a case report of atypical sarcoidosis misdiagnosed as ...tuberculosis is one of the common chronic...

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Case report A case report of atypical sarcoidosis misdiagnosed as tuberculosis Jing Yu a , Jing Shen a , Liang Wang a , Ruiping Zhu b , Jianlin Wu a, * a Department of Radiology, Affiliated Zhongshan Hospital of Dalian University, Dalian 116001, China b Department of Pathology, Affiliated Zhongshan Hospital of Dalian University, Dalian 116001, China Received 4 November 2015; revised 23 November 2015; accepted 15 December 2015 Available online 7 January 2016 Abstract Tuberculosis is one of the common chronic infectious diseases of respiratory system in China, and presents high morbidity, high drug resistance and the specific imaging characteristics. This paper focused on a case report of atypical sarcoidosis which was misdiagnosed as tuberculosis before operation. CT imaging was discussed by contrast with clinical manifestations, aiming to survey the imaging differences between atypical sarcoidosis and tuberculosis and improve the clinical differential diagnosis. © 2016 Beijing You’an Hospital affiliated to Capital Medical University. Production and hosting by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). Keywords: Tuberculosis; Atypical sarcoidosis; Medical imaging 1. Introduction Tuberculosis is one of the widespread, infectious diseases, which was caused by various strains of mycobacteria, typi- cally Mycobacterium tuberculosis. China is the second largest country in the world, in terms of morbidity of tuberculosis. As reported in 2000 by literature of the epidemiological investi- gation, approximately 550 million people have suffered from Mycobacterium tuberculosis infection, with active cases of more than 4.5 million, of which total acquired drug resistance rate had reached up to 46.5% [1]. Due to the aging and increasing number of tuberculosis cases, imaging of some of these infections showed atypical. Similarly, sarcoidosis always involved lung and mediastinal lymph nodes, showing non- caseous granulomas. For this reason, sarcoidosis sometimes would be misdiagnosed as tuberculosis. However, this misdi- agnose should be avoid for the treatments of these two dis- eases are definitely different. For the purpose of surveying the imaging differences between atypical sarcoidosis and tuberculosis, and improving the clinical differential diagnosis there between, this paper focused on a case report of atypical sarcoidosis which was misdiagnosed as tuberculosis before operation, and CT imaging was discussed by contrast with clinical manifestations. 2. Case report A 49 years old, female patient was hospitalized for abdominal discomfort, low grade fever (with the highest body temperature to be 37.8 C) and long time cough lasting for about half a month. The cough presented as irritating cough accompanied with nausea, but without phlegm and emesis. It is known that the patient is healthy ordinarily and no obvious inducement was found for these above mentioned symptoms. Therefore, a series of regular examinations were carried out, including laboratory examination, bronchoscopy, CT scan- ning, and biopsy and pathological diagnosis. The regular laboratory examination was showed in Table 1. Additionally, plenty of lymphocytes and less mesothelial cells were detected in pleural effusions, without finding heterocyst. For the bronchoscopy, no obstruction was visible within the left principal bronchus and its branches. No hyperaemia, edema and erosion were found in the lumen mucous * Corresponding author. E-mail address: [email protected] (J. Wu). Peer review under responsibility of Beijing You'an Hospital affiliated to Capital Medical University. HOSTED BY Available online at www.sciencedirect.com ScienceDirect Radiology of Infectious Diseases 3 (2016) 40e43 www.elsevier.com/locate/jrid http://dx.doi.org/10.1016/j.jrid.2016.01.005 2352-6211/© 2016 Beijing You’an Hospital affiliated to Capital Medical University. Productionand hosting by Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

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Page 1: A case report of atypical sarcoidosis misdiagnosed as ...Tuberculosis is one of the common chronic infectious diseases of respiratory system in China, and presents high morbidity,

HOSTED BY Available online at www.sciencedirect.com

ScienceDirect

Radiology of Infectious Diseases 3 (2016) 40e43

www.elsevier.com/locate/jrid

Case report

A case report of atypical sarcoidosis misdiagnosed as tuberculosis

Jing Yu a, Jing Shen a, Liang Wang a, Ruiping Zhu b, Jianlin Wu a,*a Department of Radiology, Affiliated Zhongshan Hospital of Dalian University, Dalian 116001, Chinab Department of Pathology, Affiliated Zhongshan Hospital of Dalian University, Dalian 116001, China

Received 4 November 2015; revised 23 November 2015; accepted 15 December 2015

Available online 7 January 2016

Abstract

Tuberculosis is one of the common chronic infectious diseases of respiratory system in China, and presents high morbidity, high drugresistance and the specific imaging characteristics. This paper focused on a case report of atypical sarcoidosis which was misdiagnosed astuberculosis before operation. CT imaging was discussed by contrast with clinical manifestations, aiming to survey the imaging differencesbetween atypical sarcoidosis and tuberculosis and improve the clinical differential diagnosis.© 2016 Beijing You’an Hospital affiliated to Capital Medical University. Production and hosting by Elsevier B.V. This is an open access articleunder the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

Keywords: Tuberculosis; Atypical sarcoidosis; Medical imaging

1. Introduction

Tuberculosis is one of the widespread, infectious diseases,which was caused by various strains of mycobacteria, typi-cally Mycobacterium tuberculosis. China is the second largestcountry in the world, in terms of morbidity of tuberculosis. Asreported in 2000 by literature of the epidemiological investi-gation, approximately 550 million people have suffered fromMycobacterium tuberculosis infection, with active cases ofmore than 4.5 million, of which total acquired drug resistancerate had reached up to 46.5% [1]. Due to the aging andincreasing number of tuberculosis cases, imaging of some ofthese infections showed atypical. Similarly, sarcoidosis alwaysinvolved lung and mediastinal lymph nodes, showing non-caseous granulomas. For this reason, sarcoidosis sometimeswould be misdiagnosed as tuberculosis. However, this misdi-agnose should be avoid for the treatments of these two dis-eases are definitely different. For the purpose of surveying theimaging differences between atypical sarcoidosis and

* Corresponding author.

E-mail address: [email protected] (J. Wu).

Peer review under responsibility of Beijing You'an Hospital affiliated to

Capital Medical University.

http://dx.doi.org/10.1016/j.jrid.2016.01.005

2352-6211/© 2016 Beijing You’an Hospital affiliated to Capital Medical University

the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

tuberculosis, and improving the clinical differential diagnosisthere between, this paper focused on a case report of atypicalsarcoidosis which was misdiagnosed as tuberculosis beforeoperation, and CT imaging was discussed by contrast withclinical manifestations.

2. Case report

A 49 years old, female patient was hospitalized forabdominal discomfort, low grade fever (with the highest bodytemperature to be 37.8 �C) and long time cough lasting forabout half a month. The cough presented as irritating coughaccompanied with nausea, but without phlegm and emesis. Itis known that the patient is healthy ordinarily and no obviousinducement was found for these above mentioned symptoms.Therefore, a series of regular examinations were carried out,including laboratory examination, bronchoscopy, CT scan-ning, and biopsy and pathological diagnosis.

The regular laboratory examination was showed in Table 1.Additionally, plenty of lymphocytes and less mesothelial cellswere detected in pleural effusions, without finding heterocyst.

For the bronchoscopy, no obstruction was visible within theleft principal bronchus and its branches. No hyperaemia,edema and erosion were found in the lumen mucous

. Production and hosting by Elsevier B.V. This is an open access article under

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Table 1

Results of laboratory examination.

Items Results

Leucocyte 9.7 � 109/l

Granulocyte occupancy 87.9%

Acid-fast bacilli test eErythrocyte sedimentation rate 13 mm/h

CA125 116.0 U/ml

Total protein 45.7 g/l

Rivalta test þ

41J. Yu et al. / Radiology of Infectious Diseases 3 (2016) 40e43

membrane. No stenosis and neoplasm were visible within thelumen. Mucous membrane was normal in the right upper lobebronchus while mild hyperemia was found in the mucosa ofright lower lobe bronchus, and the corresponding lumenshowed with mild distortion and obvious neoplasm develop-ment. It was shown with bronchial ridge hypertrophy andenlargement. There was apparent stenosis in the lateral bron-chus of the middle-lobe, showing with white moss-like se-cretions. The bronchial mucosa was a bit brittle and wouldbleed when touched.

For regular CT examination, irregular pulmonary consoli-dation was presented in the middle and lower lobes around theright hilum, with mean CT value of 44HU; bronchus shadowwith narrowed lumen was visible inside the lumen, andswelling lymph node shadow was likely to be visible in themediastinum; and, a small amount of pleural effusion waspresented in the right thorax, while no obvious abnormity wasvisible in the left one (Fig. 1). For the contrast enhanced CTscanning, equally enhancing lesions were presented inside thelung and around pulmonary hilus, with the mean CT value of98HU during arterial phase (Fig. 2A), and local stenosisoccurred inside the pulmonary artery; by contrast, during thevenous phase, the mean CT value was 98 HU (Fig. 2B);multiple lymphadenectasis presented as equal enhancementswere found within the right pulmonary hilus and mediastinum(behind the left atrium), while multiple small nodular shadowswere visible in the right pleura; and no obvious lymphade-nectasis was presented in the left pulmonary hilus.

Fig. 1. Regular CT scan

2.1. Biopsy and pathological examination

Biopsy was carried out for the right pleura under thoraco-scope, and it was shown that about 500 ml pale yellow effu-sion was presented in the right thorax, and there were alsopleural edema, thickening and adhesion in the right chest.Unequal multiple small nodules with diameters from about0.3 cm to about 0.5 cm was found on the right side of thepleura, mediastinum, pericardium and pulmonary surface, andlump appeared around the right pulmonary hilus (Fig. 3). Forfurther confirmation, the pathological diagnosis was carriedout, which showed granulomatous inflammation accompaniedwith small necrosis, and the possibility of tuberculosis was notexcluded. The results of acid fast stain were showed in Fig. 3.Ultrasonic bronchial endoscopy (Ebus) was carried out undergeneral anesthesia, and the lymph node on the punctured rightbronchus (region 4R) and subcarinal (region 7) lymph nodewere sent for biopsy, of which the results showed noncaseatinggranulomas (Fig. 4).

3. Discussion

Sarcoidosis is a systemic disease which is characterized bythe presence of noncaseating granulomas, with the thoracicinvolvement occurs in about 80%e90% of affected individuals[2,3]. Granulomas tend to involve pleural lymphatic, inter-lobular septa, bronchia and subpleural region. Pathologically,sarcoidosis is characterized by the gathering of plenty of activemacrophage, and T lymphocyte, Th1 cytokines, interferon-gand tumor necrosis factor were released for generating gran-uloma. The cause of sarcoidosis is still unknown, but commonpresented in 20e40 year old individuals and more in malepatients, with slight symptom or no symptom (occupy about30%e50%). Most patients would be self-healing or healedafter hormone therapy, however, about 20%e25% woulddevelop to irreversible pulmonary fibrosis, leading to respira-tory function failure, or even death (about 5%e10%) [4].

CTwas thought to be an accurate and effective examinationfor sarcoidosis and mediastinal diseases. The typical mani-festation included bilateral hilar lymphadenophathy

ning near the hilar.

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Fig. 2. Contrast-enhanced CT scanning near the hilar. A. arterial phase, B. venous phase.

Fig. 3. Pleural biopsy showing granulomatous nodules, small necrosis and acid fast stain (�) (HE � 100, �400).

Fig. 4. Biopsy for the lymph node on the punctured right bronchus (4R region) and subcarinal (region 7) lymph node. Granulomatous nodules composed of

epithelioid cell, multinuclear giant cell and lymphocytes were seen without caseous necrosis (HE � 100).

42 J. Yu et al. / Radiology of Infectious Diseases 3 (2016) 40e43

symmetrically accompanied with mediastinal lymphade-nectasis near both the pulmonary hiluses, which could bepresented in about 80%e90% of these patients [5]. Theintrapulmonary lesions included alveolar nodules, granulo-matous nodules, pulmonary fibrosis and so on [6]. The alve-olar nodules were mainly alveolar infiltrative lesions,presenting as ill-defined plaques and air bronchogram whichwas usually in upper lobes. The granulomatous nodules were

small nodules with regular shape and primary diameter of2e5 mm, which commonly distributed along the broncho-vascular bundle, interlobular septal, interlobar fissure, andsubpleural lymphatic network, and these features were moreobvious in the middle lung field and subpleural lung. At a laterstage, the patients with pulmonary fibrosis would presentbronchial distortion, honeycomb-like shadow, and diffusedlinear opacities.

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43J. Yu et al. / Radiology of Infectious Diseases 3 (2016) 40e43

Atypical sarcoidosis might present as no bilateral pulmo-nary hilar lymphadenopathy or only unilateral pulmonary hilarlymphadenopathy, or accompanied without mediastinal lym-phadenectasis or with only mediastinal lymphadenectasis[4,7,8]. Additionally, it might be also characterized by irreg-ular pulmonary consolidation with the incidence of about37.5%, which often distributed in the middle and lateral pul-monary field, among which air bronchogram was accompa-nied [9]. That sarcoidosis leading to pleural involvement wasrare, about 2.8% as reported by Huggins JT et al. [10], whichwas thought to be one of the reasons causing easy misdiag-nosis as tuberculosis. A slight or moderate extent of pleuraleffusion was visible in these cases, which could affect uni-lateral or bilateral pleura, and sometimes multiple tiny noduleswere visible on the surface of the pleura.

The imaging findings of this case were not typical, whichreflected in the following aspects: i) a large denser patchyconsolidation around the right pulmonary hilum, accompaniedwith mid-bronchial stenosis; ii) pleural effusion with multipletiny nodules in the right pleural area similar to pleuralmetastasis; iii) obvious small pulmonary nodules withoutbilateral hilar lymphadenopathy. Additionally, tuberculosiscould not be excluded according to the first thoracoscopicpleural biopsy, until results from tuberculosis-related clinicalexamination proofed that the tuberculosis was basically ruleout, and finally, the Ebus lymph node biopsy confirmed thediagnosis as ‘sarcoidosis’.

4. Conclusion

The typical manifestation of chest sarcoidosis includedbilateral hilar lymphadenophathy symmetrically accompanied

with mediastinal lymphadenectasis near both the pulmonaryhiluses. The particularity of this case prompt that it should benoted about the abovementioned atypical sarcoidosis perfor-mance in clinical work, especially pleural effusion and pleuralsmall nodules, which might present as ‘mild clinical symp-toms but severe imaging findings’. The case facilitated im-aging diagnosis of the sarcoidosis, which would aid accuratediagnosis by combining with biopsy.

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