Hepatic Legionella pneumophila Infection in an Infant With Severe Combined Immunodeficiency

Lapidot, Rotem; Alawdah, Laila; Köhler, Juliane; Paulson, Vera; Levy, Ofer

doi:10.1097/inf.0000000000001789

Cited by 4 publications

(4 citation statements)

References 11 publications

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“…A patient with L. pneumophila infection shows pneumonic as well as a variety of extrapulmonary manifestations, such as headache, encephalopathy, relative bradycardia, abdominal pain, hepatic involvement, electrolyte imbalance and other multisystemic ndings [17] . Severe extrapulmonary ndings of L. pneumophila infection are rare, especially in children [18] .…”

Section: Discussionmentioning

confidence: 99%

“…As a result, pediatric doctors do not tend to systematically pursue diagnostic testing for L. pneumophila. Pediatric data from studies suggest that severe diseases of L. pneumophila occur in children with malignant neoplasms, organ transplantation, underlying pulmonary disease and immunosuppression with corticosteroids [10][11][12] . L. pneumophila belongs to Legionella which found in the environment, such as freshwater environments, moist soil and so on [13] .…”

Section: Discussionmentioning

confidence: 99%

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Severe Sepsis With Septic Shock as a Consequence of a Severe Hospital-Acquired Pneumonia Resulting From Legionella Pneumophila in Children: A Case Series and Literature Review

Ding

Yang

2020

Preprint

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BackgroundLegionella pneumophila is responsible for hospital or community-acquired pneumonia in adults. Immunocompromised patients with Legionella pneumophila infection are associated with rapidly severe clinical course and high mortality rates. Legionella pneumophila infection is rare in children, especially combined with extrapulmonary manifestations. In this report, we describe 2 children of severe hospital-acquired pneumonia and septic shock resulting from Legionella pneumophila. Standardized metagenomics next-generation sequencing allowed early diagnosis. Appropriate antibiotic therapy and timely extracorporeal life support were effective in achieving complete recovery. This is the first report of children with septic shock related to Legionella pneumophila infections diagnosed by metagenomics next-generation sequencing and recovered successfully.Case presentationThere were 2 cases about septic shock resulting from Legionella pneumophila. One was a six-month girl with congenital biliary atresia who underwent liver transplantation. She was suspected for rejecting the liver and admitted to hospital. The other one was a five-year-old boy with Burkitt lymphoma who was in the end of early chemotherapy. They both presented with fever, cough or shortness of breath during hospitalization. And they were transferred to PICU because of worsening dyspnea and decreased blood pressure. Patients were diagnosed by severe sepsis with septic shock likely resulting from hospital-acquired pneumonia. Metagenomics next-generation sequencing indicated L. pneumophila in blood and sputum. Aggressive intravenous fluids resuscitation and vasopressors were initiated on arrival to PICU, and they were placed on mechanical ventilation and continuous renal replacement therapy. Intravenous antibiotic therapy followed by azithromycin. Finally, the patients recovered without any long-term sequelae.ConclusionsThough sepsis or sepsis shock caused by Legionella pneumophila is rare in children, it can occur at high-risk population. Metagenomics next-generation sequencing is useful for conforming hard-to-culture pathogens and severely ill patients. The report remind pediatric physicians that we should be aware that Legionella pneumophila can cause severe sepsis or sepsis shock, especially in immunocompromised children. It is significant to select appropriate samples and pathogen detection methods in the early stage of disease.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Severe Sepsis With Septic Shock as a Consequence of a Severe Hospital-Acquired Pneumonia Resulting From Legionella Pneumophila in Children: A Case Series and Literature Review

Ding

Yang

2020

Preprint

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“…In addition, several extra thoracic involvements due to Legionella species infection have been described and well-documented, known as extrapulmonary legionellosis, particularly among immunocompromised patients. Within the cardiovascular system this can cause myopericarditis and endocarditis [55][56][57]; encephalitis, brain abscess or cerebellar ataxia as neurological complications [55,58,59]; a gastrointestinal compromise with pancreatitis, colitis, liver and spleen involvement [60]; joint [61] and skin damage (cellulitis, necrotising fasciitis) [62][63][64]; and disseminated intravascular coagulation, among others. Extrapulmonary forms mainly occur secondary to a pulmonary localization due to the spread of Legionella through the blood, even though the primary focus may be outside the lung: for example, single skin infection developing after direct inoculation from the environment [61,64].…”

Section: Clinical Picturementioning

confidence: 99%

Severe Legionnaires’ disease

Rello,

Allam,

Ruiz-Spinelli

et al. 2024

Ann. Intensive Care

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Background Legionnaires’ disease (LD) is a common but under-diagnosed cause of community-acquired pneumonia (CAP), although rapid detection of urine antigen testing (UAT) and advances in molecular testing have improved the diagnosis. LD entails intensive care unit (ICU) admission in almost one-third of cases, and the mortality rate ranges from 4% to 40%. This review aims to discuss recent advances in the study of this condition and to provide an update on the diagnosis, pathogenesis and management of severe LD. Results The overall incidence of LD has increased worldwide in recent years due to the higher number of patients with risk factors, especially immunosuppression, and to improvements in diagnostic methods. Although LD is responsible for only around 5% of all-cause CAP, it is one of the three most common causes of CAP requiring ICU admission. Mortality in ICU patients, immunocompromised patients or patients with a nosocomial source of LD can reach 40% despite appropriate antimicrobial therapy. Regarding pathogenesis, no Legionella-specific virulence factors have been associated with severity; however, recent reports have found high pulmonary Legionella DNA loads, and impairments in immune response and lung microbiome in the most severe cases. The clinical picture includes severe lung injury requiring respiratory and/or hemodynamic support, extrapulmonary symptoms and non-specific laboratory findings. LD diagnostic methods have improved due to the broad use of UAT and the development of molecular methods allowing the detection of all Lp serogroups. Therapy is currently based on macrolides, quinolones, or a combination of the two, with prolonged treatment in severe cases. Conclusions Numerous factors influence the mortality rate of LD, such as ICU admission, the underlying immune status, and the nosocomial source of the infection. The host immune response (hyperinflammation and/or immunoparalysis) may also be associated with increased severity. Given that the incidence of LD is rising, studies on specific biomarkers of severity may be of great interest. Further assessments comparing different regimens and/or evaluating host-directed therapies are nowadays needed. Graphical Abstract

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“…[24,29] Multifocal, necrotic abscesses from bacterial infections such as Legionella are only described in case reports. [30] Chronic active hepatitis, neonatal jaundice with transaminitis, and acute hepatic failure from noninfectious hepatitis is seen in SCID. [31][32][33] AIH with positive anti-smooth muscle, anti-liver and kidney microsomal antibodies, and histopathologic demonstration of piecemeal necrosis and bridging fibrosis have been reported in a patient with partial ADA deficiency.…”

Section: Severe Combined Immunodeficiencymentioning

confidence: 99%

Tip of the iceberg: A comprehensive review of liver disease in Inborn errors of immunity

et al. 2022

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Inborn errors of immunity (IEIs) consist of numerous rare, inherited defects of the immune system that affect about 500,000 people in the United States.As advancements in diagnosis through genetic testing and treatment with targeted immunotherapy and bone marrow transplant emerge, increasing numbers of patients survive into adulthood posing fresh clinical challenges.A large spectrum of hepatobiliary diseases now present in those with immunodeficiency diseases, leading to morbidity and mortality in this population. Awareness of these hepatobiliary diseases has lagged the improved management of the underlying disorders, leading to missed opportunities to improve clinical outcomes. This review article provides a detailed description of specific liver diseases occurring in various inborn errors of immunity.A generalized approach to diagnosis and management of hepatic complications is provided, and collaboration with hepatologists, immunologists, and pathologists is emphasized as a requirement for optimizing management and outcomes.

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Hepatic Legionella pneumophila Infection in an Infant With Severe Combined Immunodeficiency

Cited by 4 publications

References 11 publications

Severe Sepsis With Septic Shock as a Consequence of a Severe Hospital-Acquired Pneumonia Resulting From Legionella Pneumophila in Children: A Case Series and Literature Review

Severe Sepsis With Septic Shock as a Consequence of a Severe Hospital-Acquired Pneumonia Resulting From Legionella Pneumophila in Children: A Case Series and Literature Review

Severe Legionnaires’ disease

Tip of the iceberg: A comprehensive review of liver disease in Inborn errors of immunity

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