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Journal of Obstrectic Anaesthesia and Critical Care
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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 12  |  Issue : 1  |  Page : 70-73

A rare case of fetal gas gangrene following premature rupture of membranes in the second trimester diagnosed with the aid of computed tomography


1 Department of Critical Care Medicine, Manipal Hospital, Bengaluru, Karnataka, India
2 Department of Obstetrics and Gynecology, Manipal Hospital, Bengaluru, Karnataka, India

Date of Submission10-Sep-2021
Date of Acceptance10-Dec-2021
Date of Web Publication14-Mar-2022

Correspondence Address:
Dr. K Sarath
Department of Critical Care Medicine, Manipal Hospital, Bengaluru - 560 017, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JOACC.JOACC_85_21

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  Abstract 


Introduction: To report a rare case of foetal gas gangrene following premature rupture of membranes in the second trimester diagnosed with the aid of computed tomography. Case Report: A 33-year-old lady, with G4A2L1, booked and vaccinated, developed premature rupture of membranes at 25 weeks of gestation. She developed severe abdominal pain with high-grade fever on the fourth night after expectant management with intravenous antibiotics and close monitoring. As there was a new-onset shock, she was referred to the higher centre from the obstetric care unit. The ultrasound of the abdomen and pelvis in the emergency room ruled out other causes of shock but confirmed intrauterine death. After planning for vaginal delivery, she had further deterioration requiring intubation and vasopressors. Contrast-enhanced computed tomography (CECT) of the abdomen and pelvis was performed which showed foetal gas gangrene and bilateral acute cortical necrosis. Emergency hysterotomy, performed under high-risk consent delivered macerated foetus. She had atonic uterus and required obstetric hysterectomy under general anaesthesia (GA). Her post-partum course was complicated by disseminated intravascular coagulopathy (DIC), acute respiratory distress syndrome (ARDS) and multiorgan dysfunction syndrome requiring multiple transfusions, prone ventilation and multiorgan support. However, she could not be salvaged and died on the 2nd day of surgery. Conclusion: We describe a rare fatal case of foetal gas gangrene in the second trimester following premature rupture of membranes which was diagnosed by a computed tomography (CT) scan of the abdomen and pelvis. The CT scan reliably identifies emphysematous changes in the amniotic cavity and foetal parts which helps in decision-making from the induction of labour to early surgical approach to prevent peritonitis and multiorgan failure. We recommend early CT scan in pregnancies complicated by intrauterine infections with shock which can change the line of management.

Keywords: Foetal gas gangrene, multiorgan dysfunction syndrome, PPROM


How to cite this article:
Gupta K V, Ajith Kumar A K, Bhattacharya M, Murthy PR, Sarath K. A rare case of fetal gas gangrene following premature rupture of membranes in the second trimester diagnosed with the aid of computed tomography. J Obstet Anaesth Crit Care 2022;12:70-3

How to cite this URL:
Gupta K V, Ajith Kumar A K, Bhattacharya M, Murthy PR, Sarath K. A rare case of fetal gas gangrene following premature rupture of membranes in the second trimester diagnosed with the aid of computed tomography. J Obstet Anaesth Crit Care [serial online] 2022 [cited 2022 May 19];12:70-3. Available from: https://www.joacc.com/text.asp?2022/12/1/70/339552




  Introduction Top


Infection is the third most common cause of maternal mortality only preceded by obstetric haemorrhage and hypertensive disorders, respectively.[1] However, foetal gas gangrene causing maternal mortality is extremely rare in modern obstetric practice owing to the advanced diagnostic modalities, prophylaxis and treatment of intra-amniotic infections. To our knowledge, only two case reports are available in the literature on gas gangrene of the foetus complicating pregnancy following premature rupture of the membranes at term, of which only one case has been reported in the twenty-first century.[2] Because of the rarity of the condition, consensus regarding appropriate treatment has not been established. In this context, we describe a fatal case of a foetal gas gangrene following premature rupture of the membranes in a multiparous woman in her second trimester of pregnancy which was diagnosed with the help of computed tomography (CT).


  Case Report Top


We are reporting a case of a 33-year-old multiparous woman with G4A2L1 at 25 weeks of gestation. She was fully booked and vaccinated with a few online consultations due to the Coronavirus disease 2019 (COVID-19) pandemic. She was initially admitted to an obstetric care unit with complaints of leaking per vaginum and was diagnosed as preterm premature rupture of membranes (PPROM). With this history, she travelled from a remote peripheral centre to an obstetric unit for continuity of care. She was managed conservatively with intravenous antibiotics, continuous foetal monitoring and regular monitoring of the total count, C-reactive protein with the goal of expectant management. Her condition remained uneventful until the fourth night of hospital stay when she developed a sudden severe abdominal pain which was followed by high-grade fever and shock for which she was referred to our tertiary care centre for further care.

On arrival at our emergency room, she had hypotension; tachycardia and tachypnoea. The blood pressure improved initially after fluid resuscitation; however, she later required the initiation of noradrenaline (0.1 mcg/kg/min). She was started on empirical broad-spectrum antibiotics (meropenem and metronidazole) after drawing the blood and urine cultures. The abdominal examination showed distension with no obvious crepitus. Emergency obstetric and general ultrasound of the abdomen performed at the bedside showed intrauterine foetal demise and no obvious signs of placenta previa or abruption and no obvious intra-abdominal collections. She was intubated in the emergency room in view of the progressive respiratory distress with worsening metabolic acidosis.

She was initially planned for induction of labour and was shifted to the critical care unit. As she developed further hemodynamic instability, she underwent an urgent contrast-enhanced computed tomography (CECT) of the abdomen and pelvis which showed a gravid uterus with multiple air pockets in all foetal parts and amniotic cavity suggestive of foetal gas gangrene [Figure 1], [Figure 2], [Figure 3]. These findings were consistent with foetal gas gangrene. The initial blood reports showed a total count of 5,830 cells/mm3 with left shift and thrombocytopenia 37,000 cells/mm3. The patient also had a deranged coagulation profile with hypofibrinogenemia. Acute kidney injury and high anion gap metabolic acidosis were also noted. The peripheral smear examination showed thrombocytopenia and fragmented red blood cells which suggested haemolysis with the overall picture consistent with sepsis and disseminated intravascular coagulation.
Figure 1: CT scan showing air in the fetal scrotum and gestational sac

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Figure 2: Air in Fetal brain

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Figure 3: CT scan showing air in abdomen and liver of the fetus

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Emergency hysterotomy was performed for source control, and intraoperatively, there was no major bleeding but four Fresh frozen plasma (FFPs) were transfused to correct coagulopathy. She also required minimal vasopressors (noradrenaline 0.1 microgram/kg/min) for optimal haemodynamics. Amniotomy revealed brownish foul-smelling blood and a dead male bloated and boggy foetus was delivered. The placenta and membranes delivered in toto. She was noted to have atonic uterus after the delivery of the foetus, uterotonic agents were given and B-Lynch suture was applied. The decision was taken intraoperatively to proceed with obstetric hysterectomy in view of the continued uterine atonicity. She was transferred back to the multidisciplinary Intensive Care Unit (ICU) for postoperative care.

In ICU, she was continued on noradrenaline (0.1–1.0 mcg/kg/min), vasopressin (0.01–0.04 Units/min) and adrenaline (0.1–1.o mcg/kg/min). Mechanical ventilation (volume control ventilation) and blood product transfusion (eight FFP, 10 units cryoprecipitate, one single donor platelet and two unit packed red blood cells) was carried out to correct coagulopathy. She was initiated on continuous veno-venous haemodiafiltration in view of anuria and severe metabolic acidosis. The oxygenation worsened, requiring prone ventilation. Despite aggressive organ support, the patient continued to deteriorate and passed away within 24 h of admission.


  Discussion Top


Gas gangrene is a rapidly spreading and developing infection of the muscle caused by toxin producing Clostridium species and usually accompanied by bacteraemia, hypotension and multiorgan dysfunction. Most cases of gas gangrene are caused by Clostridium perfringens and other Clostridial species such as Clostridium novyi, Clostridium histolyticum, Clostridium sordellii and Clostridium fallax.[3] Clinical and histological manifestations of gas gangrene were attributed to the production of bacterial exotoxins—mainly alpha toxin and theta toxin.

Alpha toxin is a phospholipase C which cleaves phosphatidylcholine in the cell membrane and causes microvascular thrombosis. Theta toxin (also known as perfringolysin O) is a member of the cholesterol-dependent cytolysin family which lyses red blood cells.[4]

The Clostridial species are usually commensals in the genital tract and clinical infection is rare without the presence of devitalised tissue. Lawson and colleagues[5] described the infection of the uterus in four stages. (1) Infection confined to dead foetal tissue and gas formation, (2) Infection involving endometrium and causing no systemic symptoms, (3) Damaged myometrium and possible peritoneal spread, (4) Infection established in the maternal tissue causing multiorgan failure. In our case report, she had premature rupture of the membranes in the second trimester with initially normal course with expectant management. This was complicated later by intrauterine foetal demise presented in sepsis. The inability to pick up gas gangrene on ultrasound with initial plan of induction of labour further delayed foetal delivery which might have aggravated maternal septicaemia and further worsening. In a case report by Abe et al.,[2] an unbooked patient presented at term with PROM and prolonged obstetric labour led to foetal gas gangrene and maternal mortality.

The diagnosis of gas gangrene can be delayed as the clinical condition is infrequent and it should be suspected in all the cases of foetal demise with hemodynamic instability and multiorgan failure. Physical examination findings such as crepitus over the abdominal wall and escape of gas through the cervix may be seen.[6] Patients may develop rapid-onset shock and multiorgan failure with disseminated intravascular coagulopathy (DIC). Differential diagnosis of this condition is uterine rupture, obstetric haemorrhage such as abruptio placenta or placenta previa. The X-ray of the abdomen may show gas surrounding the foetus. Intra-myometrial gas may be seen as a 'frame-like aspect' around the foetus.[6] The CT scan of the abdomen and pelvis can be used to evaluate the condition of the maternal uterus and determine the gas in the amniotic fluid cavity, placenta and foetus. To our knowledge, only one case report was published earlier demonstrating the CT findings in foetal gas gangrene.[2]

Gram stains of the cervical and vaginal smears may show large gram-positive rods and blood cultures can show the Clostridium species. In our case, the blood culture showed extended spectrum beta lactamase (ESBL) E. coli.

The treatment involves evacuation of the uterine contents at the earliest. No consensus is available to date regarding the appropriate delivery method. Habeebullah et al.[6] reported a case of foetal gas gangrene successfully treated with laparotomy without the need for hysterectomy as the infection was confined to the foetus and the margins of the uterus showed no signs of necrosis. Similarly, Jones et al.[7] reported a case of foetal gas gangrene managed by LSCS with adequate removal of the necrotic tissue. One more case with foetal gas gangrene was initially managed conservatively with the induction of labour and the patient deteriorated later and progressed to multiorgan failure. She subsequently underwent LSCS and evacuation of the foetal contents.[6] Our patient underwent LSCS and delivery of bloated foetus and post-delivery atonic uterus persisted despite the application of the B-Lynch suture and uterotonic agents. The decision of hysterectomy was taken intraoperatively. To conclude, preservation of the mother's life is the priority in cases of foetal gas gangrene and a delay in the diagnosis and foetal delivery may lead to maternal peritonitis and multiorgan failure.

Antibiotic therapy: Pending definitive etiological diagnosis, broad-spectrum empiric antibiotic treatment is warranted to cover group A Streptococcus, Clostridium species and mixed aerobes and anaerobes. Piperacillin-tazobactam plus clindamycin is the preferred treatment option. Carbapenems are an acceptable alternative to Piperacillin-tazobactam.[8] The optimal duration of the antibiotic therapy has not been defined and the duration must be tailored to the individual patients based on the final culture reports.[8] Gas gangrene antitoxin is controversial and not studied in foetal gas gangrene.[9] The use of hyperbaric oxygen therapy is controversial largely due to the lack of randomised controlled trials in humans and to divergent results from the studies in animals.[10]


  Conclusion Top


In conclusion, we describe a rare fatal case of foetal gas gangrene in the second trimester following premature rupture of the membranes which was diagnosed by the CT scan of the abdomen and pelvis. A CT scan reliably identifies the emphysematous changes in the amniotic cavity and foetal parts which helps in decision-making from induction of labour to early surgical approach to prevent peritonitis and multiorgan failure. We recommend early CT scan in pregnancies complicated by intrauterine infections with shock which can change the line of management.

Acknowledgments

We Thank Dr. Sudarshan Rawat (Department of Radiodiagnosis, Manipal hospital) for his assistance in procuring images for case report.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Say L, Chou D, Gemmill A, Tunçalp Ö, Moller AB, Daniels J, et al. Global causes of maternal death: A WHO systematic analysis. Lancet Global Health 2014;2:e323-33.  Back to cited text no. 1
    
2.
Abe K, Hamada H, Fujiki Y, Iiba M, Tenjimbayashi Y, Yoshikawa H. Radiological diagnosis of gas gangrene in a fetus at term. Taiwanese J Obstet Gynecol 2016;55:582-4.  Back to cited text no. 2
    
3.
Darke SG, King AM, Slack WK. Gas gangrene and related infection: Classification, clinical features and aetiology, management and mortality. A report of 88 cases. Br J Surg 1977;64:104-12.  Back to cited text no. 3
    
4.
Stevens DL, Bryant AE, Proft T. Clostridial gas gangrene: Clinical correlations, microbial virulence factors, and molecular mechanisms of pathogenesis. In: Norfolk PT, editor. Microbial Toxins: Molecular and Cellular Biology. UK: Horizon Bioscience; 2005. p. 313-35.  Back to cited text no. 4
    
5.
Lawson JB, Stewart DB. Obstetrics and Gynaecology in the Tropics and Developing Countries. London Arnold; 1967.  Back to cited text no. 5
    
6.
Habeebullah S, Bupathy A, Rajaram P. Two cases of gas gangrene complicating uterine rupture and fetal death at term. Asia-Oceania J Obstet Gynaecol 1994;20:191-4.  Back to cited text no. 6
    
7.
Jones DH. Gas gangrene complicating pregnancy. J Obstet Gynaecol Br Commonw 1965;72:785-90.  Back to cited text no. 7
    
8.
Stevens DL, Bisno AL, Chambers HF, Dellinger EP, Goldstein EJ, Gorbach SL, et al. Practice guidelines for the diagnosis and management of skin and soft tissue infections: 2014 update by the Infectious Diseases Society of America. Clin Infect Dis 2014;59:e10-52.  Back to cited text no. 8
    
9.
Stevens DL, Titball RW, Jepson M, Bayer CR, Hayes-Schroer SM, Bryant AE. Immunization with the C-domain of α-toxin prevents lethal infection, localizes tissue injury, and promotes host response to challenge with clostridium perfringens. J Infect Dis 2004;190:767-73.  Back to cited text no. 9
    
10.
Kaide CG, Khandelwal S. Hyperbaric oxygen: Applications in infectious disease. Emerg Med Clin North Am 2008;26:571-95.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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