Luteoma of pregnancy associated with nearly complete virilization of genetically female twins.

OBJECTIVE: To describe a pregnancy that was complicated by the virilization of the mother and two 46XX infants. METHODS: We outline the clinical presentation and diagnosis of the virilization of a mother and her twins, reviewing pertinent literature. RESULTS: We report the case of a 40-year-old Caucasian female who conceived a trichorionic triplet pregnancy through in vitro fertilization (IVF) but underwent cytoreduction at 13 weeks of gestation, leaving a diamniotic dichorionic twin pregnancy. At 16 weeks of gestation the mother experienced increasing acne, facial hair, and deepening of her voice. Due to preeclampsia, the twins were delivered via caesarean section at 33 weeks of gestation. The infants had male-appearing external genitalia (Prader score IV-V) but no palpable gonads. Congenital adrenal hyperplasia was ruled out for both twins and they were both found to have a uterus and a 46XX karyotype. Maternal testosterone level was elevated at birth (1,981 ng/dL), but the infants had normal levels. Maternal testosterone levels returned to normal after delivery, consistent with a luteoma of pregnancy, although imaging was negative for a mass. CONCLUSION: This is the second reported case of complete virilization associated with a luteoma of pregnancy. Whether or not IVF and related procedures increase the risk for a luteoma and whether or not fetal reduction procedures disrupt placental aromatases and increase the risk of virilization in the face of elevated androgen levels are questions that require further research.

Postpartum granulomatous hypophysitis with sphenoid sinus involvement: a case study.

OBJECTIVE: To report an unusual case of granulomatous hypophysitis with sphenoid sinus involvement in a woman presenting with headaches and visual field deficits approximately 2 weeks after a normal delivery. METHODS: We present the history, physical findings, hormonal assay results, pituitary imaging, surgical findings, and pathology findings at presentation and then follow-up data at several times encompassing 1 year of observation. We also performed a literature review on granulomatous hypophysitis. RESULTS: A 29-year-old woman presented with headache and visual disturbances 11 days after childbirth. Magnetic resonance imaging revealed a sellar mass with suprasellar extension, compression of the optic chiasm, possible invasion of the cavernous sinuses, and sinus mucosal thickening. A subtotal resection was performed through the transsphenoidal route. Histologic examination demonstrated extensive nonvasculitic granulomatous tissue in pituitary and sphenoid mucosal samples. Investigation for known causes of granulomatous hypophysitis was negative. She required desmopressin and levothyroxine replacement postoperatively. Sequential follow-up revealed spontaneous resolution of the residual mass in 5 months. CONCLUSION: Unique features of this case include the concomitant presence of granulomatous lesions in the pituitary gland and the sphenoid sinus, its manifestation in the early postpartum period, and the spontaneous resolution of the residual granulomatous lesions in both the sphenoid sinus and the sella turcica.

A novel noninvasive blood glucose monitor.

OBJECTIVE: To evaluate the precision and accuracy of a new advanced prototype of a noninvasive blood glucose monitor across a wide range of serum glucose concentrations. RESEARCH DESIGN AND METHODS: An advanced handheld noninvasive glucose monitor prototype was calibrated and tested using patients recruited by the General Research Center of the University of Connecticut Health Center. The monitor, developed by Infratec, uses principles of thermal emission spectroscopy. The noninvasive measurement of tympanic membrane glucose concentration was calibrated to the serum glucose concentration using 432 paired measurements from 20 subjects with insulin-requiring diabetes. This calibration was subsequently tested (results of power analyses) in a blind fashion with 126 paired measurements from six diabetic subjects who require insulin. RESULTS: In vivo measurements demonstrated the reproducibility of the methodology of the noninvasive glucose monitor. Based on the calibration model, predicted glucose concentrations for six subjects were as follows (for 126 data points): SD = 32 mg/dl, mean absolute relative error (%MARE) = 11.6, with a correlation coefficient of r = 0.87. Noninvasive glucose results were also compared with laboratory reference measurements using an error-in-variables method. Clark error grid analysis showed that 100% of the measurements fell within zones A and B (90% in zone A and 10% in zone B). The SD for all noninvasive measured concentrations was 27 mg/dl, %MARE was 8.6, and the correlation coefficient was r = 0.94. CONCLUSIONS: This first independent clinical study of an advanced noninvasive blood glucose prototype based on thermal emission in the mid-infrared spectral region has demonstrated glucose measurements with clinically acceptable accuracy but without the necessity of individual daily calibration.