scholarly journals Mycetoma Caused by Acremonium Species in a Patient with Chronic Granulomatous Disease

2016 ◽  
Vol 2016 ◽  
pp. 1-3 ◽  
Author(s):  
Richard Antrobus ◽  
Gabriel Wong ◽  
Julie Jones ◽  
Aarnoud Huissoon
Keyword(s):  
Chronic Granulomatous Disease ◽  
Fungal Infections ◽  
Antifungal Prophylaxis ◽  
Granulomatous Disease ◽  
Cutaneous Infection

Patients with chronic granulomatous disease are predisposed to fungal infections and are therefore routinely prescribed antifungal prophylaxis. We report a case where acremonium was responsible for causing a cutaneous infection (mycetoma) despite antifungal prophylaxis. Treatment with voriconazole was initiated and the infection gradually resolved. This case highlights the need for careful clinical follow-up and thorough investigation of patients who have a neutrophil immunodeficiency.

scholarly journals Posaconazole-Induced Acute Pancreatitis: A Rare Side Effect in a Child with Chronic Granulomatous Disease

2020 ◽  
Vol 8 (4) ◽  
Author(s):  
Marjan Tariverdi ◽  
Sedigheh Rafiei Tabatabaei ◽  
Ali Saffaei ◽  
Ladan Ayazkhoo ◽  
Bahador Mirrahimi
Keyword(s):  
Acute Pancreatitis ◽  
Chronic Granulomatous Disease ◽  
Fungal Infections ◽  
Epigastric Pain ◽  
Pediatric Population ◽  
Antifungal Prophylaxis ◽  
Granulomatous Disease ◽  
Drug Induced ◽  
Abdominal Sonography ◽  
First Case

Introduction: Pancreatitis is an inflammatory disease of the pancreas. Drug-induced pancreatitis is an important cause of pancreatitis. There are two pathological types of acute pancreatitis, including pancreatic edema with a mild course and pancreatic necrosis with a poor prognosis. Some agents can induce pancreatitis, but so far, posaconazole-induced pancreatitis in children has been not reported. Here, we describe the case of a child with acute pancreatitis who received posaconazole. Case Presentation: A 10-year-old girl with a three-year history of chronic granulomatous disease (CGD) was admitted to hospital due to epigastric pain, nausea, vomiting, loss of appetite, and fever for the last four days. The pain was persistent and prominent in the periumbilical area. The patient was on lifelong antifungal prophylaxis for her illness. On abdominal sonography, the head of the pancreas was inflated, which can indicate pancreatitis. All the medications were discontinued at the time of admission, and along with sufficient hydration, acetaminophen was administered for the patient’s pain. One, three, and twelve months after discharge, the patient was visited for follow-up with no signs of stomach discomfort, and the lab data was within the normal limits. CGD is a rare disease in which the phagocytes fail to produce hydrogen peroxide. Such patients are prone to bacterial and fungal infections. Conclusions: In conclusion, this is the second case of posaconazole-induced pancreatitis and the first case in children; thus, we recommend that physicians should be aware of the signs of pancreatitis in high-risk individuals like immunocompromised pediatric population.

Prostatic Abscess in a Patient with Chronic Granulomatous Disease: A Multi-Disciplinary Intervention

2017 ◽  
Vol 84 (4) ◽  
pp. 267-269 ◽  
Author(s):  
Alberto Martini ◽  
Ioannis Katafigiotis ◽  
Sofia Kalantzi ◽  
Ioannis Anastasiou ◽  
Ioannis Adamakis ◽  
...  
Keyword(s):  
Antibiotic Therapy ◽  
Chronic Granulomatous Disease ◽  
Urine Culture ◽  
Underlying Disease ◽  
Multidrug Resistant ◽  
Granulomatous Disease ◽  
Prostatic Abscess ◽  
The Past ◽  
First Case

Introduction Chronic granulomatous disease (CGD) is a rare pathology that increases patients’ susceptibility to infections, given the inability to generate oxygen radicals to fight microorganisms. In the context of CGD, primary prostatic involvement has been described only once in the past, in a pediatric patient. Case Report We report the case of a 35-year old patient with CGD presenting with persistent fever. After hospital admission, blood and urine were sent for culture and antibiotic therapy was initiated. Patient's conditions continued to deteriorate and an aggressive antibiotic therapy was administered to treat the septic scenario. Urine culture grew a multidrug-resistant Escherichia Coli. After patient's condition improved, a CT scan was performed. The depiction showed multiple abscesses within the prostate. A rectal approach was excluded given patient's underlying disease. A TURP was performed and prostatic pathology resolved. Patient was discharged on postoperative day 14. At 6-month follow-up he hasn't experienced major infections. To the best of our knowledge, this is the first case of septic shock originating from a prostatic abscess in an adult patient with CGD. Conclusions Aggressive medical therapy along with TURP resulted curative in our case. A multi-disciplinary approach was mandatory.

scholarly journals The p47phox mouse knock-out model of chronic granulomatous disease.

1995 ◽  
Vol 182 (3) ◽  
pp. 751-758 ◽  
Author(s):  
S H Jackson ◽  
J I Gallin ◽  
S M Holland
Keyword(s):  
Nadph Oxidase ◽  
Chronic Granulomatous Disease ◽  
Fungal Infections ◽  
Critical Role ◽  
Granulomatous Inflammation ◽  
Mammalian Host ◽  
Granulomatous Disease ◽  
Knock Out ◽  
Phagocyte Nadph Oxidase ◽  
Life Threatening

Chronic granulomatous disease (CGD) is caused by a congenital defect in phagocyte reduced nicotinamide dinucleotide phosphate (NADPH) oxidase production of superoxide and related species. It is characterized by recurrent life-threatening bacterial and fungal infections and tissue granuloma formation. We have created a mouse model of CGD by targeted disruption of p47phox, one of the genes in which mutations cause human CGD. Identical to the case in human CGD, leukocytes from p47phox-/- mice produced no superoxide and killed staphylococci ineffectively. p47phox-/- mice developed lethal infections and granulomatous inflammation similar to those encountered in human CGD patients. This model mirrors human CGD and confirms a critical role for the phagocyte NADPH oxidase in mammalian host defense.

Topics in Chronic Granulomatous Disease

PEDIATRICS ◽  
1991 ◽  
Vol 88 (1) ◽  
pp. 183-185
Author(s):  
SHIGENOBU UMEKI
Keyword(s):  
Nadph Oxidase ◽  
Chronic Granulomatous Disease ◽  
Host Defense ◽  
Superoxide Anion ◽  
Fungal Infections ◽  
Regulatory Elements ◽  
Granulomatous Disease ◽  
Phagocytic Cells ◽  
Oxidase System ◽  
Membrane Bound

To the Editor.— Such phagocytic cells as neutrophils and macrophages are crucial elements in the host defense against bacterial [See table in the PDF file] and fungal infections. Microbicidal activity depends to a large extent on NADPH oxidase system, which can be activated by stimuli (bacteria, fungi) and which generates the superoxide anion and other highly reactive forms of reduced oxygen.1,2 The neutrophil NADPH oxidase system is composed functionally of membrane-bound catalytic components (which consist of at least two constituents, the low potential cytochrome b5583-5 and flavoprotein5) and soluble cytosolic components6,7 which participate as either catalytic or regulatory elements.

scholarly journals Clinical observation of Сhronic granulomatous disease in a 6-year-old child

2020 ◽  
Vol 19 (4) ◽  
pp. 69-72
Author(s):  
G. A. Kharchenko ◽  
O. G. Kimirilova
Keyword(s):  
Chronic Granulomatous Disease ◽  
Clinical Observation ◽  
Fungal Infections ◽  
Genetic Defect ◽  
Clinical Manifestations ◽  
Hereditary Disease ◽  
Laboratory Method ◽  
Granulomatous Disease ◽  
Hematopoietic Stem ◽  
Nbt Test

Chronic granulomatous disease (CGD) is a hereditary disease caused by a genetic defect of violations of oxygen — dependent mechanisms of phagocytosis. Clinical manifestations of the disease are recurrent bacterial or fungal infections of the skin, hepatic abscesses, pneumonia, osteomyelitis, sepsis, meningitis et al. Most available laboratory method for the diagnosis of CGD is the test of histochemical nitro blue tetrazolium recovery (NBT-test). Allogeneic hematopoietic stem cell transplantation is considered a radical treatment for chronic granulomatous disease. The article presents a clinical observation of the manifestation of chronic granulomatous disease with an unfavorable outcome in a child aged 6 years.

scholarly journals Chronic Granulomatous Disease Presenting as Aseptic Ascites in a 2-Year-Old Child

2013 ◽  
Vol 2013 ◽  
pp. 1-5 ◽  
Author(s):  
J. F. Moreau ◽  
John A. Ozolek ◽  
P. Ling Lin ◽  
Todd D. Green ◽  
Elaine A. Cassidy ◽  
...  
Keyword(s):  
Chronic Granulomatous Disease ◽  
Fungal Infections ◽  
Novel Mutation ◽  
Nicotinamide Adenine Dinucleotide Phosphate ◽  
Unknown Origin ◽  
Granulomatous Disease ◽  
Cybb Gene ◽  
Inherited Immunodeficiency ◽  
Peritoneal Biopsy ◽  
Immunodeficiency Syndrome

Chronic granulomatous disease (CGD) is a rare inherited immunodeficiency syndrome that results from abnormal nicotinamide adenine dinucleotide phosphate (NADPH) oxidase function. This defect leads to recurrent catalase-positive bacterial and fungal infections as well as associated granuloma formation. We review the case of a 2-year-old boy who presented with ascites and fever of an unknown origin as manifestations of CGD. Cultures were negative for infection throughout his course, and CGD was suspected after identification of granulomas on peritoneal biopsy. Genetic testing revealed a novel mutation in the CYBB gene underlying his condition. This paper highlights the importance of considering CGD in the differential diagnosis of fever of unknown origin and ascites in children.

LIVER SCANS IN CHRONIC GRANULOMATOUS DISEASE OF CHILDHOOD

PEDIATRICS ◽  
1971 ◽  
Vol 48 (1) ◽  
pp. 41-50
Author(s):  
L. D. Samuels
Keyword(s):  
Chronic Granulomatous Disease ◽  
Granulomatous Disease ◽  
Recurrent Infections ◽  
Oxidase Deficiency ◽  
Liver Morphology

The biochemical defect of oxidase deficiency present in chronic granulomnatous disease of childhood is manifest clinically by recurrent infections, especially focal abscesses. In the liver these may be bacterial abscesses or characteriastic miliary, sterile granulomas. Either of these can be well-visualized and usefully followed with radioisotope liver scans. Follow-up scans of the liver have shown that chronic changes of the disease may severely distort the gross liver morphology. Cases are presented of two surviving teen-age cousins and two young sisters, subsequently deceased, whose third sibling has just presented with the disease.

scholarly journals 1129. Targeted Voriconazole Prophylaxis in Heart Transplantation Recipients

2018 ◽  
Vol 5 (suppl_1) ◽  
pp. S338-S338
Author(s):  
Michael Lin ◽  
Ignacio Echenique ◽  
Michael Angarone ◽  
Allen Anderson ◽  
Valentina Stosor
Keyword(s):  
Heart Transplantation ◽  
Fungal Infections ◽  
Single Case ◽  
Statistical Significance ◽  
Antifungal Prophylaxis ◽  
Observational Cohort Study ◽  
Observational Cohort ◽  
Univariate Analyses

Abstract Background The use of antifungal prophylaxis, targeted or universal, remains controversial and unstudied. The goal of this study is to determine the role of targeted voriconazole prophylaxis (VORI) in prevention of invasive fungal infections (IFI) after heart transplantation (HT). Methods We conducted a single-center, prospective, observational cohort study of 276 HT recipients from June 2005 to April 2017 to characterize the incidence and outcome of IFI following targeted VORI. Starting in June 2013, HT recipients with thymoglobulin (ATG) treatment received VORI for 3 months. Probable/proven IFI were defined by EORTC/MSG criteria. Descriptive frequencies and univariate analyses were performed. Results Mean duration of follow-up post-HT was 1,165 days (0–3,152 days). 149 (54%) and 70 (25%) received basiliximab and thymoglobulin induction, respectively. Thirty-one (11%) received VORI, following use of ATG in the setting of induction (68%) or rejection (32%). VORI was started at median of 6 days (0–1,008 days) post-HT for a mean duration of 97 days (5–251 days). Overall, 23 IFIs occurred in 23 recipients (8%) at mean 283 days post-HT (range 2–1,579 days), including seven Aspergillus (one occurring after VORI completion), seven invasive Candida (five with candidemia), two Rhizopus, one Cunninghamella, two histoplasma, two blastomyces, one Cryptococcus, and one multifocal cutaneous Alternaria. Conclusion Targeted VORI resulted in reduced incidences of both early and overall IFI after HT although this did not reach statistical significance. Since instituting this strategy, we have observed a single case of aspergillosis following VORI discontinuation. Overall and 1-year mortality were not impacted. The use of antifungal prophylaxis following HT requires continued investigation both to determine efficacy and toxicity in this patient population. Disclosures All authors: No reported disclosures.

scholarly journals Imaging findings of pulmonary manifestations of chronic granulomatous disease in a large single center from Shanghai, China (1999–2018)

2020 ◽  
Vol 10 (1) ◽  
Author(s):  
Qiong Yao ◽  
Qin-hua Zhou ◽  
Quan-li Shen ◽  
Zhong-wei Qiao ◽  
Xiao-chuan Wang ◽  
...  
Keyword(s):  
Chronic Granulomatous Disease ◽  
Interstitial Pneumonia ◽  
Fungal Infections ◽  
Imaging Features ◽  
Granulomatous Disease ◽  
Axillary Lymphadenopathy ◽  
Multiple Organs ◽  
Pulmonary Manifestations ◽  
Pulmonary Abnormalities ◽  
Interlobular Septal Thickening

Abstract Chronic granulomatous disease (CGD) is characterized by recurrent infections and granuloma formation in multiple organs, especially the lung. We aimed to investigate pulmonary manifestations by computed tomography (CT). In total, 100 patients with 117 episodes of pulmonary infection were included. Chest CT scans of every episode were analyzed. Random nodules were the most common findings (79.49%), followed by ground-grass opacities (74.36%), focal consolidations (62.39%), and masses (59.83%). Cavities (12.82%) and multiple small abscesses (17.09%) could be found in the consolidations and masses. CT revealed interstitial pneumonia with tree-in-bud opacities (17.09%), interlobular septal thickening (23.08%) and emphysema (35.04%), which were more severe in the bilateral upper lobes. Mediastinal and hilar lymphadenopathy (78.63%) and axillary lymphadenopathy (65.81%) were common. Fungal infection (n = 27) was the most common and presented with multiple nodules and masses. Approximately 1/4 of fungal infections had interstitial pneumonia. In Staphylococcus aureus (n = 6) and Klebsiella pneumoniae (n = 3) infections, large areas of consolidation were common. In tuberculosis infection, the pulmonary infections were more severe and complex. For Bacillus Calmette-Guérin disease, left-sided axillary lymphadenopathy was a characteristic manifestation. CT images of CGD demonstrated variable pulmonary abnormalities. The main infectious organisms have unique imaging features.

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