Inflammation and Rheumatology Section, University College London Great Ormond Street Institute of Child Health, London, United Kingdom.
National Amyloidosis Centre, Royal Free Hospital, London, United Kingdom.
Front Immunol. 2022 Sep 20;13:998967. doi: 10.3389/fimmu.2022.998967. eCollection 2022.
There is an important unmet clinical need for fast turnaround next generation sequencing (NGS) to aid genetic diagnosis of patients with acute and sometimes catastrophic inflammatory presentations. This is imperative for patients who require precise and targeted treatment to prevent irreparable organ damage or even death. Acute and severe hyper- inflammation may be caused by primary immunodeficiency (PID) with immune dysregulation, or more typical autoinflammatory diseases in the absence of obvious immunodeficiency. Infectious triggers may be present in either immunodeficiency or autoinflammation. We compiled a list of 25 genes causing monogenetic immunological diseases that are notorious for their acute first presentation with fulminant inflammation and which may be amenable to specific treatment, including hemophagocytic lymphohistiocytosis (HLH); and autoinflammatory diseases that can present with early-onset stroke or other irreversible neurological inflammatory complications. We designed and validated a pipeline that enabled return of clinically actionable results in hours rather than weeks: the Rapid Autoinflammation Panel (RAP). We demonstrated accuracy of this new pipeline, with 100% sensitivity and 100% specificity. Return of results to clinicians was achieved within 48-hours from receiving the patient's blood or saliva sample. This approach demonstrates the potential significant diagnostic impact of NGS in acute medicine to facilitate precision medicine and save "life or limb" in these critical situations.
临床上急需快速进行下一代测序(NGS),以帮助对表现为急性、有时甚至危及生命的炎症的患者进行基因诊断。这对于那些需要精确靶向治疗以防止不可逆转的器官损伤甚至死亡的患者来说至关重要。急性和严重的超炎症可能由免疫失调引起的原发性免疫缺陷(PID)引起,或者在没有明显免疫缺陷的情况下由更典型的自身炎症性疾病引起。在免疫缺陷或自身炎症中都可能存在感染诱因。我们编制了一份 25 个基因的清单,这些基因导致单基因免疫性疾病,这些疾病以其急性首发、迅速炎症为特征,可能适合特定治疗,包括噬血细胞性淋巴组织细胞增生症(HLH);以及可能表现为早发性中风或其他不可逆转的神经炎症并发症的自身炎症性疾病。我们设计并验证了一个能够在数小时内而不是数周内提供临床可操作结果的管道:快速自身炎症面板(RAP)。我们证明了这个新管道的准确性,具有 100%的敏感性和 100%的特异性。从收到患者的血液或唾液样本到将结果反馈给临床医生,这一过程在 48 小时内完成。这种方法展示了 NGS 在急性医学中的潜在重大诊断影响,以促进精准医学,并在这些危急情况下挽救“生命或肢体”。