scholarly journals Back Pain in a 23-Year-Old Male With X-Linked Chronic Granulomatous Disease

2019 ◽  
Vol 6 (11) ◽  
Author(s):  
Michelle C Sabo ◽  
Michela Blain ◽  
Denise McCulloch ◽  
Heather L Glasgow ◽  
Dhruba J Sengupta ◽  
...  
Keyword(s):  
Back Pain ◽  
Invasive Aspergillosis ◽  
Chronic Granulomatous Disease ◽  
Vertebral Osteomyelitis ◽  
Aspergillus Species ◽  
Granulomatous Disease ◽  
First Case ◽  
Increased Risk

Abstract Patients with chronic granulomatous disease are at increased risk for invasive aspergillosis. Cryptic Aspergillus species are being increasingly recognized as distinct causes of infection in this population. In this study, we describe the first case of Aspergillus udagawae vertebral osteomyelitis in a patient with X-linked chronic granulomatous disease.

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.

BACTERICIDAL FUNCTION OF HUMAN POLYMORPHONUCLEAR LEUKOCYTES

PEDIATRICS ◽  
1972 ◽  
Vol 50 (2) ◽  
pp. 264-270
Author(s):  
Paul G. Quie
Keyword(s):  
Hydrogen Peroxide ◽  
Chronic Granulomatous Disease ◽  
Polymorphonuclear Leukocytes ◽  
Bacterial Species ◽  
Bacterial Flora ◽  
Granulomatous Disease ◽  
Serum Factors ◽  
Gram Negative ◽  
Increased Risk ◽  
Human Polymorphonuclear Leukocytes

Serum from most normal persons contains specific antibodies which react with common bacterial species preparing their surfaces so that phagocytosis by leukocytes can take place. The Fab part of these antibodies reacts with immunologic specificity with antigens on the surface of bacteria. Another part of the immunoglobulin molecule termed the Fc portion is activated during the attachment of the Fab portion to bacteria and becomes a site for attachment of bacteria to receptors on the surface of phagocytic cells. This activity is greatly amplified by heat-labile serum factors. Normally bacteria are rapidly killed by human polymorphonuclear leukocytes after engulfment occurs. However staphylococci and gram-negative species of bacteria survive in the leukocytes of patients with the syndrome "Chronic Granulomatous Disease of Childhood." These patients have suffered recurrent severe infections with bacterial species that are part of the body's resident bacterial flora. By contrast these patients are not at increased risk to infection from such pyogenic bacterial species as group A streptococci or pneumococci. The leukocytes from patients with chronic granulomatous disease produce little hydrogen peroxide during phagocytosis. Catalase-producing staphylococci and gram-negative bacteria are not killed, but hydrogen peroxide-producing streptococci and pneumococci are killed. A normal metabolic response to phagocytosis as well as release of lysosonial factors are essential for the bactericidal activity of human polymorphonuclear leukocytes.

Successful Medical Therapy of Aspergillus Osteomyelitis of the Spine in an 11-Year-Old Boy With Chronic Granulomatous Disease

PEDIATRICS ◽  
1994 ◽  
Vol 93 (5) ◽  
pp. 830-835
Author(s):  
Mark W. Kline ◽  
Mary E. Paul ◽  
Howard M. Rosenblatt ◽  
William T. Shearer ◽  
Florante C. Bocobo
Keyword(s):  
Chronic Granulomatous Disease ◽  
Vertebral Osteomyelitis ◽  
Granulomatous Disease ◽  
Lipid Complex ◽  
Serious Infection ◽  
Common Causes ◽  
Oral Itraconazole ◽  
Amphotericin B Lipid Complex ◽  
Intravenous Amphotericin ◽  
Contiguous Spread

Aspergillus is one of the most common causes of serious infection and death in individuals with chronic granulomatous disease (CGD).1 Clinically evident infection often begins in the lung with pneumonia; contiguous spread or dissemination of infection may occur. Vertebral osteomyelitis in individuals with CGD usually is caused by Aspergillus.2 Unfortunately, despite aggressive medical and surgical therapy of this condition, reported cases have been associated with treatment failure, recurrence, severe disabling orthopedic or neurologic complications and death.2-5 In this report we describe a unique case in which therapy with intravenous amphotericin B lipid complex (AmBLC), oral itraconazole, and granulocyte transfusions resulted in complete clinical and radiographic resolution of Aspergillus fumigatus osteomyelitis of the spine.

scholarly journals Tissue Distribution and Penetration of Isavuconazole at the Site of Infection in Experimental Invasive Aspergillosis in Mice with Underlying Chronic Granulomatous Disease

2019 ◽  
Vol 63 (6) ◽  
Author(s):  
Annie Lee ◽  
Brendan Prideaux ◽  
Min Hee Lee ◽  
Matthew Zimmerman ◽  
Enriko Dolgov ◽  
...  
Keyword(s):  
Mass Spectrometry ◽  
Invasive Aspergillosis ◽  
Chronic Granulomatous Disease ◽  
Lung Tissue ◽  
Fungal Pathogens ◽  
High Efficiency ◽  
Wide Spectrum ◽  
Histopathological Analysis ◽  
Ionization Mass ◽  
Granulomatous Disease

ABSTRACTIsavuconazole, the active moiety of the prodrug isavuconazonium sulfate, has potent activity against a wide spectrum of fungal pathogens and is approved for the treatment of invasive aspergillosis, yet little is known about the tissue penetration of isavuconazole at the target sites of infection. Here, we explored the spatial and quantitative distribution of isavuconazole in tissue lesions in experimental pulmonary aspergillosis established in mice with chronic granulomatous disease (CGD) (gp91phox−). Matrix-assisted laser desorption ionization mass spectrometry imaging (MALDI-MSI) and laser capture microdissection (LCM)-directed high-pressure liquid chromatography coupled to tandem mass spectrometry (LC-MS/MS) were used to analyze infected lungs and brain tissues collected 1, 3, 6, and 24 h after a single oral administration of the prodrug at a dose of 256 mg/kg of body weight (corresponding to 122.9 mg/kg of isavuconazole). Drug enrichment within granulomatous lesions was observed in lung tissue at 1 h postdose, although drug levels quickly equilibrated afterwards between lesion and nonlesion areas. A prominent antifungal effect in the infected lung tissue was revealed by histopathological analysis. Isavuconazole also penetrated into the brain with high efficiency. These data further support the value of isavuconazole to treat patients with invasive aspergillosis.

Successful Treatment of Invasive Aspergillosis in Chronic Granulomatous Disease by Bone Marrow Transplantation, Granulocyte Colony-Stimulating Factor–Mobilized Granulocytes, and Liposomal Amphotericin-B

Blood ◽  
1998 ◽  
Vol 92 (8) ◽  
pp. 2719-2724 ◽  
Author(s):  
Hülya Ozsahin ◽  
Maya von Planta ◽  
Irene Müller ◽  
Hans C. Steinert ◽  
David Nadal ◽  
...  
Keyword(s):  
Bone Marrow ◽  
Bone Marrow Transplantation ◽  
Invasive Aspergillosis ◽  
Chronic Granulomatous Disease ◽  
Marrow Transplantation ◽  
Granulomatous Disease ◽  
Granulocyte Colony Stimulating Factor ◽  
Colony Stimulating Factor ◽  
Aspergillus Infection ◽  
Stimulating Factor

X-linked chronic granulomatous disease (X-CGD) is a primary immunodeficiency with complete absence or malfunction of the nicotinamide adenine dinucleotide phosphate (NADPH) oxidase in the phagocytic cells. Life-threatening infections especially with aspergillus are common despite optimal antimicrobial therapy. Bone marrow transplantation (BMT) is contraindicated during invasive aspergillosis in any disease setting. We report an 8-year-old patient with CGD who underwent HLA-genoidentical BMT during invasive multifocal aspergillus nidulans infection, nonresponsive to treatment with amphotericin-B and γ-interferon. During the first 10 days post-BMT, the patient received granulocyte colony-stimulating factor (G-CSF)–mobilized, 25 Gy irradiated granulocytes from healthy volunteers plus G-CSF beginning on day 3 to prolong the viability of the transfused granulocytes. This was confirmed in vitro by apoptosis assays and in vivo by finding nitroblue tetrazolium (NBT)-positive granulocytes in peripheral blood 12 and 36 hours after the transfusions. Clinical and biological signs of infection began to disappear on day 7 post-BMT. Positron emission tomography with F18-fluorodeoxyglucose (FDG-PET) and computed tomography (CT) scans at 3 months post-BMT showed complete disappearance of infectious foci. At 2 years post-BMT, the patient is well with full immune reconstitution and no sign of aspergillus infection. Our results show that HLA-identical BMT may be successful during invasive, noncontrollable aspergillus infection, provided that supportive therapy is optimal. © 1998 by The American Society of Hematology.

Resistant Invasive Aspergillosis in an Autosomal Recessive Chronic Granulomatous Disease

2012 ◽  
Vol 32 (4) ◽  
pp. 241-245
Author(s):  
Lamia Sfaihi ◽  
Ines Maaloul ◽  
Hela Fourati ◽  
Marie José Stasia ◽  
Zeineb Mnif ◽  
...  
Keyword(s):  
Invasive Aspergillosis ◽  
Chronic Granulomatous Disease ◽  
Autosomal Recessive ◽  
Granulomatous Disease

scholarly journals Flucloxacillin Results in Suboptimal Plasma Voriconazole Concentrations

2017 ◽  
Vol 61 (9) ◽  
Author(s):  
Eline W. Muilwijk ◽  
Bart G. J. Dekkers ◽  
Stefanie S. V. Henriet ◽  
Paul E. Verweij ◽  
Bregje Witjes ◽  
...  
Keyword(s):  
Invasive Aspergillosis ◽  
Chronic Granulomatous Disease ◽  
Granulomatous Disease ◽  
Pulmonary Aspergillosis ◽  
Gram Positive ◽  
Relevant Interaction

ABSTRACT Combining voriconazole and flucloxacillin is indicated in patient cohorts experiencing both invasive aspergillosis and Gram-positive infections (e.g., patients with chronic granulomatous disease or postinfluenza pulmonary aspergillosis). We report a highly relevant interaction between voriconazole and flucloxacillin, resulting in subtherapeutic plasma voriconazole concentrations in more than 50% of patients, that poses a severe threat if not managed properly.

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