OpenMed/multicare-articles huggingface dataset:
Antibody-Mediated Extreme Insulin Resistance: The Importance of Pre-Treatment Screening
Authors: Tina Mosaferi, Sahar Sherf, Laura Y Sue, Ines Donangelo
Journal: Case Rep Endocrinol (2019)
MeSH terms: Case Reports

We report the case of a 56 year-old Hispanic male with a 10-year history of type 2 diabetes who presented with abrupt onset of hyperglycemia resistant to escalating doses of intravenous insulin infusion (>2500 units daily). He was diagnosed with antibody-mediated insulin resistance given the presence of hyperglycemia despite receiving >200 units insulin/day, a lack of identifiable precipitants for diabetic ketoacidosis or hyperosmolar hyperglycemic state, and elevated insulin antibodies. He underwent pre-immunomodulatory therapy screening for infections, rheumatologic disorders, and malignancy, which uncovered a new diagnosis of latent tuberculosis. While concurrently being treated for latent tuberculosis, he successfully responded to immunomodulatory therapy with rituximab, dexamethasone, and cyclophosphamide. Insulin was discontinued completely, and he maintained appropriate glycemic control on oral diabetic agents (metformin and pioglitazone). This case supports the use of immunomodulatory therapy for the treatment of antibody-mediated insulin resistance and highlights the importance of pre-immunomodulatory therapy screening to uncover occult infection or identify underlying neoplastic/rheumatologic disease prior to immunosuppression.
Late Lung Metastasis in a Patient with a Clear Cell Chondrosarcoma: An Indication for a Life-Long Follow-Up?
Authors: Paulien West, Celine Jacobs, Michael Saerens, David Creytens, Gwen Sys, Lore Lapeire
Journal: Case Rep Oncol Med (2021)
MeSH terms: Case Reports

Background:
Clear cell chondrosarcoma (CCCS) is a rare subtype of chondrosarcoma and comprises between 1.6% and 2.5% of all chondrosarcoma. They are known to be chemo- and radiotherapy resistant; surgical resection is therefore the therapy of choice.
Methods:
We present a 63-year-old woman with a progressive lung nodule 20 years after initial diagnosis and treatment of a clear cell chondrosarcoma of the right os naviculare.
Results:
On serial CT scans of the chest, an asymptomatic, slowly growing nodule in the left upper lung lobe was detected. CT-guided transthoracic biopsy of this nodule confirmed the diagnosis of a chondrosarcoma lung metastasis. Video-assisted thoracoscopic wedge resection was performed with complete removal of the nodule. The patient recovered well from surgery and remains in good health during further follow-up.
Conclusion:
Given the tendency of clear cell chondrosarcoma to recur and metastasize after extended periods of time, a long-term, possibly life-long follow-up and clinical surveillance is advisable in these patients.
High-Dose Ipilimumab and High-Dose Interleukin-2 for Patients With Advanced Melanoma
Authors: Ann W Silk, Howard L Kaufman, Brendan Curti, Janice M Mehnert, Kim Margolin, David McDermott, Joseph Clark, Jenna Newman, Praveen K Bommareddy, Lisa Denzin, Saltanat Najmi, Azra Haider, Weichung Shih, Michael P Kane, Andrew Zloza
Journal: Front Oncol (2020)
Keywords: antigen presentation, combination immunotherapy, cytokine, hepatitis, interleukin-2, ipilimumab, melanoma
MeSH terms: Case Reports

High-dose ipilimumab (IPI) and high-dose interleukin-2 (IL-2) are approved agents for metastatic melanoma, but the efficacy and safety of the combination are unknown. The objective of this study was to evaluate the feasibility, safety, and efficacy of combination high-dose IPI and high-dose IL-2 in patients with histologically confirmed advanced unresectable stage III and IV melanoma. This Phase II, multicenter, open-label, single-arm trial was conducted in nine patients enrolled between 12/2014 and 12/2015. Subjects were treated with high-dose IPI 10 mg/kg intravenous (IV) every 3 weeks for four doses starting at week 1 and high-dose IL-2 (600,000 IU/kg IV bolus every 8 h for up to 14 doses) concurrently with IPI at weeks 4 and 7. After the first 12 weeks of combination therapy, maintenance IPI (10 mg/kg IV) monotherapy was administered every 12 weeks for up to 1 year. No patient had received prior PD-1 blockade, and only one received prior vemurafenib. Confirmed partial response was achieved in one (11%), stable disease in four (44%), and progressive disease in four (44%) of nine patients. Two patients achieved durable disease control of 44+ and 50+ months at the most recent follow-up without subsequent therapy. The median overall survival was not reached after a minimum 24 months of follow-up time. One-year and 2-year survival rates were 89 and 67%, respectively. Seven patients (78%) experienced grade 3 or 4 adverse events related to the study therapy, three of which were attributed to both agents. One patient discontinued the treatment due to liver and kidney toxicity. While toxicity was significant, all events were reversible, and there was no treatment-related mortality. In peripheral blood of patients with decreasing tumor burden, the ratio of the non-classical MHC-II proteins HLA-DM to HLA-DO increased 2-fold, raising the possibility of the ratio of HLA-DM:HLA-DO as a novel biomarker of response to treatment. Although the sample size was limited, combination therapy with high-dose IPI and high-dose IL-2 was feasible and associated with clinical benefit. IL-2-based compounds in combination with CTLA-4 blockade should be studied in advanced melanoma patients who fail to benefit from first-line PD-1 blockade. Clinical Trial Registration: ClinicalTrials.gov, NCT02203604. Registered 30 July 2014, https://clinicaltrials.gov/ct2/show/NCT02203604.
Functional Adrenal Collision Tumor in a Patient with Cushing's Syndrome
Authors: Cathy Zhou, Ghaneh Fananapazir, Michael J Campbell
Journal: Case Rep Endocrinol (2020)
MeSH terms: Case Reports

Adrenal collision tumors are rare and produce unique diagnostic challenges for clinicians. We report the case of a 45-year-old woman with obesity and diabetes mellitus and an incidentally-discovered adrenal mass containing macroscopic fat, thought to be a myelolipoma. A functional workup confirmed adrenocorticotropic hormone- (ACTH-) independent Cushing's syndrome. The patient underwent a successful laparoscopic adrenalectomy with pathology showing an adrenal collision tumor consisting of an adrenocortical adenoma and a myelolipoma. Postoperatively, the clinical symptoms, body mass index, and hemoglobin A1C all improved. Clinicians should consider a functional workup in patients with radiographically diagnosed myelolipomas as some may prove to be hormonally active collision tumors.
Anesthetic Management of a Down Syndrome Patient for Ventricular Septal Defect Repair: A Case Report
Authors: Haneesha Movva, Karuna Taksande
Journal: Cureus (2023)
Keywords: asd, cardiac surgery, cardiopulmonary bypass, down syndrome, vsd
MeSH terms: Case Reports

It is commonly known that Down syndrome (DS) and congenital cardiovascular abnormalities go hand in hand. Most frequently, complete atrioventricular septal abnormalities have been linked to DS. Along with DS, ventricular septal defect (VSD), atrial septal defect, tetralogy of Fallot, and patent ductus arteriosus have also been reported. We present a case of DS with VSD who underwent VSD correction. Echocardiography prompted the diagnosis, which was then confirmed by surgery. The patient was successfully transferred out of the hospital. After correcting the VSD, the survival and quality of life of the DS patient have improved.
Naloxone Alleviate the Severity of Delirium in Hospitalized Patients With Parkinsonism: Three Case Reports
Authors: Haiyan Jin, Jie Zhang, Qiongyue Hu, Junjiao Ping, Tingyun Jiang, Baoguo Du, Xin Duan
Journal: Front Psychiatry (2021)
Keywords: parkinson's disease, anti-pd medications, anticholinergic drugs, delirium, naloxone, parkinsonism
MeSH terms: Case Reports

Purpose: Delirium is common in geriatric with Parkinson's disease (PD). Treatments for delirium have generally been neuroleptics; however, antipsychotics have potential effect to block striatal dopamine D2 receptors and worsen symptom of parkinsonism. We explored whether naloxone can alleviate delirium in PD and other forms of parkinsonism. Patients and Methods: Patients with parkinsonism who met the delirium criteria of the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM-5) received naloxone infusions once or twice daily. Treatment effects were evaluated by the delirium rating scale-revised 98 (DRS-R98), including non-cognitive and cognitive subscales; the Richmond agitation-sedation scale (RASS); and the mini mental status examination (MMSE). Results: Two patients with primary parkinsonism, one with vascular PD were observed. The daily dose of naloxone was 2.08 ± 0.64 mg (range: 1-4 mg). Medication time last from 1 h to 7 days without side effects observed. Following with naloxone infusions, DRS-R98 scores decreased within 12 h and MMSE scores increased. The psychotic symptoms, disorientation, and attention deficits were alleviated significantly, while RASS scores decreased with naloxone treatment. Conclusion: Naloxone alleviated psychotic symptoms, improved cognitive dysfunction, and irritability in patients with delirium in the context of PD. The preliminary findings point out that the opioid system may be involved in the pathophysiology of delirium, which may be one of potential treat targets for delirium of PD.
Searching for the Mechanical Fingerprint of Pre-diabetes in T1DM: A Case Report Study
Authors: Flavio Di Giacinto, Linda Tartaglione, Matteo Nardini, Alberto Mazzini, Sabrina Romanò, Gaetano Emanuele Rizzo, Massimiliano Papi, Marco De Spirito, Dario Pitocco, Gabriele Ciasca
Journal: Front Bioeng Biotechnol (2020)
Keywords: afm, biomechanics, cell mechanics, diabetes, pre-type 1 diabetes, red blood cells, standard linear solid model, viscoelasticity
MeSH terms: Case Reports

We report the case of a 38 year-old Caucasian man enrolled in a study aimed at investigating the physical properties of red blood cells (RBCs) using advanced microscopy techniques, including Atomic Force Microscopy (AFM). At the time of his first enrolment in the study, he had normal Fasting Plasma Glucose (FPG) values, a BMI of 24.1, and no other symptoms of diabetes, including fatigue, high triglycerides, low HDL cholesterol, and altered inflammatory and corpuscular RBC indices. The subject reported no family history of diabetes, obesity, and cardiovascular diseases. Despite his apparently healthy conditions, the biomechanics of his RBCs was altered, showing increased values of stiffness and viscosity. More than 1 year after the mechanical measurements, the subject was admitted to the Operational Unit of Diabetology of the Policlinico Gemelli Hospital with high blood glucose and glycosylated hemoglobin (HbA1c) levels and diagnosed with type 1 diabetes (T1DM). Here, we show these data, and we discuss the hypothesis that RBC mechanical properties could be sensitive to changes occurring during the pre-diabetic phase of T1DM.
Reidel's Thyroiditis, a Diagnostic and Management Challenge: A Case Report and Review of the Literature
Authors: Yassine Er-Rahali, Mohammed Massine El Hammoumi, Jad Issouani, Colna Antonio Nfad, Souad El Moussaoui, El Hassane Kabiri, Ahmed Anass Guerboub
Journal: Case Rep Endocrinol (2021)
MeSH terms: Case Reports

Riedel's thyroiditis is a very rare inflammatory condition. It affects not only the thyroid gland but also the adjacent vital structures. It may also be associated with different forms of systemic fibrotic disorders. The exact etiology is unknown, but currently, the most favorable opinion is that it is a localized form of the systemic fibrotic process. We report the case of a 38-year-old woman, presented with a 10-month history of progressive hypothyroidism, dysphonia, and dysphagia. A Doppler ultrasound study revealed massive thyroid enlargement with multiple Eu TIRADS 3 and 4 nodules. Fine needle aspiration was noncontributive on two occasions. A hard subtotal thyroidectomy was performed. Pathological study confirmed Riedel's thyroiditis with the presence of IgG4 antibodies in immunohistochemistry. The patient was successfully treated with levothyroxine replacement and corticosteroid therapy with rapid resolution of obstructive symptoms. The case descriptions highlight the diagnostic challenge of this disease, describe the response to surgical management and corticosteroid therapy, and give a short review of the subject.
Lipschütz Genital Ulceration as Initial Manifestation of Primary Sjögren's Syndrome
Authors: Filipa de Castro Coelho, Maria Amaral, Lúcia Correia, Maria João Nunes Campos, Tereza Paula, Augusta Borges, Jorge Borrego
Journal: Case Rep Obstet Gynecol (2018)
MeSH terms: Case Reports

Genital ulcers are challenging to any clinician and causes transcend many specialties. Skin ulceration in patients with primary Sjögren's syndrome is infrequent but an established feature of cutaneous involvement. Although gynecological symptoms, such as vulvovaginal dryness, dyspareunia, and pruritus, are common in women with primary Sjögren's syndrome, patients affected by vulvar ulcers are unknown. We describe an exceptional case of necrotic aphthous-type vulvar ulceration as initial presentation of primary Sjögren's syndrome that was possibly triggered by an infectious agent. Successful healing was achieved with oral corticosteroids, despite some loss of labia minora and labia majora as sequelae of the necrotizing process. Reactive acute genital ulcers (Lipschütz ulcers) should be considered as a possible manifestation of many autoimmune/inflammatory disorders, beyond the classic associations such as Behçet's syndrome or Crohn's disease.
Costimulation pathway blockade in kidney transplant recipients with de-novo rheumatoid arthritis
Authors: Mohamed Sheta, Samy Riad, Udayakumar Deepak, Naim Issa, Mark Birkenbach, Hassan N Ibrahim, Aleksandra Kukla
Journal: Clin Nephrol Case Stud (2017)
Keywords: belatacept, kidney transplant, membranous nephropathy, rheumatoid arthritis
MeSH terms: Case Reports

The best approach to treatment of de-novo rheumatoid arthritis in solid organ transplant recipients on typical immunosuppression is not well established. The use of biologics targeting specific cell types, cytokines, and immunological pathways has been gaining interest in the treatment of both, auto- and alloimmunity. We present a case of de-novo rheumatoid arthritis in a kidney transplant recipient 10 years post-transplant while receiving cyclosporine, mycophenolate mofetil, and also prednisone. Initial presentation included features of polymyalgia rheumatica and nephrotic range proteinuria. Kidney biopsy showed membranous nephropathy. The patient was initially treated with methotrexate, while mycophenolate mofetil was discontinued. Clinical symptoms improved, but creatinine significantly increased, which led to discontinuation of methotrexate and mycophenolate mofetil was restarted. The kidney function improved, but the patient experienced a flare of rheumatoid arthritis. Costimulatory blocker, abatacept, was initiated and cyclosporine was gradually tapered off. Graft function remained stable for a follow-up period of 7 years. Joint pain, weakness, and stiffness resolved. Follow-up plain film radiographs at 5 years post initial presentation showed no new joint erosions in hands or feet. Costimulatory blockers may broaden the therapeutic choices of transplant recipients with de-novo autoimmune diseases.
Melkersson-Rosenthal syndrome in a patient with tubercular panuveitis
Authors: Kalpana Babu, Prashanta V Gundannavar, Vidya Satish, Venkatesh C Prabhakaran
Journal: Indian J Ophthalmol (2010)
MeSH terms: Melkersson-Rosenthal Syndrome / etiology, Panuveitis / complications, Tuberculosis, Ocular / complications, Case Reports, Antitubercular Agents / therapeutic use, Biopsy, Diagnosis, Differential, Humans, Male, Melkersson-Rosenthal Syndrome / diagnosis, Middle Aged, Panuveitis / diagnosis, Panuveitis / drug therapy, Tomography, X-Ray Computed, Tuberculosis, Ocular / diagnosis, Tuberculosis, Ocular / drug therapy, Antitubercular Agents

We report a rare presentation of Melkersson-Rosenthal syndrome in a patient with tubercular panuveitis. A 45-year-old male being treated with antitubercular therapy for tubercular panuveitis presented with unilateral, non-pitting right upper eyelid edema. Excision biopsy showed granulomatous inflammation involving the lymphatics. Immunohistochemistry confirmed the presence of histiocytes around the lymphatics.
Fournier's gangrene after adult male circumcision
Authors: Moses Galukande, Dennis Bbaale Sekavuga, Alex Muganzi, Alex Coutinho
Journal: Int J Emerg Med (2014)
Keywords: adult, circumcision, gangrene, male

Background:
In the advent of mass voluntary medical male circumcision (VMMC) for the partial prevention of HIV, previously rare adverse events associated with adult male circumcision are likely to be encountered with higher frequency. Fournier's gangrene, defined as a polymicrobial necrotizing fasciitis of the perineal, perianal or genital areas, is one such rare and life-threatening adverse event. In this report, we present two cases that were identified in the context of a VMMC programme over a 3-year period during which approximately 100,000 adult circumcisions were performed.
Case presentations:
Case 1: A 19-year-old male who had VMMC performed using the dorsal slit technique developed pain and blisters on the scrotal skin on the sixth postoperative day. He had no co-morbidities, and serology for HIV was negative. On examination, locally he had scrotal skin necrosis with an offensive odour and was dehydrated but afebrile. Repeated aggressive debridement was done while he stayed in a hospital for 3 weeks; at which point, he had healthy granulation tissue and was free of infection. The wound had closed spontaneously and completely by the fifth month. Case 2: A 52-year-old male who had VMMC performed with the sleeve resection method developed pain and swelling of the penis and scrotum on the fourth postoperative day. He had a low-grade fever of 37.6°C. He was not diabetic or immunosuppressed and had a negative HIV serology. He was admitted and was given IV antibiotics, and repeated aggressive debridement was performed. On the third week of hospitalization, he had healthy granulation tissue and received a split skin graft on the penile shaft. At 4 months, the scrotal defect had completely closed.
Conclusion:
Fournier's gangrene is a rare occurrence after adult male circumcision with associated high morbidity. These are the first descriptions in the VMMC era.
Case Report: Combined Liver-Kidney Transplantation to Correct a Mutation in Complement Factor B in an Atypical Hemolytic Uremic Syndrome Patient
Authors: Margarita López-Trascasa, Ángel Alonso-Melgar, Marta Melgosa-Hijosa, Laura Espinosa-Román, María Dolores Lledín-Barbancho, Eugenia García-Fernández, Santiago Rodríguez de Córdoba, Pilar Sánchez-Corral
Journal: Front Immunol (2021)
Keywords: atypical hemolytic uremic syndrome, combined liver-kidney transplantation, complement, factor b, rare diseases
MeSH terms: Atypical Hemolytic Uremic Syndrome / diagnosis, Atypical Hemolytic Uremic Syndrome / genetics, Atypical Hemolytic Uremic Syndrome / therapy, Complement Factor B / genetics, Kidney Transplantation, Liver Transplantation, Case Reports, Research Support, Non-U.S. Gov't, Gain of Function Mutation, Humans, Infant, Male, Complement Factor B

Pathogenic gain-of-function variants in complement Factor B were identified as causative of atypical Hemolytic Uremic syndrome (aHUS) in 2007. These mutations generate a reduction on the plasma levels of complement C3. A four-month-old boy was diagnosed with hypocomplementemic aHUS in May 2000, and he suffered seven recurrences during the following three years. He developed a severe hypertension which required 6 anti-hypertensive drugs and presented acrocyanosis and several confusional episodes. Plasma infusion or exchange, and immunosuppressive treatments did not improve the clinical evolution, and the patient developed end-stage renal disease at the age of 3 years. Hypertension and vascular symptoms persisted while he was on peritoneal dialysis or hemodialysis, as well as after bilateral nephrectomy. C3 levels remained low, while C4 levels were normal. In 2005, a heterozygous gain-of-function mutation in Factor B (K323E) was found. A combined liver and kidney transplantation (CLKT) was performed in March 2009, since there was not any therapy for complement inhibition in these patients. Kidney and liver functions normalized in the first two weeks, and the C3/C4 ratio immediately after transplantation, indicating that the C3 activation has been corrected. After remaining stable for 4 years, the patient suffered a B-cell non-Hodgkin lymphoma that was cured by chemotherapy and reduction of immunosuppressive drugs. Signs of liver rejection with cholangitis were observed a few months later, and a second liver graft was done 11 years after the CLKT. One year later, the patient maintains normal kidney and liver functions, also C3 and C4 levels are within the normal range. The 12-year follow-up of the patient reveals that, in spite of severe complications, CLKT was an acceptable therapeutic option for this aHUS patient.
Comparison of different final impression techniques for management of resorbed mandibular ridge: a case report
Authors: Bhupender Yadav, Manisha Jayna, Harish Yadav, Shrey Suri, Shefali Phogat, Reshu Madan
Journal: Case Rep Dent (2014)

The history of complete denture impression procedures has been influenced largely by the development of impression materials from which new techniques and ideas arose. The purpose of this study was to compare the retention of complete dentures made by using different impression techniques like conventional, admixed, all green, and functional techniques. The results showed that there was significant difference in retention between the six techniques where functional technique showed the highest mean value of retention followed by elastomeric, all green, and admixed, while cocktail and green stick compound showed the lowest mean value. However, on clinical examination, the retention produced by the six techniques was satisfactory.
Vulvar merkel carcinoma: a case report
Authors: C Iavazzo, M Terzi, P Arapantoni-Dadioti, V Dertimas, G Vorgias
Journal: Case Rep Med (2011)
MeSH terms: Case Reports

This is a new case of Merkel cell carcinoma of the vulva. It is a rare neuroendocrine carcinoma with an aggressive behavior. Because of its rarity in this location, it is not clear whether it behaves differently from the usual neuroendocrine carcinomas of the skin. A case of a 63-year-old patient with vulvar Merkel carcinoma is presented. The clinical presentation, microscopic and immunohistochemical features, and treatment are discussed.
Inferior Dislocation of Shoulder Complicated with Undisplaced Greater Tuberosity Fracture, Rupture of the Supraspinatus Tendon, and Brachial Plexus Injury in the Elderly: Case Report and Literature Review
Authors: Angelo V Vasiliadis, Christos Kalitsis, Theofanis Kantas, George Biniaris
Journal: Case Rep Orthop (2020)
MeSH terms: Case Reports

Inferior dislocation of the shoulder, also called luxatio erecta, is a rare form of the otherwise common shoulder dislocation. It appears in less than 0.5% of all shoulder dislocations. An awareness of associated potential axillary artery injury, brachial plexus complications, and rotator cuff tears is important in this rare entity and should be excluded with a high index of suspicion. In our case report, we have an 83-year-old female who inferiorly dislocated her dominant shoulder with brachial plexus injury and musculotendinous injury, which was caused by an accidental fall. The dislocation was manually reduced at the emergency department. After 18 months of conservative treatment with physical therapy, the range of motion and muscle strength of the shoulder recovered to a satisfactory mobile level according to the patient's demands.