Mesenteric Castleman Disease Mimicking Superior Mesenteric Artery Aneurysm Ji-Soo Lee, Yang Jin Park, and Young-Wook Kim, Seoul, Korea
Castleman disease (CD) is known as a lymphoproliferative disorder, which is most commonly located in the mediastinum. CD occurring in the mesentery is very rare. We report a case of CD in the mesentery, which is mimicking a superior mesenteric artery aneurysm on computed tomography image.
Castleman disease (CD) has been characterized as a lymphoproliferative disorder. Histologically, lesions of CD are divided into the hyaline vascular type, the plasma cell type, and the mixed type. The hyaline vascular type accounts for 85e90% of cases, and 90% of the cases are highly localized, and furthermore, this type is rarely multicentric.1 The hyaline vascular type of CD is most commonly located in the mediastinum, and 70% of patients with this type of CD are aged younger than 30-years.2 CD occurring in the mesentery is very rare and usually is asymptomatic.3 We describe a case of a hyaline vascular type CD in the mesentery, which is mimicking a superior mesenteric artery (SMA) aneurysm on computed tomography (CT) angiogram.
CASE REPORT A 66-year-old woman who had been taking antihypertensive medications for 20 years was referred to us with epigastric discomfort and suspected SMA aneurysm. At Conflict of Interest: No potential conflict of interest relevant to this article was reported. Division of Vascular Surgery, Department of Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea. Correspondence to: Young-Wook Kim, MD, FACS, Division of Vascular Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Irwon-ro #81, Gangnam-gu, Seoul 135-710, Korea; E-mail: [email protected] Ann Vasc Surg 2015; 29: 364.e15e364.e17 http://dx.doi.org/10.1016/j.avsg.2014.08.026 Ó 2015 Elsevier Inc. All rights reserved. Manuscript received: May 20, 2014; manuscript accepted: August 21, 2014; published online: November 22, 2014.
the time of presentation, vital signs were the following: blood pressure 121/80 mm Hg, pulse rate 71/min, body temperature 36.2 C, respiratory rate 20/min, and blood tests revealed no abnormality in liver or pancreatic enzyme levels. Contrast-enhanced abdominal CT revealed a 3.5-cm contrast-enhanced mass lesion in front of the SMA (Fig. 1A, B). On a sagittal view, it mimicked SMA aneurysm and was suspected of being connected to a branch of the SMA (Fig. 1C). On a follow-up CT scan 6 months later, the mass size was 3.3 4 cm2, which was not enlarged from the previous study. On laparotomy through the vertical midline incision, we found a 4 3 2.5-cm3-sized solid, wellcircumscribed mass close to the SMA (Fig. 2). At the gross appearance of the mass, it appeared to be a round, well-circumscribed, and hypervascular lesion. The cut surface was homogenously pink tan colored and had a rubbery consistency (Fig. 3). After complete surgical excision of the mass lesion, the patient has now been followed for 8 months without prescribing special medication.
DISCUSSION The etiology of CD is not well understood, but chronic inflammatory reaction against an unknown antigen, viral etiologies, or immunoregulation defects such as from human immunodeficiency virus (HIV) infection have been suspected causes of the excessive proliferation of B lymphocytes and plasma cells in lymphoid organs in CD patients. In the differential diagnosis of CD, lymphoma or thymoma in mediastinal CD, mesenteric lymphadenitis, tuberculosis, sarcoidosis, toxoplasmosis, cytomegalovirus, 364.e15
364.e16 Case reports
Fig. 1. Contrast-enhanced CT scan. (A) On axial view, 3.5 4 cm2-sized contrast-enhanced mass lesion was found in front of superior mesenteric artery. (B) On a reconstructed view, there shows a 3.3-cm sized
Annals of Vascular Surgery
contrast-enhanced mass lesion, which is a mimic to superior mesenteric artery aneurysm. (C) On a sagittal view, mass lesion seems to be communicating with a branch of mesenteric vessel.
Fig. 2. Operative finding: an oval-shaped, well-circumscribed, solid mesenteric mass was found close to the superior mesenteric artery, which has no vascular connection to the mesenteric vessel.
mononucleosis, HIV, and mesenteric tumors should be differentiated from mesenteric CD.4 A characteristic feature of homogenous and intense contrast enhancement of the mass lesion in unicentric hyaline vascular type CD is that it mimics aneurysm, particularly, on the reconstructed contrast-enhanced CT image. Meador and McLarney5 reported that abdominal or pelvic CD shows heterogenous appearance on CT, especially, in the case of large lesions (>5 cm).
Fig. 3. Gross and microscopic findings: (A) cut surface of the mass is pinkish colored with rubbery consistency; (B) microscopic findings (hematoxylin & eosin stain) of the mass shows a lymph node with vague lymphoid follicles with hyalinization of the germinal centers, which are suspective of hyaline vascular type of Castleman disease.
Vol. 29, No. 2, February 2015
In conclusion, localized forms of CD should be treated by complete surgical excision. Here, we report a case of mesenteric CD mimicking SMA aneurysm on contrast-enhanced CT image. REFERENCES 1. Aslan M, Bitiren M, Bolukbas C, et al. Mesenteric Castleman’s disease. Turk J Gastroenterol 2011;22:653e4. 2. Keller AR, Hochholzer L, Castleman B. Hyaline-vascular and plasma-cell types of giant lymph node hyperplasia of
Case reports 364.e17
the mediastinum and other locations. Cancer 1972;29: 670e83. 3. Gangopadhyay K, Mahasin ZZ, Kfoury H. Pathologic quiz case 2. Castleman disease (giant lymph node hyperplasia). Arch Otolaryngol Head Neck Surg 1997;123: 1137e9. 4. Inoue Y, Nakamura H, Yamazaki K, et al. Retroperitoneal Castleman’s tumors of hyaline vascular type: imaging study. Case report. Clin Imaging 1992;16:239e42. 5. Meador TL, McLarney JK. CT features of Castleman disease of the abdomen and pelvis. AJR Am J Roentgenol 2000;175: 115e8.
Castleman disease (CD) is known as a lymphoproliferative disorder, which is most commonly located in the mediastinum. CD occurring in the mesentery is very rare. We report a case of CD in the mesentery, which is mimicking a superior mesenteric artery aneurysm on computed tomography image.
Castleman disease (CD) has been characterized as a lymphoproliferative disorder. Histologically, lesions of CD are divided into the hyaline vascular type, the plasma cell type, and the mixed type. The hyaline vascular type accounts for 85e90% of cases, and 90% of the cases are highly localized, and furthermore, this type is rarely multicentric.1 The hyaline vascular type of CD is most commonly located in the mediastinum, and 70% of patients with this type of CD are aged younger than 30-years.2 CD occurring in the mesentery is very rare and usually is asymptomatic.3 We describe a case of a hyaline vascular type CD in the mesentery, which is mimicking a superior mesenteric artery (SMA) aneurysm on computed tomography (CT) angiogram.
CASE REPORT A 66-year-old woman who had been taking antihypertensive medications for 20 years was referred to us with epigastric discomfort and suspected SMA aneurysm. At Conflict of Interest: No potential conflict of interest relevant to this article was reported. Division of Vascular Surgery, Department of Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea. Correspondence to: Young-Wook Kim, MD, FACS, Division of Vascular Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Irwon-ro #81, Gangnam-gu, Seoul 135-710, Korea; E-mail: [email protected] Ann Vasc Surg 2015; 29: 364.e15e364.e17 http://dx.doi.org/10.1016/j.avsg.2014.08.026 Ó 2015 Elsevier Inc. All rights reserved. Manuscript received: May 20, 2014; manuscript accepted: August 21, 2014; published online: November 22, 2014.
the time of presentation, vital signs were the following: blood pressure 121/80 mm Hg, pulse rate 71/min, body temperature 36.2 C, respiratory rate 20/min, and blood tests revealed no abnormality in liver or pancreatic enzyme levels. Contrast-enhanced abdominal CT revealed a 3.5-cm contrast-enhanced mass lesion in front of the SMA (Fig. 1A, B). On a sagittal view, it mimicked SMA aneurysm and was suspected of being connected to a branch of the SMA (Fig. 1C). On a follow-up CT scan 6 months later, the mass size was 3.3 4 cm2, which was not enlarged from the previous study. On laparotomy through the vertical midline incision, we found a 4 3 2.5-cm3-sized solid, wellcircumscribed mass close to the SMA (Fig. 2). At the gross appearance of the mass, it appeared to be a round, well-circumscribed, and hypervascular lesion. The cut surface was homogenously pink tan colored and had a rubbery consistency (Fig. 3). After complete surgical excision of the mass lesion, the patient has now been followed for 8 months without prescribing special medication.
DISCUSSION The etiology of CD is not well understood, but chronic inflammatory reaction against an unknown antigen, viral etiologies, or immunoregulation defects such as from human immunodeficiency virus (HIV) infection have been suspected causes of the excessive proliferation of B lymphocytes and plasma cells in lymphoid organs in CD patients. In the differential diagnosis of CD, lymphoma or thymoma in mediastinal CD, mesenteric lymphadenitis, tuberculosis, sarcoidosis, toxoplasmosis, cytomegalovirus, 364.e15
364.e16 Case reports
Fig. 1. Contrast-enhanced CT scan. (A) On axial view, 3.5 4 cm2-sized contrast-enhanced mass lesion was found in front of superior mesenteric artery. (B) On a reconstructed view, there shows a 3.3-cm sized
Annals of Vascular Surgery
contrast-enhanced mass lesion, which is a mimic to superior mesenteric artery aneurysm. (C) On a sagittal view, mass lesion seems to be communicating with a branch of mesenteric vessel.
Fig. 2. Operative finding: an oval-shaped, well-circumscribed, solid mesenteric mass was found close to the superior mesenteric artery, which has no vascular connection to the mesenteric vessel.
mononucleosis, HIV, and mesenteric tumors should be differentiated from mesenteric CD.4 A characteristic feature of homogenous and intense contrast enhancement of the mass lesion in unicentric hyaline vascular type CD is that it mimics aneurysm, particularly, on the reconstructed contrast-enhanced CT image. Meador and McLarney5 reported that abdominal or pelvic CD shows heterogenous appearance on CT, especially, in the case of large lesions (>5 cm).
Fig. 3. Gross and microscopic findings: (A) cut surface of the mass is pinkish colored with rubbery consistency; (B) microscopic findings (hematoxylin & eosin stain) of the mass shows a lymph node with vague lymphoid follicles with hyalinization of the germinal centers, which are suspective of hyaline vascular type of Castleman disease.
Vol. 29, No. 2, February 2015
In conclusion, localized forms of CD should be treated by complete surgical excision. Here, we report a case of mesenteric CD mimicking SMA aneurysm on contrast-enhanced CT image. REFERENCES 1. Aslan M, Bitiren M, Bolukbas C, et al. Mesenteric Castleman’s disease. Turk J Gastroenterol 2011;22:653e4. 2. Keller AR, Hochholzer L, Castleman B. Hyaline-vascular and plasma-cell types of giant lymph node hyperplasia of
Case reports 364.e17
the mediastinum and other locations. Cancer 1972;29: 670e83. 3. Gangopadhyay K, Mahasin ZZ, Kfoury H. Pathologic quiz case 2. Castleman disease (giant lymph node hyperplasia). Arch Otolaryngol Head Neck Surg 1997;123: 1137e9. 4. Inoue Y, Nakamura H, Yamazaki K, et al. Retroperitoneal Castleman’s tumors of hyaline vascular type: imaging study. Case report. Clin Imaging 1992;16:239e42. 5. Meador TL, McLarney JK. CT features of Castleman disease of the abdomen and pelvis. AJR Am J Roentgenol 2000;175: 115e8.
