Child with dengue haemorrhagic fever complicated by ileocaecal intussusception

Abstract

Dengue fever is one of the most common neglected tropical diseases with an increasing trend seen in Sri Lanka and many other tropical countries. A number of unusual presentations and complications of dengue fever have been reported, and acute abdomen is one such presentation. However, intussusception as the cause of acute abdomen in a patient with dengue haemorrhagic fever has not been previously reported. The authors report a child who was diagnosed to have intussusception during the critical phase of dengue haemorrhagic fever while having a platelet count of 15×10³/cumm. The child had initial point-of-care ultrasound followed by detailed ultrasound of the abdomen by the radiologist, and the diagnosis of ileocaecal intussusception was confirmed. Intussusception was relieved by saline reduction and without needing laparotomy. Early diagnosis and non-operative management prevented emergency laparotomy.

Background

Dengue is a common mosquito-borne viral infection in tropical and subtropical countries.¹ Approximately, 50% of world population live in dengue endemic regions and are at risk for getting infected with dengue and having dengue haemorrhagic fever.² Dengue has a wide spectrum of symptoms that ranges from self-limited, asymptomatic infection to severe and refractory shock and is a recognised cause of mortality due to dengue shock syndrome, bleeding and other less frequent severe complications.³ Involvement of multiple organ systems and unusual presentations in dengue fever led to identification of ‘expanded dengue syndrome’ by the World Health Organisation in 2012.⁴ In expanded dengue syndrome, any organ including brain, heart, kidney, liver, pancreas, spleen and gastrointestinal tract can be involved.⁵

Acute abdomen is also a recognised unusual presentation of dengue fever.⁶ Recognition of surgical abdomen without delay is essential to prevent morbidity and mortality as non-specific abdominal pain in dengue fever can be a hindrance to early diagnosis. Further, in patients with dengue haemorrhagic fever, abdominal pain commonly occurs due to tender hepatomegaly, myalgia and sometimes due to hypovolaemia and co-occurrence of these manifestations may deviate the clinician from diagnosing surgical abdomen accurately. The authors, to the best of their knowledge, report the first account of a patient who developed acute abdomen and subsequently, was diagnosed to have intussusception during the critical phase of dengue haemorrhagic fever.

Case presentation

A 10-year-old previously healthy child presented with fever for 3 days associated with periorbital pain, myalgia and generalised body aches. She also had non-specific central abdominal pain and loose stools twice following admission. Her sister had dengue fever and was recently discharged from the local hospital. Physical examination revealed an obese (body mass index 28 kg/m²), ill looking, irritable child with congested eyes. She was haemodyanamically stable. Following confirmation of the diagnosis of dengue fever, she was commenced on monitoring and supportive care for dengue fever.

On day 4 of febrile illness, she developed nausea, vomiting and right hypochondrial pain. Intravenous fluids and antiemetics were commenced as oral intake was poor with vomiting. Packed cell volume showed a rise of more than 20% from baseline and point-of-care ultrasound confirmed right-sided pleural effusion. The onset of critical phase of dengue haemorrhagic fever was identified based on these findings. During the critical phase, she did not have clinically apparent bleeding manifestations and vital parameters remained stable. However, on day 6 of febrile illness, she developed severe, colicky right lower abdominal pain which was unusual in character for dengue haemorrhagic fever. She also had tenderness over right lower quadrant of abdomen with no rigidity.

Investigations

Full blood count on day 3 of febrile illness revealed a white cell count of 3.5×10³/ cumm and platelet count of 150×10³/cumm. Dengue NS1 antigen was positive. On day 4, she had progressive leucopenia (1.4×10³/cumm) and thrombocytopenia (95×10³/cumm). Liver functions were deranged (ALT 225 U/L, AST 325U/L). Point-of-care ultrasound confirmed evidence of fluid leakage (right-sided pleural effusion) and dengue haemorrhagic fever on day 5. On the same day, white cell count and platelet counts were 1.8×10³/cumm and 60×10³/cumm, respectively. The diagnosis of dengue haemorrhagic fever was confirmed by demonstration of dengue-specific IgM and IgG antibodies on day 7 of febrile illness.

Although she was haemodyanamically stable on day 6 of febrile illness and during the critical phase, her platelet count further dropped to 15×10³/cumm and developed colicky right lower abdominal pain. A detailed ultrasound abdomen was performed by the radiologist to exclude surgical abdomen and ultrasound confirmed the diagnosis of ileocaecal intussusception.

Differential diagnosis

Signs of acute abdomen are seen in approximately 4%–12% of patients with dengue fever.⁷ Differential diagnosis for acute abdomen in patients with dengue fever includes calculous and acalculous cholecystitis, acute appendicitis, peritonitis,⁷ subacute intestinal obstruction, splenic rupture⁴ and acute pancreatitis.⁸ The reason why dengue fever leads to subacute intestinal obstruction is unknown although intestinal wall oedema may be a predisposing factor.⁴ It is important to assess the characteristics of abdominal pain, associated symptoms and location of abdominal tenderness for clinical differentiation of these causes. In children with doubtful diagnosis and who are otherwise haemodyanamically stable, abdominal imaging helps in arriving at definitive diagnosis while avoiding unnecessary surgical interventions. Further, septicaemia is also among the differential diagnosis and can present both with thrombocytopenia and acute abdomen. Blood culture is helpful to identify the aetiology of bacteremia.

Treatment

The critical phase was managed with the WHO protocol on management of dengue haemorrhagic fever⁹ with careful monitoring of urine output and vital parameters. As no complications were seen, child was managed with saline reduction for reducing intussusception. The procedure was successful and she recovered from intussusception without needing open surgery and with no postprocedural complications. Haemodynamic monitoring was continued until she made recovery. She was notified to the Epidemiology unit of Sri Lanka as part of national surveillance of communicable diseases.

Outcome and follow-up

The child was reviewed after 3 days to assess symptoms of recurrent intussusception and the assessment revealed that she did not have any gastrointestinal symptoms. Full blood count 1 week after discharge revealed normal platelet counts indicating complete haematological recovery.

Discussion

Accurate and timely diagnosis of acute abdomen is often difficult in children with dengue fever since most children report of abdominal pain during routine evaluation and surgical abdomen in patients with dengue fever is rare. On the other hand, children with predominant symptoms of acute abdomen are initially referred to surgeons and dengue fever could be diagnosed late due to predominant initial focus on surgical abdomen distracting clinicians from the underlying aetiology.⁷ Acute abdomen is likely a co-occurrence in patients with dengue fever rather than a direct effect of dengue fever although pathophysiological processes of dengue fever can predispose to complications that can present with acute abdomen.⁷

Intussusception as a cause of acute abdomen in patients with dengue fever has not been reported hence little is known about its pathophysiology. The histological findings of enlarged mesenteric lymph nodes, serous fluid collections and oedema have been demonstrated in previously reported cases of dengue haemorrhagic fever complicated with acute appendicitis and acalculus cholecystitis.^9–11 Dengue fever further has been reported to cause mesenteric adenitis due to widespread inflammation.⁶ It is possible that an enlarged mesenteric lymph node or Peyer’s patch can act as a lead point causing invagination of intestines into one another. Thrombocytopenia and dysfunction of surviving platelets in dengue haemorrhagic fever can lead to gastrointestinal bleeding and spasms.^{12 13} The risk of bleeding is further enhanced with profound vasculopathy leading to increased vascular fragility and coagulopathy associated with dengue.¹⁴ Coagulopathy in dengue haemorrhagic fever leads to increased fibrinolysis and altered balance of procoagulation and anticoagulation factors.¹⁵ As ileocaecal region is the most common site for intussusception, it is possible that all or some of these factors were contributory in causing intussusception in the reported child.

Similar to accurate diagnosis, management of surgical abdomen is often challenging in the presence of dengue haemorrhagic fever due to high risk for haemodynamic instability, bleeding due to severe thrombocytopenia and need for surgical interventions. It is important to carefully assess the severity of both dengue haemorrhagic fever and surgical abdomen, and decide on management priorities. Haemodynamic stability should be the priority in management. Therefore, it is important that all patients with dengue haemorrhagic fever are managed with supportive care and close monitoring during the critical phase. If bacterial infections are suspected, it is important to commence on broad-spectrum antibiotics.

Although intussusception has been previously reported in association with viral infections, dengue fever as a predisposing infection for intussusception has not been reported. Further, intussusception usually presents between ages from 6 months to 2 years and presentation at 10 years is not common.¹¹ In 85%–90% of cases of ileocaecal intussusception, children respond well to ultrasound-guided pneumatic or hydrostatic reduction avoiding the need for laparotomy.¹⁶ The recurrence rate is high during the first 24 hours following reduction and patient needs to be closely observed.¹⁵ Failure of closed reduction, and observation and persistent intussusception resulting in bowel necrosis are indications for urgent laparotomy for correction of intussusception.¹⁷

Learning points.

• Expanded dengue syndrome is associated with multiple organ systems including brain, heart, liver, kidney and gastrointestinal organs.

• Although appendicitis, pancreatitis, cholecystitis, peritonitis have been very rarely reported in association with dengue fever, intussusception following dengue fever is extremely rare and has not been previously reported.

• Early accurate diagnosis and management is often challenging when acute abdomen co-occurs with dengue haemorrhagic and achievement of haemodynamic stability should be the management priority.

• Most ileo-caecal intussusceptions in children can be managed without needing laparotomy although failure of closed reduction and bowel necrosis warrant urgent laparotomy.