Cardiac arrest in a case of systemic lupus erythematosus and hepatitis-B coinfection: Can Strongyloides stercoralis be the culprit?

Strongyloides stercoralis is one of the most important helminths causing neglected tropical diseases. Its ability to cause autoinfection can lead to chronic infections and hyper infection, especially in autoimmune cases and prove to be highly fatal. We report a case of systemic lupus erythematosus (SLE) having hepatitis-B coinfection, who presented with rash all over the body, recurrent nausea, vomiting, cough, loose stool, and joint pain for 1 month and was later on found to be infected with S. stercoralis. He suffered a massive cardiac arrest during his hospital stay, which was efficiently managed. S. stercoralis infection in cases of SLE and other immunocompromised conditions must be treated at the earliest, to prevent fatal cardiac arrests and other complications.

INTRODUCTION

Strongyloides stercoralis, a soil transmitted helminth, is one of the most important causes of neglected helminthic infections. Although it is endemic worldwide, it is more prevalent in hot and humid climates as well as in resource-poor countries with improper sanitary conditions.[1] It is estimated to have infected around 30–100 million population worldwide.[2] The clinical features range from subclinical in acute and chronic infection to severe and fatal (around 90%) in hyperinfection syndrome, causing disseminated strongyloidiasis.[3] Strongyloides hyperinfection syndrome (SHS) has been reported worldwide among patients who are immunosuppressed, either due to disease or drugs. Systemic lupus erythematosus (SLE) cases may get complicated by Strongyloides stercoralis infection but majority of them show respiratory complications such as respiratory distress or failure.[45678] Here, we report a case of SLE having hepatitis-B coinfection, who was found to be infected with S. stercoralis. Along with some respiratory complications, he suffered a massive cardiac arrest, which was efficiently managed.

CASE REPORT

A 35-year-old male presented to medicine outpatient department with complains of rash all over the body, recurrent nausea, vomiting, cough, loose stool, joint pain and decreased sensation over left little finger for 1 month. There was a history of fever 1 month back. There was no history of breathlessness or decreased urine output. The patient was a nonalcoholic and a nonsmoker. He was not immunized against Hepatitis B in the past.

On reviewing his previous reports, the serological tests were positive for HBsAg (by ELISA) and antinuclear antibody (ANA) and were negative for HIV and HCV whereas the biochemical tests showed increased level of serum glutamic-oxaloacetic transaminase (SGOT-61 IU/L) and serum glutamic pyruvic transaminase (SGPT-75 IU/L) and decreased levels of albumin (2 g/dl) and total protein (3.9 g/dl). The previous ultrasonography (USG) report showed slightly thickened bowel loop, increased mesenteric echogenicity, fatty liver, and marginal splenomegaly. The treatment history revealed that he was prescribed tenofovir and some anti-leprosy drugs, which he took very irregularly.

On general examination, the patient appeared conscious and well oriented. There was absence of pallor, icterus, cyanosis, clubbing and lymphadenopathy, but there was presence of bilateral pedal edema. On systemic examination, there was decreased bilateral air entry in both lungs, the abdomen was found to be distended and there was decreased sensation in the left little finger. A presumptive diagnosis of hepatitis-B and SLE with hypoalbuminemia was made and he was admitted to the medicine ward.

On hematological examination, all parameters were normal except elevated Total leukocyte count (12,570 cells/cmm) and neutrophilia (80%). Repeat liver function tests showed increased level of SGOT (85 IU/L), SGPT (254 IU/L), and alkaline phosphatase (502 IU/L). Total protein (3.17 g/dl), albumin (1.68 g/dl), and globulin (1.49 g/dl) were further decreased. Serum lipase (10 U/L) and amylase (51 U/L) levels were normal. High-sensitivity C-reactive protein (HsCRP) level was highly raised (131.97 mg/L). Renal function tests and thyroid profile were normal. Repeat tests for viral markers were negative for HIV and HCV whereas it was positive for HBsAg by ELISA technique. Repeat test for ANA was positive. Further tests to rule out any other autoimmune disease (c-ANCA, p-ANCA, anti-histone, anti-dsDNA, U1 SM/RNP, SS-A, RO-52, anti-centromere antibody, SCL-70, PM-SCL, JO-1, nucleosome antibody, AMA-M2 antibody, Ribosomal P antibody) were all negative. The patient was diagnosed with SLE and hepatitis-B induced acute on chronic liver disease, for which medications (piperacillin-tazobactam, metronidazole, pantoprazole, drotaverine, and hydroxychloroquine) were started. Three units of albumin were also administered. The patient was also administered intravenous hydrocortisone therapy and nebulized by salbutamol sulfate and ipratropium bromide plus budesonide. As the problem of loose stools continued, a stool sample of the patient was sent for routine microscopy, which revealed the presence of multiple larvae of S. stercoralis after wet mount examination in normal saline [Figure 1a] and in Lugols iodine [Figure 1b]. Modified acid-fast staining of the stool smear also showed pink-colored larvae [Figure 2]. A second fresh stool sample was asked for, which also contained the same larvae. Tablet ivermectin was immediately administered to the patient. On the 2nd day of admission, the patient developed acute abdominal distension. USG of whole abdomen was performed which showed reduction in size of liver with coarse echotexture (signs of chronic liver disease), moderate ascites, and bilateral pleural effusion. On the 3rd day, the patient had some episodes of vomiting followed by a sudden cardiac arrest. Cardiopulmonary resuscitation (CPR) was done immediately, and suitable measures were undertaken to revive the patient, till the electrocardiogram showed sinus rhythm. On patient's request, he was referred to another hospital for further management, after which he could not be contacted.

Figure 1

Wet mount examination of stool sample showing rhabditiform larvae of Strongyloides stercoralis (short mouth, double-bulb esophagus, and genital primordium) in normal saline (a) and in Lugols iodine (b)

Figure 2

Modified Ziehl–Neelsen staining of stool sample showing pink-colored larvae of Strongyloides stercoralis

At the time of discharge, the patient was intubated with a blood pressure of 128/60 mm Hg, pulse rate 130/min, respiratory rate 30/min, and body temperature 99°F. There was absence of pallor, icterus, cyanosis, clubbing, and lymphadenopathy, but the presence of bilateral pedal edema and shifting dullness per abdomen. Bilateral air entry was reduced in infrascapular region. S1 and S2 were normally heard in the respective areas.

DISCUSSION

S. stercoralis is an intestinal nematode which has the ability to cause autoinfection, thus causing hyper infection and multiple systemic complications.[91011] This is more profound in immunocompromised conditions; however, there are reports which even establishes its fatality in immunocompetent states.[12] The patient in our case was negative for HIV and other immunocompromised conditions; however, he was a diagnosed case of SLE (ANA was positive) having hepatitis B co infection. His liver function tests were grossly deranged and on USG, features of chronic liver disease were evident.

SHS is more common in immunocompromised conditions, where the organism proliferates unchecked. This syndrome can cause exacerbation of the patient's symptoms related to an increased parasite load in the intestine and lungs.[6] In our case, the patient had SLE and he was administered steroid therapy. In addition, he was nebulized with a bronchodilator along with a steroid. Steroid therapy is known to cause immunosuppression, facilitating hyperinfection. There were several clinical features in this patient indicating hyper infection, such as abdominal pain, nausea, vomiting, diarrhea, peripheral edema and ascites secondary to hypoalbuminemia from protein losing enteropathy, absent peripheral eosinophilia, maculopapular or urticarial rash and pulmonary involvement.[3]

In this case hsCRP, which is an indicator of sepsis, was highly raised but blood culture could not be done. It has been observed that in cases of S. stercoralis infection, gut bacteria gets translocated to bloodstream, especially Streptococcus bovis and Escherichia coli, which may lead to sepsis.[1314]

Larvae of S. stercoralis has been found in different samples such as sputum, bronchial wash, bronchoalveolar lavage, duodeno-jejunal fluid, bile fluid, cerebrospinal fluid and skin tissues[3131516] but in this patient, multiple larvae were found in the wet mount of stool sample [Figure 1a and b]. Modified Ziehl–Neelsen staining done on stool smear only showed pink colored worm like structures [Figure 2]; however, the internal structures were more evident on the wet mount examination, which were in favor of rhabditiform larvae of S. stercoralis (short mouth, double-bulb esophagus, and genital primordium).[9] The patient in this case also had some respiratory complications such as respiratory distress and pleural effusion, but no pulmonary sample could be tested for the simultaneous presence of strongyloides larvae.

In cases where stool examination for the larvae is negative; but there is a strong suspicion of infection by this nematode, serological tests can be done in combination with histopathology of biopsy specimens obtained after additional endoscopy.[17] Duodenal biopsy may reveal parasites in the gastric crypts, in the duodenal glands, or eosinophilic infiltration in the lamina propria. Specialized stool exams which can be done include Baermann concentration, Harada-Mori filter paper culture, quantitative acetate concentration technique, and nutrient agar plate cultures. Polymerase chain reaction can also be done.[318]

The patient in our case underwent sudden cardiac arrest, after which he was revived with a successful CPR. Cardiac arrhythmias have been complained of in some chronic strongyloidiasis cases;[3] however, there are very few reports in literature, which shows that the patient had cardiac arrest.[1519] One of such cases had autoimmune hepatitis and purpura fulminans, and along with respiratory distress, she suffered a fatal cardiac arrest, to which she later succumbed.[15] Prolonged diarrhea in cases of untreated S. stercoralis infection might cause severe electrolyte imbalance and cardiac arrest might be one of its consequences. But to know any other possible cause of cardiac arrest in such patients, few more studies need to be done.

CONCLUSION

Patients having SLE and hepatitis-B may get infected with S. stercoralis, which can give rise to fatal complications. The patient of hyperinfection syndrome may suffer from a fatal cardiac arrest, which must be efficiently managed. All the possible samples must be collected and tested for the possibility of the presence of S. stercoralis. This nematode, which is known to complicate the cases in immunocompromised individuals, can also have fatal consequences in immunocompetent cases. Hence, the clinicians must maintain a very high degree of suspicion while treating such cases with related signs and symptoms, so that antiprotozoal treatment can be initiated at its earliest.

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.

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Conflicts of interest

There are no conflicts of interest.