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133 KUWAIT MEDICAL JOURNAL June 2012 Case Report Hypertension and Hypoglycemia in a Child with Hepatoblastoma: A Case Report Nisreen M Khalifa, Rakesh Mittal, Sahar O Khalifa Pediatric Oncology Unit, Department of Medical Oncology, Kuwait Cancer Control Center, Kuwait ABSTRACT Kuwait Medical Journal 2012; 44 (2): 133 - 134 Hepatoblastoma is the most common malignant tumor in the pediatric age group. It is treated with chemotherapy and surgery with excellent results. In majority of the cases ,the course of treatment is uncomplicated. Rarely there could be unexpected complications like hypertension due to hypersecretion of renin or severe hypoglycemia due to production of insulin like substances by tumor cells. These episodes can complicate the management of the case. We report a rare case of hepatoblastoma in an eight-yearold child who had both the complications. To the best of our knowledge, this is the first such case to be reported in literature. KEY WORDS: chemotherapy, hepatoblastoma, hypertension, hypoglycemia INTRODUCTION Hepatoblastoma is a rare aggressive childhood tumor accounting for 75% of primary liver tumors in the pediatric age group. The five-year survival has increased to nearly 70% [1, 2] with the use of cisplatinbased chemotherapy and advanced surgical techniques. Paraneoplastic syndromes which can cause metabolic complications are rarely reported. We present the case of a hepatoblastoma patient who had hypertension, hypoglycemia, hypokalemia, hyonatremia and hypomagnesemia. In hepatoblastoma, there are anecdotal case reports of hypertension due to hyperreninemia [3] or hypoglycemia due to hyperinsulinemia [4] , but to the best of our knowledge, this is the first case that had so many complications at the same time. CASE REPORT An eight-year-old Indian boy presented with right upper abdominal pain. On examination there was hepatomagaly and ascites. Computerized tomography (CT scan) revealed multiple hypoechoic lesions in both lobes of the liver, the largest measuring 4 x 4.5 cm, with some lesions showing calcification (Fig. 1). The ascitic fluid was hemorrhagic but negative for malignant cells. The alfafetoprotein (AFP) was 1,231,760 IU/ml. Needle biopsy confirmed epithelial (fetal subtype) hepatoblastoma (Fig. 2). The CT- scan of the chest was negative for pulmonary metastasis. He was started on SIOPEL 3 high risk protocol and he tolerated the first course well. During the 2 nd course of chemotherapy, he had hypertensive encephalopathy with seizures. EEG, CSF study, CT brain were normal. Renal artery Doppler study was also normal. Plasma renin was 12 pmol/l (normal range 0.264 – 2.4 pmol/l) with normal plasma aldosterone level. After few days, he had unexplained recurrent hypoglycemic episodes. Plasma insulin level was 0.2 uU/ml (normal range 2.6 – 24.9) and insulin like growth factor – I (IGF-1) was < 5 nmol/l (range 10 – 66 for P1 Tanner stage). He also had persistent asymptomatic hyponatremia (Serum Na+ < 120 mmol/l), mild hypokalemia, and hypomagnesemia. His hypertension, hypoglycemia, and electrolytes were treated appropriately. He was continued on chemotherapy with weekly AFP monitoring. Unfortunately after three cycles of chemotherapy (day 29), AFP was rising (1,716,390 IU/ml) and US abdomen showed no response with increasing ascites. His parents took him to his native country where he died. DISCUSSION In the literature, only few cases were reported with hyperreninemia in non-renal non-hepatic malignant tumors [5, 6] , and non-malignant liver tumors [7] . In the case reported by Moritake et al [3] , a hepatoblastoma child had hypertension at presentation due to hyperreninemia. The child had neoadjuvant chemotherapy, tumor resection, and postoperative chemotherapy. Renin secretion in the tumor specimen was documented. In our case, due to various logistical reasons, tumor analysis for renin secretion could not be documented. Address correspondence to: Dr. Rakesh Mittal, MD DM, Consultant & In-charge, Pediatric Oncology, Department of Medical Oncology, Kuwait Cancer Control Center, P O Box 42262, Shuwaikh 70653, Kuwait. Tel : 965-24849100 – 4532, Fax: 965 – 24810007, E-mail: rakeshmittal55@hotmail.com
June 2012 KUWAIT MEDICAL JOURNAL 134 in a series of elderly patients with hepatocellular carcinoma or gastric carcinoma. No case report or series has been published in recent time in pediatric patients describing this complication. de Groot et al [10] described this phenomenon as over-expression of the IGF-II gene by tumor. The only treatment described in the literature for this complication is surgical removal of the tumor. Unfortunately our patient could not undergo any surgery due to poor response to chemotherapy and poor performance status. Presence of hypokalemia was also described by authors [6] , which was also present in our case. However, the severe hyponatremia and hypomagnesemia seen in this case was not described earlier.. Fig. 1: Axial CT scan of the abdomen showing multiple hypodense hepatic lesions involving both liver lobes. CONCLUSIONS We report this case to highlight the paraneoplastic manifestations of hepatoblastoma like hyperreninemia and the presence of insulin-like growth factor substances. To the best of our knowledge, there is no reported case of hepatoblastoma which had both hyperreninemia and severe hypoglycemia with electrolyte abnormalities. REFERENCES Fig. 2: H & E (x 400) Trabecule of cuboidal and polygonal malignant cells with eosinophilic cytoplasm and moderate mitosis mimicking hepatocytes. Moritake et al [3] reported drop in plasma renin activity after surgery and chemotherapy, but our patient did not respond to chemotherapy, hence did not undergo surgery. Literature analysis showed curative resection as the only definitive method of bringing down the plasma renin activity [5-7] . The second serious episode in our patient was attack of severe hypoglycemia. Ha et al [4] reported a case of hepatoblastoma in a 5-year-old boy who developed hypoglycemia and they documented low endogenous insulin and increased insulin-like activity (ILA) in the serum and tissue specimen and abnormalities for carbohydrate metabolism. In their case, the child presented with an episode of severe hypoglycemia while our patient developed hypoglycemia only during the administration of the second cycle of chemotherapy. This condition was described as non-islet-cell tumor hypoglycemia (NICTH) or hypoinsulinemic hypoglycemia related to high levels of high molecular weight IGF–II [8,9] , also referred to as “Big IGF-2”. Unfortunately, our lab did not have the facility to measure this type of IGF-2. Hijuka et al [9] reported a series of 44 adult patients in which presence of serum IGF-II due to non-islets tumor led to hypoglycemia. But this condition was reported 1. Roebuck DJ, Perilongo G. Hepatoblastoma: an oncological review. Pediatr Radiol 2006 ; 36:183-186. 2. Perilongo G, Maibach R, Shafford E, et al. Cisplatin versus cisplatin plus doxorubicin for standard-risk hepatoblastoma. N Engl J Med 2009; 22:1662-1670. 3. Moritake H, Taketomi A, Kamimura S, et al. Reninproducing hepatoblastoma. J Pediatr Hematol Oncol 2000; 22:78-80. 4. Ha K, Ikeda T, Okada S, et al. Hypoglycemia in a child with hepatoblastoma. Med Pediatr Oncol 1980; 8:335- 341. 5. Pursell RN, Quinlan PM. Secondary hypertension due to a renin-producing teratoma. Am J Hypertens 2003; 16:592-595. 6. Iwamoto H, Hirata S, Honda T, Fukasawa H, Kimura N, Hoshi K. Renin-producing serous cystoadenocarcinoma of the ovary: a case report. Eur J Gynaecol Oncol 2002; 23:183-186. 7. Cox JN, Paunier L, Vallotton MB, Humbert JR, Rohner A. Epithelial liver hamartoma, systemic arterial hypertension and renin hypersecretion. Virchows Archiv 1975; 366:15-26. 8. Fukuda I, Hizuka N, Ishikawa Y, et al. Clinical features of insulin-like growth factor-II producing non-isletcell tumor hypoglycemia. Growth Horm IGF Res 2006; 16:211-216. 9. Hizuka N, Fukuda I, Takano K, Okubo Y, Asakawa- Yasumoto K, Demura H. Serum insulin-like growth factor II in 44 patients with non-islet cell tumor hypoglycemia. Endocr J 1998; 45:S61-S65. 10. de Groot JW, Rikhof B, van Doorn J, et al. Non-islet cell tumour-induced hypoglycaemia: a review of the literature including two new cases. Endocr Relat Cancer 2007 ; 14:979-993.
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