CASE REPORT Pseudohypoparathyroidism diagnosed in adulthood: maxillofacial clinical and radiographic findings A. Delantoni 1 • K. Lyroudia 1 • V. Rafailidis 2 • I. Chryssogonidis 3 Received: 19 January 2016 / Accepted: 11 June 2016 / Published online: 27 July 2016 Ó Japanese Society for Oral and Maxillofacial Radiology and Springer Japan 2016 Abstract Pseudohypoparathyroidism is a rare condition first described in 1942. Affected patients do not have a deficiency of parathyroid hormone; instead, the kidneys and bones are unable to respond to parathyroid hormone in the normal manner. Few cases of pseudohypoparathy- roidism have been reported in the dental literature, and most do not describe the advanced condition because the diagnosis is usually made, and treatment commenced, at an early age. We herein describe a patient who was diagnosed with pseudohypoparathyroidism at the age of 28 years, after completion of full facial and skeletal development. Panoramic radiography revealed a large number of impactions and incomplete root formation. Cone beam computed tomography (CBCT) showed calcification of the basal ganglia of the brain and incomplete root formation of the teeth with thin enamel and large pulp chambers. To our knowledge, this is the first patient with pseudohy- poparathyroidism to be studied with both panoramic radiography and CBCT. Pseudohypoparathyroidism is a rare condition that has not been previously studied with CBCT. The characteristic clinical and radiographic find- ings should alert dentists to this condition. Keywords CBCT Á Incidental findings Á Basal ganglia calcification Á Brain calcification Introduction Hypoparathyroidism represents a group of diseases charac- terized by hypocalcemia and hyperphosphatemia. These disorders can be the result of either defective secretion of parathyroid hormone by the parathyroid gland or failure of the target organs to respond to this hormone. The latter condition is known as pseudohypoparathyroidism (PHP) and is associated with an elevated plasma concentration of parathyroid hormone [1]. Fuller Albright described the first patients with this disorder in 1942 [2]. These patients demonstrated phenotypical features such as a rounded face, short stature, obesity, brachydactyly, and ectopic calcifica- tions. These features are typically found in patients with type Ia PHP, also known as Albright hereditary osteodystrophy. Type Ib PHP has also been described and lacks these phe- notypical characteristics. Distinction between type I and type II PHP is based on the response of cyclic adenosine monophosphate (cAMP) to parathyroid hormone; this response is abnormal in type I and normal in type II [3]. These metabolic abnormalities are very rare, with a reported prevalence of only 0.79 per 100,000 individuals [4]. We herein describe the maxillofacial and dental findings of a young patient with type Ia PHP. This is the first case report to describe the characteristics of PHP on cone beam computed tomography (CBCT). The potential value of CBCT to identify incidental findings of systemic condi- tions during routine examinations is also demonstrated. Case report A 28-year-old man presented to the Dental School of the Aristotle University of Thessaloniki. He had been recently diagnosed with type Ia PHP. The disorder was incidentally & A. Delantoni adelantoni@yahoo.com; andelant@dent.auth.gr 1 Faculty of Dentistry, Aristotle University of Thessaloniki, Agiou Dimitriou Street, 54124 Thessaloniki, Greece 2 Department of Radiology, AHEPA Hospital, Thessaloniki, Greece 3 Department of Radiology, Faculty of Medicine, Aristotle University of Thessaloniki, Thessaloniki, Greece 123 Oral Radiol (2017) 33:153–156 DOI 10.1007/s11282-016-0254-8