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Chronic idiopathic systemic capillary leak syndrome: a case report
Ahmed M Alkhunaizi1, Abdullah H Kabbani2, Mohamed A ElTigani3
11Nephrology Section, Internal Medicine Services Division, Johns Hopkins Aramco Healthcare, Saudi Aramco, Box 10955, Dhahran, 31311 Saudi Arabia.
Summary
Idiopathic systemic capillary leak syndrome (ISCLS) is a rare disorder. This case highlights a chronic, treatment-resistant form of ISCLS presenting with persistent edema and monoclonal gammopathy.
Area of Science:
- Rare diseases
- Hematology
- Immunology
Background:
- Idiopathic systemic capillary leak syndrome (ISCLS) is a rare disorder causing hypovolemic shock and edema.
- Chronic ISCLS is exceptionally rare, with limited documented cases.
Observation:
- A young woman presented with massive, persistent generalized edema, indicative of chronic ISCLS.
- Diagnostic workup revealed monoclonal gammopathy in the patient.
Findings:
- The patient's chronic ISCLS was refractory to multiple therapeutic interventions.
- Monoclonal gammopathy was a key finding in this rare presentation of ISCLS.
Implications:
- This case underscores the rarity and challenging nature of chronic ISCLS.
- Increased awareness of chronic ISCLS is crucial for potential future targeted therapies.
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