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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Systemic capillary leak syndrome &#40;SCLCS&#41; or Clarkson&#39;s Syndrome is a rare and life-threatening disorder characterized by repeated episodes of severe hypotension&#44; hypoalbuminemia and hemoconcentration&#46; Attacks are characterized by marked shifts of plasma and proteins to the extravascular space&#46; Mortality during episodes is high &#40;by persistence of shock or pulmonary oedema to reverse the syndrome&#44; in the recruitment phase&#41;&#46; In almost every case reported&#44; a monoclonal immunoglobulin has been present in the serum&#44; but its role in the disease has not been clarified&#46;<span class="elsevierStyleSup">1</span></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present a patient affected by this rare entity&#44; with a prolonged survival in which the crises have disappeared temporarily after treatment with melphalan&#8211;prednisone on two occasions&#44; and lately with intravenous immunoglobulins &#40;IVIG&#41;&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">A 48-year-old woman was first admitted to hospital in 1997 with severe hypotension&#44; hemoconcentration &#40;hemoglobin 21<span class="elsevierStyleHsp" style=""></span>g&#47;l&#41; and hypoalbuminemia &#40;23<span class="elsevierStyleHsp" style=""></span>g&#47;l&#41;&#46; The patient was managed in the Intensive Care Unit &#40;ICU&#41; with volume and dopamine&#59; she remained hypotensive for 48<span class="elsevierStyleHsp" style=""></span>h&#44; oligoanuric and with progressive oedema&#46; She finally recovered her blood pressure with progressive polyuria&#44; disappearance of oedema and normalization of hemoglobin and albumin&#46; A serum monoclonal band of Ig Kappa of 3&#46;5<span class="elsevierStyleHsp" style=""></span>g&#47;L was documented&#44; with a normal bone marrow examination&#46; She was found to be hypothyroid&#44; with a thyrotropin of 69<span class="elsevierStyleHsp" style=""></span>ng&#47;dL &#40;normal values&#58; 0&#46;8&#8211;1&#46;8&#41;&#44; thyroxine of 5&#46;3<span class="elsevierStyleHsp" style=""></span>pmol&#47;L &#40;normal values&#58; 10&#46;29&#8211;23&#46;16&#41; and positive antimicrosomal&#44; antiperoxidase and antithyreoglobulin antibodies&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The patient was diagnosed with SCLCS and treated with therbutaline and aminophylline&#46; During follow-up to year 2000 she presented 20 similar episodes&#46; In 2001&#44; given the lack of therapeutic response&#44; and after a normal new bone marrow biopsy&#44; she was treated with melphalan&#47;prednisone monthly for a year&#46; With this treatment&#44; the frequency and intensity of the episodes diminished until their total disappearance&#46; In 2005 she relapsed with 6 new episodes&#46; Serum levels of chromogranin A were found to be high &#40;188<span class="elsevierStyleHsp" style=""></span>ng&#47;mL&#41;&#44; and did not change during the crises&#44; with a NTF-&#945; that remained normal&#46; An abdominal computerized tomography&#44; nuclear magnetic resonance of the adrenal glands and scintigraphy with octeotride In111 and with Iodine123 metaiodobenzylguanidine were all within normal limits&#46; After a repeat bone marrow biopsy that was unrevealing&#44; she restarted treatment with melphalan&#47;prednisone monthly for a year&#46; The patient remained newly asymptomatic for the next 24 months&#46; The monoclonal band was greatly diminished &#40;0&#46;19 G&#47;&#41; and levels of chromogranin A remained normal &#40;60<span class="elsevierStyleHsp" style=""></span>ng&#47;mL&#41;&#46; Finally&#44; in 2008 the patient presented 2 new episodes of severe shock which reversed promptly with IVIG&#46; Because the patient had a refractory SCLS we finally decided to administer intravenous immunoglobulin &#40;2<span class="elsevierStyleHsp" style=""></span>g&#47;kg&#41; every 6 weeks&#46; Since the beginning of this treatment&#44; the patient has had complete resolution of recurrent episodes and has been asymptomatic up to the present&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">SCLS are characterized by recurrent crises of massive extravasation of plasma and its constituents to extravascular space as a result of a sudden reversible increase in capillary permeability&#46; About 120 cases have been described&#44; but recurrent episodes of shock were found in only 40 patients&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Capillary permeability is normal in quiescent periods&#46; Usually no myeloma has been documented&#44; although in four cases myeloma appeared during follow-up&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> No curative treatment is available for SCLS&#44; although different regimens have been tried with variable success&#46; The most frequently used is the combination of terbutaline and aminophylline&#44;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a> which was unsuccessful in our patient&#46; Interestingly&#44; in patients who developed myeloma during their evolution&#44; treatment with chemotherapy is accompanied by a disappearance or decrease of the attacks&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Based on these results some authors have proposed the use of chemotherapy in patients with repeated attacks&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> However to the best of our knowledge&#44; the patient reported here is the first in whom this regimen has proved effective&#44; although only temporarily&#46; More recently&#44; Lambert et al&#46; described a patient with scleroderma and chronic refractory SCLS in whom the monthly administration of IGIV was followed by disappearance of symptoms&#44; and two other patients with SLCS were also controlled with the same regimen during the acute phase of the disease&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> At least one other patient with repeated episodes of SCLCS has remained stable for three years with chronic administration of IGIV&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> More recently Gousseff et al&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> had reported a similar data in other 18 patients recruited in a European Register&#46; In conclusion&#44; our observation suggests for the first time that there is a beneficial effect from therapy with melphalan&#8211;prednisone&#44; which is reversible with the finalization of treatment&#44; and strengthens the hypothesis of the action of intravenous immunoglobulins&#46;</p></span>"
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Correspondence
Successful treatment of systemic capillary leak syndrome with intravenous immunoglobulins
Tratamiento eficaz del síndrome de extravasación capilar sistémica con inmunoglobulinas intravenosas
P. Almagroa,
Autor para correspondencia
19908pam@comb.cat

Corresponding author.
, J.M. Martíb, L. Garcia Pascualc, M. Rodriguez-Carballeiraa
a Internal Medicine, University Hospital Mútua de Terrassa, Spain
b Oncohematology, University Hospital Mútua de Terrassa, Spain
c Endocrinology Services, University Hospital Mútua de Terrassa, Spain

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