State of Alaska epidemiology bulletin: Scombroid fish poisoning: First case reports in Alaska

Abstract Background Fetus in fetu is a rare congenital anomaly. The exact etiology is unclear; one of the mostly accepted theories is the occurrence of an embryological insult occurring in a diamniotic monochorionic twin leading to asymmetrical division of the blastocyst mass. Commonly, they present in the infancy with clinical picture related to their mass effect. About 80% of cases are in the abdomen retroperitoneally. Case presentation We present two cases of this rare condition. The first case was for a 10-year-old girl that presented with anemia and abdominal mass, while the second case was for a 4-month-old boy that was diagnosed antenatally by ultrasound. Both cases had vertebrae, recognizable fetal organs, and skin coverage. Both had a distinct sac. The second case had a vascular connection with the host arising from the superior mesenteric artery. Both cases were intra-abdominal and showed normal levels of alpha-fetoprotein. Histopathological examination revealed elements from the three germ layers without any evidence of immature cells ruling out teratoma as a differential diagnosis. Conclusions Owing to its rarity, fetus in fetu requires a high degree of suspicion and meticulous surgical techniques to avoid either injury of the adjacent vital structures or bleeding from the main blood supply connection to the host. It should be differentiated from mature teratoma.

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Bittersweet: infective complications of drug-induced glycosuria in patients with diabetes mellitus on SGLT2-inhibitors: two case reports

BMC Infectious Diseases ◽

10.1186/s12879-021-05982-3 ◽

2021 ◽

Vol 21 (1) ◽

Author(s):

Caroline Bartolo ◽

Victoria Hall ◽

N. Deborah Friedman ◽

Chloe Lanyon ◽

Andrew Fuller ◽

...

Keyword(s):

Diabetes Mellitus ◽

Fungal Infections ◽

Case Reports ◽

Sglt2 Inhibitor ◽

Sglt2 Inhibitors ◽

Urogenital Tract ◽

Hypoglycemic Agents ◽

Drug Induced ◽

First Case ◽

Increased Risk

Abstract Background Sodium-glucose co-transporter 2 (SGLT2) inhibitors are novel hypoglycemic agents which reduce reabsorption of glucose at the renal proximal tubule, resulting in significant glycosuria and increased risk of genital mycotic infections (GMI). These infections are typically not severe as reported in large systematic reviews and meta-analyses of the medications. These reviews have also demonstrated significant cardiovascular benefits through other mechanisms of action, making them attractive options for the management of Type 2 diabetes mellitus (T2DM). We present two cases with underlying abnormalities of the urogenital tract in which the GMI were complicated and necessitated cessation of the SGLT2 inhibitor. Case presentations Both cases are patients with T2DM on empagliflozin, an SGLT2 inhibitor. The first case is a 64 year old man with Candida albicans balanitis and candidemia who was found to have an obstructing renal calculus and prostatic abscess requiring operative management. The second case describes a 72 year old man with Candida glabrata candidemia who was found to have prostatomegaly, balanitis xerotica obliterans with significant urethral stricture and bladder diverticulae. His treatment was more complex due to fluconazole resistance and concerns about urinary tract penetration of other antifungals. Both patients recovered following prolonged courses of antifungal therapy and in both cases the SGLT2 inhibitor was ceased. Conclusions Despite their cardiovascular benefits, SGLT2 inhibitors can be associated with complicated fungal infections including candidemia and patients with anatomical abnormalities of the urogenital tract may be more susceptible to these infections as demonstrated in these cases. Clinicians should be aware of their mechanism of action and associated risk of infection and prior to prescription, assessment of urogenital anatomical abnormalities should be performed to identify patients who may be at risk of complicated infection.

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Granulomatous interstitial nephritis in a patient with SARS-CoV-2 infection

BMC Nephrology ◽

10.1186/s12882-020-02213-w ◽

2021 ◽

Vol 22 (1) ◽

Author(s):

Katarzyna Szajek ◽

Marie-Elisabeth Kajdi ◽

Valerie A. Luyckx ◽

Thomas Hans Fehr ◽

Ariana Gaspert ◽

...

Keyword(s):

Acute Kidney Injury ◽

Interstitial Nephritis ◽

Kidney Biopsy ◽

Kidney Diseases ◽

Case Reports ◽

Kidney Injury ◽

Maculopapular Rash ◽

Tubular Injury ◽

Granulomatous Interstitial Nephritis ◽

First Case

Abstract Background Acute kidney injury (AKI) associated with severe coronavirus disease 19 (COVID-19) is common and is a significant predictor of morbidity and mortality, especially when dialysis is required. Case reports and autopsy series have revealed that most patients with COVID-19 – associated acute kidney injury have evidence of acute tubular injury and necrosis - not unexpected in critically ill patients. Others have been found to have collapsing glomerulopathy, thrombotic microangiopathy and diverse underlying kidney diseases. A primary kidney pathology related to COVID-19 has not yet emerged. Thus far direct infection of the kidney, or its impact on clinical disease remains controversial. The management of AKI is currently supportive. Case Presentation The patient presented here was positive for SARS-CoV-2, had severe acute respiratory distress syndrome and multi-organ failure. Within days of admission to the intensive care unit he developed oliguric acute kidney failure requiring dialysis. Acute kidney injury developed in the setting of hemodynamic instability, sepsis and a maculopapular rash. Over the ensuing days the patient also developed transfusion-requiring severe hemolysis which was Coombs negative. Schistocytes were present on the peripheral smear. Given the broad differential diagnoses for acute kidney injury, a kidney biopsy was performed and revealed granulomatous tubulo-interstitial nephritis with some acute tubular injury. Based on the biopsy findings, a decision was taken to adjust medications and initiate corticosteroids for presumed medication-induced interstitial nephritis, hemolysis and maculo-papular rash. The kidney function and hemolysis improved over the subsequent days and the patient was discharged to a rehabilitation facility, no-longer required dialysis. Conclusions Acute kidney injury in patients with severe COVID-19 may have multiple causes. We present the first case of granulomatous interstitial nephritis in a patient with COVID-19. Drug-reactions may be more frequent than currently recognized in COVID-19 and are potentially reversible. The kidney biopsy findings in this case led to a change in therapy, which was associated with subsequent patient improvement. Kidney biopsy may therefore have significant value in pulling together a clinical diagnosis, and may impact outcome if a treatable cause is identified.

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Proteus syndrome in pregnancy: A case report

Obstetric Medicine ◽

10.1177/1753495x20970791 ◽

2020 ◽

pp. 1753495X2097079

Author(s):

Niccole Ranaei-Zamani ◽

Mandeep K Kaler ◽

Rehan Khan

Keyword(s):

Case Report ◽

Case Reports ◽

Proteus Syndrome ◽

Cutaneous Lesions ◽

Genetic Syndrome ◽

Skeletal Tissue ◽

First Case ◽

Large Teaching Hospital ◽

Clinical Presentations ◽

In Pregnancy

Proteus syndrome is a rare, multi-system, genetic syndrome characterised by atypical and excessive growth of skeletal tissue. Clinical presentations include abnormal musculoskeletal growth and cutaneous lesions. Due to its rarity, there have been a limited number of published case reports of Proteus syndrome. This is the first case report on the management of Proteus syndrome in pregnancy. We present the case of a pregnant woman with Proteus syndrome in her first pregnancy in a large teaching hospital and discuss the considerations and challenges faced in her antenatal, intrapartum and postnatal care.

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Intraperitoneal ampicillin treatment for peritoneal dialysis- related peritonitis with Listeria monocytogenes – a case report

BMC Nephrology ◽

10.1186/s12882-020-02068-1 ◽

2020 ◽

Vol 21 (1) ◽

Author(s):

Kristina Boss ◽

Ina Wiegard-Szramek ◽

Jan Dziobaka ◽

Andreas Kribben ◽

Sebastian Dolff

Keyword(s):

Peritoneal Dialysis ◽

Case Report ◽

Listeria Monocytogenes ◽

Case Reports ◽

Chronic Kidney Disease Stage ◽

Disease Stage ◽

Serum Concentrations ◽

Duration Of Treatment ◽

First Case ◽

Listeria Infection

Abstract Background Peritoneal dialysis (PD)-related peritonitis is a rare but serious complication and is associated with increased morbidity and mortality rates. It is most commonly caused by Staphylococcus aureus or Staphylococcus epidermidis, but infection with Listeria monocytogenes may also occur. Recommendations for antibiotic treatment of a Listeria infection are currently based on a small number of case reports and suggest the administration of ampicillin. But unlike vancomycin or gentamicin, for ampicillin the route of application, the dosage, and the duration of treatment have not yet been established. We report a case in which PD-associated peritonitis due to Listeria infection was treated with ampicillin administered intravenously and intraperitoneally, separately and in combination. Case presentation A 72-year-old man with chronic kidney disease stage 5 dialysis (CKDG5D) secondary to hypertension and diabetes was hospitalised in April 2020 because of PD-related peritonitis caused by a Listeria infection. In accordance with the results of resistance tests, the patient was treated with intravenous ampicillin at a dosage of 6 g twice daily. After initial treatment the leukocyte count in the PD effluent had decreased substantially, but it was permanently reduced only with the addition of intraperitoneal ampicillin (4 g daily). Efficient serum concentrations of ampicillin were determined for both routes of administration, intravenous and intraperitoneal. Conclusion This is the first case report demonstrating that PD-related peritonitis due to Listeria monocytogenes infection can be treated with intraperitoneal ampicillin and monitored by the determination of peripheral serum concentrations of ampicillin.

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Management of Type 2B von Willebrand Disease during Pregnancy

Acta Haematologica ◽

10.1159/000453389 ◽

2017 ◽

Vol 137 (2) ◽

pp. 89-92 ◽

Cited By ~ 2

Author(s):

David McLaughlin ◽

Ron Kerr

Keyword(s):

Autosomal Dominant ◽

Case Reports ◽

Management Plan ◽

Von Willebrand Disease ◽

Best Management ◽

First Case ◽

Von Willebrand ◽

Willebrand Factor ◽

Ristocetin Cofactor ◽

Platelet Transfusions

Type 2B von Willebrand disease is a rare bleeding condition resulting in thrombocytopenia and a reduction in large VWF multimers. It usually has an autosomal dominant pattern of inheritance. We report the management of a patient with type 2B von Willebrand disease, whose diagnosis was confirmed by demonstration of a R1306W mutation, through her first pregnancy. The patient's von Willebrand factor (VWF) antigen and VWF ristocetin cofactor levels rose throughout pregnancy, with an associated drop in the platelet count. The patient was successfully managed through labour to a surgical delivery with VWF concentrate, platelet transfusions and tranexamic acid. The patient delivered a male baby who was found to have inherited type 2B von Willebrand disease and had a significant cephalhaematoma at delivery. The baby was managed with VWF concentrate and platelet transfusions and made a full recovery. There is a lack of evidence to guide the best management of pregnant patients with type 2B von Willebrand disease. We adopted a pragmatic management plan, in keeping with other published case reports. To the best of our knowledge, this is the first case report in which the child was found to have inherited type 2B von Willebrand disease and encountered bleeding problems, making this case unique amongst the published literature.

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Generalized Anxiety Disorder in Very Young Children: First Case Reports on Stability and Developmental Considerations

Case Reports in Psychiatry ◽

10.1155/2018/7093178 ◽

2018 ◽

Vol 2018 ◽

pp. 1-5

Author(s):

Michael S. Scheeringa ◽

Lauren C. Burns

Keyword(s):

Anxiety Disorder ◽

Young Children ◽

Diagnostic Criteria ◽

Generalized Anxiety Disorder ◽

Emotional Processing ◽

Case Reports ◽

Future Research ◽

Generalized Anxiety ◽

Very Young Children ◽

First Case

Generalized anxiety disorder (GAD) is purported to start in early childhood but concerns about attenuation of anxiety symptoms over time and the development of emerging cognitive and emotional processing capabilities pose multiple challenges for accurate detection. This paper presents the first known case reports of very young children with GAD to examine these developmental challenges at the item level. Three children, five-to-six years of age, were assessed with the Diagnostic Infant and Preschool Assessment twice in a test-retest reliability study. One case appeared to show attenuation of the worries during the test-retest period based on caregiver report but not when followed over two years. The other two cases showed stability of the full complement of diagnostic criteria. The cases were useful for demonstrating that the current diagnostic criteria appear adequate for this developmental period. The challenges of accurate assessment of young children that might cause missed diagnoses are discussed. Future research on the underlying dysregulation of negative emotionality and long-term follow-ups are needed to better understand the etiology, treatment, and course of GAD in this age group.

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Frontotemporal dementia and amyotrophic lateral sclerosis: Revisiting one of the first case reports with neuropathological examination

Dementia & Neuropsychologia ◽

10.1590/s1980-57642014dn81000013 ◽

2014 ◽

Vol 8 (1) ◽

pp. 83-86 ◽

Cited By ~ 2

Author(s):

Ricardo Nitrini

Keyword(s):

Amyotrophic Lateral Sclerosis ◽

Frontotemporal Dementia ◽

Motor Neurons ◽

Case Reports ◽

Rare Event ◽

Motor Impairments ◽

Late 20Th Century ◽

First Case ◽

Lateral Sclerosis ◽

Neuropathological Examination

ABSTRACT The occurrence of dementia in amyotrophic lateral sclerosis (ALS) was only widely recognized in the late 20th century. Hitherto, it was believed that dementia was a rare event due to the fortuitous association with other diseases. In 1924, Kostantin Nikolaevich Tretiakoff and Moacyr de Freitas Amorim reported a case of dementia with features of frontotemporal dementia (FTD) that preceded the motor signs of ALS. Neuropathological examination confirmed ALS and found no signs of other dementia-causing diseases. The authors hypothesized that dementia was part of ALS and recommended the search for signs of involvement of motor neurons in cases of dementia with an ill-defined clinical picture, a practice currently accepted in the investigation of cases of FTD. This was one of the first descriptions of dementia preceding the motor impairments of ALS and was published in Portuguese and French in Memórias do Hospício de Juquery.

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Septic Pulmonary Embolism: three Case Reports

Monaldi Archives for Chest Disease ◽

10.4081/monaldi.2008.400 ◽

2016 ◽

Vol 69 (2) ◽

Cited By ~ 1

Author(s):

Z. Celebi Sözener ◽

A. Kaya ◽

C. Atasoy ◽

M. Kılıckap ◽

N. Numanoglu ◽

...

Keyword(s):

Pulmonary Embolism ◽

Case Reports ◽

Pleuritic Chest Pain ◽

Radiological Findings ◽

Wound Culture ◽

Septic Embolism ◽

First Case ◽

Septic Pulmonary Embolism ◽

Fever Response ◽

Valve Operation

We present three cases of septic pulmonary embolism which occurred as a result of three different causes. The first case, was a 23 year old woman suffering from cough, sputum, hemopthisis and pleuritic chest pain. She had a right subclavian port. On her thorax computed tomography (CT) scans there were widespread bilateral, irregular parenchymal nodular infiltrates and some of them beginning to cavitate. Meticilin resistant stafilococus aureus (MRSA) was isolated from the blood culture and septic embolism was diagnosed. A month after antibiotic theraphy her parenchymal nodules have considerably decreased in size. The second case was a 40 year old woman admitted to our hospital with the same complaints. Her radiological findings were similar. Meticilin sensitive stafilococus aureus (MSSA) was isolated from the blood cultures and antibiotic theraphy was initiated. To investigate the etiology of the nodules due to septic embolism, echocardiography was performed and infective endocarditis was diagnosed. After the antibiotic theraphy and a tricuspid valve operation her parenchymal nodules disappeared. The final case involved a 51 year old man suffering from fever, fatigue, cough and pain in the left arm for one week. His general status was bad. His radiological findings were also similar to the others. Staphillococcus aureus was isolated from blood and wound culture. Following clinical and radiological findings we thought it was a case of septic pulmonary embolism and antibiotic theraphy was started. Despite the therapy we did not take fever response and he died five days after antibiotic therapy. In conclusion, septic pulmonary embolism should be considered in bilateral cavitary nodular infiltrates and must be managed fast.

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Subsequent Development of Desmoid Tumor after a Resected Gastrointestinal Stromal Tumor

Case Reports in Pathology ◽

10.1155/2018/1082956 ◽

2018 ◽

Vol 2018 ◽

pp. 1-5

Author(s):

Areen Abdulelah Murshid ◽

Hatim Q. Al-Maghraby

Keyword(s):

Gastrointestinal Stromal Tumor ◽

Desmoid Tumor ◽

Case Reports ◽

Previous History ◽

Subsequent Development ◽

Stromal Tumor ◽

Desmoid Tumors ◽

Mesenchymal Tumors ◽

First Case ◽

History Of

Desmoid tumors (deep fibromatosis) of the mesentery are rare mesenchymal tumors. They are often misdiagnosed, especially with a previous history of resection for gastrointestinal stromal tumor (GIST). Immunohistochemistry can help differentiate between these two tumors. In this article, we present a case we had encountered: a Desmoid tumor developing in a patient with a history of GIST 3 years ago. It is the first case of GIST with subsequent development of Desmoid tumor to be reported in Saudi Arabia. We discuss the two entities of Desmoid tumor and GIST by comparing their definitions, clinical presentations, histological features, immunohistochemistry stains, molecular pathogenesis, prognosis, and treatment. We also discuss the relationship between GIST and the subsequent development of Desmoid tumors and compare our case with case reports in literature.

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