Fatal systemic emphysematous infection caused by Klebsiella pneumoniae:A case report

INTRODUCTION

()is a Gram-negative pathogenic bacterium belonging to thefamily,usually causes various infections including pneumonia,urinary tract infections,hepatobiliary infections and intra-abdominal infections[1].strains are divided into the classical group and hypervirulent group.Classicalis an opportunistic pathogen and primarily infects critically ill and immunocompromised patients,and causes health-care associated infections;the hypervirulent pathotype usually infects healthy individuals and causes communityacquired infections[2-3].In recent years,several studies have reported a distinctive clinical syndrome,invasiveliver abscess syndrome(IKLAS),which typically occurs in patients with diabetes mellitus(DM)and is characterized by a liver abscess,bacteremia,and hematogenous extrahepatic infection at sites such as the eye,brain,or lung.We report a rare case of IKLAS presenting with multiple organ dysfunction syndrome(MODS),septic shock,bacteremia,numerous emphysematous liver abscesses,pneumoperitoneum,emphysematous cystitis,prostate and left seminal vesicle abscesses in a diabetic patient whose infection progressed rapidly and died within a short period.

CASE PRESENTATION

Chief complaints

A 66-year-old man was admitted to our hospital with a 14-d history of worsening fatigue,anorexia,nausea and vomiting,and a 3-d history of confusion and jaundice.

History of present illness

Fourteen days prior to hospital admission,he suffered from fatigue,anorexia,nausea and vomiting.The patient did not visit his doctor,the symptoms of nausea and vomiting gradually improved but fatigue and anorexia persisted.His family members found that he had been showing signs of confusion and jaundice 3 d earlier.He vomited 50 mL of coffee-colored gastric contents one day earlier.He had no fever,abdominal pain and lower urinary tract symptoms.

History of past illness

His past medical history included acute gastric ulcer perforation which was repaired 40 years earlier,hypertension treated with amlodipine and hydrochlorothiazide,type 2 DM without regular control treated with metformin,acarbose and insulin for 10 years,chronic prostatitis and chronic diarrhea for the past 10 years,and a cerebral infarction 8 years and 3 mo previously.He had acute calculous cholecystitis with hypotension 2 mo ago and received empirical antibiotics with ceftazidime 2.0 g and ornidazole 0.5 g intravenous route every 12 h for 10 days,blood and bile cultures and cholecystectomy were not performed.

Personal and family history

No relevant family history,travelling history or animal contact was reported.

Physical examination

Emphysematous liver abscess is a rare but life-threatening infection which is characterized by hepatic parenchymal emphysematous change.In rare circumstances,emphysematous liver abscesses are prone to spontaneous rupture resulting in secondary peritonitis and intra-abdominal sepsis which can further increase mortality rate[8].Our patient presented with pneumoperitoneum secondary to spontaneous rupture of emphysematous liver abscesses,with no evidence of hollow viscus perforation.Multiple emphysematous liver abscesses that spread throughout the liver resulted in severely destructive hepatic tissue,and extensive gas formation in the abscess was vulnerable to spontaneous rupture as gas increases the tension within the abscess cavity.Gas in the abscess is believed to be due to the fermentation of glucose into carbon dioxide byunder anaerobic conditions[9-10].In patients with numerous abscesses,it is difficult to locate and drain all the lesionspercutaneous,laparoscopic or surgical intervention,thus the mortality rate is reported to be extremely high at 27%-30%[9].Control of the infectious source is very important,failure to timely surgery or percutaneous drainage is our limitation and the lessons should be learned.

Markedly raised inflammatory parameters were found including the following: white blood cell count 18.8 × 10/L(93% neutrophils and 1% lymphocytes),C-reactive protein 315 mg/L,interleukin 6 >5000 pg/mL,and procalcitonin(PCT)70 ng/mL.Serum biochemical tests showed total bilirubin 210.9 μmol/L,direct bilirubin 165.7 μmol/L,alanine aminotransferase(ALT)940 U/L,aspartate aminotransferase(AST)3870 U/L,alkaline phosphatase 1429 U/L,lactate dehydrogenase(LDH)7852 U/L,blood urea nitrogen 48.1 mmol/L and creatinine 339.3 μmol/L.The level of blood glucose and glycosylated hemoglobin A1c(HbA1c)was 10.25 mmol/L and 8.4% respectively.A coagulation panel demonstrated a prothrombin time(PT)of 21.3 s,prothrombin time activity(PTA)39%,international normalized ratio(INR)1.9,activated partial thromboplastin time(APTT)38.7 s and D-dimer 7.25 μg/mL.Arterial blood gas analysis revealed a pH of 7.273,undetectable PCO(A fall in PCOwas beyond the range of the Point-of-Care Testing device),PO90.2 mmHg and lactate 20 mmol/L.In addition,routine urine analysis revealed numerous red blood cells(50-60/high power field),white blood cells(35-40/high power field),bilirubin and albuminuria.The patient was not anemic(blood Hb 133 g/L)and hematocrit was 39.7%.Serology showed that human immunodeficiency virus,syphilis,hepatitis B and C were all negative.

Laboratory examinations

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Imaging examinations

An abdominal computed tomography(CT)scan displayed numerous emphysematous hepatic abscesses,rupture of some liver abscesses and gas formation in the right subphrenic area(Figure 1).Pelvic CT showed intramural gas formation in the bladder and an enlarged prostate and left seminal vesicle with abnormal air accumulation(Figure 2).Chest CT revealed pulmonary infiltrates in the right lower lobes and small right pleural effusions.

FINAL DIAGNOSIS

is the common pathogen associated with emphysematous infections.Among the members of thecomplex which consists of seven-related species,is frequently misidentified asby routine clinical microbiology diagnostics in most modern laboratories[15].More recently,is recognized as a cause of emphysematous infections[16]with a higher mortality rate when compared to[17].Thus,clinicians should be aware of the potential ofinvolvement as emphysematous infections and identifyamonginfections based on mass spectrometry and genome sequencing[18-19].

TREATMENT

Empiric antimicrobial treatment with meropenem was administered along with fluid resuscitation and vasoactive support with noradrenaline.Continuous veno-venous hemofiltration was then initiated for acute kidney injury and persistent inflammatory state after adequate fluid resuscitation.A consultant hepatobiliary surgeon suggested an emergency surgical exploration but this was refused by his family.We attempted percutaneous liver abscess drainage guided by bedside ultrasound,but did not succeed due to liver abscess cavities totally occupied by air and pneumoperitoneum.His condition rapidly deteriorated,he developed emerging thrombocytopenia,decreased Hb,a progressive increase in serum enzyme levels in addition to severe metabolic acidosis,persistent renal failure and liver dysfunction,and subsequently developed respiratory failure,disseminated intravascular coagulation and coma.At that time,his laboratory examinations showed lactate 30 mmol/L,pH 7.193,HCO11.6 mmol/L,base excess −18.4 mmol/L,SO89.6%,PO/FiO159,platelets 28×10/L,Hb 86 g/L,INR 3.37,PT 39 s,PTA 21%,APTT 57.7 s,D-dimer 7.25 μg/mL,ALT 1501 U/L,AST 6012 U/L,CK 1700 U/L and LDH 12356 U/L.The patient was immediately intubated and mechanical ventilation was initiated.Despite these aggressive treatments,the patient’s condition was critical and exacerbated,with persistent MODS and hemodynamic instability despite large doses of noradrenaline(2.5 μg/kg/min).

Immigrants from Ireland, they met and married in America. Grandma was friendly, outgoing and unselfish; Grandpa was reserved, a man devoted4 to his family. But he wasn’t big on giving gifts. While he wouldn’t think twice about giving my grandma the shirt off his back, he subscribed5 to the belief that if you treated your wife well throughout the year, presents weren’t necessary; so he rarely purchased gifts for her.

OUTCOME AND FOLLOW-UP

Twenty-two hours after admission,the patient died.Two days later,cultures from peripheral blood and urine specimens revealedwith a positive string test,but antimicrobial susceptibility testing was not carried out.

When she was led to the stake, she laid the shirts on her arm, and as she stood on the pile and the fire was about to be lighted, she looked around her and saw six swans flying through the air. Then she knew that her release was at hand and her heart danced for joy. The swans fluttered round her, and hovered low so that she could throw the shirts over them. When they had touched them the swan-skins fell off, and her brothers stood before her living, well and beautiful. Only the youngest had a swan s wing instead of his left arm.52 They embraced and kissed each other, and the Queen went to the King, who was standing by in great astonishment, and began to speak to him, saying, Dearest husband, now I can speak and tell you openly that I am innocent and have been falsely accused.

DISCUSSION

The first case series of IKLAS was described in Taiwan in 1986[4]and it subsequently emerged as a global infectious disease although the majority of cases were found in southeast Asia.Our patient had a rare clinical condition with a poor prognosis and had distinctive clinical features such as ruptured emphysematous liver abscesses with concomitant pneumoperitoneum,emphysematous prostate and left seminal vesicle abscesses and emphysematous cystitis.The patient was in a critical condition complicated by MODS(kidney,liver,circulation,respiratory,coagulation)and rapidly deteriorated following admission.The fatal infection was caused by a strain of hypervirulentidentified by a positive string test,which was more virulent than classicaland capable of causing multiple sites of infection due to hematogenous spread[3].

The etiology of IKLAS is unknown.Our patient suffered from chronic diarrhea without abdominal pain and fever which may be noninfectious and functional diarrhea and is not considered the etiologic factor for IKLAS,even though gastrointestinal colonization is a major reservoir forinduced infections[5].He had acute calculous cholecystitis with hypotension 2 mo ago and received empirical antibiotics for 10 d without blood and bile cultures and cholecystectomy.Therefore,we speculated that the liver abscesses were attributable to the cholecystitis with inadequately management that led bacteria to invade the liver parenchymathe gallbladder bed.In addition,several studies have shown that DM is a significant risk factor for IKLAS[6]and poor glycemic control tends to increase the rate of disseminated infection[7].Our patient had DM for 30 years and his HbA1c was 8.4% on admission,the immunosuppression related to DM may predispose patients to the development of IKLAS.

His initial vital signs were as follows: blood pressure 70/50 mmHg,pulse 110 bpm,respiratory rate 30 breaths/min,and body temperature of 36.5 °C.The Acute Physiology and Chronic Health Evaluation II score was 23,and the Sequential Organ Failure Assessment score was 13.He demonstrated confusion,icteric sclera,normal cardiopulmonary auscultation,a soft,non-tender abdomen,and acrocyanosis with scattered marble patches on wet and cold lower limbs.

The multifocal emphysematous infections in our patient consisting of liver abscesses,cystitis,prostate and seminal vesicle abscesses are extremely rare.Emphysematous cystitis is characterized by the presence of gas in and around the bladder wall and can be treated successfully with bladder drainage and antibiotics[11].Emphysematous prostate abscess is not often diagnosed at an early stage due to non-specific symptoms and may be confirmed by CT which shows gas and abscess accumulation in the prostate[12].Surgical drainage of a prostate abscess can be performed by the transrectal,transperineal or transurethral approach[13].It is recommended in critically ill patients,such as our case,that CTguided transperineal drainage of an emphysematous prostate abscess should be performed[14].

Having travelled from land to land in this fashion without finding anything to rivet10 his attention, it occurred to him to take the form of an eagle, and in this shape he flew across many countries and arrived at length in a new and lovely spot, where the air seemed filled with the scent11 of jessamine and orange flowers with which the ground was thickly planted

But life did not depart from him- the thread would not break,but the thread of memory was severed; the thread of his mind hadbeen cut through, and what was still more grievous, a body remained- a living healthy body that wandered about like a troubled spirit.

The clinical diagnosis was IKLAS with septic shock and MODS accompanied by emphysematous prostate and left seminal vesicle abscesses and cystitis.

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CONCLUSION

the National Natural Science Foundation of China,No.81560480;Health Science Research Program of Gansu Province,No.GSWSKY 2016-19;Ph.D.Science Research Foundation of Lanzhou University Second Hospital,No.ynbskyjj 2015-1-09;and Cuiying Scientific and Technological Innovation Program of Lanzhou University Second Hospital,No.CY 2018-MS13.

FOOTNOTES

Zhang JQ and He CC designed the report;Zhang JQ wrote the manuscript;Liu R and Wang ZX collected the patient’s clinical data;Yuan B,Qi YJ,and He XN were the attending doctors and performed clinical treatment;Li YM revised the paper.

He walked about in a coat embroidered15 all over,and in the drawing-rooms of society looked just like one of those rich pearl-embroidered bell-pulls, which are only made for show; and behind them always hangs a good thick cord for use

IKLAS is a rare but severe infection which can be lethal if the diagnosis is delayed and can progress to septic shock and MODS.Spontaneous pneumoperitoneum secondary to ruptured emphysematous liver abscesses can induce intra-abdominal sepsis that further increases the mortality rate.Early diagnosis followed by efficient antibiotic therapy and surgical management are essential for these life-threatening infections.

Over the course of the year, Rose became a campus icon2 and she easily made friends wherever she went. She loved to dress up and she reveled in the attention bestowed3 up her from the other students. She was living it up. At the end of the semester we invited Rose to speak at our football banquet. I ll never forget what she taught us. She was introduced and stepped up to the podium. As she began to deliver her prepared speech, she dropped her three by five cards on the floor. Frustrated4 and a little embarrassed she leaned into the microphone and simply said, I m sorry I m so jittery5. I gave up beer for Lent and this whisky is killing6 me. I ll never get my speech back in order so let me just tell you what I know. As we laughed she cleared her throat and began: We do not stop playing because we are old; we grow old because we stop playing. There are only four secrets to staying young, being happy, and achieving success.

Informed consent has been obtained and is available from the corresponding author.

All authors declare that they have no conflict of interest.

We wrote the manuscript based on the CARE Checklist(2016).

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China

Jun-Qiang Zhang 0000-0001-9287-7552;Chan-Chan He 0000-0001-7671-7137;Bo Yuan 0000-0001-7331-6506;Rui Liu 0000-0003-2430-0588;Yu-Jing Qi 0000-0002-5774-8015;Zi-Xia Wang 0000-0002-9523-5624;Xiao-Na He 0000-0001-9680-6460;Yu-Min Li 0000-0002-9267-1412.

Ma YJ

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Ma YJ