Authors: Khaled Algashaamy, Yaohong Tan, Nicolas Mackrides, Alvaro Alencar, Jing-Hong Peng, Joseph Rosenblatt, Juan P Alderuccio, Izidore S Lossos, Francisco Vega, Jennifer Chapman
Journal: Case Rep Hematol (2018)
MeSH terms: Case Reports

Prolymphocytic transformation is a concept usually applied in the context of chronic lymphocytic leukemia/small lymphocytic lymphoma to describe the presence of a high percentage of prolymphocytes in peripheral blood (usually more than 55%). Prolymphocytic transformation has also been reported in mantle cell lymphoma (MCL) but only rarely in splenic marginal zone lymphoma (SMZL). We present two splenic B-cell lymphomas presenting in the leukemic phase and with increased prolymphocytes, both classified as SMZL with prolymphocytic transformation. One case clinically simulated B-prolymphocytic leukemia (B-PLL). Both lymphomas were very unusual because the tumor cells diffusely and strongly expressed cyclin D1 despite lacking the t(11; 14)(q13; q32) as detected by several approaches including next-generation sequencing, fluorescence in situ hybridization using CCND1 break apart probe and fusion probes for t(11; 14)(q13; q32), and conventional karyotyping. These cases therefore simulated prolymphocytic variants of MCL. The incidence of this phenomenon is unknown, and awareness of this potential alternate protein expression pattern is important in order to avoid diagnostic errors.
PRaG Therapy of Refractory Metastatic Gastric Cancer: A Case Report
Authors: Hong Xu, Zhihui Hong, Meiling Xu, Yuehong Kong, Yifu Ma, Chanchan Shan, Pengfei Xing, Liyuan Zhang
Journal: Front Immunol (2022)
Keywords: gm-csf, case report, gastric cancer, immunotherapy, pmmr, radiotherapy
MeSH terms: Adenocarcinoma / drug therapy, Antineoplastic Agents / therapeutic use, Stomach Neoplasms / drug therapy, Case Reports, Research Support, Non-U.S. Gov't, DNA Mismatch Repair, Humans, Prognosis, Antineoplastic Agents

Patients with metastatic gastric cancer had limited treatments and often had a somber prognosis, especially when patients were unable to tolerate high-intensity cytotoxic treatment due to poor physical condition or organ dysfunction after the failure of standard therapy. Here, we reported a metastatic and proficient mismatch repair (pMMR) gastric adenocarcinoma patient with the Eastern Cooperative Oncology Group (ECOG) performance status score of 2 associated with hypoproteinemia and fatigue, and poor appetite that was unable to tolerate high-intensity therapy after several chemotherapy regimens and anti-angiogenic therapy. After receiving novel triple-combination therapy, which consists of PD-1 inhibitor, Radiotherapy and Granulocyte-macrophage colony-stimulating factor (GM-CSF) therapy (PRaG for short), the patient achieved a complete response (CR) with a progression-free survival time of 14 months, and ECOG performance status score improved from 2 to 0. A significant systemic effect was observed in this case and the PRaG triple-combination therapy might provide a novel treatment strategy for metastatic pMMR gastric cancer patients.
Tuberculosis presenting as immune thrombocytopenic purpura
Authors: Fahir Ozkalemkas, Ridvan Ali, Atilla Ozkan, Tulay Ozcelik, Vildan Ozkocaman, Esra Kunt-Uzaslan, Beril Bahadir-Erdogan, Halis Akalin
Journal: Ann Clin Microbiol Antimicrob (2004)

Background:
Although various hematologic abnormalities are seen in tuberculosis, immune thrombocytopenic purpura is a rare event.
Case presentation:
We report a case of a 29 year-old male who was presented with immune thrombocytopenia-induced hemoptysis, macroscopic hematuria and generalized petechiae. The patient was found to have clinical, microbiological and radiological evidence of active pulmonary tuberculosis. The immune thrombocytopenic purpura was successfully treated with anti-tuberculous drugs combined with corticosteroids and high dose immune globulin therapy.
Conclusion:
Immune thrombocytopenic purpura can be one of the hematological manifestations of tuberculosis which has a global prevalence with increasing incidence secondary to HIV infection.
A single coronary artery with left circumflex artery crossing right ventricular outflow tract in tetralogy of Fallot with absent left pulmonary artery
Authors: Vivek Jaswal, Shyam Kumar Singh Thingnam, Vikas Kumar, Ruchit Patel, Ganesh Kumar Munirathinam, Dheemta Toshkhani
Journal: J Cardiovasc Thorac Res (2021)
Keywords: absent left pulmonary artery, left circumflex artery, right ventricular outflow tract, single coronary artery, tetralogy of fallot
MeSH terms: Case Reports

Tetralogy of Fallot (TOF) with unilateral absence of pulmonary artery and the anomalous coronary artery is a rare combination. Detailed preoperative evaluation of coronary artery anatomy is must to prevent injury to the major vessels crossing right ventricular outflow tract. We report a rare association of single coronary artery with left circumflex artery crossing right ventricular outflow tract close to the pulmonary annulus in tetralogy of Fallot with absent left pulmonary artery in 11-year-old girl. Though there is a great diversity of coronary anomalies in tetralogy of Fallot, the prepulmonic course of left circumflex artery crossing the right ventricular outflow tract (RVOT) close to the pulmonary annulus has rarely been described in the literature. The patient underwent successful primary single lung intracardiac repair. Right ventricular outflow tract obstruction was treated by handmade valved pericardial autologous conduit and release of the tethering of hypoplastic native unicuspid pulmonary valve leaflet maintaining its integrity.
Overinterpretation and Overtreatment of Low-Titer Antibodies Against Contactin-Associated Protein-2
Authors: Christian G Bien
Journal: Front Immunol (2018)
Keywords: cell-based assays, contactin-associated protein-2 antibodies, depression, diagnostic specificity, immunotherapy, neural antibodies
MeSH terms: Autoantibodies / immunology, Autoantigens / immunology, Autoimmune Diseases of the Nervous System / diagnosis, Autoimmune Diseases of the Nervous System / immunology, Encephalitis / diagnosis, Encephalitis / immunology, Membrane Proteins / immunology, Nerve Tissue Proteins / immunology, Case Reports, Adult, Autoimmune Diseases of the Nervous System / drug therapy, Biomarkers, Electroencephalography, Encephalitis / drug therapy, Female, Humans, Membrane Proteins / antagonists & inhibitors, Nerve Tissue Proteins / antagonists & inhibitors, Autoantibodies, Autoantigens, Biomarkers, CNTNAP2 protein, human, Membrane Proteins, Nerve Tissue Proteins

Antibodies (abs) against neural or glial antigens have become important diagnostic markers of autoimmune encephalitides. A key requirement for interpretation of any test in clinical medicine is specificity. In this work, a 35-year-old female patient with low-titer contactin-associated protein-2 abs not satisfying clinical criteria of autoimmune encephalitis is reported. The patient had a recurrent depressive disorder and, at the time of the ab study, a moderate depressive episode. Overinterpretation and misinterpretation of patient's complaints and paraclinical study results fueled the idea of an autoimmune encephalitis. It is suggested to check patients with supposedly positive ab test results critically for clinical criteria, titer cutoffs, and ab-typical epidemiological features like age and sex.
Idiopathic Bilateral Suprachoroidal Haemorrhage: A Rare Case Presentation
Authors: Komal Saluja, Mayuresh Naik, Rajshekhar Vemparala, Anuj Mehta
Journal: Case Rep Ophthalmol Med (2017)
MeSH terms: Case Reports

55-year-old male presented with sudden onset painful diminution of vision in both eyes. On local examination, his visual acuity was FC at 2 metres in right eye and FC at 1 m in left eye. The IOP in right eye was 46 mm Hg and 44 mm Hg in left eye. The patient was admitted and started on injection mannitol, oral syrup glycerol, and oral acetazolamide. Locally, timolol maleate and brimonidine were also started. The next day, his IOP was 17 mm Hg bilaterally but his visual acuity deteriorated to FC 1 m in right eye and hand movement in left eye with inaccurate projection of rays in both eyes. USG B-scan was performed which revealed bilateral choroidal detachment. The echotexture of fluid was suggestive of haemorrhage. As the IOP was controlled, systemic hyperosmotic/antiglaucoma agents were withdrawn in stepwise fashion over next two days. The patient was started on oral prednisolone. At 2 weeks, the visual acuity in both eyes was only perception of light, with inaccurate PR. IOP was 10 mm Hg in both eyes. USG B-scan revealed resorption of the hemorrhage, with partial resolution of the choroidal detachment. The final BCVA was 6/18 and 6/12 in right and left eye.
Acute respiratory failure as presentation of late-onset Pompe disease complicating the diagnostic process as a labyrinth: a case report
Authors: Francesco Menzella, Luca Codeluppi, Mirco Lusuardi, Carla Galeone, Franco Valzania, Nicola Facciolongo
Journal: Multidiscip Respir Med (2018)
Keywords: enzyme, glycogen, muscle biopsy, respiratory failure, ventilation
MeSH terms: Case Reports

Background:
Acute respiratory failure can be triggered by several causes, either of pulmonary or extra-pulmonary origin. Pompe disease, or type II glycogen storage disease, is a serious and often fatal disorder, due to a pathological accumulation of glycogen caused by a defective activiy of acid α-glucosidase (acid maltase), a lysosomal enzyme involved in glycogen degradation. The prevalence of the disease is estimated between 1 in 40,000 to 1 in 300,000 subjects.
Case presentation:
This case report describes a difficult diagnosis of late-onset Pompe disease (LOPD) in a 52 year old Caucasian woman with acute respiratory failure requiring orotracheal intubation and subsequent tracheostomy for long-term mechanical ventilation 24 h/day. Despite a complex diagnostic process including several blood tests, bronchoscopy with BAL, chest CT, brain NMR, electromyographies, only a muscle biopsy allowed to reach the correct diagnosis.
Discussion:
The most frequent presentation of myopathies, including LOPD, is proximal limb muscle weakness. Respiratory related symptoms (dyspnea on effort, reduced physical capacity, recurrent infections, etc.) and respiratory failure are often evident in the later stages of the diseases, but they have been rarely described as the onset symptoms in LOPD. In our case, a third stage LOPD, the cooperation between pulmonologists and neurologists was crucial in reaching a correct diagnosis despite a very complex clinical scenario due to different confounding co-morbidities as potential causes of respiratory failure and an atypical presentation. In this patient, enzyme replacement therapy with infusion of alglucosidase alfa was associated with progressive reduction of ventilatory support to night hours, and recovery of autonomous walking.
CT and MR Unilateral Brain Features Secondary to Nonketotic Hyperglycemia Presenting as Hemichorea-Hemiballism
Authors: Víctor Manuel Suárez-Vega, Carlos Sánchez Almaraz, Ana Isabel Bernardo, Ricardo Rodríguez-Díaz, Ana Díez Barrio, Leticia Martín Gil
Journal: Case Rep Radiol (2016)

Hemichorea-hemiballism is an unusual hyperkinetic movement disorder characterized by continuous involuntary movements of an entire limb or both limbs on one side of the body. The acute onset of this disorder occurs with an insult in contralateral basal ganglia. Ischemic events represent the most common cause. Nonketotic hyperglycemia comes in second place. Nonketotic hyperglycemic hemichorea-hemiballism (NHH) is a rare cause of unilateral brain abnormalities on imaging studies confined to basal ganglia (mainly putaminal region as well as caudate nucleus). Subtle hyperdensity in striatal region can be found on CT studies whereas brain MR imaging typically shows T1 hyperintensity and T2 hypointensity in the basal ganglia contralateral to the movements. Diagnosis is based on both glucose levels and neuroimaging findings. Elevated blood glucose and hemoglobin A1c levels occur with poorly controlled diabetes. In this case report, our aim is to present neuroimaging CT and MR unilateral findings in an elderly woman secondary to nonketotic hyperglycemia presenting as hemichorea-hemiballism.
Concurrent colonic mucosa-associated lymphoid tissue lymphoma and adenoma diagnosed after a positive fecal occult blood test: a case report
Authors: Pei-Chiang Lin, Jinn-Shiun Chen, Po Deng, Chih-Wei Wang, Chiung-Huei Huang, Reiping Tang, Jy-Ming Chiang, Chien-Yuh Yeh, Pao-Shiu Hsieh, Wen-Sy Tsai, Sum-Fu Chiang
Journal: J Med Case Rep (2016)
MeSH terms: Adenoma / diagnosis, Colonic Neoplasms / diagnosis, Lymphoma, B-Cell, Marginal Zone / diagnosis, Neoplasms, Multiple Primary / diagnosis, Occult Blood, Case Reports, Adenoma / surgery, Colonic Neoplasms / surgery, Colonoscopy, Humans, Intestinal Mucosa / surgery, Lymphoma, B-Cell, Marginal Zone / surgery, Male, Middle Aged, Neoplasms, Multiple Primary / surgery

Background:
Colonic lymphoma is an uncommon presentation of extranodal lymphoma. Colonic mucosa-associated lymphoid tissue lymphoma is a different entity from gastric mucosa-associated lymphoid tissue lymphoma, and very rare. The presentation and management of colonic mucosa-associated lymphoid tissue are highly variable in the literature.
Case presentation:
We report the case of a 59-year-old Taiwanese man who underwent a colonoscopy after a positive test for fecal occult blood. His past history included hypertension and hyperthyroidism. The colonoscopy revealed an adenomatous polyp and mucosa-associated lymphoid tissue lymphoma. We successfully performed a polypectomy and endoscopic mucosal resection. The lymphoma was staged according to the Ann Arbor system modified by Musshoff as E-I. Our patient showed no lymphoma recurrence over a 3-year follow-up.
Conclusions:
Endoscopic mucosal resection for colonic mucosa-associated lymphoid tissue lymphoma without disseminated disease may be feasible. We successfully used colonoscopic treatment without adjuvant therapy to treat early-stage pathogen-free colonic mucosa-associated lymphoid tissue lymphoma.
Bilateral Postoperative Cyst after Maxillary Sinus Surgery: Report of a Case and Systematic Review of the Literature
Authors: Boris-Mark Niederquell, Peter A Brennan, Michael Dau, Maximilian Moergel, Bernhard Frerich, Peer Wolfgang Kämmerer
Journal: Case Rep Dent (2016)

Purpose. We present a case of a bilateral postoperative maxillary cyst (PMC) and discuss this with a systemic review. Case Report and Literature Review. A 68-year-old female with pain and swelling on the right side of the face. MRI and CT showed a cystic tumors of the right and left maxillary sinus. Radical maxillary surgery via a Caldwell-Luc procedure had been performed 55 years ago and bilateral PMC was diagnosed. The PubMed database was searched for PMC within the last 30 years. Results. Together with the current case, we found 23 reports including 284 patients describing PMC. It was diagnosed at a mean time of 22 years after causal surgery at a mean age of 47 years. Initial symptoms were mostly pain with or without swelling. The main radiological sign was a unilocular radiolucency with a slight preference for the left side. Discussion. PMC is a long-term complication that can occur after maxillary sinus surgery and a second surgical approach is required in order to stop cystic expansion. Therefore, patients' informed consent on this complication as well as a prolonged follow-up is recommended. Simple paranasal ultrasound or paranasal sinus plain radiography may lead to an earlier detection reducing interventional morbidity.
Autoimmune Diseases in Patients with Cushing's Syndrome after Resolution of Hypercortisolism: Case Reports and Literature Review
Authors: Luigi Petramala, Federica Olmati, Maria Gabriella Conforti, Antonio Concistré, Valeria Bisogni, Nikita Alfieri, Gino Iannucci, Giorgio de Toma, Claudio Letizia
Journal: Int J Endocrinol (2018)

Introduction:
Cushing's syndrome (CS) is a clinical condition characterized by excessive cortisol production, associated with metabolic complications, such as diabetes mellitus, dyslipidemia, metabolic syndrome, hypertension, and cardiovascular diseases. Nowadays, the occurrence of autoimmune diseases in CS have not been completely evaluated in the previous studies.
Objective:
The aim of this study was to evaluate the occurrence of autoimmune diseases in CS patients after successfully treated.
Materials and methods:
From January 2001 to December 2017, in our Secondary Hypertension Unit, we evaluated 147 CS patients (91 with ACTH-independent disease, 54 with ACTH-dependent disease, and 2 patients with ectopic ACTH production.
Results:
109 CS patients (74.1%) were surgically treated (67 ACTH-independent CS patients (61.5%) undergone adrenalectomy and 42 ACTH-dependent CS (38.5%) undergone transsphenoidal surgery) and evaluated after 6, 12, and 24 months after clinical and biochemical remission of disease. In 9 (8.3%) of overall treated CS patients (8.3%), during follow-up, we observed the onset of some manifestations of autoimmune diseases. In particular, one patient had a systemic lupus erythematosus, one patient had rheumatoid arthritis, 4 patients reported autoimmune thyroiditis (Basedow-Graves' disease and Hashimoto's thyroiditis), one patient had clinical features of psoriasis, one patient showed myasthenia gravis, and one patient had giant cell arteritis.
Conclusions:
Our results demonstrate that patients successfully treated for CS could develop autoimmune diseases. Therefore, after treatment, CS patients need to be strictly monitored in order to evaluate the possible onset of autoimmune diseases.
Laparoscopic plug removal for chronic pain after inguinal hernia repair using the plug-and-patch technique: A case report
Authors: Tatsuya Tazaki, Masaru Sasaki, Mohei Kohyama, Yoichi Sugiyama, Ryuta Shintakuya, Toshinori Hirano, Shinya Takahashi, Atsushi Nakamitsu
Journal: Int J Surg Case Rep (2019)
Keywords: case report, chronic pain, inguinal hernia, postoperative pain

Introduction:
The use of tension-free mesh repair techniques for inguinal hernias has led to uniformly low recurrence rates. The main associated morbidity is chronic postoperative inguinal pain. Mesh removal and triple neurectomy is the indicated procedure; there is insufficient evidence to support mesh removal alone without neurectomy in patients with chronic postoperative inguinal pain.
Presentation of case:
A 76-year-old man previously underwent repair of a right inguinal direct hernia using the plug-and-patch technique. Two years later, he experienced groin pain requiring the use of pain medication. Five years after surgery, he expressed the desire to remove the mesh because of chronic pain, rated 8 out of 10 on a numeric rating scale. We suspected that he was experiencing nociceptive pain caused by a plug meshoma, so we performed a laparoscopic plug extraction. His inguinal pain improved to 2 out of 10 on the second postoperative day, and he stopped taking pain medication by 10 months after surgery.
Discussion:
The laparoscopic approach to plug removal is safe and simple. We successfully avoided causing new-onset pain by not using a groin incision to remove the mesh plug.
Conclusion:
Laparoscopic plug removal for nociceptive pain due to a plug meshoma is effective. However, since there is insufficient evidence to recommend mesh removal without triple neurectomy, informed consent and further consideration of techniques and diagnostic methods are needed.
Pediatric Anti-N-methyl-D-aspartate Receptor Encephalitis Mimicking Glutaric Aciduria Type 1: A Case Report
Authors: Daniel Almeida do Valle, Mara Lúcia Schmitz Ferreira Santos, Michelle Silva Zeny, Mara L Cordeiro
Journal: Front Neurol (2020)
Keywords: anti-n-methyl-d-aspartate receptor encephalitis, antibody, child, dystonia, glutaric acidemia i
MeSH terms: Case Reports

Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is an immune-mediated disease that induces a wide spectrum of symptoms, especially in toddlers. These include acute-onset movement disorders, with neurological regression, and other associated neurological symptoms. Anti-NMDAR encephalitis remains a diagnostic challenge, especially in toddlers, with better prognosis associated with early treatment. We report the case of a 15-months-old boy who initially presented with vomiting and later with acute-onset dystonia after the administration of antiemetics. Within 14 days, the patient developed neuropsychomotor developmental regression and worsening dystonia. After ruling out an acute dystonic reaction and glutaric acidemia type 1 (GA-1), a final diagnosis of anti-NMDAR encephalitis was made. The patient responded well to immunomodulatory therapy. The present case underscores the importance of early treatment for patient prognosis and of including anti-NMDAR encephalitis in the differential diagnosis of acute-onset movement disorders.