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Table of Contents
CASE REPORT
Year : 2019  |  Volume : 21  |  Issue : 1  |  Page : 69-71

Diagnosis and treatment of coincident Hodgkin's lymphoma and hamartoma by endobronchial methods: A Case report


1 Department of Interventional Pulmonology, Atatürk Chest Diseases and Thoracic Surgery Research and Education Hospital, Ankara, Turkey
2 Department of Pulmonology, Atatürk Chest Diseases and Thoracic Surgery Research and Education Hospital, Ankara, Turkey
3 Department of Pathology, Atatürk Chest Diseases and Thoracic Surgery Research and Education Hospital, Ankara, Turkey

Date of Submission15-Feb-2018
Date of Decision09-Mar-2018
Date of Acceptance02-May-2018
Date of Web Publication30-Apr-2019

Correspondence Address:
Dr. Sezgi Şahin Duyar
Department of Pulmonology, Atatürk Chest Diseases and Thoracic Surgery Research and Education Hospital, 06280 Keçiören, Ankara
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ejop.ejop_23_19

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  Abstract 


Endobronchial ultrasound-transbronchial needle aspiration (EBUS-TBNA) is recommended for the diagnosis of malign and benign mediastinal lymphadenopathies and lesions adjacent to the central airways. However the diagnostic yield of EBUS-TBNA in diagnosis of lymphoma is weak. Additionally, the challenge of cathcing Reed-Sternberg cells in such a small sample size lowers the sensitivitiy of EBUS-TBNA for diagnosis of Hodgkin lymphoma. EBUS-TBNA can be performed with rigid bronchoscopy. A 64 years old male patient with multiple abdominal and mediastinal lymphadenopathies with coinciding hamartoma and Hodgkin lymphoma is reported for presenting diagnostic and therapeutic interventional methods performed for this unique coincidance.

Keywords: Endobronchial ultrasound-guided transbronchial needle aspirate, hamartoma, Hodgkin's lymphoma


How to cite this article:
Ozturk A, Aktaş Z, Duyar S&, Yılmaz A, Demiraǧ F. Diagnosis and treatment of coincident Hodgkin's lymphoma and hamartoma by endobronchial methods: A Case report. Eurasian J Pulmonol 2019;21:69-71

How to cite this URL:
Ozturk A, Aktaş Z, Duyar S&, Yılmaz A, Demiraǧ F. Diagnosis and treatment of coincident Hodgkin's lymphoma and hamartoma by endobronchial methods: A Case report. Eurasian J Pulmonol [serial online] 2019 [cited 2019 May 27];21:69-71. Available from: http://www.eurasianjpulmonol.com/text.asp?2019/21/1/69/257449




  Introduction Top


Interventional bronchoscopy has rapidly gained a wide area in diagnostic and therapeutic approaches in both malignant and nonmalignant lesions during the last decades. Flexible bronchoscopy which is widely available can access distal airways without general anesthesia, but rigid bronchoscopy can be superior in situ ations when more secured airway is needed. Endobronchial ultrasound-guided transbronchial needle aspirate (EBUS-TBNA) can be used for diagnosis of malignant hilar and mediastinal lymph nodes with a sensitivity of 85%–100%.[1] However, its role in the diagnosis of suspected lymphoma is still being debated over.


  Case Report Top


A 64-year-old male patient admitted to our clinic with complaints of dyspnea and fatigue for 6 months. His medical history was irrelevant except that he was an ex-smoker with a 50 packs/year smoking history. His physical examination was normal and oxygen saturation in room air was measured as 94% via a pulse oximeter.

Routine biochemistry was totally normal. Complete blood count was as follows: white blood cells: 5900/mm3, hemoglobin: 7.1 g/dl, and platelet: 404,000/mm3. Sedimentation was 40 mm/h; C-reactive protein was 5.5 mg/dl. After these routine tests, the medical investigation was focused on the cause of anemia. Hematological parameters including levels of serum iron, iron-binding capacity, ferritin, Vitamin B12, and folate were normal. Hepatosplenomegaly was not evident on abdominal ultrasonography, but abdominal tomography revealed multiple lymphadenopathy, which of the biggest is 2 cm in diameter. Upper gastrointestinal endoscopy and colonoscopy were performed, and neither sign of cancer nor hemorrhage was observed.

The patient underwent pulmonary function test (PFT) and computed tomography (CT) of the thorax for differential diagnosis of chronic dyspnea. PFT resulted in restrictive dysfunction. The CT of the thorax demonstrated multiple conglomerate mediastinal lymphadenopathies in paratracheal, aortopulmonary, subcarinal, and right paraesophageal regions [Figure 1].
Figure 1: Coronal section of thorax computed tomography: mediastinal lymphadenopathies (the circle) and the endobronchial hamartoma in the apical segment of left lower lobe (the arrow)

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Initially, fiber-optic bronchoscopy (FOB) under topical airway anesthesia was planned. However, the patient could not tolerate the procedure, so it was switched to FOB under deep sedation. A polypoid lesion was visualized during FOB under deep sedation, and the patient underwent rigid bronchoscopy. A polypoid lesion in the apical segment of the left lower lobe was totally extracted by cryoprobe [Figure 2]. EBUS was performed for differential diagnosis of the mediastinal lymph nodes through the rigid bronchoscope, and subcarinal hypoechoic lymph node with unruffled borders was sampled [Figure 3]. Informed consent was taken before the interventions and for disclosing the patient's records for scientific purposes.
Figure 2: Bronchoscopic images of polypoid lesion projecting from the orifice of the apical segment of left lower lobe

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Figure 3: The images from endobronchial ultrasound: subcarinal hypoechoic lymph node with unruffled borders

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Pathologic evaluation of cryobiopsy pointed out the diagnosis of lipomatous hamartoma, whereas EBUS-TBNA of the lymph node was coherent with Hodgkin's lymphoma (HL). The patient was referred to the oncology center for treatment and follow-up of HL.


  Discussion Top


The diagnostic procedure of this patient is special in many aspects. The pathological diagnosis and subtyping of lymphoma by EBUS-TBNA are very difficult because of small sample size, leading to discordance between cytological and histological samples. In spite of these difficulties, Kennedy et al. presented a retrospective study of 25 patients with suspected lymphoma who were diagnosed by EBUS-TBNA with a sensitivity of 90.9% and a specificity of 100%.[2] However, The British Thoracic Society Interventional Bronchoscopy Guideline Group indicated that there was not sufficient evidence to recommend EBUS-TBNA for routine use in the diagnosis of lymphoma in 2011.[1] Since the publication of this guideline, ongoing researches have tried to shine a light on this topic; however, because of wide range of sensitivity, this issue remains a controversy.[3],[4] However, in the study of Moonim et al., high diagnostic yield of EBUS-TBNA for HL was observed with a sensitivity of 79%.[4]

As it was stated in the CHEST guideline of EBUS-TBNA published in 2016, EBUS-TBNA which is a minimally invasive procedure can be the first step for patients with suspected lymphoma but negative results, especially for HL, must be reevaluated by mediastinoscopy because it is difficult to detect Reed–Sternberg cells in fine-needle aspiration biopsies and also larger sample size can be beneficial for flow cytometry and immunohistochemical staining.[5] We also managed to reach the diagnosis of HL by EBUS-TBNA.

EBUS-TBNA can be performed with rigid bronchoscopy in some special cases requiring a secured airway for performing additional therapeutic procedures. Ozkan et al. reported that EBUS-TBNA with rigid bronchoscopy, jet ventilation, and general anesthesia was performed efficiently and safely in 105 cases.[6] We performed EBUS-TBNA with the same technique because of the accompanying hamartoma which was extracted by cryobiopsy in the first place. The patient was recovered from hamartoma and was diagnosed as HL in the same session.

To our knowledge, the coincidence of endobronchial hamartoma (EH) and HL has not been reported before. A few cases of lung cancer concomitant with EH were reported to raise awareness for excluding a metastatic lesion to conduct correct treatment.[7],[8]

Despite high recurrence rates, bronchoscopic methods including mechanical resection, laser, cryotherapy, and argon plasma coagulation are considered as the first choice for the treatment of EH. As repeated bronchoscopic management is effective with less morbidity, surgical resection must be an alternative for the patients with end-stage lung damage.[9],[10]


  Conclusion Top


The usage of different bronchoscopic techniques in this unique coincidence of EH and HL resulted in treatment and diagnosis of these diseases, respectively. This case report represents the importance of patient-tailored approach in interventional pulmonology.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Du Rand IA, Barber PV, Goldring J, Lewis RA, Mandal S, Munavvar M. British Thoracic Society guideline for advanced diagnostic and therapeutic flexible bronchoscopy in adults. Thorax 2011;66 Suppl 3:iii1-21.  Back to cited text no. 1
    
2.
Kennedy MP, Jimenez CA, Bruzzi JF, Mhatre AD, Lei X, Giles FJ. Endobronchial ultrasound-guided transbronchial needle aspiration in the diagnosis of lymphoma. Thorax 2008;63:360-5.  Back to cited text no. 2
    
3.
Erer OF, Erol S, Anar C, Aydoǧdu Z, Özkan SA. Diagnostic yield of EBUS-TBNA for lymphoma and review of the literature. Endosc Ultrasound 2017;6:317-22.  Back to cited text no. 3
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4.
Moonim MT, Breen R, Fields PA, Santis G. Diagnosis and subtyping of de novo and relapsed mediastinal lymphomas by endobronchial ultrasound needle aspiration. Am J Respir Crit Care Med 2013;188:1216-23.  Back to cited text no. 4
    
5.
Wahidi MM, Herth F, Yasufuku K, Shepherd RW, Yarmus L, Chawla M. Technical aspects of endobronchial ultrasound-guided transbronchial needle aspiration: CHEST guideline and expert panel report. Chest 2016;149:816-35.  Back to cited text no. 5
    
6.
Ozkan F, Khan A, Freitag L, Darwiche K. EBUS TBNA with rigid bronchoscopy, jet ventilation and general anesthesia – A review of procedural, anesthesia and post recovery times. Eur Respir J 2014;44:3721.  Back to cited text no. 6
    
7.
Oshima Y, Niiya Y, Minakata T, Himuro N, Tomita Y, Kataoka D, et al. Lung cancer associated with hamartoma; Report of a case. Kyobu Geka 2018;71:142-5.  Back to cited text no. 7
    
8.
Chen SS, Zhou H, Tong B, Yu LL, Fan SS, Xiao ZK, et al. Endobronchial hamartoma mimicking malignant lung tumor contralateral endobronchial metastasis: A case report. Medicine (Baltimore) 2017;96:e9085.  Back to cited text no. 8
    
9.
Abdel Hady SM, Elbastawisy SE, Hassaballa AS, Elsayed HH. Is surgical resection superior to bronchoscopic resection in patients with symptomatic endobronchial hamartoma? Interact Cardiovasc Thorac Surg 2017;24:778-82.  Back to cited text no. 9
    
10.
Kim SA, Um SW, Song JU, Jeon K, Koh WJ, Suh GY, et al. Bronchoscopic features and bronchoscopic intervention for endobronchial hamartoma. Respirology 2010;15:150-4.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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