A Cohort of 169 Chronic Granulomatous Disease Patients Exposed to BCG Vaccination: a Retrospective Study from a Single Center in Shanghai, China (2004–2017)

Zhou, Qinhua; Hui, Xiaoying; Ying, Wenjing; Hou, Jia‐Woei; Wang, Wenjie; Liu, Danru; Wang, Ying; Yu, Yizhen; Wang, Jingyi; Sun, Jinqiao; Zhang, Qian; Wang, Xiaochuan

doi:10.1007/s10875-018-0486-y

Cited by 57 publications

(66 citation statements)

References 54 publications

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“…In addition, previous studies have shown that 30%‐40% of CGD patients have a family history of CGD, whereas in our study, only 21% had a positive family history of CGD. In our study, nearly 64% of the CGD pediatric patients suffered from abnormal responses to BCG vaccination, which was similar to Zhou et al's study . They reported a cohort of 169 CGD patients exposed to BCG vaccination and showed a high incidence (59%) of BCG infection in China .…”

Section: Discussionsupporting

confidence: 90%

“…In our study, nearly 64% of the CGD pediatric patients suffered from abnormal responses to BCG vaccination, which was similar to Zhou et al's study. 11 They reported a cohort of 169 CGD patients exposed to BCG vaccination and showed a high incidence (59%) of BCG infection in China. 11 In Hong Kong and Iran, 47% and 56% of CGD patients develop BCGitis.…”

Section: Discussionmentioning

confidence: 99%

“…11 They reported a cohort of 169 CGD patients exposed to BCG vaccination and showed a high incidence (59%) of BCG infection in China. 11 In Hong Kong and Iran, 47% and 56% of CGD patients develop BCGitis. 12,13 Apart from CGD, BCG infections are reported in other primary immunodeficiency diseases (PID), including severe combined immunodeficiency syndrome (SCID), Mendelian susceptibility to mycobacterial diseases (MSMD), and X-linked hyper-IgM syndrome (XHIM).…”

Section: Discussionmentioning

confidence: 99%

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Clinical and genetic characteristics of Chinese pediatric patients with chronic granulomatous disease

Gao

Yin

Tong

et al. 2019

Pediatric Allergy Immunology

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BackgroundChronic granulomatous disease (CGD) is a rare disease in China, and very little large‐scale studies have been conducted to date. We aimed to investigate the clinical and genetic features of CGD in Chinese pediatric patients.MethodsPediatric patients with CGD from Beijing Children's Hospital, Capital Medical University, China, were enrolled from January 2006 to December 2016.ResultsA total of 159 pediatric patients with CGD were enrolled. The median age of clinical onset was 1.4 months, and 73% (116/159) had clinical onset symptoms before the 1 year of age. The most common site of invasion was the lungs. The lymph nodes, liver, and skin were more frequently invaded in X‐linked (XL) CGD patients than in autosomal recessive (AR) CGD patients (P < 0.05). Approximately 64% (92/144) of the pediatric patients suffered from abnormal response to BCG vaccination. The most frequent pathogens were Aspergillus and Mycobacterium tuberculosis. Gene analysis indicated that 132 cases (89%, 132/147) harbored CYBB pathogenic variants, 7 (5%, 7/147) carried CYBA pathogenic variants, 4 (3%, 4/147) had NCF1 pathogenic variants, and 4 (3%, 4/147) had NCF2 pathogenic variants. The overall mortality rate in this study was 43%, particularly the patients were males, with CYBB mutant and did not receive HSCT treatment.ConclusionsChronic granulomatous disease is a rare disease affecting Chinese children; however, it is often diagnosed at a later age, and thus, the mortality rate is relatively high. The prevalence and the severity of disease in XL‐CGD are higher than AR‐CGD.

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Section: Discussionsupporting

confidence: 90%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Clinical and genetic characteristics of Chinese pediatric patients with chronic granulomatous disease

Gao

Yin

Tong

et al. 2019

Pediatric Allergy Immunology

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“…In Comèl‐Netherton syndrome, 8/9 described patients showed recurrent or persistent S. aureus skin infections once skin lesions had developed . Lastly, S. aureus was isolated in 1/4 patients with CGD and suppurative dermatitis …”

Section: Resultsmentioning

confidence: 99%

Skin disorders are prominent features in primary immunodeficiency diseases: A systematic overview of current data

Wit

Brada

Veldhuizen

et al. 2018

Allergy

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Primary immunodeficiency diseases (PIDs) are characterized by an increased risk of infections, autoimmunity, autoinflammation, malignancy, and allergic disorders. Skin disorders are also common clinical features in PIDs and may be among the presenting manifestations. Recognition of specific PID-associated skin conditions in combination with other clinical features as described in the currently used warning signs could raise suspicion of an underlying PID. We aimed to provide a systematically obtained overview of skin disorders and their prevalence in PIDs. Secondary, the prevalence of Staphylococcus (S.) aureus-associated skin disorders and atopy was reviewed, as these are the most prominent skin features in PIDs. A systematic search was performed in EMBASE, MEDLINE, Web of Science, Cochrane, and Google Scholar (up to May 9, 2018). All original observational and experimental human studies that address the presence of skin disorders in PIDs were selected. We rated study quality using the Institute of Health Economics Quality Appraisal Checklist for Case Series Studies. Sixty-seven articles (5030 patients) were included. Study quality ranged from 18.2% to 88.5%. A broad spectrum of skin disorders was reported in 30 PIDs, mostly in single studies with a low number of included patients. An overview of associated PIDs per skin disorder was generated. Data on S. aureus-associated skin disorders and atopy in PIDs were limited. In conclusion, skin disorders are prominent features in PIDs. Through clustering of PIDs per skin disorder, we provide a support tool to use in clinical practice that should raise awareness of PIDs based on presenting skin manifestations.

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“…Este último método tem como vantagens a necessidade de uso de pouco volume de sangue periférico de pacientes, a rapidez na aquisição dos resultados e a confiabilidade (Kim, Kim et al 2013). (Fattahi, Badalzadeh et al 2011;Yang, Cambier et al 2012;Koker, Camcioglu et al 2013;Zhou, Hui et al 2018 (Margolis, Melnick et al 1990;Holland 2010).…”

Section: Introductionunclassified

Caracterização clínica e genética de pacientes brasileiros com doença granulomatosa crônica e susceptibilidade mendeliana a infecções por micobactérias

Zurro¹

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A Cohort of 169 Chronic Granulomatous Disease Patients Exposed to BCG Vaccination: a Retrospective Study from a Single Center in Shanghai, China (2004–2017)

Abstract: Similar to other non-consanguineous populations, X-linked CGD accounted for the majority of the cases in China. However, BCG infections were a clinical challenge unique to China. In addition, severe infections were the major cause of death and the overall mortality was still high in China.

Cited by 57 publications

References 54 publications

Clinical and genetic characteristics of Chinese pediatric patients with chronic granulomatous disease

Clinical and genetic characteristics of Chinese pediatric patients with chronic granulomatous disease

Skin disorders are prominent features in primary immunodeficiency diseases: A systematic overview of current data

Caracterização clínica e genética de pacientes brasileiros com doença granulomatosa crônica e susceptibilidade mendeliana a infecções por micobactérias

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