Haemolytic Uremic Syndrome Associated with Citrobacter freundii in a Young Boy with X-Linked Agammaglobulinemia
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LETTER TO EDITOR
Haemolytic Uremic Syndrome Associated with Citrobacter freundii in a Young Boy with X-Linked Agammaglobulinemia Murugan Sudhakar 1 & Manpreet Arora 1 & Lesa Dawman 1 & Dharmagat Bhattarai 1 & Pratap Kumar Patra 1 & Madhubala Sharma 1 & Ankur Kumar Jindal 1 & Ritambhra Nada 2 & Amit Rawat 1 & Karalanglin Tiewsoh 1 Received: 2 September 2020 / Accepted: 29 September 2020 # Springer Science+Business Media, LLC, part of Springer Nature 2020
To the Editor, X-linked agammaglobulinemia (XLA) is one of the commonest inborn error of immunity that results in defective antibody production. XLA is characterized by panhypogammaglobulinemia resulting in recurrent infections with encapsulated bacteria (such as Streptococci, Staphylococci, and Haemophilus influenzae), enteroviruses (such as poliovirus, echovirus, and coxsackievirus), hepatitis viruses and Giardia. Citrobacter freundii is a normal gut commensal that has mostly been reported as healthcare-associated infection (HCAI) and also in immunocompromised individuals [1, 2]. Renal manifestations in patients with XLA are extremely rare and limited to 2 cases of membranoproliferative glomerulonephritis [3] and 1 case of membranous nephropathy [4]. However, the pathophysiology of renal manifestations in XLA is unclear. No case of thrombotic microangiopathy (TMA) has been reported in these patients. Herein, we report a young boy with XLA with myriad clinical manifestations of the disease including TMA.
An 8-year-old boy presented with history of loose stools and vomiting for 5 days. He also developed reduced urine output and periorbital puffiness for 2 days prior to hospitalization. In the past, he was symptomatic since the age of 9 months when he developed acute flaccid paralysis with anterior horn myelitis. He was suspected to have vaccineassociated paralytic poliomyelitis (VAPP) leading to sequalae of thoracolumbar spine kyphoscoliosis (Supplementary Figure 1A). However, stool examination did not reveal vaccine-derived poliovirus strain. He had had 5 episodes of pneumonia since the age of 2 years (Supplementary Figure 1B, C, and D) requiring IV antimicrobials, 1 episode of persistent diarrhoea caused by Giardia lamblia, chronic bilateral ear discharge, and 1 episode of bacterial meningitis. At the age of 8, he developed septic arthritis of the right elbow, and pus culture grew Streptococcus pneumoniae. His family history revealed death of 3 maternal uncles and 3 maternal grand uncles in early childhood due to repeated sinopulmonary infections, suggestive of an X-linked recessive
Electronic supplementary material The online version of this article (https://doi.org/10.1007/s10875-020-00882-8) contains supplementary material, which is available to authorized users. * Lesa Dawman [email protected]
Ankur Kumar Jindal [email protected] Ritambhra Nada [email protected]
Murugan Sudhakar [email protected]
Amit Rawat [email protected]
Manpreet Arora [email protected]
Karalanglin Tiewsoh [email protected]
Dharmagat Bha
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