OpenMed/multicare-articles huggingface dataset:
Alpha II Antiplasmin Deficiency Complicating Pregnancy: A Case Report
Authors: Brenda Dawley
Journal: Obstet Gynecol Int (2011)
MeSH terms: Case Reports

Background. Alpha II antiplasmin is a protein involved in the inhibition of fibrinolysis. A deficiency in this protein leads to increased hemorrhage. It is inherited in an autosomal recessive fashion. Case. 30-year-old Gravida 1, Para 0, presented for prenatal care with her first and subsequently her second pregnancy. Her medical history was significant for a known deficiency in alpha II antiplasmin. Her first and second pregnancies were complicated by nonobstetrical hemorrhage requiring transfusions and severe preeclampsia requiring preterm deliveries. Conclusion. Alpha II antiplasmin deficiency resulted in multiple episodes of nonobstetrical hemorrhages requiring transfusion and ultimately preterm deliveries due to severe preeclampsia. Both infants and mother had a good outcome. The presence of this disorder may require a multidisciplinary team approach involving obstetricians, pediatricians, and hematologists. Precis. Alpha II antiplasmin deficiency is a rare autosomal recessive disorder leading to increased fibrinolysis and hemorrhage. We present a case report of a pregnancy complicated by this disorder.
Exome-first approach identified novel INDELs and gene deletions in Mowat-Wilson Syndrome patients
Authors: Maria Florencia Gosso, Cristian Rohr, Bianca Brun, Guadalupe Mejico, Fernanda Madeira, Fabian Fay, Melina Klurfan, Martin Vazquez
Journal: Hum Genome Var (2018)
MeSH terms: Case Reports

Mowat-Wilson syndrome (MWS) is characterized by severe intellectual disability, absent or impaired speech and microcephaly, with a gradual post-natal onset. The syndrome is often confused with other Angelman-like syndromes (ALS) during infancy, but in older children and adults, the characteristic facial gestalt of Mowat-Wilson syndrome allows it to be distinguished easily from ALS. We report two cases in which an exome-first approach of patients with MWS identified two novel deletions in the ZEB2 gene ranging from a 4 base deletion (case 1) to at least a 573 Kb deletion (case 2).
Epithelial-Myoepithelial Carcinoma of the Base of Tongue with Possible Lung Metastases
Authors: Michael Y Chen, Vipul Vyas, Ryan Sommerville
Journal: Case Rep Otolaryngol (2017)
MeSH terms: Case Reports

Background:
Epithelial-myoepithelial carcinomas are rare neoplasms usually arising from the salivary glands. There is limited evidence in the literature on their prognosis in the base of the tongue but other cases have resolved without recurrence.
Methods:
The patient underwent biopsies demonstrating the diagnosis of epithelial-myoepithelial carcinoma of the base of tongue and a PET scan showed multiple bilateral rounded pulmonary nodules.
Results:
The patient declined chemotherapy and radiotherapy to maximise his quality of life and passed away under management from palliative care several months later.
Discussion:
This is the only case in the literature of this type of carcinoma in the base of the tongue resulting in metastases and a poor prognosis. The case highlights the importance of checking for metastases in such lesions and their potentially serious outcomes if left untreated.
Nodulocystic basal cell carcinoma on the limb masquerading as vascular malformation on imaging
Authors: Paul Brian Ng Hung Shin, Bibiana Tie, Vijay Kanagarajah, Ailin Tan
Journal: J Surg Case Rep (2023)
Keywords: basal cell carcinoma, nodulocystic, vascular malformation
MeSH terms: Case Reports

Nodulocystic basal cell carcinoma (BCC) is a cystic variant of BCC, which can easily be misdiagnosed. We report a case of a 52-year-old man with a nodulocystic BCC that appeared at the site of a previously excised BCC 9 years prior. It examined as a benign cyst with a radiological signature of a vascular malformation. It was histologically confirmed on fine needle aspirate (FNA) and excisional biopsy to be a nodulocystic BCC. BCC is one of the most common paraneoplastic neoplasms affecting photo-exposed areas and displaying many variants. Nodulocystic BCC is a rarer variant that may have more than one dermoscopic face and can appear macroscopically benign. Given its malignant potential, it is imperative that it is accurately diagnosed. We highlighted that nodular cystic BCC can easily be misdiagnosed. Careful history and FNA are key differentiators to establish the correct diagnosis.
Severe carvedilol toxicity without overdose - caution in cirrhosis
Authors: Satish Maharaj, Karan Seegobin, Julio Perez-Downes, Belinda Bajric, Simone Chang, Pramod Reddy
Journal: Clin Hypertens (2017)
Keywords: beta blocker, carvedilol, cirrhosis, glucagon, toxicity, treatment
MeSH terms: Case Reports

Background:
Carvedilol is used in the management of hypertension, ischemic heart disease, heart failure and most recently, portal hypertension. It has been associated with improved outcomes regarding variceal bleeding, hepatic decompensation and death when compared to propranolol and endoscopic band ligation. The main cause of portal hypertension is cirrhosis and therefore carvedilol is increasingly used in these patients. Due to its extensive hepatic metabolism, carvedilol is contraindicated in severe hepatic impairment. However, there are no dosage adjustments in the manufacturer's labelling for mild to moderate hepatic impairment.
Case presentation:
We present a case of cardiogenic shock that occurred after carvedilol 25 mg orally was administered to a patient with cirrhosis. As there was no overdose, the diagnosis was based on clinical recognition of the toxidrome. The patient was successfully treated with glucagon 5 mg bolus followed by infusion.
Conclusions:
Patients with cirrhosis represent a special at-risk group for beta blocker toxicity. The typical threshold for carvedilol toxicity in overdose is 50 mg but in patients with cirrhosis this is not applicable. Nurses and physicians need to recognize the toxidrome early. Hospitals where carvedilol is used in patients with cirrhosis should have glucagon in formulary at doses to treat toxicity (bolus and infusion). Finally, dose adjustment and slow uptitration of carvedilol in cirrhosis is recommended.
Aortosternal Venous Compression: A Review of Two Cases
Authors: Victoria Giglio, Zain Badar, Yasovineeth Bhogadi, Brian Van Adel, Gordon Yip
Journal: Case Rep Med (2022)
MeSH terms: Case Reports

Aortosternal venous compression (AVC) is a rare venous compression syndrome that involves brachiocephalic venous compression due to its positioning between the sternum and the aorta. One of the features of AVC involves compression of the left innominate vein with variability in luminal caliber on inspiration and expiration. Imaging modalities such as computed tomography (CT) examination can aid in initial diagnosis; however, venography can be utilized for confirmatory diagnosis due to its higher specificity during the inspiratory and expiratory phases. Through findings demonstrated during venography, we herein present two cases of confirmed AVC secondary to an aberrant right subclavian artery. Characteristic imaging features in the diagnosis of AVC and its etiology are discussed.
Pediatric-isolated auricular erythromelalgia: a case report
Authors: Kelly Grandy, Gerard Corsten, Paul Hong
Journal: Case Rep Pediatr (2012)

Erythromelalgia is a rare disorder that typically affects the skin of the feet, hands, or both, that is characterized by red skin, warmth, and a burning quality of pain. It usually affects both sides of the body, but may manifest unilaterally. Cooling of the affected areas usually results in symptom relief. We report a case of a young boy with erythromelalgia of the ears.
Video-assisted Thoracoscopic Resection of a Giant Bulla in Vanishing Lung Syndrome: case report and a short literature review
Authors: Kobe Van Bael, Mark La Meir, Hans Vanoverbeke
Journal: J Cardiothorac Surg (2014)
MeSH terms: Pulmonary Emphysema / surgery, Thoracic Surgery, Video-Assisted / methods, Case Reports, Review, Adult, Diagnosis, Differential, Follow-Up Studies, Humans, Male, Pleura / surgery, Pulmonary Emphysema / diagnosis, Radiography, Thoracic, Syndrome, Tomography, X-Ray Computed

A 36-year-old Caucasian man was admitted to our hospital with acute onset of left-sided chest pain. Computed Tomography confirmed the presence of a giant bulla on the apex of the lower lobe of the left lung. A video-assisted thoracic surgery (VATS) with bullectomy was performed using two linear endostaplers. Additionally pleurectomy was performed. No serious complications occurred in the postoperative course, as the patient showed good lung re-expansion and no prolonged air leakage.VATS bullectomy is a suitable and eminent technique to approach giant bullous emphysema and definitely fulfils a role in its treatment.
An unusual case of ectomesenchymal chondromyxoid tumour of the left tonsillar bed: imaging and histopathologic features
Authors: Alessandro Stecco, Martina Quagliozzi, Massimiliano Pino, Paolo Spina, Franco Pia, Renzo Boldorini, Alessandro Carriero
Journal: BJR Case Rep (2016)
MeSH terms: Case Reports

We describe a case of a rare soft-palatal and parapharyngeal mass in an adult woman. A 71-year-old female presented with a huge mass protruding from the soft palate, complaining about difficulty in swallowing for the past 4 months. After inspection and ear nose and throat fibroscopy, in which the mass appeared regular-shaped and with a regular mucosa, the patient underwent a CT scan and MRI examination. The CT scan showed an oval, not-infiltrating, hypodense mass including a discrete irregular-shaped central calcification, while the MRI examination confirmed the non-infiltrating growth and showed that around the calcified core, the lesion was surrounded by fat. After surgical removal, the histopathologic diagnosis was that of an "ectomesenchymal chondromyxoid tumour". Ectomesenchymal chondromyxoid tumour is a rare benign neoplasm arising from the tongue. Although only 45 cases have been reported in the literature, there are several unique features that define this lesion. Ours is the first case with a complete CT scan and MRI with diffusion imaging description.
Detection of a cardiac mass by [18F]FDG-PET/CT: a rare case
Authors: A Mallia, Ll Travaini, G Trifiro, G Paganelli
Journal: Ecancermedicalscience (2009)
MeSH terms: Case Reports

Intra-cardiac masses present an important problem in cardiology. The differential diagnoses includes tumours, which may be primary (benign or malignant) or metastatic, and infected mural thrombi.Myxomas, sarcomas, breast, lung and renal cancer represent the commonest causes of primary benign, malignant and metastatic intra-cardiac masses, respectively.Recent studies have shown that cardiac involvement in malignant lymphoma is common but under-investigated.Diagnostic imaging techniques for detection of cardiac masses include echocardiography, CT and MRI, with echocardiography having the highest sensitivity. We propose that 18-F-PET/CT may play an important role in the detection and evaluation of intra-cardiac masses.
Primary ciliary dyskinesia complicated with diffuse panbronchiolitis: a case report and literature review
Authors: Wei Chen, Changzhou Shao, Yuanlin Song, Chunxue Bai
Journal: Clin Respir J (2014)
Keywords: case report, clinical profiles, diffuse panbronchiolitis, literature review, primary ciliary dyskinesia
MeSH terms: Bronchiolitis / complications, Bronchiolitis / diagnosis, Haemophilus Infections / complications, Haemophilus Infections / diagnosis, Kartagener Syndrome / complications, Kartagener Syndrome / diagnosis, Case Reports, Review, Adult, Bronchiolitis / therapy, Female, Haemophilus Infections / therapy, Humans, Kartagener Syndrome / therapy, Diffuse panbronchiolitis

Background:
Fifty percent of patients with primary ciliary dyskinesia (PCD) have situs inversus. Diffuse panbronchiolitis (DPB) might be one of the characteristic features of the lung in PCD.
Methods:
We reported a case of PCD without situs inversus, yet complicated with DPB, and did literature review.
Results:
A 34-year-old nonsmoking Chinese woman with 6-year primary infertility suffered from recurrent episodes of respiratory tract infections since childhood. Lung auscultation revealed end-inspiratory coarse crackles. Pulmonary function tests demonstrated mild obstructive ventilation functional impairment. Lung biopsy showed respiratory bronchiolitis. Nasal mucosa cilia showed the absence of both outer and inner dynein arms of the microtubules. Saccharin test was positive. Chest images showed bronchiectasis and bronchiolitis but no situs inversus. Paranasal sinus computed tomography (CT) showed maxillary sinusitis and ethmoid sinusitis. A culture of bronchoalveolar lavage fluid was positive for Pseudomonas aeruginosa. Her conditions improved in clinical symptoms and CT images after 2 months of treatment with azithromycin. Literature review revealed that very rare patients were diagnosed as PCD complicated with diffuse DPB, and all of them had situs inversus.
Conclusions:
The association of DPB might be one of the characteristic features of the lung in PCD. Further studies on the concurrence of these two diseases are suggested so as to elucidate the mechanism of both.
Successful Treatment of Cutaneous Curvularia geniculata, Nocardia niigatensis, and Viral Papillomatosis in a Dog During the Therapeutic Management of Immune-Mediated Hemolytic Anemia
Authors: Emily Strzok, Chris Siepker, Abigail Armwood, Elizabeth Howerth, Joanne Smith, Frane Banovic
Journal: Front Vet Sci (2019)
Keywords: canine, dermatology, immunosuppression, opportunistic skin infections, skin
MeSH terms: Case Reports

Opportunistic infections represent a major cause of mortality in immunocompromised patients. Discontinuation or reduction of immunosuppressive medications is recommended with the development of opportunistic infections, which may cause a relapse or worsening of the immune-mediated disease. A 7.5-year-old, spayed female great Dane was diagnosed with immune-mediated hemolytic anemia with initial immunosuppressive therapy consisting of oral prednisone, ciclosporin and mycophenolate mofetil. The patient developed diffuse right forelimb pyogranulomatous fungal dermatitis with deep draining tracts 6 weeks into immunosuppressive treatment with Curvularia geniculata growth. Oral once daily terbinafine and itraconazole were initiated; ciclosporin was immediately discontinued and the mycophenolate mofetil/prednisone doses were reduced. The right forelimb skin lesions resolved after 4 weeks, but the patient presented with a diffuse severe neutrophilic dermatitis on the left forelimb; 16S rRNA sequencing identified Nocardia niigatensis. Cutaneous nocardiosis was treated with oral enrofloxacin and doxycycline; systemic immunosuppressive therapies were continued for immune-mediated hemolytic anemia control. One month later, the left forelimb lesions completely resolved but the patient developed several multifocal, exophytic warts; the clinical features and histopathology were consistent with viral papillomas. Within the following 4 weeks, the patient developed severe diffuse papillomatosis of the left forelimb, which was successfully treated with 2 weeks of every other day topical imiquimod administration. In this case, successful treatment of cutaneous opportunistic bacterial, fungal and viral infection was possible with proper treatment even though the immunosuppressive drug treatments could not be discontinued.
Papular granuloma annulare of palms and soles: case report of a rare presentation
Authors: Sidharth Sonthalia, Rahul Arora, Rashmi Sarkar, Uday Khopkar
Journal: F1000Res (2014)

We report the case of a 44-year-old Indian male patient who presented with mildly tender isolated papular lesions confined to the palms of the hands and soles of the feet. The histopathology was characteristic of granuloma annulare. There was an excellent response with 4-week treatment with a potent topical steroid ointment and no recurrence was reported at the follow-up one year later. This report is interesting because of the rare presentation of a common disease.
Case report: a 37-year-old male with telangiectasias, polycythemia vera, perinephric fluid collections, and intrapulmonary shunting
Authors: Javed Khan, David B Sykes
Journal: BMC Hematol (2014)
Keywords: erythrocytosis, erythropoietin, monoclonal gammopathy, perinephric fluid, polycythemia, tempi
MeSH terms: Case Reports

Background:
The TEMPI syndrome was recently described in 2011, and is characterized by the constellation of five hallmarks: Telangiectasias, Erythrocytosis and elevated Erythropoietin, Monoclonal gammopathy, Perinephric fluids collections, and Intrapulmonary shunting. The underlying pathophysiology is unknown, though it has been postulated that the monoclonal gammopathy may play a causal role.
Case presentation:
A 37-year-old non-smoking male presented to our institution with a fever and the sensation of fullness in the right flank. His exam was notable for telangiectasias, clubbing of the fingernails, plethora, and a palpable bulge in the right flank. Renal ultrasound demonstrated bilateral perinephric fluid collections. Laboratory evaluation revealed erythrocytosis with low serum erythropoietin, and testing for the JAK2V617F mutation was positive, confirming a diagnosis of polycythemia vera. Though his room air saturation was normal at rest, it decreased dramatically with exercise, felt to be secondary to microscopic intrapulmonary shunting. The patient's presentation is very similar to that of the TEMPI syndrome, a very rare syndrome of which there have been six published cases. In contrast to the TEMPI syndrome where the erythrocytosis is driven by highly elevated serum erythropoietin, our patient was found to have polycythemia vera. Also in contrast to the other patients with TEMPI syndrome, our patient did not have an identifiable monoclonal gammopathy. Our patient responded to treatment with hydroxyurea. His erythrocytosis, perinephric fluid collections, and telangiectasias resolved over the course of six months. The intrapulmonary shunting has continued to gradually improve with treatment, suggesting that this is an entirely reversible process.
Conclusion:
Our case is the first to describe the combination of polycythemia vera, telangiectasias, perinephric fluid collections, and intrapulmonary shunting. The presentation is highly similar to the previously described TEMPI syndrome, though calls into question the potential importance of the monoclonal gammopathy. Our patient demonstrated a response to treatment with hydroxyurea, while patients with the TEMPI syndrome have shown responses to plasma-cell directed therapies such as bortezomib.
A case report of ruptured ectopic pregnancy plus massive hemoperitoneum on a heterotrophic pregnancy in a resource-poor setting, Mbengwi, Cameroon
Authors: Nzozone Henry Fomukong, Edwin Ngouagna, Mandeng Ma Linwa Edgar, Claude Ngwayu Nkfusai, Fala Bede, Frankline Sevidzem Wirsiy, Samuel Nambile Cumber
Journal: Pan Afr Med J (2021)
Keywords: heterotopic, case report, ectopic, pregnancy, rupture
MeSH terms: Hemoperitoneum / etiology, Pregnancy, Heterotopic / diagnostic imaging, Salpingectomy / methods, Case Reports, Abdominal Pain / etiology, Adult, Cameroon, Female, Humans, Laparotomy / methods, Pregnancy, Rupture, Spontaneous / diagnostic imaging, Ultrasonography, Prenatal

Heterotopic pregnancy is a rare obstetrics phenomenon and carries significant maternal morbidity and mortality due to the risk of rupture of the ectopic pregnancy. Physicians tend to feel comfortable and relieved when an intrauterine gestation sac is seen. This results in an inadequate inspection of the adnexae and remaining structures during emergency bedside ultrasound despite a strong initial clinical suspicion of ectopic pregnancy. We present a case report of ruptured ectopic pregnancy and massive hemoperitoneum in a patient with heterotopic pregnancy. The diagnosis was done on bedside ultrasonography in a clinically unstable 32-year-old patient with a history of infertility. She presented with acute abdominal pain, body weakness, and amenorrhea. She underwent emergency laparotomy and salpingectomy. In our context where ultrasound is not readily available, practitioners carrying out salpingectomy for ruptured ectopic pregnancies should bear in mind the plausibleness of heterotopic pregnancy to properly handle the uterus.
Malignant Hyperthermia and Idiopathic HyperCKemia
Authors: Pashtoon Murtaza Kasi
Journal: Case Rep Med (2011)
MeSH terms: Case Reports

Malignant hyperthermia (MH) is a rare but life-threatening condition that is more frequently encountered and discussed within the anesthesia literature. Here we through a case specifically discuss the susceptibility of individuals and/or families with asymptomatic unexplained elevations of creatine kinase (CK), also frequently referred to as hyperCKemia or idiopathic hyperCKemia (IHCK) in recent reports. The clinical implications would be to underscore the importance of this as a susceptibility to developing MH and highlight the importance of genetic susceptibility testing in such cases. Anesthesiologists and critical care intensivists as well as primary care physicians should keep this in mind when seeing patients with asymptomatic hyperCKemia and potentially inform them about the possibility of developing MH if exposed to triggering agents. Genetic susceptibility testing should be considered if available and family members should also receive nontriggering agents when undergoing anesthesia and wear Medic Alert tags.
How Do Institutional Conflicts of Interest Between Pharmaceutical Companies and the Healthcare Sector Become Corrupt? A Case Study of Scholarship Donations Between Department of Clinical Anesthesiology, Mie University, and Ono Pharmaceutical in Japan
Authors: Akihiko Ozaki, Anju Murayama, Kayo Harada, Hiroaki Saito, Toyoaki Sawano, Tetsuya Tanimoto, Piotr Ozieranski
Journal: Front Public Health (2022)
Keywords: japan, mie university, anesthesiology, conflicts of interest, ethics, pharmaceutical companies
MeSH terms: Anesthesiology, Conflict of Interest, Drug Industry, Fellowships and Scholarships, Health Care Sector, Humans, Japan, Pharmaceutical Preparations, Universities, Pharmaceutical Preparations

Institutional conflicts of interest (ICOIs) with pharmaceutical companies can bias internal operation of healthcare organizations. Naturally, a scholarship donation-which is a donation scheme unique to Japan, provided to healthcare organizations and their subunits to encourage educational and academic activities related to the development of new drugs-fall into the ICOI category. While anecdotal evidence exists that scholarship donations have been used as bribes by pharmaceutical companies, there has been little case study research that would illuminate the workings of this "gray area" mechanism. From this perspective, we offer an in-depth analysis of a recent scandal involving the Department of Clinical Anesthesiology, Mie University and Ono Pharmaceutical, where a scholarship donation was used by a pharmaceutical company to increase the prescription of one of its key drugs at a hospital department. Available evidence also suggests that a professor based within the department originally requested a scholarship donation from the company, which became an initial trigger of the scandal. We argue that by scrutinizing scholarship donations we can gain insight into problems specific to ICOIs between the pharmaceutical companies and the healthcare sector in Japan. In addition, scholarship donations can be understood as a form of "gifts" which have been found to underpin certain forms of pharmaceutical companies' promotional activities in Japan but also in other countries. We conclude by highlighting potential institutional remedies, which may alleviate ICOIs and corrupt behavior affecting the healthcare sector.