Primary hyperparathyroidism presenting as acute psychosis secondary to hypercalcaemia requiring curative parathyroidectomy

Primary hyperparathyroidism presenting as acute psychosis secondary to hypercalcaemia requiring... Primary hyperparathyroidism (PHPT) can cause hypercalcaemia secondary to a pathologically high secretion of parathyroid hormone. Rarely this can first manifest as acute psychosis. It is imperative to exclude organic causes of psychosis before labelling the psychosis as primarily psychological. If hypercalcaemia is revealed, investigation is required to elucidate the underlying cause whilst instigating treatment to lower serum calcium levels. If PHPT is the underlying pathology, subse- quent treatment involves surgical exploration and resection of the parathyroid adenoma or hyperplasia. mood and amnesia for 2 weeks. She was sleeping poorly and INTRODUCTION had reduced appetite. A collateral history from her daughter Hypercalcaemia often presents with subtle clinical symptoms revealed that she had been laughing to herself, wandering such as polyuria, polydipsia, weakness, irritability, abdominal without orientation and not attending work. There was no his- pain and nausea. This vague clinical picture can delay accurate tory of alcohol or drug consumption or smoking, and no per- diagnosis until more significant complications arise such as tinent family history. acute pancreatitis, fractures and arrhythmias. Rarely, hypercal- She was orientated in time, place and person and her men- caemia can manifest as acute psychosis. tal state examination revealed a well-kempt lady who main- We report the case of acute psychosis secondary to hyper- tained eye contact and spoke coherently. calcaemia caused by primary hyperparathyroidism (PHPT) and On initial presentation, a psychiatric assessment was done its subsequent management with parathyroidectomy. but her serum calcium levels were not checked. She was started on an antidepressant (sertraline 50 mg) and an antipsychotic (olanzapine 10 mg) and discharged. She attended hospital two CASE REPORT weeks later for worsening polyuria, polydipsia, anxiety and confu- Our patient is a middle-aged Afro-Caribbean lady who was seen by sion. Investigations revealed raised levels of serum calcium (3.0 the liaison psychiatry team after experiencing intimidating third- mmol/l), raised parathyroid hormone (PTH) (14 ng/l), low vitamin person auditory hallucinations, paranoid delusions, confusion, low D (8 nmol/l), low serum phosphate (0.6 mmol/l) and a small left Received: January 5, 2018. Accepted: February 4, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy023/4898377 by Ed 'DeepDyve' Gillespie user on 16 March 2018 2 P. Singh et al. sided neck lump. Thyroid function tests, chest X-ray, CT-head extracellular fluid. PTH also increases conversion of 25- and MRI brain were all normal. hydroxychloecalciferol to the more active 1,25-dihydroxychole- From these results PHPT was suspected and the hypercalcae- calciferol via the 1-hydroxylase enzyme in renal proximal mia treated with intravenous fluids and a pamidronate infusion. tubules, thereby increasing calcium and phosphate absorption Calcium levels normalized over the next few days along with her from the kidneys and alimentary canal. psychiatric symptoms suggesting a causative relationship. An Negative feedback is integrated into this system through ultrasound neck revealed an 11 mm lesion with echogenicity and inhibition of PTH secretion by high serum calcium levels acting internal vascularity, likely to be an atypical parathyroid adenoma directly on the parathyroid gland, thereby maintaining calcium (Fig. 1). A Sestamibi scintigraphy scan was inconclusive in correlat- levels within the normal range. In PHPT, PTH is autonomously ing with the ultrasound or clinical impression, however, evidence secreted independently of this negative feedback, resulting in was sufficient to warrant surgical exploration with removal of the high PTH and calcium levels, and often low phosphate and adenoma as a definitive treatment for the PHPT and psychosis. vitamin D levels. Up to 30% of cases of hypercalcaemia in PHPT are asymptom- atic at presentation [6] with detection usually from coincidental blood tests. Clues in the history relating to symptomatic hypercal- DISCUSSION caemia should prompt a full neck examination for parathyroid Psychosis may be the first manifestation of endocrine abnor- gland masses. Investigations should include anECG whichmay malities such as Cushing’s syndrome [1], hyperthyroidism [2], reveal a shortened QT interval, J-waves or even ventricular hypothyroidism [3] and rarely PHPT [4]. arrhythmias. Blood tests may demonstrate high calcium, high A direct mechanism between hypercalcaemia and psychosis PTH, low phosphate and low vitamin D levels indicating primary has not yet been elucidated. Calcium is known to have a role in or tertiary hyperparathyroidism. Urea and electrolyte tests may determining monoamine metabolism in the central nervous indicate if the patient has chronic kidney disease which can system through modulation of dopaminergic and cholinergic result in tertiary hyperparathyroidism. Urinalysis may reveal cal- metabolism and the release of neurotransmitters at synaptic ciuresis and if urine calcium levels are low, Familal hypocalciuric junctions. Increased serum calcium has been observed during hypercalcaemia (FHH) may be suspected. catatonic episodes in Schizophrenia patients [5]. Evidence for a Neck ultrasound is the first line imaging modality and can correlation between severity of psychosis and degree of hyper- illustrate abnormal masses. PHPT is caused by adenomas in 85% calcaemia is inconclusive. of cases, hyperplasia in 15% and carcinoma in under 1% of cases Hypercalcaemia has a multitude of causes including bone [7]. Ultrasound can help to differentiate the cause through asses- metastases, myeloma, a paraneoplastic response to PTH-related sing location, shape, echogenicity and vascularity. peptide from squamous cell carcinomas; iatrogenic complications Further imaging involves Sestamibi scintigraphy wherein the from thiazides, vitamin A or D toxicity; inherited conditions includ- radionucleotide is more rapidly absorbed by hyperfunctioning ing infantile hypercalcaemia and FHH; granulomatous dis- parathyroid tissue than normal tissue allowing localization with a eases including sarcoidosis; neuroendocrine syndromes including gamma camera. However, evidence suggests that Sestamibi scans multiple endocrine neoplasia, hyper or hypothyroidism, and are unreliable for small adenomas or multiglandular disease, with hyperparathyroidism. false negative rates of 22% and overall sensitivity 77% [8]. PTH is normally secreted from the parathyroid glands in In the acute setting hypercalcaemia is treated with intraven- response to low serum calcium levels; it acts on the kidneys ous fluids, loop diuretics and bisphosphonates. Haemodialysis and bone to increase serum calcium levels, and enhances the may be used when patients are unable to tolerate high volume effects of vitamin D. It increases renal calcium reabsorption in hydration or with refractory hypercalcaemia [9]. The definitive the ascending loop of Henlé, distal tubule and collecting tubule, treatment is surgical excision of the parathyroid adenoma. Pre- and increases phosphate excretion in the proximal tubule. PTH surgical localization imaging and hand-held intraoperative gam- acts on bone by stimulating osteoclasts directly and indirectly ma probes allow for minimally invasive parathyroidectomy. via osteoblastic cytokines to pump calcium from bone fluid into Intraoperative PTH assays prior to and after adenoma excision with a decrease of >50% at 10 min post excision indicates suc- cessful resection, otherwise another adenoma or hyperplasia may be present, necessitating further exploration [10]. Acute psychosis is a rare manifestation of hypercalcaemia. We suggest investigation and exclusion of organic causes of psychosis before commencing psychiatric medication, as per the ICD-10 criteria, and where hyperparathyroidism is causa- tive, only surgical excision is curative. CONFLICT OF INTEREST STATEMENT None. FUNDING None. INFORMED CONSENT Informed consent received from the patient for the production of this manuscript and the use of any accompanying images. Figure 1: Ultrasound neck image illustrating the parathyroid adenoma measur- ing 11 mm with echogenicity and internal vasculature. Consent is available for review if required. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy023/4898377 by Ed 'DeepDyve' Gillespie user on 16 March 2018 Hyperparathyroidism presenting as psychosis 3 4. Babar G, Alemzadeh R. A case of acute psychosis in an ado- AUTHOR’S CONTRIBUTION lescent male. Case Rep Endocrinol 2014;2014:937631. All authors contributed equally to the production of this article. 5. Athanassenas G, Papadopoulos E, Kourkoubas A. Serum cal- cium and magnesium levels in chronic schizophrenics. PRESENTATION J Clin Psychopharmacol 1983;3:212–6. 6. Silverberg SJ, Walker MD, Bilezikian JP. Asymptomatic This article was presented as an international oral presentation at primary hyperparathyroidism. J Clin Densitom 2013;16: the European Society of Surgical Research, July 2017, Amsterdam, 14–21. Netherlands. 7. Thompson NW, Eckhauser FE, Harness JK. The anatomy of primary hyperparathyroidism. Surgery 1982;92:814–21. REFERENCES 8. Merlino JI, Ko K, Minotti A, McHenry CR. The false nega- 1. Tang A, O’Sullivan AJ, Diamond T, Gerard A, Campbell P. Psy- tive technetium-99m-sestamibi scan in patients with pri- chiatric symptoms as a clinical presentation of Cushing’ssyn- mary hyperparathyroidism: correlation with clinical factors drome. Ann Gen Psychiatry 2013;12:23. and operative findings. Am Surg 2003;69:225–9. discussion 2. Mahmood A, Ashton AK, Hina FH. Psychotic symptoms 9-30. with underlying graves disease: a case report. Prim Care 9. Argiles A, Kerr PG, Canaud B, Flavier JL, Mion C. Calcium Companion J Clin Psychiatry 2005;7:311–2. kinetics and the long-term effects of lowering dialysate cal- 3. Heinrich TW, Grahm G. Hypothyroidism presenting as psych- cium concentration. Kidney Int 1993;43:630–40. osis: myxedema madness revisited. Prim Care Companion J Clin 10. Suliburk JW, Perrier ND. Primary hyperparathyroidism. Psychiatry 2003;5:260–6. Oncologist 2007;12:644–53. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy023/4898377 by Ed 'DeepDyve' Gillespie user on 16 March 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Primary hyperparathyroidism presenting as acute psychosis secondary to hypercalcaemia requiring curative parathyroidectomy

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Abstract

Primary hyperparathyroidism (PHPT) can cause hypercalcaemia secondary to a pathologically high secretion of parathyroid hormone. Rarely this can first manifest as acute psychosis. It is imperative to exclude organic causes of psychosis before labelling the psychosis as primarily psychological. If hypercalcaemia is revealed, investigation is required to elucidate the underlying cause whilst instigating treatment to lower serum calcium levels. If PHPT is the underlying pathology, subse- quent treatment involves surgical exploration and resection of the parathyroid adenoma or hyperplasia. mood and amnesia for 2 weeks. She was sleeping poorly and INTRODUCTION had reduced appetite. A collateral history from her daughter Hypercalcaemia often presents with subtle clinical symptoms revealed that she had been laughing to herself, wandering such as polyuria, polydipsia, weakness, irritability, abdominal without orientation and not attending work. There was no his- pain and nausea. This vague clinical picture can delay accurate tory of alcohol or drug consumption or smoking, and no per- diagnosis until more significant complications arise such as tinent family history. acute pancreatitis, fractures and arrhythmias. Rarely, hypercal- She was orientated in time, place and person and her men- caemia can manifest as acute psychosis. tal state examination revealed a well-kempt lady who main- We report the case of acute psychosis secondary to hyper- tained eye contact and spoke coherently. calcaemia caused by primary hyperparathyroidism (PHPT) and On initial presentation, a psychiatric assessment was done its subsequent management with parathyroidectomy. but her serum calcium levels were not checked. She was started on an antidepressant (sertraline 50 mg) and an antipsychotic (olanzapine 10 mg) and discharged. She attended hospital two CASE REPORT weeks later for worsening polyuria, polydipsia, anxiety and confu- Our patient is a middle-aged Afro-Caribbean lady who was seen by sion. Investigations revealed raised levels of serum calcium (3.0 the liaison psychiatry team after experiencing intimidating third- mmol/l), raised parathyroid hormone (PTH) (14 ng/l), low vitamin person auditory hallucinations, paranoid delusions, confusion, low D (8 nmol/l), low serum phosphate (0.6 mmol/l) and a small left Received: January 5, 2018. Accepted: February 4, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy023/4898377 by Ed 'DeepDyve' Gillespie user on 16 March 2018 2 P. Singh et al. sided neck lump. Thyroid function tests, chest X-ray, CT-head extracellular fluid. PTH also increases conversion of 25- and MRI brain were all normal. hydroxychloecalciferol to the more active 1,25-dihydroxychole- From these results PHPT was suspected and the hypercalcae- calciferol via the 1-hydroxylase enzyme in renal proximal mia treated with intravenous fluids and a pamidronate infusion. tubules, thereby increasing calcium and phosphate absorption Calcium levels normalized over the next few days along with her from the kidneys and alimentary canal. psychiatric symptoms suggesting a causative relationship. An Negative feedback is integrated into this system through ultrasound neck revealed an 11 mm lesion with echogenicity and inhibition of PTH secretion by high serum calcium levels acting internal vascularity, likely to be an atypical parathyroid adenoma directly on the parathyroid gland, thereby maintaining calcium (Fig. 1). A Sestamibi scintigraphy scan was inconclusive in correlat- levels within the normal range. In PHPT, PTH is autonomously ing with the ultrasound or clinical impression, however, evidence secreted independently of this negative feedback, resulting in was sufficient to warrant surgical exploration with removal of the high PTH and calcium levels, and often low phosphate and adenoma as a definitive treatment for the PHPT and psychosis. vitamin D levels. Up to 30% of cases of hypercalcaemia in PHPT are asymptom- atic at presentation [6] with detection usually from coincidental blood tests. Clues in the history relating to symptomatic hypercal- DISCUSSION caemia should prompt a full neck examination for parathyroid Psychosis may be the first manifestation of endocrine abnor- gland masses. Investigations should include anECG whichmay malities such as Cushing’s syndrome [1], hyperthyroidism [2], reveal a shortened QT interval, J-waves or even ventricular hypothyroidism [3] and rarely PHPT [4]. arrhythmias. Blood tests may demonstrate high calcium, high A direct mechanism between hypercalcaemia and psychosis PTH, low phosphate and low vitamin D levels indicating primary has not yet been elucidated. Calcium is known to have a role in or tertiary hyperparathyroidism. Urea and electrolyte tests may determining monoamine metabolism in the central nervous indicate if the patient has chronic kidney disease which can system through modulation of dopaminergic and cholinergic result in tertiary hyperparathyroidism. Urinalysis may reveal cal- metabolism and the release of neurotransmitters at synaptic ciuresis and if urine calcium levels are low, Familal hypocalciuric junctions. Increased serum calcium has been observed during hypercalcaemia (FHH) may be suspected. catatonic episodes in Schizophrenia patients [5]. Evidence for a Neck ultrasound is the first line imaging modality and can correlation between severity of psychosis and degree of hyper- illustrate abnormal masses. PHPT is caused by adenomas in 85% calcaemia is inconclusive. of cases, hyperplasia in 15% and carcinoma in under 1% of cases Hypercalcaemia has a multitude of causes including bone [7]. Ultrasound can help to differentiate the cause through asses- metastases, myeloma, a paraneoplastic response to PTH-related sing location, shape, echogenicity and vascularity. peptide from squamous cell carcinomas; iatrogenic complications Further imaging involves Sestamibi scintigraphy wherein the from thiazides, vitamin A or D toxicity; inherited conditions includ- radionucleotide is more rapidly absorbed by hyperfunctioning ing infantile hypercalcaemia and FHH; granulomatous dis- parathyroid tissue than normal tissue allowing localization with a eases including sarcoidosis; neuroendocrine syndromes including gamma camera. However, evidence suggests that Sestamibi scans multiple endocrine neoplasia, hyper or hypothyroidism, and are unreliable for small adenomas or multiglandular disease, with hyperparathyroidism. false negative rates of 22% and overall sensitivity 77% [8]. PTH is normally secreted from the parathyroid glands in In the acute setting hypercalcaemia is treated with intraven- response to low serum calcium levels; it acts on the kidneys ous fluids, loop diuretics and bisphosphonates. Haemodialysis and bone to increase serum calcium levels, and enhances the may be used when patients are unable to tolerate high volume effects of vitamin D. It increases renal calcium reabsorption in hydration or with refractory hypercalcaemia [9]. The definitive the ascending loop of Henlé, distal tubule and collecting tubule, treatment is surgical excision of the parathyroid adenoma. Pre- and increases phosphate excretion in the proximal tubule. PTH surgical localization imaging and hand-held intraoperative gam- acts on bone by stimulating osteoclasts directly and indirectly ma probes allow for minimally invasive parathyroidectomy. via osteoblastic cytokines to pump calcium from bone fluid into Intraoperative PTH assays prior to and after adenoma excision with a decrease of >50% at 10 min post excision indicates suc- cessful resection, otherwise another adenoma or hyperplasia may be present, necessitating further exploration [10]. Acute psychosis is a rare manifestation of hypercalcaemia. We suggest investigation and exclusion of organic causes of psychosis before commencing psychiatric medication, as per the ICD-10 criteria, and where hyperparathyroidism is causa- tive, only surgical excision is curative. CONFLICT OF INTEREST STATEMENT None. FUNDING None. INFORMED CONSENT Informed consent received from the patient for the production of this manuscript and the use of any accompanying images. Figure 1: Ultrasound neck image illustrating the parathyroid adenoma measur- ing 11 mm with echogenicity and internal vasculature. Consent is available for review if required. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy023/4898377 by Ed 'DeepDyve' Gillespie user on 16 March 2018 Hyperparathyroidism presenting as psychosis 3 4. Babar G, Alemzadeh R. A case of acute psychosis in an ado- AUTHOR’S CONTRIBUTION lescent male. Case Rep Endocrinol 2014;2014:937631. All authors contributed equally to the production of this article. 5. Athanassenas G, Papadopoulos E, Kourkoubas A. Serum cal- cium and magnesium levels in chronic schizophrenics. PRESENTATION J Clin Psychopharmacol 1983;3:212–6. 6. Silverberg SJ, Walker MD, Bilezikian JP. Asymptomatic This article was presented as an international oral presentation at primary hyperparathyroidism. J Clin Densitom 2013;16: the European Society of Surgical Research, July 2017, Amsterdam, 14–21. Netherlands. 7. Thompson NW, Eckhauser FE, Harness JK. The anatomy of primary hyperparathyroidism. Surgery 1982;92:814–21. REFERENCES 8. Merlino JI, Ko K, Minotti A, McHenry CR. The false nega- 1. Tang A, O’Sullivan AJ, Diamond T, Gerard A, Campbell P. Psy- tive technetium-99m-sestamibi scan in patients with pri- chiatric symptoms as a clinical presentation of Cushing’ssyn- mary hyperparathyroidism: correlation with clinical factors drome. Ann Gen Psychiatry 2013;12:23. and operative findings. Am Surg 2003;69:225–9. discussion 2. Mahmood A, Ashton AK, Hina FH. Psychotic symptoms 9-30. with underlying graves disease: a case report. Prim Care 9. Argiles A, Kerr PG, Canaud B, Flavier JL, Mion C. Calcium Companion J Clin Psychiatry 2005;7:311–2. kinetics and the long-term effects of lowering dialysate cal- 3. Heinrich TW, Grahm G. Hypothyroidism presenting as psych- cium concentration. Kidney Int 1993;43:630–40. osis: myxedema madness revisited. Prim Care Companion J Clin 10. Suliburk JW, Perrier ND. Primary hyperparathyroidism. Psychiatry 2003;5:260–6. Oncologist 2007;12:644–53. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy023/4898377 by Ed 'DeepDyve' Gillespie user on 16 March 2018

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Journal of Surgical Case ReportsOxford University Press

Published: Feb 1, 2018

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