Liver International. 2018;38:1875. wileyonlinelibrary.com/journal/liv © 2018 John Wiley & Sons A/S. | 1875
Published by John Wiley & Sons Ltd DOI: 10.1111/liv.13898
L I V E R I N T E R N A T I O N A L I M A G E S
Recurrent hepatic mesenchymal hamartoma with osseous metaplasia
Nur Rahadiani
1| Marini Stephanie
1| Juan Putra
21Department of Anatomical Pathology, Cipto Mangunkusumo Hospital, University of Indonesia, Jakarta, Indonesia
2Department of Pathology, Harvard Medical School, Boston Children’s Hospital, Boston, MA, USA
Correspondence
Juan Putra, Department of Pathology, Boston Children’s Hospital, Harvard Medical School, Boston, MA, USA.
Email: [email protected]
Per our institutional policy, an informed consent was not required for this article as the patient was anonymous without identifiable or unique characteristics mentioned in the manuscript.
A 7- year- old Asian male presented with abdominal pain and en- larging abdomen status post- partial hepatectomy for mesenchymal hamartoma (MH) 2 years ago. The lesion was present at the resec- tion margin during the initial procedure. Current imaging studies revealed a 4 cm hepatic lesion in segment 5 and 8, suggestive of recurrent MH. The patient underwent partial hepatectomy and
macroscopic examination of the surgical specimen demonstrated a well- circumscribed, tan- white lesion with both solid and cystic components (Figure 1A). Histologically, the lesion showed a myx- omatous mesenchymal cell proliferation with cystic dilatation of bile ducts and entrapped hepatocytes, characteristic of hepatic MH (Figure 1B). Focal white and firm area was identified at the edge of the lesion (Figure 2A) which histologically corresponded to lamellar bone with marrow formation, consistent with an osseous metaplasia.
Hepatic MH is a benign tumour which is predominantly seen within the first 2 years of life.1 Its pathogenesis is unclear, however, it shares re- current chromosomal abnormalities involving chromosome 19q13.4 with its malignant counterpart, undifferentiated embryonal sarcoma of the liver.1,2 Resection with a clear margin is the treatment of choice whenever possible. To our knowledge, this is the first case of recurrent hepatic MH with osseous metaplasia. The heterotropic bone formation is most likely a secondary reaction from previous procedure3 as it was located at the periphery of the lesion and bone is not known to be a MH component.
Recognizing this phenomenon is important to distinguish it from other entities, particularly mesenchymal component of hepatoblastoma.
CONFLIC T OF INTEREST
The authors have no financial disclosure or conflict of interest.
ORCID
Juan Putra http://orcid.org/0000-0002-3487-4641
REFERENCES
1. Siddiqui MA, McKenna BJ. Hepatic mesenchymal hamartoma: a short review. Arch Pathol Lab Med. 2006;130:1567-1569.
2. Putra J, Ornvold K. Undifferentiated embryonal sarcoma of the liver:
a concise review. Arch Pathol Lab Med. 2015;139:269-273.
3. Hogan NM, Caffrey E, Curran S, Sheehan M, Joyce MR. Heterotropic ossification of the abdominal wall. Int J Surg Case Rep. 2012;3:489-491.
F I G U R E 2 (A) Further sectioning demonstrates a focal firm, white area (arrow) at the periphery of the lesion; (B) histologically, this area corresponds to lamellar bone and marrow formation, in keeping with a heterotropic ossification (Hematoxylin and eosin, 10X)
(A) (B)
F I G U R E 1 A, Serial sectioning of the hepatectomy specimen shows a well- demarcated, unencapsulated, tan- white, multinodular hepatic lesion with solid and cystic components. B, The hepatic lesion is predominantly comprised of a loose mesenchymal proliferation with cystically dilated bile ducts and entrapped hepatocytes (arrowheads) (Haematoxylin and eosin, 4X)
(A) (B)
