Clin J Gastroenterol (2014) 7:68–73 DOI 10.1007/s12328-013-0451-5
CASE REPORT
A case of peripancreatic tuberculous lymphadenitis diagnosed by endoscopic ultrasound-guided fine-needle aspiration Hiroto Furuhashi • Hiroshi Abe • Kai Yoshizawa • Yuki Hirose • Yukiko Miura Nobuyoshi Seki • Tamihiro Miyazaki • Tomonori Sugita • Yuta Aida • Munenori Itagaki • Haruya Ishiguro • Satoshi Sutoh • Yoshio Aizawa
•
Received: 25 June 2013 / Accepted: 15 December 2013 / Published online: 10 January 2014 Ó Springer Japan 2014
Abstract Tuberculous lymphadenitis is a rare cause of obstructive jaundice. Here, we report the case of a 33-yearold male with obstructive jaundice caused by tuberculous lymphadenitis around the pancreatic head. The patient was born in China and had immigrated to Japan at 12 years of age. He presented with acute abdominal pain and jaundice. Findings from ultrasonography, computed tomography, and endoscopic retrograde cholangiopancreatography were suggestive of a stenosis of the distal common bile duct caused by multiple low-density masses around the pancreatic head with a contrast-enhanced solid rim. We successfully diagnosed the mass as tuberculous lymphadenitis using endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA). The patient was treated with anti-tuberculous combination chemotherapy for 6 months, and subsequently exhibited clinical improvement. Thus, we found that EUSFNA was a valuable minimally invasive method for diagnosing masses that cause icterus. Keywords Tuberculous lymphadenitis Obstructive jaundice Endoscopic ultrasound-guided fine-needle aspiration
Introduction Although tuberculosis is one of the most common diseases found worldwide, abdominal lymphadenitis of the pancreas or peripancreatic region due to tuberculous infection is a rare cause of obstructive jaundice [1–3]. The majority of reported cases of tuberculous lymphadenitis around the common bile duct (CBD) have been incidentally diagnosed following laparotomy or laparoscopy since it mimics pancreatic and biliary carcinoma, which are the most common causes of such jaundice [4]. Successful diagnosis of tuberculous lymphadenitis without laparotomy or laparoscopy has been reported only once to date [5]. Recently, endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA) has been used for the pathological diagnosis of swollen abdominal lymph nodes as a minimally invasive method. Here, we report a case of obstructive jaundice caused by tuberculous lymphadenitis around the pancreatic head that was successfully diagnosed using EUS-FNA. We also review the literature concerning obstructive jaundice caused by tuberculous lymphadenitis.
Case report H. Furuhashi (&) H. Abe Y. Hirose Y. Miura N. Seki T. Miyazaki T. Sugita Y. Aida M. Itagaki H. Ishiguro S. Sutoh Y. Aizawa Division of Gastroenterology and Hepatology, Department of Internal Medicine, Jikei University School of Medicine Katsushika Medical Center, 6-41-2 Aoto, Katsushika-ku, Tokyo 125-8506, Japan e-mail: [email protected] K. Yoshizawa Department of Gastroenterology, Machida Municipal Hospital, Tokyo, Japan
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A 33-year-old previously health male without a history of treatment for tuberculosis presented with acute abdominal pain and jaundice. The patient was born in Shanghai, China, and had immigrated to Japan at the age of 12 years. He had no history of tuberculin skin testing or Bacille de Calmette et Gue´rin vaccination prior to admission. General physical examination revealed no remarkable findings other than icterus. Laboratory examination at the time of admission revealed total bilirubin 5.6 mg/dL, direct bilirubin 4.5
Clin J Gastroenterol (2014) 7:68–73
69
Table 1 Laboratory data on admission WBC
5.8 9 103/lL
RBC
4.44 9 106/lL
Hb
14.4 g/dL
Plt
210 9 103/lL
PT
91 %
PT-INR
1.05
APTT
29.8 s
Fbg
442 mg/dL
HPT
100 %
T-Bil
5.6 mg/dL
D-Bil
4.5 mg/dL
AST
262 IU/L
ALT
375 IU/L
LDH
270 IU/L
ALP c-GTP
1,499 IU/L 2,226 IU/L
TP
7.5 g/dL
Alb
4.2 g/dL
Amy
34 IU/L
UN
9 mg/dL
Cre
0.75 mg/dL
CRP
1.3 mg/dL
CEA
2.2 ng/mL
CA19-9
57 IU/mL
SCC
0.6 ng/mL
NSE
13.2 ng/mL
DUPAN-2
320 IU/mL
SIL-2R
511 IU/mL
mg/dL, alkaline phosphatase 1,499 IU/L, gamma glutamyl transferase 2,226 IU/L, aspartate aminotransferase 262 IU/L, and alanine aminotransferase 375 IU/L. Serum tumor marker levels were CEA 2.2 ng/mL, CA19-9 57 U/mL, SCC 0.6 ng/mL, NSE 13.2 ng/mL, DUPAN-2 320 U/mL, and soluble interleukin-2 receptor 511 U/mL (Table 1). Serological tests for human immunodeficiency virus (HIV) p24 antigen and HIV antibody were negative. Plain chest radiographs and chest computed tomography (CT) scans showed no abnormalities. Transabdominal ultrasonography revealed dilation of the intrahepatic bile ducts (IHBDs) and CBD, with the presence of hypoechoic multinodular masses around the CBD; gallstones and choledocholithiasis were not observed (Fig. 1). An enhanced CT scan showed dilatation of the IHBD and CBD along with stenosis of the distal CBD caused by multiple low-density masses around the pancreatic head with a contrast-enhanced solid rim (Fig. 2); this finding was characteristic of abdominal lymphadenitis. No swelling of other lymph nodes was observed in any other location. No space-occupying lesion was detected in the pancreas, liver, gallbladder, kidneys, or spleen.
Fig. 1 Abdominal ultrasonography showing hypoechoic multinodular masses around the pancreatic head (arrow)
Fig. 2 Enhanced computed tomography showing multiple lowdensity masses around the pancreatic head with a contrast-enhanced solid rim (arrow)
Subsequent esophagogastroduodenoscopy and colonoscopy showed no abnormalities. Endoscopic retrograde cholangiopancreatography (ERCP) was then performed, revealing a 3-cm smooth stenosis in the distal CBD due to extrinsic compression (Fig. 3). We therefore initiated endoscopic retrograde biliary drainage (ERBD) with a single 7-cm 7-Fr polyethylene stent. Cytological examination of the bile duct was not performed. EUS was initially performed by an experienced endoscopist using a radial echo-endoscope EU-ME1 (Olympus Ltd., Tokyo, Japan). EUS showed a multinodular hypoechoic lesion with a heterogenous pattern (Fig. 4). To confirm the histological diagnosis, EUS-FNA was performed. We used a convex array echo-endoscope GFUCT240-AL5 (Olympus) to assess the low-density masses, with 5 passes, using a 22-gauge manually operated needle device NA-10 J-1 (Olympus), as described in a previous report [6], under Doppler guidance to avoid intervening vascular structures. The transduodenal route was used to
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lymphadenitis. Acid-fast bacilli were clearly identified on Ziehl–Neelsen staining (Fig. 5c, d). Polymerase chain reaction of the tissue was positive for Mycobacterium tuberculosis DNA. A standard tuberculin skin test showed a [20-mm infiltrate. After a few weeks, culture of the obtained tissue tested positive for M. tuberculosis. Therefore, anti-tuberculous therapy was initiated, and the patient was discharged. The patient received a full course of anti-tuberculous drugs with isoniazid, rifampicin, ethambutol, and pyrazinamide for 2 months, followed by isoniazid, rifampicin, and ethambutol for 4 months. After the full course of antituberculous therapy was completed, CT scans showed marked shrinking of the masses. Subsequently, the biliary stent was removed, and no sign of recurrence was noted. The patient was found to have remained in a good condition at the 12-month follow-up examination.
Discussion Fig. 3 Endoscopic retrograde cholangiopancreatography showing a 3-cm smooth defect in the distal common bile duct due to extrinsic compression (arrow)
Fig. 4 Endoscopic ultrasonography showing a multinodular hypoechoic lesion with a heterogenous pattern (arrow heads)
obtain samples of lymph nodes around the pancreatic head; however, an image at the time of puncture could not be obtained. Histopathological examination demonstrated the presence of epithelioid granulomatous inflammation with large necrotic areas and Langhans giant cells (Fig. 5a, b). These findings were strongly suggestive of tuberculous
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Tuberculous lymphadenitis should be considered as a differential diagnosis for obstructive jaundice, especially in patients from areas that are endemic for tuberculosis as well as in immigrant patients from endemic countries. In developed countries, *70 % of the reported cases of tuberculous lymphadenitis have occurred in immigrants [7–9], with a median patient age of *40 years [7, 9, 10]. The active incidence rate of tuberculosis in Shanghai in 1987–1990 (54.5/100,000) was considerably higher than in Japan (39.3/100,000). These findings strongly suggested that, in the present case, tuberculous lymphadenitis developed a considerable amount of time after the first latent infection, which itself developed when the patient was living in Shanghai. However, the specific period of the first tuberculous infection could not be precisely confirmed. Even if tuberculous lymphadenitis is strongly suspected as a cause of obstructive jaundice, it is not easy to confirm this diagnosis without invasive techniques, such as laparotomy or laparoscopy [11–13]. Because histological confirmation of epithelioid granulomas or detection of M. tuberculosis in the local lymph node is a gold standard for the diagnosis of tuberculous lymphadenitis, histopathological examination of the involved lymph node is indispensable [14]. The imaging findings for tuberculous lymphadenitis may mimic a malignant stricture caused by cystic neoplasms of the pancreas, bile duct carcinoma, malignant lymphoma, or metastatic lymph node [15–18]. However, multiple low-density masses around the pancreatic head with a contrast-enhanced solid rim on contrast enhanced CT scan, as in the present case, appear to be a characteristic feature of lymphadenitis. This finding was reported to
Clin J Gastroenterol (2014) 7:68–73
71
Fig. 5 Histopathological findings of endoscopic ultrasound-guided fine-needle aspiration samples showing epithelioid granulomatous inflammation with large necrotic areas (a hematoxylin and eosin stain,
9100) and a Langhans giant cell (b hematoxylin and eosin stain, 9200). In addition, acid-fast bacilli were clearly observed (c Ziehl– Neelsen stain, 9400; d 91,000)
represent central caseous necrosis with peripheral active inflammation of swollen lymph nodes that are infected with tuberculosis [19]. Therefore, we believed that this lymph node-associated disease was more likely to be tuberculous lymphadenitis than malignant lymphoma or malignant lymph node metastasis from a primary lesion. To confirm the histopathological diagnosis of tuberculous lymphadenitis, we subsequently performed EUS-FNA, through which the diagnosis was successfully established. We searched the PubMed database (1993–2013) for similar case reports using the keywords ‘tuberculous’, ‘lymphadenitis’, and ‘jaundice’. Cases where the diagnosis was histologically accomplished based on the findings of concurrent extra-abdominal lymph nodes were excluded. A total of 27 cases (mean age 38.8 years; range 18–68 years) with jaundice caused by tuberculous lymphadenitis were found [5, 11–13, 15–18, 20]. In these 27 cases, laparotomy was performed in 21 cases [11–13, 15–18, 20], laparoscopy was conducted in 1 case [17], and EUS-FNA was performed with successful histological diagnosis in only 1 case [5]. In the remaining 4 cases, the methodology for obtaining histological diagnosis was not described [11]. These findings strongly suggested that diagnosing tuberculous
lymphadenitis is considerably difficult without laparotomy or laparoscopy. EUS-FNA is a minimally invasive technique for obtaining histological samples from swollen abdominal lymph nodes [5]. However, there are several disadvantages to using EUS-FNA. One of the disadvantages is the possibility of tumor seeding during aspiration biopsy from a ‘malignant’ mass. Therefore, EUS-FNA may be contraindicated if the possibility of a malignant tumor has not been ruled out. However, many recent reports have suggested that the possibility of FNA-related tumor seeding by EUS is considerably lower than that by percutaneous ultrasonography [21–27]. Another disadvantage of EUS-FNA is the difficulty in obtaining an adequate sample for accurate diagnosis; the diagnostic accuracy of EUS-FNA is variable at 60–90 % [28–30]. Although variously sized needles (19–25 gauge) have been used for EUS-FNA, there is no evidence that using larger-sized needles can achieve a more accurate diagnosis. It is possible that the number of samples obtained by EUSFNA, rather than the size, may be associated with the accuracy of histological diagnosis. A previous report has suggested that at least 5 passes are required to ensure
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adequate cellularity for lymph nodes with a diagnostic sensitivity and specificity of 77 and 100 %, respectively [6]. We therefore performed EUS-FNA with 5 passes, and obtained an accurate histological diagnosis. In conclusion, we have presented here a case of obstructive jaundice caused by tuberculous lymphadenitis. Young age and a history of emigration from a country endemic for tuberculosis are diagnostic clues for tuberculous lymphadenitis in developed countries. EUS-FNA is a safe and minimally invasive method for histological confirmation of diagnosis and should be considered as the first choice for obtaining samples from lymph nodes around the CBD. Disclosures Conflict of Interest of interest.
The authors declare that they have no conflict
Human/Animal Rights All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2008(5). Informed Consent Informed consent was obtained from all patients for being included in the study.
References 1. Itaba S, Yoshinaga S, Nakamura K, Mizutani T, Honda K, Takayanagi R, et al. Endoscopic ultrasound-guided fine-needle aspiration for the diagnosis of peripancreatic tuberculous lymphadenitis. J Gastroenterol. 2007;42:83–6. 2. Boujaoude JD, Honein K, Yaghi C, Ghora C, Abadjian G, Sayegh R. Diagnosis by endoscopic ultrasound guided fine needle aspiration of tuberculous lymphadenitis involving the peripancreatic lymph nodes: a case report. World J Gastroenterol. 2007;13:474–7. 3. Song TJ, Lee SS, Park do H, Lee TY, Lee SO, Seo DW, et al. Yield of EUS-guided FNA on the diagnosis of pancreatic/ peripancreatic tuberculosis. Gastrointest Endosc. 2009;69:484–91. 4. Reisman Y, Gips CH, Lavelle SM, Wilson JH. Clinical presentation of (subclinical) jaundice—the Euricterus project in The Netherlands. Hepatogastroenterology. 1996;43:1190–5. 5. Macias-Garcia F, Iglesias-Garcia J, Abdulkader I, Larino-Noia J, Forteza-Vila J, Dominguez-Munoz JE. Tuberculous lymph node at the porta hepatis: diagnosis by EUS-guided FNA. Gastrointest Endosc. 2011;74:437–9. 6. LeBlanc JK, Ciaccia D, Al-Assi MT, McGrath K, Imperiale T, Tao LC, et al. Optimal number of EUS-guided fine needle passes needed to obtain a correct diagnosis. Gastrointest Endosc. 2004;59:475–81. 7. Geldmacher H, Taube C, Kroeger C, Magnussen H, Kirsten DK. Assessment of lymph node tuberculosis in northern Germany: a clinical review. Chest. 2002;121:1177–82. 8. Fain O, Lortholary O, Djouab M, Amoura I, Babinet P, Beaudreuil J, et al. Lymph node tuberculosis in the suburbs of Paris: 59 cases in adults not infected by the human immunodeficiency virus. Int J Tuberc Lung Dis. 1999;3:162–5.
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9. Wark P, Goldberg H, Ferson M, McKenzie D, Lau E, Rivas K. Mycobacterial lymphadenitis in eastern Sydney. Aust NZ J Med. 1998;28:453–8. 10. Mert A, Tabak F, Ozaras R, Tahan V, Oztu¨rk R, Aktug˘lu Y. Tuberculous lymphadenopathy in adults: a review of 35 cases. Acta Chir Belg. 2002;102:118–21. 11. Xia F, Poon RT, Wang SG, Bie P, Huang XQ, Dong JH. Tuberculosis of pancreas and peripancreatic lymph nodes in immunocompetent patients: experience from China. World J Gastroenterol. 2003;9:1361–4. 12. Obama K, Kanai M, Taki Y, Nakamoto Y, Takabayashi A. Tuberculous lymphadenitis as a cause of obstructive jaundice: report of a case. Surg Today. 2003;33:229–31. 13. Turan M, Sen M, Koyuncu A, Aydin C, Elaldi N, Arici S. Pancreatic pseudotumor due to peripancreatic tuberculous lymphadenitis. Pancreatology. 2002;2:561–4. 14. Glassroth J. Diagnosis of tuberculosis. In: Reichmann LB, Hershfield ES, editors. Tuberculosis: a comprehensive international Approach. New York: Marcel Dekker; 1993. p. 149–62. 15. Colovic R, Gubor N, Jesic R, Micev M, Jovanovic T, Colovic N, et al. Tuberculous lymphadenitis as a cause of obstructive jaundice: a case report and literature review. World J Gastroenterol. 2008;14:3098–100. 16. Sen M, Turan M, Karadayi K, Aslan M, Elagoz S. Peripancreatic tuberculous lymphadenitis mimicking carcinoma: report of a case. Acta Chir Belg. 2004;104:338–40. 17. Poon RT, Lo CM, Fan ST. Diagnosis and management of biliary obstruction due to periportal tuberculous adenitis. Hepatogastroenterology. 2001;48:1585–7. 18. Caroli-Bosc FX, Conio M, Maes B, Chevallier P, Hastier P, Delmont JP. Abdominal tuberculous involving hepatic hilar lymph nodes. A cause of portal vein thrombosis and portal hypertension. J Clin Gastroenterol. 1997;25:541–3. 19. Pereira JM, Madureira AJ, Vieira A, Ramos I. Abdominal tuberculosis: imaging features. Eur J Radiol. 2005;55:173–80. 20. Peyre CG, Wakim M, Mateo R, Genyk Y, Singh G, Selby RR, et al. Unusual cases of jaundice secondary to non-neoplastic bile duct obstruction. Am Surg. 2004;70:620–4. 21. Micames C, Jowell PS, White R, Paulson E, Nelson R, Morse M, et al. Lower frequency of peritoneal carcinomatosis in patients with pancreatic cancer diagnosed by EUS-guided FNA vs. percutaneous FNA. Gastrointest Endosc. 2003;58:690–5. 22. Katanuma A, Maguchi H, Hashigo S, Kaneko M, Kin T, Yane K, et al. Tumor seeding after endoscopic ultrasound-guided fineneedle aspiration of cancer in the body of the pancreas. Endoscopy. 2012;44:E160–1. 23. Shah JN, Fraker D, Guerry D, Feldman M, Kochman ML. Melanoma seeding of an EUS-guided fine needle track. Gastrointest Endosc. 2004;59:923–4. 24. Paquin SC, Garie´py G, Lepanto L, Bourdages R, Raymond G, Sahai AV. A first report of tumor seeding because of EUS-guided FNA of a pancreatic adenocarcinoma. Gastrointest Endosc. 2005;61:610–1. 25. Doi S, Yasuda I, Iwashita T, Ibuka T, Fukushima H, Araki H, et al. Needle tract implantation on the esophageal wall after EUSguided FNA of metastatic mediastinal lymphadenopathy. Gastrointest Endosc. 2008;67:988–90. 26. Hirooka Y, Goto H, Itoh A, Hashimoto S, Niwa K, Ishikawa H, et al. Case of intraductal papillary mucinous tumor in which endosonography-guided fine-needle aspiration biopsy caused dissemination. J Gastroenterol Hepatol. 2003;18:1323–4. 27. Chong A, Venugopal K, Segarajasingam D, Lisewski D. Tumor seeding after EUS-guided FNA of pancreatic tail neoplasia. Gastrointest Endosc. 2011;74:933–5. 28. Wiersema MJ, Vilmann P, Giovannini M, Chang KJ, Wiersema LM. Endosonography-guided fine-needle aspiration biopsy:
Clin J Gastroenterol (2014) 7:68–73 diagnostic accuracy and complication assessment. Gastroenterology. 1997;112:1087–95. 29. Gress FG, Hawes RH, Savides TJ, Ikenberry SO, Lehman GA. Endoscopic ultrasound-guided fine-needle aspiration biopsy using linear array and radial scanning endosonography. Gastrointest Endosc. 1997;45:243–50.
73 30. Gress F, Gottlieb K, Sherman S, Lehman G. Endoscopic ultrasonography-guided fine-needle aspiration biopsy of suspected pancreatic cancer. Ann Intern Med. 2001;134:459–64.
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CASE REPORT
A case of peripancreatic tuberculous lymphadenitis diagnosed by endoscopic ultrasound-guided fine-needle aspiration Hiroto Furuhashi • Hiroshi Abe • Kai Yoshizawa • Yuki Hirose • Yukiko Miura Nobuyoshi Seki • Tamihiro Miyazaki • Tomonori Sugita • Yuta Aida • Munenori Itagaki • Haruya Ishiguro • Satoshi Sutoh • Yoshio Aizawa
•
Received: 25 June 2013 / Accepted: 15 December 2013 / Published online: 10 January 2014 Ó Springer Japan 2014
Abstract Tuberculous lymphadenitis is a rare cause of obstructive jaundice. Here, we report the case of a 33-yearold male with obstructive jaundice caused by tuberculous lymphadenitis around the pancreatic head. The patient was born in China and had immigrated to Japan at 12 years of age. He presented with acute abdominal pain and jaundice. Findings from ultrasonography, computed tomography, and endoscopic retrograde cholangiopancreatography were suggestive of a stenosis of the distal common bile duct caused by multiple low-density masses around the pancreatic head with a contrast-enhanced solid rim. We successfully diagnosed the mass as tuberculous lymphadenitis using endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA). The patient was treated with anti-tuberculous combination chemotherapy for 6 months, and subsequently exhibited clinical improvement. Thus, we found that EUSFNA was a valuable minimally invasive method for diagnosing masses that cause icterus. Keywords Tuberculous lymphadenitis Obstructive jaundice Endoscopic ultrasound-guided fine-needle aspiration
Introduction Although tuberculosis is one of the most common diseases found worldwide, abdominal lymphadenitis of the pancreas or peripancreatic region due to tuberculous infection is a rare cause of obstructive jaundice [1–3]. The majority of reported cases of tuberculous lymphadenitis around the common bile duct (CBD) have been incidentally diagnosed following laparotomy or laparoscopy since it mimics pancreatic and biliary carcinoma, which are the most common causes of such jaundice [4]. Successful diagnosis of tuberculous lymphadenitis without laparotomy or laparoscopy has been reported only once to date [5]. Recently, endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA) has been used for the pathological diagnosis of swollen abdominal lymph nodes as a minimally invasive method. Here, we report a case of obstructive jaundice caused by tuberculous lymphadenitis around the pancreatic head that was successfully diagnosed using EUS-FNA. We also review the literature concerning obstructive jaundice caused by tuberculous lymphadenitis.
Case report H. Furuhashi (&) H. Abe Y. Hirose Y. Miura N. Seki T. Miyazaki T. Sugita Y. Aida M. Itagaki H. Ishiguro S. Sutoh Y. Aizawa Division of Gastroenterology and Hepatology, Department of Internal Medicine, Jikei University School of Medicine Katsushika Medical Center, 6-41-2 Aoto, Katsushika-ku, Tokyo 125-8506, Japan e-mail: [email protected] K. Yoshizawa Department of Gastroenterology, Machida Municipal Hospital, Tokyo, Japan
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A 33-year-old previously health male without a history of treatment for tuberculosis presented with acute abdominal pain and jaundice. The patient was born in Shanghai, China, and had immigrated to Japan at the age of 12 years. He had no history of tuberculin skin testing or Bacille de Calmette et Gue´rin vaccination prior to admission. General physical examination revealed no remarkable findings other than icterus. Laboratory examination at the time of admission revealed total bilirubin 5.6 mg/dL, direct bilirubin 4.5
Clin J Gastroenterol (2014) 7:68–73
69
Table 1 Laboratory data on admission WBC
5.8 9 103/lL
RBC
4.44 9 106/lL
Hb
14.4 g/dL
Plt
210 9 103/lL
PT
91 %
PT-INR
1.05
APTT
29.8 s
Fbg
442 mg/dL
HPT
100 %
T-Bil
5.6 mg/dL
D-Bil
4.5 mg/dL
AST
262 IU/L
ALT
375 IU/L
LDH
270 IU/L
ALP c-GTP
1,499 IU/L 2,226 IU/L
TP
7.5 g/dL
Alb
4.2 g/dL
Amy
34 IU/L
UN
9 mg/dL
Cre
0.75 mg/dL
CRP
1.3 mg/dL
CEA
2.2 ng/mL
CA19-9
57 IU/mL
SCC
0.6 ng/mL
NSE
13.2 ng/mL
DUPAN-2
320 IU/mL
SIL-2R
511 IU/mL
mg/dL, alkaline phosphatase 1,499 IU/L, gamma glutamyl transferase 2,226 IU/L, aspartate aminotransferase 262 IU/L, and alanine aminotransferase 375 IU/L. Serum tumor marker levels were CEA 2.2 ng/mL, CA19-9 57 U/mL, SCC 0.6 ng/mL, NSE 13.2 ng/mL, DUPAN-2 320 U/mL, and soluble interleukin-2 receptor 511 U/mL (Table 1). Serological tests for human immunodeficiency virus (HIV) p24 antigen and HIV antibody were negative. Plain chest radiographs and chest computed tomography (CT) scans showed no abnormalities. Transabdominal ultrasonography revealed dilation of the intrahepatic bile ducts (IHBDs) and CBD, with the presence of hypoechoic multinodular masses around the CBD; gallstones and choledocholithiasis were not observed (Fig. 1). An enhanced CT scan showed dilatation of the IHBD and CBD along with stenosis of the distal CBD caused by multiple low-density masses around the pancreatic head with a contrast-enhanced solid rim (Fig. 2); this finding was characteristic of abdominal lymphadenitis. No swelling of other lymph nodes was observed in any other location. No space-occupying lesion was detected in the pancreas, liver, gallbladder, kidneys, or spleen.
Fig. 1 Abdominal ultrasonography showing hypoechoic multinodular masses around the pancreatic head (arrow)
Fig. 2 Enhanced computed tomography showing multiple lowdensity masses around the pancreatic head with a contrast-enhanced solid rim (arrow)
Subsequent esophagogastroduodenoscopy and colonoscopy showed no abnormalities. Endoscopic retrograde cholangiopancreatography (ERCP) was then performed, revealing a 3-cm smooth stenosis in the distal CBD due to extrinsic compression (Fig. 3). We therefore initiated endoscopic retrograde biliary drainage (ERBD) with a single 7-cm 7-Fr polyethylene stent. Cytological examination of the bile duct was not performed. EUS was initially performed by an experienced endoscopist using a radial echo-endoscope EU-ME1 (Olympus Ltd., Tokyo, Japan). EUS showed a multinodular hypoechoic lesion with a heterogenous pattern (Fig. 4). To confirm the histological diagnosis, EUS-FNA was performed. We used a convex array echo-endoscope GFUCT240-AL5 (Olympus) to assess the low-density masses, with 5 passes, using a 22-gauge manually operated needle device NA-10 J-1 (Olympus), as described in a previous report [6], under Doppler guidance to avoid intervening vascular structures. The transduodenal route was used to
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lymphadenitis. Acid-fast bacilli were clearly identified on Ziehl–Neelsen staining (Fig. 5c, d). Polymerase chain reaction of the tissue was positive for Mycobacterium tuberculosis DNA. A standard tuberculin skin test showed a [20-mm infiltrate. After a few weeks, culture of the obtained tissue tested positive for M. tuberculosis. Therefore, anti-tuberculous therapy was initiated, and the patient was discharged. The patient received a full course of anti-tuberculous drugs with isoniazid, rifampicin, ethambutol, and pyrazinamide for 2 months, followed by isoniazid, rifampicin, and ethambutol for 4 months. After the full course of antituberculous therapy was completed, CT scans showed marked shrinking of the masses. Subsequently, the biliary stent was removed, and no sign of recurrence was noted. The patient was found to have remained in a good condition at the 12-month follow-up examination.
Discussion Fig. 3 Endoscopic retrograde cholangiopancreatography showing a 3-cm smooth defect in the distal common bile duct due to extrinsic compression (arrow)
Fig. 4 Endoscopic ultrasonography showing a multinodular hypoechoic lesion with a heterogenous pattern (arrow heads)
obtain samples of lymph nodes around the pancreatic head; however, an image at the time of puncture could not be obtained. Histopathological examination demonstrated the presence of epithelioid granulomatous inflammation with large necrotic areas and Langhans giant cells (Fig. 5a, b). These findings were strongly suggestive of tuberculous
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Tuberculous lymphadenitis should be considered as a differential diagnosis for obstructive jaundice, especially in patients from areas that are endemic for tuberculosis as well as in immigrant patients from endemic countries. In developed countries, *70 % of the reported cases of tuberculous lymphadenitis have occurred in immigrants [7–9], with a median patient age of *40 years [7, 9, 10]. The active incidence rate of tuberculosis in Shanghai in 1987–1990 (54.5/100,000) was considerably higher than in Japan (39.3/100,000). These findings strongly suggested that, in the present case, tuberculous lymphadenitis developed a considerable amount of time after the first latent infection, which itself developed when the patient was living in Shanghai. However, the specific period of the first tuberculous infection could not be precisely confirmed. Even if tuberculous lymphadenitis is strongly suspected as a cause of obstructive jaundice, it is not easy to confirm this diagnosis without invasive techniques, such as laparotomy or laparoscopy [11–13]. Because histological confirmation of epithelioid granulomas or detection of M. tuberculosis in the local lymph node is a gold standard for the diagnosis of tuberculous lymphadenitis, histopathological examination of the involved lymph node is indispensable [14]. The imaging findings for tuberculous lymphadenitis may mimic a malignant stricture caused by cystic neoplasms of the pancreas, bile duct carcinoma, malignant lymphoma, or metastatic lymph node [15–18]. However, multiple low-density masses around the pancreatic head with a contrast-enhanced solid rim on contrast enhanced CT scan, as in the present case, appear to be a characteristic feature of lymphadenitis. This finding was reported to
Clin J Gastroenterol (2014) 7:68–73
71
Fig. 5 Histopathological findings of endoscopic ultrasound-guided fine-needle aspiration samples showing epithelioid granulomatous inflammation with large necrotic areas (a hematoxylin and eosin stain,
9100) and a Langhans giant cell (b hematoxylin and eosin stain, 9200). In addition, acid-fast bacilli were clearly observed (c Ziehl– Neelsen stain, 9400; d 91,000)
represent central caseous necrosis with peripheral active inflammation of swollen lymph nodes that are infected with tuberculosis [19]. Therefore, we believed that this lymph node-associated disease was more likely to be tuberculous lymphadenitis than malignant lymphoma or malignant lymph node metastasis from a primary lesion. To confirm the histopathological diagnosis of tuberculous lymphadenitis, we subsequently performed EUS-FNA, through which the diagnosis was successfully established. We searched the PubMed database (1993–2013) for similar case reports using the keywords ‘tuberculous’, ‘lymphadenitis’, and ‘jaundice’. Cases where the diagnosis was histologically accomplished based on the findings of concurrent extra-abdominal lymph nodes were excluded. A total of 27 cases (mean age 38.8 years; range 18–68 years) with jaundice caused by tuberculous lymphadenitis were found [5, 11–13, 15–18, 20]. In these 27 cases, laparotomy was performed in 21 cases [11–13, 15–18, 20], laparoscopy was conducted in 1 case [17], and EUS-FNA was performed with successful histological diagnosis in only 1 case [5]. In the remaining 4 cases, the methodology for obtaining histological diagnosis was not described [11]. These findings strongly suggested that diagnosing tuberculous
lymphadenitis is considerably difficult without laparotomy or laparoscopy. EUS-FNA is a minimally invasive technique for obtaining histological samples from swollen abdominal lymph nodes [5]. However, there are several disadvantages to using EUS-FNA. One of the disadvantages is the possibility of tumor seeding during aspiration biopsy from a ‘malignant’ mass. Therefore, EUS-FNA may be contraindicated if the possibility of a malignant tumor has not been ruled out. However, many recent reports have suggested that the possibility of FNA-related tumor seeding by EUS is considerably lower than that by percutaneous ultrasonography [21–27]. Another disadvantage of EUS-FNA is the difficulty in obtaining an adequate sample for accurate diagnosis; the diagnostic accuracy of EUS-FNA is variable at 60–90 % [28–30]. Although variously sized needles (19–25 gauge) have been used for EUS-FNA, there is no evidence that using larger-sized needles can achieve a more accurate diagnosis. It is possible that the number of samples obtained by EUSFNA, rather than the size, may be associated with the accuracy of histological diagnosis. A previous report has suggested that at least 5 passes are required to ensure
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adequate cellularity for lymph nodes with a diagnostic sensitivity and specificity of 77 and 100 %, respectively [6]. We therefore performed EUS-FNA with 5 passes, and obtained an accurate histological diagnosis. In conclusion, we have presented here a case of obstructive jaundice caused by tuberculous lymphadenitis. Young age and a history of emigration from a country endemic for tuberculosis are diagnostic clues for tuberculous lymphadenitis in developed countries. EUS-FNA is a safe and minimally invasive method for histological confirmation of diagnosis and should be considered as the first choice for obtaining samples from lymph nodes around the CBD. Disclosures Conflict of Interest of interest.
The authors declare that they have no conflict
Human/Animal Rights All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2008(5). Informed Consent Informed consent was obtained from all patients for being included in the study.
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