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Adult Onset Still's Disease Accompanied by Acute Respiratory Distress Syndrome: a Case Report

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Adult Onset Still's Disease Accompanied by Acute Respiratory Distress Syndrome: a Case Report EXPERIMENTAL AND THERAPEUTIC MEDICINE 12: 1817-1821, 2015 Adult onset Still's disease accompanied by acute respiratory distress syndrome: A case report XIAO-TU XI1, MAO‑JIE WANG2, RUN‑YUE HUANG2 and BANG‑HAN DING1 1Emergency Department; 2Rheumatology Department, The Second Affiliated Hospital of Guangzhou University of Chinese Medicine (Guangdong Provincial Hospital of Chinese Medicine), Guangzhou, Guangdong 510006, P.R. China Received July 17, 2015; Accepted May 20, 2016 DOI: 10.3892/etm.2016.3512 Abstract. Adult onset Still's disease (AOSD) is a systemic vascular permeability and a loss of aerated lung tissue (8). inflammatory disorder characterized by rash, leukocytosis, ARDS occurs in 7.2‑38.9/100,000 individuals (9,10), and has fever and arthralgia/arthritis. The most common pulmo- a mortality rate of ~40% (11). Patient mortality varies based nary manifestations associated with AOSD are pulmonary on numerous factors, including age, etiology of the lung injury infiltrates and pleural effusion. The present study describes and non‑pulmonary organ dysfunction. The most common a 40‑year‑old male with AOSD who developed fever, sore risk factors for ARDS are pneumonia, sepsis, aspiration and throat and shortness of breath. Difficulty breathing promptly trauma (11). Multiple predisposing risk factors, in addition to developed, and the patient was diagnosed with acute respira- some secondary factors such as alcohol abuse and obesity, also tory distress syndrome (ARDS). The patient did not respond increase the risk (4). In addition to patient support provided by to antibiotics, including imipenem, vancomycin, fluconazole, improving oxygen delivery to the tissues and preventing multi- moxifloxacin, penicillin, doxycycline and meropenem, but was organ system failure, early recognition and correction of the sensitive to glucocorticoid treatment, including methylpred- underlying cause is important in ARDS treatment. A thorough nisolone sodium succinate. ARDS accompanied by AOSD has patient assessment searching for the underlying cause should been rarely reported in the literature. In conclusion, in a patient be undertaken; once identified, it should be managed appro- with ARDS who does not respond to antibiotic treatment, the priately. Potential sources of infection should also be sought involvement of AOSD should be considered. to initiate timely and appropriate antibiotic therapy to improve sepsis‑associatted outcomes (4,11). ARDS accompanied by Introduction AOSD is sensitive to high dose corticosteroid therapy. The present study reports the case of a 40‑year old male diagnosed Adult‑onset Still's disease (AOSD) is a multisystemic inflam- with AOSD accompanied by ARDS. matory disorder characterized by a high spiking fever, a transient rash and arthritis/arthralgia (1). Other clinical Case report symptoms include sore throat, myalgia, lymphadenopathies, splenomegaly and neutrophilic leukocytosis (2). The etiology A 40‑year‑old Chinese male was admitted to The Second of AOSD remains unclear, but an autoimmune pathogenesis Affiliated Hospital of Guangzhou University of Chinese has been suggested (3). The most common pulmonary mani- Medicine (Guangzhou, China) with a four‑day history of festations associated with AOSD are pulmonary infiltrates intermittent fever accompanied by with shortness of breath and pleural effusion (4). However, acute respiratory distress for one day. The patient was a driver who did not smoke or syndrome (ARDS) accompanied by AOSD has rarely been drink, and had no recent travel or contact history of infec- reported (5‑7). ARDS is a form of acute diffuse lung injury tious diseases. On June 3, 2014, the patient had a temperature characterized by severe inflammation, increased pulmonary of 39˚C and developed fever, cough, a stuffy, runny nose and sore throat. On June 5, 2014, the patient was admitted to the Emergency Department at The Second Affiliated Hospital of Guangzhou University Chinese Hospital where laboratory analyses was performed. The patient had an Correspondence to: Dr Bang‑Han Ding, Emergency Department, initial white blood cell (WBC) count of 33.25x109 cells/l The Second Affiliated Hospital of Guangzhou University of Chinese 9 Medicine (Guangdong Provincial Hospital of Chinese Medicine), (normal range, 4‑10x10 cells/l) with 92.1% neutrophils 111 Dade Road, Guangzhou, Guangdong 510006, P.R. China (normal range, 50‑70%), and a routine urine test revealed E‑mail: [email protected] positive urine protein (1‑2 g, 2+) and leukocyte esterase (+). From these results, the patient was diagnosed with a urinary Key words: adult onset Still's disease, acute respiratory distress tract infection, and was treated with levofloxacin [100 ml syndrome, spiking fever (0.5 g), q.d.; Daiichi Sankyo, Co., Ltd., Shanghai, China] and antipyretic therapy (ibuprofen suspension; 20 ml, p.o., t.i.d.; Jiangsu Hengrui Medicine Co., Ltd., Jiangsu, China). 1818 XI et al: AOSD ACCOMPANIED BY ARDS: A CASE REPORT However, the body temperature of the patient continued to ventricular enlargement. Furthermore, a small quantity of peri- fluctuate between 38.9 and 39.2˚C, accompanied by mild cardial effusion was demonstrated; iv) splenauxe; v) increased shortness of breath and headache. exudation around both kidneys compared with the results of On June 6, 2014, further examination was performed and the CT scan on June 6, 2014, and a small quantity of effusion results indicated that the respiratory status of the patient had was observed in the bilateral paracolic sulci the abdomen; and worsened (Table I). Computerized axial tomography (CT) vi) pelvic effusion. Furthermore, blood gas analyses (PaO2, scanning of the abdomen showed splenomegaly and bilateral 66.5 mmHg; PaCO2, 32.1 mmHg) indicated that the breathing kidney inflammatory effusions Fig.( 1). As a result, the patient problem was severe. The patient was diagnosed with ARDS, was diagnosed with sepsis, acute respiratory distress syndrome and symptomatic treatments, including morphine and propofol (ARDS), multiple organ dysfunction syndrome (including the treatment, were immediately performed. heart, lungs and liver), infective fever (perirenal infection), fatty Over a number of days, laboratory test results were liver and splenomegaly. The patient was subsequently treated obtained (Table II) and the therapeutic strategy was subse- with tienam (1,000 mg, b.i.d.; EMD Millipore, Billerica, MA, quently modified. Propofol and morphine were administered USA), polyene phosphatidylcholine (456 mg, t.i.d.; Sanofi S.A., for sedation and analgesia, and esmolol was used to reduce Paris, France), plavix (75 mg, q.d.; Sanofi S.A.), aspirin (100 mg, the heart rate. In addition, anti‑infection therapy was q.d.; Bayer AG, Leverkusen, Germany) and lipitor. However, expanded and a number of antibiotics were administered, the results obtained on the following day suggested that the including imipenem, vancomycin (500 mg, q. 8 h; Eli Lilly patient's condition was deteriorating; therefore, the patient was Japan K.K., Kobe, Japan), fluconazole (400 mg, q.d.; Pfizer, immediately admitted to the Intensive Care Unit (ICU). Inc., New York, NY, USA ), moxifloxacin (400 mg, q.d.; Following admission to ICU (June 7, 2014), the vital signs of Bayer AG), penicillin, doxycycline (200 mg, q.d.; Guang- the patient were as follows: Body temperature, 38.6˚C (normal dong Weilun Biological Co., Ltd., Guangdong, China) and range, 36.0‑37.0˚C); pulse rate, 121 beats/min (normal range, meropenem (1,000 mg, q. 8 h; Sumitomo Dainippon Pharma 60‑100 beats/min); respiratory rate, 32 breaths/min (normal Co., Ltd., Osaka, Japan). Other symptomatic treatments, such range, 70‑75 breaths/min); and blood pressure, 131/72 mmHg as protecting the liver and stomach, nourishing the myocar- (normal range, 90‑139/60‑89 mmHg). Physical examination dium, correcting anemia and improving the immune system revealed bilateral coarse breath sounds over the lung fields were administered. On June 14th, 2014, the oxygenation index without dry or wet rale, mild yellow dye of the systemic skin was improved (>200), therefore, the trachea intubation was mucous membrane and sclera, pharyngeal hyperemia, and removed and a non‑invasive ventilator for assisted ventilation 121 beats/min heart rate with the cardiac dull field expanded, was adopted. Hydrocortisone (100 mg i.v.d. b.i.d.; Tianjin a sign of pericardial effusion. The abdomen was soft, while Biochemical Pharmaceutical Co., Ltd., Tianjin, China) was the spleen could be touched below the left rib with ~2 fingers administered for two days as the result of recurrent fever on in width. In addition, the spleen felt tough with smooth edges June 20, 2014. As a result of the fever, all antibiotic treat- with no haphalgesia. Laboratory test results are presented in ment was discontinued on June 24, 2014. The breathing and Table II. The results of chest radiography were as follows: liver function of the patient were improved, although pyrexia Pulmonary congestion, heart enlargement and a small quan- remained. The patient was, therefore, moved to the general tity of pleural effusion on both sides. This led to a diagnosis inpatient area for further treatment. of cardiac insufficiency and infection in the lower right lung. Following transfer to the general inpatient area, a number of The therapeutic strategy comprised of ventilation using a examinations were performed, and the results were as follows: non‑invasive ventilator, anti‑infection therapy using imipenem Epstein‑Barr virus (EBV)‑DNA, 7.50x103 IU/ml;
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