How a bite on the cheek of a 29-year-old female patient with Crohn’s disease turned out to be tuberculosis

How a bite on the cheek of a 29-year-old female patient with Crohn’s disease turned out to be... Crohn’s disease, orofacial tuberculosis, infliximab, travel, Myanmar To the Editor: Tumour necrosis factor inhibitors [anti-TNFs] are associated with an increased risk of tuberculosis [TB] infection and re-activation.1 However, patients with inflammatory bowel disease who are stably adjusted to anti-TNFs are increasingly travelling on long-distance trips to the tropics and subtropics with enhanced exposure to infectious agents such as Mycobacterium tuberculosis.2 Here we report on the development of orofacial TB after a leisure trip to South East Asia. A 29-year-old German student with ileocolonic Crohn’s disease since 2008 and a drug history of prednisolone, budesonide, azathioprine and 6-mercaptopurine was switched to anti-TNFs [adalimumab, followed later by infliximab] in June 2014 after a negative T-SPOT®.TB test and inconspicuous chest X-ray. After a trip to Myanmar and Thailand for 4 weeks in April 2016 [during an anti-TNF-free period], the patient was hospitalized with a high fever and influenza B infection. TB diagnostic tests were not performed at this time. In November 2016, she reported a right-side cheek bite injury due to local mucosal inflammation. Over the next 6 months, the patient developed a progressive ulceration and swelling of the orofacial mucosa with spreading to the lower lip [Figure 1A] and pronounced cervical and submandibular swelling of the regional lymph nodes with a restriction of mouth opening. Figure 1. View largeDownload slide Figure 1. View largeDownload slide A biopsy taken from the cheek mucosa in May 2017 showed chronic and active focal erosive inflammation with subepithelial granulomas composed of macrophages, epithelioid histiocytes and multinucleated giant cells. This finding could correspond to an oral manifestation of Crohn’s disease,3 but PCR of the biopsy specimen and sputum revealed M. tuberculosis complex. Computed tomography showed flat compactions in both lungs and a calcified nodule in the right lung apex, as well as multiple perimandibular imprints in the soft tissue around teeth 42–47, accompanied by numerous enlarged regional lymph nodes [Figure 1B]. This suggested secondary TB of the cheek. Infliximab was immediately stopped; there has been no flare of Crohn’s disease as of now. Standard TB treatment [rifampicin, isoniazid, pyrazinamide, ethambutol] was initiated. Resistance to isoniazid and streptomycin was demonstrated in phenotypic resistance testing. Consequently, isoniazid was switched to levofloxacin.4 After 2 months of quadruple therapy, sputum conversion and complete healing of oral ulcers, treatment was de-escalated to rifampicin, ethambutol and levofloxacin for a further 7 months with a planned continuity phase. Orofacial TB usually occurs secondarily to open pulmonary TB and is a rare differential diagnosis of oral ulcers or nodules.5 To establish this diagnosis, mucosal biopsy with histopathological and microbiological work-up is necessary. This patient’s course demonstrates that TB diagnostics should be thoroughly repeated in patients with anti-TNFs if new oral ulcerations have occurred, especially after travel to a high-risk region. Funding This study had no funding source. Conflict of Interest None. Author Contributions HT, NT and CL cared for the patient and drafted the manuscript. All authors read and approved the final manuscript. Acknowledgments We thank the patient for consenting to the submission of this letter. References 1. Carpio D , Jauregui-Amezaga A , de Francisco R et al. ; GETECCU . Tuberculosis in anti-tumour necrosis factor-treated inflammatory bowel disease patients after the implementation of preventive measures: compliance with recommendations and safety of retreatment . J Crohns Colitis 2016 ; 10 : 1186 – 93 . Google Scholar CrossRef Search ADS PubMed 2. Soonawala D , van Eggermond AM , Fidder H , Visser LG . Pretravel preparation and travel-related morbidity in patients with inflammatory bowel disease . Inflamm Bowel Dis 2012 ; 18 : 2079 – 85 . Google Scholar CrossRef Search ADS PubMed 3. Singh B , Kedia S , Konijeti G et al. Extraintestinal manifestations of inflammatory bowel disease and intestinal tuberculosis: frequency and relation with disease phenotype . Indian J Gastroenterol 2015 ; 34 : 43 – 50 . Google Scholar CrossRef Search ADS PubMed 4. Schaberg T , Bauer T , Brinkmann F et al. Tuberculosis guideline for adults – guideline for diagnosis and treatment of tuberculosis including LTBI testing and treatment of the german central committee (DZK) and the German Respiratory Society (DGP) . Pneumologie 2017 ; 71 : 325 – 97 . Google Scholar CrossRef Search ADS PubMed 5. Bansal R , Jain A , Mittal S . Orofacial tuberculosis: clinical manifestations, diagnosis and management . J Family Med Prim Care 2015 ; 4 : 335 – 41 . Google Scholar CrossRef Search ADS PubMed Copyright © 2018 European Crohn’s and Colitis Organisation (ECCO). Published by Oxford University Press. All rights reserved. For permissions, please email: journals.permissions@oup.com This article is published and distributed under the terms of the Oxford University Press, Standard Journals Publication Model (https://academic.oup.com/journals/pages/about_us/legal/notices) http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Crohn's and Colitis Oxford University Press

How a bite on the cheek of a 29-year-old female patient with Crohn’s disease turned out to be tuberculosis

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Publisher
Elsevier Science
Copyright
Copyright © 2018 European Crohn’s and Colitis Organisation (ECCO). Published by Oxford University Press. All rights reserved. For permissions, please email: journals.permissions@oup.com
ISSN
1873-9946
eISSN
1876-4479
D.O.I.
10.1093/ecco-jcc/jjy013
Publisher site
See Article on Publisher Site

Abstract

Crohn’s disease, orofacial tuberculosis, infliximab, travel, Myanmar To the Editor: Tumour necrosis factor inhibitors [anti-TNFs] are associated with an increased risk of tuberculosis [TB] infection and re-activation.1 However, patients with inflammatory bowel disease who are stably adjusted to anti-TNFs are increasingly travelling on long-distance trips to the tropics and subtropics with enhanced exposure to infectious agents such as Mycobacterium tuberculosis.2 Here we report on the development of orofacial TB after a leisure trip to South East Asia. A 29-year-old German student with ileocolonic Crohn’s disease since 2008 and a drug history of prednisolone, budesonide, azathioprine and 6-mercaptopurine was switched to anti-TNFs [adalimumab, followed later by infliximab] in June 2014 after a negative T-SPOT®.TB test and inconspicuous chest X-ray. After a trip to Myanmar and Thailand for 4 weeks in April 2016 [during an anti-TNF-free period], the patient was hospitalized with a high fever and influenza B infection. TB diagnostic tests were not performed at this time. In November 2016, she reported a right-side cheek bite injury due to local mucosal inflammation. Over the next 6 months, the patient developed a progressive ulceration and swelling of the orofacial mucosa with spreading to the lower lip [Figure 1A] and pronounced cervical and submandibular swelling of the regional lymph nodes with a restriction of mouth opening. Figure 1. View largeDownload slide Figure 1. View largeDownload slide A biopsy taken from the cheek mucosa in May 2017 showed chronic and active focal erosive inflammation with subepithelial granulomas composed of macrophages, epithelioid histiocytes and multinucleated giant cells. This finding could correspond to an oral manifestation of Crohn’s disease,3 but PCR of the biopsy specimen and sputum revealed M. tuberculosis complex. Computed tomography showed flat compactions in both lungs and a calcified nodule in the right lung apex, as well as multiple perimandibular imprints in the soft tissue around teeth 42–47, accompanied by numerous enlarged regional lymph nodes [Figure 1B]. This suggested secondary TB of the cheek. Infliximab was immediately stopped; there has been no flare of Crohn’s disease as of now. Standard TB treatment [rifampicin, isoniazid, pyrazinamide, ethambutol] was initiated. Resistance to isoniazid and streptomycin was demonstrated in phenotypic resistance testing. Consequently, isoniazid was switched to levofloxacin.4 After 2 months of quadruple therapy, sputum conversion and complete healing of oral ulcers, treatment was de-escalated to rifampicin, ethambutol and levofloxacin for a further 7 months with a planned continuity phase. Orofacial TB usually occurs secondarily to open pulmonary TB and is a rare differential diagnosis of oral ulcers or nodules.5 To establish this diagnosis, mucosal biopsy with histopathological and microbiological work-up is necessary. This patient’s course demonstrates that TB diagnostics should be thoroughly repeated in patients with anti-TNFs if new oral ulcerations have occurred, especially after travel to a high-risk region. Funding This study had no funding source. Conflict of Interest None. Author Contributions HT, NT and CL cared for the patient and drafted the manuscript. All authors read and approved the final manuscript. Acknowledgments We thank the patient for consenting to the submission of this letter. References 1. Carpio D , Jauregui-Amezaga A , de Francisco R et al. ; GETECCU . Tuberculosis in anti-tumour necrosis factor-treated inflammatory bowel disease patients after the implementation of preventive measures: compliance with recommendations and safety of retreatment . J Crohns Colitis 2016 ; 10 : 1186 – 93 . Google Scholar CrossRef Search ADS PubMed 2. Soonawala D , van Eggermond AM , Fidder H , Visser LG . Pretravel preparation and travel-related morbidity in patients with inflammatory bowel disease . Inflamm Bowel Dis 2012 ; 18 : 2079 – 85 . Google Scholar CrossRef Search ADS PubMed 3. Singh B , Kedia S , Konijeti G et al. Extraintestinal manifestations of inflammatory bowel disease and intestinal tuberculosis: frequency and relation with disease phenotype . Indian J Gastroenterol 2015 ; 34 : 43 – 50 . Google Scholar CrossRef Search ADS PubMed 4. Schaberg T , Bauer T , Brinkmann F et al. Tuberculosis guideline for adults – guideline for diagnosis and treatment of tuberculosis including LTBI testing and treatment of the german central committee (DZK) and the German Respiratory Society (DGP) . Pneumologie 2017 ; 71 : 325 – 97 . Google Scholar CrossRef Search ADS PubMed 5. Bansal R , Jain A , Mittal S . Orofacial tuberculosis: clinical manifestations, diagnosis and management . J Family Med Prim Care 2015 ; 4 : 335 – 41 . Google Scholar CrossRef Search ADS PubMed Copyright © 2018 European Crohn’s and Colitis Organisation (ECCO). Published by Oxford University Press. All rights reserved. For permissions, please email: journals.permissions@oup.com This article is published and distributed under the terms of the Oxford University Press, Standard Journals Publication Model (https://academic.oup.com/journals/pages/about_us/legal/notices)

Journal

Journal of Crohn's and ColitisOxford University Press

Published: Feb 1, 2018

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