Stillbirth as the primary manifestation of disseminated tuberculosis in a young immunocompetent mother with multiple perforations of the ileum

  1. Deepak Rajput 1,
  2. Amit Gupta 1,
  3. Ravi Roshan 1 and
  4. Arvind Kumar 2
  1. 1 General Surgery, All India Institute of Medical Sciences - Rishikesh, Rishikesh, Uttarakhand, India
  2. 2 Pathology, All India Institute of Medical Sciences - Rishikesh, Rishikesh, Uttarakhand, India
  1. Correspondence to Dr Deepak Rajput; deepakrajputsjh@gmail.com

Publication history

Accepted:28 Jan 2021
First published:09 Feb 2021
Online issue publication:09 Feb 2021

Case reports

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Abstract

Tuberculosis (TB), a significant cause of morbidity and mortality worldwide, is particularly relevant in low/middle-income countries like India, where the disease is endemic. The female reproductive system is very vulnerable to this infection with, the clinical presentation being utterly silent in most patients. Symptoms of TB in pregnancy may initially be attributed to the gravidity itself besides temporary concealment of associated weight loss by the normally occurring weight gain during the pregnancy. Untreated TB may cause pregnancy loss by either placental damage or direct harm to both the mother and child. We report a case of latent disseminated TB in a young immunocompetent female that was revealed in the postpartum state (after full-term stillbirth delivery at home) as 20 ileal perforations secondary to intestinal TB. Due to ongoing sepsis and delayed presentation to the hospital, the patient could not be salvaged despite the best possible efforts.

Background

Tuberculosis (TB) is the second-leading cause of death globally among all communicable diseases. The prevalence of TB, especially extrapulmonary TB (EPTB), is increasing worldwide. TB, directly and indirectly, affects female reproductive health and can present in different ways resembling other diverse disorders to tax the best diagnostician’s ingenuity. Pregnancy usually does not influence the course of pulmonary or EPTB but may aggravate the condition. Although pulmonary TB in pregnant women poses risk for their infants, EPTB outcomes are not well known during the pregnancy. Congenital TB though rare, resulting in either due to haematogenous spread or by ingestion and aspiration of infected amniotic fluid, is associated with high perinatal mortality.

Case presentation

A 21-year-old female postpartum state (first pregnancy), without comorbidity and any significant medical history, presented to the emergency department with acute abdomen and respiratory distress of 5 days duration, developing after full-term stillbirth delivery at home. There was no history of fever, cough, weight loss or altered bowel habits. Menstrual history before the pregnancy was unremarkable.

Pallor, dry tongue and bilateral pedal oedema were the positive findings on general physical examination. Her recorded vitals at presentation were: Pulse 100 beats/min; Blood pressure 100/60 mm Hg; Respiratory rate 24 breaths/min; Oxygen saturation (SpO2) 93% on room air. Abdominal examination revealed fullness over the lower abdomen associated with tenderness and auscultable bowel sounds. No guarding or rebound tenderness could be elicited. The patient was admitted and intravenous fluids, analgesics, empirical antibiotics and moist oxygen were initiated.

Investigations

Because of the ongoing COVID-19 pandemic and the patient having breathing difficulty, nasal and oropharyngeal swabs were taken which reported negative for coronavirus on real-time reverse transcription-polymerase chain reaction (RT-PCR) testing. Routine laboratory workup done on the morning of the day of admission revealed haemoglobin: 0.8 g/L, leucocytes: 16×109/L, platelet count: 90×109/L, serum albumin: 25 g/L, international normalised ratio: 1.2 and serum creatinine: 123 µmol/L. An abdominal radiograph showed dilated small bowel loops but no evidence of free air under the right dome of the diaphragm. Both lung fields showed basal opacities on the radiograph. Ultrasound abdomen showed an extensive pelvic collection. After initial fluid bolus resuscitation and lowering of creatinine value to within normal range, contrast-enhanced computed tomography (CECT) of the torso was planned. CT scan of the chest done on the night of admission showed few solid centrilobular nodules giving tree-in-bud pattern with extensive consolidation involving bilateral lung parenchyma suggestive of active infection likely tubercular (figure 1). Abdominal tomography demonstrated gross hydropneumoperitoneum and bulky uterus postpartum state (figure 2).

Figure 1

CECT chest showing tree-in-bud pattern’ and patchy consolidation areas involving bilateral lung parenchyma.

Figure 2

CECT abdomen showing gross hydropneumoperitoneum (yellow arrow) and bulky uterus postpartum status (white arrow).

Differential diagnosis

The patient had no routine prenatal laboratory tests. For evaluation of the antecedent stillbirth, hepatitis panel, human immunodeficiency virus (HIV) and venereal disease research laboratory (VDRL) tests were ordered that turned out to be negative. Various differentials thought of included puerperal sepsis, appendicular perforation, abdominal TB and complicated enteric fever. Widal test was also negative. As the patient gave no history of fever, acute illnesses were ruled out. CT abdomen findings pointing towards hollow viscous perforation augmented by chest sections suggesting a tubercular infection strongly favoured the possible diagnosis of disseminated TB.

Treatment

An emergency exploratory laparotomy under high-risk consent was performed during the early morning hours of the subsequent day following admission. It revealed around one litre of fecopurulent contamination with multiple perforations (figure 3), a total of 20 in number, spanning almost the whole of the ileum for which resection of the involved segment was done (figure 4), and two viable ends of small bowel exteriorised. Intraoperatively, the patient landed into refractory hypotension. The anaesthetist started dual ionotropic support: inj. norepinephrine 8 mg @ 12 mL/hour and inj. vasopressin @ two units/hour and one unit of packed red blood cells was transfused. Second-line injectable antitubercular drugs (levofloxacin 500 mg/100 mL intravenous 12 hourly and amikacin 750 mg intravenous 24 hourly) were started postoperatively and the patient shifted to the intensive care unit on ventilatory support.

Figure 3

Intraoperative photograph showing multiple perforations in the ileum depicted by black arrows.

Figure 4

The resected segment of ileum in kidney tray.

Outcome and follow-up

The patient had a stormy postoperative period and remained on ionotropic support in the intensive care unit. Oliguria ensued progressively over subsequent 48 hours leading to a rise in serum creatinine level to 250 µmol/L. She underwent haemodialysis for the same but urine output did not improve. Her requirement of ionotropic support gradually increased and the stoma remained oedematous and non-functional even on the third postoperative day. She subsequently developed acute respiratory distress syndrome and finally succumbed to multiorgan dysfunction on postoperative day five. The histopathological examination of the resected bowel revealed granulomatous inflammation and extensive serositis (figure 5). Ziehl-Neelsen staining of the tissue demonstrated acid-fast bacilli (figure 6).

Figure 5

Microphotograph of the specimen: 10x view after H&E stain showing granulomatous inflammation (white arrow).

Figure 6

Ziehl-Neelsen stain demonstrating acid-fast bacilli (black arrow) in the tissue.

Discussion

Active TB during the pregnancy leads to increased maternal morbidity but maternal mortality is rare, particularly in immunocompetent pregnant women with active TB. Perinatal mortality, four times more frequent in babies born to mothers with active TB, is a serious complication of untreated active disseminated TB during the pregnancy.1 EPTB, which constitutes about 15%–27% of all cases of TB, has a poor outcome and leads to increased morbidity and mortality.2 EPTB in pregnancy, especially ulcerative gastrointestinal variant leading to intestinal perforation, is rare and that too occurs mostly in immunosuppressed individuals.

In low/middle-income countries, the prevalence of TB in pregnant women parallels that of the general population, and TB is the most common cause of mortality in women of reproductive age than all causes combined. According to the WHO estimates, the incidence of TB is 8.6 million cases of which 80% of cases are concentrated in just 22 countries and India is ranked as the country with the highest burden of TB.3

Early diagnosis and treatment of active TB especially disseminated TB is important for better perinatal and maternal outcomes. Active EPTB is clinically difficult to diagnose during the pregnancy because of its non-specific symptoms, that mimic physiological changes of pregnancy. Amenorrhoea caused by abdominal TB can be misconstrued as pregnancy. Similarly, vomiting and increased urinary frequency can be related to physiological changes in pregnancy, leading to diagnostic delays.4 In developed countries, due to the early diagnosis and treatment of active TB, the outcome of pregnancy is rarely influenced.5 But in low/middle-income nations, it worsens pregnancy outcomes significantly. A recent worldwide increase in multidrug resistant/extensively drug resistant tuberculosis (MDR/XDR TB) has once again increased the incidence of pregnancy-associated TB and its adverse effects.

EPTB including female genital TB is difficult to diagnose due to its non-specific presentation, varied results of imaging and less accurate diagnostic tests. Biopsy and surgical intervention during the pregnancy may not be possible because of the risk of preterm labour, reduced accessibility of the lesions and anaesthetic risk to the foetus.6 The diagnostic test for EPTB should be accurate, fast, easy-to-implement, sustainable and affordable. Interferon-gamma release assays are likely comparable to tuberculin skin tests and are used during the pregnancy.

Tuberculous lymphadenitis, the most common form of EPTB, has minimal effect on maternal and fetal outcomes as it facilitates early detection.7 Gastrointestinal TB with multiple perforations, being rare and associated with worse outcomes, should be suspected and diagnosed early. Endoscopic or fine-needle aspiration biopsy may help to diagnose some intestinal TB patients, without a formal laparotomy.8

Progressive TB should be diagnosed early and antitubercular drugs prescribed as soon as the diagnosis is made but with caution as some of them can cause potential teratogenic effects. Isoniazid, ethambutol and rifampicin are relatively safe to use during the pregnancy and many international organisations, including the International Union Against Tuberculosis and Lung Diseases, British Thoracic Society, American Thoracic Society, the WHO, as well as the Revised National Tuberculosis Control Programme of India, now recommend the use of pyrazinamide.9 10

Perinatal morbidity and mortality increase in mothers with active TB especially EPTB. Preterm birth, fetal growth retardation and infants with low Apgar scores after birth are frequently seen.11

Our patient presented after a full-term stillbirth with multiple ileal perforations (twenty in number) that led to septic shock and a CT scan suggested active pulmonary TB. This is a rare case of undiagnosed disseminated TB with pulmonary, gastrointestinal and genitourinary tract involvement presenting after a full-term pregnancy.

The patient was asymptomatic during the whole pregnancy but an acute onset and rapidly worsening clinical course of the disease led to the patient’s demise. Hence, a proactive suspicion, an early diagnosis and management are necessary for a better perinatal and maternal outcome. In India and other nations with a high prevalence of active TB in the reproductive age group, antenatal screening for active TB during the pregnancy would be useful in decreasing maternal and perinatal morbidity and mortality.

Learning points

  • Tuberculosis (TB) is a substantial imitator, disguising various clinical conditions.

  • Although pulmonary TB in pregnant women poses risk for their infants, extrapulmonary TB outcomes are not well known during the pregnancy.

  • Extrapulmonary TB’s effects on pregnancy depend on the sites involved, severity and duration of the disease and pregnancy-associated complications.

  • This case report emphasises the need for early diagnosis and treatment of TB, preferably before pregnancy and regular antenatal follow-up for an excellent obstetric and perinatal outcome.

Acknowledgments

We are grateful to Dr Navin Kumar for his contribution to patient care. CT images were provided, courtesy of the department of Radio diagnosis, AIIMS Rishikesh.

Footnotes

  • Twitter @DeepakR42189905

  • Contributors DR was the treating clinician and wrote the first draft of the manuscript. AG and RR researched literature and conceived the study. AK reported the findings on histopathological examination. All authors reviewed and edited the manuscript and approved the final version of the manuscript.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Patient consent for publication Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.

References

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