CASE REPORT

Embolisation for caecal bleeding in a child with typhlitis

Manou S. de Lijster & Anne M. Smets &

Henk van den Berg & Jim A. Reekers

Received: 25 January 2014 /Revised: 26 April 2014 /Accepted: 14 May 2014# Springer-Verlag Berlin Heidelberg 2014

Abstract A 16-year-old girl being treated for a relapse ofpromyelocytic leukaemia developed typhlitis of the caecumand ascending colon related to Klebsiella septicaemia duringthe neutropenic phase, 2 weeks after the start of inductiontreatment with chemotherapy. After 10 days of treatmentwith parenteral feeding and antibiotics, massive rectal bloodloss occurred, causing haemodynamic instability. Contrast-enhanced abdominal CT showed contrast extravasationin the caecal lumen. This life-threatening situation promptedvisceral angiography, which confirmed a contrast blush in thecaecum. Subsequent embolisation resulted in haemodynamicstability.

Keywords Neutropenic colitis . Klebsiella septicaemia .

Intestinal haemorrhage . Image-guided embolisation .

Adolescent

Introduction

One of the complications of chemotherapy is typhlitis,also called neutropenic enterocolitis. With the use ofintensive chemotherapy regimens for various tumour types,typhlitis has become an increasingly frequent complicationof oncological therapy,mainly in neutropenic patients. The exactpathophysiology of typhlitis is unknown, but a combinationof immunosuppression and direct cytotoxicity to the bowelwall resulting inmucosal injury are thought to play an importantrole. Secondary infection from bacterial invasion resulting insepsis and bowel perforation are severe complications [1]. Inmost cases the bowel wall of the ileocecal segment is affected,showing transmural inflammation. Typhlitis is often associatedwith bone marrow aplasia or neutropenia.

Complications of typhlitis that need immediate treatmentare perforation and gastrointestinal haemorrhage. Classictreatment for these complications is surgical exploration [2].

Case report

A 16-year-old girl diagnosed with acute myeloid leukaemiarelapse was treated with fludarabine, cytarabine, liposomaldaunorubicin and intrathecal chemotherapy. Fourteen daysafter this initial treatment the girl developed septicaemia fromextended spectrum beta lactamase Klebsiella pneumoniae.

Antibiotics were switched from ceftazidim and gentamycinto meropenem and ciproxin. At this time the girl developedfever, right lower quadrant pain and diarrhoea. Abdominal USshowed thickening of the caecal and ascending colon wall upto the hepatic flexure and a small amount of free intraperitoneal

M. S. de Lijster (*)Department of Radiology, Academic Medical Centre,Meibergdreef 9, 1105AZ Amsterdam, The Netherlandse-mail: m.s.delijster@amc.uva.nl

A. M. SmetsDepartment of Pediatric Radiology, Academic Medical Centre,Amsterdam, The Netherlands

H. van den BergDepartment of Pediatrics, Academic Medical Centre,Amsterdam, The Netherlands

J. A. ReekersDepartment of Interventional Radiology, Academic Medical Centre,Amsterdam, The Netherlands

Pediatr RadiolDOI 10.1007/s00247-014-3059-0

fluid. Clinical symptoms in combination with US findingssuggested the diagnosis of neutropenic enterocolitis/typhlitis.

During the following 6 days, antibiotic treatment was con-tinued and the girl received parenteral nutrition. On the 7thday, enteral feeding was resumed and gradually increased.The 11th day after the initial onset of abdominal discomfortthe girl had massive rectal bleeding and became haemody-namically unstable. Her condition stabilized with blood trans-fusions and saline and dextrose infusions.

Contrast-enhanced abdominal CT showed contrast extrav-asation in the caecal lumen (Fig. 1). Emergency visceralangiography of the superior mesenteric artery then showedcontrast material entering the caecal lumen, indicating thesource of active bleeding (Fig. 2). After super-selective cath-eterisation of the superior mesenteric artery branch supplyingthe caecum, a blush was shown again (Fig. 3). Trans-catheterembolisation was performed using two micro-coils. Controlangiography at the end of the procedure showed completeocclusion of the vessel (Fig. 4).

Clinically, gastrointestinal blood loss stopped after theembolisation. Follow-up sonography was performed regularlyto monitor bowel wall thickness. Broad-spectrum antibiotictreatment (metronidazole) was continued per os for as long asbowel wall thickening was seen. After 1 week the girl wasdischarged from the hospital. During the month after

discharge, she developed several complications related totreatment of her acute non-lymphoblastic leukaemia, but rec-tal blood loss did not reappear. The girl died 6 months laterfrom a fungal infection after bone marrow transplant.

Discussion

The overall incidence of typhlitis or neutropenic enterocolitisin oncological patients is reported to be 1–26% [1]. In childrentreated for acute lymphoblastic leukaemia (ALL) or acutemyeloid leukaemia (AML) the incidence of typhlitis is 8–12% [2]. Treatment with chemotherapy for haematologicalmalignancies, mucositis and stem cell transplantation are riskfactors for developing typhlitis [3].

Fig. 1 Axial contrast-enhanced abdominal CT in a 16-year-old girl, whowas being treated for leukaemia and had suspected typhlitis, showscontrast extravasation (arrow) in the caecal lumen

Fig. 2 Emergency visceral angiography of the superior mesenteric arteryin the same 16-year-old girl shows contrast material entering the caecallumen (arrow), indicating the source of active bleeding

Fig. 3 Angiography in the 16-year-old girl shows a blush (arrow) aftersuper-selective catheterisation of the superior mesenteric artery branchsupplying the caecum

Fig. 4 Control angiography in the 16-year-old girl shows completeocclusion of the vessel after trans-catheter embolisation performed usingtwo micro-coils

Pediatr Radiol

The pathophysiology is not fully understood, but severalmechanisms are associated with the development of typhlitisin immunocompromised patients. Direct cytotoxicity inducedby chemotherapeutic drugs results in weakness of the mucosalbarrier. Overgrowth of bacterial flora or secondary infectioncan result in severe damage to the bowel wall [1, 4, 5]. Thecaecum and ascending colon have a marginal blood supply,which makes them susceptible to the development of typhlitis.However the transverse colon and descending colon andrectum can also be involved. In the literature the most fre-quently reported symptoms experienced by children withtyphlitis are abdominal pain (91%), fever (84%), abdominaltenderness (82%) and diarrhoea (72%) [6].

High alertness is necessary to recognise the diagnosis,because of the non-specificity of the symptoms. During theneutropenic phase after treatment with chemotherapy, abdom-inal symptoms can be related to several causes. Typhlitis, butalso perforation and appendicitis, which need to be treatedsurgically, must be considered.

US is the modality of choice to explore the abdomen.Increased bowel wall thickness (>3 mm) in combination withthe clinical symptoms of abdominal pain and fever is highlysuspicious for typhilitis [6, 7].

Treatment of typhlitis is initially conservative with bowelrest, total parenteral nutrition and broad-spectrum antibiotics.Complications of typhlitis such as bowel perforation andnecrosis are treated surgically [6]. Typhlitis can cause severebleeding resulting in haemodynamic instability, as in ourpatient. Surgery has always been the treatment of choice in

cases of life-threatening haemorrhage, but the increasingavailability of interventional radiology and its minimal inva-siveness make it a very good option for first-line treatment ofthis life-threatening complication [8].

Conflicts of interest None

References

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