Bicytopenia - a rare hematological manifestation secondary to hypothalamic dysfunction in a case of craniopharyngioma: A case report

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Abstract

Introduction: Hypothalamic dysfunction can result in a wide range of endocrine and non-endocrine manifestations, including pituitary hormone deficiencies, disorders of water balance, obesity, thermoregulatory disturbances, autonomic dysfunction, behavioral changes, and sleep abnormalities. These complications often require long-term follow-up and periodic reassessment. Case Presentation: We present the case of an 11-year-old girl with craniopharyngioma who, following surgical resection and radiotherapy, developed multiple pituitary hormone deficiencies, adipsic diabetes insipidus, hyperphagia, cognitive disturbances, and delayed puberty. Unique to this case was the occurrence of cyclic bicytopenia mirroring thermal dysregulation episodes, in the absence of an identifiable hematological cause. As body temperature normalized, hematological parameters improved, suggesting a potential link between thermal dysregulation and cytopenia. Management involved hormone replacement, fluid and sodium balance monitoring, topical estrogen therapy, and lifestyle interventions. We address and review the mechanisms proposed for cytopenias related to thermoregulation and propose possible mechanisms for the other findings while discussing the therapeutic challenges in managing such complex cases. Conclusion: Postoperative management of craniopharyngioma is often complicated by involvement of multiple hypothalamic domains. This case emphasizes the importance of recognizing uncommon and underreported manifestations, such as thermoregulation-associated cytopenia and adipic diabetes insipidus. Early diagnosis and multidisciplinary management are essential for improving outcomes and long-term quality of life in affected children.

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