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Mesenchymal hamartoma of the liver in adulthood: immunohistochemical profiles, clinical and histopathological features in two patients

Journal of Hepato-Biliary-Pancreatic Surgery volume 12, pages 502–507 (2005)Cite this article

Abstract

Mesenchymal hamartoma is an uncommon cystic mass of the liver which occurs primarily in children. There are a few reports of its occurrence in adulthood. Here, we present two cases in female patients, 54 and 51 years old. Radiological examinations in both patients showed multiple cystic lesions in the liver. Surgically, total cystectomy was performed in the first patient, while an unroofing procedure was done in the second patient (due to misdiagnosis of the lesion as a simple cyst of the liver). On microscopic examinations of the lesion in each patient, a multilocular cyst was observed, lined by flattened epithelium and surrounded by a mesenchymal component composed of mature connective tissue, arterial and venous vascular structures, peripheral nerve bundles, and ductal structures. An immunohistochemical panel consisting of desmin, smooth-muscle actin, S-100, vimentin, CD34, carcinoembryonic antigen, pancytokeratin, cytokeratin 7, cytokeratin 8, cytokeratin 17, cytokeratin 18, cytokeratin 19, and cytokeratin 20 was applied to paraffin sections. Immunoreactivity for cytokeratin 7 and cytokeratin 19 was observed in cystic epithelium and ductal structures. Focal and patchy desmin immunoreactivity was observed in connective tissue. S-100 was positive only in peripheral nerve bundles. In conclusion, mesenchymal hamartoma of the liver in adulthood is a localized tumoral abnormality that precedes birth, and which has delayed clinical presentation. These lesions seems to be related to a maturation process. During this period of maturation, immature edematous stroma rich in mucopolysaccharides may convert to mature paucicellular hyalinized connective tissue. This maturation process may be also related to loss of premalignant potential of these tumors.

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References

  1. CA De Maioribus KP Lally K Sim H Isaacs GH Mahour (1990) ArticleTitleMesenchymal hamartoma of the liver. A 35-year review Arch Surg 125 598–600 Occurrence Handle1:STN:280:By%2BB3szptlM%3D

    CAS  Google Scholar 

  2. JT Stocker KG Ihsak (1983) ArticleTitleMesenchymal hamartoma of the liver: report of 30 cases and review of the literature Pediatr Pathol 1 245–67 Occurrence Handle6687279 Occurrence Handle1:STN:280:BiqD3sfivFE%3D

    PubMed  CAS  Google Scholar 

  3. PP Anthony (2000) Tumors and tumor-like lesions of the liver and biliary tract, aetiology, epidemiology and pathology RNM MacSween AD Burt BC Portman KG Ishak PJ Scheuer PP Anthony (Eds) Pathology of the liver Churchill Livingstone London 711–75

    Google Scholar 

  4. JR Cook JD Pfeifer LP Dehner (2002) ArticleTitleMesenchymal hamartoma of the liver in the adult: association with distinct clinical features and histological changes Hum Pathol 33 893–8 Occurrence Handle12378513 Occurrence Handle10.1053/hupa.2002.127442

    PubMed  Article  Google Scholar 

  5. G Papastratis H Margaris GN Zografos D Korkolis Z Mannika (2000) ArticleTitleMesenchymal hamartoma of the liver in an adult: a review of the literature Int J Clin Pract 54 552–4 Occurrence Handle11198739 Occurrence Handle1:STN:280:DC%2BD3M7gsl2itA%3D%3D

    PubMed  CAS  Google Scholar 

  6. S Megremis E Sfakianaki A Voludaki N Chroniaris (1994) ArticleTitleThe ultrasonographic appearance of a cystic mesenchymal hamartoma of the liver observed in middle-aged woman J Clin Ultrasound 22 338–41 Occurrence Handle8046044 Occurrence Handle1:STN:280:ByuA387ls1I%3D

    PubMed  CAS  Google Scholar 

  7. H Edmundson (1956) ArticleTitleDifferential diagnosis of tumors and tumorlike lesions of liver in infancy and childhood Am J Dis Child 91 168–86

    Google Scholar 

  8. K Washington (2004) Masses of the liver SE Mills D Carter JK Greenson HA Oberman V Reutwer MH Stoler (Eds) Sternberg's diagnostic surgical pathology, volume 2 EditionNumberFourth ed. Lippincott Williams & Wilkins Philadelphia 1705–74

    Google Scholar 

  9. CM Jennings CR Merril DN Slater (1987) ArticleTitleThe computed tomographic appearances of benign hepatic hamartoma Clin Radiol 38 103–4 Occurrence Handle3816057 Occurrence Handle10.1016/S0009-9260(87)80429-4 Occurrence Handle1:STN:280:BiiC3s3htV0%3D

    PubMed  Article  CAS  Google Scholar 

  10. T Brkic I Hrstic B Vucelic J Jakic-Razumovic M Skegro B Romic et al. (2003) ArticleTitleBenign mesenchymal liver hamartoma in an adult male: a case report and review of the literature Acta Med Austriaca 30 134–7 Occurrence Handle15055160 Occurrence Handle1:STN:280:DC%2BD2c7mt1Kjuw%3D%3D

    PubMed  CAS  Google Scholar 

  11. CB Drachenberg JC Papadimitriou MA Rivero Wood C. Distinctive (1991) ArticleTitlecase. Adult mesenchymal hamartoma of the liver: report of a case with light microscopic, FNA cytology, immunohistochemistry, and ultrastructural studies and review of the literature Mod pathol 4 392–5 Occurrence Handle2068067 Occurrence Handle1:STN:280:By6B1MnntlA%3D

    PubMed  CAS  Google Scholar 

  12. OH Gutierrez FA Burgener (1998) ArticleTitleMesenchymal hamartoma of the liver in an adult: radiologic diagnosis Gastrointest Radiol 13 341–4

    Google Scholar 

  13. PJ Grases M Matos-Villalobos F Arcia-Romero V Lecuna-Torres (1979) ArticleTitleMesenchymal hamartoma of the liver Gastroenterology 76 1466–9 Occurrence Handle437445 Occurrence Handle1:STN:280:CSaC2s%2FlvFY%3D

    PubMed  CAS  Google Scholar 

  14. KY Chau JW Ho PC Wu Yuen WK. Mesenchymal (1994) ArticleTitleHamartoma of liver in a man: comparison with cases in infants J Clin Pathol 47 864–6 Occurrence Handle7962662 Occurrence Handle1:STN:280:ByqD2M7mslM%3D

    PubMed  CAS  Google Scholar 

  15. TL Gramlich BW Killough AJ Garvin (1998) ArticleTitleMesenchymal hamartoma of the liver: report of a case in a 28-year-old Hum Pathol 19 991–2

    Google Scholar 

  16. A Alanen K Katevuo S Toikkanen (1987) ArticleTitleA non-cystic mesenchymal hamartoma of the liver — an unusual case of an unusual entity. Case report and review of the literature Bildgebung 56 181–4 Occurrence Handle3078435

    PubMed  Google Scholar 

  17. JH Chung KJ Cho DW Choi BH Lee JG Chi (1999) ArticleTitleSolid mesenchymal hamartoma of the liver in adult J Korean Med Sci 14 335–7 Occurrence Handle10402180 Occurrence Handle1:STN:280:DyaK1MzivVKgsg%3D%3D

    PubMed  CAS  Google Scholar 

  18. M Wada E Ohashi H Jin M Nishikawa S Shintani M Yamashita et al. (1992) ArticleTitleMesenchymal hamartoma of the liver: report of an adult case and review of the literature Intern Med 31 1370–5 Occurrence Handle1300174 Occurrence Handle1:STN:280:ByyB3sjms1w%3D

    PubMed  CAS  Google Scholar 

  19. KE Bove RI Blough S Soukup (1998) ArticleTitleThird report of t(19q)(13,4) in mesenchymal hamartoma of liver with comments on link to embryonal sarcoma Pediatr Dev Pathol 1 438–42 Occurrence Handle9688769 Occurrence Handle10.1007/s100249900060 Occurrence Handle1:STN:280:DyaK1MzjsFKgtA%3D%3D

    PubMed  Article  CAS  Google Scholar 

  20. GY Lauwers LD Grant WH Donnely AM Meloni RM Foss AA Sanberg et al. (1997) ArticleTitleHepatic undifferentiated (embryonal) sarcoma arising in a mesenchymal hamartoma Am J Surg Pathol 21 1248–54 Occurrence Handle9331300 Occurrence Handle10.1097/00000478-199710000-00018 Occurrence Handle1:STN:280:ByiH2s%2FosVQ%3D

    PubMed  Article  CAS  Google Scholar 

  21. WJ Lennington GF Gray SuffixJr DL Page (1993) ArticleTitleMesenchymal hamartoma of liver: a regional ischemic lesion of a sequestered lobe Am J Dis Child 147 193–6 Occurrence Handle8427244 Occurrence Handle1:STN:280:ByyC2M7it1M%3D

    PubMed  CAS  Google Scholar 

  22. JT Mascarello HF Krous (1992) ArticleTitleSecond report of a translocation involving 19q13,4 in a mesenchymal hamartoma of the liver Cancer Genet Cytogenet 58 141–2 Occurrence Handle1551077 Occurrence Handle10.1016/0165-4608(92)90100-M Occurrence Handle1:STN:280:By2B3c%2FjsVE%3D

    PubMed  Article  CAS  Google Scholar 

  23. F Speleman V De Telder KR De Potter (1989) ArticleTitleCytogenetic analysis of a mesenchymal hamartoma of the liver Cancer Genet Cytogenet 40 29–32 Occurrence Handle2758398 Occurrence Handle10.1016/0165-4608(89)90142-8 Occurrence Handle1:STN:280:BiaA3snms1Q%3D

    PubMed  Article  CAS  Google Scholar 

  24. MJ O'Sullivan PE Swanson J Knoll EM Taboada LP Dehner (2000) ArticleTitleUndifferentiated embryonal sarcoma with unusual features arising within mesenchymal hamartoma of the liver: report of a case and review of the literature Int J Clin Pract 54 482–9

    Google Scholar 

  25. D Von Schweinitz BG Dammeier S Gluer (1999) ArticleTitleMesenchymal hamartoma of the liver — new insight into histogenesis J Pediatr Surg 34 552–4

    Google Scholar 

  26. GM Azar N Kutin E Kahn (2003) ArticleTitleUnusual hepatic tumor with features of mesenchymal hamartoma and congenital solitary nonparasitic cyst Pediatr Dev Pathol 6 265–9 Occurrence Handle12679872 Occurrence Handle10.1007/s10024-003-7073-8

    PubMed  Article  Google Scholar 

  27. N Katkhouda E Mavor J Gugenheim J Mouiel (2000) ArticleTitleLaparoscopic management of benign cystic lesions of the liver J Hepatobiliary Pancreat Surg 7 212–7 Occurrence Handle10982616 Occurrence Handle10.1007/s005340050178 Occurrence Handle1:STN:280:DC%2BD3cvotFyqug%3D%3D

    PubMed  Article  CAS  Google Scholar 

  28. LP Dehner SL Ewing HW Summer (1975) ArticleTitleInfantile mesenchymal hamartoma of the liver: histologic and ultrastructural observations Arch Pathol 99 379–82 Occurrence Handle1170838 Occurrence Handle1:STN:280:CSqB3cvmtV0%3D

    PubMed  CAS  Google Scholar 

  29. M Yamamoto H Hagihara M Mogaki (1994) ArticleTitleAdult mesenchymal hamartoma of the liver mimicking bile duct cystadenoma J Gastroenterol 29 518–24 Occurrence Handle7951866 Occurrence Handle1:STN:280:ByqD3sbls1E%3D

    PubMed  CAS  Google Scholar 

  30. H Sawai T Manabe Y Yamanaka S Kurahashi A Kamiya (1998) ArticleTitleAngiomyolipoma of the liver: case report and collective review of cases diagnosed from fine needle aspiration biopsy specimens J Hepatobiliary Pancreat Surg 5 333–8 Occurrence Handle9880784 Occurrence Handle10.1007/s005340050055 Occurrence Handle1:STN:280:DyaK1M%2FpvFahtQ%3D%3D

    PubMed  Article  CAS  Google Scholar 

  31. JW Catto M Madan DJ Alexander (1999) ArticleTitleHepatobiliary cystadenoma presenting with intermittent inferior vena caval obstruction J Hepatobiliary Pancreat Surg 6 324–6 Occurrence Handle10526070 Occurrence Handle10.1007/s005340050125 Occurrence Handle1:STN:280:DyaK1Mvlslegsw%3D%3D

    PubMed  Article  CAS  Google Scholar 

  32. JSJ Brooks (2004) Disorders of soft tissue SE Mills D Carter JK Greenson HA Oberman V Reutwer MH Stoler (Eds) Sternberg's diagnostic surgicla pathology, volume 2 EditionNumberFourth ed. Lippincott Williams & Wilkins Philadelphia 137–222

    Google Scholar 

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Author information

Affiliations

  1. Pathology Department, Kocaeli University School of Medicine, Kocaeli, Turkey

    Gurbuz Yesim & Turan Gupse

  2. General Surgery Department, Kocaeli University School of Medicine, Kocaeli, Turkey

    Utkan Zafer & Alponat Ahmet

Authors
  1. Gurbuz Yesim
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  2. Turan Gupse
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  3. Utkan Zafer
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  4. Alponat Ahmet
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Yesim, G., Gupse, T., Zafer, U. et al. Mesenchymal hamartoma of the liver in adulthood: immunohistochemical profiles, clinical and histopathological features in two patients. J Hepatobiliary Pancreat Surg 12, 502–507 (2005). https://doi.org/10.1007/s00534-005-1025-9

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  • DOI: https://doi.org/10.1007/s00534-005-1025-9

Key words

  • Liver
  • Mesenchymal hamartoma
  • Immunohistochemistry