OpenMed/multicare-articles huggingface dataset:
Subcutaneous glucagon infusion and continuous glucose monitoring enable effective management of hypoglycemia in a patient with IGF-2-producing hemangiopericytoma
Authors: Eric D Buras, Emily Weatherup, Jennifer Wyckoff
Journal: Clin Diabetes Endocrinol (2018)
Keywords: continuous glucose monitoring, glucagon, hemangiopericytoma, insulin like growth factor (igf)-2
MeSH terms: Case Reports

Background:
Ectopic insulin-like growth factor (IGF)-2 production is a rare complication of an array of epithelial and mesenchymal tumors, and can clinically manifest as life-threatening hypoglycemia.
Case presentation:
A 49-year-old woman with 13-year history of metastatic hemangiopericytoma, previously treated with multiple rounds of chemotherapy and palliative radiation, presented to the emergency department after a hypoglycemic seizure. On arrival, glucose was 18 mg/dL (1.0 mmol/L) and required continuous dextrose infusion for maintenance within normal limits. Insulin was <2.0 μU/mL, C-peptide 0.1 ng/mL, and beta-hydroxybutyrate <0.2 mmol/L. Random cortisol was 21 μg/dL; sulfonylurea screen, and insulin antibodies were negative. IGF-2 level was 1320 ng/mL; IGF-1 was within normal limits and IGF binding protein (BP)-3 suppressed. Dexamethasone, started at 6 mg twice daily, allowed discontinuation of the glucose infusion. Given concern for nocturnal hypoglycemia, and patient interest in steroid-sparing anti-hypoglycemic regimen, she was also started on overnight continuous subcutaneous glucagon infusion via insulin pump. She was discharged with instructions to maintain a diet high in complex carbohydrates during the day, while utilizing glucagon pump at night. She was also started on continuous glucose monitoring system (CGMS) with an alarm to warn of hypoglycemia. Glucagon infusion rate was later titrated based on CGMS readings. Abdominal CT revealed increasing size of a right upper quadrant mass not previously subjected to radiotherapy. After radiation to this area, hypoglycemia improved, allowing further glucagon titration. In parallel, IGF-2 level declined to 380 ng/mL.
Conclusions:
Ectopic IGF-2 production is a rare but often fatal complication of many cancers, and should be considered on the differential diagnosis in patients with malignancy and unexplained hypoglycemia. Once hypoglycemia is diagnosed, patients often have end-stage disease. While treatment of the causative tumor is the only definitive intervention, anti-hypoglycemia therapy is a life-saving, temporizing measure. In this case, the patient attained euglycemia and survived 3 months after presentation before ultimately succumbing to other malignancy-related complications. Given efficacy in management of hypoglycemia while awaiting definitive tumor-directed therapy, we submit nighttime subcutaneous glucagon infusion and CGMS are valuable additions to the physician's armamentarium in managing this condition.
Occupational Neurobrucellosis Mimicking a Brain Tumor: A Case Report and Review of the Literature
Authors: Hussein Algahtani, Bader Shirah, Dina Abdulghani, Roiya Farhan, Raghad Algahtani
Journal: Case Rep Infect Dis (2017)
MeSH terms: Case Reports

Brucellosis is a zoonotic bacterial infection which is transmitted to humans from infected animals and is endemic in many parts of the world including Saudi Arabia. In this article, we report a case of occupational neurobrucellosis that presented with a space-occupying lesion mimicking a brain tumor. We stress on the importance of obtaining detailed social history including occupation to reach the diagnosis in several conditions including brucellosis. We also stress on taking universal precautions when handling any specimens. It may be advisable that manipulation of all unknown specimens arriving at the laboratory should occur in biological safety cabinet until a highly infectious organism is ruled out. Neurobrucellosis should be included in the differential diagnosis in patients presenting with solitary mass lesion mimicking brain tumor especially in endemic areas or high occupational risk group.
Primary vesical actinomycosis in a 23-year-old man
Authors: Tevfik Ziypak, Senol Adanur, Fatih Ozkaya, Muhammet Calık, Ozkan Polat, Yilmaz Aksoy, Isa Ozbey
Journal: Case Rep Urol (2014)

Introduction. Actinomycosis can affect any organ of the body, although cutaneous fistulas are common in actinomycotic infections, and other organs such as the bladder are only rarely involved. Case Presentation. Herein we report and discuss a young male patient with primary vesical actinomycosis. A 23-year-old man was hospitalized complaining of intermittent gross hematuria over a 6-month duration. The patient underwent a cystoscopic examination under general anesthesia; an edematous, hyperemic, wide-based mass, which protruded from the dome of the bladder, was seen and incompletely resected. The histopathological examination of the material showed Actinomyces organisms surrounded by inflammation and a photomicrograph showed the microorganism. After confirmation of bladder actinomycosis, the patient received penicillin. A CT scan of the abdomen and pelvis showed no evidence of the mass at the postoperative 6th month. Cystoscopic examination showed complete healing of the transurethral resection area at the dome of the bladder. Conclusion. In conclusion, we believe that the gold standard treatment for vesical actinomycosis should include the combination of a transurethral resection of the mass and long-term penicillin treatment.
Expression of endothelial nitric oxide synthase and endothelin-1 in skin tissue from amputated limbs of patients with complex regional pain syndrome
Authors: J George Groeneweg, Claudia Heijmans Antonissen, Frank J P M Huygen, Freek J Zijlstra
Journal: Mediators Inflamm (2008)
MeSH terms: Complex Regional Pain Syndromes / metabolism, Endothelin-1 / metabolism, Nitric Oxide Synthase Type III / metabolism, Skin / metabolism, Case Reports, Research Support, Non-U.S. Gov't, Adult, Amputation, Surgical, Chronic Disease, Complex Regional Pain Syndromes / pathology, Extremities / blood supply, Extremities / surgery, Female, Humans, Immunohistochemistry, Middle Aged, Platelet Endothelial Cell Adhesion Molecule-1 / metabolism, Skin / blood supply, Endothelin-1, Platelet Endothelial Cell Adhesion Molecule-1, Nitric Oxide Synthase Type III

Background and objectives:
Impaired microcirculation during the chronic stage of complex regional pain syndrome (CRPS) is related to increased vasoconstriction, tissue hypoxia, and metabolic tissue acidosis in the affected limb. Endothelial dysfunction is suggested to be the main cause of diminished blood flow. The aim of this study was to examine the distribution of endothelial nitric oxide synthase (eNOS) and endothelin-1(ET-1) relative to vascular density represented by the endothelial marker CD31-immunoreactivity in the skin tissue of patients with chronic CRPS.
Methods:
We performed immunohistochemical staining on sections of skin specimens obtained from the amputated limbs (one arm and one leg) of two patients with CRPS.
Results:
In comparison to proximal specimens we found an increased number of migrated endothelial cells as well as an increase of eNOS activity in distal dermis specimens.
Conclusions:
We found indications that endothelial dysfunction plays a role in chronic CRPS.
Fatal immune-related hepatitis with intrahepatic cholestasis and pneumonia associated with camrelizumab: A case report and literature review
Authors: Youwen Tan, Yun Ye, Li Chen
Journal: Open Med (Wars) (2021)
Keywords: camrelizumab, immune-related hepatitis, immune-related pneumonia, intrahepatic cholestasis
MeSH terms: Case Reports

Camrelizumab (SHR-1210), a human monoclonal antibody against programmed death receptor 1 (PD-1), blocks the binding of PD-1 to PD-L1, consequently inhibiting immune system evasion by tumor cells. A 65-year-old man underwent radical esophagectomy 5 months ago following the diagnosis of esophageal cancer by gastroscopy. Approximately 40 days later, capecitabine was administered at a dosage of 1.5 g Po bid for 14 days, and anti-PD-1 (camrelizumab 200 mg) was administered twice. Around 20 days later, abnormal liver function was detected. He received a diagnosis of drug-induced liver injury. Chest computed tomography scanning revealed interstitial inflammatory lesions in both lower lungs. Liver biopsy revealed immune injury with ductopenia. Therefore, the diagnosis was revised as immune-related pneumonia and hepatitis associated with camrelizumab. The treatment regimen of methylprednisolone was adjusted to 40 mg/day and gradually increased to 80 mg/day. Mycophenolate mofetil was administered at a dose of 2 g/day. Consequently, chest tightness and shortness of breath resolved, and pulmonary inflammation improved. However, jaundice did not improve and continued to exacerbate. The last measured prothrombin time was 41 s, prothrombin activity was 19%, and the international normalized ratio was 4.03. The cause of death was diagnosed as liver failure, cardiopulmonary failure, and septic shock.
Azithromycin-Induced Thrombocytopenia: A Rare Etiology of Drug-Induced Immune Thrombocytopenia
Authors: Muhammad Umer Butt, Ahmad Jabri, Samy Claude Elayi
Journal: Case Rep Med (2019)
MeSH terms: Case Reports

Drug-induced thrombocytopenia requires a high suspicion for diagnosis and a broad investigation to exclude other etiologies of low platelets. Cessation of the offending agent often results in recovery of platelet counts. Many medications are known to cause a degree of thrombocytopenia. We present a rare case of severe thrombocytopenia associated with administration of azithromycin.
Spontaneous retropharyngeal haematoma: a case report
Authors: Arvind Singh, Enyi Ofo, Vincent Cumberworth
Journal: J Med Case Rep (2008)

Introduction:
Spontaneous retropharyngeal haematoma is an unusual condition. It has multiple aetiological factors and can present to a number of specialists including the otolaryngologist.
Case presentation:
We describe a case of spontaneous retropharyngeal haematoma which demonstrates the dramatic presentation and emphasises the need for a conservative approach.
Conclusion:
It is important to be aware of this unusual condition with its distinct presentation. Surgical intervention should be resisted unless a treatable aetiological factor is found or airway compromise occurs. Most cases will resolve with conservative management.
H. capsulatum: A Not-So-Benign Cause of Pericarditis
Authors: Paolo K Soriano, Muhammad Iqbal, Shakthishri Kandaswamy, Sami Akram, Abhishek Kulkarni, Tamer Hudali
Journal: Case Rep Cardiol (2017)
MeSH terms: Case Reports

The common causes of pericarditis and its course are benign in the majority of cases. Thus, further testing is usually not pursued and treatment for a presumptive viral etiology with nonsteroidal agents and steroids has been an accepted strategy. We present a patient with pericarditis who was unresponsive to first-line therapy and was subsequently found to have necrotizing granulomas of the pericardium with extensive adhesions and fungal elements seen on tissue biopsy. Serologic testing confirms active H. capsulatum infection, and he responded well to Itraconazole treatment. In patients with pericarditis who fail standard therapy with NSAIDs and steroids, it is suggested that they undergo thorough evaluation and that histoplasmosis be considered as an etiology, especially in endemic regions.
Immediate Dental Implants Enriched with L-PRF in the Esthetic Zone
Authors: Manoti Sehgal, Lovleen Puri, Sapna Yadav, Puja Malhotra, Sumit Singh Phukela, Bhupender Yadav, Bharti Raina
Journal: Case Rep Dent (2018)
MeSH terms: Case Reports

The aim of this article is to present the clinical application of immediate implant placement with L-PRF and immediate prosthetic loading in anterior esthetic region. A 24-year-old healthy female patient reported with a chief complaint of poor esthetics in the upper front tooth region with retained deciduous teeth. On oral examination, there were retained deciduous teeth (52, 53, and 63) with congenitally missing permanent successors. The retained deciduous teeth were extracted, and immediate implant placement was done in the extraction sockets along with L-PRF membranes in one surgical session under local anesthesia. Immediate temporization was performed with composite crowns on immediately placed dental implants. After 3 months of the healing period, the final implant-level impressions were made and the temporary composite crowns were replaced with the final zirconia porcelain crowns. A 12-month follow-up was made, and satisfactory esthetic and functional results were obtained.
Unusual presentation of lung carcinoma with pituitary metastasis: a challenging diagnosis and sodium management dilemmas
Authors: Poh Shean Wong, Subashini Rajoo, Hairuddin Achmad Sankala, Mohamed Badrulnizam Long Bidin
Journal: Endocr Oncol (2022)
MeSH terms: Case Reports

Summary:
Pituitary metastasis (PM) is a rare complication of an advanced malignancy. Albeit rare, PM can be more detected and achieve a longer survival rate through frequent neuroimaging and newer oncology therapies. Lung cancer is the most frequent primary site, followed by breast and kidney cancers. Patients with lung cancer generally present with respiratory symptoms and are commonly diagnosed at an advanced stage already. Nevertheless, physicians should be mindful of other systemic manifestations as well as signs and symptoms related to metastatic spread and paraneoplastic syndromes. Herein, we report the case of a 53-year-old woman who presented with PM as the first sign of an undiagnosed lung cancer. Initially, her condition was a challenging diagnosis and was even complicated with diabetes insipidus (DI), which can present as severe hyponatremia when coexisting with adrenal insufficiency. This case also highlights that treating DI with antidiuretic hormone (ADH) replacement was complicated by extreme difficulties in attaining satisfactory sodium and water balance during the clinical course, with the possibility of coexistent DI and syndrome of inappropriate ADH secretion because of the underlying lung cancer.
Learning points:
When patients present with pituitary mass and diabetes insipidus (DI), pituitary metastasis should be considered as an initial differential diagnosis. DI caused by pituitary adenoma is rare and is typically a late finding.DI can present as severe hyponatremia when coexisting with adrenal insufficiency.Cortisol can directly inhibit endogenous antidiuretic hormone (ADH) secretion. Patients with adrenocorticotropic hormone deficiency will have increased tonic ADH activity and subsequently reduced capacity for free-water excretion. However, when on steroid therapy, patients should be monitored for possible DI because steroids can restore free-water excretion.A substantial change in serum sodium after desmopressin treatment should eliminate the possibility of desmopressin overdose or coexistence of DI and syndrome of inappropriate ADH secretion in patients with lung cancer. Therefore, frequent monitoring of serum sodium concentrations is crucial.
Necrotizing fasciitis due to Vibrio cholerae non-O1/non-O139 after exposure to Austrian bathing sites
Authors: Sonja Hirk, Steliana Huhulescu, Franz Allerberger, Sarah Lepuschitz, Sonja Rehak, Sandra Weil, Elisabeth Gschwandtner, Michael Hermann, Stephanie Neuhold, Alexander Zoufaly, Alexander Indra
Journal: Wien Klin Wochenschr (2016)
Keywords: bathing sites, climate change, drought, heat wave, necrotizing fasciitis, vibrio cholerae
MeSH terms: Fasciitis, Necrotizing / diagnosis, Fasciitis, Necrotizing / microbiology, Ponds / microbiology, Vibrio Infections / diagnosis, Vibrio Infections / microbiology, Vibrio cholerae non-O1 / isolation & purification, Case Reports, Research Support, Non-U.S. Gov't, Aged, Aged, 80 and over, Anti-Bacterial Agents / therapeutic use, Austria, Baths, Environmental Exposure, Fasciitis, Necrotizing / drug therapy, Fatal Outcome, Female, Humans, Male, Vibrio Infections / drug therapy, Water Microbiology, Anti-Bacterial Agents

We report on two cases of necrotizing fasciitis of the lower leg due to nontoxigenic Vibrio cholerae (V. cholerae). A 73-year-old woman (case 1) and an 80-year-old man (case 2) were hospitalized with symptoms of necrotizing fasciitis on July 18 and August 15, 2015, respectively. In both cases, symptoms started the day after swimming in local ponds. Swabs gained intraoperatively and a blood culture from the male patient, yielded V. cholerae non-O1/non-O139, negative for cholera toxin gene ctx and positive for hemolysin genes hlyA and hlyB. Water samples taken from pond A on August 17, 2015 (32 days after exposure of case 1) and from pond B on August 20, 2015 (7 days after exposure of case 2) yielded non-O1/non-O139 V. cholerae in most-probable numbers of > 11,000 per 100 ml each. The occurrence of two cases of necrotizing fasciitis within a 1 month period related to two Austrian non-saline bathing waters, previously not known to harbor V. cholerae, is probably linked to the prevailing extreme weather conditions (heat wave, drought) this summer in Austria. While case 1 was discharged in good clinical condition after 73 days, case 2 died after four months of hospitalization. Public health authorities are challenged to assess the effects of long-term climate change on pathogen growth and survival in continental bodies of fresh water.
Comprehensive orthodontic treatment of adult patient with cleft lip and palate
Authors: Noemí Leiva Villagra, Miguel Muñoz Domon, Sebastian Véliz Méndez
Journal: Case Rep Dent (2014)

The aim of the paper is to present full orthodontic treatment of an operated cleft lip adult patient. Case Report. An 18-year-old patient consulted for severe crowded teeth. He comes from a poor family. At that time he already had four operations (velum, palate, lip, and myringotomy). Treatment included maxillary expansion, tooth extraction, and fixed orthodontic, as well as kinesiology and speech therapy treatment. A multidisciplinary approach allowed us to achieve successfully an excellent result for this patient and gave him a harmonic smile and an optimal function without orthognathic surgery. Two years after treatment, occlusion remains stable.
An advanced understanding of the heterogeneous clinical features of "non-criteria" obstetric antiphospholipid syndrome: Two case reports and a literature review
Authors: Xue Peng, Xi Tan, Aiyun Xing
Journal: Front Immunol (2023)
Keywords: antiphospholipid antibodies, antiphospholipid syndrome, obstetric antiphospholipid syndrome, pregnancy, seronegative antiphospholipid syndrome, “non-criteria” obstetric antiphospholipid syndrome
MeSH terms: Abortion, Habitual / etiology, Antiphospholipid Syndrome / diagnosis, Autoimmune Diseases, Premature Birth, Review, Case Reports, Research Support, Non-U.S. Gov't, Antibodies, Antiphospholipid, Female, Humans, Infant, Newborn, Pregnancy, Antibodies, Antiphospholipid

Antiphospholipid syndrome (APS) is an acquired autoimmune disorder characterized by recurrent venous and/or arterial thrombosis and/or pregnancy complications, in the presence of elevated antiphospholipid (aPL) antibodies. APS in pregnant women is referred to as "obstetrical" APS (OAPS). The diagnosis of definite OAPS requires the presence of one or more typical clinical criteria and persistent aPL antibodies at least 12 weeks apart. However, the classification criteria for OAPS have generated wide discussion, with a growing impression that certain patients not fully meeting these criteria might be inappropriately excluded from the classification, which is known as "non-criteria" OAPS. We present here two unique cases of potentially lethal "non-criteria" OAPS, complicating severe preeclampsia, fetal growth restriction (FGR), liver rupture, preterm birth, refractory recurrent miscarriages, or even stillbirth. We further share our diagnostic search and analysis, treatment adjustment, and prognosis for this unusual antenatal event. We will also present a short review of an advanced understanding of the pathogenetic mechanisms of this disease, heterogeneous clinical features, and potential significance.
Pseudoaneurysm of the Perforating Peroneal Artery following Ankle Arthroscopy
Authors: Ichiro Tonogai, Eiki Fujimoto, Koichi Sairyo
Journal: Case Rep Orthop (2018)
MeSH terms: Case Reports

The use of standard anterolateral and anteromedial portals in ankle arthroscopy results in reduced risk of vascular complications. Anatomical variations of the arterial network of the foot and ankle might render the vessels more susceptible to injury during procedures involving the anterior ankle joint. The literature, to our knowledge, reports only one case of a pseudoaneurysm involving the peroneal artery after ankle arthroscopy. Here, we report the unusual case of a 48-year-old man in general good health with the absence of the anterior tibial artery and posterior tibial artery. The patient presented with a pseudoaneurysm of the perforating peroneal artery following ankle arthroscopy for traumatic osteoarthritis associated with nonunion of the medial malleolus. The perforating peroneal artery injury was repaired by performing end-to-end anastomosis. The perforating peroneal artery is at higher risk for iatrogenic injury during ankle arthroscopy in the presence of abnormal arterial variations of the foot and ankle, particularly the absence of the anterior tibial artery and posterior tibial artery. Before ankle arthroscopy, surgeons should therefore carefully observe the course of the perforating peroneal artery on enhanced 3-dimensional computed tomography, especially in patients with a history of trauma to the ankle joint.
Gastric duplication cyst: two case reports and review of the literature
Authors: Jai P Singh, Heena Rajdeo, Kalyani Bhuta, John A Savino
Journal: Case Rep Surg (2013)

Background. Duplication of the alimentary tract is a rare congenital anomaly. Gastric duplication cysts (GDCs) represent 4% of all alimentary tract duplications, and approximately 67% manifest within the first year of life. Duplication cysts in adults are generally encountered as incidental findings at endoscopy or laparotomy. Herein, we report two rare cases of symptomatic GDC presenting in adults. Case 1. A 27-year-old male presented with a five-month history of back pain. Exam revealed mild epigastric tenderness with a vague palpable mass in left upper abdomen. CT scan showed 8 × 7.4 × 6 cm homogenous, nonseptated cystic mass posterosuperior to pancreatic tail. On laparotomy, a cystic mass measuring 11 × 8 cm was found, which was densely adherent to posterior wall of stomach suggestive of GDC. Case 2. A 28-year-old woman presented with epigastric pain associated with vomiting for 2 months. Exam revealed mild epigastric tenderness. CT scan showed four cystic lesions in the medial wall of distal stomach measuring approximately one cm each suggestive of duplication cysts. Exploratory laparotomy with antrectomy and truncal vagotomy with Billroth II reconstruction were performed. Pathology in both patients was diagnostic of GDC. Conclusion. GDC is a rare anomaly, and its presentation in adults is even rarer.