Sjögren's syndrome concurrent with protein‐losing gastroenteropathy with secondary systemic capillary leak syndrome : A case report

Watanabe, Kei; Murakami, Shinichiro; Misago, M; Yoshikawa, Manabu; Tamai, Daisuke; Nakao, Shinichiro; Ueoka, Takato; Ito, Mototoshi; Shinomura, Yasuhisa; Kajiwara, Nobuyuki

doi:10.1002/ccr3.1675

Cited by 4 publications

(2 citation statements)

References 12 publications

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“…Currently, there are many studies on the treatment of CLS[ 17 ]. However, there is still a lack of specific treatment measures, and the treatment of the primary disease is time-consuming[ 18 ]. In the leakage stage of CLS, small molecular proteins in the plasma leak out of the blood vessels, the plasma colloid osmotic pressure decreases, the effective circulating blood volume decreases, and the blood pressure drops progressively.…”

Section: Discussionmentioning

confidence: 99%

Veno-venous-extracorporeal membrane oxygenation treatment for severe capillary leakage syndrome: A case report

Nong¹,

Lv²,

Lu³

2021

WJCC

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BACKGROUND Capillary leak syndrome (CLS) is characterized by the leakage of large amounts of fluid and plasma proteins into the interstitial space, resulting in hypoalbuminemia, hypovolemic shock, elevated blood concentration, systemic progressive edema, and multiple serosal cavity effusion. Clinical syndromes such as cavity effusion pose a grave threat to the life and health of the patient. CASE SUMMARY A 58-year-old female patient was admitted to the hospital after being in a coma for 6 h following accidental ingestion of a pesticide. She was treated with phencyclidine hydrochloride and pralidoxime iodide for detoxification, mechanical ventilation to maintain oxygen supply, continuous renal replacement therapy to maintain the internal environment, and hemoperfusion to promote the excretion of toxins. She also received a transfusion of red blood cells and massive fluid resuscitation. However, her blood pressure was not maintained. The patient was diagnosed with CLS due to pesticide poisoning. Oxygenation was difficult to maintain under full ventilator support; therefore, veno-venous-extracorporeal membrane oxygenation (VV-ECMO) treatment was given 13 h after admission. Her oxygenation level improved, but a large amount of ascites and pleural effusion soon became apparent. We continued drainage with an indwelling drainage tube, and the ECMO flow stabilized. The leakage gradually decreased, and ECMO was discontinued 3 d later. On the 6 th day, the patient recovered from unconsciousness, but on gastroscopic evaluation, severe erosions were found in her entire stomach. With the family’s consent, treatment was stopped, and the patient was discharged from the hospital on the 7 th day. CONCLUSION ECMO, liquid resuscitation and management, and improvement in plasma colloidal osmotic pressure, circulation, and tissue oxygen supply are crucial in treating CLS.

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Section: Discussionmentioning

confidence: 99%

Veno-venous-extracorporeal membrane oxygenation treatment for severe capillary leakage syndrome: A case report

Nong¹,

Lv²,

Lu³

2021

WJCC

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“…We identified a total of 13 articles describing 14 cases of PLGE associated with SS in a PubMed search, as listed in Table 1. [7][8][9][10][11][12][13][14][15][16][17] Most cases were reported from Asian countries. Compared with the previous cases, the present case involved one of the youngest patients and most severe cases with the lowest serum albumin level.…”

Section: Discussionmentioning

confidence: 99%

Protein‐losing gastroenteropathy with severe hypoalbuminemia associated with Sjögren’s syndrome: A case report and review of the literature

Akaishi

Yasaka

Abe

et al. 2019

J of Gen and Family Med

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A 30‐year‐old man with severe hypoalbuminemia (serum albumin: 0.9 g/dL) was admitted with severe bilateral leg edema and unilateral pleural effusion. Serum anti‐SS‐A and SS‐B antibody levels were abnormally elevated, and his symptoms fulfilled the diagnostic criteria for Sjögren's syndrome. Technetium‐99m albumin scintigraphy revealed protein leakage from a large area of the small intestine. Immunohistochemistry revealed perivascular deposition of C1q, C3d, and immunoglobulin G in the duodenal mucosa. The patient was diagnosed with protein‐losing gastroenteropathy associated with Sjögren's syndrome. Within 2 months of treatment with oral prednisolone and mycophenolate mofetil, the clinical symptoms of hypoalbuminemia and Sjögren's syndrome disappeared completely.

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Sjögren syndrome associated with protein-losing enteropathy: case-based review

et al. 2020

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Sjögren's syndrome concurrent with protein‐losing gastroenteropathy with secondary systemic capillary leak syndrome : A case report

Cited by 4 publications

References 12 publications

Veno-venous-extracorporeal membrane oxygenation treatment for severe capillary leakage syndrome: A case report

Veno-venous-extracorporeal membrane oxygenation treatment for severe capillary leakage syndrome: A case report

Protein‐losing gastroenteropathy with severe hypoalbuminemia associated with Sjögren’s syndrome: A case report and review of the literature

Sjögren syndrome associated with protein-losing enteropathy: case-based review

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