OpenMed/multicare-articles huggingface dataset:
Fatal strongyloides hyperinfection complicating a gram-negative sepsis after allogeneic stem cell transplantation: a case report and review of the literature
Authors: Isabel Izquierdo, Javier Briones, Rafael Lluch, Cristina Arqueros, Rodrigo Martino
Journal: Case Rep Hematol (2013)

Strongyloides stercoralis is an intestinal nematode that causes strongyloidiasis, which affects 30 to 100 million people worldwide. Risk factors for hyperinfection and disseminated disease include immunosuppressive drug therapy, human T-lymphotropic virus-1 (HTLV-1) infection, solid organ and bone marrow transplantation, hematologic malignant diseases, hypogammaglobulinemia, and severe malnutrition and associated conditions. The diagnosis can be difficult because a single stool examination fails to detect larvae in up to 70% of the cases, and the symptoms are nonspecific. Although eosinophilia is a common finding in patients with chronic Strongyloides infection, it is an unreliable predictor of hyperinfection. Furthermore, the lack of eosinophilia while receiving immunosuppressive therapy cannot reliably exclude the underlying chronic Strongyloides infection. We report here a fatal Strongyloides hyperinfection in a patient receiving allogeneic stem cell transplantation; risk factors and outcome in this clinical setting are discussed.
Luteoma of Pregnancy Presenting with Severe Maternal Virilisation: A Case Report
Authors: Vincenzo Rapisarda, Francesco Pedalino, Veronica Concetta Santonocito, Giorgia Cavalli, Giuseppe Zarbo
Journal: Case Rep Obstet Gynecol (2016)

Luteoma of pregnancy is a rare, benign condition characterized by a tumor-like mass of the ovary that emerges during pregnancy and regresses spontaneously after delivery. It is usually asymptomatic and the diagnosis is generally incidental. Luteoma arises from the proliferation of luteinised cell under the influence of β-hCG and can be hormonally active, with production of androgens resulting in maternal and fetal hirsutism and virilisation. We report a case of a 25-year-old primigravida who presented at 28 weeks of gestation with virilisation symptoms. Serum androgen levels were seven-hundred-fold higher than normal. A diagnosis of pregnancy luteoma was made at the time of caesarean section. The ovarian mass, serum androgen levels, and the condition of the patient improved after delivery.
Transient Dexmedetomidine Bolus-Induced Excessive Urination Intraoperatively in a 68-Year-Old Male
Authors: Joseph A McGuire, Summer A Niazi, Daniel C Sizemore
Journal: Case Rep Anesthesiol (2020)
MeSH terms: Case Reports

Excessive urination can be a perioperative challenge for providers due to the possibility of secondary hypernatremia. Dexmedetomidine has previously been reported by several groups to induce a polyuric-like syndrome; however, the exact mechanism in humans remains unclear. In this report, we discuss a case of intraoperative, transient dexmedetomidine bolus-induced excessive urination and suggest a potential mechanism by which this may occur in a subset of the population.
Prolonged Myelosuppression due to Progressive Bone Marrow Fibrosis in a Patient with Acute Promyelocytic Leukemia
Authors: Yuta Inagawa, Yukiko Komeno, Satoshi Saito, Yuji Maenohara, Tetsuro Yamagishi, Hiroyuki Kawashima, Taku Saito, Keiko Abe, Kuniko Iihara, Yasumasa Hatada, Tomiko Ryu
Journal: Case Rep Hematol (2019)
MeSH terms: Case Reports

A 34-year-old woman was diagnosed with acute promyelocytic leukemia. Chemotherapy was administered following the JALSG APL204 protocol. Induction therapy with all-trans retinoic acid resulted in complete remission on day 49. She developed coccygeal pain from day 18, which spread to the spine and cheekbones and lasted 5 weeks. She had similar bone pain on days 7-10 of the first consolidation therapy and on days 4-12 of the second consolidation therapy. Oral loxoprofen was prescribed for pain relief. On day 33 of the third consolidation, white blood cell and neutrophil counts were 320/μL and 20/μL, respectively. After she developed epigastralgia and hematemesis, she developed septic shock. Gastroendoscopy revealed markedly thickened folds and diffusely damaged mucosa with blood oozing. Computed tomography revealed thickened walls of the antrum and the pylorus. Despite emergency treatments, she died. Bacterial culture of the gastric fluid yielded Enterobacter cloacae and enterococci growth. Collectively, she was diagnosed with phlegmonous gastritis. Retrospective examination of serial bone marrow biopsy specimens demonstrated progressive bone marrow fibrosis, which may have caused prolonged myelosuppression. Thus, evaluation of bone marrow fibrosis by bone marrow biopsy after each treatment cycle might serve as a predictor of persistent myelosuppression induced by chemotherapy.
Successful Regional Anesthetic for a Parturient with Moyamoya Syndrome
Authors: Anjalena Pasam, Akshatha Kamath, Hattiangadi Sangeetha Kamath, Joel Yarmush, Khaja Ahmed
Journal: Case Rep Anesthesiol (2020)
MeSH terms: Case Reports

Anesthesia for Cesarean section could be challenging due to the physiological changes during pregnancy, but it can be more complicated if associated with sickle cell disease and moyamoya disease. The moyamoya syndrome is nothing but sickle cell disease complicated by cerebral vasculopathy. Incidence of moyamoya disease in the USA is 0.086/100,000 people. We report a case of a pregnant woman with sickle cell disease and moyamoya syndrome, who underwent a successful spinal epidural for primary cesarean section, with careful monitoring of blood pressure.
A rare case of squamous cell carcinoma of the bladder presenting as a metastatic right ventricular mass
Authors: Joanna M Bonsall, Rhome Hughes, Mario Mosunjac, David Harrison, Habib Samady
Journal: Case Rep Med (2010)
MeSH terms: Case Reports

A 74-year-old woman presented with bilateral lower extremity swelling, worsening dyspnea on exertion, and mild hemoptysis. An echocardiogram at time of admission showed a mass in the right ventricle. The pathology of a sample obtained via transvenous biopsy was consistent with squamous cell carcinoma; no primary source could initially be identified. Severe thrombocytopenia, likely consumptive, precluded surgical intervention, so the patient underwent palliative radiation. Unfortunately, she developed fatal respiratory failure. Upon autopsy, the bladder was found to contain polyps of invasive squamous cell carcinoma, similar in morphology to the tumor mass in the heart. Her lungs contained multiple tumor emboli at different stages, which was likely the final cause of her death. Squamous cell carcinoma metastases to the endocardium are extremely rare and without defined treatment. Surgery can improve prognosis in those with primary tumors that are benign or without metastases. In those with symptomatic metastatic tumors, palliative debulking can done although generally will not improve prognosis. It is currently unknown whether radiation improves survival. In this case, irradiation did destroy a portion of the tumor as the final pathology showed extensive necrosis of the tumor; unfortunately, it did not change her symptoms and did not change the final outcome.
Congenital bronchial atresia presenting as a cavitary lesion on chest radiography: a case report
Authors: Kostas Psathakis, Danai Eleftheriou, Panagiotis Boulas, Charalampos Mermigkis, Kostas Tsintiris
Journal: Cases J (2009)

Background:
Congenital bronchial atresia is a rare anomaly, which usually presents in adulthood as an incidental finding on routine examinations.
Case presentation:
In this report we present a patient with a cavitary lesion at his right upper lobe, found by chance on chest radiography. Computed tomography of the chest revealed the characteristic findings of a mucocele with distal oligemia and hyperlucency of the affected lung parenchyma. Further examination including bronchoscopy virtually excluded other possible disorders and the diagnosis of congenital bronchial atresia was established.
Conclusion:
The radiological presentation of congenital bronchial atresia may occasionally mimic serious lung diseases. The procedure of choice for the diagnosis is the computed tomography of the chest. Bronchoscopy is not diagnostic but is valuable in doubtful cases to exclude different disorders.
Polypoid Dysplasia in Barrett's Esophagus: Diagnosis, Management, and Very Different Outcomes in Two Consecutive Cases
Authors: Megan Murphy, Christina Tofani, Kunjal Gandhi, Anthony Infantolino
Journal: Case Rep Gastrointest Med (2016)

Background. Barrett's esophagus is associated with an increased risk of adenocarcinoma. Dysplasia in Barrett's esophagus is a precursor to adenocarcinoma. Rarely, dysplastic polypoid lesions are superimposed on Barrett's esophagus. Most reported cases of polypoid dysplasia in Barrett's esophagus have been advanced on presentation and treated with esophagectomy. We describe two cases of polypoid changes in Barrett's esophagus and treatment with polypectomy followed by radiofrequency ablation. Cases. A 75 yo male presented with esophageal polyps, which on biopsy showed gastric cardia/foveolar mucosa with focal intestinal metaplasia without dysplasia. Biopsy of intervening flat mucosa was consistent with nondysplastic Barrett's esophagus. Extensive hot snare polypectomies were performed followed by RFA. One year later, repeat EGD revealed no evidence of Barrett's esophagus. A 61 yo male presented with esophageal polyps, which on biopsy showed gastric cardia/foveolar mucosa with intestinal metaplasia and foci of low-grade dysplasia. Extensive hot snare polypectomies were performed followed by RFA. At repeat EGD, four months later, an esophageal mass was found. Biopsy of the mass showed invasive adenocarcinoma. The patient was referred for esophagectomy. Conclusion. This case series shows two outcomes, one with successful eradication of dysplasia and the other with disease progression to invasive adenocarcinoma requiring esophagectomy.
Case Report: Exceptional Response to Second Line Temozolomide Therapy in a Patient With Metastatic Adrenocortical Carcinoma
Authors: Deborah Cosentini, Antonella Turla, Ornella Carminati, Salvatore Grisanti, Vittorio Domenico Ferrari, Marta Laganà, Giovanni Rosti, Sandra Sigala, Alfredo Berruti
Journal: Front Endocrinol (Lausanne) (2021)
Keywords: acc, adrenal tumor, alkylating drug, progesterone, treatment
MeSH terms: Adrenal Cortex Neoplasms / drug therapy, Adrenocortical Carcinoma / drug therapy, Antineoplastic Combined Chemotherapy Protocols / therapeutic use, Case Reports, Research Support, Non-U.S. Gov't, Adrenal Cortex Neoplasms / pathology, Adrenocortical Carcinoma / secondary, Adult, Cisplatin / administration & dosage, Doxorubicin / administration & dosage, Etoposide / administration & dosage, Female, Humans, Mitotane / administration & dosage, Prognosis, Remission Induction, Temozolomide / administration & dosage, Young Adult, Etoposide, Mitotane, Doxorubicin, Cisplatin, Temozolomide

Background:
In a recently published retrospective case series, Temozolomide was found active as second line approach in advanced ACC patients. The disease control rate obtained, however, was short-lived. We report here an ACC patient with extensive metastatic disease who obtained a remarkable long lasting response with this alkylating agent.
Case presentation:
a 22-year-old female patient with ACC presented at our Medical Oncology Department in poor general condition due the presence of extensive metastatic pulmonary involvement. The disease had progressed to etoposide, doxorubicin and cisplatin plus mitotane therapy. Second line temozolomide therapy was prescribed leading to a progressive improvement of patient general conditions. The disease restaging after 12 cycles revealed a complete response of lung lesions and the patient was free from progression for 14+ months.
Conclusion:
Temozolomide therapy could be exceptionally efficacious in the management of ACC patients. The molecular mechanisms of sensitivity and resistance to this drug should be carefully studied, in order to select the patients destined to obtain a significant clinical benefit to the drug.
Anti-contactin-associated protein-like 2 antibody-associated encephalitis in children: A case report and literature review
Authors: Qingyang Dou, Renke Li, Xiaomei Shu
Journal: Front Pediatr (2022)
Keywords: antibody, autoimmune encephalitis, caspr2, children, prognosis
MeSH terms: Case Reports

Background:
Anti-Contactin-associated protein-like 2 (CASPR2) antibody-associated encephalitis is a rare group of autoimmune diseases that causes extensive damage to the central and/or peripheral nervous system.
Case presentation:
Here, we reported a case of anti-CASPR2 antibody-associated encephalitis in a 12-year-old male patient with symptoms of headache, consciousness disturbance, mental abnormalities, urinary incontinence, fasciculations in the extremity muscles, and involuntary movements. The testing for autoimmune encephalitis-associated antibodies showed that CASPR2-associated antibodies were positive, and electroencephalography showed diffuse slow waves. No tumor was found after screening for malignancies. The child's status significantly improved after receiving immunotherapy with intravenous methylprednisolone and immunoglobulin.
Conclusions:
Anti-CASPR2 antibody-associated encephalitis has been rarely reported in children. It has a complex clinical presentation and a low incidence of tumor. Most pediatric patients have a favorable prognosis and relapse is uncommon.
Longitudinal Characterization of Cytokine Overproduction: A Case Report in Critically Ill COVID-19 Patients With Hyperinflammation in Bronchoalveolar Lavage
Authors: Zhen Luo, Chengliang Zhu, Zhihui Ruan, Xianghua Cui, Muhammad Adnan Shereen, Pan Pan, Jingtao Huang, Fubing Wang, Hanwen Su, Yuchen Xia, Jianguo Wu
Journal: Front Med (Lausanne) (2021)
Keywords: covid-19, sars-cov-2, bronchoalveolar lavage fluid, cytokine overproduction, hyperinflammation, poor outcomes
MeSH terms: Case Reports

Objectives: The longitudinal characterization and risk of poor outcomes related to cytokine overproduction in critical coronavirus disease 2019 (COVID-19) patients with hyperinflammation in bronchoalveolar lavage requires further investigation. Methods: We enrolled two critically ill patients with comorbidities diagnosed with severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) detected by RT-PCR during hospitalization. Clinical characteristics, longitudinal immunological, and biochemical parameters of each critical COVID-19 case were collected. Main Results: The clinical characteristics and laboratory results of each case demonstrated critical symptoms of COVID-19 with poor outcomes. Both nasopharyngeal swabs and bronchoalveolar lavage fluid (BALF) samples tested positive for SARS-CoV-2. Two patients received targeted treatments against pathogen infection and inflammation in addition to interventional therapies, except for Patient 2, who received an additional artificial liver system treatment. Hyperinflammation with a dominantly high level of IL-6 was observed in BALF samples from both critical cases with decreased T cell populations. High levels of cytokines and pathological parameters were successively maintained in Patient 1, but rapidly reduced at the late treatment stage in Patient 2. The outcome of Patient 1 is death, whereas the outcome of Patient 2 is recovery. Conclusions: This case report suggests that a high risk of poor outcomes was related to a heavily hyperinflammatory milieu in both the blood and lungs of critical COVID-19 patients. The artificial liver intervention on cytokines overproduction might be beneficial for the recovery of critical COVID-19 patients as a reliable therapy that can be coordinated with targeted treatments, which ought to be further tested in adequately designed and powered clinical trials.
Mature Cystic Teratoma Hiding in the Retroperitoneum of an Adult
Authors: Yardesh Singh, Shamir O Cawich, Thivy Kuruvilla, Sidiyq Mohammed, Ammiel Arra
Journal: Case Rep Oncol Med (2018)
MeSH terms: Case Reports

We report a rare case of a mature cystic teratoma found in the retroperitoneum of a 28-year-old woman with vague symptomatology. We review the radiologic and pathologic features of this rare lesion.
Fulminant Cerebral Fat Embolism: Case Description and Review of the Literature
Authors: Giorgio Berlot, Rossana Bussani, Vennus Shafiei, Nadia Zarrillo
Journal: Case Rep Crit Care (2018)
MeSH terms: Case Reports

The release of fat and bone marrow fragments is a common occurrence following traumatic and nontraumatic events. In most cases, they go symptomless or cause only minor disturbances, but occasionally they can determine a multiorgan dysfunction whose severity ranges from mild to fatal. The authors describe the case of a patient who became deeply comatose and ultimately died after a traffic accident in which he suffered the exposed right femoral and tibial fracture in the absence of other injuries. He underwent the external fixation of the fractured bones 2 hours after the admission under general anesthesia. Three hours later, he failed to awake at the suspension of the anesthetic agents and became anisocoric; a CT scan demonstrated a diffuse cerebral edema with the herniation of the cerebellar tonsils; these abnormalities were unresponsive to the treatment and the brain death was one day later. The causes, the mechanisms, the symptoms, the prevention, and the treatment of the syndrome are reviewed and discussed.
Case report: Intraoperative thrombosis cardiac arrest in extended right hepatectomy involving use of local haemostatic agent in intraoperative cell salvage (ICS) and administration of recombinant activated factor VII (rFVIIa)
Authors: Lee S Kyang, Andrew Howard, Nayef A Alzahrani, David L Morris
Journal: Int J Surg Case Rep (2019)
Keywords: intraoperative cell salvage, liver resection, mortality, recombinant activated factor vii, thrombosis, rfviia

Introduction:
In modern surgical era, local haemostatic agents and blood components such as recombinant activated factor VII (rFVIIa) have expanded surgeons' armamentarium in controlling "surgical" and "nonsurgical bleeding". We report a case of intraoperative thrombosis and cardiac arrest involving use of local haemostatic agent in intraoperative cell salvage and rFVIIa administration in extended right hepatectomy.
Presentation of case:
A 46-year-old lady underwent extended right hepatectomy using cardiopulmonary bypass (CPB) and autotransfusion with ICS for metastatic gastrointestinal stromal tumour. She became extremely coagulopathic following weaning of CPB despite an array of fluid and blood products replacements. Decision to administer rFVIIa as a measure to arrest bleeding was unsuccessful. Extensive systemic thrombosis occurred which resulted in cardiac arrest and mortality.
Discussion:
The thromboembolic event was unclear but likely multifactorial. Two important hypotheses were the administration of rFVIIa and use of local haemostatic agent in ICS.
Conclusion:
Reported incidence of thromboembolism with use of rFVIIa in refractory bleeding is variable. More randomised controlled trials are needed to ascertain the efficacy and safety profile of the haemostatic agent. At present, off-label use of rFVIIa should be guided by the risk:benefit profile on a case-to-case basis. The authors also feel strongly against the use of local haemostatic gel in conjunction with ICS due to potential systemic circulation of the thrombin.
Case report: Preimplantation genetic testing for X-linked alport syndrome caused by variation in the COL4A5 gene
Authors: Nengqing Liu, Xiaojun Wen, Zhanhui Ou, Xiaowu Fang, Jing Du, Xiufeng Lin
Journal: Front Pediatr (2023)
Keywords: col4a5, x-linked alport syndrome, next-generation sequencing, preimplantation genetic testing, type iv collagen
MeSH terms: Case Reports

X-Linked Alport Syndrome (XLAS) is an X-linked, dominant, hereditary nephropathy mainly caused by mutations in the COL4A5 gene, found on chromosome Xq22. In this study, we reported a pedigree with XLAS caused by a COL4A5 mutation. This family gave birth to a boy with XLAS who developed hematuria and proteinuria at the age of 1 year. We used next-generation sequencing (NGS) to identify mutations in the proband and his parents and confirmed the results using Sanger sequencing. This testing showed there was a single nucleotide missense variation, c.3659G>A (p.Gly1220Asp) (NM_033380.3), in the COL4A5 gene. To prevent the inheritance of the syndrome, we used eight embryos for trophoblast biopsy after assisted reproductive technology treatment, and whole genome amplification (WGA) was performed using multiple annealing and looping-based amplification cycles (MALBAC). Embryos were subjected to Preimplantation Genetic Testing (PGT) procedures, including Sanger sequencing, NGS-based single nucleotide polymorphism (SNP) haplotype linkage analysis, and chromosomal copy number variation (CNV) analysis. The results showed that three embryos (E1, E2, and E4) were free of CNV and genetic variation in the COL4A5 gene. Embryo E1 (4AA) was transferred after consideration of the embryo growth rate, morphology, and PGT results. Prenatal diagnosis in the second trimester showed that the fetus had a normal karyotype and did not carry the COL4A5 mutation (c.3659G>A). Ultimately, a healthy boy was born and did not carry the pathogenic COL4A5 mutation, which indicated that PGT prevented the intergenerational transmission of the causative mutation of XLAS.
Potential benefit of hormonal therapy for non-uterine soft tissue sarcoma (STS) - a case report and literature review
Authors: Li Li, Isaiah P Schuster, Robin Jacob, Kenneth H Hupart, Vladimir Gotlieb
Journal: Springerplus (2013)
Keywords: estrogen receptor, hormonal therapy, lms, soft tissue sarcoma

The expression of hormone receptors (HR) is considered a good prognostic marker in uterine sarcoma. Hormonal therapy is widely employed in the therapy of HR positive breast and gynecologic cancers, however, there is little information concerning hormonal therapy in HR positive extrauterine sarcoma. A 55-60 year age group female presented with an estrogen receptor positive metastatic retroperitoneal leiomyosarcoma (LMS). She was treated with four cycles of a combination of Gemcitabine and Paclitaxel. Her disease remained stable for 29 months when tamoxifen was initiated. The patient succumbed to an unrelated malignancy after a total of 44 months of treatment. Despite emerging reports about the potential benefit of hormonal therapy, selective estrogen and progesterone receptor modulators and aromatase inhibitors, for uterine sarcoma, there is a paucity of information regarding the application of these therapies to sarcomas arising at other sites. Our patient survived significantly longer than expected with metastatic retroperitoneal sarcoma. In part this may be due to the survival benefit associated with HR positive tumors, but it may also indicate a role for hormonal therapy which has yet to be explored.