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Bilateral Nephroblastoma with Dilated Cardiomyopathy As an Indication for Off-Protocol Treatment: a Case Report

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Bilateral Nephroblastoma with Dilated Cardiomyopathy As an Indication for Off-Protocol Treatment: a Case Report International Journal of Environmental Research and Public Health Case Report Bilateral Nephroblastoma with Dilated Cardiomyopathy as an Indication for Off-Protocol Treatment: A Case Report Patrycja Sosnowska-Sienkiewicz 1,*, Ewelina Gowin 2, Katarzyna Jo ´nczyk-Potoczna 3 , Przemysław Ma ´nkowski 1, Jan Godzi ´nski 4 and Danuta Januszkiewicz-Lewandowska 5 1 Department of Pediatric Surgery, Poznan University of Medical Sciences, 60-572 Poznan, Poland; [email protected] 2 Department of Health Promotion, Poznan University of Medical Sciences, 60-572 Poznan, Poland; [email protected] 3 Department of Pediatric Radiology, Poznan University of Medical Sciences, 60-572 Poznan, Poland; [email protected] 4 Department of Pediatric Traumatology and Emergency Medicine, Wroclaw Medical University, 50-041 Wroclaw, Poland; [email protected] 5 Department of Pediatric Oncology, Hematology and Transplantology, Poznan University of Medical Sciences, 60-572 Poznan, Poland; [email protected] * Correspondence: [email protected]; Tel.: +48-61-8491-578; Fax: +48-61-8491-228 Received: 9 November 2020; Accepted: 16 December 2020; Published: 18 December 2020 Abstract: Patients with a Wilms tumor are often admitted to the hospital accidentally, with an abdominal mass causing asymmetry of the abdominal wall. Hypertension accompanying a Wilms tumor occurs in about 10–27% of children, but cardiomyopathy associated with a Wilms tumor is very rarely described. This publication presents a case of a 9-month-old girl with a bilateral Wilms tumor accompanied by dilated cardiomyopathy since her initial cancer diagnosis, as well as her off-protocol treatment. The severe condition of the child forced the application of off-protocol treatment, i.e., accelerated resection of a larger tumor, which enabled the improvement of heart performance and made subsequent therapy possible. In the course of the presented treatment, a gradual normalization of cardiac ventricular function and contractility was observed. In conclusion, a massive abdominal tumor associated with abdominal compartment syndrome compromised the functioning of the cardiovascular system in the young child. Therefore, earlier removal of Wilms tumors in patients with heart failure should be considered. This may result in the improvement of cardiovascular function and the possibility of further therapy. Keywords: cardiomyopathy; children; cardiotoxicity; hypertension; Wilms tumor 1. Introduction Wilms tumors (nephroblastoma) are the most common kidney cancer in children [1]. More often, the tumor occurs in children with hemihypertrophy or children with another malformation of the genitourinary, cardiovascular, or musculoskeletal system [1]. Usually, the disease affects one kidney, but in 5–10% of cases, it is bilateral [2,3]. Wilms tumors are genetically heterogeneous and mutations in WT1, WTX, and CTNNB1 underlie the genetic basis of about one-third of Wilms tumors. Another gene mutation found in Wilms tumors is the AMER1 gene. Changes on the short arm of chromosome 11 where IGF2 and H19 genes are located were also associated with development of a Wilms tumor. Int. J. Environ. Res. Public Health 2020, 17, 9483; doi:10.3390/ijerph17249483 www.mdpi.com/journal/ijerph Int. J. Environ. Res. Public Health 2020, 17, x 2 of 6 Int.Patients J. Environ. with Res. PublicWilms Health tumors2020, 17 are, 9483 often admitted to the hospital accidentally with an abdominal 2 of 6 mass causing asymmetry of the abdominal wall. Other symptoms present at the time of diagnosis of a Wilms Patientstumor, such with Wilmsas malaise, tumors loss are of often appetite, admitted abdominal to the hospital pain accidentallyor hematuria, with occur an abdominal in 20–30% of patientsmass [1]. causing Hypertension asymmetry accomp of the abdominalanying a Wilms wall. Other tumor symptoms occurs in present about at 10–27% the time of of children diagnosis [4,5]. Cardiomyopathyof a Wilms tumor, associated such as with malaise, a Wilms loss of tumor appetite, is very abdominal rarely paindescribed or hematuria, [6–8]. occur in 20–30% ofChemotherapy patients [1]. Hypertension and surgery, accompanying and in some a cases Wilms also tumor radiation occurs intherapy, about 10–27% are an ofeffective children treatment [4,5]. [1]. ForCardiomyopathy bilateral tumors, associated nephron-sparing with a Wilms tumorsurgery, is very preferably rarely described bilateral, [6 –or8]. otherwise performed at least on theChemotherapy less involved and side, surgery, is the and current in some standard cases also radiation[9]. therapy, are an effective treatment [1]. ForThis bilateral publication tumors, presents nephron-sparing the case surgery,of a girl preferably with bilateral bilateral, Wilms or otherwise tumors accompanied performed at least by dilated on the less involved side, is the current standard [9]. cardiomyopathy since her initial cancer diagnosis, as well as her off-protocol treatment. This publication presents the case of a girl with bilateral Wilms tumors accompanied by dilated cardiomyopathy since her initial cancer diagnosis, as well as her off-protocol treatment. 2. Case Report 2. Case Report A 9-month-old girl was admitted to the Pediatric Oncology Department due to the abdominal mass noticedA 9-month-old by her parents girl was two admitted weeks to earlier. the Pediatric She had Oncology been born Department from a duesecond to the pregnancy abdominal at 39 weeksmass of gestation, noticed by herwith parents birth twoweight weeks of earlier. 3790 g She and had the been APGAR born from score a secondof 10. The pregnancy pregnancy at 39 weeks had gone withoutof gestation, any complications. with birth weight Her ofparents 3790 g and theolder APGAR brother score are of healthy. 10. The pregnancy had gone without anyA large complications. mass crossing Her parents the andmidline older of brother the ab aredomen healthy. was palpable at admission. The child A large mass crossing the midline of the abdomen was palpable at admission. The child presented presented low body weight (8600 g), lack of appetite, increased respiratory and heart rate, and low body weight (8600 g), lack of appetite, increased respiratory and heart rate, and increased blood increased blood pressure. Computer tomography (CT) of the abdomen showed bilateral kidney pressure. Computer tomography (CT) of the abdomen showed bilateral kidney tumors. On the right 3 tumors.side, On a heterogeneous the right side, pathological a heterogeneous mass of 11.2pathological10.4 15.8 mass cm (979.1of 11.2 cm ×3 10.4; acc. × to 15.8 calculation cm (979.1 formula cm ; acc. × × to calculationV = D cm formulaS cm W V cm= D 0.532)cm × S was cm extending × W cm from× 0.532) the diaphragmwas extending to the pelvis.from the The diaphragm tumor shifted to the × × × pelvis.the The liver tumor to the shifted left side. the In liver the middle to the partleft ofside. the In left the kidney, middle three part solid of pathologicalthe left kidney, lesions three of asolid pathologicalsimilar nature lesions were of visible—thea similar nature first one were located visible—the in the upper-middle first one located pole, 3.0 in 1.8the upper-middle3.2 cm (9.2 cm3), pole, × × 3.0 ×the 1.8 second× 3.2 cm one (9.2 located cm3), laterally the second from one the first,located 2.2 laterally1.6 1.9 from cm (3.6 the cm first,3), and 2.2 the× 1.6 third × 1.9 one cm slightly (3.6 cm3), × × 3 lower and on the side, measuring 0.6 1.0 1.2 cm (0.4 cm ) (Figure1). 3 and the third one slightly lower and on× the ×side, measuring 0.6 × 1.0 × 1.2 cm (0.4 cm ) (Figure 1). FigureFigure 1. 1.CTCT scan scan of of the the abdomen abdomen on admissionadmission to to the the hospital. hospital. LungLung CT CTdid did not not show show metastases. metastases. Echocardiograp Echocardiographyhy presented presented dilatation dilatation of cardiacof cardiac ventricles ventricles withwith reduced reduced ventricular ventricular function function and and impaired impaired cardiaccardiaccontractility—left contractility—left ventricle ventricle (LV) (LV) transverse transverse shortening fraction (FS%) was 10–12% (normal range 28–44%; mean 36%), and LV ejection fraction shortening fraction (FS%) was 10–12% (normal range 28–44%; mean 36%), and LV ejection fraction (EF%) was 38–40% (normal range 64–83%; average 74%). Coronary artery abnormalities were excluded. (EF%) was 38–40% (normal range 64–83%; average 74%). Coronary artery abnormalities were Electrocardiography (ECG) at rest showed normal sinus rhythm. The heart rate at rest was 170–180 beats excluded.per minute. Electrocardiography Twenty-four-hour (ECG) Holter at monitoringrest showed showed normal no sinus abnormalities. rhythm. The The heart girl hasrate rated at rest at was 170–1807–8 pointsbeats (Rossper minute. Heart Failure Twenty-four-hour Classification), Holter which indicates monitoring moderately showed severe no abnormalities. heart failure. The girl has ratedChemotherapy at 7–8 points with(Ross vincristine Heart Failure and actinomycin Classification), was prescribed which indicates (VCR 1.5 moderately mg/m2, Actinomycin severe heart failure.0.045 mg/kg, all drug doses due to body weight below 12 kg according to SIOP scheme were reduced to 67%).Chemotherapy Despite chemotherapy with vincristine and cardiac and acti treatmentnomycin including was prescribed spironolactone, (VCR captopril 1.5 mg/m and2,carvedilol, Actinomycin 0.045the mg/kg, child stillall drug presented doses tachycardia, due to body tachypnea weight be andlow moderate 12 kg according hypertension to SIOP with increasingscheme were levels reduced of to 67%). Despite chemotherapy and cardiac treatment including spironolactone, captopril and carvedilol, the child still presented tachycardia, tachypnea and moderate hypertension with increasing levels of B-type natriuretic peptide (BNP), lactate dehydrogenase (LDH), high-sensitivity troponin T (hscTnT) and plasma renin activity (PRA). Int. J. Environ. Res. Public Health 2020, 17, 9483 3 of 6 Int. J. Environ.B-type natriureticRes. Public Health peptide 2020 (BNP),, 17, x lactate dehydrogenase (LDH), high-sensitivity troponin T (hscTnT)3 of 6 and plasma renin activity (PRA).
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