A rare case of ectopic ACTH syndrome with rhabdomyolysis
Abstract Background Manifestations of hypokalaemia in ectopic adrenocorticotropic hormonesyndrome(EAS) vary from mild muscle weakness to life-threatening arrhythmia. Herein, we present a rare case of EAS with concomitant rhabdomyolysis(RM) as a result of intractable hypokalaemia. Case presentation A...
| Main Authors: | , , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
BMC
2021-05-01
|
| Series: | BMC Endocrine Disorders |
| Subjects: | |
| Online Access: | https://doi.org/10.1186/s12902-021-00755-0 |
| _version_ | 1818733508088037376 |
|---|---|
| author | Wei Qiang Sucai Song Tianjun Chen Zhe Wang Jun Feng Jiaojiao Zhang Hui Guo |
| author_facet | Wei Qiang Sucai Song Tianjun Chen Zhe Wang Jun Feng Jiaojiao Zhang Hui Guo |
| author_sort | Wei Qiang |
| collection | DOAJ |
| description | Abstract Background Manifestations of hypokalaemia in ectopic adrenocorticotropic hormonesyndrome(EAS) vary from mild muscle weakness to life-threatening arrhythmia. Herein, we present a rare case of EAS with concomitant rhabdomyolysis(RM) as a result of intractable hypokalaemia. Case presentation A 64-year-old man was admitted for limb weakness and facial hyperpigmentation for 2 weeks. Lab tests revealed intractable hypokalaemia (lowest at 1.8 mmol/L) and metabolic alkalosis. The diagnosis of RM was based on a creatine kinase(CK)level of 5 times the upper limit. The elevated CK and myohemoglobin (Mb) levels returned to within the normal range after the alleviation of hypokalaemia. The patient was diagnosed with ACTH-dependent Cushing’s syndrome (CS) based on unsuppressed serum cortisol after a low-dose dexamethasone suppression test(LDDST) and remarkably elevated ACTH levels. The diagnosis of EAS was made based on the results of a high-dose dexamethasone suppression test(HDDST) and bilateral inferior petrosal sinus sampling(BIPSS). Multiple lymph nodes in the left supraclavicular fossa, right root of neck, mediastinum and bilateral hili of the lung were found with abnormal uptake of 68Ga-DOTA-NOC. Mediastinoscopic lymph node biopsy was performed. The pathological diagnosis was small-cell and large-cell neuroendocrine carcinoma with positive ACTH staining. The patient was prescribed mifepristone and received one cycle of chemotherapy. The patient could not tolerate subsequent chemotherapy and died of dyscrasia. Conclusions RM is a rare complication of EAS with insidious onset and atypical clinical manifestations. Serum potassium levels should be vigilantly monitored to avoid RM in EAS. |
| first_indexed | 2024-12-17T23:50:35Z |
| format | Article |
| id | doaj.art-0e4e0be89b8a4741b4fd86bf6c45be10 |
| institution | Directory Open Access Journal |
| issn | 1472-6823 |
| language | English |
| last_indexed | 2024-12-17T23:50:35Z |
| publishDate | 2021-05-01 |
| publisher | BMC |
| record_format | Article |
| series | BMC Endocrine Disorders |
| spelling | doaj.art-0e4e0be89b8a4741b4fd86bf6c45be102022-12-21T21:28:13ZengBMCBMC Endocrine Disorders1472-68232021-05-012111810.1186/s12902-021-00755-0A rare case of ectopic ACTH syndrome with rhabdomyolysisWei Qiang0Sucai Song1Tianjun Chen2Zhe Wang3Jun Feng4Jiaojiao Zhang5Hui Guo6Department of Endocrinology and Metabolism, The First Affiliated Hospital of Xi’an Jiaotong UniversityDepartment of Endocrinology and Metabolism, The First Affiliated Hospital of Xi’an Jiaotong UniversityDepartment of Respiratory and Critical Medicine, The First Affiliated Hospital of Xi’an Jiaotong UniversityDepartment of Thoracic Surgery, The First Affiliated Hospital of Xi’an Jiaotong UniversityDepartment of Vascular Surgery, The First Affiliated Hospital of Xi’an Jiaotong UniversityDepartment of Pathology, The First Affiliated Hospital of Xi’an Jiaotong UniversityDepartment of Endocrinology and Metabolism, The First Affiliated Hospital of Xi’an Jiaotong UniversityAbstract Background Manifestations of hypokalaemia in ectopic adrenocorticotropic hormonesyndrome(EAS) vary from mild muscle weakness to life-threatening arrhythmia. Herein, we present a rare case of EAS with concomitant rhabdomyolysis(RM) as a result of intractable hypokalaemia. Case presentation A 64-year-old man was admitted for limb weakness and facial hyperpigmentation for 2 weeks. Lab tests revealed intractable hypokalaemia (lowest at 1.8 mmol/L) and metabolic alkalosis. The diagnosis of RM was based on a creatine kinase(CK)level of 5 times the upper limit. The elevated CK and myohemoglobin (Mb) levels returned to within the normal range after the alleviation of hypokalaemia. The patient was diagnosed with ACTH-dependent Cushing’s syndrome (CS) based on unsuppressed serum cortisol after a low-dose dexamethasone suppression test(LDDST) and remarkably elevated ACTH levels. The diagnosis of EAS was made based on the results of a high-dose dexamethasone suppression test(HDDST) and bilateral inferior petrosal sinus sampling(BIPSS). Multiple lymph nodes in the left supraclavicular fossa, right root of neck, mediastinum and bilateral hili of the lung were found with abnormal uptake of 68Ga-DOTA-NOC. Mediastinoscopic lymph node biopsy was performed. The pathological diagnosis was small-cell and large-cell neuroendocrine carcinoma with positive ACTH staining. The patient was prescribed mifepristone and received one cycle of chemotherapy. The patient could not tolerate subsequent chemotherapy and died of dyscrasia. Conclusions RM is a rare complication of EAS with insidious onset and atypical clinical manifestations. Serum potassium levels should be vigilantly monitored to avoid RM in EAS.https://doi.org/10.1186/s12902-021-00755-0Cushing’s syndromeEctopic ACTH syndromeHypokalaemiaRhabdomyolysis |
| spellingShingle | Wei Qiang Sucai Song Tianjun Chen Zhe Wang Jun Feng Jiaojiao Zhang Hui Guo A rare case of ectopic ACTH syndrome with rhabdomyolysis BMC Endocrine Disorders Cushing’s syndrome Ectopic ACTH syndrome Hypokalaemia Rhabdomyolysis |
| title | A rare case of ectopic ACTH syndrome with rhabdomyolysis |
| title_full | A rare case of ectopic ACTH syndrome with rhabdomyolysis |
| title_fullStr | A rare case of ectopic ACTH syndrome with rhabdomyolysis |
| title_full_unstemmed | A rare case of ectopic ACTH syndrome with rhabdomyolysis |
| title_short | A rare case of ectopic ACTH syndrome with rhabdomyolysis |
| title_sort | rare case of ectopic acth syndrome with rhabdomyolysis |
| topic | Cushing’s syndrome Ectopic ACTH syndrome Hypokalaemia Rhabdomyolysis |
| url | https://doi.org/10.1186/s12902-021-00755-0 |
| work_keys_str_mv | AT weiqiang ararecaseofectopicacthsyndromewithrhabdomyolysis AT sucaisong ararecaseofectopicacthsyndromewithrhabdomyolysis AT tianjunchen ararecaseofectopicacthsyndromewithrhabdomyolysis AT zhewang ararecaseofectopicacthsyndromewithrhabdomyolysis AT junfeng ararecaseofectopicacthsyndromewithrhabdomyolysis AT jiaojiaozhang ararecaseofectopicacthsyndromewithrhabdomyolysis AT huiguo ararecaseofectopicacthsyndromewithrhabdomyolysis AT weiqiang rarecaseofectopicacthsyndromewithrhabdomyolysis AT sucaisong rarecaseofectopicacthsyndromewithrhabdomyolysis AT tianjunchen rarecaseofectopicacthsyndromewithrhabdomyolysis AT zhewang rarecaseofectopicacthsyndromewithrhabdomyolysis AT junfeng rarecaseofectopicacthsyndromewithrhabdomyolysis AT jiaojiaozhang rarecaseofectopicacthsyndromewithrhabdomyolysis AT huiguo rarecaseofectopicacthsyndromewithrhabdomyolysis |