{"title":"<i>Actinotignum schaalii</i> Can Be an Uropathogen of \"Culture-Negative\" Febrile Urinary Tract Infections in Children with Urinary Tract Abnormalities.","authors":"Mami Washio, Nobutaka Harada, Daisuke Nishima, Megumi Takemoto","doi":"10.1159/000526398","DOIUrl":"https://doi.org/10.1159/000526398","url":null,"abstract":"<p><p>Accurate diagnosis and treatment of febrile urinary tract infections (UTI) during childhood are important for the prevention of renal parenchymal damage and functional loss, and detection of underlying diseases related to chronic kidney disease (CKD). <i>Actinotignum schaalii</i> (<i>A. schaalii</i>)-related febrile UTI in children is rare, and its incidence and risk factors remain unclear. A 3-year-old boy with a history of UTI presented with fever and vomiting. Although the culture of his urine specimen in air was negative, <i>A. schaalii</i> was observed in a 5% carbon dioxide (CO<sub>2</sub>) culture condition, as well as an anaerobic one. A diagnosis of febrile UTI was made, and he recovered with antibiotic therapy. He was found to have CKD associated with vesicoureteral reflux (VUR) after further investigations. <i>A. schaalii</i> is one of the causative agents of febrile UTI in children with urinary tract abnormalities. Although the culture in the air could show negative results, urine culture in 5% CO<sub>2</sub> and anaerobic conditions is useful for diagnosis. Our case is the youngest and the first known case of <i>A. schaalii</i>-related febrile UTI associated with VUR in children.</p>","PeriodicalId":9599,"journal":{"name":"Case Reports in Nephrology and Dialysis","volume":"12 3","pages":"150-156"},"PeriodicalIF":0.7,"publicationDate":"2022-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/dc/59/cnd-0012-0150.PMC9743144.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10813546","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Michaël Mekeirele, Silke Verheyen, Ruth Van Lancker, Stephanie Wuyts, Tim Balthazar
{"title":"Hemadsorption: A New Therapeutic Option for Selected Cases of Bromazepam Intoxication.","authors":"Michaël Mekeirele, Silke Verheyen, Ruth Van Lancker, Stephanie Wuyts, Tim Balthazar","doi":"10.1159/000526323","DOIUrl":"https://doi.org/10.1159/000526323","url":null,"abstract":"<p><p>Benzodiazepine ingestion is frequent in patients admitted to ICU for intoxications. Generally, a supportive approach by securing the airway, breathing, and circulation is sufficient. Flumazenil is a well-known antidote for benzodiazepines but does not influence its elimination. Following preclinical data, we applied for the first time in humans a hemadsorption filter in a patient with a bromazepam intoxication. This technique proved to be effective in eliminating bromazepam in a patient with CHILD-C cirrhosis. We conclude that hemadsorption is a viable option to reduce length of ICU stay or intubation in slow metabolizers without contraindications.</p>","PeriodicalId":9599,"journal":{"name":"Case Reports in Nephrology and Dialysis","volume":"12 3","pages":"163-166"},"PeriodicalIF":0.7,"publicationDate":"2022-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/f7/2d/cnd-0012-0163.PMC9743138.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10813549","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Rituximab, Mycophenolic Acid, and Calcineurin Inhibitors Achieve Long-Term Remission in Pediatric Focal Segmental Glomerulosclerosis with Steroid-Resistant and Frequently Relapsing Nephrotic Syndrome: A Report of Two Cases.","authors":"Cahyani Gita Ambarsari, Meilania Saraswati, Genta Syaifrin Laudza","doi":"10.1159/000525776","DOIUrl":"https://doi.org/10.1159/000525776","url":null,"abstract":"<p><p>Studies investigating the effect of rituximab in children with nephrotic syndrome (NS) due to focal segmental glomerulosclerosis (FSGS) have reported conflicting results, with some concluding that patients may require additional immunosuppressive therapy to achieve and/or maintain long-term remission. We report successful treatment of pediatric FSGS with rituximab infusions, followed by maintenance immunosuppression with mycophenolic acid (MPA) and a calcineurin inhibitor (CNI) in 1 patient with refractory steroid-resistant NS (SRNS), and one with frequently relapsing NS (FRNS). Case 1 is a patient with refractory SRNS due to FSGS. MPA and tacrolimus induced complete remission within 6 months following rituximab treatment. Remission was maintained for over 2 years, and the patient's kidney function and body height also returned to normal ranges within this time. Case 2 is a patient with FRNS due to FSGS, who was treated with rituximab followed by MPA and cyclosporine, which successfully prevented relapses for 18 months, that is, at the end point of the observation. Our case report demonstrates that rituximab and a combination of CNIs and MPA can be effective in achieving complete remission in pediatric refractory SRNS and sustaining remission in pediatric FSGS with FRNS and SRNS for several years. This treatment regimen has the advantage of eliminating the need for long-term high-dose steroid treatments, allowing 1 patient to achieve normal growth and recover from other adverse steroid effects.</p>","PeriodicalId":9599,"journal":{"name":"Case Reports in Nephrology and Dialysis","volume":"12 3","pages":"167-177"},"PeriodicalIF":0.7,"publicationDate":"2022-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/1c/cb/cnd-0012-0167.PMC9743143.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10813548","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Lucas Jacobs, Francesco Feoli, Pascal Bruderer, Semra Top, Ivan Grozdev, Edouard Cubilier, Frederic Collart
{"title":"Severe Bullous Pemphigoid Onset after Jugular Catheter Placement in a Patient on Hemodialysis.","authors":"Lucas Jacobs, Francesco Feoli, Pascal Bruderer, Semra Top, Ivan Grozdev, Edouard Cubilier, Frederic Collart","doi":"10.1159/000524903","DOIUrl":"https://doi.org/10.1159/000524903","url":null,"abstract":"<p><p>Pruritus is highly prevalent in the dialysis population. Its etiology however remains often unclear with uremic pruritus primarily suspected unless compelling evidence of another cause. Although bullous pemphigoid (BP) is considered idiopathic, there are growing data in the literature on BP provoked by different factors, such as medications or surgical procedures. These secondary dermatoses are described as rather mild conditions and more frequent in the elderly Caucasian. We herein describe a newly dialyzed African man of 76 years old, treated by a sulfonylurea such as an antidiabetic drug, who developed a severe BP after jugular catheter placement.</p>","PeriodicalId":9599,"journal":{"name":"Case Reports in Nephrology and Dialysis","volume":"12 2","pages":"138-144"},"PeriodicalIF":0.7,"publicationDate":"2022-08-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/c7/57/cnd-0012-0138.PMC9459599.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"33485339","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Vena Cava Superior Syndrome Six Years after Central Venous Catheter Removal in a Patient on Hemodialysis.","authors":"Michelle Janssen, Susan Logtenberg","doi":"10.1159/000525795","DOIUrl":"https://doi.org/10.1159/000525795","url":null,"abstract":"<p><p>Vena cava superior (VCS) syndrome is rarely seen as a complication of central-venous-catheter placement. Usually, the syndrome appears when the presence of the catheter causes intraluminal obstruction or thrombosis. In this case report, however, we describe a patient on intermittent hemodialysis who had been free of any venous central line for over 6 years, presented with a VCS syndrome. The CT scan showed an absent VCS without extravascular compression. Previous catheter placement was diagnosed as the case of the VCS syndrome. It is important to realize that VCS syndrome can occur late after removal of central venous catheters, and thus, clinicians should be aware of its symptoms in any patient who has had an upper central line in the past medical history.</p>","PeriodicalId":9599,"journal":{"name":"Case Reports in Nephrology and Dialysis","volume":"12 2","pages":"132-137"},"PeriodicalIF":0.7,"publicationDate":"2022-08-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/59/0c/cnd-0012-0132.PMC9459634.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"33485333","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Maha Mohamed, Muhammad Waseem Athar, Yaasir Mamoojee, Alison Brown, Frances Dowen, Jim Macfarlane
{"title":"Rifabutin-Induced SIADH and Leucopenia in a Renal Transplant Recipient with Genitourinary Tract Tuberculosis: A Case Report and Review of the Literature.","authors":"Maha Mohamed, Muhammad Waseem Athar, Yaasir Mamoojee, Alison Brown, Frances Dowen, Jim Macfarlane","doi":"10.1159/000525921","DOIUrl":"https://doi.org/10.1159/000525921","url":null,"abstract":"<p><p>Tuberculosis (TB) infection of the genitourinary tract (GU TB) is rare in renal transplant recipients, with only a few published case series. GU TB is difficult to diagnose with or without immunosuppression but must always be suspected in any patient with unexplained sterile pyuria. As GU TB is associated with graft rejection, prompt diagnosis and treatment are vital. Treatment is challenging, as rifampicin, the most effective drug used to treat tuberculosis, is a significant inducer of cytochrome P-450 3A metabolism, with the potential to cause significant reductions in the serum levels of calcineurin inhibitors. For this reason, rifabutin, a weaker cytochrome P-450 3A inducer, with similar efficacy against TB, is sometimes used as an alternative to rifampicin in transplant recipients. We present a renal transplant patient diagnosed with GU TB, treated with a regime containing rifabutin, who subsequently developed profound hyponatremia and leucopenia. Serum and urine biochemistry was consistent with a diagnosis of the syndrome of inappropriate antidiuretic hormone secretion (SIADH). Both SIADH and leucopenia resolved with rifabutin cessation. This is the first report of biochemically proven, idiosyncratic SIADH and leucopenia associated with the use of rifabutin in the treatment of GU TB in a renal transplant recipient.</p>","PeriodicalId":9599,"journal":{"name":"Case Reports in Nephrology and Dialysis","volume":"12 2","pages":"117-123"},"PeriodicalIF":0.7,"publicationDate":"2022-08-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/3c/26/cnd-0012-0117.PMC9459626.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"33485336","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Four Cases of Serum Copper Excess in Patients with Renal Anemia Receiving a Hypoxia-Inducible Factor-Prolyl Hydroxylase Inhibitor: A Possible Safety Concern.","authors":"Hironori Nakamura, Shigekazu Kurihara, Mariko Anayama, Yasushi Makino, Masaki Nagasawa","doi":"10.1159/000525735","DOIUrl":"https://doi.org/10.1159/000525735","url":null,"abstract":"<p><p>Copper is an indispensable trace metal element and is mainly absorbed in the stomach and small intestine and excreted into the bile. Hypoxia-inducible factor-prolyl hydroxylase inhibitors (HIF-PHIs) have emerged as a novel approach for renal anemia management. Many intestinal genes, including <i>divalent metal transporter 1, duodenal cytochrome B</i>, and copper transporter ATPase7A, related to iron absorption are transactivated by HlF-α, during iron deficiency. We first report 4 cases of patients with renal anemia who showed excess in serum copper level during roxadustat or daprodustat treatment, which were decreased to the normal level after discontinuing HIF-PHIs and changing the drug to darbepoetin alfa, suggesting that HIF-PHI is associated with serum copper excess. HIF-PHI modulates iron metabolism, such as iron absorption, sequestration, and mobilization, and may increase serum copper levels by increasing copper absorption and/or redistribution of copper in tissues. Therefore, it is urgent to examine the correlation between HIF-PHI use and serum copper levels because copper excess might be involved in several acute or chronic adverse events.</p>","PeriodicalId":9599,"journal":{"name":"Case Reports in Nephrology and Dialysis","volume":"12 2","pages":"124-131"},"PeriodicalIF":0.7,"publicationDate":"2022-08-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/f7/57/cnd-0012-0124.PMC9459559.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"33485334","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Acute Kidney Injury following Exposure to Formaldehyde-Free Hair-Straightening Products.","authors":"Nabil Abu-Amer, Natalie Silberstein, Margarita Kunin, Sharon Mini, Pazit Beckerman","doi":"10.1159/000525567","DOIUrl":"https://doi.org/10.1159/000525567","url":null,"abstract":"<p><p>Formaldehyde-free hair-straightening products are hair-smoothening solutions widely used by professional beauty salons. Formaldehyde-free hair straighteners do not technically contain formaldehyde; however, they contain other chemicals such as glyoxyloyl carbocysteine which releases formaldehyde upon contact with heat. Moreover, its by-product glyoxylate may convert to oxalate; both compounds have potential nephrotoxic effect. Here, we report a case of a 41-year-old woman who presented to the emergency room with weakness, nausea, vomiting, and stage 3 acute kidney injury (AKI) according to Kidney Disease: Improving Global Outcomes (KDIGO) acute kidney injury staging shortly after exposure to formaldehyde-free hair-straightening product; other causes of AKI were excluded such as preceding acute illness, drug history, or other nephrotoxic agent exposure. On physical examination, the patient was pale, and her vital signs were normal. The urine microscopy and serologic workup were not indicative. Kidney core biopsy revealed interstitial edema, acute interstitial nephritis, and oxalate crystal nephropathy. Kidney function completely recovered after a short course of steroid therapy. In this case, AKI was a complication caused by exposure to hair-straightening products branded as formaldehyde free but actually containing other chemical products which release formaldehyde and other toxic chemicals when heated during the straightening procedure and may cause systemic toxicity, particularly kidney injury. Different cosmetic products are widely in use, but not all are under tight regulation, and therefore, it is important to raise the awareness among both medical teams and consumers of possible adverse health effects of different cosmetic products.</p>","PeriodicalId":9599,"journal":{"name":"Case Reports in Nephrology and Dialysis","volume":"12 2","pages":"112-116"},"PeriodicalIF":0.7,"publicationDate":"2022-07-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/b5/ab/cnd-0012-0112.PMC9386411.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"33486248","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Propylthiouracil-Induced Antineutrophil Cytoplasmic Antibodies-Associated Vasculitis with Renal and Lung Involvement.","authors":"Albert Hing Wong, Wei-Kei Wong, Lai-Meng Looi, Jeyakantha Ratnasingam, Soo-Kun Lim","doi":"10.1159/000525182","DOIUrl":"https://doi.org/10.1159/000525182","url":null,"abstract":"<p><p>Propylthiouracil (PTU)-induced antineutrophil cytoplasmic antibodies (ANCA)-associated vasculitis (AAV) is a rare and heterogeneous disease. Moreover, optimal treatment is still lacking. We described the case of a 44-year-old lady with underlying Graves' disease who had cough, blood-streaked sputum, and impaired renal function. A strongly positive anti-myeloperoxidase antibody (>200 U/mL) along with pauci-immune glomerulonephritis and pulmonary hemorrhage resulted in the diagnosis of PTU-induced AAV, given that the patient had been on PTU for 3 years. PTU withdrawal, therapeutic plasma exchanges, and oral cyclophosphamide provided favorable clinical and biochemical outcomes. She remained well on azathioprine 50 mg daily as maintenance therapy and clinically euthyroid with carbimazole 2.5 mg daily. The effective treatment for drug-induced ANCA vasculitis remains controversial, but rapid withdrawal of the offending medication should be the mainstay of treatment. In severe drug-induced ANCA vasculitis with pulmonary hemorrhage and/or life-threatening organ involvement such as kidney failure requiring dialysis, therapeutic plasma exchange with immunosuppressants is often required. In this case, we have shown that patient achieved remission after therapeutic plasma exchange with cyclophosphamide in the acute stage of treatment and remained symptom-free with azathioprine in the maintenance phase of treatment for 24 months.</p>","PeriodicalId":9599,"journal":{"name":"Case Reports in Nephrology and Dialysis","volume":" ","pages":"105-111"},"PeriodicalIF":0.7,"publicationDate":"2022-06-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/b3/f8/cnd-0012-0105.PMC9251458.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40601516","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Membranous Nephropathy-Like Apolipoprotein E Deposition Disease with Apolipoprotein E Toyonaka and Homozygous Apolipoprotein E2/2 without Dyslipidemia, with Characteristic Electron-Dense Deposits.","authors":"Akihiko Koshino, Chikako Takaeda, Takahiro Matsuno, Shinji Kitajima, Yasunori Iwata, Norihiko Sakai, Kiyotaka Nagahama, Yo Niida, Takao Saito, Hitoshi Yokoyama, Takashi Wada","doi":"10.1159/000525086","DOIUrl":"https://doi.org/10.1159/000525086","url":null,"abstract":"<p><p>Recently, several cases of novel apolipoprotein E (apoE)-related glomerular disease known as membranous nephropathy (MN)-like apoE deposition disease with apoE Toyonaka (Ser197Cys) and homozygous apoE2/2 have been reported. However, the clinical and pathological characteristics are uncertain due to the small number of reports. Here, we report an additional case with various clinical and pathological characteristics. A 28-year-old Japanese man with mild proteinuria and hematuria underwent a kidney biopsy. Examination under a light microscope revealed mesangial proliferation, mesangial matrix expansion, and segmental spike lesion. An immunofluorescence study showed no immunoglobulin or complement depositions. In the electron microscopic (EM) examination, massive deposits with various electron densities in the subepithelial, subendothelial, and paramesangial areas were more prominent than those reported in previous cases, which resembled microbubbles or microcysts on higher magnification. The glomerular basement membrane (GBM) structure was partly degenerated by these deposits. Serum triglyceride and cholesterol levels were within the normal range. However, the serum apoE concentration was significantly high, and glomerular apoE accumulation was detected in immunohistochemistry. The DNA sequence revealed apoE Toyonaka and homozygous apoE2/2 similar to that of the previous cases with MN-like apoE deposition disease. MN-like apoE deposition disease can manifest as only mild hematuria and proteinuria without dyslipidemia. Various characteristic deposits associated with GBM degeneration can be observed in the EM study.</p>","PeriodicalId":9599,"journal":{"name":"Case Reports in Nephrology and Dialysis","volume":" ","pages":"96-104"},"PeriodicalIF":0.7,"publicationDate":"2022-06-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/88/ce/cnd-0012-0096.PMC9247525.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40601514","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}