Back to Search Start Over

Pseudohypoparathyroidism type 1B (PHP1B), a rare disorder encountered in adolescence.

Authors :
Vlachopapadopoulou EA
Anagnostou E
Dikaiakou E
Hanna P
Tsolia M
Michalacos S
Linglart A
Karavanaki K
Source :
Journal of pediatric endocrinology & metabolism : JPEM [J Pediatr Endocrinol Metab] 2020 Nov 26; Vol. 33 (11), pp. 1475-1479.
Publication Year :
2020

Abstract

Objectives The objective of this paper is to report a peculiar case of a patient with pseudohypoparathyroidism type 1b (PHP1B). Pseudohypoparathyroidism (PHP) refers to a group of disorders characterized by hypocalcemia, hyperphosphatemia, and elevated parathyroid hormone (PTH) concentrations as the result of end-organ unresponsiveness to PTH. Case presentation We present a 14-year-old boy, who was admitted with severe symptomatic hypocalcaemia, absence of dysmorphic features and Albright's hereditary osteodystrophy features. Laboratory investigations revealed markedly low serum calcium, high phosphate, markedly elevated PTH levels and vitamin D insufficiency, while magnesium, albumin, ALP and TSH were normal. The clinical and laboratory findings were consistent with PHP1B. Molecular analysis revealed loss of methylation at the AB DMR of the GNAS locus, confirming the diagnosis. Yet no STX16 deletion was detected. Conclusions It is possible that delSTX16- patients carry a defect in an element that controls the methylation both at the GNAS-A/B DMR and at the GNAS-AS2. This rare case emphasizes the need of individualized molecular analysis in PHP1B patients in order to elucidate the possible molecular defect.

Details

Language :
English
ISSN :
2191-0251
Volume :
33
Issue :
11
Database :
MEDLINE
Journal :
Journal of pediatric endocrinology & metabolism : JPEM
Publication Type :
Academic Journal
Accession number :
33027051
Full Text :
https://doi.org/10.1515/jpem-2020-0192