Retention of aberrant cortisol secretion in a patient with bilateral macronodular adrenal hyperplasia after unilateral adrenalectomy

Figures & data

Figure 1 Imaging of adrenal glands.

Notes: The arrows indicate the nodule in the right (yellow) and left (red) adrenal gland. (A) Image of CT. (B) Merged image of CT and 131-I adosterol SPECT. (C) Image of 131-I adosterol SPECT.
Abbreviations: CT, computed tomography; SPECT, single photon emission computed tomography.

Figure 2 Aberrant responses of cortisol secretion preoperatively.

Notes: Vertical axis indicates serum cortisol level. Horizontal axis indicates time course after exogenous stimuli.
Abbreviations: ACTH, adrenocorticotropic hormone; GnRH, gonadotropin-releasing hormone.

Figure 3 Cortisol secretion on metoclopramide challenge at postoperative day 8.

Notes: Vertical axis indicates serum cortisol level. Horizontal axis indicates time course after metoclopramide administration. Numerical values are also shown with trajectory.

Figure 4 Time course of morning serum cortisol before and after left adrenalectomy.

Notes: Vertical axis indicates morning serum cortisol level. Horizontal axis indicates time course before and after left adrenalectomy (week 0). Three data points collected before surgery, and data collected at postoperative days 8, 72 and 109 are shown. Regimen of steroid coverage at day 8 was hydrocortisone 15 mg/day (10 and 5 mg after breakfast and dinner, respectively). Regimen of steroid coverage at days 72 and 109 was hydrocortisone 12.5 mg/day, 10 and 2.5 mg after breakfast and dinner, respectively. Numerical values are also shown with trajectory.

Table 1 Review of BMAH cases with paradoxical cortisol secretion response on metoclopramide challenge

Case	Author	Sex	Age (years)	BMI (kg/m^2)	Function	Maleifestation	Nodule size (mm)		U-Cor (µg/day)	Degree of increase (%)
							Right	Left
1	Libé R	Male	53	31.2	CS	Cushing’s sign	35	45	293	133
2		Female	34	30.0	CS	Cushing’s sign	45	30	373	180
3		Female	70	25.0	CS	Incidentaloma	50	30	122	204
4		Female	45	21.3	SCS	Incidentaloma	60	30	44	438
5		Female	37	30.5	SCS	Incidentaloma	33	15	26	185
6		Male	59	25.6	SCS	Hypertension	30	60	31	426
7		Female	66	27.8	SCS	Incidentaloma	14	30	29	209
8		Female	35	27.4	SCS	Incidentaloma	26	27	18	180
9		Female	67	28.6	SCS	Incidentaloma	14	40	56	209
10		Female	54	28.2	SCS	Incidentaloma	18	44	36	137
11		Female	53	26.7	SCS	Incidentaloma	35	27	83	310
12		Male	74	23.3	SCS	Incidentaloma	20	25	85	160
13		Male	51	20.6	SCS	Incidentaloma	27	40	63	206
14		Male	52	27.0	SCS	Incidentaloma	55	46	50	138
15		Male	51	24.0	SCS	Incidentaloma	40	37	47	183
16		Female	43	22.3	SCS	Incidentaloma	45	15	46	154
17		Female	55	33.6	SCS	Hypertension	30	24	26	183
18	Bourdeau I	Female	51	NA	SCS	Hypertension, weight gain	44	50	NA	145
19		Male	61	NA	SCS	NA	61	73	NA	191
20		Male	53	25.3	SCS	Hypertension	35	40	NA	300
21	Vezzosi D	Female	56	NA	CS	Hypertension, Centripetal obesity, facial plethora, dorsal fat pad supraclavicular fat pad, glucose intolerance, mild hirsutism	NA	NA	34	270
22		Female	54	NA	CS	Hypertension, centripetal obesity, facial plethora, easy bruising, diabetes mellitus	NA	NA	50	301
23	Feelders RA	Female	60	24.4	CS	Centripetal obesity, facial plethora, muscle weakness, easy bruising depression, skin atrophy	35	50	199–228 326–652	289 195
24		Female	40	32.8	CS	Hypertension, centripetal obesity, facial plethora, weight gain, muscle weakness, easy bruising, dorsal fat pad, supraclavicular fat pad, depression	50	30
25	Lacroix A	Female	63	28.9	CS	Hypertension, weight gain, muscle weakness, numbness, hot flushes, decrease in concentration and memory	40	40	279	257

Note: Clinical features of cases with BMAH reported to be associated with paradoxical response of cortisol secretion on metoclopramide challenge are shown together with the percentage increase of cortisol level.

Abbreviations: BMAH, bilateral macronodular adrenal hyperplasia; BMI, body mass index; CS, Cushing’s syndrome; NA, not applicable; SCS, subclinical Cushing’s syndrome; U-Cor, urinary cortisol excretion.