Description
An early 30s woman with a history of bipolar disorder presented to our hospital, having abdominal pain for the previous 4 days. Her body weight had increased over the years, and type 2 diabetes and hypertension developed 2 years ago. Her medication includes as follows: tofogliflozin 20 mg, metformin 1000 mg, telmisartan 40 mg, alprazolam 2.4 mg, lithium carbonate 600 mg and escitalopram oxalate 20 mg. There was no history of exogenous glucocorticoid administration. Her blood glucose control had worsened, and oral semaglutide was administered 6 days prior to the visit. Physical examination revealed central obesity with abdominal violaceous striae (figure 1), but other pathological findings that indicated intra-abdominal inflammation were not noted. Blood examination showed no abnormalities in C reactive protein, transaminase and amylase. Abdominal ultrasound examination and gastrointestinal endoscopy did not point to any abnormalities that could cause the abdominal pain. Her symptoms were attributed to semaglutide use and the discontinuation of this medication lead to resolution of the abdominal complaints.
Figure 1.
Wide and purple abdominal skin striae and central obesity can be observed.
The hormonal evaluations were performed to determine the cause of central obesity with abdominal violaceous striae. A serum adrenocorticotropic hormone (ACTH) concentration of 50.8 pg/mL (6.8–63.2) and a serum cortisol level of 18.6 µg/dL (4.0–18.3) were detected. A 24-hour urinary free cortisol level was 264 µg/dL (4.3–176) and 1 mg dexamethasone suppression test (DST) showed a cortisol level of 13.4 µg/dL (<1.8). Pituitary MRI showed no pituitary adenoma. Serum cortisol after 8 mg DST was 2.4 µg/dL. Corticotropin-releasing hormone (CRH) test showed an elevation in ACTH and cortisol levels of 290% and 150%, respectively, from the baseline. Serum cortisol level after dexamethasone-CRH test was 16.5 µg/dL (<1.4). Inferior petrosal sinus sampling was not performed because the patient did not consent. The patient was diagnosed with Cushing’s disease and treatment of metyrapone was started as the patient did not consent to surgical treatment.
Various physical findings, such as central obesity, rounded face, red cheeks, dorsocervical fat pad and abdominal violaceous striae, are observed in patients with Cushing’s syndrome (CS). However, some of these symptoms overlap with more common clinical conditions such as metabolic syndrome. Therefore, diagnosis of CS is often delayed, and the mean duration from the first symptom of CS to the diagnosis is approximately 34 months.1 However, as prolonged exposure to excess glucocorticoids increases the occurrence of metabolic and cardiovascular complications and mortality,2 diagnosing CS promptly is important. Among various physical features observed in patients with CS, abdominal violaceous striae are the most specific physical finding, which is present in approximately 50% of patients with CS.3 Skin striae owing to hypercortisolism are often wide and purple, in contrast to the narrow and pale or pink striae by rapid weight gain.4 CS should be suspected in a patient with violaceous abdominal striae and metabolic comorbidities even if they presented to the hospital for a different reason.
Patient’s perspective.
I never thought to be diagnosed with such a rare disease at the visit for abdominal symptoms. I wasn't convinced that over the years I’d gained weight and been diagnosed with diabetes and high blood pressure. The diagnosis of CS connected me to everything that had happened to me over the years. I would like to thank the doctor who diagnosed me and all the staff at the hospital.
Learning points.
Diagnosing Cushing’s syndrome (CS) can be challenging due to the lack of a unique sign of hypercortisolism and an overlap with more common clinical conditions such as metabolic syndrome.
Abdominal violaceous striae are one of the more specific physical findings of CS.
CS should be suspected in a patient with violaceous abdominal striae and metabolic comorbidities even if they presented to the hospital for a different reason.
Footnotes
Twitter: @yQkHAFnVtQmwwpu
Contributors: The following authors were responsible for drafting of the text, sourcing and editing of clinical images, investigation results, drawing original diagrams and algorithms, and critical revision for important intellectual content: TS, MK, SK and TY. The following authors gave final approval of the manuscript: TS, MK, SK and TY.
Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.
Competing interests: None declared.
Provenance and peer review: Not commissioned; externally peer reviewed.
Ethics statements
Patient consent for publication
Consent obtained directly from patient(s).
References
- 1. Rubinstein G, Osswald A, Hoster E, et al. Time to diagnosis in Cushing’s syndrome: a meta-analysis based on 5367 patients. J Clin Endocrinol Metab 2020;105:dgz136. 10.1210/clinem/dgz136 [DOI] [PubMed] [Google Scholar]
- 2. Lambert JK, Goldberg L, Fayngold S, et al. Predictors of mortality and long-term outcomes in treated Cushing’s disease: a study of 346 patients. J Clin Endocrinol Metab 2013;98:1022–30. 10.1210/jc.2012-2893 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 3. Kreitschmann-Andermahr I, Psaras T, Tsiogka M, et al. From first symptoms to final diagnosis of Cushing’s disease: experiences of 176 patients. Eur J Endocrinol 2015;172:285–9. 10.1530/EJE-14-0766 [DOI] [PubMed] [Google Scholar]
- 4. Savas M, Mehta S, Agrawal N, et al. Approach to the patient: diagnosis of Cushing syndrome. J Clin Endocrinol Metab 2022;107:3162–74. 10.1210/clinem/dgac492 [DOI] [PMC free article] [PubMed] [Google Scholar]