scholarly journals Patellar Osteomyelitis in a 9-Year-Old Patient with Chronic Granulomatous Disease: A Case Report

Children ◽  
2022 ◽  
Vol 9 (1) ◽  
pp. 76
Author(s):  
Yonggeun Park ◽  
Seungjin Yoo ◽  
Yongyeon Chu ◽  
Chaemoon Lim
Keyword(s):  
Chronic Granulomatous Disease ◽  
Fungal Infections ◽  
Pediatric Population ◽  
Clinical Manifestations ◽  
Granulomatous Disease ◽  
Advanced Imaging ◽  
Hematogenous Osteomyelitis ◽  
First Case ◽  
Acute Hematogenous Osteomyelitis ◽  
High Index Of Suspicion

Hematogenous osteomyelitis is commonly reported in long tubular bones in the pediatric population. Acute osteomyelitis involving the patella is extremely uncommon in children, and its diagnosis is frequently delayed due to its rarity and variable clinical manifestations. Chronic granulomatous disease (CGD) is a rare genetic immunodeficiency disorder characterized by severe recurrent bacterial and fungal infections. The most commonly affected sites of infection are the lungs, lymph nodes, skin, liver, and gastrointestinal tract. Acute hematogenous osteomyelitis of the patella associated with CGD has never been reported. Our report describes the first case of acute hematogenous patellar osteomyelitis in a pediatric patient with CGD. Her clinical manifestations were similar to other possible differentials such as septic arthritis; however, use of advanced imaging confirmed the diagnosis, and the patient was successfully managed surgically. Since hematogenous osteomyelitis in children is uncommon, a high index of suspicion and advanced imaging may help with its diagnosis, and in cases where antibiotic treatment proves to be insufficient, prompt surgical management is imperative.

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.

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.

Disseminated Infection Due to Chrysosporium zonatum in a Patient with Chronic Granulomatous Disease and Review of Non-Aspergillus Fungal Infections in Patients with This Disease

1999 ◽  
Vol 37 (1) ◽  
pp. 18-25 ◽  
Author(s):  
Emmanuel Roilides ◽  
Lynne Sigler ◽  
Evangelia Bibashi ◽  
Helen Katsifa ◽  
Nicolas Flaris ◽  
...  
Keyword(s):  
Chronic Granulomatous Disease ◽  
Amphotericin B ◽  
Liposomal Amphotericin ◽  
Clinical Laboratory ◽  
Fungal Infections ◽  
National Committee ◽  
Granulomatous Disease ◽  
First Case ◽  
Mating Tests ◽  
Yellowish White

We report the first case of Chrysosporium zonatuminfection in a 15-year-old male with chronic granulomatous disease who developed a lobar pneumonia and tibia osteomyelitis while on prophylaxis with gamma interferon. The fungus was isolated from sputum and affected bone, and hyphae were observed in the bone by histopathology. Therapy with amphotericin B eradicated the osteomyelitis and pneumonia, but pneumonia recurred in association with pericarditis and pleuritis during therapy with itraconazole. These manifestations subsided, and no recurrences occurred with liposomal amphotericin B therapy. Infections caused by Chrysosporium species are very rare, andC. zonatum has not previously been reported to cause mycosis in humans. This species, the anamorph of the heterothallic ascomycete Uncinocarpus orissi (family Onygenaceae), is distinguished by its thermotolerance, by colonies which darken from yellowish white to buff, and by club-shaped terminal aleurioconidia borne at the ends of short, typically curved stalks. The case isolate produced fertile ascomata in mating tests with representative isolates. The median (range) MICs for our isolate as well as those for two other human isolates and a nonhuman isolate determined by the National Committee for Clinical Laboratory Standards method adapted for moulds were ≤0.06 μg/ml (≤0.06 to 0.25 μg/ml) for amphotericin B, 0.687 μg/ml (0.25 to 2 μg/ml) for itraconazole, >128 μg/ml (>128 μg/ml) for flucytosine, and 48 μg/ml (32 to >128 μg/ml) for fluconazole.

scholarly journals Granulomatous inflammation in the manifestation of chronic granulomatous disease: a clinical case report

2019 ◽  
Vol 18 (4) ◽  
pp. 99-104 ◽  
Author(s):  
D. V. Yukhacheva ◽  
D. E. Pershin ◽  
N. G. Uskova ◽  
G. V. Tereshchenko ◽  
N. B. Kuzmenko
Keyword(s):  
Chronic Granulomatous Disease ◽  
Clinical Case ◽  
Fungal Infections ◽  
Interleukin 1 ◽  
Clinical Manifestations ◽  
Granulomatous Inflammation ◽  
Personal Data ◽  
Unrelated Donor ◽  
Granulomatous Disease ◽  
Hematopoietic Stem

Chronic granulomatous disease is a primary immunodeficiency, characterized by a violation of the oxygen-dependent mechanisms of phagocytosis. Mutations in the genes encoding proteins of the NADPH-oxidase complex lead to a violation of the respiratory burst. Clinical manifestations are recurrent bacterial and fungal infections, and the development of granulomatous complications due to a defect in autophagy, accompanied by an increase in the level of interleukin-1 inthe blood. The treatment of this disease is continuous preventive antimicrobial therapy, and specific therapy for the treatment of granulomatous complications. Hematopoietic stem cell transplantation from an allogeneic or unrelated donor is currently considered to be only radical treatment for CGD. This article presents a clinical case of the manifestation of an X-linked form of chronic granulomatous disease with granulomatous manifestations in the absence of an infectious history. Parents patient agreed to use personal data in research and publications.

scholarly journals A Case of COVID-19-Related Thrombocytopenia and Leukopenia in an Adolescent with Mild Symptoms

Children ◽  
2021 ◽  
Vol 8 (6) ◽  
pp. 509
Author(s):  
Lydia Kossiva ◽  
Athanasios Thirios ◽  
Eleni Panagouli ◽  
Alexandros Panos ◽  
Stavroula Lampidi ◽  
...  
Keyword(s):  
Bone Marrow ◽  
Viral Infection ◽  
Nasal Congestion ◽  
Pediatric Population ◽  
Clinical Manifestations ◽  
Bone Marrow Aspiration ◽  
White Blood Count ◽  
Chronic Obstructive ◽  
First Case ◽  
The Impact

Since the beginning of the COVID-19 pandemic, there have been numerous reports and reviews on the complications caused by the disease, analyzing the acute and chronic consequences. The main symptoms of SARS-CoV-2 are dry cough, fever, and fatigue. COVID-19 appears to affect all systems, including renal, cardiovascular, circulatory, and respiratory systems, causing chronic obstructive pulmonary disease. We report on a 14-year-old male adolescent, who presented with thrombocytopenia (platelet count 92 × 109 /L) and leukopenia (white blood count 4.2 × 103 /μL) that was observed two months ago. Ten days before the first blood test, a viral infection with nasal congestion and runny nose was reported, without other accompanying symptoms. Viral antibodies screening revealed positivity for all the three specific COVID-19 antibodies. Further haematological evaluation with bone marrow aspiration revealed non-specific dysplastic features of the red cell and megakaryocyte progenitors. Although haematological alterations due to COVID-19 infection are available from adult patients’ reports, the effect of COVID-19 infection in the pediatric population is underestimated and this is the first case with such haematological involvement. Noteworthy, in the current case, the impact of the COVID-19 infection was not related to the severity of the disease, as the symptoms were mild. In similar cases, bone marrow aspiration would not be performed as a part of routine work-up. Thus, it is important when evaluating pediatric patients with COVID-19 infection to search and report those alterations in order to better understand the impact and the spectrum of clinical manifestations of the specific viral infection in children and adolescents.

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.

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 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.

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