Received: 12 April 2023  | Revised: 27 July 2023  | Accepted: 10 September 2023
DOI: 10.1002/ijgo.15157  
R E V I E W  A R T I C L E
O b s t e t r i c s
Disseminated adult Wilms tumor in pregnancy: Leveraging 
multidisciplinary care in a low- resource setting
Perez Sepenu1  |   Alim Swarray-D een2 |   Aba Scott3 |   Theodore K. Boafor2  |    
Winfred K. Baah4 |   Mathew K. Kyei5 |   Jerry Coleman2
1Department of Obstetrics & Gynecology, 
Maternal- Fetal Medicine Unit, Korle Bu Abstract
Teaching Hospital, Accra, Ghana Wilms tumor (WT) occurring in adults is rare and even much more rarely found to 
2Department of Obstetrics & Gynecology, 
University of Ghana Medical School, coexist with pregnancy. Clinical outcome in adults is worse overall compared with 
Accra, Ghana pediatric patients with WT and is often misdiagnosed with no standardized protocols 
3National Centre for Radiotherapy, for care guided by high- evidence clinical trials. We present a case of a 23- year- old 
Oncology and Nuclear Medicine, Korle Bu 
Teaching Hospital, Accra, Ghana woman diagnosed with WT who was found to be pregnant immediately following 
4Department of Medicine & Therapeutics, nephrectomy. Workup findings showed that she had disseminated disease but was 
University of Ghana Medical School, 
Accra, Ghana successfully managed in a multidisciplinary team setting with modified intrapartum 
5Department of Surgery, Urology Unit, chemotherapy followed by postpartum chemotherapy. In low-r esource settings, man-
University of Ghana Medical School, agement protocols for adult patients with WT can be individualized by multidiscipli-
Accra, Ghana
nary teams to leverage available resources for best outcomes.
Correspondence
Theodore K. Boafor, Department of K E Y W O R D S
Obstetrics and Gynecology, University of adult Wilms tumor, antenatal chemotherapy, multidisciplinary care, nephroblastoma, 
Ghana Medical School, College of Health pregnancy, renal tumor
Sciences, PO Box 4236, Korle Bu, Accra, 
Ghana.
Email: theodoreboafor@gmail.com, 
tboafor@ug.edu.gh
1  |  INTRODUC TION rarely seen in adults, with an incidence of 0.2 cases per million,1 and 
predominantly occurs in younger adults with a median diagnosis at 
It is rare for malignancies to coexist with pregnancy, with an inci- age 34 years.5 Although adult WT is histologically similar to pediatric 
dence of 1 in 1000 to 1 in 6000 pregnancies in high-r esource coun- nephroblastoma, the clinical presentation tends to differ, with adults 
tries.1 A coexisting malignancy during pregnancy can pose seemingly having a higher stage at diagnosis and poorer overall prognosis.6 
impossible clinical decisions for the remainder of the gestation pe- Women tend to have a higher survival rate compared with men.7
riod. Women face the choice between continuing the pregnancy, The coexistence of WT and pregnancy is extremely rare, with 
which can complicate treatment and allow for further disease only few cases have been reported in the literature.1
progression and or harm to the growing fetus, or terminating the Management of WT in adults is not currently standardized. 
pregnancy to allow for the timely administration of lifesaving treat- Generally, it follows the treatment modalities for children as rec-
ment.2 Although rare, the most common cancers diagnosed during ommended by Children's Oncology Group (COG) and International 
pregnancy include but are not limited to breast, melanoma, thyroid, Society of Pediatric Oncology (SIOP) protocols.5 When it occurs in 
cervical cancers, lymphomas, and very rarely Wilms tumor (WT).2–4 the setting of pregnancy, it becomes a quagmire of complex man-
WT, or nephroblastoma, constitutes 6% to 7% of all pediatric tu- agement choices that is not eased by preexisting treatment guide-
mors and is the most common primary renal tumor in children.5 It is lines or clinical trials to dictate the standard of care. We present a 
© 2023 International Federation of Gynecology and Obstetrics.
Int J Gynecol Obstet. 2023;00:1–6.  wileyonlinelibrary.com/journal/ijgo  | 1
2  |    SEPENU et al.
case of disseminated WT in a pregnant 23- year- old woman who was from renal function test were unremarkable, with a creatinine level 
managed by a multidisciplinary team (MDT) where chemotherapy of 59 mmol/L. Findings from liver function test and clotting profile 
was initiated antenatally followed by successful delivery and disease were both normal.
management. A left radical nephrectomy was performed with pathology no-
table for WT. Macroscopy included a left nephrectomy specimen 
of 1 kg and measuring 21 × 13 × 9 cm with the cut surface showing 
2  |  C A SE REPORT a firm yellow-g rey tumor measuring 14 × 11 × 10 cm in diameter; 
A 23-y ear- old Ghanaian woman of African ancestry initially pre-
sented to a peripheral health facility with a year's history of left 
flank swelling. She did not seek any medical care when she noticed 
the mass, until symptoms became associated with fever, chills, and 
weight loss. She was initially diagnosed as having a urinary tract in-
fection and treated with antibiotics. She was eventually referred to 
the urologist when symptoms worsened and also became associated 
with frank hematuria.
Findings from an abdomino-p elvic computed tomography (CT) 
scan with contrast showed a large mass involving the upper and mid-
dle poles of the left kidney measuring 11 × 11 × 16 cm. The rest of the 
abdominal organs (liver, gallbladder, pancreas, spleen, right kidney) 
were normal (Figures 1–3 ).
Results from CT renal angiography without and with intravenous 
contrast showed a large necrotic mass lesion involving the upper 
part of the left kidney. Lobar branches were seen arising from the 
main left renal artery outside the kidney contour with the tumor 
seen stretching the lobar branches. A tiny accessory renal arterial 
branch was seen arising from the proximal part of the main renal 
artery (Figure 4).
A full blood cell count showed a hemoglobin level of 10.3 g/ F I G U R E  2  Abdomino-p elvic computed tomography scan 
dL with white cell and platelet counts within normal limits. Results showing left Wilms tumor (coronal view).
F I G U R E  1  Abdomino- pelvic computed tomography scan F I G U R E  3  Abdomino-p elvic computed tomography scan 
showing left Wilms tumor (axial view). showing left Wilms tumor (sagittal view).
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SEPENU et al.     |  3
the tumor appeared to have broken through the capsule, and there by tightly bound cells, embryonal-l ooking rosettes, and pseudo ro-
was evidence of tumor rupture. Findings from microscopy showed settes. These features of epithelial cells with minimal blastema and 
a predominantly tubular pattern with luminal borders separated stroma were in keeping with nephroblastoma (WT). No unfavorable 
features were reported.
Further immunohistochemistry results were positive for paired 
box gene 8 (PAX8), p53, Wilms' tumor suppressor gene (WT1), epi-
thelial membrane antigen (EMA), pan cytokeratin (PanCK), neuron- 
specific enolase (NSE), and CD56 and showed predominantly 
epithelial cells with minimal blastema and stroma (Figure 5).
Due to persistent postoperative vomiting for 5 days, a urine 
pregnancy test was performed and noted to be positive. Pelvic ul-
trasound confirmed the presence of a 6- week gestational sac.
An MDT consisting of maternal- fetal medicine specialists, on-
cologists, urologists, renal physicians, neonatologists, anesthesiol-
ogists, a dietician, and clinical psychologists was formed to guide 
her management. The initial management plan of the MDT was to 
offer adjuvant radiotherapy after radical nephrectomy preceded 
by termination of pregnancy with ovarian cryopreservation. This 
decision was made because of the disease stage (stage III, due to 
tumor rupture) and gestational age. The patient was counseled on 
her diagnosis and the MDT recommendation. She, however, declined 
the ovarian cryopreservation because of financial constraints and 
refused termination of pregnancy, and hence could not have the ra-
F I G U R E  4  Computed tomography renal angiography showing a diation therapy. She was then scheduled to undergo follow-u p in the 
large mass lesion of the left kidney. obstetric renal high- risk clinic.
F I G U R E  5  Immunohistochemistry report.
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4  |    SEPENU et al.
She defaulted her follow- ups and presented 4 months later to the Three weeks after delivery when the wound was fully healed 
obstetric renal high- risk clinic with complaints of recurrent left flank and the patient was fully recovered, she was worked up again to 
pain. A repeat abdominopelvic ultrasound demonstrated a huge restart chemotherapy using the full pediatric WT protocol consist-
retroperitoneal mass in the left renal bed with associated satellite ing of cyclophosphamide, doxorubicin, vincristine, dactinomycin, 
masses suggestive of tumor recurrence with intra- abdominal dis- and adriamycin. She has since then received weekly intravenous 
semination and metastasis to the left hepatic lobe, as well as a live vincristine for 10 weeks followed by a three- weekly intravenous 
pregnancy at 24 weeks 6 days gestation. She continued to express vincristine regimen for five cycles (one dose given so far), intrave-
the desire to continue her pregnancy. A fetal anomaly scan was per- nous dactinomycin on day 28 of initiation of vincristine followed 
formed and results were unremarkable. by 6- weekly dactinomycin for five cycles (one dose given so far). 
The multidisciplinary team, in consultation with the pediatric Reassessment ultrasonography of the abdomen showed no intra- 
oncology team, counseled the patient and provided a workup for abdominal free fluid, lymphadenopathy, or mass lesions and two 
her to receive palliative chemotherapy with doxorubicin (adriamycin) heterogeneous lesions at segment 6 and 4B of the liver measuring 
50 mg/m2 and cyclophosphamide 450 mg/m2 for three cycles three 5.2 and 3.2 cm.
weekly, until 34 to 35 weeks of gestation, to allow recovery from She was offered follow- up of her gonadal function using evalua-
side effects of chemotherapy, especially neutropenia, with an aim tion of levels of her anti- AMH, FSH, LH, estradiol, and progesterone 
of delivery of the baby at 38 weeks of gestation. She was to have at 6 and 12 months postchemotherapy. She is currently doing very 
optimum fetal surveillance with biweekly growth scans, daily fetal well at her last radio-o ncology review and has given her consent for 
kick counts, twice- daily fetal Dopplers, umbilical artery Dopplers as this case report.
indicated, maturation of fetal lungs at 28 weeks and 34 weeks using 
intramuscular dexamethasone, and delivery via induction of labor at 
34 weeks. 3  |  DISCUSSION
Following delivery, she was to have the full pediatric chemo-
therapy protocol for WT, which included vincristine, adriamycin and Only approximately 300 cases of nephroblastoma (WT) in adults 
dactinomycin in addition to the doxorubicin and cyclophosphamide have been documented in the literature with varying terminologies 
followed after cycle 6 by irradiation to the whole liver as 19.8 Gy in and confusing histological diagnosis.5 Nephroblastoma presents as 
11 fractions. Due to the chemotherapy, she was also counseled not a triphasic embryonal tumor with various proportions of blastema, 
to breastfeed the baby. stroma, and epithelial cells.8 Blastemal cells are the most malignant 
She was mostly managed on an outpatient basis except in the im- tumor component as they are the least differentiated with rapid mi-
mediate postchemotherapy period when she was admitted for fetal totic activity. Epithelial components tend to be well differentiated 
surveillance (daily fetal kick count, twice daily fetal heart rate moni- with abortive tubules or glomeruli- like structure. Stromal compo-
toring, and weekly nonstress test) and maternal monitoring. nents have moderate malignant potential with varying undifferenti-
Intramuscular dexamethasone 6 mg 12 hourly for 48 h was ad- ated to partially differentiated mesenchymal cells.5 This patient had 
ministered for fetal lung maturation at 28 and 34 weeks. a monophasic tumor predominantly epithelial, which has a better 
The planned third cycle of adriamycin and cyclophosphamide prognosis because it is the least malignant component. Immunohis-
was suspended due to persistent pancytopenia at 34 weeks of ges- tochemistry staining in African children with WT children show a 
tation to allow platelet levels to improve as it was close to the time preponderance of p53 mutation ranging from 8.3% to 60.3% com-
of delivery. pared with Caucasians (between 0% and 13.4%).9 This patient had a 
The patient requested an elective cesarean section to be positive p53 mutation in addition to other mutations. This mutation 
followed by contraception after she had been counseled on the is associated with more clinically aggressive disease even with fa-
possible options including induction of labor. She delivered via vorable histology.9
an uneventful elective cesarean section at 37 weeks 6 days to a The clinical presentation of WT is usually weight loss, flank pain, 
live female neonate with Apgar scores of 8 and 9 at 1 and 5 min- flank mass, or hematuria.10 All of these symptoms were present in 
utes, respectively and a birth weight of 2435 g (third percentile our patient.
for gestational age). A postplacental copper intrauterine device Abdominal ultrasound is the first diagnostic modality of choice to 
was inserted for family planning. The baby was examined by the confirm a renal mass. CT scan and magnetic resonance imaging help 
neonatologist immediately following delivery and found to be to differentiate WT from other renal masses such as neuroblastoma, 
healthy. Due to the antenatal exposure of the fetus to chemo- which can further be differentiated from WT through urine analysis 
therapy, gonadal function assessment of the baby was planned for catecholamines and metaiodobenzylguanidine scintigraphy. This 
within 6 months after delivery via assessment of levels of the anti- patient's diagnosis was made using an abdominal ultrasound and CT 
Mullerian hormone (AMH), follicle- stimulating hormone (FSH), scan. Supportive tests included a full blood cell count to screen for 
luteinizing hormone (LH), and estradiol by a pediatric endocrinol- tumor- associated anemia and thrombocytopenia, kidney function 
ogist. The patient was discharged on postoperative day 3 in stable test for potential derangement in kidney function, liver function test 
condition. for altered liver enzymes in liver metastasis, and clotting profile for 
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SEPENU et al.     |  5
disrupted coagulation such as occurs in WT- acquired von Willebrand to ensure earlier presentation, social support, improved supportive 
disease.11 care, a locally adapted treatment guideline, and dedicated multidisci-
Currently, Kilton et al.'s diagnostic criteria is widely used to diag- plinary management may be key to improve outcomes.18
nose adult WT and includes: (a) a primary renal neoplasm; (b) primi- Clinical psychologist care is also important in enhancing com-
tive blastomatous spindle or round cell component; (c) formation of pliance to treatment. This case had psychological support through-
abortive or embryonal tubular or glomeruloid structures; (d) no areas out her care, ensuring compliance to therapy. One of the important 
of hypernephroma; (e) pictorial confirmation of histologic findings; questions that informed her choice of keeping the pregnancy was 
and (f) age older than 15 years.5,12,13 concerning her fertility after chemotherapy, for which she needed 
Due to the absence of clear guidelines in adults, treatment is psychological support. Chemotherapy using alkylating agents and 
adapted from the WT protocols for children. In developed coun- irradiation of the abdomen and pelvis potentially reduces primor-
tries, multidisciplinary team management, appropriate patient dial follicles with resultant temporary or permanent ovarian function 
stratification using histological type to select chemotherapeutic loss.15 Symptoms of ovarian insufficiency may arise during treat-
agents, and research- driven care are core to treatment success.14 ment, shortly afterwards, or later on as premature menopause.15 
The two most commonly applied treatment regimens for WT are There is higher risk with cyclophosphamide, intermediate risk with 
derived from the National Wilms Tumor Study Group/COG and Adriamycin, and low risk with vincristine, all of which were used in 
SIOP, which involves surgery, chemotherapy, and irradiation.11 In the management of this case. Gonadal function status of premeno-
metastatic disease, usually involving lymph node, lung, and liver, pausal women who receive chemotherapy can be assessed at 6 and 
escalated therapy and whole- lung radiation therapy is the treat- 12 months after chemotherapy using levels of AMH, FSH, LH, estra-
ment of choice. This patient, managed under resource constraints, diol, and progesterone, with AMH said to be the most sensitive.20,21 
had both disseminated disease to the para- aortic lymph node and Neonates exposed antenatally to gonadotoxic chemotherapy may be 
liver at the time of presentation and also a wanted pregnancy. An followed up in the minipuberty phase within 6 to 9 months of deliv-
MDT was constituted to formulate a plan of care in a high- risk ery by the pediatric endocrinologist using assessment of AMH, FSH, 
pregnancy clinic setting. Patient-a dapted and risk- modified treat- LH, and estradiol levels.22 A review of studies by Del et al., however, 
ment is a strategy of choice, where chemotherapy not fatally toxic suggest that there are no long- term effects on child gonadal func-
to the fetus was initiated during the pregnancy and escalated im- tion after antenatal exposure to gonadotoxic chemotherapy.23
mediately postpartum guided by the histological type. Concerns Contraception is important to postpone pregnancy at least until 
regarding the possible effect of chemotherapy on the offspring 2 years after treatment to allow enough time to confirm remission 
of such women include congenital anomalies, genetic diseases, and rule out secondary effects of chemotherapy that may compli-
low birth weight, and prematurity.15 However, apart from low cate future pregnancy such as diabetes or hepatic, renal, or cardiac 
birth weight, the neonate of this patient had a good outcome, disease. An MDT during the prenatal period that includes an oncol-
highlighting the fact that with MDT care, the appropriate choice ogist and a maternal fetal medicine specialist is essential to set the 
of life- saving chemotherapeutic agents, and fetal surveillance best time to get pregnant.15 Our patient chose postplacental intra-
can promote good outcomes even in pregnant women managed uterine device insertion as contraception.
in low-r esource settings. Presently, regimens for chemotherapy 
used in WT protocols after the first trimester, such as cyclophos-
phamide, vincristine, doxorubicin, are considered relatively safe.16 4  |  CONCLUSION
Avilés and Neri reported that offspring of children exposed in 
utero to such chemotherapeutic agents on follow-u p had no con- In summary, we present a rare case of an adult patient diagnosed 
genital or neurologic abnormalities, no reported malignancies, and with disseminated WT peripartum. She was found to be pregnant 
normal educational performance.17 However, antenatal chemo- soon after radical nephrectomy and before postsurgical chemother-
therapeutic protocols must be implemented after multidisciplinary apy could be commenced. She preferred to keep the pregnancy and 
discussions and appropriate patient counseling prior to treatment was managed in a multidisciplinary high- risk obstetric renal clinic 
initiation.16 Abdomino-p elvic irradiation on the other hand is con- with antenatal and postpartum chemotherapy. Further research is 
traindicated as it directly affects the growing fetus and hence is needed to outline protocols for resource- adapted and risk- profiled 
generally avoided antenatally, as was done in this case. therapy in adult pregnant patients with WT prior to delivery.
Barriers to care have been studied in childhood nephroblastoma, 
especially in low- resource settings with strategies for improved care AUTHOR CONTRIBUTIONS
clearly defined.18,19 Although this has not been studied in adult WT, Perez Sepenu, Jerry Coleman, and Theodore K. Boafor: conception, 
it offers a starting basis to outline care modalities to improve out- drafting of the manuscript, MDT meetings, clinical management of 
comes. The major barriers were late presentation to a mainstream patients, editing the manuscript, and approval of the final manu-
hospital for definitive care and poor compliance to treatment.18 In script. Alim Swarray- Deen, Aba Scott, Mathew K. Kyei, and Winfred 
this presented case, there was a delay to seek definitive care as well K. Baah: MDT meetings, clinical management of patients, editing of 
as a delay in diagnosis at the initial care centers. Public education the manuscript, and approval of the final manuscript.
 18793479, 0, Downloaded from https://obgyn.onlinelibrary.wiley.com/doi/10.1002/ijgo.15157 by University of Ghana - Accra, Wiley Online Library on [02/10/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
6  |    SEPENU et al.
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