Syndrome d’activation macrophagique et grossesse : à propos d’un cas

Hannebicque-Montaigne, K.; Roch, Alexandra M.; Launay, David; Coulon, C.; Deruelle, Philippe; Langlois, Sylvie

doi:10.1016/j.annfar.2011.11.023

Cited by 11 publications

(3 citation statements)

References 10 publications

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“…In our patient, whose HLH developed in the context of pregnancy, it is possible that the immune changes and cytokine activation associated with pregnancy could have contributed to the development of this syndrome in a genetically predisposed individual. 25 , 26 Although HLH is commonly associated with adult-onset Still’s disease in the literature, we did not observe any such cases in our series. This is consistent with previous studies that have reported cases of HLH in patients with other underlying inflammatory conditions.…”

Section: Discussioncontrasting

confidence: 63%

“…Further studies are needed to explore this potential link and its implications for the diagnosis and management of HLH. 26,27 There is currently no standardized treatment protocol for HLH in adults, and treatment approaches are typically based on retrospective case series, case reports, or extrapolated from treatment guidelines for other conditions. Symptomatic treatment is the mainstay of therapy, with specific treatment indicated only in cases of extreme severity where the underlying pathology is controlled.…”

Section: Discussionmentioning

confidence: 99%

“…Further studies are needed to explore this potential link and its implications for the diagnosis and management of HLH. 26 , 27 …”

Section: Discussionmentioning

confidence: 99%

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Uncovering the Spectrum of Hemophagocytic Lymphohistiocytosis: A Nephrology Department’s Analysis of 14 Cases

Hajji,

Barbouch,

Kaaroud

et al. 2023

Clin Med Insights Case Rep

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Introduction: Hemophagocytic lymphohistiocytosis (HLH) is a disease of multi-organ dysfunction due to excessive immune activation causing widespread inflammation and tissue destruction. It is a severe condition associated with high morbidity and mortality. Early identification is crucial for prompt treatment. The objective of this case series is to underscore the intricacy of managing HLH in individuals with renal dysfunction. Methods: This is a retrospective study of patients diagnosed with HLH in a nephrology department over a period of 30 years. We retrospectively reviewed the medical files by applying the Revised HLH-2004 criteria. Results: Among the 14 female patients included, the mean age was 45.2 years (range 23-78). Nine patients presented with sudden onset of fever and chills. Physical examination revealed purpura in 3 cases, hepatomegaly and splenomegaly in 6 and 5 cases respectively, and peripheral lymphadenopathy in 1 case. Hemorrhagic complications were observed in 5 cases, hypertriglyceridemia in 9 cases, and hyperferritinemia in all cases. Hypothyroidism was observed in all cases, and impaired renal function was detected in 11 of them, with 5 experiencing it as a result of lupus nephritis, and 1 case attributed to pre-eclampsia. Hemophagocytosis was confirmed through sternal puncture in 11 cases. Treatment involved etiological therapy with corticosteroids and immunosuppressants and/or anti-infectives. Intravenous immunoglobulins were administered in 6 cases, while 2 cases required coagulation factor transfusions. Unfortunately, 9 patients did not survive. Conclusion: The study highlights the need for increased awareness and prompt recognition of HLH, particularly in patients with associated renal complications.

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Section: Discussioncontrasting

confidence: 63%

Section: Discussionmentioning

confidence: 99%

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Uncovering the Spectrum of Hemophagocytic Lymphohistiocytosis: A Nephrology Department’s Analysis of 14 Cases

Hajji,

Barbouch,

Kaaroud

et al. 2023

Clin Med Insights Case Rep

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Hemophagocytic syndrome in pregnancy: case report, diagnosis, treatment, and prognosis

Rousselin

Alavi

Moigne

et al. 2017

Clinical Case Reports

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Adult-onset Still’s disease complicated by macrophage activation syndrome during pregnancy: a case-based review

Wise

Zell

2023

Clin Rheumatol

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Adult-onset Still’s disease is a rare, systemic inflammatory rheumatic disease characterized by recurrent fevers, arthritis, and an evanescent rash. One of the most serious hematologic derangements that can be seen with adult-onset Still’s disease is macrophage activation syndrome. Macrophage activation syndrome is characterized by activation of lymphocytes, resulting in a cytokine storm and hemophagocytosis in the bone marrow, along with multi-organ failure. Adult-onset Still’s disease with macrophage activation syndrome first presenting during pregnancy is exceptionally rare; here, we report two unique cases of such a presentation and review the pertinent literature. Both of our cases presented critically ill with end-organ failure, and responded to immunosuppression; fetal demise was present in one and an emergency caesarean section with a viable fetus was performed in the other patient. Maternal outcomes were favorable in both cases and both patients did well long-term with systemic therapy. Systemic immunosuppression, particularly anti-IL1 therapy, may be considered as treatment for this rare and life-threatening condition when presenting during pregnancy.

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Syndrome d’activation macrophagique et grossesse : à propos d’un cas

Cited by 11 publications

References 10 publications

Uncovering the Spectrum of Hemophagocytic Lymphohistiocytosis: A Nephrology Department’s Analysis of 14 Cases

Uncovering the Spectrum of Hemophagocytic Lymphohistiocytosis: A Nephrology Department’s Analysis of 14 Cases

Hemophagocytic syndrome in pregnancy: case report, diagnosis, treatment, and prognosis

Adult-onset Still’s disease complicated by macrophage activation syndrome during pregnancy: a case-based review

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