Showing papers in "Journal of Medical Case Reports in 2016"

Removal of dental implant displaced into maxillary sinus by combination of endoscopically assisted and bone repositioning techniques: a case report

Abstract: Background Accidental displacement of a dental implant into the maxillary sinus is an infrequent although not uncommon complication encountered in dental clinical practice, with the main cause thought to be inadequate bone height in the posterior maxilla. We report a case of migration of a dental implant into the maxillary sinus, and discuss the benefits of its removal by a combination of endoscopically assisted and bone repositioning techniques.

Pyomyositis is not only a tropical pathology: a case series

Abstract: Pyomyositis is an acute bacterial infection of skeletal muscle that results in localized abscess formation. This infection was thought to be endemic to tropical countries, and is also known as “tropical pyomyositis”. However, pyomyositis is increasingly recognized in temperate climates and is frequently associated with an immunosuppressive condition, such as human immunodeficiency virus, malignancy, and diabetes mellitus. It is also found in healthy and athletic people after strenuous or vigorous exercise or following localized and possibly unnoticed trauma. It can be primary or secondary to neighboring or remote infection. Primary pyomyositis is a rare condition that can affect children and adolescents. Diagnosis can be delayed because the affected muscle is deeply situated and local signs are not apparent. This delay in diagnosis can result in increased morbidity and a significant mortality rate. The pediatric population, which comprises 35% of the reported pyomyositis cases, is an especially difficult subset of patients to diagnose. In our series, we describe the cases of four previously healthy Caucasian children who were admitted to our Pediatric Department with different clinical presentations. Pyomyositis in our patients was related to factors affecting the muscle itself, including strenuous exercise and direct muscle trauma. Therapy was started with a cephalosporin antibiotic and teicoplanin was subsequently added. The minimum length of therapy was 3 weeks. The diagnosis of pyomyositis in our patients, none of whom were immune-compromised, is confirmation that this disease is not an exclusive pathology of tropical countries and demonstrates that there is an increasing prevalence of pyomyositis in temperate climates.

Case reports in medical education: a platform for training medical students, residents, and fellows in scientific writing and critical thinking.

Abstract: A case report is a detailed narrative that usually illustrates a diagnostic or therapeutic problem experienced by one or several patients. Case reports commonly serve as the first line of evidence for new interventions or they function as alarms that an issue exists with an already established therapy. Case reports are of minor importance in evidence-based medicine; however, they make meaningful contributions to both the knowledge and education of medical students, residents, and fellows. Case reports are written with the goal of sharing information for medical, scientific, or educational purposes. They often serve as medical or even undergraduate students’ first experience with medical writing and they provide a solid foundation for manuscript preparation and publication. In the last few decades, there has been an exponential increase in medical student research, specifically in the number of manuscripts published by medical students. It is important to foster this academic spirit among students by encouraging them to become involved in research. This editorial will focus on the value and educational benefits of writing case reports for medical students, university students, residents, and fellows.

A case-control study of prevalence of anemia among patients with type 2 diabetes.

Abstract: Anemia is defined as a reduction in the hemoglobin concentration of blood, which consequently reduces the oxygen-carrying capacity of red blood cells such that they are unable to meet the body’s physiological needs. Several reports have indicated that anemia mostly occurs in patients with diabetes with renal insufficiency while limited studies have reported the incidence of anemia in people with diabetes prior to evidence of renal impairment. Other studies have also identified anemia as a risk factor for the need for renal replacement therapy in diabetes. Understanding the pathogenesis of anemia associated with diabetes may lead to the development of interventions to optimize outcomes in these patients. The aim of this study was therefore to determine the prevalence of anemia among patients with type 2 diabetes. A total of 100 (50 with type 2 diabetes and 50 controls) participants were recruited for our study. Participants’ blood samples were analyzed for fasting blood glucose, full blood count and renal function tests among others. The prevalence of anemia was then determined statistically. A high incidence of anemia was observed in the cases. Of the patients with diabetes, 84.8 % had a hemoglobin concentration that was significantly less (males 11.16±1.83 and females 10.41±1.49) than the controls (males 14.25±1.78 and females 12.53±1.14). Renal insufficiency determined by serum creatinine level of >1.5 mg/dL, estimated glomerular filtration rate <60 ml/minute/1.73 m2, and erythropoietin levels was also observed to be high in the cases (54.0 %; with mean creatinine concentration of 3.43±1.73 and erythropoietin 6.35±1.28 mIU/mL). A significantly increased fasting blood glucose, urea, sodium, potassium, and calcium ions were observed in the cases (7.99±1.30, 5.19±1.99, 140.90±6.98, 4.86±0.53 and 1.47±0.31 respectively) as compared to the controls (4.66±0.54, 3.56±2.11, 135.51±6.84, 4.40±0.58 and 1.28±0.26 respectively). Finally, a significant association between hemoglobin concentration and fasting blood glucose was also observed in the cases. The findings suggest that a high incidence of anemia is likely to occur in patients with poorly controlled diabetes and in patients with diabetes and renal insufficiency.

Primary hyperparathyroidism in pregnancy treated with cinacalcet: a case report and review of the literature

Abstract: The efficacy and safety of various modes of medical treatment for primary hyperparathyroidism in pregnancy are largely unknown. We report the case of a 34-year-old white woman with primary hyperparathyroidism symptomatic for nephrolithiasis. Her serum calcium was 3.15 mmol/l and parathyroid hormone was 109.0 ng/L. Neck imaging found no pathological parathyroid tissue. Cinacalcet and cholecalciferol were started. She became pregnant 17 months later. The calcimimetic was stopped. During pregnancy, she was admitted for hydration administered intravenously two to three times per week. In her 24th week of pregnancy, cinacalcet was restarted. In her 32nd week, a cesarean section was carried out as planned. Only three cases of primary hyperparathyroidism in women on cinacalcet therapy in pregnancy have been published in the literature. In the present case, hydration was useful in controlling serum calcium. Cinacalcet therapy helped to control serum calcium.

Simvastatin-induced cognitive dysfunction: two case reports

Abstract: Background Simvastatin is commonly prescribed for hypercholesterolemia to reduce vascular risk in patients. Some of these patients have dementia with cognitive defects of several domains. Although protective effects seem to be present, there is emerging evidence that statins cause cognitive impairment. The role of cholesterol in cognitive function is complex. This is reflected in the effects that statins show on cognition functions. The reduction in cholesterol levels seen with statins is effective in improving learning and memory in some patients. However, there is emerging evidence that statins may worsen cognitive function. Similarly, there are major concerns over whether statins alleviate or worsen cognitive problems. The correlation between cholesterol levels and cognitive function is still controversial, mainly due to a lack of robust evidence.

Tapia's syndrome: pathogenetic mechanisms, diagnostic management, and proper treatment: a case series.

Abstract: Background Tapia’s syndrome is an uncommon disease described in 1904 by Antonio Garcia Tapia, a Spanish otolaryngologist. It is characterized by concomitant paralysis of the hypoglossal (XIIth) and pneumogastric (Xth) nerves. Only 69 cases have been described in the literature. Typically, the reported patients presented with a history of orotracheal intubation. Common symptoms are dysphonia, tongue deviation toward the affected side, lingual motility disturbance, and swallowing difficulty.

Considerations for management of interstitial ectopic pregnancies: two case reports

Abstract: Conventional treatment of interstitial pregnancies includes systemic methotrexate, direct methotrexate injection, wedge resection, or hysterectomy. We present two cases of interstitial pregnancies that were successfully managed by different minimally invasive surgical techniques. We also report the novel use of hysteroscopic urologic stone retrieval forceps in the transvaginal removal of persistent products of conception after systemic methotrexate for an interstitial pregnancy. Case 1 was a 28-year-old gravida 1 white woman at 8 weeks gestation; she was diagnosed with a left interstitial pregnancy. After laparoscopic confirmation of the interstitial pregnancy, successful ultrasound-guided suction dilation and curettage was performed. Case 2 was a 33-year-old gravida 3 para 1021 (one term pregnancy, no preterm pregnancies, one ectopic pregnancy and one spontaneous miscarriage, and one living child) Hispanic woman with persistent products of conception after systemic methotrexate for a left interstitial pregnancy. She underwent hysteroscopic-guided removal of the persistent products of conception, which was possible due to novel use of urologic stone retrieval forceps. Successful minimally invasive treatment of interstitial pregnancies may be possible in certain cases. Collaboration between different specialties continues to be important for improving minimally invasive options.

Successful thrombolysis with recombinant tissue plasminogen activator after antagonizing dabigatran by idarucizumab: a case report.

Abstract: Effective anticoagulation routinely precludes patients from receiving intravenous thrombolysis with recombinant tissue plasminogen activator to reverse severe symptoms of ischemic stroke. We report what we believe to be the first case of ischemic stroke successfully treated with recombinant tissue plasminogen activator after antagonizing dabigatran with the monoclonal antibody idarucizumab, recently approved worldwide. A 75-year-old Caucasian man presented to our hospital with severe aphasia and mild hemiparesis. After providing written consent, he received two doses of 2.5 g of idarucizumab over 20 minutes followed by standard protocol in-label recombinant tissue plasminogen activator application. All symptoms resolved within 1 h. Applying a recombinant tissue plasminogen activator after antagonizing dabigatran with idarucizumab is feasible and easy to manage in an emergency room or stroke unit. Thus, idarucizumab represents a new therapeutic option for patients receiving dabigatran treatment, reestablishing their eligibility for recombinant tissue plasminogen activator thrombolysis.

Massive adrenal vein aneurysm mimicking an adrenal tumor in a patient with hemophilia A: a case report and review of the literature

Abstract: Visceral venous aneurysms are exceedingly rare, and until now, there have been no reports of this phenomenon in the adrenal vasculature. This report details the first adrenal venous aneurysm reported in the literature. The aneurysm presented as an 18-cm mass that was initially suspected to be a hematoma or tumor on the basis of the complex medical history of the patient, which included hemophilia A and testicular cancer. After surgical excision, pathologic examination confirmed this mass to be a 15.9-cm adrenal vein aneurysm, the largest aneurysm of any type or location recorded in the medical literature. A 58-year-old caucasian male with hemophilia A presented to the emergency room of another institution with abdominal pain, blood in the stool, and a history of diverticulosis and symptomatic hemorrhoids. A large, left-sided adrenal mass was detected by computed tomography, and because of the patient’s hemophilia A and imaging consistent with a hemorrhagic mass, a hematoma was initially suspected. The patient was transferred to our institution, monitored for further bleeding with a stable hospital course, and discharged from the hospital under close monitoring. After 7–8 weeks with no change in the size of the mass, concerns grew regarding increasing symptoms of both satiety and mass effects from the large anomaly, as well as about the patient’s complicated medical history, which also included cancer. Surgical excision was recommended because of the concerns about increasing symptoms and the possibility of a malignancy. Correction and maintenance of factor VIII levels were incorporated pre-, intra-, and postoperatively, and en bloc surgical resection was performed to minimize bleeding and provide oncologic extirpation of the mass. A bowling ball-sized mass was removed, and careful pathologic examination revealed the mass to be a venous adrenal aneurysm. After a brief hospital stay, the patient made a full recovery. Extensive review of the literature revealed 11 reports of adrenal artery aneurysms but no reported case of an adrenal aneurysm arising from the venous system. Several case reports suggest a correlation between hemophilia and aneurysms. In patients with inherited clotting disorders such as hemophilia A, aneurysms may present in atypical fashions and should be carefully ruled out.

Sternoclavicular joint septic arthritis with chest wall abscess in a healthy adult: a case report

Abstract: Septic arthritis of the sternoclavicular joint is rare. It can be associated with serious complications such as osteomyelitis, chest wall abscess, and mediastinitis. In this report, we describe a case of an otherwise healthy adult with septic arthritis of the sternoclavicular joint with chest wall abscess. A 68-year-old Japanese man presented to our hospital complaining of pain and erythema near the right sternoclavicular joint. Despite 1 week of oral antibiotics, his symptoms did not improve. Computed tomography revealed an abscess with air around the right pectoralis major muscle. After being transferred to a tertiary hospital, emergency surgery was performed. Operative findings included necrotic tissue around the right sternoclavicular joint and sternoclavicular joint destruction, which was debrided and packed open. Methicillin-susceptible Staphylococcus aureus was identified in blood and wound cultures. Negative pressure wound therapy and hyperbaric oxygen therapy were performed for infection control and wound healing. The patient’s general condition improved, and good granulation tissue developed. The wound was closed using a V-Y flap on hospital day 48. The patient has been free of relapse for 3 years. Septic arthritis of the sternoclavicular joint is an unusual infection, especially in otherwise healthy adults. Because it is associated with serious complications such as chest wall abscess, prompt diagnosis and appropriate treatment are required.

Intravascular large B-cell lymphoma associated with silicone breast implant, HLA-DRB1*11:01, and HLA-DQB1*03:01 manifesting as macrophage activation syndrome and with severe neurological symptoms: a case report.

Abstract: Silicone implants have been successfully used for breast augmentation and reconstruction in millions of women worldwide. The reaction to the silicone implant is highly variable; it can lead to local inflammatory symptoms, and sometimes to systemic symptoms and disease. Over 80 cases of anaplastic lymphoma kinase-negative anaplastic large cell lymphoma have been reported in patients with silicone breast implants and have been accepted as a new clinical entity. To the best of our knowledge, an intravascular large B-cell lymphoma associated with a silicone breast implant has not been reported previously. A 48-year-old Caucasian woman who presented with high fever was found to have splenomegaly on physical examination. A laboratory diagnosis revealed pancytopenia, hypertriglyceridemia, and hyperferritinemia. She developed signs of altered sensorium, hemiparesis, aphasia, and cauda equina syndrome. On further evaluation, she fulfilled the necessary five out of eight criteria for diagnosis of macrophage activation syndrome/hemophagocytic lymphohistiocytosis. Dexamethasone administration was followed by prompt improvement; however, 3 days later she again manifested high fever, which persisted despite administration of immunoglobulin and cyclosporine A. Her silicone breast implant was considered a possible contributor to her macrophage activation syndrome and was therefore removed. A histological examination of the capsule tissue showed an extensive lymphohistiocytic/giant cell foreign body reaction suggestive of autoimmune/inflammatory syndrome induced by adjuvants. However, the histological examination unexpectedly also revealed an intravascular large B-cell lymphoma. The genetic background of our patient with silicone breast implants might have predisposed her to three rare and difficult to diagnose syndromes/diseases: macrophage activation syndrome/hemophagocytic lymphohistiocytosis, autoimmune/inflammatory syndrome induced by adjuvants, and intravascular large B-cell lymphoma. The simultaneous manifestation of all three syndromes suggests causal interrelationships. Human leukocyte antigen testing in all women who undergo silicon breast implantation could in the future enable us to better evaluate the risk of potential side effects.

Effect of deoxyribonuclease I treatment for dementia in end-stage Alzheimer’s disease: a case report

Abstract: Alzheimer's disease is the most common cause of dementia and is characterized by a progressive loss of brain tissue leading to amyloid-β accumulation and severe decline in cognitive function. The cause of Alzheimer’s disease is poorly understood, and available treatments are limited in their efficacy, particularly for patients with more severe symptoms. We report the case of a 77-year-old Caucasian man with severe dementia and behavioral disturbance secondary to Alzheimer’s disease treated with memantine who began adjunct treatment with deoxyribonuclease I. Prior to initiation of deoxyribonuclease I treatment, our patient appeared to be in a stuporous state, with a Mini-Mental State Examination score of 3 and a Functional Assessment Staging Test score of 7. After obtaining informed consent from family members, we started administration of 120 mg of deoxyribonuclease I per day (1500 KU/mg) for treatment of severe cognitive impairment. Our patient began to demonstrate rapid, considerable improvement in cognitive function 2 days following initiation of deoxyribonuclease I treatment. After 2 months of continued treatment, Mini-Mental State Examination and Functional Assessment Staging Test scores were 18 and 4, respectively.

Vertebral defect, anal atresia, cardiac defect, tracheoesophageal fistula/esophageal atresia, renal defect, and limb defect association with Mayer-Rokitansky-Küster-Hauser syndrome in co-occurrence: two case reports and a review of the literature

Abstract: The vertebral defect, anal atresia, cardiac defect, tracheoesophageal fistula/esophageal atresia, renal defect, and limb defect association and Mayer-Rokitansky-Kuster-Hauser syndrome are rare conditions. We aimed to present two cases with the vertebral defect, anal atresia, cardiac defect, tracheoesophageal fistula/esophageal atresia, renal defect, and limb defect association and Mayer-Rokitansky-Kuster-Hauser co-occurrence from our local surgical center and through a systematic literature search detect published cases. Furthermore, we aimed to collect existing knowledge in the embryopathogenesis and genetics in order to discuss a possible link between the vertebral defect, anal atresia, cardiac defect, tracheoesophageal fistula/esophageal atresia, renal defect, and limb defect association and Mayer-Rokitansky-Kuster-Hauser syndrome. Our first case was a white girl delivered by caesarean section at 37 weeks of gestation; our second case was a white girl born at a gestational age of 40 weeks. A co-occurrence of vertebral defect, anal atresia, cardiac defect, tracheoesophageal fistula/esophageal atresia, renal defect, and limb defect association and Mayer-Rokitansky-Kuster-Hauser syndrome was diagnosed in both cases. We performed a systematic literature search in PubMed ((VACTERL) OR (VATER)) AND ((MRKH) OR (Mayer-Rokitansky-Kuster-Hauser) OR (mullerian agenesis) OR (mullerian aplasia) OR (MURCS)) without limitations. A similar search was performed in Embase and the Cochrane library. We added two cases from our local center. All cases (n = 9) presented with anal atresia and renal defect. Vertebral defects were present in eight patients. Rectovestibular fistula was confirmed in seven patients. Along with the uterovaginal agenesis, fallopian tube aplasia appeared in five of nine cases and in two cases ovarian involvement also existed. The co-occurrence of the vertebral defect, anal atresia, cardiac defect, tracheoesophageal fistula/esophageal atresia, renal defect, and limb defect association and Mayer-Rokitansky-Kuster-Hauser syndrome is extremely rare. This group of patients has unusual phenotypic characteristics. The long-term outcome after treatment of defects is not well reported. A single unifying cause is not known and the etiology probably includes both genetic and non-genetic causes. We stress the importance of future studies to optimized treatment, follow-up, and etiology.

How to review a case report.

Abstract: Sharing individual patient experiences with clinical colleagues is an essential component of learning from each other. This sharing of information may be made global by reporting in a scientific journal. In medicine, patient management decisions are generally based on the evidence available for use of a particular investigation or technology [1]. The hierarchical rank of the evidence signifies the probability of bias. The higher up the hierarchy, the better its reliability and thus its clinical acceptance (Table 1). Though case reports remain lowest in the hierarchy of evidence, with meta-analysis representing the highest level, they nevertheless constitute important information with regard to rare events and may be considered as anecdotal evidence [2] (Table 1). Case reports may stimulate the generation of new hypotheses, and thus may support the emergence of new research. Table 1 Levels of evidence The definition of a case report or a case series is not well defined in the literature and has been defined variously by different journals and authors. However, the basic definition of a case report is the detailed report of an individual including aspects like exposure, symptoms, signs, intervention, and outcome. It has been suggested that a report with more than four cases be called a case series and those with fewer than four a case report [3]. A case series is descriptive in design. Other authors describe “a collection of patients” as a case series and “a few patients” as a case report [4]. We suggest that should more than one case be reported, it may be defined as a case series—a concept proposed by other authors [5].

Lung disease associated with filamin A gene mutation: a case report.

Abstract: Mutations in the gene encoding filamin A (FLNA) lead to diseases with high phenotypic diversity including periventricular nodular heterotopia, skeletal dysplasia, otopalatodigital spectrum disorders, cardiovascular abnormalities, and coagulopathy. FLNA mutations were recently found to be associated with lung disease. In this study, we report a novel FLNA gene associated with significant lung disease and unique angiogenesis. Here, we describe a 1-year-old Saudi female child with respiratory distress at birth. The child then had recurrent lower respiratory tract infections, bilateral lung emphysema with basal atelectasis, bronchospasm, pulmonary artery hypertension, and oxygen and mechanical ventilation dependency. Molecular testing showed a new pathogenic variant of one copy of c.3153dupC in exon 21 in the FLNA gene. Our data support previous reports in the literature that associate FLNA gene mutation and lung disease.

Management of anterior chamber dislocation of a dexamethasone intravitreal implant: a case report

Abstract: Ozurdex is a 700 mcg dexamethasone intravitreal implant, approved for the management of macular edema secondary to retinal vein occlusion, and other related pathoglogiesAnterior chamber dislocation of Ozurdex represents an uncommon complication of the intravitreal injection, which can be managed by repositioning the implant into the vitreous cavity. We describe the case of a successful repositioning of an Ozurdex implant by mobilization and subsequent balanced saline solution injection in the anterior chamber. An 83-year-old white woman presented to our Emergency Unit complaining of pain and vision loss in herright eye lasting a week. Her anamnesis revealed a history of persistent cystoid macular edema after phacoemulsification with scleral-fixated posterior chamber intraocular lens implantation, recently treated with an intravitreal Ozurdex implant. She also took a long-distance flight 2 days after the injection. An anterior segment examination showed corneal edema and the rod implant adherent to corneal endothelium. To avoid corneal decompensation, we opted for a implant repositioning. Under topical anesthesia, a 30-gauge needle was introduced through a limbar incisionto mobilize the dislocated rod. Balanced saline solution was injected, with a successful repositioning of the implant into the vitreous cavity. Topical 5 % hypertonic saline solution and 0.2 % betamethasone associated with 0.5 % chloramphenicol drops were administered four times a day. To prevent redislocation of the Ozurdex implant, she was instructed to avoid prone position, any kind of physical effort, and not to undertake long-distance flights during the first postoperative week. One week after surgery, an anterior segment examination showed an improvement of corneal edema. Funduscopy showed that the Ozurdex implant was settled into the vitreous cavity. Anterior chamber dislocation of Ozurdex from the vitreous cavity is rare. In our patient, in addition to the posterior capsule tearing, the long-distance flight could have contributed to implant dislocation. Repositioning of the implant is necessary to avoid endothelial decompensation. It can be carried out by using saline balanced solution with the same efficacy as other surgical procedures reported in the literature. A possible disadvantage of this procedure could be implant migration.

Laryngeal inflammatory myofibroblastic tumor (IMT): a case report and review of the literature

Abstract: This case report is interesting as cases of children with laryngeal inflammatory myofibroblastic tumor are not common and previously had been presented as isolated case reports. This is the first case report in Asia describing a laryngeal inflammatory myofibroblastic tumor and its removal using an endoscopic approach. Our patient is a 12-year-old Malay girl from Singapore who presented with hoarseness without respiratory distress. The initial impression was that of a granuloma or a papilloma. We did a biopsy, which confirmed the histology to be inflammatory myofibroblastic tumor, and a magnetic resonance imaging scan showed a contrast-enhanced lesion. The lesion was excised completely using an endoscopic approach. The child was discharged well on the first postoperative day and she has been on follow-up for a year in the clinic. This report highlights the importance of understanding the differential diagnosis for a child with hoarseness. It is not uncommon for a pediatrician, a general practitioner, and a pediatric otolaryngologist to see a child presenting with hoarseness. In most cases, the diagnosis made would be screamer’s nodules, which is commonly seen in children. In a small group, recurrent respiratory papillomatosis form the diagnosis. Over the past few years, the cases of recurrent respiratory papillomatosis have decreased significantly. Laryngeal tumors are not common in children. However, we must maintain a high index of suspicion when we have a child with hoarseness who does not improve with speech therapy and watchful waiting. In such situations, a stroboscope is usually necessary to diagnose the voice problems and to rule out pathological conditions such as laryngeal tumors. If left untreated, the lesion can grow with time and result in a life-threatening airway condition. We also demonstrate our endoscopic technique in this report, and it has proven to be safe with no increased recurrence and much lower morbidity.

Nail gun injuries to the head with minimal neurological consequences: a case series.

Abstract: An estimated 3700 individuals are seen annually in US emergency departments for nail gun-related injuries. Approximately 45 cases have been reported in the literature concerning nail gun injuries penetrating the cranium. These cases pose a challenge for the neurosurgeon because of the uniqueness of each case, the dynamics of high pressure nail gun injuries, and the surgical planning to remove the foreign body without further vascular injury or uncontrolled intracranial hemorrhage. Here we present four cases of penetrating nail gun injuries with variable presentations. Case 1 is of a 33-year-old white man who sustained 10 nail gunshot injuries to his head. Case 2 is of a 51-year-old white man who sustained bi-temporal nail gun injuries to his head. Cases 3 and 4 are of two white men aged 22 years and 49 years with a single nail gun injury to the head. In the context of these individual cases and a review of similar cases in the literature we present surgical approaches and considerations in the management of nail gun injuries to the cranium. Case 1 presented with cranial nerve deficits, Case 2 required intubation for low Glasgow Coma Scale, while Cases 3 and 4 were neurologically intact on presentation. Three patients underwent angiography for assessment of vascular injury and all patients underwent surgical removal of foreign objects using a vice-grip. No neurological deficits were found in these patients on follow-up. Nail gun injuries can present with variable clinical status; mortality and morbidity is low for surgically managed isolated nail gun-related injuries to the head. The current case series describes the surgical use of a vice-grip for a good grip of the nail head and controlled extraction, and these patients appear to have a good postoperative prognosis with minimal neurological deficits postoperatively and on follow-up.

Acute cholestatic hepatitis induced by Epstein-Barr virus infection in an adult: a case report.

Abstract: Acute cholestatic hepatitis without features of infectious mononucleosis is a rare presentation of primary Epstein–Barr infection, with only several cases previously reported in the medical literature. Early investigation for Epstein–Barr virus in febrile patients with deranged liver function tests and no demonstrable biliary obstruction on imaging can expedite both diagnosis and treatment, thereby avoiding costly or invasive procedures such as liver biopsy. A 59-year-old white woman of Anglo-Saxon descent presenting with a febrile illness was noted to have a cholestatic picture of deranged liver function tests. Over the following week a progressive obstructive jaundice developed, with no evidence of choledocholithiasis on ultrasound or magnetic resonance cholangiopancreatography. Specific immunoglobulin M antibodies against Epstein–Barr virus were detected in her serum and the diagnosis of Epstein–Barr hepatitis was confirmed by polymerase chain reaction testing. Supportive treatment was implemented and her liver function had normalized 3 months after presentation. Epstein–Barr virus is associated with a wide variety of clinical manifestations and can present as cholestatic hepatitis with or without features of infectious mononucleosis. While the diagnosis is often suggested by serological testing, Epstein–Barr virus polymerase chain reaction is a new non-invasive laboratory study that can help identify infection in cases where the clinical presentation is atypical.

Hemolytic Uremic Syndrome Associated With Escherichia Coli O157:H7 Infection in Older Adults: A Case Report and Review of the Literature

Abstract: Hemolytic uremic syndrome associated with Shiga toxin-producing Escherichia coli O157:H7 has been widely known as a common cause of acute renal failure in children. There are only a few reports of sporadic Shiga toxin-producing Escherichia coli-hemolytic uremic syndrome in adults in the USA. Analyses from the 2011 outbreak of hemolytic uremic syndrome associated with Escherichia coli O104:H4 reported that mortality rates are highest in those patients with age >60-years old. Therefore, recognizing Shiga toxin-producing Escherichia coli-hemolytic uremic syndrome in older people can help early introduction of the appropriate therapy. We describe an 86-year-old Caucasian woman, initially treated as suspected thrombotic thrombocytopenic purpura, with worsening neurological and renal functions despite plasmapheresis (plasma exchange). A subsequent normal ADAMTS13 activity level and positive stool sample for Escherichia coli O157:H7 confirmed the diagnosis of Shiga toxin-associated hemolytic uremic syndrome. We shifted our management towards aggressive supportive care. Despite conventional treatment, hemolytic uremic syndrome unfortunately led to her death. Our case demonstrates the importance of recognizing Shiga toxin-producing Escherichia coli-hemolytic uremic syndrome as an etiology of microangiopathic hemolytic anemia in older people. According to the current literature, supportive care is the best approach for Shiga toxin-producing Escherichia coli-hemolytic uremic syndrome. Therapies such as plasma exchange and eculizumab (a complement inhibitor) are not shown to be effective in Shiga toxin-producing Escherichia coli-hemolytic uremic syndrome. There is a dire need to continue research to find better treatment options in this disease entity with a high mortality, particularly in older people.

State-of-the-art monitoring in treatment of dengue shock syndrome: a case series

Abstract: Early recognition and treatment of circulatory volume loss is essential in the clinical management of dengue viral infection. We hypothesized that a novel computational algorithm, originally developed for noninvasive monitoring of blood loss in combat casualties, could: (1) indicate the central volume status of children with dengue during the early stages of “shock”; and (2) track fluid resuscitation status. Continuous noninvasive photoplethysmographic waveforms were collected over a 5-month period from three children of Thai ethnicity with clinical suspicion of dengue. Waveform data were processed by the algorithm to calculate each child’s Compensatory Reserve Index, where 1 represents supine normovolemia and 0 represents the circulatory volume at which hemodynamic decompensation occurs. Values between 1 and 0 indicate the proportion of reserve remaining before hemodynamic decompensation. This case report describes a 7-year-old Thai boy, another 7-year-old Thai boy, and a 9-year-old Thai boy who exhibited signs and symptoms of dengue shock syndrome; all the children had secondary dengue virus infections, documented by serology and reverse transcriptase polymerase chain reaction. The three boys experienced substantial plasma leakage demonstrated by pleural effusion index >25, ascites, and >20 % hemoconcentration. They received fluid administered intravenously; one received a blood transfusion. All three boys showed a significantly low initial Compensatory Reserve Index (≥0.20), indicating a clinical diagnosis of “near shock”. Following 5 days with fluid resuscitation treatment, their Compensatory Reserve Index increased towards “normovolemia” (that is, Compensatory Reserve Index >0.75). The results from these cases demonstrate a new variation in the diagnostic capability to manage patients with dengue shock syndrome. The findings shed new light on a method that can avoid possible adverse effects of shock by noninvasive measurement of a patient’s compensatory reserve rather than standard vital signs or invasive diagnostic methods.

Papillon-Lefèvre syndrome: a series of five cases among siblings.

Abstract: Papillon–Lefevre syndrome is a rare autosomal recessive disorder characterized by palmoplantar hyperkeratosis and aggressively progressing periodontitis leading to premature loss of deciduous and permanent dentition. The etiopathogenesis of the syndrome is relatively obscure, and immunologic, genetic, or possible bacterial etiologies have been proposed. A series of five cases of Papillon–Lefevre syndrome among the siblings in a family is presented here: a 3-year-old Arab girl, a 4-year-old Arab boy, a 11-year-old Arab boy, a 12-year-old Arab boy, and a 14-year-old Arab boy. The patients presented with severe gingival inflammation and mobility of teeth. The clinical manifestations were typical of Papillon–Lefevre syndrome and the degree of involvement of the oral and skin conditions varied among them. This case series stresses the consanguinity in the family as an etiologic factor. All siblings in the family were affected with Papillon–Lefevre syndrome which makes this a rare case. A multidisciplinary approach with the active participation of a dental surgeon, dermatologist, and pediatrician is essential for the management of cases of Papillon–Lefevre syndrome.

Spontaneous splenic rupture associated with apixaban: a case report

Abstract: Spontaneous splenic rupture associated with anticoagulant use is a rare but potentially lethal disorder. Lack of prompt recognition can be associated with poor patient outcomes. The use of novel oral anticoagulants is becoming more common and thus consideration of this disorder while evaluating a patient who presents with abdominal pain while using these agents is extremely important. This is the first reported case of spontaneous splenic rupture associated with apixaban. We describe the clinical case of an 83-year-old white man who complained of sudden severe abdominal pain 5 days into a hospital stay for acute-on-chronic congestive heart failure and exacerbation of chronic obstructive pulmonary disease. Neither he nor his wife reported any significant trauma for the past 6 months prior to his admission. His medical history included chronic atrial fibrillation treated with medications including apixaban 2.5 mg twice daily. An urgent abdominal computed tomography scan demonstrated a large splenic hematoma and evidence of intraperitoneal bleeding from which he rapidly declined, developing hypovolemic shock. An emergency splenic arteriogram displayed a patent splenic artery and an embolization was successful in stabilizing him. Due to evidence of recurrent bleeding, an exploratory laparotomy and splenectomy was subsequently performed the following day. The diagnosis of spontaneous splenic rupture is important to consider in a patient using apixaban who presents with abdominal pain and associated signs of hypotension and anemia. For hemodynamically unstable patients, prompt treatment to stop significant bleeding through splenic artery embolization or splenectomy is warranted and may be lifesaving.

IgG4-related disease presenting with an epidural inflammatory pseudotumor: a case report

Abstract: Inflammatory pseudotumor is a rare clinical condition that can be related to immunoglobulin G4 disease. Only a few cases of spinal inflammatory pseudotumors have been reported in the literature and an association with immunoglobulin G4 disease was not conclusive in any of them. We describe what we believe to be the first biopsy-proven case of an epidural inflammatory pseudotumor related to immunoglobulin G4 disease. A 57-year-old Caucasian woman presented to our hospital with severe paraparesis, gait disturbance, and sensory loss secondary to a relapsing epidural mass. Examination of a biopsy specimen revealed a lymphoplasmacytic infiltration with fibrosis and an immunoglobulin G4-positive plasma cell ratio of over 50 %, which are compatible with a diagnosis of immunoglobulin G4-related inflammatory pseudotumor. Our patient was successfully treated with systemic and epidural administration of glucocorticoids. Immunoglobulin G4-related disease is an emerging clinical condition in which central nervous system involvement is still uncommon. We describe the case of a patient with an epidural mass with medullar compression, which was proved to be an immunoglobulin G4-related epidural inflammatory pseudotumor. Our findings suggest a new manifestation of immunoglobulin G4-related disease. This disorder should be considered in the differential diagnosis of spinal tumors as a potentially treatable condition with glucocorticoids.

Eccrine porocarcinoma of the vulva: a case report and review of the literature

Abstract: Malignant tumors arising from the vulva account for only 0.6 % of all cancers in female patients. The predominant histologic type, representing about 90 % of these malignancies, is squamous cell carcinoma. Eccrine porocarcinoma is a rare malignant tumor arising from sweat glands. The incidence of eccrine porocarcinoma is estimated at 0.005–0.01 % of all cutaneous tumors. To the best of our knowledge, only seven previous cases of vulvar eccrine porocarcinoma have been reported in the English-language literature. We present the case of a patient with eccrine porocarcinoma of the vulva, and we summarize the clinical features of this disease using seven previously reported cases. A 54-year-old Japanese woman visited a local hospital complaining of fever and left vulvar pain for 2 months. An initial examination revealed a 1 × 1 cm, firm, ulcerative mass in the inner aspect of the left labium minorum. With a preoperative diagnosis of vulvar squamous cell carcinoma, we performed a radical local excision followed by bilateral inguinal lymphadenectomy. Histological examination showed eccrine porocarcinoma, stage IB (T1bN0M0). Radiation therapy with weekly cisplatin administration was then given as adjuvant therapy. One month after treatment was completed, computed tomography revealed multiple metastases in the bilateral lungs and in the sacral bone. The patient received three courses of chemotherapy (paclitaxel and carboplatin) and underwent palliative radiation therapy to the sacrum. She died of her disease 12 months after surgery. We report the case of a patient with eccrine porocarcinoma of the vulva and summarize the clinical features and the treatment options of eccrine porocarcinoma from a few retrospective case reports. Although eccrine porocarcinoma is a rare disease, clinicians and pathologists should be aware of its clinical and histological features and its biological behavior.

Osseous choristoma of the tongue: two case reports

Abstract: Osseous choristoma is a very rare, benign lesion in the maxillofacial region. It appears as a benign mass of normally matured bony tissue covered by the normal epithelium of the tongue. It is usually seen in front of the foramen cecum of the tongue. Surgical excision is the treatment of choice with an excellent prognosis and there have been very few cases of recurrence. Here we present two cases of osseous choristoma on the dorsum of the tongue. Case 1 was a 15-year-old Japanese girl who presented with a painless but gradually growing swelling on the dorsum of her tongue approximately 1 year before her admission. Case 2 was a 21-year-old Japanese woman with a complaint of pain in the lower left, posterior side of her mouth. Histological findings showed that both lesions were composed of well-organized, mature, compact bone beneath the oral mucosal membrane. Subsequent to simple surgical excision, no recurrence of the lesions was observed after the follow-up period. Previous literatures have proposed both malformation and trauma hypotheses as the etiopathologies of osseous choristoma. However, the histopathological findings of the two cases in the present study do not support the trauma hypothesis. Although osseous choristoma is clinically a benign condition, the underlying histopathological processes are important. The outcome of aberrant formation of calcified tissue in the vicinity of vital structures such as nerves and blood vessels may be of clinical significance.

Anti-N-methyl-D-aspartate receptor encephalitis with an imaging-invisible ovarian teratoma: a case report.

Abstract: Background Anti-N-methyl-D-aspartate receptor encephalitis is a recently discovered disease entity of paraneoplastic limbic encephalitis. It largely affects young women and is often associated with an ovarian teratoma. It is a serious yet treatable condition if diagnosed early. Its remedy involves immunotherapy and surgical removal of the teratoma of the ovaries. This case of anti-N-methyl-D-aspartate receptor encephalitis involves an early surgical intervention with bilateral oophorectomy, despite negative imaging evidence of a teratoma.

Pancreatic metastases from ocular malignant melanoma: the use of endoscopic ultrasound-guided fine-needle aspiration to establish a definitive cytologic diagnosis: a case report.

Abstract: When encountering solid pancreatic lesions, nonpancreatic primary metastases are rare and differentiating a metastasis from a primary neoplastic lesion is challenging. The clinical presentation and radiologic features can be similar and the possibility of a pancreatic metastasis should be considered when the patient refers to a history of a different primary cancer. Endoscopic ultrasound offers a key anatomical advantage in accessing the pancreas and endoscopic ultrasound-guided fine-needle aspiration has become the gold standard method for diagnosing pancreatic lesions. A 58-year-old white Hispanic woman with a history of uveal malignant melanoma, presented with abdominal pain and jaundice. On admission, laboratory tests were performed (her total bilirubin was 6.37 mg/dL with a direct fraction of 5.30 mg/dL). Cross-sectional, abdominal computed tomography with contrast, showed a low-attenuating lesion localized in the pancreatic head (measuring 4 × 3 cm) and a thinner section of the distal bile duct suspicious for compression. Our patient was scheduled for an endoscopic ultrasound-guided fine-needle aspiration to establish a diagnosis. Endoscopic ultrasound showed a solid, hypoechoic, well-defined lesion with regular contours (measuring 3.17 × 2.61 cm), localized between the head and neck of the pancreas. Endoscopic ultrasound-guided fine-needle aspiration was performed with a 22G needle and cytology confirmed the diagnosis of metastatic melanoma. Our patient subsequently underwent right orbital exenteration, followed by duodenopancreatectomy without complications. At the moment our patient is receiving adjuvant chemotherapy at an outside oncology clinic. To the best of our knowledge, this is a very rare presentation of an ocular malignant melanoma with an isolated pancreatic metastasis causing symptomatic biliary obstruction. Endoscopic ultrasound-guided fine-needle aspiration has proven to be the best method to diagnose solid pancreatic lesions. In this particular case, cytology was essential in confirming the diagnosis and guiding the most adequate therapy, which was a pancreatic resection, ocular exenteration of the melanoma, followed by adjuvant chemotherapy.

Subependymal giant cell astrocytoma (SEGA): a case report and review of the literature

Abstract: Background Subependymal giant cell astrocytoma is a rare tumor that occurs in the wall of the lateral ventricle and foramen of Monro and, rarely, in the third ventricle. It is one of the intracranial lesions found in tuberous sclerosis complex (which include subependymal nodules, cortical tubers, retinal astrocytoma and subependymal giant cell astrocytoma), but cases without such lesions have also been reported in the literature. It was described for the first time in 1908 by Vogt as part of the typical triad of tuberous sclerosis complex. At the 2012 Washington Consensus Conference, it was decided by the invited expert panel to document the definition of subependymal giant cell astrocytoma as a lesion at the caudothalamic groove with either a size of more than 1 cm in any direction or a subependymal lesion at any location that has shown serial growth on consecutive imaging regardless of size. Most subependymal giant cell astrocytomas will show avid enhancement after contrast administration; however, a growing subependymal lesion even in the absence of enhancement should be considered a subependymal giant cell astrocytoma.