This case report shares the management experience of a patient with pregnancy combined with adrenal adenoma causing ACTH-independent Cushing's syndrome (CS), accompanied by obstetric antiphospholipid syndrome (OAPS) and severe pre-eclampsia. The case was a 26-year-old that presented with typical clinical symptoms and signs of CS. The patient had a history of 4 spontaneous abortions in the last 4 years. The 24-hour urinary free cortisol was significantly increased, an abnormal cortisol circadian rhythm was demonstrated by a high late-night salivary cortisol, blood ACTH was suppressed (< 1ng/dL), anticardiolipin antibody was positive, and imaging examination showed an adrenal tumor. The patient underwent laparoscopic adrenal tumor resection under general anesthesia at 23 weeks of gestation. The tumor was pathologically confirmed to be an adrenocortical adenoma. The patient underwent a cesarean section at 39 weeks of gestation to give birth to a healthy baby girl with an Apgar score of 10. Pregnancy complicated by CS is clinically rare, easily masked by normal physiological changes of pregnancy, and is difficult to diagnose. The determination of 24-hour urinary free cortisol, the circadian rhythm of serum cortisol, ultrasound, and MRI can be helpful in the diagnosis of CS during pregnancy. Surgery is the first choice for the treatment of CS during pregnancy. As a subtype of antiphospholipid syndrome, patients with OAPS are prone to thrombotic events and recurrent miscarriages if not treated accordingly. To our knowledge no cases of CS with OAPS and severe pre-eclampsia have been reported. We summarize the experience of the treatment of this patient and review the literature to improve clinicians' awareness of this disease.
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http://dx.doi.org/10.3389/fendo.2023.1147316 | DOI Listing |
Oman Med J
July 2024
Family Medicine General Foundation Program, Oman Medical Specialty Board, Muscat, Oman.
We report a rare case of a middle-aged Omani woman who was known to have primary antiphospholipid syndrome, glucose-6-phosphate dehydrogenase deficiency, and iron deficiency anaemia. Cannulation attempts caused bulla which progressed to ulceration. A pathergy phenomenon with high suspicion of pyoderma gangrenosum was postulated.
View Article and Find Full Text PDFTher Adv Musculoskelet Dis
December 2024
Grupo de Patología Musculoesquelética, Hospital Clínico San Carlos, Instituto de Investigación Sanitaria San Carlos, Madrid, Spain.
Background: Rheumatology has experienced notable changes in the last decades. New drugs, including biologic agents and Janus kinase (JAK) inhibitors, have blossomed. Concepts such as window of opportunity, arthralgia suspicious for progression, or difficult-to-treat rheumatoid arthritis (RA) have appeared; and new management approaches and strategies such as treat-to-target have become popular.
View Article and Find Full Text PDFSci Rep
December 2024
Laboratory of Molecular and Cellular Immunology, Institute of Molecular Biology NAS RA, 7 Hasratyan Str., Yerevan, 0014, Armenia.
Antiphospholipid syndrome (APS) is associated with recurrent pregnancy morbidity, yet the underlying mechanisms remain elusive. We performed multifaceted characterization of the biological and transcriptomic signatures of mouse placenta and uterine natural killer (uNK) cells in APS. Histological analysis of APS placentas unveiled placental abnormalities, including disturbed angiogenesis, occasional necrotic areas, fibrin deposition, and nucleated red blood cell enrichment.
View Article and Find Full Text PDFRev Med Interne
December 2024
Service de médecine interne et inflammation, département inflammation-immunopathologie-biothérapie (DMU I3), CEREMAIAA, hôpital Saint-Antoine, AP-HP, Sorbonne université, Paris, France.
Hydroxychloroquine (HCQ), a synthetic antimalarial, is recognized for its immunomodulatory, anti-inflammatory and vascular-protective effects. In 20-30% of cases of primary obstetrical antiphospholipid syndrome (APS), the combination of antiplatelet aggregation and prophylactic anticoagulation fails to prevent obstetrical complications, a situation referred to as refractory obstetrical APS. This is partly due to the pro-inflammatory effects of antiphospholipid antibodies (aPL) binding to decidual and trophoblastic cells, which compromise embryonic implantation and placentation.
View Article and Find Full Text PDFThis article presents a clinical case of ocular thrombotic microangiopathy of mixed origin (antiphospholipid syndrome, malignant arterial hypertension, multigenic thrombophilia). Multimodal imaging of the fundus provides a detailed assessment of its structures. Pathological changes in the choroid, the "retinal pigment epithelium - Bruch's membrane" complex, and the neurosensory retina, identified using fundus photography, short-wavelength autofluorescence, optical coherence tomography (OCT), and OCT angiography, are described as nonspecific in nature.
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