OpenMed/multicare-articles huggingface dataset:
Refractory Immune Thrombocytopenic Purpura and Cytomegalovirus Infection: A Call for a Change in the Current Guidelines
Authors: Alexei Shimanovsky, Devbala Patel, Jeffrey Wasser
Journal: Mediterr J Hematol Infect Dis (2016)
MeSH terms: Case Reports

Immune thrombocytopenic purpura (ITP) is characterized by a decreased platelet count caused by excess destruction of platelets and inadequate platelet production. In many cases, the etiology is not known, but the viral illness is thought to play a role in the development of some cases of ITP. The current (2011) American Society of Hematology ITP guidelines recommend initial diagnostic studies to include testing for HIV and Hepatitis C. The guidelines suggest that initial treatment consist of observation, therapy with corticosteroids, IVIG or anti D. Most cases respond to the standard therapy such that the steroids may be tapered and the platelet counts remain at a hemostatically safe level. Some patients with ITP are dependent on long-term steroid maintenance, and the thrombocytopenia persists with the tapering of the steroids. Recent case reports demonstrate that ITP related to cytomegalovirus (CMV) can persist in spite of standard therapy and that antiviral therapy may be indicated. Herein we report a case of a 26-year-old female with persistent ITP that resolved after the delivery of a CMV-infected infant and placenta. Furthermore, we review the current literature on CMV-associated ITP and propose that the current ITP guidelines be amended to include assessment for CMV, even in the absence of signs and symptoms, as part of the work-up for severe and refractory ITP, especially prior to undergoing an invasive procedure such as splenectomy.
Left atrial mass found on cardiac ultrasound leading to emergent thrombectomy
Authors: Nathalie van der Rijst, Tejas Sinha, Parth Rali, Parag Desai, Sebastian Iturra, Aamir Ajmeri
Journal: Respir Med Case Rep (2022)
MeSH terms: Case Reports

Atrial myxomas, though the most common primary cardiac neoplasm, remain a rare disease occurring in about 0.03% of the population. While clinically benign, they are considered functionally malignant as they can cause life-threatening embolic events. Here we present a patient with a high-risk intermediate pulmonary embolism where bedside ultrasound revealed significant right ventricular dysfunction with an associated large left atrial mass. These findings combined with the patient's instability allowed her to be rushed to surgery for definitive treatment.
A Case Report of Acute Esotropia in a Young Woman following Heroin Withdrawal
Authors: Bethel Shiferaw, Ebisa Bekele, Sara Syed, Lu Fan, Nirav Patel, Samia Qazi, Nicolas Biro
Journal: Case Rep Med (2015)

Introduction. Esotropia is a form of strabismus that can give the affected individual a "cross-eyed" appearance. Acute onset of esotropia is an uncommon form; in the vast majority of cases, no underlying neurological etiology is found. Case Presentation. A 22-year-old female with a long history of opiate abuse presented with acute onset of diplopia. She noted her eyes were crossing and started seeing double. She stopped using heroin 11 days prior to presentation. There was large inward deviation of her left eye. Convergence was difficult and accompanied by horizontal nystagmus. Diplopia resolved by covering each eye. Further investigations including imaging studies were normal. Discussion. Acute onset esotropia is rare and must be investigated right away to exclude central nervous system pathologies, where no opiates use is reported. Diplopia in the form of acute esotropia may manifest in up to 30% of individuals undergoing heroin withdrawal. Evaluating acute esotropia requires detailed information of medical history with an emphasis on drug use. Conclusion. Acute onset esotropia with double vision can be caused by abrupt withdrawal of opiates. This case should serve to raise awareness among health care professionals, to avoid costly and unnecessary diagnostic evaluations and interventions.
Root Resorption of the Permanent Central Incisor through Ectopic Eruption of the Maxillary Canine
Authors: Maria Pires Machado Paixão, Kelly Maria Silva Moreira, Ricardo Scarparo Navarro, Suzana Cavalcanti Monteiro de Oliveira, José Carlos Pettorossi Imparato, Juliana Braga Reis
Journal: Case Rep Dent (2023)
MeSH terms: Case Reports

Although ectopic eruption of the maxillary canine has a low prevalence, a late diagnosis can have serious consequences. A careful clinical examination, assisted with radiographic examination, ensures early diagnosis, facilitates planning, and minimizes possible adverse consequences. This study reports a case of ectopic eruption of the permanent maxillary canine, with complete root resorption of the central permanent incisor, the consequences of which caused functional, aesthetic, and psychological harms to the patient. The procedures used included canine ectopic remodeling of the ectopic canine in the central incisor and orthodontic correction, which treated the anomaly and rebuilt the patient's self-esteem.
Reexpansion Pulmonary Edema following Laparoscopy-Assisted Distal Gastrectomy for a Patient with Early Gastric Cancer: A Case Report
Authors: Kazuhito Yajima, Tatsuo Kanda, Ryo Tanaka, Yu Sato, Takashi Ishikawa, Shin-Ichi Kosugi, Tadayuki Honda, Katsuyoshi Hatakeyama
Journal: Case Rep Surg (2012)

We report here a case of reexpansion pulmonary edema following laparoscopy-assisted distal gastrectomy (LADG) for early gastric cancer. A 57-year-old Japanese woman with no preoperative comorbidity was diagnosed with early gastric cancer. The patient underwent LADG using the pneumoperitoneum method. During surgery, the patient was unintentionally subjected to single-lung ventilation for approximately 247 minutes due to intratracheal tube dislocation. One hour after surgery, she developed severe dyspnea and produced a large amount of pink frothy sputum. Chest radiography results showed diffuse ground-glass attenuation and alveolar consolidation in both lungs without cardiomegaly. A diagnosis of pulmonary edema was made, and the patient was immediately intubated and received ventilatory support with high positive end-expiratory pressure. The patient gradually recovered and was weaned from the ventilatory support on the third postoperative day. This case shows that single-lung ventilation may be a risk factor for reexpansion pulmonary edema during laparoscopic surgery with pneumoperitoneum.
A Case of New Onset Cervical Dystonia in Pregnancy
Authors: Eoghan Donlon, Pat Moloney, Darragh Frier, Robert McFarlane, Shane Smyth, Fiona Molloy, Richard Walsh, Tim Lynch, Eavan McGovern
Journal: Tremor Other Hyperkinet Mov (N Y) (2022)
Keywords: dystonia, dystonia gravidarum, botulinum toxin, pregnancy
MeSH terms: Dystonic Disorders, Movement Disorders, Restless Legs Syndrome, Torticollis, Case Reports, Female, Humans, Movement, Pregnancy

Background:
Though uncommon, primary movement disorders can occur in pregnancy, the most common being restless legs syndrome and chorea gravidarum [1]. New onset dystonia in pregnancy has been reported four times previously with a resolution of symptoms within six months of delivery [2345]. Exacerbation of pre-existing movement disorders and the onset of de novo movement disorders during pregnancy support the hypothesis that female sex hormones play an important role in the regulation of basal ganglia circuitry.
Case report:
Here we describe a case of new-onset cervical dystonia during pregnancy with persistence of symptoms after delivery.
Discussion:
The phenotypic overlap between this case and previously reported cases further establishes dystonia gravidarum as a distinct clinical entity.
A high-value, low-cost bubble continuous positive airway pressure system for low-resource settings: technical assessment and initial case reports
Authors: Jocelyn Brown, Heather Machen, Kondwani Kawaza, Zondiwe Mwanza, Suzanne Iniguez, Hans Lang, Alfred Gest, Neil Kennedy, Robert Miros, Rebecca Richards-Kortum, Elizabeth Molyneux, Maria Oden
Journal: PLoS One (2013)
MeSH terms: Bronchiolitis / therapy, Continuous Positive Airway Pressure / instrumentation, Oxygen Inhalation Therapy / instrumentation, Respiratory Distress Syndrome, Newborn / therapy, Case Reports, Research Support, Non-U.S. Gov't, Research Support, U.S. Gov't, Non-P.H.S., Acute Disease, Bronchiolitis / pathology, Continuous Positive Airway Pressure / economics, Developing Countries, Humans, Infant, Infant, Newborn, Oxygen Inhalation Therapy / economics, Respiratory Distress Syndrome, Newborn / pathology, Treatment Outcome

Acute respiratory infections are the leading cause of global child mortality. In the developing world, nasal oxygen therapy is often the only treatment option for babies who are suffering from respiratory distress. Without the added pressure of bubble Continuous Positive Airway Pressure (bCPAP) which helps maintain alveoli open, babies struggle to breathe and can suffer serious complications, and frequently death. A stand-alone bCPAP device can cost $6,000, too expensive for most developing world hospitals. Here, we describe the design and technical evaluation of a new, rugged bCPAP system that can be made in small volume for a cost-of-goods of approximately $350. Moreover, because of its simple design--consumer-grade pumps, medical tubing, and regulators--it requires only the simple replacement of a <$1 diaphragm approximately every 2 years for maintenance. The low-cost bCPAP device delivers pressure and flow equivalent to those of a reference bCPAP system used in the developed world. We describe the initial clinical cases of a child with bronchiolitis and a neonate with respiratory distress who were treated successfully with the new bCPAP device.
Case Report: A Novel ABCC8 Variant in a Chinese Pedigree of Maturity-Onset Diabetes of the Young
Authors: Chaoyan Tang, Liheng Meng, Ping Zhang, Xinghuan Liang, Chaozhi Dang, Hui Liang, Junfeng Wu, Haiyun Lan, Yingfen Qin
Journal: Front Endocrinol (Lausanne) (2021)
Keywords: abcc8, maturity-onset diabetes of the young (mody), sulfonylureas, treatment, variant
MeSH terms: Diabetes Mellitus, Type 2 / genetics, Sulfonylurea Receptors / genetics, Case Reports, Research Support, Non-U.S. Gov't, Adolescent, Adult, Aged, Diabetes Mellitus, Type 2 / drug therapy, Female, Humans, Hypoglycemic Agents / administration & dosage, Middle Aged, Mutation, Missense, ABCC8 protein, human, Hypoglycemic Agents, Sulfonylurea Receptors, Mason-Type Diabetes

Background:
We aimed to analyze a novel ABCC8 variant of a Chinese patient with suspected maturity-onset diabetes of the young (MODY) and to provide evidence for precise diagnosis and appropriate treatment.
Method:
A Chinese family with suspected MODY was recruited in this study, which included a 15-year-old female patient with diabetes. Clinical data and blood samples were collected from the proband and other family members. All of the living relatives were given an oral glucose tolerance test. Next-generation sequencing was performed to identify the mutated genes in the proband. Sanger sequencing was utilized to confirm the location of the pathogenic variant in all subjects. Further treatment was referred to targeted family members according to genetic testing.
Results:
The proband was found to have a random blood glucose level of 244.8 mg/dl and an HbA1c level of 9.2%. Before this investigation, her grandparents had been diagnosed with diabetes. The second uncle, two aunts, mother, and cousin of the proband were diagnosed with diabetes by abnormal HbA1C (6.5-12.1%) and fasting blood glucose (FBG, 91.4-189.7 mg/dl). The second aunt of the proband had impaired glucose homeostasis (HbA1C = 6.4% and FBG = 88.0 mg/dl). One novel missense variant c.1432G>A (p.A478T) in exon 9 of the ABCC8 gene was detected in the proband with suspected MODY. The variant was also found in six family members with diabetes or impaired glucose homeostasis, including her second uncle, two aunts, mother, and cousin. After the treatment was switched to glimepiride, the fasting blood glucose was adjusted to 99.54 mg/dl, the 2-h postprandial blood glucose was 153.54 mg/dl, serum fructosamine was 259 μmol/l, and HbA1c was 5.8%. The glycemic control remained optimal, and no hypoglycemic episodes were observed in the living relatives.
Conclusion:
This study revealed one novel missense variant of the ABCC8 gene in Chinese families. The present findings indicated that the members of this family responded to treatment with sulfonylureas as previously seen in ABCC8 MODY.
Unusual Presentation of Prostate Cancer Metastatic to the Cricoid Cartilage and Oral Cavity
Authors: Marlene Olvera, Miriam Delgado, Melchor Vázquez, José Zavala, Verónica Macedo, Martha Puentes
Journal: Case Rep Med (2018)
MeSH terms: Case Reports

In Mexico, prostate cancer is the second leading cause of death in men. Prostate cancer usually presents metastasis to the regional lymph nodes and bone. Hereby, we present an unusual case of metastatic prostate cancer, with affectation to the cricoid cartilage and oral cavity, being the first case to have ever been reported in Mexico. A 68-year-old Mexican man was diagnosed with prostate cancer and cribriform architecture histology with low serum level of prostate-specific antigen, debuting with laryngeal stridor. The biopsy came back positive for metastatic prostate carcinoma. During treatment with radiotherapy, metastasis developed to the oral cavity.
Malignant peritoneal mesothelioma: clinicopathological characteristics of two cases
Authors: Mustafa Cem Algın, Faik Yaylak, Zülfü Bayhan, Figen Aslan, Nilüfer Araz Bayhan
Journal: Case Rep Surg (2014)

Introduction. Peritoneal mesothelioma is a rare tumor, presenting difficulties in diagnosis and treatment. Peritoneum is the second most common area of the mesothelioma after pleura, and even synchronous pleural and peritoneal mesotheliomas are observed in 30-45% of all cases. The diagnosis may be difficult due to lack of specific symptoms and clinical findings. In addition, a delay in the diagnosis is not rare especially in the absence of previous asbestos exposure. Here we report two cases of malignant peritoneal mesotheliomas. The diagnostic and therapeutic approaches for these rare neoplasms are discussed. Case Presentation. The cases were two men (one aged 54 years old and the other 40 years old). Prolonged abdominal pain and swelling were the primary presentation symptoms and findings. The mesotheliomas were developed in the right upper quadrant of abdomen in both of the cases. Both cases were treated with surgical resection. Final diagnosis were possible with histological and immunohistochemical documentation of tumor characteristics, which were consistent with dictating a mesothelial origin. No history of asbestos exposure was reported. Conclusion. Peritoneal mesotheliomas are rare clinical entities. However, patients with prolonged abdominal pain and abdominal masses should be considered to have atypical pathologies such as peritoneal mesotheliomas.
Stormorken Syndrome Caused by a Novel STIM1 Mutation: A Case Report
Authors: Li-Jun Jiang, Xue Zhao, Zhi-Yan Dou, Qing-Xiao Su, Zan-Hua Rong
Journal: Front Neurol (2021)
Keywords: stim1, mutation, stormorken syndrome, thrombocytopenia, tubular aggregate myopathy
MeSH terms: Case Reports

Objective: To identify the gene mutation of Stormorken syndrome and review the published Stromal Interaction Molecule 1 (STIM1) mutation phenotype. Methods: We described the clinical and molecular aspects of a Chinese female with Stormorken syndrome by laboratory tests, muscle biopsies, and genetic analysis. We used this information to summarize all the mutation sites reported in the literature. We also reviewed the clinical features of published cases with a gain of function mutations of STIM1. Results: A 12-year-old Chinese female presented with skin purpura in the lower limbs and stroke-like episodes. Muscle biopsy and microscopic examination revealed atrophy in her skeletal muscle. Genetic analysis identified a novel heterozygous missense mutation, a c.1095G>C transition (NM_003156.3), which caused a p.K365N amino acid substitution in the protein and affected a STIM1-orai1-activation region (SOAR). Conclusions: The novel variant c.1095G>C transition (NM_003156.3) was located in the SOAR, which expands the phenotypic spectrum of STIM1 variants in human disorders and may define the molecular basis of Stormorken syndrome.
An Interesting Unknown Combined Pathology in a Patient with Acute Balance Problem
Authors: Sertac Yetiser, Kutlay Karaman
Journal: Case Rep Otolaryngol (2019)
MeSH terms: Case Reports

A 54-year-old woman with acute-onset nausea and vomiting presented to outpatient clinic. She had headache for 3 weeks. She had difficulty during tandem gait and was falling to the right. Otherwise, her neurological examination was normal. She had normal hearing. VNG analysis revealed spontaneous nystagmus beating to the left with optical fixation. However, she had horizontal and slightly down-beating gaze-evoked nystagmus at primary gaze position. Temporal bone CT and MRI showed widespread encephalitis of the right side of the brain and isolated destruction of the right superior semicircular canal. The patient was treated with high-dose combined antibiotics. She had remarkable recovery within 3 weeks.
Primary Hepatic Neuroendocrine Tumor with Unusual Thyroid Follicular-Like Morphologic Characteristics
Authors: Mohd Elmugtaba Ibrahim, Kerolos Abadeer, Qihui Jim Zhai, Aziza Nassar
Journal: Case Rep Pathol (2017)
MeSH terms: Case Reports

We describe a primary hepatic neuroendocrine tumor of a 57-year-old Thai woman who presented in 2004 with a suspicious mass in the left hepatic lobe. She underwent left hepatectomy for the 10.5-cm mass, called intermediate grade neuroendocrine carcinoma of unknown origin, likely metastatic. The tumor recurred in 2007, then called recurrent primary hepatic neuroendocrine tumor (PHNET), and the patient underwent liver transplant. Because of similarity between the neuroendocrine tumor and a thyroid tumor-specifically, follicular-like characteristics-immunohistochemical stains for thyroglobulin, TTF1, and calcitonin were performed. However, all were negative. All imaging studies revealed no evidence of a primary lesion other than the liver mass. In 2008, the patient's liver transplant failed because of ischemic cholangiopathy, and she underwent a second liver transplant. Seven years later, in 2015, she presented with metastatic neuroendocrine tumor of intermediate grade to the lung, consistent with metastatic PHNET. She underwent left upper-lobe wedge resection to remove the tumor. The patient is alive with no evidence of disease at 13 years after initial diagnosis. This rare variant of PHNET had thyroid-like morphologic characteristics but there is no evidence of primary thyroid tumor or thyroid markers in the primary and recurrent hepatic tumors and lung metastasis.
Acute pseudo-septic arthritis following viscosuplementation of the knee
Authors: Zineb Idrissi, Karima Benbouazza, Maryam Fourtassi, Hanae Raissouni, Meriem El Aadmi, Fatima Zanat, Najia Hajjaj-Hassouni
Journal: Pan Afr Med J (2012)
Keywords: hyaluronic acid, pseudoseptic arthritis, complicaton, knee, viscosupplementation
MeSH terms: Arthritis, Infectious / chemically induced, Hyaluronic Acid / adverse effects, Knee Joint, Viscosupplementation / adverse effects, Viscosupplements / adverse effects, Case Reports, Acute Disease, Aged, Female, Humans, Viscosupplements, Hyaluronic Acid

A 70-year-old woman with a history of medial femoro-tibial compartment of knee osteoarthritis was admitted for acute arthritis six days after a second intra-articular injection of Hyaluronic acid. The joint fluid was inflammatory, with no crystals, and laboratory tests showed marked inflammation leading to antibiotic treatment for suspected septic arthritis. The persistent symptoms and negative results of joint fluid and blood cultures led to discontinuation of the antibiotic therapy after 10 days. Anti-inflammatory with rehabilitation therapy of the knee relieved the symptoms, and the patient was discharged home 3 weeks after her admission. Aseptic arthritis induced by repeated Hyaluronic acid injection is the most likely diagnosis. Physicians should be conscious of this extremely severe complication.
Case reports: Using Good Psychiatric Management (GPM) conceptualizations in the dimensional assessment and treatment of personality disorders
Authors: Martin Blay, Ines Benmakhlouf, Miguel Duarte, Nader Perroud, Christian Greiner, Patrick Charbon, Lois Choi-Kain, Mario Speranza
Journal: Front Psychiatry (2023)
Keywords: gpm, good psychiatric management, borderline personality disorder, case report, narcissistic personality disorder, obsessive-compulsive personality disorder, personality assessment
MeSH terms: Case Reports

Good Psychiatric Management (GPM) is a generalist clinical management approach for borderline personality disorder that incorporates common ingredients of good standard care for any psychiatric diagnosis with what works from prevailing specialist psychotherapies. Similar to all validated therapies for BPD, it relies on a specified formulation of the disorder' symptoms as arising from interpersonal hypersensitivity, to dynamically describe typical patterns of daily self- and interpersonal issues that drive the instability that defines the general personality dysfunction characteristic of the disorder. Recent adaptations of GPM have been proposed for narcissistic personality disorder and obsessive-compulsive personality disorder, with development of similar dynamic models for both (intrapsychic coherence model and model of overcontrol). New dimensional models of personality disorder diagnosis have been developed to address limitations of categorical approach, but the incorporation of these models into usage in the delivery of clinical services (where categorical approach remains the most used) is limited. This paper describes an adaptation of GPM to two cases of personality disorder that illustrate the usefulness of GPM models for dynamic representation of complex daily fluctuations in internal psychic coherence and interpersonal functioning. Specialist psychotherapies will never meet the demands of public health needs to treat personality dysfunction, and incorporation of new dimensional models of diagnosis are needed for treatments that can provide a minimal standard of care for providers and patients.
Glycogenic Hepatopathy in Type 1 Diabetes Mellitus
Authors: Murat Atmaca, Rifki Ucler, Mehmet Kartal, Ismet Seven, Murat Alay, Irfan Bayram, Sehmus Olmez
Journal: Case Reports Hepatol (2015)

Glycogenic hepatopathy is a rare cause of high transaminase levels in type 1 diabetes mellitus. This condition, characterized by elevated liver enzymes and hepatomegaly, is caused by irreversible and excessive accumulation of glycogen in hepatocytes. This is a case report on a 19-year-old male case, diagnosed with glycogenic hepatopathy. After the diagnosis was documented by liver biopsy, the case was put on glycemic control which led to significant decline in hepatomegaly and liver enzymes. It was emphasized that, in type 1 diabetes mellitus cases, hepatopathy should also be considered in the differential diagnoses of elevated liver enzyme and hepatomegaly.