On Becoming Empowered

Posted on January 1, 2026 by MaryO

This is kind of a “cheat” post since it’s a compilation of other posts, web pages, message board posts and some original thoughts.  

For all of my early life, I was the good, compliant, patient.  I took whatever pills the doctor prescribed, did whatever tests h/she (most always a he) wrote for.  Believed that whatever he said was the absolute truth.  He had been to med school.  He knew what was wrong with me even though he didn’t live in my body 24/7 and experience what I did.

I know a lot of people are still like this.  Their doctor is like a god to them.  He can do no wrong – even if they don’t feel any better after treatment, even if they feel worse.  “But the doctor said…”

Anyway, I digress.

All this changed for me in 1983.

At first I noticed I’d stopped having my periods and, of course, I thought I was pregnant. I went to my Gynecologist who had no explanation. Lots of women lose their periods for a variety of reasons so no one thought that this was really significant.

Then I got really tired, overly tired. I would take my son to a half hour Choir rehearsal and could not stay awake for the whole time. I would lie down in the back of the van, set an alarm and sleep for the 30 minutes.

A whole raft of other symptoms started appearing – I grew a beard (Hirsuitism), gained weight even though I was on Weight Watchers and working out at the gym nearly every day, lost my period, everything hurt, got what is called a “moon face” and a “buffalo hump” on the back of my neck. I also got stretch marks. I was very depressed but it’s hard to say if that was because of the hormone imbalance or because I felt so bad and no one would listen to me.

I came across a little article in the Ladies Home Journal magazine which said “If you have these symptoms…ask your doctor about Cushing’s”. After that, I started reading everything I could on Cushing’s and asking my doctors. Due to all my reading at the library and medical books I bought, I was sure I had Cushing’s but no one would believe me. Doctors would say that Cushing’s Disease is too rare, that I was making this up and that I couldn’t have it.

I asked doctors for three years – PCP, gynecologist, neurologist, podiatrist – all said the now-famous refrain.  It’s too rare.  You couldn’t have Cushing’s.  I kept persisting in my reading, making copies of library texts even when I didn’t understand them, keeping notes.  I just knew that someone, somewhere would “discover” that I had Cushing’s.

My husband was on the doctors’ sides.  He was sure it was all in my mind (as opposed to all in my head!) and he told me to just think “happy thoughts” and it would all go away.

A Neurologist gave me Xanax. Since he couldn’t see my tumor with his Magnetic Resonance Imaging (MRI) machine there was “no possibility” that it existed. Boy was he wrong!

Later in 1986 I started bruising incredibly easily. I could touch my skin and get a bruise. On New Year’s Day of 1987 I started bleeding under the skin. My husband made circles around the outside perimeter each hour with a marker, like the rings of a tree. When I went to my Internist the next day he was shocked at the size. He now thought I had a blood disorder so he sent me to a Hematologist/Oncologist.

Fortunately, the Hematologist/Oncologist ran a twenty-four hour urine test and really looked at me. Both he and his partner recognized that I had Cushing’s. Of course, he was sure that he did the diagnosis.  No matter that I had been pursuing this with other doctors for 3 years.

It was not yet determined if it was Cushing’s Disease (Pituitary) or Syndrome (Adrenal). However, he couldn’t help me any further so the Hematologist referred me to an Endocrinologist.

The Endocrinologist, of course, didn’t trust the other tests I had had done so I was back to square one. He ran his own multitude of tests. He had to draw blood at certain times like 9 AM. and 5 PM. There was a dexamethasone suppression test where I took a pill at 10 p.m. and gave blood at 9 am the next day. I collected gallons of urine in BIG boxes (Fun in the fridge!). Those were from 6 a.m. to 6 a.m. to be delivered to his office by 9 a.m. same day. I was always worried that I’d be stopped in rush hour and the police would ask about what was in that big container. I think I did those for a week. He also did standard neurological tests and asked lots of questions.

When the endo confirmed that I had Cushing’s in 1987 he sent me to a local hospital where they repeated all those same tests for another week and decided that it was not my adrenal gland (Cushing’s Syndrome) creating the problem. The doctors and nurses had no idea what to do with me, so they put me on the brain cancer ward.

When I left this hospital after a week, we didn’t know any more than we had before.

As luck would have it, NIH (National Institutes of Health, Bethesda, Maryland) was doing a clinical trial of Cushing’s. I live in the same area as NIH so it was not too inconvenient but very scary at first to think of being tested there. At that time I only had a choice of NIH, Mayo Clinic and a place in Quebec to do this then-rare pituitary surgery called a Transsphenoidal Resection. I chose NIH – closest and free. After I was interviewed by the Doctors there, I got a letter that I had been accepted into the clinical trial. The first time I was there was for 6 weeks as an inpatient. More of the same tests.

There were about 12 of us there and it was nice not to be alone with this mystery disease. Many of these Cushies (mostly women) were getting bald, couldn’t walk, having strokes, had diabetes. One was blind, one had a heart attack while I was there. Towards the end of my testing period, I was looking forward to the surgery just to get this whole mess over with. While I was at NIH, I was gaining about a pound a day!

The MRI still showed nothing, so they did a Petrosal Sinus Sampling Test. That scared me more than the prospect of surgery. (This test carries the risk of stroke and uncontrollable bleeding from the incision points.) Catheters were fed from my groin area to my pituitary gland and dye was injected. I could watch the whole procedure on monitors. I could not move during this test or for several hours afterwards to prevent uncontrolable bleeding from a major artery. The test did show where the tumor probably was located. Also done were more sophisticated dexamethasone suppression tests where drugs were administered by IV and blood was drawn every hour (they put a heplock in my arm so they don’t have to keep sticking me). I got to go home for a weekend and then went back for the surgery – the Transsphenoidal Resection. I fully expected to die during surgery (and didn’t care if I did) so I signed my will and wrote last letters to those I wanted to say goodbye to. During the time I was home just before surgery, a college classmate of mine (I didn’t know her) did die at NIH of a Cushing’s-related problem. I’m so glad I didn’t find out until a couple months later!

November 3, 1987, the surgeon, Dr. Ed Oldfield, cut the gum above my front teeth under my upper lip so there is no scar. He used tiny tools and microscopes. My tumor was removed successfully. In some cases (not mine) the surgeon uses a plug of fat from the abdomen to help seal the cut. Afterwards, I was in intensive care overnight and went to a neurology ward for a few days until I could walk without being dizzy. I had some major headaches for a day or two but they gave me drugs (morphine) for those. Also, I had cotton plugs in my nostrils. It was a big day when they came out. I had diabetes insipidus (DI) for a little while, but that went away by itself – thank goodness!

I had to use a foam product called “Toothies” to brush my teeth without hitting the incision. Before they let me go home, I had to learn to give myself an injection in my thigh. They sent me home with a supply of injectible cortisone in case my level ever fell too low (it didn’t). I was weaned gradually off cortisone pills (scary). I now take no medications. I had to get a Medic Alert bracelet. I will always need to tell medical staff when I have any kind of procedure – the effects of my excess cortisone will remain forever.

I went back to the NIH for several follow-up visits of a week each where they did all the blood and urine testing again. After a few years NIH set me free. Now I go to my “outside” endocrinologist every year for the dexamethasone suppression test, 24-hour urine and regular blood testing.

As I get further away from my surgery, I have less and less chance that my tumor will grow back. I have never lost all the weight I gained and I still have the hair on my chin but most of my other symptoms are gone. I am still and always tired and need a nap most days. I do not, however, still need to take whole days off just to sleep.

I consider myself very lucky that I was treated before I got as bad as some of the others on my floor at NIH but think it is crazy that these symptoms are not taken seriously by doctors.

My story goes on and if you’re interested some is on this blog and some is here:

Forbes Magazine | MaryO’s bio | Cushing’s and Cancer Blog | Cushing’s Awareness Day Testimonial Archive |

Because of this experience in getting a Cushing’s diagnosis – and later, a prescription for growth hormone – I was concerned that there were probably other people not being diagnosed with Cushing’s. When I searched online for Cushing’s, all the sites that came up were for dogs and horses with Cushing’s.  Not what I was looking for!

In July of 2000, I was talking with my dear friend Alice, who ran a wonderful menopause site, Power Surge, wondering why there weren’t many support groups online (OR off!) for Cushing’s.  This thought percolated through my mind for a few hours and I realized that maybe this was my calling.  Maybe I should be the one to start a network of support for other “Cushies” to help them empower themselves.

I wanted to educate others about the awful disease that took doctors years of my life to diagnose and treat – even after I gave them the information to diagnose me.  I didn’t want anyone else to suffer for years like I did.  I wanted doctors to pay more attention to Cushing’s disease.

The first website (http://www.cushings-help.com) went “live” July 21, 2000.  It was just a single page of information. The message boards began September 30, 2000 with a simple message board which then led to a larger one, and a larger.  Today, in 2010, we have over 7 thousand members.  Some “rare disease”!

The message boards are stillactive and we have weekly online text chats, weekly live interviews, local meetings, conferences, email newsletters, a clothing exchange, a Cushing’s Awareness Day Forum, podcasts, phone support and much more. Because I wanted to spread the word to others not on “the boards” we have extended out to social networking sites – twitter groups, facebook groups, twines, friendfeeds, newsletters, websites, chat groups, multiply.com, and much, much more.

People are becoming more empowered and participating in their own diagnoses, testing and treatment.  This have changed a lot since 1983!

When I had my Cushing’s over 40 years ago (AARRGGHH!), I never thought that I would meet another Cushing’s patient in real life or online. Back then, I’d never even been aware that there was anything like an “online”. I’m so glad that people struggling with Cushing’s today don’t have to suffer anymore thinking that they’re the only one who deals with this.

Because of my work on the websites – and, believe me it is a ton of work! – I have had the honor of meeting over a hundred other Cushies personally at local meetings, conferences, at NIH (the National Institutes of Health in Bethesda, MD where I had my final diagnosis and surgery). It occurred to me once that this is probably more than most endocrinologists will ever see in their entire career. I’ve also talked to countless others on the phone. Amazing for a “rare” disease!

I don’t know what pushed me in 1983, how I got the confidence and self-empowerment to challenge these doctors and their non-diagnoses over the years.  I’m glad that I didn’t suffer any longer than I did and I’m glad that I have a role in helping others to find the medical help that they need.

What do *YOU* think?  How are you becoming empowered?

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Consecutive Resections of Double Pituitary Adenoma for Resolution of Cushing Disease

Posted on December 20, 2025 by MaryO

BACKGROUND

Double pituitary adenomas are rare presentations of two distinct adenohypophyseal lesions seen in <1% of surgical cases. Increased rates of recurrence or persistence are reported in the resection of Cushing microadenomas and are attributed to the small tumor size and localization difficulties. The authors report a case of surgical treatment failure of Cushing disease because of the presence of a secondary pituitary adenoma.

OBSERVATIONS

A 32-year-old woman with a history of prolactin excess and pituitary lesion presented with oligomenorrhea, weight gain, facial fullness, and hirsutism. Urinary and nighttime salivary cortisol elevation were elevated. Magnetic resonance imaging confirmed a 4-mm3 pituitary lesion. Inferior petrosal sinus sampling was diagnostic for Cushing disease. Primary endoscopic endonasal transsphenoidal resection was performed to remove what was determined to be a lactotroph-secreting tumor on immunohistochemistry with persistent hypercortisolism. Repeat resection yielded a corticotroph-secreting tumor and postoperative hypoadrenalism followed by long-term normalization of the hypothalamic-pituitary-adrenal axis.

LESSONS

This case demonstrates the importance of multidisciplinary management and postoperative hormonal follow-up in patients with Cushing disease. Improved strategies for localization of the active tumor in double pituitary adenomas are essential for primary surgical success and resolution of endocrinopathies.

ABBREVIATIONS

ACTH = adrenocorticotrophic hormone;  BMI = body mass index;  DHEA-S = dehydroepiandrosterone sulfate;  FSH = follicle-stimulating hormone;  GH = growth hormone;  IHC = immunohistochemical;  IPSS = inferior petrosal sinus sampling;  LH = luteinizing hormone;  MRI = magnetic resonance imaging;  POD = postoperative day;  T4 = thyroxine;  TF = transcription factor;  TSH = thyroid-stimulating hormone;  UFC = urinary free cortisol

Pituitary adenomas are adenohypophyseal tumors that can cause endocrinopathies, such as pituitary hormone hypersecretion or anterior hypopituitarism. Cell lineages are used to classify these tumors on the basis of immunohistochemical (IHC) staining of transcription factors, hormones, and other biomarkers.1 Pituitary adenomas differentiate from pluripotent stem cells along one of three lineage pathways, depending on the following active transcription factors (TFs): pituitary transcription factor 1 (PIT-1), T-box transcription factor (TPIT), or steroidogenic factor-1 (SF-1). Rarely, two or more discrete pituitary adenomas from different lineages are identified in patients; however, the etiology remains unclear.2 The incidence of multiple pituitary adenomas has been reported to be 1%–2% of all resected pituitary adenomas but is likely underestimated based on data from large autopsy series.1–4 Pluri-hormonal adenomas are also rare entities in which a single tumor contains multiple TF lineages with one or more hormonal excesses.1–3 Preoperative recognition of multiple or pluri-hormonal pituitary adenomas is rare, and most tumors are discovered incidentally upon autopsy, intraoperatively, or on histological analysis.2,3,5

In cases of multiple synchronous pituitary adenomas, only one hormone excess syndrome is most frequently evident on clinical presentation and endocrine workup. Silent pituitary tumors positive for prolactin on immunohistochemistry are the most prevalent additional, incidentally found tumor in cases of multiple pituitary adenomas.5 This is particularly true in Cushing disease.6,7 It is important to recognize the presence of multiple pituitary adenomas especially in the setting of hormonally active pituitary adenomas to provide optimal management for this subset of patients. Complete resection is curative for Cushing disease with the standard of care achieved through a transsphenoidal approach. Localization of the tumor presents a challenge because of suboptimal sensitivity of magnetic resonance imaging (MRI) in demonstrating microadenomas, the inconsistency of lateralization with inferior petrosal sinus sampling (IPSS), and delays in pathological analysis.1,8,9 Additionally, the presence of an additional pituitary adenoma can obscure the microtumor through its large size and mass effect and can act as a “decoy lesion” during MRI, IPSS, and resection.6

Consideration of multiple pituitary tumors is necessary for successful resection. In a patient with a biochemical picture of Cushing disease, the demonstration of an adenoma with negative adrenocorticotrophic hormone (ACTH) immunostaining and the absence of postoperative hypoadrenalism may indicate the existence of a double adenoma. Few cases have described a lack of remission of an endocrinopathy after transsphenoidal resection due to the presence of an additional adenoma,2,6,10 and even less so in the instance of the persistence of Cushing disease.6 We present a rare case of double pituitary adenomas in a patient presenting with Cushing disease who underwent two endoscopic endonasal transsphenoidal resections and immunostaining for prolactin and ACTH, respectively, with long-term normalization of her hypothalamic-pituitary-adrenal (HPA) axis.

Illustrative Case

History and Presentation

A 32-year-old female, gravida 0 para 0, with a history of a pituitary lesion and hyperprolactinemia presented to our institution for the evaluation for Cushing disease. Ten years earlier, the patient had presented to a gynecologist with hirsutism, galactorrhea, and oligomenorrhea. Her endocrine workup was remarkable for an elevated prolactin at 33.8 ng/mL (2.3–23.3 ng/mL), while follicle-stimulating hormone (FSH), luteinizing hormone (LH), and thyroid-stimulating hormone (TSH) levels were normal. No ACTH or cortisol levels were available. MRI demonstrated a 5 × 6 × 5–mm T1-weighted isointense pituitary lesion protruding into the suprasellar cistern due to a small sella size. She was treated with bromocriptine 2.5 mg daily for 5 years, with normalization of her prolactin level. Subsequent MRI demonstrated a stable lesion size and T1 and T2 hyperintensity in the region of the known pituitary lesion, considered to be posttreatment cystic change with proteinaceous contents and blood. After the normalization of her prolactin levels, she continued to have oligomenorrhea and abnormal hair growth. Polycystic ovaries were not visualized on ultrasound. She was started on oral contraceptives and then switched to the etonorgestrel implant.

A decade after initial presentation, she presented to endocrinology at our institution with 3 years of weight gain, hirsutism, and potential oligomenorrhea. Vital signs were stable (blood pressure: 122/86; heart rate: 72 beats/min), and facial fullness and striae on her bilateral breasts were appreciated on physical examination. She was taking isoniazid and pyridoxine for a recent diagnosis of latent tuberculosis and had discontinued bromocriptine 5 years earlier. Her weight was 66.3 kg and body mass index (BMI) was 23.9 kg/m2. She reported that her maternal uncle had a pituitary tumor. Laboratory analysis was positive for elevated urinary free cortisol (UFC) of 109 µg per 24 hours (2.5–45 µg/24 h; Table 1) and nighttime salivary cortisol of 142 ng/mL (<100 ng/dL) with high-normal prolactin of 22.8 ng/mL (2.3–23.3 ng/dL) and normal FSH, LH, TSH, and thyroxine (T4). Dehydroepiandrosterone sulfate (DHEA-S) was 128 µg/dL (98.8–340.0 µg/dL). Imaging demonstrated a 4 × 4 × 4–mm pituitary lesion with decreased T1-weighted and increased central T2-weighted signal intensity in the left lateral pituitary (Fig. 1A–C). Desmopressin (Ferring Pharmaceuticals DDAVP) stimulation increased a basal ACTH of 49.9 pg/mL to ACTH of 91.2 pg/mL, and cortisol increased from 13.7 µg/dL to 21.2 µg/dL, consistent with neoplastic hypercortisolism. IPSS was performed, which showed a right-sided, central-to-peripheral ACTH gradient (Table 2). The patient elected to undergo endoscopic endonasal resection with the initial target as the left-lateral pituitary mass to achieve a cure for Cushing disease.

TABLE 1Urinary free cortisol at baseline and 3, 5, and 7 months after the primary resection

Variable Baseline 3 Mos 5 Mos 7 Mos on Osilodrostat
Urinary free cortisol (4–50 µg/24 hrs) 109 134.2 125.4 40.3
Urinary creatinine (0.5–2.5 g/24 hrs) 0.995 1.17 1.42 1.11
Urinary vol (mL) 1950 2300 2100 2125
FIG. 1
FIG. 1

Preoperative coronal precontrast (A) and postcontrast (B) T1-weighted magnetic resonance imaging (MRI) and T2-weighted MRI (C) demonstrated a 4-mm3 lesion (arrows) with decreased T1 and increased central T2 signal intensity in the left lateral pituitary. Two days after surgery, coronal precontrast (D) and postcontrast T1-weighted (E) and T2-weighted (F) MRI demonstrated the unchanged adenoma.

TABLE 2Preoperative inferior petrosal sinus sampling with corticorelin ovine triflutate 68 µg

Time (mins) ACTH (pg/mL) Prolactin (ng/mL)
Peripheral Petrosal Sinus ACTH Ratio Peripheral Petrosal Sinus Prolactin Ratio
Rt Lt Rt Lt Rt Lt Rt Lt
−5 50.6 225 1586 4.45 31.34 21 124 295 5.90 14.05
0 48.8 389 1376 7.97 28.20 22.2 185 198 8.33 8.92
3 69.8 4680 1333 67.05 19.1 22.1 396 32.5 17.92 1.47
5 80.9 4590 1623 56.74 20.06 22.1 436 32.2 19.73 1.46
10 112 4160 1660 37.14 14.82 20.2 367 42 17.90 2.05

ACTH or prolactin ratio = inferior petrosal sinus ACTH or prolactin/peripheral blood ACTH or prolactin.

Primary Resection and Outcomes

During the primary resection, abnormal tissue was immediately visible after a linear incision along the bottom of the dura, with an excellent plane of dissection. The inferomedial adenoma was distinct from the known left lateral lesion, and the resection was considered complete by the primary neurosurgeon. Subsequently, the left-sided adenoma was not pursued because of the historical prolactinoma diagnosis and an assumption that the newly discovered adenoma was the cause of ACTH hypersecretion. However, pathology of the inferomedial tumor was strongly and diffusely positive for prolactin (Fig. 2B), synaptophysin, and cytokeratin, with an Mindbomb Homolog-1 (MIB-1) proliferative index of 2.4%. ACTH, growth hormone (GH), FSH, LH, and TSH immunostaining were negative. TF immunohistochemistry was not available. On postoperative day (POD) 1, pituitary MRI was performed and demonstrated the unchanged 4-mm3 T1-weighted hypointense lesion with small central T2-weighted hyperintensity in the left lateral gland (Fig. 1D–F). Cortisol levels ranged from 9.7 to 76.2 µg/dL (4.8–19.5 µg/dL), and ACTH was 19.5 pg/mL (7.2–63.3 pg/mL) on POD 1.

FIG. 2
FIG. 2

Histological examination of surgical specimens from the inferomedial (A–C) and left lateral (D–F) lesions. The initial resection (hematoxylin and eosin [H&E], A) was strongly and diffusely positive for prolactin (B) with normal reticulin levels (C) indicating a lactotrophic pituitary adenoma. The second operation (H&E, D) was diagnostic for a corticotropic pituitary adenoma with diffusely positive adrenocorticotrophic hormone (ACTH) (E) and decreased reticulin (F). Original magnification ×100.

Early reoperation was discussed with the patient based on the pathology and persistent hypercortisolism; however, she elected to pursue conservative management with close follow-up. Postoperative cortisol nadir was 4.8 µg/dL (4.8–19.5 µg/dL) on POD 2 during her 4-day hospital stay. DHEA-S was significantly decreased from baseline at 22.3 µg/dL (98.8–340.0 µg/dL) and a prolactin level of 3.4 ng/mL (2.3–23.3 ng/dL) was low-normal. No glucocorticoids were administered during her hospital course. There was no clinical evidence of vasopressin deficiency while she was an inpatient.

Three months postoperatively, the patient reported insomnia, poor hair quality, fatigue, nocturnal sweating, and continued increasing weight gain with fat accumulation in the supraclavicular and dorsal cervical area. She had one spontaneous menstrual period despite the use of etonogestrel implant. UFC was increased at 134.2 µg/24 hours (4–50 µg/24 h; Table 1). The 8:00 am serum cortisol was 10.2 µg/dL (5.0–25.0 µg/dL). She was started on osilodrostat 2 mg twice daily for her persistent hypercortisolism, and she reported some clinical improvement; however, she had continued elevation in her late-night salivary cortisol levels up to 7.0 nmol/L. Other endocrine lab work was normal, with a prolactin of 13.5 ng/mL (2.8–23.3 ng/mL) and TSH of 3.67 µIU/mL (0.4–4.0 µIU/mL). Her weight had increased by 4.9 kg to 71.2 kg with a BMI of 25.3 kg/m2. Approximately 6 months postoperatively, she was amenable to a secondary resection targeting the remaining left lateral pituitary adenoma.

Secondary Resection and Outcomes

After obtaining adequate exposure for the secondary resection, the lesion in the left lateral aspect of the pituitary was targeted. The tumor was clearly identified and completely resected without intraoperative complication. IHC staining was diffusely positive for ACTH (Fig. 2E), synaptophysin, and cytokeratin with decreased reticulin and an MIB-1 index of 3.3%. Prolactin, GH, TSH, LH, and FSH immunostaining were negative. Postoperative cortisol monitoring demonstrated decreased levels, with a nadir of 2.0 µg/dL on POD 0. Levels of ACTH and DHEA-S were decreased at 4.4 pg/mL (7.2–63.3 pg/mL) and 13.3 µg/dL (98.8–340 µg/dL), respectively, on POD 1. Prolactin remained within the normal range at 8.2 ng/mL (2.8–23.3 ng/mL). The patient was started on intravenous hydrocortisone 50 mg every 8 hours for adrenal insufficiency. Postoperative symptoms of nausea, headache, and muscle weakness resolved with hydrocortisone administration. She was discharged on hydrocortisone 60 mg daily in divided doses for adrenal insufficiency and had no signs of vasopressin deficiency during her 2-day hospital course.

By 3 months, the patient reported decreased fatigue, myalgia, and insomnia and improved overall well-being and physical appearance. She was weaned down to a total daily dose of 20 mg of hydrocortisone and had lost 5.2 kg. Her menstruation returned while having an etonogestrel implant. Rapid ACTH stimulation was abnormal, with decreased cortisol at 30 minutes of 4.1 µg/dL (7.2–63.3 pg/mL) demonstrating continued adrenal insufficiency. Follow-up MRI demonstrated miniscule remaining left pituitary adenoma (Fig. 3). Seven months after her second surgery, she was started on 50 µg levothyroxine for primary hypothyroidism in the setting of slightly elevated TSH of 4.1 µIU/mL (0.4–4.0 µIU/mL) and a low-normal T4 of 0.8 ng/dL (0.7–1.5 ng/dL).

FIG. 3
FIG. 3

Postoperative imaging 3 months after the second operation demonstrates near gross-total resection (yellow arrows: surgical cavity) of the left lateral pituitary adenoma on coronal precontrast (A) and postcontrast T1-weighted (B) and T2-weighted (C) MRI.

Two years after the second resection, the patient lost 10.1 kg (weight, 61.1 kg; BMI, 21.76 kg/m2). Her ACTH stimulation test became normal, and hydrocortisone therapy was discontinued. At the 2-year time point, the patient and her husband successfully conceived a child.

Patient Informed Consent

The necessary patient informed consent was obtained in this study.

Discussion

Double or multiple pituitary adenomas are discovered in 0.37%–2.6% of resected pituitary lesions.3,4,6,11,12 A majority of multiple pituitary adenomas are not suspected before surgery with an inconclusive clinical presentation or endocrine laboratory workup.6 The presentation of multiple synchronous neoplasms is thought to be more common than having a single neoplasm with multiple lineages.1 Studies have shown that additional pituitary adenomas are seen at a rate of 1.6%–3.3% in Cushing disease in studies including both contiguous and noncontiguous double pituitary adenomas.6 Additional pituitary adenomas that are hormonally active make up 40% of resected double pituitary adenomas, with most staining for gonadotroph adenoma.13 Overall, the most common incidental pituitary adenoma is prolactinoma,6 which occurs most frequently with GH or ACTH adenomas.5 In very rare instances, Cushing cases can present with hyperprolactinemia and Cushing synchronously.6 Hormonal secretion and clinical presentation are variable, with the pathology most often attributed to only one component of double pituitary adenoma.3,14 The multiple-hit theory is the most common hypothesis for double pituitary adenoma etiology with coincidental monoclonal expansion of two or more lineages, which present with separate pseudo-capsules for each lesion.15

Observations

On presenting with Cushing disease, the differential diagnosis before the initial operation considered that the known left lateral pituitary adenoma could be a mixed tumor with both prolactin and ACTH lineages. Therefore, it was the initial target of the resection until discovering the second adenoma intraoperatively. With two distinct adenomas, the inferomedial adenoma was presumed to be the source of the ACTH hypersecretion and was subsequently resected. The left lesion was thought to be a prolactinoma and hormonally inactive after historical dopaminergic therapy and thus was not pursued during the initial surgery. However, pathology confirmed that the opposite was true. Few cases have also involved incidental pituitary tumors that look like the hormonally active adenoma and encourage resection of it, leaving the primary pituitary adenoma behind.6,7 It has been reported that these “decoy lesions” can cause surgical failure and require secondary operations.6,7,10,16 Intraoperative localization and confirmation of the adenoma classification may have also been helpful during the case, including tissue-based ACTH antibody assay,9 plasma ACTH measurements with a immunochemiluminometric method,17 or intraoperative ultrasound.5,6

The inferomedial second tumor was not appreciated or reported throughout her serial MRI studies from 2010 to 2020. Interestingly, imaging did demonstrate the left pituitary adenoma that was medically treated as a prolactinoma, although it was later diagnosed as an ACTH-secreting lesion on IHC staining. Preoperative visualization of a pituitary adenoma in Cushing disease is reported to be limited, with a reported 50% incidence with negative MRI with standard 1.5 T.1,18,19 MRI technical refinements in magnet strength, slice thickness, or enhanced spin sequences have increased sensitivity, but one-third of patients with Cushing disease still have negative scans.20 Small prolactinomas, especially those near the cavernous sinus, are also notoriously difficult to visualize on MRI, although recent advances using co-registration of 11C-methionine positron emission tomography–computed tomography with MRI (Met-PET/MRICR) may prove useful.21 Difficulty with preoperative visualization complicates a diagnosis of multiple adenomas, with or without multiple endocrinopathies, and negatively affects surgical planning. In a single-institution retrospective review of MRI in all cases of double pituitary tumors, only one of eight patients (12.5%) over 16 years of age had a positive MRI for double pituitary tumors and was diagnosed preoperatively.2

The patient’s preoperative IPSS demonstrated a right central-to-peripheral gradient. This was incongruent with the MRI demonstrating the single left-sided tumor. While IPSS is useful in confirming Cushing disease, its sensitivity for lateralization has been reported at only 59%–71%.9 With this in mind and a known left-sided adenoma on MRI, exploration of the right side of the pituitary was not originally planned. Ultimately, the left-sided adenoma was the source of ACTH hypersecretion, which remains incongruent with preoperative IPSS. It has been suggested that multiple pituitary adenomas in Cushing disease could further decrease its accuracy.1,6

The patient’s initial historical prolactin levels (33.8 ng/dL) were lower than reported levels of 100–250 ng/dL for microadenoma and >250 ng/dL in cases of macroadenoma. Normally, in active single prolactinoma, prolactin secretion is correlated to size. We do not suspect that the presence of more than one pituitary adenoma would affect the level of prolactin hypersecretion.6 Slight elevations in prolactin can be attributed to causes such as pituitary stalk effect, medications, and physiological stimulation. During the 5 years of bromocriptine therapy, the effect on the inferomedial prolactinoma was unknown, as it was not appreciated on MRI. There are reports of prolactinomas being less responsive to dopaminergic agonist therapy in cases of double adenomas.14,22 Upon resection of the inferomedial prolactinoma during the initial operation, there was no further change in the patient’s prolactin levels, which could most likely be attributed to prior dopaminergic therapy. Unfortunately, the initial endocrine laboratory workup did not include levels of ACTH or cortisol. In addition to hyperprolactinemia, Cushing disease can also present with changes in menstruation. After the secondary resection and removal of the ACTH-secreting pituitary adenoma, the patient’s oligomenorrhea resolved and she achieved pregnancy. Retrospectively, it remains unclear if the prolactinoma was once truly active hormonally.

Lessons

The rare presence of two pituitary adenomas can complicate the diagnosis, medical and surgical management, and long-term outcomes for patients. A complete endocrine workup is essential when a pituitary adenoma is suspected and can help screen for pluri-hormonal and multiple pituitary adenomas. In our patient, it is unknown when the onset of hypercortisolism was with the limited initial hormonal workup.

Currently, localizing and resecting the hormonally active adenoma in double or multiple pituitary adenomas remain a challenge, with limitations in preoperative imaging and intraoperative measures. After encountering the additional inferomedial lesion during surgery, resection of both adenomas during the initial surgery may have been prudent to ensure the resolution of Cushing disease. Although exploration for additional pituitary adenomas is not usually recommended, it could be considered in cases of multiple pituitary adenomas and uncertainty of the culprit of Cushing disease.

The current characterization of pituitary tumors by the World Health Organization includes immunohistochemistry for both transcription factors and pituitary hormones, with clinical usefulness to be determined by future studies. Multiple lineages can occur mixed in a single pituitary adenoma or across different noncontiguous adenomas and can only be determined by TF immunostaining. The left ACTH-staining lesion in our patient had some shrinkage and MRI changes, which may have been a response to dopaminergic therapy. Full characterization of the tumor cell lineages in this case remains undetermined without staining for TFs.

In conclusion, we report a rare case of Cushing disease concurrent with a prolactinoma leading to the need for repeat resection. This is one of the few reported cases of a double pituitary adenoma leading to a lack of biochemical remission of hypercortisolism after the initial surgery. Strategies for localization of the active tumor in double pituitary adenomas are essential for primary surgical success and the resolution of endocrinopathies.

Author Contributions

Conception and design: Zwagerman, Tavakoli, Shah, Findling. Acquisition of data: Zwagerman, Armstrong, Tavakoli, Shah, Ioachimescu, Findling. Analysis and interpretation of data: Zwagerman, Armstrong, Tavakoli, Shah, Coss, Ioachimescu, Findling. Drafting of the article: Zwagerman, Armstrong, Shah. Critically revising the article: Zwagerman, Armstrong, Tavakoli, Shah, Ioachimescu, Findling. Reviewed submitted version of the manuscript: Zwagerman, Armstrong, Tavakoli, Shah, Laing, Ioachimescu, Findling. Approved the final version of the manuscript on behalf of all authors: Zwagerman. Statistical analysis: Armstrong, Shah. Administrative/technical/material support: Zwagerman, Armstrong, Shah. Study supervision: Zwagerman, Tavakoli, Shah, Laing.

References

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Misconceptions About Cushing’s

Posted on December 14, 2025 by MaryO

Cushing’s.  So many people are confused by what it is and what it isn’t.  They may have heard of it because a dog they know has it – or, these days, a horse, ferret, rat.  Seems it’s way more common in lots of animals but not people.

If people have heard of the “animal version” they might say “Yeah, my dog had that and it was easy to diagnose. We just gave him medication…”

When we first started having bios on the website, sometimes people would say that they had Cushions Disease.  At first I wondered about that but then it started to make more sense.  If you’ve never heard of the disease,  the doctor mumbles something.  You know you’re a little “fluffy” and cushions makes a lot of sense.

Twice in the last week I’ve seen Cushing’s described as Crushings Disease.  That sort of makes sense, too.  Cushing’s crushes your plans, relationships, credibility, pretty much everything.

Other misconceptions involve Cushing’s symptoms.  Others, especially doctors, will see you gain weight and assume you’re eating too much and a good diet will fix everything.  Or see that you’re depressed (who wouldn’t be!) and offer anti-depressants.

Doctors may say that Cushing’s is too rare, that they’ll never see a case of it in their practice.  But rare doesn’t mean that no one gets it.  Rare doesn’t mean that doctors shouldn’t test for it.

Then, the anatomy just isn’t right.  People say that they have a brain tumor instead of a pituitary tumor.  I just read this on another site: The pituitary gland is on the bottom of the brain… Umm – not exactly ON the bottom of the brain but maybe close enough for people to get an idea.

What sorts of things about Cushing’s/Cushions/Crushings that just weren’t quite right?

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Co-Occurrence of Endogenous and Exogenous Cushing’s Syndromes: Does “Double Cushing Syndrome” Really Exist? A Case Report

Posted on December 1, 2025 by MaryO

ABSTRACT

Double Cushing syndrome exists: exogenous steroid use can mask concurrent adrenal hypercortisolism. When symptoms persist and cortisol remains high after tapering or stopping prescribed glucocorticoids, an endogenous source is likely. Early recognition with ACTH testing, dexamethasone suppression, and adrenal imaging reduces misdiagnosis, favors timely surgery, and supports safe tapering.

1 Introduction

Cushing syndrome (CS) is a non-physiological increase in plasma glucocorticoids [1]. In most cases, the source of increased plasma glucocorticoids is caused by exogenous steroid administration, which is quite common, and about 1% of the world population is on long-term (more than 3 months) oral glucocorticoids [1, 2]. On the contrary, endogenous overproduction of glucocorticoids is rare, and annually, only two to eight per million people are diagnosed with endogenous CS [3]. The simultaneous occurrence of endogenous and exogenous CS is an exceptionally uncommon phenomenon. This dual manifestation has been reported in a few case reports, highlighting its rarity and the complex diagnostic and therapeutic challenges it poses [4, 5]. Therefore, in this study, we discuss a patient who presented with cushingoid features and was simultaneously diagnosed with both endogenous and exogenous CS or, as it is called, double CS.

2 Case Presentation

The patient was a 46-year-old male with a history of new-onset hypertension and recurrent deep vein thrombosis (DVT) who was referred to our endocrinology clinic with a chief complaint of hip pain and weakness of the lower limbs. In the past 3 years, the patient had been receiving 50 mg/day of oral prednisolone and inhalation powder of Umeclidinium and Vilanterol (62.5/25 μg/dose) because of respiratory complications that started after Coronavirus Disease 2019 (COVID-19) vaccination. After 3 months of corticosteroid treatment, he experienced DVT for the first time when he was started on rivaroxaban. However, while he was on treatment, the second DVT occurred 1 month before his referral, and therefore, rivaroxaban was changed to warfarin 5 mg/day.

The patient also mentioned weight gain with his body mass index (BMI) rising from 26 to 31 kg/m2, progressive weakness of proximal muscles, easy bruising, decreased libido, mood changes with mostly euphoric mood, and irritability during the last 2 years. Moreover, multiple osteoporotic fractures of ribs, clavicle, sternum, and lumbar vertebrae were added to his symptoms in the past 5 months. At that time, he underwent bone densitometry, which revealed osteopenia of the left hip with a Z-score of −1.3 and severe osteoporosis of total lumbar spine with a T-score of −3.9. He started taking calcium and vitamin D3 supplements and received a single injection of 750 μg/3 mL teriparatide 30 days before his referral to our center.

Two months ago, the patient gradually reduced the dosage of prednisolone by tapering the dose to 12.5 mg/day. However, a month later, the hip pain and muscle weakness worsened to such an extent that the patient was unable to walk. Due to his signs and symptoms, the patient was referred to our center for further evaluation of CS. The patient also mentioned a history of nephrolithiasis, new-onset hypertension, and lower limb edema, for which he was started on eplerenone 25 mg and furosemide 20 mg tablets once daily. In his family history, the patient’s mother had type 2 diabetes mellitus, and his two sisters had a history of nephrolithiasis. The patient did not mention any history of allergies to medications or foods. He was addicted to opium and had 15 pack-years of smoking, but he did not mention alcohol consumption.

Upon admission, the patient presented with a blood pressure of 150/83 mmHg, heart rate of 74 bpm, respiratory rate of 20/min, temperature of 36.5°C, oxygen saturation of 93%, and BMI of 31 kg/m2. He was sitting in a wheelchair due to weakness and severe pain in the hip. On physical examination, the patient showed the features of CS, including moon face, buffalo hump, central obesity, facial plethora, thin and brittle skin, acne, and purple stretch marks (striae) on the flanks (Figure 1). Proximal muscle weakness in the lower limbs with a muscle force grade of 4/5 and 3+ edema was also observed. Laboratory investigations are shown in Table 1.

Details are in the caption following the image

De-identified clinical photographs illustrating the Cushingoid phenotype. (A) Overall habitus with marked central (truncal) adiposity. (B) Rounded plethoric face (“moon facies”). (C) Relatively slender distal extremities compared with truncal obesity. (D) Dorsocervical fat pad (“buffalo hump”). (E) Upper thoracic/supraclavicular fat accumulation. (F) Protuberant abdomen with wide violaceous striae.
TABLE 1. Laboratory findings of case report.
Laboratory test Patient value (in-hospital) Patient value (follow-up) Reference range
On admission
Hemoglobin (g/dL) 16.6 13.6 13.5–17.5
Hematocrit (%) 49.5 42.1 42–52
WBC (white blood cells; 103/μL) 11.8 7.1 4.0–11.0
PLT (platelet count; 103/μL) 286 294 150–450
BUN (blood urea nitrogen; mg/dL) 10 11 7–18
Cr (creatinine; mg/dL) 0.9 0.9 0.7–1.3
ALP (alkaline phosphatase; IU/L) 1016 129 44–147
AST (aspartate aminotransferase; IU/L) 48 30 < 31
ALT (alanine transaminase; IU/L) 88 21 < 31
CRP (C-reactive protein; mg/dL) 31 3 < 5
ESR (erythrocyte sedimentation rate; mm/h) 63 24 < 15
Sodium (mEq/L) 148 141 136–145
Potassium (mEq/L) 4.8 4.3 3.5–5
FBS (fasting blood glucose; mg/dL) 97 89 80–100
TC (total cholesterol; mg/dL) 267 182 < 200
TG (triglyceride; mg/dL) 148 104 < 200
LDL (low-density lipoprotein; mg/dL) 138 98 < 130
HDL (high-density lipoprotein; mg/dL) 64 55 30–70
In hospital
Cortisol 8 a.m. fasting (μg/dL) 20.2 14.1 4.3–24.9
ACTH (adrenocorticotropic hormone; pg/mL) < 1 — 7.2–63.3
1 mg Overnight dexamethasone suppression test (μg/dL) 16.5 — < 1.8

3 Methods (Differential Diagnosis, Investigations, and Treatment)

Initially suspected of having exogenous-induced CS, the patient’s prednisolone was on hold for 3 days. Cortisol 8 a.m. fasting level, measured with Electrochemiluminescence (ECL) and adrenocorticotropic hormone (ACTH) test, was 20.2 μg/dL (585.4 nmol/L) and < 1 pg/mL, respectively. Due to the lack of suppression of serum cortisol despite not using oral glucocorticoids, the absence of adrenal insufficiency symptoms, and the fact that the patient’s symptoms remained unchanged during this period, co-occurrence of endogenous CS was suspected.

A 1 mg overnight dexamethasone suppression test was performed to confirm endogenous CS diagnosis, and the results were reported as 16.5 μg/dL (normal range < 1.8 μg/dL). Considering the possibility of an ACTH-independent CS, the patient underwent an abdominopelvic multidetector computed tomography (MDCT) of abdominopelvic with adrenal protocol, which revealed a well-defined lesion with an approximate size of 32.8 × 38.6 mm in the left adrenal gland with a radiodensity of 90 Hounsfield units and a normal right adrenal gland (Figure 2). Moreover, evidence of previous old fractures as multiple callus formation was seen involving the clavicles, sternum, bilateral ribs, ischium, and pelvic bones. Multilevel old stable compression fractures of thoracic and lumbar vertebral bodies were also present. The differential diagnoses were glucocorticoid secretory adrenal tumors, including adrenal cell carcinoma and lipid-poor adenoma. In order to rule out pheochromocytoma, 24-h urine catecholamines were measured, and the results were negative.

Details are in the caption following the image

Abdominopelvic multidetector computed tomography (MDCT) with adrenal protocol showing a well-defined lesion with an approximate size of 32.8 × 38.6 mm in the left adrenal gland; radiodensity 90 HU. (A) Transverse plane. (B) Coronal plane. (C) Sagittal plane.

Finally, the patient underwent left adrenalectomy and corticosteroid replacement therapy due to the suppression of the other adrenal gland. According to the post-operative pathological investigations, immunohistochemistry markers reported as negative chromogranin, positive melan-A and inhibin, less than 3% Ki-67 marker, and lipid-poor adrenal cortical adenoma without invasions were diagnosed (Figure 3).

Details are in the caption following the image

Immunohistochemistry of the adrenal lesion (all panels acquired with a 100× oil-immersion objective; 10× eyepiece; original magnification ×1000). (A) Positive inhibin, (B) Positive Melan-A, (C) Less than 3% Ki-67 marker, and (D) Negative chromogranin.

4 Results (Outcome and Follow-Up)

Within 3 months after the operation, the patient’s corticosteroid was tapered and then discontinued due to the normalization of the cortisone serum test (14.1 μg/dL). Proximal limb weakness and hip pain, which had deprived the patient of the ability to move, gradually improved so that he could walk easily and perform daily activities. The signs and symptoms related to CS, including the patient’s mood, skin signs, and general appearance, returned to normal. The patient has been followed up for 6 months after the surgery. The patient’s BMI decreased to 24 kg/m2, and he stopped his anti-hypertensive medications with a blood pressure of 100/60 mmHg without previously prescribed drugs. So far, the laboratory tests have been within the normal range, and he has no complaints (Table 1).

5 Discussion

The described case was diagnosed with a cortisol-producing adrenocortical adenoma accompanied by exogenous CS. CS is an uncommon clinical condition caused by prolonged exposure to increased cortisol levels, which can be due to endogenous or exogenous factors [6]. Endogenous CS is infrequent and is classified as ACTH-dependent (80% of cases) or ACTH-independent (20% of cases) [7]. In the ACTH-independent category, adrenal adenoma accounts for 60% of cases and only 12% of cases of endogenous CS [7, 8]. Exogenous CS mainly occurs due to prolonged administration of glucocorticoids, which are used to manage a broad spectrum of diseases such as inflammatory, autoimmune, or neoplastic disorders and are the most common cause of CS worldwide [9]. Multiple factors, including formulation, duration of administration, pharmacokinetics, affinity, and potency of exogenous glucocorticoids, affect the probability of exogenous CS, but all forms of glucocorticoids can induce CS [10].

In the setting of cushingoid clinical features with chronic administration of high-dose glucocorticoids, especially oral prednisolone, the probability of exogenous CS is remarkably high; therefore, CS diagnostic approaches suggest that the first step after confirmation of cortisol excess is ruling out exogenous glucocorticoid administration [7, 8, 10]. Therefore, the possibility of co-occurrence of endogenous CS with iatrogenic CS is extremely low, and the diagnosis requires high clinical suspicion [4].

Differentiating endogenous and exogenous CS based on clinical features can be challenging and far-fetched. However, a few points can help physicians distinguish between these two. First, exogenous CS symptoms tend to be more striking, while endogenous CS appears more gradually. Second, hypertension, hypokalemia, and features of androgen excess, such as acne and hirsutism, are more common in endogenous CS [4, 10]. In addition, endogenous CS should be suspected if the patient’s symptoms continue after corticosteroid discontinuation or if the serum cortisol level is high despite corticosteroid cessation. In our case, the patient had a high cortisol level despite stopping prednisolone for 3 days, and he did not have any symptoms of adrenal insufficiency despite stopping prednisolone suddenly. Consequently, it was suspected that glucocorticoids might come from an endogenous source. Because ACTH was suppressed concurrently with elevated cortisol, non-ACTH-dependent CS was suspected, and MDCT of abdominopelvic confirmed it.

So far, few similar cases of simultaneous endogenous and exogenous CS have been reported. The first case was a 23-year-old woman with juvenile idiopathic arthritis who was administered high doses of triamcinolone for 16 years [4]. The development of cushingoid features that favored endogenous CS, such as hirsutism and acne, strengthened the suspicion of endogenous CS, and a CT scan revealed hypercortisolism with a bulky and nodular left adrenal gland, and a double CS was confirmed [4]. The second case was a 66-year-old woman diagnosed with exogenous CS after consumption of Traditional Chinese medicine (TCM) for a year [5]. The cessation of TCM did not significantly improve her cushingoid features, and she developed additional CS complications, including hypertension, diabetes mellitus, and osteoporotic fractures over the next 8 years. CS workup revealed a right-sided adrenal adenoma, and after the adrenalectomy, her clinical cushingoid features markedly improved [5]. These cases suggest that exogenous and endogenous CS can exist simultaneously in the same person. Although it is very rare, it should be considered in a person who still complains of CS symptoms after corticosteroid cessation. We suggest clinicians evaluate the patients for the disappearance of exogenous CS symptoms after tapering and stopping glucocorticoids. If the symptoms remain, they should be evaluated for endogenous CS.

6 Conclusion

The co-occurrence of an endogenous CS in the setting of an exogenous CS is curious. The diagnosis is based on a high clinical suspicion. Clinicians should evaluate patients for symptom resolution after tapering and discontinuing corticosteroids. Clinical cushingoid features that do not resolve after discontinuing exogenous glucocorticoids and high cortisol levels despite discontinuing corticosteroids should raise clinicians’ suspicion of the co-occurrence of exogenous and endogenous CS.

Author Contributions

Reza Amani-Beni: investigation, methodology, writing – original draft, writing – review and editing. Atiyeh Karimi Shervedani: methodology, writing – original draft. Bahar Darouei: conceptualization, validation, writing – review and editing. Matin Noroozi: methodology, writing – original draft. Maryam Heidarpour: conceptualization, supervision, validation, writing – review and editing.

Acknowledgments

The authors have nothing to report.

Consent

Written informed consent was obtained from the patient to publish this report, including de-identified clinical photographs, in accordance with the journal’s patient consent policy.

Conflicts of Interest

The authors declare no conflicts of interest.

Data Availability Statement

The data that supports the findings of this study are available on request of the corresponding author. The data are not publicly available due to privacy restrictions.

https://onlinelibrary.wiley.com/doi/10.1002/ccr3.71419

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“The Healthcare System Did Fail Me Repeatedly”: a Qualitative Study On Experiences Of Healthcare Among Canadian Women With Cushing’s Syndrome

Posted on October 4, 2024 by MaryO

Abstract

Background

As a rare endocrine disorder, Cushing’s Syndrome (Cushing’s) is characterized by numerous symptoms and a non-specific presentation, leading to a delay to diagnosis for patients with this disease. To date, research examining the lived experiences of patients with Cushing’s in healthcare is absent in the literature. This preliminary inquiry into the healthcare experiences of women with Cushing’s aimed to examine the utility of this line of inquiry to support the patient centered care of individuals with Cushing’s.

Methods

Seven women from across Canada with endogenous Cushing’s participated in the study. Semi-structured interviews were conducted examining participants’ healthcare and body-related experiences with Cushing’s. Results pertaining to healthcare experiences were analyzed for the current study using reflexive thematic analysis.

Results

Four themes emerged whereby women with Cushing’s experienced (1) a lack of patient centered care, characterized by provider miscommunication and medical gaslighting; (2) a misunderstanding of their symptoms as related to weight gain; (3) weight stigma in healthcare encounters; and (4) a shift in their quality of care following diagnosis.

Conclusions

The results highlight the importance of patient centered care as well as the negative impact of commonly reported barriers to patient centered care. Cushing’s specific barriers to patient centered care may include weight stigma as well as the rare incidence of Cushing’s. Further research is needed to better understand the healthcare experiences of people with Cushing’s in Canada.

Peer Review reports

Cushing Syndrome (Cushing’s) is a rare disorder caused by an increase in circulating free cortisol [1]. As receptors for glucocorticoids are widespread throughout the body, the effect of this increase in circulating cortisol is prolific [2]. Symptoms include uncontrollable weight gain, dorsocervical fat pad, facial plethora, purple striae, easy bruising, fatigue, proximal myopathy, hypertension, as well as menstrual irregularities and hirsutism among women [1, 3]. While Cushing’s is most often caused by corticosteroids (i.e., exogenous Cushing’s) with a more easily identifiable cause, this research is focused on endogenous Cushing’s, which occurs at an approximate rate of 3.2 cases per million per year globally [4]. Endogenous Cushing’s is divided into two types: adrenocorticotropic hormone (ACTH) dependent and ACTH independent [2]. ACTH dependent Cushing’s comprise approximately 80% of endogenous Cushing’s cases and is caused by ACTH secreting tumours most often on the pituitary (i.e., Cushing’s disease), or an ectopic ACTH secreting tumour found elsewhere in the body [2]. ACTH Independent Cushing’s cases account for the remaining 20% and are most often caused by adrenal cortical adenomas or hyperplasia [5].

The rare rate of occurrence, coupled with the broad symptomology contributes to the challenges with diagnosing Cushing’s, with a delay to diagnosis of two to six years [6, 7]. Although individuals may experience few symptoms its early stages, disease progression that occurs with delayed diagnosis can lead to significant impairment in daily life as well as hypertension, metabolic diseases, and other complications [8]. Given that the incidence of Cushing’s is higher among women compared to men, [9]. as well as the gendered nature of weight and appearance expectations for women in society (i.e., thinness), [10] understanding the impact of this disease on women is an important, but unexplored, direction for women’s health research.

The non-specific presentation and rare occurrence of endogenous Cushing’s is also important to consider in the context of patient centered care, defined as relationship-based care that meets the needs, priorities, and values of patients [11, 12]. Patient centered care focuses on the creation of a positive therapeutic alliance between patient and provider, with shared power and responsibility in decision making [11, 12]. A recent systematic literature review identified that most barriers to patient centered care occur at the provider level, including lack of training, physician burnout, and poor-quality communication [12]. One potential barrier to patient centered care for individuals with Cushing’s is weight stigma (i.e., stereotypes and negative attitudes about people with higher weights), which is prevalent in healthcare, [13, 14] and is associated with consequences for healthcare utilization [15, 16].

Although researchers have documented the clinical presentation of Cushing’s, [7] approaches to working with patients with a possible diagnosis, [1] as well as treatment approaches and outcomes, [17, 18] there is an absence of research on the lived experience of patients with Cushing’s in healthcare. Given the established delay to diagnosis of Cushing’s [6, 7] and the possible impact of weight stigma on patient centered care, we sought to conduct a preliminary inquiry into the patient experience in primary care. Thus, the aim of this study was to examine the healthcare experiences of women with Cushing’s in Canada, as women experience greater incidence of Cushing’s [9] as well as greater appearance-based sociocultural pressures [10].

Methods

Research team background and epistemological underpinnings

We recognize that our identities and positionalities as researchers have a significant impact on the research we conduct [19]. All authors identify as White cisgender women with lived experiences with weight-related issues, and SCJ has lived experience of Cushing’s Disease. SCJ led this research project as part of her degree requirements in a double-major biology and psychology undergraduate program. SN is a weight stigma researcher with a background in qualitative research. JFS conducts research on body image and eating disorder recovery, with a background in qualitative methods. Further, this research was undertaken from a social constructionist epistemological position, recognizing the power of sociocultural discourses related to appearance, weight, and health on the experiences of higher-weight people in society broadly as well as healthcare specifically [20,21,22]. These sociocultural discourses position weight as an accurate indicator of health that is within individual control, with lower body weights considered healthiest (i.e., “normal” weight body mass index) [20,21,22].

Participants

Participants were seven women from across Canada (see Table 1 for demographics). The mean age of the sample was 44.14 (SD = 13.32) and mean self-reported time to Cushing’s diagnosis after first seeking medical care was 2.14 years (SD = 0.58). Six participants experienced Adrenocorticotropic hormone (ACTH) dependent Cushing’s, while one patient experienced ACTH independent adrenal adenoma. Of the six participants with ACTH dependent Cushing’s, five had an ACTH secreting pituitary adenoma and one participant had an ectopic ACTH producing lung carcinoma. All participants self-declared that they exhibited clinically significant 24-hour urine free cortisol levels, which contributed to their diagnosis.

Table 1 Participant demographics and pseudonyms

Procedure

This study received research ethics approval from the first and second authors’ institution (#21–0507). Convenience sampling occurred in February 2022 via a shareable recruitment post on five Facebook support groups for Cushing’s Syndrome. Potential participants were directed to reach out to the first author to indicate their interest in the study. Participation was limited to cisgender women over the age of 18 who had been diagnosed with endogenous Cushing’s and received care in the Canadian healthcare system. Nine individuals from one Facebook support group contacted the researcher, two of whom did not receive treatment in Canada. The remaining seven participants were provided with a consent form and demographics survey, which they completed prior to scheduling a semi-structured interview.

Following the completion of informed consent, interviews took place over zoom, in a private location of the participant’s choosing. To obtain a rich understanding of experiences with Cushing’s, we asked a series of questions about participants’: (1) journey to diagnosis, (2) healthcare experience pre- and post-diagnosis, and (3) perception of body and weight pre- and post-diagnosis. Interview questions were open-ended and provided opportunity for participants to describe positive and/or negative experiences. Please see the Appendix for full interview protocol. Interviews lasted approximately one hour with $30 compensation, were recorded, and were transcribed verbatim by the first author. Following transcription, participants had the opportunity to review their transcript and remove any data they were uncomfortable including in the analysis (i.e., member checking). One participant removed a small portion of her transcript, which did not impact the analysis.

Analysis

Following transcription, anonymization, and member checking, the first and second author used a qualitative descriptive approach [23] in the preliminary data analysis stages to become familiar with the data and the experiences of the seven women interviewed. Two main content areas predominated: (1) negative experiences in healthcare and (2) women’s body image as connected to Cushing’s-related changes. Given these divergent content areas, the research team engaged in a two-pronged approach to qualitative data analysis, one for the negative healthcare experiences (reported here) and one for women’s body image.

The data pertaining to negative experiences in healthcare were analysed using reflexive thematic analysis [24, 25]. This method allowed for a rich and flexible understanding of similarities in individual experiences. To generate themes, the first and second authors read the transcripts line-by-line and assigned descriptive codes, which were then reviewed and re-coded at a more interpretive level to create themes in the data, both latent (i.e., underlying) and semantic (i.e., explicitly stated) [24, 25]. Themes were reviewed and approved by all authors. Throughout data collection and analysis, effort was made to discuss reactions to the data and potential biases.

Results

The results of the inductive thematic analysis indicated four overarching themes in the data: (1) lack of patient centered care prior to diagnosis; (2) patient misunderstanding of symptoms; (3) experienced weight stigma; and (4) diagnosis as a golden ticket to treatment and stigma-reduced care. Each of these themes is reviewed below.

Lack of patient-centered care

Lack of patient-centered care prior to diagnosis often took two forms: (1) provider miscommunication, and (2) medical gaslighting. These sub-themes are described below.

Provider miscommunication. In the beginning stages of their investigations, participants reported difficulties in communication with their primary care providers. Honey stated that she was “going back and forth continually, feeling like I wasn’t being heard, feeling like I had to fight for every… For every feeling. I had to try to justify everything I was saying.” Similarly, Abby noted: “I’d started to wake up in the morning with little bruises on my body like almost like a fingerprint. And I thought that was weird and I brought it up to her … And she was like oh that, there’s nothing wrong with that, it’s fine.” Participants described confusion and worry when they perceived that something was wrong but did not perceive they were being taken seriously by their primary care provider.

Sometimes, miscommunication made the diagnostic process even more challenging, as was the case when Bethany was asked about stretch marks by her family doctor:

I remember my family doctor asking me at one point, do you have stretch marks? And I said, ‘no, I don’t have stretch marks. Last time I had them was when I was pregnant. And that was many years ago.’ But he didn’t describe the stretch marks. I was thinking of the pregnancy stretch marks with the fine white lines. And he must have thought about Cushing’s with the striae that are wide and purple. But there was no description of them. If I had [a description], I would have shown them immediately!

Christy noted how phone appointments may have negatively impacted her diagnostic process:

All of this went down in the [COVID-19] pandemic, so I actually never met a doctor in person about this ever. … I explain the weight gain I’m like kay I was 110. Now I’m 230 and these are the bursts. These are when it happened. Um, but they actually never saw me, not once, not even over zoom it was all over the phone … When I explained how much I gained they were just like, no.

In general, participants felt like they were not receiving the care they hoped for from their primary care provider. For example, Abby said that “Cushing’s patients have a lot of things going on and most regular doctors when they see you…they think you’re a hypochondriac because there’s always something wrong with [you].” Participants perceived their primary care providers as missing the big picture. Darby recognized that “I think each time I went into the doctor for the different things, … I would go in for specific things, but we never sort of put them all together as to what it was.” In describing her frustration with this process, Emily said: “And I’m just like how many other people are struggling with stuff like this, and they just get [dismissed].” Together, this miscommunication contributed, in part, to the delayed diagnosis, as it took time for providers to recognize the possible diagnosis of Cushing’s.

Medical gaslighting. Medical gaslighting is the process by which medical professionals will downplay, dismiss, or silence a patients’ view of their illness [26]. Abby described her experience with the first primary care provider she consulted: “he basically told me that I had mental baggage, and it was that I was a head case and that was my problem.” After moving to a different city, she described the next provider she met with as having a similarly dismissing response: “I also explained to her what was going on. I said I think there’s something wrong, I exercise and the harder the exercise more I gain weight. She basically said you’re not trying hard enough and suggested the South Beach diet to me.” Besides their weight, other symptoms were also dismissed. Emily said:

It was always passed off as oh, your high-stress job, high-stress family life, you’re depressed; those were always the comments to me. You’re just depressed, you’re just anxious because of this stuff and blah blah, blah. … As time went on the symptoms started multiplying. So, I’m like it’s not just not sleeping and depressed and anxious I’m like… my resting heart rate is in the 160’s without moving um, my vision’s starting to get impaired, I feel like I’m crazy, um and then the weight gain started. And once again they just said oh you know take up, take up cycling. That’s what the doctor told me.

In describing her experiences with feeling downplayed or dismissed, Honey stated:

I had swollen hands and feet, too, so he said, Well, you’re gonna have to get your diet under control, and I said well my diet is really good actually and I’ve been going to weight watchers for a year, haven’t dropped a pound, so there’s an issue there. And again, he would just chalk it up to PMS.

Honey also experienced skin sensitivities and was told she simply needed to use sunscreen more frequently. During the diagnostic process, Christy experienced medical gaslighting when completing her 24-hour urine free cortisol test:

I did the test and waited months again for my results. And then it came back super high. And so, he accused me of doing the test wrong. He said there’s no way um this makes sense. You need to do the test again. So, then I did the test again. And it was high again. And he accused me of doing it wrong again. And I was like, I’m telling you like I don’t know what you, I’ve got the instructions in front of me. … And so I did it again, and it came back like again in the thousands. … I did feel like I was going nuts, the same way when he kept telling me I was doing the test wrong.

Patient misunderstanding of symptoms

Participants also described misunderstanding their symptoms, which highlights a further breakdown in patient-provider communication about their disease, as these misperceptions were not clarified for them upon diagnosis and treatment. Participants appeared to hyper-fixate on, and misunderstand, their weight gain. They also misunderstood other symptoms as related to weight. Emily noted this fixation on weight directly when she said: “In the very beginning I noticed the weight gain first to be honest.” Abby shared that she decided to approach a health professional because of her weight gain:

I was a happy, healthy active athletic person, and all of a sudden something in my life happened where I just kept gaining weight and gaining weight. No matter how much like exercising or eating properly that I had done… The catalyst for me going to the doctor was, um, I noticed a bunch of stretch marks on my stomach and to me that was a sign of like rapid weight gain in such a small amount of time.

Like Abby, other participants misunderstood their weight gain and attempted to engage in behavioural changes to reverse this process. Bethany said:

Thankfully I did not have a doctor that told me to go and exercise and eat less. But when you did that, you still gained weight. The more I exercised, the more walking I did, the less I ate, the more I gained and the rounder the face got. And the more hair I lost.

Similarly, Christy described her weight gain as the catalyst behind her decision to hire a personal trainer:

I kind of decided based on super significant weight gain that I was going to invest in a personal trainer… So, I was with the personal trainer for about three months, and was only gaining weight, like I was cutting calories and working out I think it was like 12 times a week. … But everything was just getting worse. Like I was weaker, I couldn’t do things the same way that I used to, like the stretch marks were nuts I kept gaining weight I didn’t know why, despite still I was still walking every day and doing what I could.

When describing their experience of symptom onset prior to receiving their diagnosis, participants understood many of their physical changes as connected to their weight gain. This was especially true for those experiences that surrounded muscle weakening and movement, despite these being Cushing’s symptoms that are independent from weight. Bethany recalls how, when called for her appointment in her primary care provider’s waiting room, she “staggered to stand up and was almost immobile to a point, I was so big,” an experience she attributed to her weight gain. As symptoms worsened, participants felt disconnected from their bodies. Emily said: “I can’t trust [my] own body,” and Christy felt “so exhausted” because “of course that’s exhausting. Like, carrying all that [weight] around is tiring. Like, if you’re not exhausted, that would be weird.”

Participants also noted a misunderstanding of other Cushing’s symptoms, both alongside and independent of their weight gain. In recognizing the impact of stress on her symptoms, Honey said:

I opened a business, and the stress came back because of that, and then everything came to a head. Then I got the bruising, then I got the buffalo shoulders, then I got the big moon face. … People were starting to say, like is your whole life okay? It was kind of insulting. And then I got the big belly, and it just got bigger and bigger, and within 3 weeks I had gained 25lbs.

Darby noted that, in the fullness of her busy life, it was easy to explain away her symptoms:

I didn’t acknowledge what was going on it, I just didn’t push to find out why. I was always able to find a reason in my own brain or my own thinking as to all, you know. You know, I’m just I’m working too much and this will pass, and then it’ll get better. Or, you know, when I was falling down for no reason, it was like oh man you just got to pay more attention. All right, you know, I need to eat better because I’m not, you know, exercising. When I was gaining the weight and went to weight watchers and some reason the weight wasn’t coming off. … And I’m thinking, I’m doing all the right stuff.

Similarly, Franny described how she perceived her symptoms to be related to anxiety:

Oh, it’s just anxiety you’re always… It’s like the palpitations Oh, it’s just you’re nervous about something. … I guess I just… in my case, like I haven’t… maybe they caught it so soon enough that, like I haven’t really got to the point where like I’m I’m feeling a lot of symptoms?

Experienced weight stigma

Participants described numerous experiences of weight stigma in healthcare while seeking a diagnosis. Invalidation occurred when patients’ personal accounts of their illness were dismissed or not taken seriously, as described in the Lack of Patient Centered Care sub-themes. While many participant experiences of medical gaslighting can also be regarded as experiences of weight stigma, these themes were differentiated by whether or not participants reported they were dismissed or silenced, consistent with the definition of gaslighting [21].

Participants contributed their negative experiences, at least in part, to their weight. Participant’s felt like there was a significant difference in how they were treated by practitioners before the onset of their Cushing’s symptoms. Christy contrasted her experiences in healthcare pre- and post- Cushing’s: “When I went to the doctor and I was concerned about something as a kid, nobody ever doubted me like. And yeah, since then … people in general just don’t get [taken] seriously looking the way that I do. And don’t respect me as much it seems.” Although Christy spoke of “people in general,” this sentiment included her primary healthcare provider. Similarly, Honey described a perceived change in her relationship with her primary care provider pre- and post- Cushing’s onset and felt that her weight contributed to this change. She described her once positive relationship as one that is now associated with hurt:

Prior to Cushing’s my doctor was very forthcoming. I would tell him what I needed if I needed, felt I needed blood work, or felt I needed anything, or suggested that I…. maybe he’s not the one to help me that I need to see a specialist he would send me on to that specialist. So, yes, I always felt hurt by him.

Abby summarized participants’ experiences when she said: “Judging books by covers that’s, that’s what the healthcare system seems to do and I do feel that, like some of my treatment was definitely due to my body [size] and what, what they preconceived that I was a fat person, so it was my fault.” Franny described having requested her entire medical file and that seeing herself repeatedly referred to as a “morbidly obese female” in emails and other communication had a negative impact on her body image and self-esteem.

Diagnosis as a golden ticket

Post-diagnosis, participants experienced a profound sense of relief and validation, perceived themselves to be lucky to have received a diagnosis, and were thankful for the subsequent treatment they received. Bethany said: “once you get a diagnosis, it doesn’t matter what the diagnosis is, you’ve got a diagnosis and now we work towards that. Let’s correct it”. Christy felt like “Having that diagnosis under my belt was like a golden ticket … everything was easier.” Abby declared that the doctor who officially diagnosed her saved her life: “She had taken the time to like, say I believe you. Instead of just seeing a fat person and blaming it on me, which is really what the healthcare system does. Really it ruins people.” After waiting three years for a diagnosis, Darby stated that she felt “very lucky” and said:

From when my doctor said to me ‘I think it’s Cushing’s’ to when the actual diagnosis was, it was fast. It was weeks. It was, it was very, very quick. …Once we actually knew what it was to actually being able to have the surgery to have the tumor removed and start reversing the process of what was going on with my body. So. I was very lucky.

In hindsight, participants noticed there was a difference in how they were treated by healthcare professionals pre- and post-diagnosis. Abby said: “I firmly believe that there is a correlation there between how I was treated before, and then how I was treated after.” She elaborated to say that: “people have to believe me now, because a specialist has said, ‘This is what I have, and you may not understand it, but I do’.” Emily noted that “after I got the diagnosis my family doctor was very, very open, and caring with moving forward. And he commended me for pushing and knowing that things were wrong, and he was really good about it, and he apologised that it took long to get figured out.” Honey also noted” “I’d have to say like during the diagnosis, not great, but since I’ve had my pituitary tumor removed great, the aftercare has been fantastic.”

Discussion

Across seven semi-structured interviews with women with endogenous Cushing’s, the results of our preliminary inquiry into the lived experience of patients suggest that both patients and providers struggle to understand the progressive and individualized presentation of Cushing’s. This struggle can come, unintentionally, at the expense of patient centered care in the form of provider miscommunication, medical gaslighting, and weight stigma. Our results also suggest that diagnosis is viewed by patients as an important turning point for both effective treatment as well as reduced stigma.

Overall, the results of our study highlight the importance of patient centered care. Participants reported miscommunication, invalidation, weight stigma, and gaslighting in their healthcare encounters, all of which are contrary to the goals of patient centered communication [11, 12]. These findings are consistent with previous research identifying structural, educational, and resourcing barriers to patient centered communication [11, 27]. These barriers include understaffing, specific healthcare settings (i.e., acute care), limited time, insufficient communication, lack of training in patient centered communication, and inadequate patient education [11, 27]. Given that patient engagement is associated with improved outcomes, a better understanding of their condition, and more awareness of resources, [27] identifying strategies to enhance patient centered communication are of critical importance. Researchers have identified staffing longevity as associated with greater engagement and less burnout [27] and have identified provider training and professional development as significant in the implementation of patient centered communication [12]. Thus, in addition to patient centered communication skills, providers require knowledge of weight stigma and Cushing’s as well as appropriately resourced work environments to enact patient-centered care. However, an important responsibility falls to broader healthcare systems to empower providers by providing comprehensive training, addressing provider burnout, and changing healthcare culture and systems to allow for increased engagement in patient centered communication [12].

The rare occurrence [6, 7] and non-specific presentation [8] of endogenous Cushing’s is a unique barrier to patient centered communication. Participants in this study recognized the impact that the rare occurrence of their disease had on their provider’s misunderstanding of their symptoms. Bethany noted one interaction whereby her primary care provider mentioned being told in medical school that they would never see a case of Cushing’s in their careers. However, knowledge of Cushing’s is a pre-requisite for early detection [6]. Such knowledge is important, as overlooking endogenous Cushing’s is associated with increased mortality rates due to hypertension, metabolic diseases, and bone-related complications, with continued cardiovascular risk persisting after treatment [8].

An additional barrier to patient centered care highlighted in our results is weight stigma. Our findings are consistent with previous qualitative research on experienced weight stigma in healthcare contexts [28,29,30,31]. In previous research, patients have consistently reported the attribution of presenting concerns by healthcare providers to weight without a full exploration of the issue as well as poor verbal and non-verbal communication [28,29,30,31]. Such experiences with poor quality communication and stigmatization from healthcare providers are associated with subsequent patient healthcare delay and avoidance, [15, 17] and may contribute to increased internalization of weight stigma [32]. Our results highlight that, for patients with endogenous Cushing’s, weight stigma may be a contributing factor in the consistently reported delays to diagnosis, [6, 7] due to stereotyped assumptions about weight gain coupled with a lack of knowledge about Cushing’s, among providers as well as patients. Although participants in this study reported symptoms beyond weight gain, they perceived their weight as having an impact on their healthcare encounters.

Our findings related to weight stigma highlight the potential harm of weight- and appearance-focused sociocultural discourses [21, 22] on Cushing’s-related healthcare. For patients, internalization of these discourses may have contributed to the extent to which they understood symptoms such as fatigue and muscle weakening as caused by their weight gain. For providers, this may have contributed to their clinical impressions and decision-making. Researchers have previously identified weight and appearance discourses as contributing to a weight-centric practice paradigm, whereby higher weights are regarded as within individual control as well as the cause of poor health [33]. Within a weight-centric paradigm, recommending weight loss is regarded as an effective health-promoting solution and failure to achieve or sustain weight loss is regarded as the fault of the patient. Criticisms of the unintended consequences of a weight-centric paradigm have prompted researchers to call for a weight-neutral approach to healthcare that recognizes and addresses weight stigma, assesses cardiometabolic and lifestyle health risks in patients across the weight spectrum, and seeks to promote the health of patients independent of changes in weight status [33].

This research was conducted as a preliminary inquiry into the lived experience of patients with Cushing’s, given the lack of such research in the literature. Our findings suggest that patient experiences are an important, but unexplored, line of inquiry and that more research is needed in this area. Given that lived experiences may differ across contexts and may vary by an individual’s culture, race, relationship status, and/or geographical location, further research is needed with larger samples to elucidate the potential impact of provider miscommunication, medical gaslighting, and weight stigma on the experiences of patients with Cushing’s throughout the diagnostic and treatment process. Future research examining Cushing’s-related healthcare experiences access gender and racial identities is also needed to fully understand the broad experiences of patients with this disease.

Limitations

To our knowledge, this is the second study to examine the lived experiences of people with Cushing’s, with only one other qualitative study examining the impact of Cushing’s on quality of life, published in Italian [34]. However, this is the first study to examine patients’ lived experiences in healthcare, as this previously published Italian study examined patients’ perceived quality of life. Despite this novelty and strength, this research is not without its limitations. First, as only seven women with Cushing’s were recruited, this preliminary inquiry is limited by sample size. Thus, the results are not generalizable to all women with Cushing’s, their healthcare providers, or their experiences in healthcare. Second, given that our participants were recruited from an online support group, their reported experiences may be different from those who have not sought such support from the online patient community. Third, our sample was limited in demographics. All participants identified as white women but did represent a broad range in age and socioeconomic status. Finally, as our participants reported a delay to diagnosis of 1.5 to 3 years, their experiences may not represent those of individuals who experienced a longer delay to diagnosis.

Conclusion

The findings of this research indicate that a lack of patient centered care may have an impact on the recognition and referral/diagnosis of Cushing’s in primary healthcare settings. Our results highlight an important area of inquiry that warrants further attention. Given the small sample size, further research is needed to clarify and to expand on these findings. However, these results highlight the importance of patient centered care and curious investigation into the causes of unexpected and uncontrollable weight gain in patient encounters.

Data availability

Data for this study is available on reasonable request to the corresponding author.

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Acknowledgements

Not applicable.

Funding

SCJ was supported by a Jamie Cassels Undergraduate Research Award.

Author information

Authors and Affiliations

  1. Department of Psychology, University of Victoria, 3800 Finnerty Rd, Victoria, BC, V8P 5C2, Canada

    Sarah C Jones

  2. Educational Psychology and Leadership Studies, University of Victoria, 3800 Finnerty Rd, Victoria, BC, V8P 5C2, Canada

    Sarah Nutter

  3. Institute on Aging and Lifelong Health, University of Victoria, 3800 Finnerty Rd, Victoria, BC, V8P 5C2, Canada

    Sarah Nutter

  4. Brigham and Women’s Hospital, 75 Francis St., Boston, MA, 02115, USA

    Jessica F Saunders

Contributions

SCJ and SN conceptualized the study. All authors contributed to the study design and SCJ conducted all interviews and transcriptions. Data analysis was conducted by SCJ and SN with support from JFS. All authors contributed to manuscript preparation.

Corresponding author

Correspondence to Sarah Nutter.

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Ethics approval and consent to participate

This study received ethics approval from the Human Research Ethics Board at the University of Victoria (#21–0507). Informed consent was received from all participants.

Consent for publication

Not applicable.

Competing interests

The authors declare no competing interests.

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