Benign Multicystic Mesothelioma by shade1314


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CASE 146

                                                                                  Case 146: Benign Multicystic

                     Phillip J. Koo, MD
                                                                                            History      A 27-year-old gravida 1, para 1 woman who had a history
                     John S. Wills, MD
                                                                                                         of pelvic inflammatory disease presented to the emergency
                                                                                                         department with lower abdominal pain, nausea, vomiting,
                                                                                                         and chills. She began menstruating the night before pre-
                                                                                                         sentation and reported an acute exacerbation of her
                                                                                                         chronic abdominal pain, which typically worsened with
                                                                                                         her menses. Her surgical history included one caesarean
                                                                                                         section and laparoscopic excision of a tubo-ovarian ab-
                                                                                                         scess. She was afebrile. Physical examination revealed
                                                                                                         mild diffuse abdominal tenderness and tenderness of the
                                                                                                         cervix. Laboratory findings revealed a white blood cell
                                                                                                         count of 17 000 cells per cubic millimeter. (A normal white
                                                                                                         blood cell count is between 4800 and 11 000 cells per cubic
                                                                                                            Ultrasonography (US) of the pelvis was performed. A
                                                                                                         computed tomographic (CT) examination of the abdomen
                                                                                                         and pelvis was subsequently performed after intravenous
                                                                                                         administration of 125 mL of iohexol (Omnipaque; Amer-
                                                                                                         sham Health, Princeton, NJ). We acquired 5-mm axial
                                                                                                         sections and performed 3-mm coronal reconstruction.

                                                                                                                                 This finding was consistent with a diagno-
                                                                                   Imaging Findings
                                                                                                                                 sis of benign multicystic mesothelioma.
                                                                                  US of the pelvis revealed a multicystic             Benign multicystic mesothelioma,
                                                                                  mass within the left adnexa posterior to       also referred to as cystic mesothelioma or
                                                                                  the uterus (Fig 1). The left ovary was         multilocular peritoneal inclusion cyst, is
                                                                                  normal. Doppler US revealed no vascu-          an intermediate-grade neoplasm of the
                                                                                  lar flow to the mass, which extended to         mesothelial cells of the peritoneum. It
                                                                                  the right lower quadrant of the abdo-          tends to recur locally, making it more ag-
                                                                                  men (Fig 2). Oral and intravenous con-         gressive than benign adenomatoid tu-
                                                                                  trast material– enhanced CT of the ab-         mors, which arise from mesothelial cells
                                                                                  domen and pelvis depicted a complex            in the genital tract; however, it is more
                                                                                  low-attenuation multicystic mass in the        benign than malignant peritoneal me-
                     Part one of this case appeared 4 months previously and may
                                                                                  pelvis, with noncalcified septa, that ex-       sotheliomas (1). Unlike malignant me-
                     contain larger images.
                                                                                  tended to the right upper quadrant of          sothelioma, cystic mesothelioma is not as-
                     Published online                                             the abdomen (Figs 3, 4). The coronal           sociated with prior asbestos exposure
                     10.1148/radiol.2513071235                                    image shows the displacement of multi-         (1). Although the tumor does not metas-
                     Radiology 2009; 251:944 –946                                 ple small-bowel loops to the left, with no     tasize, there is a high recurrence rate that
                                                                                  scalloping of the liver or spleen (Fig 4).     has been reported to be 27%–75% in the
                       From the Department of Radiology, Pennsylvania Hospi-
                                                                                                                                 3 months to 19 years after initial resec-
                     tal, University of Pennsylvania Health System, 800 Spruce
                     St, Philadelphia, PA 19107. Received July 13, 2007; revi-                                                   tion (2).
                     sion requested September 10; revision received October                                                           Multicystic mesothelioma has a ten-
                     8; accepted December 17; final version accepted Febru-        Laparotomy revealed a widespread thin-         dency to occur in young to middle-aged
                     ary 19, 2008. Address correspondence to P.J.K.               walled mass with multiple thin-walled          women, often those who present with
                     (e-mail: ).                              fluid-filled cysts attached to multiple peri-    abdominal or pelvic pain, distention,
                     Authors stated no financial relationship to disclose.         toneal surfaces (Fig 5). Pathologic analy-     and/or mass (2). Men are also affected.
                                                                                  sis revealed cysts lined with mesothelial      However, there is a substantial female
                         RSNA, 2009                                               cells that contained watery secretions.        predominance, as Takenouchi et al (3)

                     944                                                                                       ▪ Radiology: Volume 251: Number 3—June 2009
DIAGNOSIS PLEASE: Benign Multicystic Mesothelioma                                                                                                         Koo and Wills

  Figures 1, 2

  Figure 1: Sagittal transabdominal US image of the left adnexa reveals a multi-    Figure 2: Axial transabdominal Doppler US image of the right lower quadrant of the
  cystic mass (arrows) and a normal left ovary ( ).                                 abdomen shows the multicystic mass, with no color flow to the multiple septa (arrow).

  Figure 3                                                                                                          Figure 4

  Figure 3: Oral (20 mL iohexol diluted with 430 mL of water) and intravenous contrast-enhanced axial CT
  image of the pelvis reveals multiple septa (arrowheads) within the fluid-attenuation mass.

reported 81.2% of cases occurred in                         nant peritoneal mesothelioma, primary
women. There are no proved risk fac-                        papillary serous carcinoma of the peri-
tors for cystic mesothelioma, but cysts                     toneum, lymphangioma, teratoma, and                     Figure 4: Oral and intravenous contrast-en-
are commonly found at sites of prior                        other mesenteric cysts. Loculated as-                   hanced coronal CT image of the abdomen and
surgery or pelvic inflammatory disease                       cites, which occurs secondary to adhe-                  pelvis shows multiple septa (arrows) in the fluid-
(4). Hormonal sensitivity of the disease                    sions or peritonitis, usually demon-                    attenuation mass, which extends from the pelvis to
has been discussed in the literature,                       strates irregular borders and is sur-                   the right upper quadrant of the abdomen and dis-
since the disease is most often seen in                     rounded by bowel loops and abdominal                    places multiple small-bowel loops to the left. No
women of childbearing age and since                         and/or pelvic organs, as opposed to                     scalloping of the liver or spleen is shown.
cyst size has been shown to decrease                        cysts, which tend to displace adjacent
after treatment with a gonadotropin-re-                     structures (6). Peritoneal carcinomato-
leasing hormone analogue agonist and                        sis would likely contain more associated              liver and spleen has been described as a
tamoxifen (5).                                              imaging findings—such as adenopathy,                   diagnostic sign in the identification of
    The differential diagnosis included                     peritoneal tumor deposits, and/or le-                 the mucinous fluid seen in patients with
loculated ascites, peritoneal carcinoma-                    sions—that can be localized to the pri-               pseudomyxoma peritonei (7). Malig-
tosis, pseudomyxoma peritonei, malig-                       mary neoplastic organ. Scalloping of the              nant peritoneal mesothelioma is associ-

Radiology: Volume 251: Number 3—June 2009 ▪                                                                                                   945
DIAGNOSIS PLEASE: Benign Multicystic Mesothelioma                                                                                                Koo and Wills

 Figure 5                                              Wilkinson EJ. Cystic mesothelioma of the         Bradley S. Gluck, MD, Southampton, NY
                                                       peritoneum. Radiology 1989;170(2):333–           Navraj S. Grewal, MD, Elmhurst, Ill
                                                       337.                                             Pramod K. Gupta, MD, Plano, Tex
                                                                                                        Yuusuke Hirokawa, MD, Kyoto, Japan
                                                    3. Takenouchi Y, Oda K, Takahara O, et al.
                                                                                                        Teeranan Intharapat, MD, Hat-Yai, Songkhla,
                                                       Report of a case of benign cystic mesotheli-
                                                       oma. Am J Gastroenterol 1995;90(7):1165–
                                                                                                        Takanori Kikuchi, MD, PhD, Matsuyama,
                                                                                                              Ehime, Japan
                                                    4. Datta RV, Paty PB. Cystic mesothelioma of        Stefanos Lachanis, MD, Athens, Greece
                                                       the peritoneum. Eur J Surg Oncol 1997;           Mario A. Laguna, MD, Milwaukee, Wis
                                                       23(5):461– 462.                                  Jaume Llauger, MD, Barcelona, Spain
                                                                                                        Chikara Maeda, MD, Kyoto, Japan
                                                    5. Jerbi M, Hidar S, Ziadi S, Khairi H. Benign
                                                                                                        Yoji Maetani, MD, Kyoto, Japan
                                                       multicystic peritoneal mesothelioma. Int J
                                                                                                        Naganathan B. Mani, MD, Clayton, Mo
                                                       Gynaecol Obstet 2006;93(3):267–268.
                                                                                                        Andrew C. Mason, MBBCh, Vancouver, British
                                                    6. Hanbidge AE, Lynch D, Wilson SR. US of the             Columbia, Canada
                                                       peritoneum. RadioGraphics 2003;23(3):663–        Arash Meshksar, MD, Shiraz, Fars, Islamic Repub-
                                                       684.                                                   lic of Iran
                                                                                                        Manabu Minami, MD, PhD, Yokohama, Japan
 Figure 5: Intraoperative photograph shows          7. Sulkin TV, O’Neill H, Amin AI, Moran B. CT       Masayuki Miyajima, Fukushima, Japan
 multiple watery fluid-filled cysts (arrows) along       in pseudomyxoma peritonei: a review of 17        Aldo Morra, Sr, MD, PhD, Albignasego, Padova,
 the peritoneal surface.                               cases. Clin Radiol 2002;57(7):608 – 613.               Italy
                                                    8. Puvaneswary M, Proietto T. Primary papil-        Seyed Ali Nabavizadeh, MD, Shiraz, Fars, Islamic
                                                       lary serous carcinoma of the peritoneum:               Republic of Iran
ated with prior asbestos exposure most                 four cases and review of computed tomogra-       Kyoko Nagai, Yokohama, Japan
                                                       phy findings. Australas Radiol 2004;48(3):        Tammam N. Nehme, MD, East Wenatchee,
often in middle-aged men and manifests                                                                        Wash
                                                       421– 425.
in the form of pleural abnormalities and                                                                Mizuki Nishino, MD, Boston, Mass
                                                    9. Stafford-Johnson DB, Bree RL, Francis IR,        Hiroshi Nobusawa, MD, PhD, Ota, Tokyo, Japan
peritoneal, omental and/or mesenteric
                                                       Korobkin M. CT appearance of primary papil-      Hakmin Park, MD, Ann Arbor, Mich
masses (8). Primary papillary serous                   lary serous carcinoma of the peritoneum. AJR     Ilias Primetis, MD, Athens, Greece
carcinoma of the peritoneum is associ-                 Am J Roentgenol 1998;171(3):687– 689.            Mantosh S. Rattan, MD, Miami, Fla
ated with ascites, peritoneal and omen-            10. Wong WL, Johns TA, Herlihy WG, Martin            Matthew C. Rheinboldt, MD, Nashville, Tenn
tal masses without ovarian masses, and                 HL. Best cases from the AFIP: multicystic me-    Tsutomu Sakamoto, MD, Tokyo, Japan
no detectable primary site elsewhere                   sothelioma. RadioGraphics 2004;24(1):247–        Steven M. Schultz, MD, Fort Worth, Tex
(8). Peritoneal and omental calcifica-                  250.                                             Matthew P. Shapiro, MD, Charlottesville, Va
                                                                                                        Muneesh Sharma, MD, Nassau, Bahamas
tions have also been described as indic-
                                                   Congratulations to the 51 individuals                Hideki Shima, MD, Tokyo, Japan
ative of this entity (9). The radiologic                                                                Taro Shimono, MD, Osaka, Sayama, Japan
                                                   and three resident groups that submit-
appearance of lymphangioma is often                                                                     James D. Sprinkle, Jr, MD, Spotsylvania, Va
                                                   ted the most likely diagnosis (benign
identical to that of multicystic mesothe-                                                               Evan Stein, MD, PhD, New York, NY
                                                   multicystic mesothelioma) for Diagnosis
lioma; however, lymphangioma occurs                                                                     Yumiko Oishi Tanaka, MD, Tsukuba, Ibaraki,
                                                   Please, Case 146. The names and loca-                      Japan
in younger patients, and the cysts are
                                                   tions of the individuals and resident                Yoshito Tsushima, MD, Maebashi, Gunma, Japan
known to be filled with chylous fluid
                                                   groups, as submitted, are as follows:                Publio C. Viana, MD, Sao Paulo, Brazil
(10). Fat and calcifications can be de-                                                                  Silvio A. Vollmer, MD, Cipolletti, Rio Negro,
tected in patients with teratomas; how-            Individual responses                                       Argentina
ever, they are not seen in patients with                                                                Navid A. Zenooz, MD, Hamden, Conn
                                                   Guis S. Astacio, MD, Rio de Janeiro, Brazil
multicystic mesothelioma. Mesenteric                                                                    Resident group responses
                                                   Fahad Azzumeea, MBBS, Toronto, Ontario,
cysts are more commonly unilocular,                     Canada
with no detectable septa (10). Thus, the                                                                Baylor University Medical Center Radiology Res-
                                                   Eric L. Bressler, MD, Minnetonka, Minn
                                                                                                             idents, Dallas, Tex
most likely diagnosis, given this pa-              Douglas C. Brown, MD, Virginia Beach, Va
                                                                                                        University Hospital La Paz Residents, Madrid,
tient’s history and imaging findings, was           Rogerio Caldana, MD, Sao Paulo, Brazil
benign multicystic mesothelioma.                   Marco A. Cura, MD, San Antonio, Tex
                                                                                                        Virginia Commonwealth University Radiology
                                                   Thaworn Dendumrongsup, MD, Songkhla, Thai-
                                                                                                             Residents, Richmond, Va
References                                              land
                                                                                                        Yasushi Kawata, MD, Akita, Japan, submitted
 1. Weiss SW, Tavassoli FA. Multicystic meso-      Mohamed Eltomey, MD, Tanta, Elgharbia, Egypt
                                                                                                             the most likely diagnosis for Diagnosis
    thelioma: an analysis of pathologic findings    Eduardo P. Eyheremendy, MD, Buenos Aires,
                                                                                                             Please, Case 142 (Susac syndrome) prior to
    and biologic behavior in 37 cases. Am J Surg        Argentina
                                                                                                             the deadline for submissions for that case
    Pathol 1988;12(10):737–746.                    Fabricio S. Feltrin, MD, Sao Paulo, Brazil
                                                                                                             but was inadvertently left off the list.
                                                   Ann S. Fulcher, MD, Midlothian, Va
 2. O’Neil JD, Ros PR, Storm BL, Buck JL,          Gilles Genin, Annecy, France

946                                                                                   ▪ Radiology: Volume 251: Number 3—June 2009

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