Cutaneous Mycobacterium szulgai infection in a patient with Cushing's syndrome: a case report and literature review

Abstract Background Opportunistic infection is an under-recognized complication of Cushing’s syndrome, with infection due to atypical mycobacterium rarely reported. Mycobacterium szulgai commonly presents as pulmonary infection, with cutaneous infection seldom reported in the literature. Case Presen...

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Main Authors: Haiyan Ye, Vanessa C. Harris, Kelvin Hei-Yeung Chiu, Shuang Chen, Fanfan Xing, Linlin Sun, Chaowen Deng, Jin Yang, Jasper Fuk-Woo Chan, Kwok-Yung Yuen
Format: Article
Language:English
Published: BMC 2023-04-01
Series:BMC Infectious Diseases
Subjects:
Online Access:https://doi.org/10.1186/s12879-023-08253-5
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author Haiyan Ye
Vanessa C. Harris
Kelvin Hei-Yeung Chiu
Shuang Chen
Fanfan Xing
Linlin Sun
Chaowen Deng
Jin Yang
Jasper Fuk-Woo Chan
Kwok-Yung Yuen
author_facet Haiyan Ye
Vanessa C. Harris
Kelvin Hei-Yeung Chiu
Shuang Chen
Fanfan Xing
Linlin Sun
Chaowen Deng
Jin Yang
Jasper Fuk-Woo Chan
Kwok-Yung Yuen
author_sort Haiyan Ye
collection DOAJ
description Abstract Background Opportunistic infection is an under-recognized complication of Cushing’s syndrome, with infection due to atypical mycobacterium rarely reported. Mycobacterium szulgai commonly presents as pulmonary infection, with cutaneous infection seldom reported in the literature. Case Presentation 48-year-old man with a newly-diagnosed Cushing’s syndrome secondary to adrenal adenoma presented with a subcutaneous mass on the dorsum of his right hand, was diagnosed with cutaneous Mycobacterium szulgai infection. The most likely source of the infection was through minor unnoticed trauma and inoculation from a foreign body. The patient’s Cushing’s syndrome, high serum cortisol levels and secondary immune suppression facilitated mycobacterial replication and infection. The patient was successfully treated with adrenalectomy, surgical debridement of cutaneous lesion, and a combination of rifampicin, levofloxacin, clarithromycin, and ethambutol for 6 months. There were no signs of relapse one year after cessation of anti-mycobacterial treatment. A literature review on cutaneous M. szulgai infection to further characterize the clinical characteristics of this condition, identified 17 cases of cutaneous M. szulgai infection in the English literature. Cutaneous M. szulgai infections with subsequent disease dissemination are commonly reported in immunocompromised hosts (10/17, 58.8%), as well as in immunocompetent patients with a history of breached skin integrity, such as invasive medical procedures or trauma. The right upper extremity is the most commonly involved site. Cutaneous M. szulgai infection is well controlled with a combination of anti-mycobacterial therapy and surgical debridement. Disseminated infections required a longer duration of therapy than localized cutaneous infections. Surgical debridement may shorten the duration of antibiotics. Conclusions Cutaneous M. szulgai infection is a rare complication of adrenal Cushing’s syndrome. Further studies are needed to provide evidence-based guidelines on the best combination of anti-mycobacterial and surgical therapy for managing this rare infective complication.
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spelling doaj.art-d43bf1668a8c40dc99850b82ba7a0e7f2023-04-30T11:08:42ZengBMCBMC Infectious Diseases1471-23342023-04-012311710.1186/s12879-023-08253-5Cutaneous Mycobacterium szulgai infection in a patient with Cushing's syndrome: a case report and literature reviewHaiyan Ye0Vanessa C. Harris1Kelvin Hei-Yeung Chiu2Shuang Chen3Fanfan Xing4Linlin Sun5Chaowen Deng6Jin Yang7Jasper Fuk-Woo Chan8Kwok-Yung Yuen9Department of Infectious Disease and Microbiology, The University of Hong Kong-Shenzhen HospitalDepartment of Internal Medicine, Division of Infectious Diseases and Department of Global Health, Amsterdam University Medical Centers, University of AmsterdamDepartment of Microbiology, Queen Mary Hospital, Hong Kong Special Administrative RegionDepartment of Pathology, The University of Hong Kong-Shenzhen HospitalDepartment of Infectious Disease and Microbiology, The University of Hong Kong-Shenzhen HospitalDepartment of Infectious Disease and Microbiology, The University of Hong Kong-Shenzhen HospitalDepartment of Infectious Disease and Microbiology, The University of Hong Kong-Shenzhen HospitalDepartment of Infectious Disease and Microbiology, The University of Hong Kong-Shenzhen HospitalDepartment of Infectious Disease and Microbiology, The University of Hong Kong-Shenzhen HospitalDepartment of Infectious Disease and Microbiology, The University of Hong Kong-Shenzhen HospitalAbstract Background Opportunistic infection is an under-recognized complication of Cushing’s syndrome, with infection due to atypical mycobacterium rarely reported. Mycobacterium szulgai commonly presents as pulmonary infection, with cutaneous infection seldom reported in the literature. Case Presentation 48-year-old man with a newly-diagnosed Cushing’s syndrome secondary to adrenal adenoma presented with a subcutaneous mass on the dorsum of his right hand, was diagnosed with cutaneous Mycobacterium szulgai infection. The most likely source of the infection was through minor unnoticed trauma and inoculation from a foreign body. The patient’s Cushing’s syndrome, high serum cortisol levels and secondary immune suppression facilitated mycobacterial replication and infection. The patient was successfully treated with adrenalectomy, surgical debridement of cutaneous lesion, and a combination of rifampicin, levofloxacin, clarithromycin, and ethambutol for 6 months. There were no signs of relapse one year after cessation of anti-mycobacterial treatment. A literature review on cutaneous M. szulgai infection to further characterize the clinical characteristics of this condition, identified 17 cases of cutaneous M. szulgai infection in the English literature. Cutaneous M. szulgai infections with subsequent disease dissemination are commonly reported in immunocompromised hosts (10/17, 58.8%), as well as in immunocompetent patients with a history of breached skin integrity, such as invasive medical procedures or trauma. The right upper extremity is the most commonly involved site. Cutaneous M. szulgai infection is well controlled with a combination of anti-mycobacterial therapy and surgical debridement. Disseminated infections required a longer duration of therapy than localized cutaneous infections. Surgical debridement may shorten the duration of antibiotics. Conclusions Cutaneous M. szulgai infection is a rare complication of adrenal Cushing’s syndrome. Further studies are needed to provide evidence-based guidelines on the best combination of anti-mycobacterial and surgical therapy for managing this rare infective complication.https://doi.org/10.1186/s12879-023-08253-5Mycobacterium szulgaiCushing’s syndromeHypercortisolismCutaneous infectionCase report
spellingShingle Haiyan Ye
Vanessa C. Harris
Kelvin Hei-Yeung Chiu
Shuang Chen
Fanfan Xing
Linlin Sun
Chaowen Deng
Jin Yang
Jasper Fuk-Woo Chan
Kwok-Yung Yuen
Cutaneous Mycobacterium szulgai infection in a patient with Cushing's syndrome: a case report and literature review
BMC Infectious Diseases
Mycobacterium szulgai
Cushing’s syndrome
Hypercortisolism
Cutaneous infection
Case report
title Cutaneous Mycobacterium szulgai infection in a patient with Cushing's syndrome: a case report and literature review
title_full Cutaneous Mycobacterium szulgai infection in a patient with Cushing's syndrome: a case report and literature review
title_fullStr Cutaneous Mycobacterium szulgai infection in a patient with Cushing's syndrome: a case report and literature review
title_full_unstemmed Cutaneous Mycobacterium szulgai infection in a patient with Cushing's syndrome: a case report and literature review
title_short Cutaneous Mycobacterium szulgai infection in a patient with Cushing's syndrome: a case report and literature review
title_sort cutaneous mycobacterium szulgai infection in a patient with cushing s syndrome a case report and literature review
topic Mycobacterium szulgai
Cushing’s syndrome
Hypercortisolism
Cutaneous infection
Case report
url https://doi.org/10.1186/s12879-023-08253-5
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