JBR–BTR, 2015, 98: 85-87.

Cysts in a Brunner’s Gland : A Clue to Diagnosis

Y.-K. Fan, Y.-P. Liu, Y.-L. Lin, W.-K. Su1

The appearance of cystic Brunner’s gland (BGHs) on computed tomography (CT) or magnetic resonance imaging (MRI) has only been reported in a very small number of cases. Imaging diagnosis of cystic BGHs is usually difficult. We present a case of cystic BGH and characterize it in conjunction with previously reported cases. We found that the of BGHs are smaller than those of other cystic duodenal lesions. The presence of cysts in BGHs can limit the differential diagnosis to cystic duodenal lesions, and our observations may assist others in the discrimina- tion of cystic BGHs from other cystic duodenal lesions.

Key-words: Brunner’s gland – Hamartoma – Duodenum — .

Brunner’s gland hamartomas (BGHs) are uncommon duodenal le- sions, and account for approximate- ly 5% of all duodenal masses (1). BGHs contain cysts on rare occa- sions, and only a very small number of cases of cystic BGH with accom- panying computed tomography (CT) or magnetic resonance imaging (MRI) documentation have been re- ported (2-4). Imaging diagnosis of BGHs is usually a challenge, due to their nonspecific appearance. The presence of cysts can limit the differ- ential diagnosis to cystic duodenal lesions. Characterization of the cysts in BGHs may facilitate their differen- tiation from other cystic duodenal le- sions. Herein, we present a case of duodenal cystic BGH and review the cases reported to date, to determine the differences between the cysts of Fig. 1. — Endoscopic findings. A submucosal tumor with BGHs and those of other cystic duo- ­intact mucosa (asterisk) in the second portion of the duodenum denal lesions. causes luminal narrowing.

Case report

A 39-year-old man who had been that the cells and lobules of the Discussion experiencing postprandial epigastric ­Brunner’s glands were enlarged, but pain and for three days vis- otherwise normal in appearance Brunner’s glands are normally lo- ited our emergency department. The with no cellular atypia (Fig. 3). On the cated in the duodenal submucosa, results of his physical examination, basis of the histologic findings, a and their concentration gradually de- laboratory tests, and abdominal so- ­diagnosis of a Brunner’s gland creases towards the distal part of the nography were unremarkable. An ­hamartoma was made. During hos- duodenum. These glands produce endoscopic examination of the up- pitalization, oral intake was withheld an alkaline secretion to protect the per revealed a and esomeprazole and metoclo- duodenum from by gastric shallow gastric ulcer and a submu- pramide were administered intrave- acid and optimize the pH for pancre- cosal tumor in the second portion of nously. Three days after admission, atic enzymes. The pathogenesis of the duodenum causing marked his and vomiting re- BGHs is unknown. BGHs are glandu- ­luminal narrowing (Fig. 1). Contrast- solved, and the patient was dis- lar or hamartomas, enhanced abdominal CT showed a charged from the hospital. Surgical rather than true . Because 2.2 × 4.5 cm mass with multifocal, excision of the duodenal hamartoma these lesions do not have features of cyst-like low densities occupying the was not performed. The patient has cellular atypia, the terms Brunner’s second portion of the duodenum followed up twice in the outpatient gland “” or “hamarto- (Fig. 2). An endoscopic biopsy of the department in the 1 month since his ma” are preferable to “.” duodenal tumor was performed and discharge, and remains symptom- The distinction between Brunner’s pathologic examination revealed free. gland hyperplasia and Brunner’s gland hamartoma is arbitrary. Ac- cording to the guidelines used at the Armed Forces Institute of Pathology (Washington DC, USA), if the lesion From: 1. Dpt of Radiology, Mackay Memorial Hospital, Taiwan, R.O.C. Address for correspondence: Dr Y.-K. Fan, Dpt of Radiology, Mackay Memorial Hospi- is less than 5 mm in size it is called a tal, N° 690, Sec. 2, Guangfu Rd, East Dist., Hsinchu, Taiwan 300, R.O.C. hyperplasia, and if it is greater than E-mail: [email protected] 5 mm it is called a hamartoma (5). 86 JBR–BTR, 2015, 98 (2)

Fig. 2. — Axial and coronal contrast-enhanced CT images. A mass (white arrows) in the second portion of duodenum contains multiple small cysts. Duodenal lumen: black and white arrowheads, : asterisks.

A B

Fig. 3. — Photomicrographs of the BGH (hematoxylin and eosin, original magnification× 80 in A and ×200 in B) shows proliferation of Brunner’s glands and cystic dilatation of a gland (asterisk in B).

BGHs may be symptomatic, and ­literature to date (2-4). These cysts agnosis because the differential di- symptomatic lesions are often larg- are dilated acini or ducts of Brunner’s­ agnosis is limited to cystic duodenal er. The most common presentations glands. The CT findings of the previ- lesions. Cysts in BGHs differ from in symptomatic patients are gastro- ously reported cases and the current those in other cystic duodenal le- intestinal hemorrhage (37%) and case are summarized in Table I. The sions. Duplication cysts and lymph- ­obstructive symptoms (37%) (6). In a smaller hamartomas (< 2 cm) con- angiomas are thin-walled cystic series of 27 patients, 70% of BGHs tain a solitary cyst, and the larger masses without mural nodules or were located in the duodenal bulb, ones (≥ 4 cm) manifest as a soft tis- soft tissue components. These cysts 26% in the second portion of the sue mass containing multiple cysts. are larger than those in cystic ­duodenum, and 4% in the third These cysts are round, ovoid, or BGHs (7, 8). In contrast with conven- ­portion, and most (89%) were pe- elongated in shape. We found that tional soft tissue and central nervous dunculated (6). the short axes of all cysts in these system schwannomas, cysts are un- Cysts can only be identified in BGHs do not exceed the diameter of usual in gastrointestinal schwanno- BGHs via CT or MRI on rare occa- the abdominal aorta at the same lev- mas (9). If present, they are fewer in sions. To our knowledge, only 4 cases el, in axial images. Diagnosis of number and larger than those in demonstrating the CT or MRI ap- BGHs is usually difficult, due to non- BGHs of similar size (4, 10). Cystic pearance of cystic BGHs have been specific imaging findings. However, change is not uncommon in gastro- reported in the English-language the presence of cysts is helpful in di- intestinal stromal tumors (GISTs), CYSTS IN A BRUNNER’S GLAND HAMARTOMA — FAN et al 87

Table I. — Summary of the CT findings of cystic BGHs in the current case and the literature.

Case Age/Sex Size of tumors (cm)a Number of cysts Reference 1 39/M 4.5 7 Current case 2 30/M 4.0 ≥ 10c (2) 3 49/F 1.8 1 (3) 4 66/F 6.0 ≥ 5c (3) 5 48/M < 2.0b 1 (4) a Largest dimension. b The exact size of the BGH is not available in case 5. However, the largest dimension of the hamartoma approximates the diameter of abdominal aorta, and the diameter of abdominal aorta is estimated to be less than 2 cm. c Because limited images are available in these cases, exact number of cysts cannot be determined.

and is more commonly seen in large References 8. Levy A.D., Cantisani V., Miettinen M.: tumors. Cysts in GISTs are signifi- Abdominal lymphangiomas: imaging cantly larger than those in BGHs (11, 1. Botsford T.W., Crowe P., Crocker D.W.: features with pathologic correlation. 12). The duodenum is an uncommon Tumors of the small intestine. A AJR Am J Roentgenol, 2004, 182: site for review of experience with 115 cases 1485-1491. 9. Levy A.D., Quiles A.M., Miettinen M., ­formation. In the cases reported by including a report of a rare case of malignant hemangio-endothelioma. Sobin L.H.: Gastrointestinal schwan­ McCowin and Federle (13), these Am J Surg, 1962, 103: 358-365. nomas:­ CT features with clinico­ pseudocysts were solitary and larger 2. Park B.J., Kim M.J., Lee J.H., Park pathologic correlation. AJR Am J than the cysts of BGHs, and in all S.S., Sung D.J., Cho S.B.: Cystic Roentgenol, 2005, 184: 797-802. ­patients, CT revealed other evidence Brunner’s gland hamartoma in the 10. Bayraktutan U., Kantarci M., of in addition to duode- duodenum: a case report. World J Ozgokce M., Aydinli B., nal pseudocysts. Cystic dystrophy in Gastroenterol, 2009, 15: 4980-4983. Atamanalp S.S., Sipal S.: Education heterotopic pancreas (CDHP), most 3. Hur S., Han J.K., Kim M.A., Bae J.M., and Imaging. Gastrointestinal: benign commonly located in the second Choi B.I.: Brunner’s gland hamartoma: cystic schwannoma localized in the gastroduodenal ligament, a rare case. portion of the duodenum, has a simi- computed tomographic findings with histopathologic correlation in 9 cases. J Gastroenterol Hepatol, 2012, 27: lar appearance to cystic BGHs. These J Comput Assist Tomogr, 2010, 34: 985. cysts are multiple and small, and are 543-547. 11. King D.M.: The radiology of gastro­ very similar to those seen in larger 4. Lee J., Park C.M., Kim K.A., Lee C.H., intestinal stromal tumours (GIST). cystic BGHs. However, inflammatory Choi J.W., Shin B.K., et al.: Cystic imaging, 2005, 5: 150-156. changes such as periduodenal ­lesions of the gastrointestinal tract: 12. Naitoh I., Okayama Y., Hirai M., ­edema, periduodenal effusion, and multimodality imaging with patho- Kitajima Y., Hayashi K., Okamoto T., enlarged lymph nodes are observed logic correlations. Korean J Radiol, et al.: Exophytic pedunculated in most cases with CDHP (14). 2010, 11: 457-468. gastrointestinal stromal tumor with 5. Patel N.D., Levy A.D., Mehrotra A.K., remarkable cystic change. J Gastro­ Sobin L.H.: Brunner’s gland hyper­ enterol, 2003, 38: 1181-1184. Conclusion plasia and hamartoma: imaging fea- 13. McCowin M.J., Federle M.P.: tures with clinicopathologic correla- Computed tomography of pancreatic The size of cysts is the key to diag- tion. AJR Am J Roentgenol, 2006, pseudocysts of the duodenum. AJR nosing cystic BGHs. The cysts in 187: 715-722. Am J Roentgenol, 1985, 145: 1003- BGHs are smaller than the cysts of 6. Levine J.A., Burgart L.J., Batts K.P., 1007. other cystic duodenal lesions, except Wang K.K.: Brunner’s gland hamarto- 14. Vullierme M.P., Vilgrain V., Flejou J.F., CDHP. When a mass containing a mas: clinical presentation and patho- Zins M., O’Toole D., Ruszniewski P., small solitary cyst or multiple small logical features of 27 cases. Am J et al.: Cystic dystrophy of the duodenal wall in the heterotopic cysts is found in the first and second Gastroenterol, 1995, 90: 290-294. 7. Macpherson R.I.: Gastrointestinal pancreas: radiopathological correla­ portion of the duodenum, without tract duplications: clinical, pathologic, tions. J Comput Assist Tomogr, 2000, evidence of periduodenal inflamma- etiologic, and radiologic consider- 24: 635-643. tory change or pancreatitis, cystic ations. Radiographics, 1993, 13: 1063- BGH should be considered. 1080.