Distributive shock due to systemic capillary leak syndrome treated with high-dose immunosuppression
A female patient in her 60s presented with a history of malaise, chills, headache and vomiting. She was in shock on presentation with a high haematocrit and a low albumin with evidence of rhabdomyolysis. Severe limb and truncal oedema developed with worsening hypotension leading to intensive care un...
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| creator | Sheehan, James Robert Keating, Liza Chan, Antoni Walden, Andrew |
| description | A female patient in her 60s presented with a history of malaise, chills, headache and vomiting. She was in shock on presentation with a high haematocrit and a low albumin with evidence of rhabdomyolysis. Severe limb and truncal oedema developed with worsening hypotension leading to intensive care unit admission for multiple organ support. Extensive radiological, microbiological and immunological work up was negative with the exception of a monoclonal gammopathy. A review of patient investigations led to a diagnosis of Clarkson's disease. Treatment with high-dose methylprednisolone and intravenous immunoglobulins led to a rapid decline in the creatine kinase (CK) level and vasopressor requirements. The patient was discharged home on long-term terbutaline and has made a good recovery. |
| doi_str_mv | 10.1136/bcr-2013-009048 |
| format | Article |
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She was in shock on presentation with a high haematocrit and a low albumin with evidence of rhabdomyolysis. Severe limb and truncal oedema developed with worsening hypotension leading to intensive care unit admission for multiple organ support. Extensive radiological, microbiological and immunological work up was negative with the exception of a monoclonal gammopathy. A review of patient investigations led to a diagnosis of Clarkson's disease. Treatment with high-dose methylprednisolone and intravenous immunoglobulins led to a rapid decline in the creatine kinase (CK) level and vasopressor requirements. 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She was in shock on presentation with a high haematocrit and a low albumin with evidence of rhabdomyolysis. Severe limb and truncal oedema developed with worsening hypotension leading to intensive care unit admission for multiple organ support. Extensive radiological, microbiological and immunological work up was negative with the exception of a monoclonal gammopathy. A review of patient investigations led to a diagnosis of Clarkson's disease. Treatment with high-dose methylprednisolone and intravenous immunoglobulins led to a rapid decline in the creatine kinase (CK) level and vasopressor requirements. The patient was discharged home on long-term terbutaline and has made a good recovery.</description><subject>Abdomen</subject><subject>Blood pressure</subject><subject>Capillary Leak Syndrome - complications</subject><subject>Capillary Leak Syndrome - diagnosis</subject><subject>Capillary Leak Syndrome - drug therapy</subject><subject>Diagnosis, Differential</subject><subject>Edema</subject><subject>Female</subject><subject>Glucocorticoids - therapeutic use</subject><subject>Glucose</subject><subject>Hemoglobin</subject><subject>Humans</subject><subject>Immunoglobulins, Intravenous - therapeutic use</subject><subject>Immunologic Factors - therapeutic use</subject><subject>Kinases</subject><subject>Methylprednisolone - therapeutic use</subject><subject>Middle Aged</subject><subject>Potassium</subject><subject>Rare Disease</subject><subject>Shock - etiology</subject><subject>United Kingdom</subject><subject>White</subject><issn>1757-790X</issn><issn>1757-790X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2013</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><recordid>eNqFkc9L3jAYx8PYmOI87yYBL2PQmd9JL4I43QbCLhO8hTZ5avPaNjVpHf73y8vrRHdZLgnJJ1-eLx-EPlLyhVKuTlqXKkYorwipiTBv0D7VUle6JjdvX5z30GHOG1IWp8II_h7tMS61UoruI_c15CWFdl3CA-DcR3eH_Qp4iTg_5gXG4LBr5jAMTXrEAzR35X7yKY6FSdAs4PHvsPS4D7d95WMGHMZxnWJe5zlBziFOH9C7rhkyHD7tB-j68uLX-ffq6ue3H-dnV1UrBDVVzYjmUgKTriO-Ns5L4lUnW1orAdI4oqTyRDReQOcJE2BAKG1qIQ1ow_gBOt3lzms7gncwLakZ7JzCWIa3sQn29csUensbHyxXQjJpSsCnp4AU71fIix1DdlC6TxDXbClnSnNK6y16_A-6iWuaSj1LteHMMMVooU52lEsx5wTd8zCU2K1DWxzarUO7c1h-HL3s8Mz_NVaAzzugHTf_TfsDca6mRQ</recordid><startdate>20130409</startdate><enddate>20130409</enddate><creator>Sheehan, James Robert</creator><creator>Keating, Liza</creator><creator>Chan, Antoni</creator><creator>Walden, Andrew</creator><general>BMJ Publishing Group LTD</general><general>BMJ Publishing Group</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7RV</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>BTHHO</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>KB0</scope><scope>M0S</scope><scope>M1P</scope><scope>NAPCQ</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>20130409</creationdate><title>Distributive shock due to systemic capillary leak syndrome treated with high-dose immunosuppression</title><author>Sheehan, James Robert ; 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She was in shock on presentation with a high haematocrit and a low albumin with evidence of rhabdomyolysis. Severe limb and truncal oedema developed with worsening hypotension leading to intensive care unit admission for multiple organ support. Extensive radiological, microbiological and immunological work up was negative with the exception of a monoclonal gammopathy. A review of patient investigations led to a diagnosis of Clarkson's disease. Treatment with high-dose methylprednisolone and intravenous immunoglobulins led to a rapid decline in the creatine kinase (CK) level and vasopressor requirements. The patient was discharged home on long-term terbutaline and has made a good recovery.</abstract><cop>England</cop><pub>BMJ Publishing Group LTD</pub><pmid>23576661</pmid><doi>10.1136/bcr-2013-009048</doi><oa>free_for_read</oa></addata></record> |
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| subjects | Abdomen Blood pressure Capillary Leak Syndrome - complications Capillary Leak Syndrome - diagnosis Capillary Leak Syndrome - drug therapy Diagnosis, Differential Edema Female Glucocorticoids - therapeutic use Glucose Hemoglobin Humans Immunoglobulins, Intravenous - therapeutic use Immunologic Factors - therapeutic use Kinases Methylprednisolone - therapeutic use Middle Aged Potassium Rare Disease Shock - etiology United Kingdom White |
| title | Distributive shock due to systemic capillary leak syndrome treated with high-dose immunosuppression |
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