Received Date: 05/03/2019; Accepted Date: 26/03/2019; Published Date: 02/04/2019
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Basidiobolomycosis is a rare fungal infection caused by Basidiobolus ranarum that mainly can lead to subcutaneous infection and it's unusual to involve the gastrointestinal tract. Gastrointestinal basidiobolomycosis is difficult to diagnose primarily due to its non-specific clinical presentation in which it can mimic appendicitis or malignancy. This case report describes a child with severe gastrointestinal basidiobolomycosis in which it leads to fatality.
Basidiobolus ranarum (B. ranarum) is a fungus of zygomycetes class found in soil and the dung of reptiles, amphibians and bats that can be transmitted through traumatic inoculation which causes basidiobolomycosis a rare fungal infection and it's well known to cause subcutaneous infection but rarely it can lead to gastrointestinal manifestations [1,2]. Gastrointestinal basidiobolomycosis mostly involve the colon and it's difficult to diagnose due to its non-specific presentation as the patient may have abdominal pain, fever, constipation or diarrhea and weight loss but it should be a differential especially in pediatric patients present with abdominal mass and eosinophilia as gastrointestinal basidiobolomycosis can be misdiagnosed as appendicular mass, intestinal tuberculosis or malignancy [2-5]. Basidiobolomycosis has been reported in both children and adults.
We report a case of 6 years old, Saudi, male patient presented to Emergency department (ER) with abdominal pain especially in the right lower quadrant, fever and vomiting, based on investigations he was diagnosed as inflammatory bowel disease secondary to intra-abdominal abscess due to basidiobolomycosis.
A previously healthy 6 years old boy presented to the ER complaining of abdominal pain of few weeks duration, that was on and off, mainly in the right lower quadrant of the abdomen and progressed to be severe in the last 10 days. Associated with fever and vomiting, with no history of nausea, anorexia, change in bowel habit or urinary symptoms. Few weeks prior to our hospital visit, the patient sought medical advice where he was given IV antibiotic and discharged home on oral antibiotic for suspected complicated appendicitis along with urinary tract infection. Computed tomography (CT) abdomen was done at that time and showed:
1) Enlarged tubular structures in the right iliac fossa region measuring 8 cm X 2.5 cm related to the cecum,
2) Suspected appendicular lesions
3) Free fluid collection
4) Multiple enlarged mesenteric and right iliac lymph nodes.
On physical examination, patient was conscious, alert, oriented and cooperative. His vital signs upon presentation, Pulse 120 beat per minute, blood pressure 93/66 mmHg, Temperature 36.7 Celsius. The abdomen was soft, lax, not distended with tenderness in the right iliac fossa and mass felt in the right lower abdomen and positive bowel sound. Initial laboratory workup revealed: White blood cells (WBCs) 20.4 k/uL, neutrophils (NE) 61.5%, hemoglobin (HB) 10.1 g/dL, hematocrit (HCT) 32%, platelet (PLT) 589.000/mm3, sodium (Na) 132 mmol/L, potassium (K) 4.1 mmol/L, Urea 0.2 mmol/L, Creatinine 39 umol/L. Imaging studies including abdominal ultrasound (US) and CT abdomen with oral and intravenous (IV) contrast were done. Abdominal US showed hepatomegaly, Hypo echoic mass seen at right iliac fossa measures 4.9 cm X 4.2 cm, fluid collection with appendicular mass (Figure 1).
While CT abdomen demonstrated large pelvic right iliac fossa complex lesion connected to a tubular enhancing lesion likely matching the clinical diagnosis of appendicular abscess (Figure 2).
Based on these result, the decision was to operate on the patient. Intraoperative findings revealed large amount of pus, multiple intraperitoneal masses related to terminal ileum, paracecal and proximal ileal loops, inflamed mass at paracecal region with necrotic wall of cecum and appendix. Ileocecectomy and specimen for histopathology were sent with 2 drains inserted. Postoperative course was smooth at the beginning, patient was extubated and shifted to the ward and received 3rd generation of cephalosporin’s and metronidazole and kept nil per os (NPO) on nasogastric tube (NGT). Day 6 postoperative, oral fluid diet was reintroduced gradually and was well tolerated which lead us to resumption of normal diet. Postoperative day 8 the result of histopathology revealed large Cecal ulcer 5 cm with ileocecal mass with focal cecal perforation, wall thickening rich in eosinophil’s and thick fungal hyphae Basidiobolomycosis. Afterwards, the patient started to have surgical wound infection with positive C/S of Pseudomonas aeruginosa, bedside wound opening was done with daily irrigation and dressing and antibiotics changed according to the sensitivity. Day 15 postoperative, he started to deteriorate with history of fever and vomiting. On the subsequent day he developed tachycardia, abdominal distention, constipation and spikes of fever. The patient was kept NPO and NGT was inserted which brought moderate amount of brownish content. Abdominal X-ray, which was ordered to be done on daily basis, showed multiple air fluid level (Figure 3).
Emergent abdominal US revealed minimal fluid in right iliac fossa, most likely post-surgical status. Blood workup result demonstrated WBCs 23.8 k/uL, NE 88.1%, HB 11.2 g/dL and normal urea and electrolytes. On the next day, HB dropped to 9.6 g/ dL with persistent bilious gastric aspiration, abdominal distension and tenderness. Postoperative day 18, the patient was pushed to operation room for re-exploration. Intraoperative findings included multiple intra-abdominal abscesses eroding the small bowel with inflamed bowel wall, multiple necrotic segments, severe adhesions with inflammatory masses invading the recto sigmoid region, small ileostomy and colostomy were performed and drains inserted.
Post-operative, the patient pushed to PICU while still intubated, sedated and hemodynamically stable. Later on the same day, 40 min of CPR was performed after he arrested but he couldn't be resuscitated.
Zygomycosis is a rare fungal infection caused by two forms: Mucormycosis and Entomophthoromycosis . Conidiobolus coronatus and B. ranarum are the two major species causing entomophthoromycosis [2,5]. The first isolation of B. ranarum was in 1955 from crumbling plants . The first recorded human infection caused by B. ranarum was in 1956 . Gastrointestinal basidiobolomycosis was diagnosed for the first time in 1980 in 4 years old child that died soon after the diagnosis due to disseminated fungal infection and the first culture proves gastrointestinal basidiobolomycosis reported in 1986 in the United States [4,8]. Basidiobolomycosis is known to cause soft tissue infections especially in tropical areas [4,5]. Recently, an increase in the incidence of basidiobolomycosis infection has been reported in which it may be attributed to the increase of immune compromised state of general populations around the world . The risk factors for basidiobolomycosis remain unclear and It's not obvious how the fungus get inside the gastrointestinal tract of the patients in case of gastrointestinal basidiobolomycosis but it's suspected that ingestion of food contaminated by soil or animal feces is the source . In our case, the patient presented with severe abdominal pain in the right lower quadrant, fever and vomiting, these complaints may misdiagnosed as appendicitis or inflammatory bowel disease that's why the diagnosis of gastrointestinal basidiobolomycosis poses some difficulties. The diagnosis of basidiobolomycosis infection was made based on surgical specimen. The management was a combination between surgical intervention and medical treatment. As based on literature, the optimal way to manage gastrointestinal basidiobolomycosis is by surgical resection of all necrotic bowel and debridement for any involved tissue followed by 3 months of antifungal treatment . The best antifungal agent remain debatable as previously reported treatment failure with amphotericin B but the use of itraconazole considered an effective choice in gastrointestinal basidiobolomycosis [8-11].
This case report describes a child with severe gastrointestinal basidiobolomycosis in which it leads to fatality.