respiratory medicine case reports · 2017. 1. 9. · includes malignant lesions, sarcoidosis,...

4
Case report Cerebral tuberculomas e A clinical challenge Regina Monteiro a, * , José Carlos Carneiro a , Claúdia Costa b , Raquel Duarte a, c, d a Pulmonology Department, Hospital Center of Vila Nova de Gaia/Espinho (CHVNG/E), Porto, Portugal b Internal Medicine Department, CHVNG/E, Portugal c Pulmonology Diagnostic Center and Multi-Drug Resistant Tuberculosis (MDR TB) Reference Center for the North Region of Portugal, Vila Nova de Gaia, Porto, Portugal d Faculty of Medicine, University of Porto, Porto, Portugal article info Article history: Received 20 November 2012 Received in revised form 2 April 2013 Accepted 16 April 2013 Keywords: Cerebral tuberculomas Disseminated tuberculosis abstract Cerebral tuberculomas are a rare and serious form of tuberculosis (TB) due to the haematogenous spread of Mycobacterium Tuberculosis (MT). Symptoms and radiologic features are nonspecic, leading some- times to misdiagnosis. Anti-TB drugs are essential for the successful treatment of cerebral tuberculomas but there is no agreement regarding the duration of therapy. The authors present a case of a 55 years old male, presented to the emergency room with sudden onset of diplopia. Cerebral computerized tomography revealed multiple brain lesions, with contrast enhancement and peri-lesional oedema. The patient was HIV negative and because of previous malig- nancy the rst suspicion was metastatic disease. Cultural exam of the bronchial wash showed MT sen- sitive to all rst-line drugs. The patient started antituberculosis treatment with 4 drugs (HRZE) for 2 months, followed by maintenance therapy (HR). Treatment was prolonged for 24 months because at 12th and 18th months of treatment one of the brain lesions, although signicantly smaller, still showed contrast enhancement. Even though it is not clear if contrast enhancement lesions represent active lesions or just inam- mation, continuing treatment until total resolution of the tuberculomas is probably prudent. Ó 2013 Elsevier Ltd. 1. Background Central nervous system (CNS) tuberculosis (TB) is a serious form of TB, due to haematogenous spread of Mycobacterium tuberculosis (MT). Manifesting as meningitis, cerebritis and tuberculous ab- scesses or tuberculomas, it occurs in approximately 1% of all pa- tients with TB, affecting disproportionately children and immunosuppressed patients. Other risk factors include malnutri- tion, alcoholism and malignancies. 1 Intracranial tuberculomas are the least common presentation of CNS TB, found in 1% of these patients. 2 They are multiple in only 15%e33% of the cases. 3 Tuberculomas often present with symptoms and signs of focal neurological decit without evidence of systemic disease. 4 The radiologic features are also nonspecic and differential diagnosis includes malignant lesions, sarcoidosis, pyogenic abscess, toxo- plasmosis and cysticercosis. 1,4,5 It is universally accepted that anti-TB drugs are essential for the successful treatment of intracranial tuberculomas but there is no agreement regarding the duration of therapy. 3,6,7 2. Case report A 55 years old white male, ex-smoker for 19 years (6 pack- years), working as an air conditioning installer, came to the Emer- gency Room in May 2010 with sudden onset of diplopia. He also complained of headache, loss of weight (about 10% over the last month) and of back pain over the last year, that he thought to be related with an accident. He denied any respiratory symptom or other. His medical history included a right colon cancer, treated with surgery and chemotherapy 20 years ago (he had abandoned follow- up after 10 years), and pulmonary TB 4 years ago, treated with 2 months of Isoniazid, Rifampicin, Pyrazinamide and Ethambutol (HRZE) follow by 4 months of Isoniazid and Rifampicin (HR) under directly observed therapy (DOT) e after that he went abroad and missed follow-up. He was not on any medication and denied any allergies. On examination the patient was underweight and walked with difculty because of the diplopia. His physical examination was * Corresponding author. E-mail address: [email protected] (R. Monteiro). Contents lists available at SciVerse ScienceDirect Respiratory Medicine Case Reports journal homepage: www.elsevier.com/locate/rmcr 2213-0071 Ó 2013 Elsevier Ltd. http://dx.doi.org/10.1016/j.rmcr.2013.04.003 Respiratory Medicine Case Reports 9 (2013) 34e37 Open access under CC BY-NC-ND license. Open access under CC BY-NC-ND license.

Upload: others

Post on 27-Jan-2021

1 views

Category:

Documents


0 download

TRANSCRIPT

  • at SciVerse ScienceDirect

    Respiratory Medicine Case Reports 9 (2013) 34e37

    Contents lists available

    Respiratory Medicine Case Reports

    journal homepage: www.elsevier .com/locate /rmcr

    Case report

    Cerebral tuberculomas e A clinical challenge

    Regina Monteiro a,*, José Carlos Carneiro a, Claúdia Costa b, Raquel Duarte a,c,d

    a Pulmonology Department, Hospital Center of Vila Nova de Gaia/Espinho (CHVNG/E), Porto, Portugalb Internal Medicine Department, CHVNG/E, Portugalc Pulmonology Diagnostic Center and Multi-Drug Resistant Tuberculosis (MDR TB) Reference Center for the North Region of Portugal, Vila Nova de Gaia, Porto, Portugald Faculty of Medicine, University of Porto, Porto, Portugal

    a r t i c l e i n f o

    Article history:Received 20 November 2012Received in revised form2 April 2013Accepted 16 April 2013

    Keywords:Cerebral tuberculomasDisseminated tuberculosis

    * Corresponding author.E-mail address: [email protected] (R. Mon

    2213-0071 � 2013 Elsevier Ltd.http://dx.doi.org/10.1016/j.rmcr.2013.04.003

    Open access under CC BY

    a b s t r a c t

    Cerebral tuberculomas are a rare and serious form of tuberculosis (TB) due to the haematogenous spreadof Mycobacterium Tuberculosis (MT). Symptoms and radiologic features are nonspecific, leading some-times to misdiagnosis. Anti-TB drugs are essential for the successful treatment of cerebral tuberculomasbut there is no agreement regarding the duration of therapy.

    The authors present a case of a 55 years old male, presented to the emergency roomwith sudden onsetof diplopia. Cerebral computerized tomography revealed multiple brain lesions, with contrastenhancement and peri-lesional oedema. The patient was HIV negative and because of previous malig-nancy the first suspicion was metastatic disease. Cultural exam of the bronchial wash showed MT sen-sitive to all first-line drugs. The patient started antituberculosis treatment with 4 drugs (HRZE) for 2months, followed by maintenance therapy (HR). Treatment was prolonged for 24 months because at 12thand 18th months of treatment one of the brain lesions, although significantly smaller, still showedcontrast enhancement.

    Even though it is not clear if contrast enhancement lesions represent active lesions or just inflam-mation, continuing treatment until total resolution of the tuberculomas is probably prudent.

    � 2013 Elsevier Ltd. Open access under CC BY-NC-ND license.

    1. Background

    Central nervous system (CNS) tuberculosis (TB) is a serious formof TB, due to haematogenous spread ofMycobacterium tuberculosis(MT). Manifesting as meningitis, cerebritis and tuberculous ab-scesses or tuberculomas, it occurs in approximately 1% of all pa-tients with TB, affecting disproportionately children andimmunosuppressed patients. Other risk factors include malnutri-tion, alcoholism and malignancies.1

    Intracranial tuberculomas are the least common presentation ofCNS TB, found in 1% of these patients.2 They are multiple in only15%e33% of the cases.3

    Tuberculomas often present with symptoms and signs of focalneurological deficit without evidence of systemic disease.4 Theradiologic features are also nonspecific and differential diagnosisincludes malignant lesions, sarcoidosis, pyogenic abscess, toxo-plasmosis and cysticercosis.1,4,5

    teiro).

    -NC-ND license.

    It is universally accepted that anti-TB drugs are essential for thesuccessful treatment of intracranial tuberculomas but there is noagreement regarding the duration of therapy.3,6,7

    2. Case report

    A 55 years old white male, ex-smoker for 19 years (6 pack-years), working as an air conditioning installer, came to the Emer-gency Room in May 2010 with sudden onset of diplopia. He alsocomplained of headache, loss of weight (about 10% over the lastmonth) and of back pain over the last year, that he thought to berelated with an accident. He denied any respiratory symptom orother.

    His medical history included a right colon cancer, treated withsurgery and chemotherapy 20 years ago (he had abandoned follow-up after 10 years), and pulmonary TB 4 years ago, treated with 2months of Isoniazid, Rifampicin, Pyrazinamide and Ethambutol(HRZE) follow by 4 months of Isoniazid and Rifampicin (HR) underdirectly observed therapy (DOT) e after that he went abroad andmissed follow-up. He was not on any medication and denied anyallergies.

    On examination the patient was underweight and walked withdifficulty because of the diplopia. His physical examination was

    mailto:[email protected]/science/journal/22130071http://www.elsevier.com/locate/rmcrhttp://dx.doi.org/10.1016/j.rmcr.2013.04.003http://dx.doi.org/10.1016/j.rmcr.2013.04.003http://dx.doi.org/10.1016/j.rmcr.2013.04.003http://creativecommons.org/licenses/by-nc-nd/3.0/http://creativecommons.org/licenses/by-nc-nd/3.0/

  • R. Monteiro et al. / Respiratory Medicine Case Reports 9 (2013) 34e37 35

    unremarkable besides an ophthalmoplegia. Cerebral computerizedtomography (CT) revealed multiple brain lesions, with contrastenhancement and peri-lesional oedema.

    He was admitted for investigation and treatment. Brain mag-netic resonance imaging (MRI) confirmed 4 space occupying le-sions with central necrosis, irregular outlines with peripheralcontrast enhancing and moderate peri-lesional oedema (Fig. 1).

    Fig. 1-. Brain MR before treatment: 4 space occupying lesions in the brain with central necroedema.

    Lumbar puncture was performed but was not diagnostic (cere-brospinal fluid was negative for malignant cells or microorgan-isms). Biochemical analyses such as nucleic acid amplification oradenosine deaminase measurement were not performed. Chest X-ray was considered normal. Serologies to human immunodefi-ciency virus (HIV) and toxoplasmosis were negative. Thoracic andabdominal CT revealed peri-centimetric mediastinal lymph nodes,

    osis, irregular outlines with peripheral contrast enhancing and moderate peri-lesional

  • Fig. 2-. Thoracic CT before treatment: peri-centimetric mediastinal lymph nodes;bilateral lung micronodules and a small condensation area in the right middle lobewith discrete air bronchogram; lytic lesion at the left 8th rib-vertebral joint.

    Fig. 4-. First and last brain MR (after 24 months of treatment): total resolution of thetuberculomas.

    R. Monteiro et al. / Respiratory Medicine Case Reports 9 (2013) 34e3736

    bilateral pulmonarymicronodules, a small condensation area in theright middle lobe with discrete air bronchogram and a lytic lesionat the 8th left rib-vertebral joint (Fig 2). Bronchoscopy showed nochanges in the bronchial tree besides anthracotic plates. Therewereno malignant cells in the bronchial wash and direct exam wasnegative for TB. Endoscopies did not showany sign ofmalignancy. Apositron emission tomography (PET) was performed and revealedabnormal enhancement in multiple small foci in both lung fields,mediastinal and abdominal lymph nodes, bone lesions (3rd and 8thleft ribs and right iliac wing), both adrenals and brain (Fig. 3). Thebiopsy of the rib lesion presented only epithelioid granulomas. TBwas confirmed by cultural exam of the bronchial wash that showedMT sensitive to all first-line drugs.

    Fig. 3-. PET scan: abnormal enhancement in multiple small foci in both lung field

    The patient had already started corticosteroids (prednisolone1 mg/kg) at admission for cerebral oedema and antituberculoustreatment was added when TB was found. The patient was dis-charged but continued treatment with 4 drugs (HRZE) in form of

    s; lymph nodes and bone lesions (3rd and 8th left ribs and right iliac wing).

  • R. Monteiro et al. / Respiratory Medicine Case Reports 9 (2013) 34e37 37

    DOT and completed 8 weeks of corticosteroids with clinicalimprovement. After 2 months the patient was completely asymp-tomatic and treatment was reduced to maintenance therapy with 2drugs (HR).

    Radiologic revaluations were made at 6th, 12th, 18th and 24thmonths after the diagnosis and beginning of the treatment. Thelung micronodules and mediastinal lymph nodes remained stableand some calcified. Bone lesions also stabilized. The brain MRI atthe 18th month of treatment one of the lesions, although signifi-cantly smaller still showed contrast enhancement so treatment wasprolonged for 24 months. In the last brain MRI no contrastenhancement lesions were seen (Fig. 4).

    3. Discussion

    Due to its rarity, nonspecific symptoms and radiological find-ings, intracranial tuberculomas remain a clinical challenge.

    Misdiagnosis of tuberculomas as malignant diseases have beendescribed in literature.4,5,7,8 Our patient was HIV negative, had amedical history of malignancy and presented with multiple brainlesions so the first suspicion was also metastatic disease. Tuber-culomas must however be always included in differential diagnosisof cerebral space occupying lesions.

    As the patient missed follow-up after his first TB infection thismay be the case of a reactivation but reinfection is also a possibilityand genotype was not performed.

    Regarding treatment, the Center for Disease Control and Pre-vention recommends 12 months of treatment for CNS TB when theMT strain is sensitive to all drugs.9 However numerous variablescan affect the response of the disease to therapy and it has beensuggested that treatment duration should be tailored to theradiological response.6 After 12 months of treatment more thantwo-thirds of the patients still have contrast enhancing lesions.Although it is not clear if this represents an active lesion or justinflammation, continuing treatment is probably prudent. Totalresolution of the tuberculoma is observed when scans demonstrateno enhancing lesions or only an area of calcification.6

    Systemic corticosteroids as adjuvant therapy are indicatedwhenthere is peri-lesional oedema or paradoxical progression duringtreatment. Surgical intervention may be necessary in situationswith acute complications or when the diagnosis is notensured.2,3,6,10

    Acknowledgements

    We would like to thank the careful editing of Flávio Monteiro,Papworth Hospital (Cambridge, England).

    References

    1. Rock RB, Olin M, Baker CA, Molitor TW, Peterson PK. Central nervous systemtuberculosis: pathogenesis and clinical aspects. Clinical Microbiology Reviews2008;21(2):243e61.

    2. Pimentel MLV, Alves SMV, Novis SAP, Brandão RZ, Neto EB. Intracranialtuberculomas developing during treatment of pulmonary tuberculosis: casereport. Arq Neuropsiquiatri 2000;58(2-B):572e7.

    3. Hejazi N, Hassler W. Multiple intracranial tuberculomas with atypical responseto tuberculostatic chemotherapy: literature review and a case report. Infection1997;25(4):41e6.

    4. Sahaiu-Srivastava S, Jones B. Brainstem tuberculoma in the immunocompetent:case report and literature review. Clinical Neurology and Neurosurgery2008;110:302e4.

    5. Ogbole GI, Bassey OS, Okolo CA, Ukperi SO, Ogunseyinde AO. Testiculartuberculosis presenting with metastatic intracranial tuberculomas: a casereport. Journal of Medical Case Reports 2011;5:100.

    6. Poonnoose SI, Rajshekhar V. Rate of resolution of histologically verified intra-cranial tuberculomas. Neurosurgery 2003;53:873e9.

    7. Suslu HT, Bozbuga M, Bayindir C. Cerebral tuberculoma mimicking high gradeglial tumor. Turkish Neurosurgery 2011;21(3):427e9.

    8. Álvarez-Salgado JA, Ruiz-Ginés JA, Gonzales-Sejas AG, Belinchón-Diego JM,Fernández-Mesa FGL, Lope-Llorca AR. Tuberculoma intracraneal simulandoneoplasia maligna. Caso clinico y revision de la literatura. Neurocirugía2011;22:600e4.

    9. Centers for Disease Control and Prevention. Treatment of Tuberculosis,American Thoracic Society, CDC, and Infectious Diseases Society of America.MMWR 2003; 52 (No. RR-11).

    10. Jain SK, Kwon P, Moss WJ. Management and outcomes of intracranial tuber-culomas developing during antituberculous therapy: case report and review.Clinical Pediatrics 2005;44:443e50.

    http://refhub.elsevier.com/S2213-0071(13)00017-8/sref1http://refhub.elsevier.com/S2213-0071(13)00017-8/sref1http://refhub.elsevier.com/S2213-0071(13)00017-8/sref1http://refhub.elsevier.com/S2213-0071(13)00017-8/sref1http://refhub.elsevier.com/S2213-0071(13)00017-8/sref2http://refhub.elsevier.com/S2213-0071(13)00017-8/sref2http://refhub.elsevier.com/S2213-0071(13)00017-8/sref2http://refhub.elsevier.com/S2213-0071(13)00017-8/sref2http://refhub.elsevier.com/S2213-0071(13)00017-8/sref3http://refhub.elsevier.com/S2213-0071(13)00017-8/sref3http://refhub.elsevier.com/S2213-0071(13)00017-8/sref3http://refhub.elsevier.com/S2213-0071(13)00017-8/sref3http://refhub.elsevier.com/S2213-0071(13)00017-8/sref4http://refhub.elsevier.com/S2213-0071(13)00017-8/sref4http://refhub.elsevier.com/S2213-0071(13)00017-8/sref4http://refhub.elsevier.com/S2213-0071(13)00017-8/sref4http://refhub.elsevier.com/S2213-0071(13)00017-8/sref5http://refhub.elsevier.com/S2213-0071(13)00017-8/sref5http://refhub.elsevier.com/S2213-0071(13)00017-8/sref5http://refhub.elsevier.com/S2213-0071(13)00017-8/sref6http://refhub.elsevier.com/S2213-0071(13)00017-8/sref6http://refhub.elsevier.com/S2213-0071(13)00017-8/sref6http://refhub.elsevier.com/S2213-0071(13)00017-8/sref7http://refhub.elsevier.com/S2213-0071(13)00017-8/sref7http://refhub.elsevier.com/S2213-0071(13)00017-8/sref7http://refhub.elsevier.com/S2213-0071(13)00017-8/sref8http://refhub.elsevier.com/S2213-0071(13)00017-8/sref8http://refhub.elsevier.com/S2213-0071(13)00017-8/sref8http://refhub.elsevier.com/S2213-0071(13)00017-8/sref8http://refhub.elsevier.com/S2213-0071(13)00017-8/sref8http://refhub.elsevier.com/S2213-0071(13)00017-8/sref9http://refhub.elsevier.com/S2213-0071(13)00017-8/sref9http://refhub.elsevier.com/S2213-0071(13)00017-8/sref9http://refhub.elsevier.com/S2213-0071(13)00017-8/sref9

    Cerebral tuberculomas – A clinical challenge1. Background2. Case report3. DiscussionAcknowledgementsReferences