A novel approach to intraoperative diagnosis of primary pigmented nodular adrenocortical disease
Cushing syndrome (CS) is an endocrine disorder with far-reaching complications that extend beyond the disease remission. Diagnosis of the aetiology of CS can be challenging, whether it is dependent or independent of adrenocorticotrophic hormone (ACTH). Here, we describe a case of ACTH-independent CS...
Saved in:
| Main Authors: | , , , , , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
Bioscientifica
2025-02-01
|
| Series: | Endocrinology, Diabetes & Metabolism Case Reports |
| Subjects: | |
| Online Access: | https://edm.bioscientifica.com/view/journals/edm/2025/1/EDM-24-0079.xml |
| Tags: |
Add Tag
No Tags, Be the first to tag this record!
|
| _version_ | 1823858277060968448 |
|---|---|
| author | Nethrani Sameera Wijesekara Pathirana Hasitha Udayakumara A S K Banagala M R Sumanatilleke A V T Damayanthi S R Constantine G W Katulanda Z T M Thowfeek T Sasikanth S Pathmanathan |
| author_facet | Nethrani Sameera Wijesekara Pathirana Hasitha Udayakumara A S K Banagala M R Sumanatilleke A V T Damayanthi S R Constantine G W Katulanda Z T M Thowfeek T Sasikanth S Pathmanathan |
| author_sort | Nethrani Sameera Wijesekara Pathirana |
| collection | DOAJ |
| description | Cushing syndrome (CS) is an endocrine disorder with far-reaching complications that extend beyond the disease remission. Diagnosis of the aetiology of CS can be challenging, whether it is dependent or independent of adrenocorticotrophic hormone (ACTH). Here, we describe a case of ACTH-independent CS due to primary pigmented nodular adrenocortical disease (PPNAD) in a 33-year-old female patient with several complications of CS, including diabetes, hypertension, osteoporosis and severe depression with suicidal ideation. In this case, following the demonstration of ACTH independence of CS, it was challenging to localise the lesion as there were bilateral adrenal lesions. Furthermore, preoperative efforts in localisation in the form of adrenal venous sampling (AVS) failed. However, the diagnosis of PPNAD was confirmed using an intraoperative frozen section and macroscopy, and the patient underwent bilateral adrenalectomy during the same surgery. This case highlights a novel approach to diagnosing and managing PPNAD intraoperatively in a resource-limited setting where preoperative localisation studies have failed. |
| format | Article |
| id | doaj-art-8500912e3c7444a3be79163035aaefdf |
| institution | Kabale University |
| issn | 2052-0573 |
| language | English |
| publishDate | 2025-02-01 |
| publisher | Bioscientifica |
| record_format | Article |
| series | Endocrinology, Diabetes & Metabolism Case Reports |
| spelling | doaj-art-8500912e3c7444a3be79163035aaefdf2025-02-11T12:32:06ZengBioscientificaEndocrinology, Diabetes & Metabolism Case Reports2052-05732025-02-012025110.1530/EDM-24-00791A novel approach to intraoperative diagnosis of primary pigmented nodular adrenocortical diseaseNethrani Sameera Wijesekara Pathirana0Hasitha Udayakumara1A S K Banagala2M R Sumanatilleke3A V T Damayanthi4S R Constantine5G W Katulanda6Z T M Thowfeek7T Sasikanth8S Pathmanathan9Diabetes and Endocrinology Unit, National Hospital of Sri Lanka, Colombo, Sri LankaNational Hospital of Sri Lanka, Colombo, Sri LankaNational Hospital of Sri Lanka, Colombo, Sri LankaDiabetes and Endocrinology Unit, National Hospital of Sri Lanka, Colombo, Sri LankaDepartment of Histopathology, National Hospital of Sri Lanka, Colombo, Sri LankaDepartment of Histopathology, National Hospital of Sri Lanka, Colombo, Sri LankaDepartment of Chemical Pathology, National Hospital of Sri Lanka, Colombo, Sri LankaDepartment of Chemical Pathology, National Hospital of Sri Lanka, Colombo, Sri LankaDiabetes and Endocrinology Unit, National Hospital of Sri Lanka, Colombo, Sri LankaDiabetes and Endocrinology Unit, National Hospital of Sri Lanka, Colombo, Sri LankaCushing syndrome (CS) is an endocrine disorder with far-reaching complications that extend beyond the disease remission. Diagnosis of the aetiology of CS can be challenging, whether it is dependent or independent of adrenocorticotrophic hormone (ACTH). Here, we describe a case of ACTH-independent CS due to primary pigmented nodular adrenocortical disease (PPNAD) in a 33-year-old female patient with several complications of CS, including diabetes, hypertension, osteoporosis and severe depression with suicidal ideation. In this case, following the demonstration of ACTH independence of CS, it was challenging to localise the lesion as there were bilateral adrenal lesions. Furthermore, preoperative efforts in localisation in the form of adrenal venous sampling (AVS) failed. However, the diagnosis of PPNAD was confirmed using an intraoperative frozen section and macroscopy, and the patient underwent bilateral adrenalectomy during the same surgery. This case highlights a novel approach to diagnosing and managing PPNAD intraoperatively in a resource-limited setting where preoperative localisation studies have failed.https://edm.bioscientifica.com/view/journals/edm/2025/1/EDM-24-0079.xmlprimary pigmented nodular adrenocortical diseasecushing syndrome |
| spellingShingle | Nethrani Sameera Wijesekara Pathirana Hasitha Udayakumara A S K Banagala M R Sumanatilleke A V T Damayanthi S R Constantine G W Katulanda Z T M Thowfeek T Sasikanth S Pathmanathan A novel approach to intraoperative diagnosis of primary pigmented nodular adrenocortical disease Endocrinology, Diabetes & Metabolism Case Reports primary pigmented nodular adrenocortical disease cushing syndrome |
| title | A novel approach to intraoperative diagnosis of primary pigmented nodular adrenocortical disease |
| title_full | A novel approach to intraoperative diagnosis of primary pigmented nodular adrenocortical disease |
| title_fullStr | A novel approach to intraoperative diagnosis of primary pigmented nodular adrenocortical disease |
| title_full_unstemmed | A novel approach to intraoperative diagnosis of primary pigmented nodular adrenocortical disease |
| title_short | A novel approach to intraoperative diagnosis of primary pigmented nodular adrenocortical disease |
| title_sort | novel approach to intraoperative diagnosis of primary pigmented nodular adrenocortical disease |
| topic | primary pigmented nodular adrenocortical disease cushing syndrome |
| url | https://edm.bioscientifica.com/view/journals/edm/2025/1/EDM-24-0079.xml |
| work_keys_str_mv | AT nethranisameerawijesekarapathirana anovelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT hasithaudayakumara anovelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT askbanagala anovelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT mrsumanatilleke anovelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT avtdamayanthi anovelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT srconstantine anovelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT gwkatulanda anovelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT ztmthowfeek anovelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT tsasikanth anovelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT spathmanathan anovelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT nethranisameerawijesekarapathirana novelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT hasithaudayakumara novelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT askbanagala novelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT mrsumanatilleke novelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT avtdamayanthi novelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT srconstantine novelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT gwkatulanda novelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT ztmthowfeek novelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT tsasikanth novelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease AT spathmanathan novelapproachtointraoperativediagnosisofprimarypigmentednodularadrenocorticaldisease |