Bisoffi Zeno, Leoni Stefania, Buonfrate Dora, Lodesani Claudia, Eseme Franklin Esoka, Monteiro Geraldo Badona, Marocco Stefania, Guerriero Massimo
Centre for Tropical Diseases, Sacro Cuore-Don Calabria Hospital, 37024, Negrar, Verona, Italy.
Medici senza Frontiere Italia, Via Magenta 5, 00186, Roma, Italy.
Malar J. 2015 Dec 2;14:487. doi: 10.1186/s12936-015-1015-6.
The hyperreactive malarial splenomegaly (HMS) represents a chronic, potentially fatal complication of malaria. Case definition includes: gross splenomegaly, high level of anti-malarial antibody and IgM, response to long-term anti-malarial prophylaxis. In this study, a large series of patients not fully meeting the case definition was tentatively classified as early hyperreactive malarial splenomegaly (e-HMS). The main research questions was: does "e-HMS" tend to evolve to the full-blown syndrome? And if so, what are the main factors influencing this evolution?
Retrospective, longitudinal study. The patient database was searched to retrieve all potentially eligible patients. e-HMS was defined by splenomegaly of any size (with or without raised IgM), high anti-malarial antibody titre and exclusion of other causes of splenomegaly. The clinical outcome at following visits was analysed in relation to re-exposure to malaria, and to treatment (only part of the patients with e-HMS were treated with a single anti-malarial treatment and advised to follow an effective anti-malarial prophylaxis, if re-exposed). The association of the outcome with the main independent variables was first assessed with univariate analysis. A stepwise logistic regression model was then performed to study the association of the outcome with the main independent variables.
One hundred and twenty-six subjects with e-HMS were retrieved. Eighty-one had at least one follow-up visit. Of 46 re-exposed to malaria for a variable period, 21 (46 %) had progressed, including 10/46 (22 %) evolving to full-blown HMS, while of 29 patients not re-exposed, 24 (93 %) had improved or cured and five (7 %) progressed (p < 0.001). At logistic regression re-exposure was confirmed as a major risk factor of progression (OR 9.458, CI 1.767-50.616) while treatment at initial visit was protective (OR 0.187, CI 0.054-0.650).
e-HMS should be regarded as a clinical condition predisposing to HMS. Although the case definition may include false positives, e-HMS should be treated just as the full-blown syndrome. A single anti-malarial treatment is probably adequate, followed by effective prophylaxis for patients exposed again to malaria transmission.
高反应性疟疾脾肿大（HMS）是疟疾的一种慢性、潜在致命并发症。病例定义包括：脾脏显著肿大、高水平抗疟疾抗体和IgM、对长期抗疟疾预防措施有反应。在本研究中，一系列未完全符合病例定义的患者被暂定为早期高反应性疟疾脾肿大（e-HMS）。主要研究问题是：“e-HMS”是否倾向于发展为典型综合征？如果是，影响这种发展的主要因素是什么？
回顾性纵向研究。检索患者数据库以获取所有可能符合条件的患者。e-HMS的定义为任何程度的脾肿大（无论IgM是否升高）、高抗疟疾抗体滴度以及排除其他脾肿大原因。分析随访时的临床结局与再次接触疟疾以及治疗的关系（只有部分e-HMS患者接受了单一抗疟疾治疗，并被建议如果再次接触疟疾应采取有效的抗疟疾预防措施）。首先通过单因素分析评估结局与主要自变量的关联。然后进行逐步逻辑回归模型以研究结局与主要自变量的关联。
共检索到126例e-HMS患者。81例患者至少有一次随访。在46例再次接触疟疾不同时间段的患者中，21例（46%）病情进展，其中10/46例（22%）发展为典型HMS，而在29例未再次接触疟疾的患者中，24例（93%）病情改善或治愈，5例（7%）病情进展（p<0.001）。逻辑回归分析证实再次接触疟疾是病情进展的主要危险因素（OR 9.458，CI 1.767 - 50.616），而初次就诊时的治疗具有保护作用（OR 0.187，CI 0.054 - 0.650）。
e-HMS应被视为易发展为HMS的临床状态。尽管病例定义可能包括假阳性，但e-HMS应与典型综合征同样对待。单一抗疟疾治疗可能就足够了，随后应对再次接触疟疾传播的患者采取有效的预防措施。
