Gastrointestinal basidiobolomycosis with hepatic dissemination: a case report

Abstract

Introduction:Gastrointestinal basidiobolomycosis (GIB) is an emerging fungal infection with a few cases reported worldwide. It is caused by Basidiobolus ranarum, which does not usually invade blood vessels and rarely disseminates.Case presentation:We present a rare case of GIB with hepatic dissemination in a 12-year-old Yemeni boy living in southwestern Saudi Arabia. The initial provisional diagnosis was intestinal lymphoma, and a right hemicolectomy was carried out, but histopathological assessment ruled out lymphoma and suggested intestinal tuberculosis. Two weeks after starting antituberculous medications, the patient was referred to our hospital because of fever and right upper abdominal discomfort. There was leukocytosis with marked eosinophilia, and a liver biopsy showed evidence of B. ranarum infection. A second opinion by histopathological examination of resected tissues diagnosed colonic basidiobolomycosis. The patient was treated successfully with itraconazole.Conclusion:GIB is an emerging disease in southwestern Saudi Arabia and should be considered in a patient with an abdominal mass and eosinophilia coming from this region. Persistent elevation of leukocytes and eosinophils after surgical resection of the affected tissue could be used as a predictor of fungal dissemination. Further research is needed for a better understanding of GIB.

Disseminated angioinvasive basidiobolomycosis with a favourable outcome

“…Angioinvasion does not usually occur in GIB [16] , [17] , [18] , [19] , [20] . The presence of portal vein occlusion and vascular invasion, as seen in our patient, is rare; the concomitant liver and colonic masses indicating dissemination are also unusual for GIB [13] . Disseminated serious disease has been described in fatal cases of concomitant liver and pulmonary dissemination.…”

“…Intraabdominal malignancy was the initial provisional diagnosis in the majority of cases (43%), followed by inflammatory bowel disease (16%), and diverticulitis [3] . In endemic areas, patients might be treated for abdominal tuberculosis, leading to a delay in definitive diagnosis and treatment, with increases in morbidity and mortality [13] .…”

Human Pathogenic Entomophthorales

“…Infections caused by C. incongruus have been diagnosed in both apparently healthy and immunocompromised hosts (32,49,172,174,184). In contrast to C. coronatus, C. incongruus has been recovered in culture more often from cases of systemic infections than from cases of localized subcutaneous disease (31, 35, (42); fever, anorexia, pleuritic and chest pain, fever, severe wt loss with lung involvement, and dissemination to blood vessels and brain are the main findings (165,169,171) Unusual cases involving subcutaneous tissues in anatomical areas other than the face have been reported (45,209) There is a rare report without culture suggesting disseminating infection due to C. coronatus from the face to liver, kidney, and small intestine (210) Conidiobolus incongruus Cases in humans are rare; patients with this infection showed refractory fever, cellulitis of the forehead with sinusitis, obstruction of the nares, periorbital edema, and orbital inflammation (82,172,174,180) Systemic infections are common in immunocompromised hosts (35,47); involvement of the lungs was a common feature in these patients; anorexia, persistent cough with or without hemoptysis, fever, and wt loss; invasion of internal organs, including with intestinal infection with dissemination to liver, small bowel, gallbladder, pancreas, kidney, or retroperitoneum have been reported (142,185,186,197,227); pulmonary infection spread from a cutaneous lesion was also recorded (191) Reported to occur in apparently healthy hosts; lesions are painless and found around the neck, trunk, limbs, buttocks, and, less frequently, other sites (51,53,183); usually, edematous extensive single granulomatous lesions are observed; in the infected areas, there is moderate to severe pruritus around nodular lesions with eroded to ulcerative granulomas (184,188) Reported to occur in apparently healthy individuals; common clinical signs include abdominal pa...…”

“…13A to D). 207; soft tissue thickening (126) Frontal hypodensity suggesting intracerebral abscesses, pansinusitis, periorbital edema, orbital inflammation (172) Thickening of the intestines with single or multiple tumor-like masses of the colon or rectum; in cases of disseminated infection, similar masses are found in liver, small bowel, gallbladder, pancreas, kidney, and/or peritoneum (192,203,210) MRI Increase in T2 signals of the nose and soft tissues of the upper lips; involvement of the dorsal cavity and infiltration of maxillary sinuses and nasal concha 167A study of a case of pericarditis caused by C. incongruus found large pericardial effusion, causing pericardial tamponade; irregular echogenic densities were also found near the left ventricle (35) Circumferential thickening of cecum and rectum with significant luminal narrowing; retroperitoneal lymphoadenopathies (211) Traditional X ray Presence of soft tissue and opacities due to nasal and facial swelling, with obstruction of the airway passages of left or right antrum; frontal and maxillary sinusitis; no signs of bone involvement (208) Generalized cardiac enlargement, bilateral pulmonary infiltration, posterior mediastinal tumorlike masses (40); progressive low infiltration of the lungs that soon evolves to adjacent sections of the affected lungs (35,42,50) Abnormal collection of gas in the descending colon region; upper gastrointestinal thickened folds in the fundus and body of the stomach and the proximal duodenum (212) Ultrasound Recorded only for the right foot, attributed to C. incongruus (184) ND Detection of tumor-like masses in abdomen and pelvis and thickening of the colon and rectum (120); heterogeneous mass around the abdominal aorta (203) a Shown are the most common findings with each of the selected procedures. ND, not determined (no reports).…”

“…Likewise, these tools were instrumental in determining the extent of damage in cases of disseminated conidiobolomycosis involving the brain (172) and the lungs (32,35,42,50,209) (Table 4). These methodologies have also been extensively used for cases of intestinal and disseminated basidiobolomycosis (120,140,192,203,(210)(211)(212). Thickening of the intestine and the presence of tumor-like masses in the colon, rectum and, in cases of disseminated infection, liver, gallbladder, pancreas, and kidney were typical findings.…”