Primary hyperparathyroidism (PHPT) is a disorder of calcium homeostasis. We report the case of a 17-year-old adolescent male, who presented with an acute psychosis coinciding with severe hypercalcemia and markedly elevated intact parathyroid hormone (iPTH) level and low vitamin D level. A Sestamibi scan showed a positive signal inferior to the left lobe of the thyroid gland. He had only a partial response to the initial medical and psychiatric management. The enlarged parathyroid gland was resected surgically and postoperatively serum calcium and iPTH levels normalized. The histopathology was compatible with a benign adenoma. Patient’s acute psychotic symptoms resolved gradually after surgery; however he remained under psychiatric care for the behavioral issues for about 6 months after surgery. While psychosis is a rare clinical manifestation of hypercalcemia secondary to PHPT in pediatric population, it should be considered as a clinical clue in an otherwise asymptomatic pediatric patient. 1. Introduction Primary hyperparathyroidism (PHPT) is a disorder of calcium homeostasis. It is rare in pediatric population (age < 18 years) and is usually caused by a single parathyroid adenoma (PA) [1–3]. PA has an incidence of 2–5 per 100,000 children [1]. Elevated levels of intact parathyroid hormone (iPTH) lead to persistent hypercalcemia. PA commonly involves the inferior parathyroid glands, although it can be found in several ectopic sites including thyroid gland, thymus, pericardium, and retroesophageal areas [4]. Hypercalcemia due to PA presents initially as nonspecific symptoms like polyuria, weakness, fatigue, irritability, anorexia, abdominal pain, nausea, emesis, and weight loss. Therefore, the diagnosis of hypercalcemia is often delayed due to subtle clinical symptoms until the complications emerge [1]. These include renal stones, osteopenia and osteoporosis, osteitis fibrosa cystica, peptic ulcer disease, pancreatitis, constipation, hypertension, arrhythmias (shortening of QT interval) [5], and neuropsychiatric diseases [6]. PHPT should be included in the differential diagnosis of all cases of hypercalcemia. Common causes of hypercalcemia include William’s syndrome, idiopathic infantile hypercalcemia, malignancy, and toxicity with drugs like thiazides and vitamin A. Rare causes include familial hypocalciuric hypercalcemia, multiple endocrine neoplasia I and II syndromes, subcutaneous fat necrosis, granulomatous disorders, hypervitaminosis D, malignancies, adrenal insufficiency [7], hyperthyroidism, hypothyroidism, and limb fracture with immobilization
References
[1]
J. Kollars, A. E. Zarroug, J. van Heerden et al., “Primary hyperparathyroidism in pediatric patients,” Pediatrics, vol. 115, no. 4, pp. 974–980, 2005.
[2]
J. M. Ruda, C. S. Hollenbeak, and B. C. Stack Jr., “A systematic review of the diagnosis and treatment of primary hyperparathyroidism from 1995 to 2003,” Otolaryngology: Head and Neck Surgery, vol. 132, no. 3, pp. 359–372, 2005.
[3]
D. Melliere, D. Berrahal, L. Perlemuter, E. Hindie, and D. Simon, “Primary hyperparathyroidism. Symptoms related to age, sex, calcaemia, anatomic lesions and weight of the glands,” Presse Medicale, vol. 24, no. 39, pp. 1889–1893, 1995.
[4]
T. R. S. Prasad and V. Bhatnagar, “Giant solitary parathyroid adenoma presenting with bone disease,” Indian Pediatrics, vol. 39, no. 11, pp. 1044–1047, 2002.
[5]
M. F. Carroll and D. S. Schade, “A practical approach to hypercalcemia,” American Family Physician, vol. 67, no. 9, pp. 1959–1966, 2003.
[6]
M. A. Levine, “Primary hyperparathyroidism: 7,000 years of progress,” Cleveland Clinic Journal of Medicine, vol. 72, no. 12, pp. 1084–1098, 2005.
[7]
A. Montoli, G. Colussi, and L. Minetti, “Hypercalcaemia in Addison's disease: calciotropic hormone profile and bone histology,” Journal of Internal Medicine, vol. 232, no. 6, pp. 535–540, 1992.
[8]
V. I. Reus, “Behavioral disturbances associated with endocrine disorders,” Annual Review of Medicine, vol. 37, pp. 205–214, 1986.
[9]
D. Hirsch, G. Orr, V. Kantarovich, H. Hermesh, E. Stern, and L. Blum, “Gushing's syndrome presenting as a schizophrenia-like psychotic state,” Israel Journal of Psychiatry and Related Sciences, vol. 37, no. 1, pp. 46–50, 2000.
[10]
C.-L. Lu, Y.-C. Lee, S.-J. Tsai, P.-G. Hu, and C.-B. Sim, “Psychiatric disturbances associated with hyperthyroidism: an analysis report of 30 cases,” Zhonghua Yi Xue Za Zhi (Taipei), vol. 56, no. 6, pp. 393–398, 1995.
[11]
V. Bhatara, M. G. Alshari, P. Warhol, J. M. McMillin, and A. Bhatara, “Coexistent hypothyroidism, psychosis, and severe obsessions in an adolescent: a 10-year follow-up,” Journal of Child and Adolescent Psychopharmacology, vol. 14, no. 2, pp. 315–323, 2004.
[12]
B. Spivak, M. Radvan, R. Ohring, and A. Weizman, “Primary hyperparathyroidism, psychiatric manifestations, diagnosis and management,” Psychotherapy and Psychosomatics, vol. 51, no. 1, pp. 38–44, 1989.
[13]
G. R. Geffken, H. E. Ward, J. P. Staab, S. L. K. Carmichael, and D. L. Evans, “Psychiatric morbidity in endocrine disorders,” Psychiatric Clinics of North America, vol. 21, no. 2, pp. 473–489, 1998.
[14]
P. J. Velasco, M. Manshadi, K. Breen, and S. Lippmann, “Psychiatric aspects of parathyroid disease,” Psychosomatics, vol. 40, no. 6, pp. 486–490, 1999.
[15]
G. Athanassenas, E. Papadopoulos, A. Kourkoubas et al., “Serum calcium and magnesium levels in chronic schizophrenics,” Journal of Clinical Psychopharmacology, vol. 3, no. 4, pp. 212–216, 1983.
[16]
S. H. Snyder, “Molecular mechanism of psychotropic drug action,” in Proceedings of the 136th American Psychiatric Association Annual Meeting, New York, NY, USA, 1983.
[17]
C. Joborn, J. Hetta, H. Johansson et al., “Psychiatric morbidity in primary hyperparathyroidism,” World Journal of Surgery, vol. 12, no. 4, pp. 476–481, 1988.
[18]
R. D. Alarcon and J. A. Franceschini, “Hyperparathyroidism and paranoid psychosis: case report and review of the literature,” British Journal of Psychiatry, vol. 145, pp. 477–486, 1984.
[19]
J. A. van Heerden and C. S. Grant, “Surgical treatment of primary hyperparathyroidism: an institutional perspective,” World Journal of Surgery, vol. 15, no. 6, pp. 688–692, 1991.
[20]
C. R. Harman, J. A. van Heerden, D. R. Farley, C. S. Grant, G. B. Thompson, and K. Curlee, “Sporadic primary hyperparathyroidism in young patients: a separate disease entity?” Archives of Surgery, vol. 134, no. 6, pp. 651–656, 1999.
[21]
C. B. Huang, S. C. Huang, F. F. Chou, and W. J. Chen, “Primary hyperparathyroidism in children: report of a case and a brief review of the literature,” Journal of the Formosan Medical Association, vol. 92, no. 12, pp. 1095–1098, 1993.
[22]
D. H. Fulmer, A. B. Dimich, E. O. Rothschild, and W. P. Myers, “Treatment of hypercalcemia. Comparison of intravenously administered phosphate, sulfate, and hydrocortisone,” Archives of Internal Medicine, vol. 129, no. 6, pp. 923–930, 1972.
[23]
U. Querfeld, I. B. Salusky, and R. N. Fine, “Treatment of severe hypercalcemia with peritoneal dialysis in an infant with end-stage renal disease,” Pediatric Nephrology, vol. 2, no. 3, pp. 323–325, 1988.
[24]
H. Lauschke, M. Kaminski, H. Stratmann, and A. Hirner, “The parathyrotoxic crisis—a surgical emergency,” Chirurg, vol. 71, no. 1, pp. 58–62, 2000.
[25]
Y. Ohata, T. Yamamoto, Y. Kitai et al., “A case of primary hyperparathyroidism in childhood found by a chance hematuria,” Clinical Pediatric Endocrinology, vol. 16, no. 1, pp. 11–16, 2007.
[26]
Y. Fujikawa, M. Koga, H. Sugao et al., “Neurobehavioral symptoms in mild primary hyperparathyroidism: related to hypercalcemia but not improved by parathyroidectomy,” Henry Ford Hospital Medical Journal, vol. 35, no. 4, pp. 211–215, 1987.
[27]
C. K. Smith, J. Barish, J. Correa, and R. H. Williams, “Psychiatric disturbance in endocrinologic disease,” Psychosomatic Medicine, vol. 34, no. 1, pp. 69–86, 1972.
