RESUMEN
OBJECTIVE: Granulosa cell ovarian tumors are known to secrete estrogen. Herein we report a patient presenting with primary amenorrhea and virilization with markedly high androgen levels, all thought to be disproportional to be attributed to polycystic ovary syndrome (PCOS) alone. Bilateral oophorectomy revealed a rare androgen-secreting granulosa cell ovarian tumor and bilateral cysts (PCOS) both contributing to manifestations. METHODS: Description of a case and discussion of the literature. RESULTS: A 25-year-old female presented with primary amenorrhea, male pattern baldness and hirsutism of the face and chest, clitoral hypertrophy, cystic acne on the chest and shoulders, and type 2 diabetes. Diagnosis of PCOS was made at age 13 years. The concurrent presence of congenital adrenal hyperplasia was also considered. She was receiving metformin, oral contraceptives, and/or spironolactone with no improvement in manifestations at presentation to the endocrine clinic. She reported several elevated serum testosterone and androstenedione levels. Diagnosis of PCOS was established by the presence of enlarged cystic ovaries on ultrasound examination. The patient reported worsening manifestations including progression of diabetes despite therapy with metformin. She shaved her face, chest, and breasts daily for cosmetic reasons. Laboratory testing demonstrated markedly elevated total testosterone level (234 ng/dL), and normal cortisol and adrenocorticotropic hormone levels. Dexamethasone suppression and human chorionic gonadotropin stimulation indicated ovaries as a probable source of excessive circulating androgen levels. A computed tomography scan of the abdomen and pelvis showed multiple cysts in both ovaries, the largest being 1.9 cm in the right ovary. Due to the severity of manifestations, the patient was counseled to undergo fluoroscopically guided blood sampling of bilateral adrenal and ovarian veins, which confirmed both ovaries to be the source of the excess androgen production with a greater contribution by the right ovary. The patient underwent bilateral oophorectomy. Pathologic examination confirmed the presence of bilateral polycystic ovaries as well as an androgen-secreting granulosa cell tumor in the right ovary which apparently contributed to greater androgen levels in a right ovarian venous blood sample. Peripheral venous androgen levels normalized promptly after bilateral oophorectomy. Gradual resolution of clinical manifestations followed. CONCLUSION: A unique presentation of granulosa cell ovarian tumor with concurrent PCOS contributing to the extremely excessive production of androgens in a young woman manifesting primary amenorrhea and masculinization at the onset of puberty with marked gradual resolution of manifestations following bilateral oophorectomy.
RESUMEN
INTRODUCTION: Annually, >50% of the US population reports musculoskeletal (MSK) pain to a provider, with direct healthcare costs exceeding $185 billion. The number of MSK complaints and the associated costs are projected to rise, increasing demand for and burden on providers. Establishing new care models to decrease inefficiencies may lower costs and optimise care delivery. The purpose of the Integration of Musculoskeletal Physical Therapy Care in the Patient-Centred Medical Home (IMPaC) study is to compare initial evaluation by a physical therapist (PT) integrated into primary care versus initial evaluation by a primary care provider (PCP) for patients with an MSK complaint. METHODS AND ANALYSIS: This single-site, randomised clinical trial will test the hypothesis that a PT within a primary care facility as the initial evaluating provider for patients with an MSK complaint will lower costs, improve utilisation (ie, reduced opioid prescriptions, imaging, physical therapy, emergency department visits and missed appointments) and increase patient satisfaction within 90 days of the index visit compared with PCP evaluation in the same location. Participants aged ≥18 years will be randomised with equal allocation and stratified by pain site (ie, back, knee, upper extremity and other). In the initial PT evaluation arm, patients will be assessed, treated and then instructed to complete a home exercise programme. The PCP cohort will undergo a usual PCP evaluation, and if a referral to physical therapy is made, patients will be randomised to onsite versus offsite physical therapy. Differences will be calculated and tested across the two arms. ETHICS AND DISSEMINATION: Approval was received from the Duke University Institutional Review Board (01 May 2017) and the National Institutes of Health, National Centre for Advancing Translational Sciences (01 January 2017). Findings will be communicated via quarterly reports to funding bodies and disseminated through scientific publications. TRIAL REGISTRATION NUMBER: NCT03110211; Pre-results.
