1.Beyond Mitotane in a Patient With Highly Aggressive Adrenocortical Carcinoma
Muhammad Shukri Johar ; Siti Sanaa Wan Azman ; Dorothy Maria Anthony Bernard ; Foo Siew Hui
Journal of the ASEAN Federation of Endocrine Societies 2026;41(S1):30-31
Introduction:
Adrenocortical carcinoma (ACC) is an aggressive malignancy with high rates of recurrence even after surgical
resection. Surgery remains the mainstay of treatment,
while adjuvant options are limited. Mitotane is the only
approved systemic therapy. Current guidelines recommend
stereotactic body radiotherapy (SBRT) alongside adjuvant
mitotane therapy in Rx, R1, R2 resections and in locally
advanced disease.
Case:
We present a case of a 40-year-old female who presented
with abdominal pain and was found to have a large
heterogeneous left adrenal mass measuring 8.2 × 8.5 × 9.5
cm (Hounsfield Unit 63) on computed tomography (CT)
imaging. Clinically, she was obese with a body mass index
of 33.7 kg/m². No discriminatory feature of Cushing’s was
present. Hormonal evaluation demonstrated autonomous
cortisol secretion with failure of suppression on both
overnight and low-dose dexamethasone suppression tests
at 301 nmol/L and 313.6 nmol/L, respectively. DHEA,
testosterone, and urinary metanephrine were within range.
Hemoglobin A1c was 6.6%. She underwent open left
adrenalectomy. Intra-operatively, a 12 × 10 cm adrenal tumor
was identified with multiple areas of tumor rupture and
spillage during mobilization. HPE confirmed high-grade
ACC with high Weiss score of 8, Ki-67 index 60–80%, and
mitotic count 54/50 hpf (pT2Nx). Post-operative CT imaging
demonstrated a residual soft tissue lesion in the left adrenal
bed (largest diameter 3.8 cm) with fluorodeoxyglucose
avidity. We commenced adjuvant mitotane therapy,
titrated to 2 g TDS with supraphysiological hydrocortisone
replacement. Mitotane level was within therapeutic range
(16 mcg/mL). She was deemed unsuitable for repeat surgery
due to the proximity of the residual mass to the adjacent
vessel and was planned for SBRT therapy after a multidisciplinary team discussion.
Conclusion
High-risk ACC with suspected residual disease remains a
therapeutic challenge. While mitotane remains the cornerstone of adjuvant therapy, SBRT may represent a promising
adjunctive local treatment modality in carefully selected
patients. Further studies are required to define its role in
improving local control and outcomes in ACC.
Adrenocortical Carcinoma
;
Mitotane
2.A rare occurrence of Premature Birth and Recurrent Acute Pulmonary Oedema in the mother due to Cushing’s Syndrome: A case report
Dorothy Maria A/p Anthony Bernard ; Ooi Xin Yi ; Hema Lata A/p Veerasamy ; Mohamed Badrulnizam Long Bidin ; See Chee Keong
Journal of the ASEAN Federation of Endocrine Societies 2021;36(2):200-204
Presentation of Cushing’s syndrome during pregnancy is extremely rare. We report a 21-year-old female with Cushing’s syndrome diagnosed at 23 weeks of gestation and had recurrent acute pulmonary oedema during the antepartum and postpartum period. She delivered prematurely via emergency caesarean section at 28 weeks of gestation. This case highlights the rare occurrence of recurrent acute pulmonary oedema during pregnancy and consequential premature birth in a patient with adrenal Cushing’s. She was diagnosed with adrenal Cushing’s during the postpartum period based on unsuppressed serum cortisol after overnight and low-dose dexamethasone suppression test with a suppressed ACTH. CT scan of the adrenal glands revealed a right adrenal cortical adenoma. The risk of complications in infants and mothers who suffer from Cushing’s syndrome needs to be handled carefully. The diagnosis of Cushing’s syndrome in pregnant women often overlaps and is difficult to establish in early pregnancy.
Pregnancy


Result Analysis
Print
Save
E-mail