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Focal nodular hyperplasia in pediatric allogeneic hematopoietic cell transplant: case series

Bone Marrow Transplantation volume 45, pages 1357–1359 (2010)Cite this article

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Focal nodular hyperplasia (FNH) is an uncommon benign hepatic tumor. In the general population it is difficult to quantify the incidence; however, it is estimated to account for 8% of all primary liver tumors1 and 0.02% of pediatric liver tumors.2 FNH is often found incidentally, although some patients may present with vague abdominal complaints. It is rarely reported in children. The etiology is not clearly understood. It is speculated that it develops as a hyperplastic response to a pre-existing arterial malformation. Diagnosis based on radiographic findings can be difficult and needle biopsy may be necessary to confirm the diagnosis.1 It is unclear whether patients who have received chemotherapy and TBI as conditioning for hematopoietic SCT (HSCT) are at increased risk for the development of FNH. However, it is important for clinicians caring for these patients to be aware of this entity, as HSCT patients are at risk for hepatic complications. The following case series describes three pediatric patients post HSCT patients with FNH.

A 20-year-old female patient had been diagnosed with ALL at 3 years of age. She was treated on the Pediatric Oncology Group (POG) protocol 8699. She relapsed 3 years after the completion of therapy, was re-induced with chemotherapy and achieved a second remission. She underwent an allogeneic HSCT with a seven of eight HLA-matched unrelated donor. Conditioning consisted of cytarabine (18 gm/m2), CY (2.7 gm/m2) and TBI to a total dose of 1400 cGy with partial shielding to the lungs, the liver and the kidneys. GVHD prophylaxis was with partial T-cell depletion of the BM graft with Ab/C′ and CYA, as previously described by Casper et al.3 She had no evidence of sinusoidal obstruction syndrome post HSCT. She had stage 1 acute skin GVHD and chronic mild, limited skin GVHD, which resolved completely. Fourteen years after HSCT she presented with abdominal distention, which began 5 months before her clinic visit. Her medications included chlordiazepoxide, clidinium bromide, calcium, fiber, multi-vitamin and oral contraceptives. She had an abdominal computed tomography (CT) and magnetic resonance imaging (MRI) of her liver, which showed a 4 cm × 5 cm mass in her left lobe, with a central scar and a nodular lesion surrounding the scar. A second lesion was found in the right lobe, which enhanced with contrast on both the CT and MRI. A small liver cyst and small lesion in the right lobe consistent with a hemangioma were also seen. Owing to her history of malignancy and risk of secondary neoplasm, an open liver biopsy was performed. Pathology confirmed the diagnosis of FNH, multi focal. Owing to continued abdominal pain and the inflammatory response noted, complete resection of these lesions by a formal left lateral segmentectomy was performed. The patient tolerated these procedures without complications and made a full recovery. She remains off oral contraception because of the possible association between FNH and oral contraceptions. She had a follow-up MRI of her liver 6 months post-op, which was normal without focal abnormality.

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Acknowledgements

We would like to thank the entire Blood and Marrow transplant team for their continued care and support of HSCT patients post transplant. We would also like to thank the Midwest Athletes Against Cancer (MACC) Fund for support of this work.

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  1. Division of Pediatric Blood and Marrow Transplant, Division of Pediatric Radiology, Medical College of Wisconsin, Children's Hospital of Wisconsin, Milwaukee, WI, USA

    L Anderson, D Gregg, D Margolis, J Casper & J Talano

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  1. L Anderson
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  2. D Gregg
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  3. D Margolis
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  4. J Casper
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  5. J Talano
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Correspondence to L Anderson.

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Anderson, L., Gregg, D., Margolis, D. et al. Focal nodular hyperplasia in pediatric allogeneic hematopoietic cell transplant: case series. Bone Marrow Transplant 45, 1357–1359 (2010). https://doi.org/10.1038/bmt.2009.336

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