Disseminated Bacillus Calmette-Guérin (BCG) infection and acute exacerbation of interstitial pneumonitis: an autopsy case report and literature review.
Administration, Intravesical
Aged
Antitubercular Agents
/ therapeutic use
Autopsy
BCG Vaccine
/ administration & dosage
Carcinoma in Situ
/ drug therapy
Fatal Outcome
Humans
Lung Diseases, Interstitial
/ drug therapy
Male
Mycobacterium Infections, Nontuberculous
/ diagnosis
Mycobacterium bovis
/ genetics
Negative Results
Neoplasm Recurrence, Local
/ drug therapy
Pulse Therapy, Drug
Steroids
/ therapeutic use
Symptom Flare Up
Treatment Outcome
Urinary Bladder Neoplasms
/ drug therapy
BCG infection
Bacillus Calmette–Guérin
Exanthema
Hepatosplenomegaly
Interstitial pneumonitis
Mycobacterium bovis
Myelosuppression
Journal
BMC infectious diseases
ISSN: 1471-2334
Titre abrégé: BMC Infect Dis
Pays: England
ID NLM: 100968551
Informations de publication
Date de publication:
29 Sep 2020
29 Sep 2020
Historique:
received:
27
04
2020
accepted:
06
09
2020
entrez:
30
9
2020
pubmed:
1
10
2020
medline:
21
10
2020
Statut:
epublish
Résumé
Intravesical administration of Bacillus Calmette-Guérin (BCG) has proven useful for treatment and prevention of recurrence of superficial bladder cancer and in situ carcinoma. However, fatal side effects such as disseminated infections may occur. Early diagnosis and accurate therapy for interstitial pneumonitis (IP) are important because exacerbation of IP triggered by infections is the major cause of death. Although some fatality reports have suggested newly appeared IP after intravesical BCG treatment, to our knowledge, there are no reports which have demonstrated acute exacerbation of existing IP. Moreover, autopsy is lacking in previous reports. We report the case of a patient with fatal IP exacerbation after BCG instillation and the pathological findings of the autopsy. A 77-year-old man with a medical history of IP was referred to our hospital because of fever and malaise. He had received an intravesical injection of BCG 1 day before the admission. His fever reduced after the use of antituberculosis drugs, so he was discharged home. He was referred to our hospital again because of a high fever 7 days after discharge. On hospitalisation, he showed high fever and systemic exanthema. Hepatosplenomegaly and myelosuppression were also observed. Biopsies revealed multiple epithelioid cell granulomas with Langhans giant cells of the liver and bone marrow. Biopsy DNA analyses of Mycobacterium bovis in the bone marrow, sputum, and blood were negative. His oxygen demand worsened drastically, and the ground-glass shadow expanded on the computed tomography scan. He was diagnosed with acute exacerbation of existing IP. We recommenced the antituberculosis drugs with steroid pulse therapy, but he died on day 35 because of respiratory failure. The autopsy revealed a diffuse appearance of multiple epithelioid cell granulomas with Langhans giant cells in multiple organs, although BCG was not evident. We report the first case of acute exacerbation of chronic IP by BCG infection. This is also the first case of autopsy of a patient with acute exacerbation of existing IP induced by intravesical BCG treatment. Whether the trigger of acute IP exacerbation is infection or hypersensitivity to BCG is still controversial, because pathological evidence confirming BCG infection is lacking. Physicians who administer BCG against bladder cancer should be vigilant for acute exacerbation of IP.
Sections du résumé
BACKGROUND
BACKGROUND
Intravesical administration of Bacillus Calmette-Guérin (BCG) has proven useful for treatment and prevention of recurrence of superficial bladder cancer and in situ carcinoma. However, fatal side effects such as disseminated infections may occur. Early diagnosis and accurate therapy for interstitial pneumonitis (IP) are important because exacerbation of IP triggered by infections is the major cause of death. Although some fatality reports have suggested newly appeared IP after intravesical BCG treatment, to our knowledge, there are no reports which have demonstrated acute exacerbation of existing IP. Moreover, autopsy is lacking in previous reports. We report the case of a patient with fatal IP exacerbation after BCG instillation and the pathological findings of the autopsy.
CASE PRESENTATION
METHODS
A 77-year-old man with a medical history of IP was referred to our hospital because of fever and malaise. He had received an intravesical injection of BCG 1 day before the admission. His fever reduced after the use of antituberculosis drugs, so he was discharged home. He was referred to our hospital again because of a high fever 7 days after discharge. On hospitalisation, he showed high fever and systemic exanthema. Hepatosplenomegaly and myelosuppression were also observed. Biopsies revealed multiple epithelioid cell granulomas with Langhans giant cells of the liver and bone marrow. Biopsy DNA analyses of Mycobacterium bovis in the bone marrow, sputum, and blood were negative. His oxygen demand worsened drastically, and the ground-glass shadow expanded on the computed tomography scan. He was diagnosed with acute exacerbation of existing IP. We recommenced the antituberculosis drugs with steroid pulse therapy, but he died on day 35 because of respiratory failure. The autopsy revealed a diffuse appearance of multiple epithelioid cell granulomas with Langhans giant cells in multiple organs, although BCG was not evident.
CONCLUSIONS
CONCLUSIONS
We report the first case of acute exacerbation of chronic IP by BCG infection. This is also the first case of autopsy of a patient with acute exacerbation of existing IP induced by intravesical BCG treatment. Whether the trigger of acute IP exacerbation is infection or hypersensitivity to BCG is still controversial, because pathological evidence confirming BCG infection is lacking. Physicians who administer BCG against bladder cancer should be vigilant for acute exacerbation of IP.
Identifiants
pubmed: 32993546
doi: 10.1186/s12879-020-05396-7
pii: 10.1186/s12879-020-05396-7
pmc: PMC7523392
doi:
Substances chimiques
Antitubercular Agents
0
BCG Vaccine
0
Steroids
0
Types de publication
Case Reports
Journal Article
Review
Langues
eng
Sous-ensembles de citation
IM
Pagination
708Références
J Urol. 2006 Jun;175(6):2004-10
pubmed: 16697786
Nihon Kokyuki Gakkai Zasshi. 2008 Oct;46(10):803-7
pubmed: 19044030
BMC Pulm Med. 2014 Mar 05;14:35
pubmed: 24593234
Am Rev Respir Dis. 1987 Mar;135(3):763-5
pubmed: 3826901
Nefrologia. 2016 Nov - Dec;36(6):711-713
pubmed: 27445097
J Urol. 1992 Mar;147(3):695-7
pubmed: 1538462
J Urol. 1992 Mar;147(3):596-600
pubmed: 1538436
Nihon Hinyokika Gakkai Zasshi. 2011 Sep;102(5):691-5
pubmed: 22191278
N Engl J Med. 2015 Nov 26;373(22):2162-72
pubmed: 26605931
Medicine (Baltimore). 2014 Oct;93(17):236-54
pubmed: 25398060
Scott Med J. 1994 Apr;39(2):49-50
pubmed: 8720760
J Investig Allergol Clin Immunol. 2009;19(3):230-2
pubmed: 19610267
Arch Bronconeumol. 2016 Aug;52(8):445-6
pubmed: 26920491
Int Braz J Urol. 2004 Sep-Oct;30(5):400-2
pubmed: 15610574
Nihon Kyobu Shikkan Gakkai Zasshi. 1997 Dec;35(12):1383-8
pubmed: 9567086
Arch Intern Med. 1970 Apr;125(4):691-5
pubmed: 5437894
Thorax. 2012 Oct;67(10):933-4
pubmed: 22407891
Hinyokika Kiyo. 1999 Jul;45(7):493-5
pubmed: 10466068
J Urol. 1976 Aug;116(2):180-3
pubmed: 820877
Hinyokika Kiyo. 2017 Oct;63(10):427-430
pubmed: 29103257