COVID-19 in Chronic Granulomatosis Disease: A Case Report

Esmaeilzadeh, Hossein; Dehghani, Seyed Sina; Shahhoseini, Babak; Alyasin, Soheila; Nabavizadeh, Seyed Hesamedin; Askari, Aida

doi:10.18502/ijaai.v21i4.10295

Cited by 4 publications

(12 citation statements)

References 23 publications

(26 reference statements)

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“…The detailed characteristics of the included studies are shown in Additional file 2 : Table S3. There were 73 case report [ 4 – 6 , 8 , 9 , 25 – 32 , 37 – 39 , 44 , 45 , 49 , 51 , 53 – 60 , 67 – 69 , 72 , 76 – 79 , 82 – 84 , 86 – 88 , 90 , 91 , 93 , 94 , 96 , 99 – 107 , 109 – 114 , 116 , 119 – 123 , 126 , 127 , 129 – 131 ], 38 cohort [ 7 , 10 , 19 , 33 – 36 , 40 – 43 , 46 – 48 , 50 , 52 , 61 – 66 , 70 , 71 , 73 , 74 , 80 , 81 , 92 , 95 , 97 , 98 , 108 , 117 , 118 , 124 , 125 , 132 ], 4...…”

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

“…Phagocytic diseases were the sixth most-common IEIs in children who experienced COVID-19 (n = 54, 7.6%) [ 19 , 40 , 43 , 46 , 48 , 52 , 53 , 61 – 63 , 65 , 74 , 77 , 81 , 90 , 92 , 95 , 98 , 101 , 107 , 117 , 124 , 127 ] (see Additional file 2 : Table S3). Among them, 26 have chronic granulomatous disease (48.1% of all phagocytic diseases) [ 40 , 43 , 46 , 48 , 53 , 62 , 65 , 74 , 81 , 90 , 92 , 95 , 98 , 124 ], 8 have Shwachman-Diamond syndromes (14.8%) [ 61 , 95 , 101 ], 6 have HAX1 deficiencies (11.1%) [ 74 , 77 , 124 ], 2 have Glycogen storage diseases type 1b (3.7%) [ 62 ], and 2 have Elastase deficiency (3.7%) [ 81 , 127 ]. The remaining 10 patients have JAGN1 deficiency (n = 1) [ 117 ]; poikiloderma with neutropenia (n = 1) [ 107 ]; cystic fibrosis (n = 1) [ 19 ]; leukocyte adhesion deficiency type 3 (n = 1) [ 65 ]; GATA2 deficiency (n = 1) [ 95 ]; undefined leukopenia (n = 1) [ 46 ]; and unspecified phagocytic diseases (n = 4) [ 52 , 63 , 101 , 124 ].…”

Section: Resultsmentioning

confidence: 99%

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Severity of SARS-CoV-2 infection in children with inborn errors of immunity (primary immunodeficiencies): a systematic review

Alhumaid,

Al Mutared,

Al Alawi

et al. 2023

Allergy Asthma Clin Immunol

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Background Inborn errors of immunity (IEIs) are considered significant challenges for children with IEIs, their families, and their medical providers. Infections are the most common complication of IEIs and children can acquire coronavirus disease 2019 (COVID-19) even when protective measures are taken. Objectives To estimate the incidence of severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection in children with IEIs and analyse the demographic parameters, clinical characteristics and treatment outcomes in children with IEIs with COVID-19 illness. Methods For this systematic review, we searched ProQuest, Medline, Embase, PubMed, CINAHL, Wiley online library, Scopus and Nature through the Preferred Reporting Items for Systematic Reviews and Meta Analyses (PRISMA) guideline for studies on the development of COVID-19 in children with IEIs, published from December 1, 2019 to February 28, 2023, with English language restriction. Results Of the 1095 papers that were identified, 116 articles were included in the systematic review (73 case report, 38 cohort 4 case-series and 1 case–control studies). Studies involving 710 children with IEIs with confirmed COVID-19 were analyzed. Among all 710 IEIs pediatric cases who acquired SARS-CoV-2, some children were documented to be admitted to the intensive care unit (ICU) (n = 119, 16.8%), intubated and placed on mechanical ventilation (n = 87, 12.2%), suffered acute respiratory distress syndrome (n = 98, 13.8%) or died (n = 60, 8.4%). Overall, COVID-19 in children with different IEIs patents resulted in no or low severity of disease in more than 76% of all included cases (COVID-19 severity: asymptomatic = 105, mild = 351, or moderate = 88). The majority of children with IEIs received treatment for COVID-19 (n = 579, 81.5%). Multisystem inflammatory syndrome in children (MIS-C) due to COVID-19 in children with IEIs occurred in 103 (14.5%). Fatality in children with IEIs with COVID-19 was reported in any of the included IEIs categories for cellular and humoral immunodeficiencies (n = 19, 18.6%), immune dysregulatory diseases (n = 17, 17.9%), innate immunodeficiencies (n = 5, 10%), bone marrow failure (n = 1, 14.3%), complement deficiencies (n = 1, 9.1%), combined immunodeficiencies with associated or syndromic features (n = 7, 5.5%), phagocytic diseases (n = 3, 5.5%), autoinflammatory diseases (n = 2, 3%) and predominantly antibody deficiencies (n = 5, 2.5%). Mortality was COVID-19-related in a considerable number of children with IEIs (29/60, 48.3%). The highest ICU admission and fatality rates were observed in cases belonging to cellular and humoral immunodeficiencies (26.5% and 18.6%) and immune dysregulatory diseases (35.8% and 17.9%) groups, especially in children infected with SARS-CoV-2 who suffered severe combined immunodeficiency (28.6% and 23.8%), combined immunodeficiency (25% and 15%), familial hemophagocytic lymphohistiocytosis (40% and 20%), X-linked lymphoproliferative diseases-1 (75% and 75%) and X-linked lymphoproliferative diseases-2 (50% and 50%) compared to the other IEIs cases. Conclusion Children with IEIs infected with SARS-CoV-2 may experience higher rates of ICU admission and mortality in comparison with the immunocompetent pediatric populations. Underlying immune defects does seem to be independent risk factors for severe SARS-CoV-2 infection in children with IEIs, a number of children with SCID and CID were reported to have prolonged infections–though the number of patients is small–but especially immune dysregulation diseases (XLP1 and XLP2) and innate immunodeficiencies impairing type I interferon signalling (IFNAR1, IFNAR2 and TBK1).

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

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Severity of SARS-CoV-2 infection in children with inborn errors of immunity (primary immunodeficiencies): a systematic review

Alhumaid,

Al Mutared,

Al Alawi

et al. 2023

Allergy Asthma Clin Immunol

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“…It has been hypothesized that defective respiratory burst and impaired neutrophil extracellular trap formation in CGD may prevent exaggerated lung tissue damage seen in severe COVID-19 infection, and probably has led to better outcomes in this subset of IEIs [ 39 ]. Nevertheless, complications including mortalities have also been reported in certain studies [ 40 – 42 ]. Castano‐Jaramillo et al reported a fatal MIS-C-like illness in a 16-year-old boy with CYBB mutation [ 41 ].…”

Section: Discussionmentioning

confidence: 99%

Impact of COVID-19 Pandemic on Clinical Care of Patients and Psychosocial Health of Affected Families with Chronic Granulomatous Disease: an Observational Study from North India

et al. 2023

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Day-to-day clinical management of patients with inborn errors of immunity, including chronic granulomatous disease (CGD), has been affected by the coronavirus disease-2019 (COVID-19) pandemic. There is a dearth of information on impact of this pandemic on clinical care of children with CGD and psychological profile of the caretakers. Among the 101 patients with CGD followed up in our center, 5 children developed infection/complications associated with COVID-19. Four of these children had a mild clinical course, while 1 child developed features of multisystem inflammatory syndrome in children (MISC) requiring intravenous glucocorticoids. Parents and caretakers of CGD patients ( n = 21) and 21 healthy adults with similar ages and genders were also evaluated on the following scales and questionnaires: COVID-19 Fear Scale (FCV 19S), Impact of Event Scale (IES-R), Depression, Anxiety, and Stress Scale (DASS 21), Preventive COVID-19 Behavior Scale (PCV 19BS), and a “COVID-19 Psychological wellbeing questionnaire.” Median age of the parents/caregivers was 41.76 years (range: 28–60 years). Male:female ratio was 2:1. In the study group, 71.4% had higher IES scores compared to 14.3% in controls. The caregivers had a high prevalence of stress, anxiety, avoidance behavior, and depression compared to controls ( p < 0.001). Children with CGD have had predominantly mild infection with COVID-19; however, caregivers/parents of these children were at risk of developing psychological distress. The COVID-19 pandemic has brought to light the importance of patients’ and caretakers’ mental health which needs periodic assessment and appropriate interventions. Supplementary Information The online version contains supplementary material available at 10.1007/s10875-023-01524-5.

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“…However, CGD patients have normal immunity to most viruses. Two patients with severe COVID-19 have been reported in the literature, which suggests that COVID-19 might have a different pathogenesis than other viruses [ 46 ].…”

Section: Pathogens and Cgd-related Infectious Diseasesmentioning

confidence: 99%

Chronic Granulomatous Disease (CGD): Commonly Associated Pathogens, Diagnosis and Treatment

Justiz-Vaillant,

Williams-Persad,

Arozarena-Fundora

et al. 2023

Microorganisms

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Chronic granulomatous disease (CGD) is a primary immunodeficiency caused by a defect in the phagocytic function of the innate immune system owing to mutations in genes encoding the five subunits of the nicotinamide adenine dinucleotide phosphatase (NADPH) oxidase enzyme complex. This review aimed to provide a comprehensive approach to the pathogens associated with chronic granulomatous disease (CGD) and its management. Patients with CGD, often children, have recurrent life-threatening infections and may develop infectious or inflammatory complications. The most common microorganisms observed in the patients with CGD are Staphylococcus aureus, Aspergillus spp., Candida spp., Nocardia spp., Burkholderia spp., Serratia spp., and Salmonella spp. Antibacterial prophylaxis with trimethoprim-sulfamethoxazole, antifungal prophylaxis usually with itraconazole, and interferon gamma immunotherapy have been successfully used in reducing infection in CGD. Haematopoietic stem cell transplantation (HCT) have been successfully proven to be the treatment of choice in patients with CGD.

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COVID-19 in Chronic Granulomatosis Disease: A Case Report

Cited by 4 publications

References 23 publications

Severity of SARS-CoV-2 infection in children with inborn errors of immunity (primary immunodeficiencies): a systematic review

Severity of SARS-CoV-2 infection in children with inborn errors of immunity (primary immunodeficiencies): a systematic review

Impact of COVID-19 Pandemic on Clinical Care of Patients and Psychosocial Health of Affected Families with Chronic Granulomatous Disease: an Observational Study from North India

Chronic Granulomatous Disease (CGD): Commonly Associated Pathogens, Diagnosis and Treatment

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