Disseminated Macrolide-Resistant Mycobacterium intracellulare Infection in a Child with Autoimmune Lymphoproliferative Disorder: A Case Report and Literature Review
Issued Date
2024-11-06
Resource Type
eISSN
14761645
Scopus ID
2-s2.0-85208772494
Pubmed ID
39226904
Journal Title
The American journal of tropical medicine and hygiene
Volume
111
Issue
5
Start Page
1051
End Page
1055
Rights Holder(s)
SCOPUS
Bibliographic Citation
The American journal of tropical medicine and hygiene Vol.111 No.5 (2024) , 1051-1055
Suggested Citation
Nithirungruang P., Tanpowpong P., Getsuwan S., Boonsathorn S. Disseminated Macrolide-Resistant Mycobacterium intracellulare Infection in a Child with Autoimmune Lymphoproliferative Disorder: A Case Report and Literature Review. The American journal of tropical medicine and hygiene Vol.111 No.5 (2024) , 1051-1055. 1055. doi:10.4269/ajtmh.24-0076 Retrieved from: https://repository.li.mahidol.ac.th/handle/20.500.14594/102083
Title
Disseminated Macrolide-Resistant Mycobacterium intracellulare Infection in a Child with Autoimmune Lymphoproliferative Disorder: A Case Report and Literature Review
Author's Affiliation
Corresponding Author(s)
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Abstract
A 4-year-old girl, previously treated with prednisolone and cyclosporin A due to autoimmune lymphoproliferative syndrome, presented with acute fever, progressive pancytopenia, intermittent abdominal pain, and acute episode of hematemesis. Esophagogastroduodenoscopy revealed diffuse erythematous gastric mucosa and blunt duodenal villi. Histopathology of the duodenal mucosa was notable for numerous acid-fast bacilli within foamy macrophages in the lamina propria, and nontuberculous Mycobacterium DNA was detected by polymerase chain reaction of duodenal tissue. Azithromycin, rifampicin, and ethambutol were started while waiting for species identification and drug susceptibility testing. Macrolide-resistant Mycobacterium intracellulare was demonstrated from blood culture, indicating disseminated infection. The patient died of overwhelming infection, despite receiving newly adjusted regimen (rifampicin, ethambutol, clofazimine, and amikacin). This case highlights the importance of considering disseminated Mycobacterium avium complex (MAC) in the differential diagnosis of immunocompromised, non-HIV patients presenting with gastrointestinal manifestations. Early identification of macrolide-resistant MAC is crucial for guiding appropriate treatment and potentially improve patient outcomes.