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Abstract Hemophagocytic Lymphohistiocytosis syndrome is fatal hyper‐inflammatory condition due to over‐activation of the immune system, being of primary and secondary types. This case report emphasizes the difficulty and challenge in and of the HLH diagnosis, and therapy should be employed promptly given the high mortality associated with HLH.

Celiac disease is a multisystem disorder in children and adults; it is considered as one of the most important malabsorption syndromes related to an auto-immune mediated response to gluten-containing food in a genetically predisposed patient. The most common involved system is the Gastrointestinal tract. Association with other autoimmune diseases such as thyroid disease and diabetes mellitus type 1 is well known but autoimmune mediated hemolytic anemia that responded to a gluten-free diet was interesting to be found. A six years old male previously healthy child, presented with pallor and reduced activity, dark-colored urine, with clinical and laboratory features of hemolytic anemia, and multiple blood transfusions within a year of medical care-seeking to finally diagnosed with celiac disease and responded to a gluten-free diet of about 8 months now with no need of blood transfusion.