When Thirst Ceases to Exist: A Case Report and Literature Review of Adipsic Diabetes Insipidus Following Coil Embolization of a Ruptured Anterior Communicating Artery Aneurysm.

Diabetes insipidus is a condition characterized by inappropriately dilute urine in the setting of serum hyperosmolality. The two predominant subtypes include central (from lack of vasopressin production) and nephrogenic diabetes insipidus (from renal resistance to circulating vasopressin). A common manifestation is the significant pursuant thirst from excessive polyuria. We present a case report and literature review of an infrequent variation of central diabetes insipidus known as adipsic (hypothalamic) diabetes insipidus, characterized by the absence of thirst, secondary to coiling of a ruptured anterior communicating artery aneurysm. Due to the loss of thirst, patients are at a heightened risk for hypernatremia and complications secondary to dehydration. Our patient's course was complicated by recurrent polyuria and hypernatremia, requiring a fixed-dose desmopressin regimen. On follow-up, only partial thirst sensation was restored. We provide a literature review to compare our case report to the scant literature available to broaden the awareness of this infrequent, perilous, manifestation.

A Scoping Review of the Apparent Phenomenon of the Improvement in Hypoparathyroidism in Pregnant and Postpartum Females.

Hypoparathyroidism requires management with both calcium supplementation and active vitamin D to avert a state of hypocalcemia. During late gestation and the postpartum period (specifically lactation), there is an under-recognized, yet intriguing occurrence of apparent 'pseudohyperparathyroidism', whereby supplementation dosages may need to either be reduced or discontinued, to prevent hypercalcemia. The explanation for this apparent phenomenon of improved parathyroid status ('remission' or 'resolution') is incompletely understood; the purpose of this review is to analyze the case reports of this enigma within the medical (and grey) literature, providing an overall pathophysiological explanation and recommendation for the management of such patients. A literature search was conducted through PubMed/Medline, CINAHL, Cochrane Library Database, Scopus, UpToDate, Google Scholar, and the grey literature without a time-restricted period, analyzing all available articles within the literature describing an apparent improvement in parathyroid status in late-gestation and postpartum (lactating) females. Non-hypoparathyroid case reports were also included to further analyze and synthesize an overall likely pathophysiological explanation. Through the literature search, 24 papers were identified covering such a phenomenon in patients with hypoparathyroidism, alongside multiple additional reports of a similar occurrence in patients without underlying hypoparathyroidism. The pathophysiology is believed to occur due to the placental production of parathyroid hormone-related peptide (PTHrP) during gestation, with further production from the lactating mammary glands during the postpartum period. A typical pattern is observed, with increased PTHrP and suppressed PTH throughout both gestation and lactation (present in both normal and hypoparathyroid subjects). The concept of PTHrP-induced hypercalcemia is further demonstrated in patients without hypoparathyroidism, including subjects with placental hypersecretion and mammary gland enlargement. It is evident that patients with hypoparathyroidism may require a dosage reduction during late gestation and lactation, due to the risk for hypercalcemia. In addition to patients with hypoparathyroidism, this pathophysiological phenomenon occurs in unsuspecting patients, demonstrating the need for all clinicians in contact with pregnant females to be aware of this uncommon - yet perilous - occurrence.

Association Between Primary Hyperparathyroidism and Secondary Diabetes Mellitus: Findings From a Scoping Review.

An ill-defined association exists between diabetes mellitus (insulin resistance) and primary hyperparathyroidism. This article explores this phenomenon while providing an explanation for such a relationship and reviewing the evidence regarding the response to insulin sensitivity following a parathyroidectomy. Primary hyperparathyroidism may increase the risk of developing insulin resistance; peculiarly, this is not present in all patients. It is likely that both intracellular hypercalcemia and hypophosphatemia alter the insulin receptor expression and response; the contribution of parathyroid hormone is less clear. Following parathyroidectomy, patients may demonstrate improvement in their insulin sensitivity, while others have no response or a detrimental effect. A varied phenotype exists among patients, and furthermore, it is unclear why certain patients demonstrate improvement in insulin sensitivity following a parathyroidectomy, whereas others fail to do so. While this review provides a broad overview of the general endocrine community, it is imperative to note that clinical applicability is limited until further studies address these remaining uncertainties. Due to the lack of understanding regarding this endocrinological enigma, the presence of insulin resistance, at this present time, should not be a criterion for a parathyroidectomy.

The Unforeseen Diagnosis: Hyperparathyroidism-Jaw Tumour Syndrome Case Report and Review of the Literature.

Hypercalcaemia and its systemic sequelae are a relatively common finding amongst patients in the field of endocrinology. Primary hyperparathyroidism, a frequent cause of hypercalcaemia, is often seen among middle-aged female patients, typically resulting from an underlying single-gland adenoma. Although patients may present with symptoms (nephrolithiasis, musculoskeletal discomfort, dehydration, or mood disturbance, to name a few), hypercalcaemia is rather frequently identified incidentally. In younger patients, a familial form of primary hyperparathyroidism must be considered, with a positive diagnosis mandating familial screening. Hyperparathyroidism-jaw tumour syndrome is one such autosomal dominant familial disorder, characterised by a mutation in the cell division cycle 73 (CDC73; also known as HRPT-2) tumour suppressor gene. This disorder is characterised by multiple pleiotropic phenomena, including recurrent primary hyperparathyroidism (and the effects of hypercalcaemia), neoplasms (such as uterine, renal, mandibular, and maxillary), and infertility. A patient not conforming to the classic candidacy for primary hyperparathyroidism requires consideration for a familial cause. Case Description. We present a rare diagnostic entity-hyperparathyroidism-jaw tumour (HPT-JT) syndrome-in a 36-year-old female with recurrent primary hyperparathyroidism, frequent nephrolithiasis, and infertility for 18 years prior to the diagnosis. We aim to promote awareness amongst medical professionals of this rare, but nonetheless essential differential diagnosis through a case report and review of the literature. Conclusion. Medical professionals must avoid diagnostic overshadowing and display a low threshold for genetic testing in younger patients with primary hyperparathyroidism. The importance of proper identification extends beyond the patient to their relatives and offspring.