Browse

You are looking at 1 - 7 of 7 items

Carmina Teresa Fuss Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

Search for other papers by Carmina Teresa Fuss in
Google Scholar
PubMed
Close
,
Stephanie Burger-Stritt Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

Search for other papers by Stephanie Burger-Stritt in
Google Scholar
PubMed
Close
,
Silke Horn Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

Search for other papers by Silke Horn in
Google Scholar
PubMed
Close
,
Ann-Cathrin Koschker Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

Search for other papers by Ann-Cathrin Koschker in
Google Scholar
PubMed
Close
,
Kathrin Frey Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

Search for other papers by Kathrin Frey in
Google Scholar
PubMed
Close
,
Almuth Meyer Division of Endocrinology and Diabetology, Department of Internal Medicine, Helios Klinikum Erfurt, Erfurt, Germany

Search for other papers by Almuth Meyer in
Google Scholar
PubMed
Close
, and
Stefanie Hahner Division of Endocrinology and Diabetology, Department of Medicine I, University Hospital Würzburg, Würzburg, Germany

Search for other papers by Stefanie Hahner in
Google Scholar
PubMed
Close

Summary

Standard treatment of hypoparathyroidism consists of supplementation of calcium and vitamin D analogues, which does not fully restore calcium homeostasis. In some patients, hypoparathyroidism is refractory to standard treatment with persistent low serum calcium levels and associated clinical complications. Here, we report on three patients (58-year-old male, 52-year-old female, and 48-year-old female) suffering from severe treatment-refractory postsurgical hypoparathyroidism. Two patients had persistent hypocalcemia despite oral treatment with up to 4 µg calcitriol and up to 4 g calcium per day necessitating additional i.v. administration of calcium gluconate 2–3 times per week, whereas the third patient presented with high frequencies of hypocalcemic and treatment-associated hypercalcemic episodes. S.c. administration of rhPTH (1–34) twice daily (40 µg/day) or rhPTH (1–84) (100 µg/day) only temporarily increased serum calcium levels but did not lead to long-term stabilization. In all three cases, treatment with rhPTH (1–34) as continuous s.c. infusion via insulin pump was initiated. Normalization of serum calcium and serum phosphate levels was observed within 1 week at daily 1–34 parathyroid hormone doses of 15 µg to 29.4 µg. Oral vitamin D and calcium treatment could be stopped or reduced and regular i.v. calcium administration was no more necessary. Ongoing efficacy of this treatment has been documented for up to 7 years so far. Therefore, we conclude that hypoparathyroidism that is refractory to both conventional treatment and s.c. parathyroid hormone (single or twice daily) may be successfully treated with continuous parathyroid hormone administration via insulin pump.

Learning points:

  • Standard treatment of hypoparathyroidism still consists of administration of calcium and active vitamin D.

  • Very few patients with hypoparathyroidism also do not respond sufficiently to standard treatment or administration of s.c. parathyroid hormone once or twice daily.

  • In those cases, continuous s.c. administration of parathyroid hormone via insulin pump may represent a successful treatment alternative.

Open access
Mawson Wang Nepean Blue Mountains Local Health District, Katoomba, New South Wales, Australia

Search for other papers by Mawson Wang in
Google Scholar
PubMed
Close
,
Catherine Cho Nepean Blue Mountains Local Health District, Katoomba, New South Wales, Australia

Search for other papers by Catherine Cho in
Google Scholar
PubMed
Close
,
Callum Gray Nepean Blue Mountains Local Health District, Katoomba, New South Wales, Australia

Search for other papers by Callum Gray in
Google Scholar
PubMed
Close
,
Thora Y Chai Department of Endocrinology, Nepean Blue Mountains Local Health District, Kingswood, New South Wales, Australia
Faculty of Medicine and Health, The University of Sydney, Sydney, New South Wales, Australia

Search for other papers by Thora Y Chai in
Google Scholar
PubMed
Close
,
Ruhaida Daud Nepean Blue Mountains Local Health District, Katoomba, New South Wales, Australia

Search for other papers by Ruhaida Daud in
Google Scholar
PubMed
Close
, and
Matthew Luttrell Department of Endocrinology, Nepean Blue Mountains Local Health District, Kingswood, New South Wales, Australia

Search for other papers by Matthew Luttrell in
Google Scholar
PubMed
Close

Summary

We report the case of a 65-year-old female who presented with symptomatic hypercalcaemia (corrected calcium of 4.57 mmol/L) with confusion, myalgias and abdominal discomfort. She had a concomitant metabolic alkalosis (pH 7.46, HCO3 - 40 mmol/L, pCO2 54.6 mmHg). A history of significant Quick-Eze use (a calcium carbonate based antacid) for abdominal discomfort, for 2 weeks prior to presentation, suggested a diagnosis of milk-alkali syndrome (MAS). Further investigations did not demonstrate malignancy or primary hyperparathyroidism. Following management with i.v. fluid rehydration and a single dose of i.v. bisphosphonate, she developed symptomatic hypocalcaemia requiring oral and parenteral calcium replacement. She was discharged from the hospital with stable biochemistry on follow-up. This case demonstrates the importance of a detailed history in the diagnosis of severe hypercalcaemia, with MAS representing the third most common cause of hypercalcaemia. We discuss its pathophysiology and clinical importance, which can often present with severe hypercalcaemia that can respond precipitously to calcium-lowering therapy.

Learning points:

  • Milk-alkali syndrome is an often unrecognised cause for hypercalcaemia, but is the third most common cause of admission for hypercalcaemia.

  • Calcium ingestion leading to MAS can occur at intakes as low as 1.0–1.5 g per day in those with risk factors.

  • Early recognition of this syndrome can avoid the use of calcium-lowering therapy such as bisphosphonates which can precipitate hypocalcaemia.

Open access
Florence Gunawan Barwon Health, Geelong University Hospital, Geelong, Victoria, Australia

Search for other papers by Florence Gunawan in
Google Scholar
PubMed
Close
,
Elizabeth George Barwon Health, Geelong University Hospital, Geelong, Victoria, Australia

Search for other papers by Elizabeth George in
Google Scholar
PubMed
Close
, and
Mark Kotowicz Barwon Health, Geelong University Hospital, Geelong, Victoria, Australia

Search for other papers by Mark Kotowicz in
Google Scholar
PubMed
Close

Summary

Denosumab is a fully human MAB that acts as a potent anti-resorptive by inhibiting activation of osteoclasts by inhibiting the receptor activator of nuclear factor-kappa B (RANK) ligand. Hypocalcaemia has been reported as one of the serious adverse sequelae of use of denosumab. We present a case of refractory hypocalcaemia following administration of a single dose of denosumab in a patient with metastatic castrate-resistant prostate cancer. The patient’s serum calcium and vitamin D concentrations and renal function were normal prior to denosumab administration. Serum alkaline phosphatase (ALP) level was however elevated pre-morbidly consistent with known bone metastases. The patient was treated with high-dose oral and IV calcium without any appreciable response in serum calcium. During his 30-day hospital admission, he demonstrated disease progression with development of new liver metastases and bone marrow involvement. Normocalcaemia was not achieved despite 1 month of aggressive therapy. Given the patient was asymptomatic and prognosis guarded, he was eventually discharged for ongoing supportive care under the palliative care team.

Learning points:

  • Denosumab is a potent anti-resorptive therapy and hypocalcaemia is one of the known adverse effects.

  • Serum calcium and vitamin D concentrations must be replete prior to administration of denosumab to reduce the risk of hypocalcaemia.

  • Denosumab has been proven to be more effective than zoledronic acid in preventing skeletal-related adverse effects in patients with metastatic castrate-resistant prostate cancer.

Open access
Maria P Yavropoulou 1st Propaedeutic Department of Internal Medicine, LAIKO General Hospital of Athens

Search for other papers by Maria P Yavropoulou in
Google Scholar
PubMed
Close
,
Efstathios Chronopoulos 2nd Orthopaedic Department, Konstantopouleio General Hospital

Search for other papers by Efstathios Chronopoulos in
Google Scholar
PubMed
Close
,
George Trovas Laboratory for Research of the Musculoskeletal System, Th Garofalidis, National and Kapodistrian University of Athens, Athens, Greece

Search for other papers by George Trovas in
Google Scholar
PubMed
Close
,
Emmanouil Avramidis 2nd Orthopaedic Department, Konstantopouleio General Hospital

Search for other papers by Emmanouil Avramidis in
Google Scholar
PubMed
Close
,
Francesca Marta Elli Endocrinology Unit, Department of Clinical Sciences and Community Health, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, University of Milan, Milan, Italy

Search for other papers by Francesca Marta Elli in
Google Scholar
PubMed
Close
,
Giovanna Mantovani Endocrinology Unit, Department of Clinical Sciences and Community Health, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, University of Milan, Milan, Italy

Search for other papers by Giovanna Mantovani in
Google Scholar
PubMed
Close
,
Pantelis Zebekakis 1st Department of Internal Medicine, AHEPA University Hospital, Thessaloniki, Greece

Search for other papers by Pantelis Zebekakis in
Google Scholar
PubMed
Close
, and
John G Yovos 1st Department of Internal Medicine, AHEPA University Hospital, Thessaloniki, Greece

Search for other papers by John G Yovos in
Google Scholar
PubMed
Close

Summary

Pseudohypoparathyroidism (PHP) is a heterogeneous group of rare endocrine disorders characterised by normal renal function and renal resistance to the action of the parathyroid hormone. Type 1A (PHP1A), which is the most common variant, also include developmental and skeletal defects named as Albright hereditary osteodystrophy (AHO). We present two cases, a 54- and a 33-year-old male diagnosed with PHP who were referred to us for persistently high levels of serum calcitonin. AHO and multinodular goitre were present in the 54-year-old male, while the second patient was free of skeletal deformities and his thyroid gland was of normal size and without nodular appearance. We performed GNAS molecular analysis (methylation status and copy number analysis by MS-MLPA) in genomic DNA samples for both patients. The analysis revealed a novel missense variant c.131T>G p.(Leu44Pro) affecting GNAS exon 1, in the patient with the clinical diagnosis of PHP1A. This amino acid change appears to be in accordance with the clinical diagnosis of the patient. The genomic DNA analysis of the second patient revealed the presence of the recurrent 3-kb deletion affecting the imprinting control region localised in the STX16 region associated with the loss of methylation (LOM) at the GNAS A/B differentially methylated region and consistent with the diagnosis of an autosomal dominant form of PHP type 1B (PHP1B). In conclusion, hypercalcitoninaemia may be encountered in PHP1A and PHP1B even in the absence of thyroid pathology.

Learning points:

  • We describe a novel missense variant c.131T>G p.(Leu44Pro) affecting GNAS exon 1 as the cause of PHP1A.

  • Hypercalcitoninaemia in PHP1A is considered an associated resistance to calcitonin, as suggested by the generalised impairment of Gsα-mediated hormone signalling.

  • GNAS methylation defects, as in type PHP1B, without thyroid pathology can also present with hypercalcitoninaemia.

Open access
Carine Ghassan Richa Rafic Hariri University Hospital, Beirut, Lebanon
Department of Medicine, Endocrinology Division, Lebanese University, Hadath, Lebanon
Endocrinology Department, Rafic Hariri University Hospital, Beirut, Lebanon

Search for other papers by Carine Ghassan Richa in
Google Scholar
PubMed
Close
,
Khadija Jamal Saad Rafic Hariri University Hospital, Beirut, Lebanon
Department of Medicine, Endocrinology Division, Lebanese University, Hadath, Lebanon
Endocrinology Department, Mount Lebanon Hospital, Beirut, Lebanon

Search for other papers by Khadija Jamal Saad in
Google Scholar
PubMed
Close
,
Ali Khaled Chaaban Rafic Hariri University Hospital, Beirut, Lebanon
Department of Radiology, Beirut Governmental University Hospital, Beirut, Lebanon
Diagnostic Radiology, Radiology Department

Search for other papers by Ali Khaled Chaaban in
Google Scholar
PubMed
Close
, and
Mohamad Souheil El Rawas Rafic Hariri University Hospital, Beirut, Lebanon
Department of Medicine, Endocrinology Division, Lebanese University, Hadath, Lebanon
Clinical Endocrinology, Endocrinology Department, Rafic Hariri University Hospital, Beirut, Lebanon

Search for other papers by Mohamad Souheil El Rawas in
Google Scholar
PubMed
Close

Summary

The objective of the study is to report a case of acute pancreatitis secondary to hypercalcemia induced by primary hyperparathyroidism in a pregnant woman at the end of the first trimester. The case included a 32-year-old woman who was diagnosed with acute pancreatitis and severe hypercalcemia refractory to many regimens of medical therapy in the first trimester of pregnancy. She was successfully treated with parathyroidectomy in the early second trimester with complete resolution of hypercalcemia and pancreatitis. Neonatal course was unremarkable. To our best knowledge, this is a rare case when primary hyperparathyroidism and its complications are diagnosed in the first trimester of pregnancy. In conclusion, primary hyperparathyroidism is a rare life-threatening condition to the fetus and mother especially when associated with complications such as pancreatitis. Early therapeutic intervention is important to reduce the morbidity and mortality. Parathyroidectomy performed in the second trimester can be the only solution.

Learning points:

  • Learning how to make diagnosis of primary hyperparathyroidism in a woman during the first trimester of pregnancy.

  • Understanding the complications of hypercalcemia and be aware of the high mortality and sequelae in both fetus and mother.

  • Providing the adequate treatment in such complicated cases with coordinated care between endocrinologists and obstetricians to ensure optimal outcomes.

Open access
Naweed Alzaman Division of Endocrinology, Diabetes, and Metabolism, Tufts Medical Center, 800 Washington Street, Boston, Massachusetts, 02111, USA

Search for other papers by Naweed Alzaman in
Google Scholar
PubMed
Close
,
Anastassios G Pittas Division of Endocrinology, Diabetes, and Metabolism, Tufts Medical Center, 800 Washington Street, Boston, Massachusetts, 02111, USA

Search for other papers by Anastassios G Pittas in
Google Scholar
PubMed
Close
,
Miriam O'Leary Department of Otolaryngology, Tufts Medical Center, 800 Washington Street, Boston, Massachusetts, 02111, USA

Search for other papers by Miriam O'Leary in
Google Scholar
PubMed
Close
, and
Lisa Ceglia Division of Endocrinology, Diabetes, and Metabolism, Tufts Medical Center, 800 Washington Street, Boston, Massachusetts, 02111, USA

Search for other papers by Lisa Ceglia in
Google Scholar
PubMed
Close

Summary

Transient hypocalcemia after thyroidectomy is not uncommon and the risk increases with the extent of neck surgery. We report a case of severe and prolonged hypocalcemia after total thyroidectomy complicated by thoracic duct injury. Hypoparathyroidism and thoracic duct injury are potential complications following total thyroidectomy with extensive lymph node dissection. This case suggested that having both conditions may complicate treatment of hypoparathyroid-induced hypocalcemia by way of losses of calcium and vitamin D in the chyle leak.

Learning points

  • This report highlights chyle leak as an uncommon cause of prolonged hypocalcemia in patients who have undergone extensive neck surgery.

  • Chyle has an electrolyte concentration similar to that of plasma.

  • Medical treatment options for a chyle leak include fat-free oral diet or parenteral nutrition without oral intake, pharmacological treatment (primarily octreotide).

Open access
Jingjing Jiang Department of Endocrinology and Metabolism, Zhongshan Hospital, Fudan University, Shanghai, 200032, People's Republic of China

Search for other papers by Jingjing Jiang in
Google Scholar
PubMed
Close
,
Mei Zhang Department of Endocrinology, The First Affiliated Hospital of Nanjing Medical University, 300 Guangzhou Road, Nanjing, 210029, People's Republic of China

Search for other papers by Mei Zhang in
Google Scholar
PubMed
Close
,
Ronghua He Department of Endocrinology, The First Affiliated Hospital of Nanjing Medical University, 300 Guangzhou Road, Nanjing, 210029, People's Republic of China

Search for other papers by Ronghua He in
Google Scholar
PubMed
Close
,
Meiping Shen Department of General Surgery, The First Affiliated Hospital of Nanjing Medical University, Nanjing, 210029, People's Republic of China

Search for other papers by Meiping Shen in
Google Scholar
PubMed
Close
, and
Wei Liu Department of Nuclear Medicine, The First Affiliated Hospital of Nanjing Medical University, Nanjing, 210029, People's Republic of China

Search for other papers by Wei Liu in
Google Scholar
PubMed
Close

Summary

Functional parathyroid cysts are a rare cause of primary hyperparathyroidism and are often mistaken for thyroid cysts. Systemic lupus erythematosus (SLE) is also a very rare cause of hypercalcemia. We report the case of a 62-year-old woman, who was diagnosed with SLE 30 years ago, presenting with clinical and biochemical features of primary hyperparathyroidism. Laboratory investigation revealed increased serum calcium and parathyroid hormone (PTH) levels; neck ultrasonography (USG) revealed 40×34×26 mm cystic mass in the left lobe of thyroid gland. PTH level in the cysts was >2500 pg/ml, determined by USG-guided fine-needle aspiration (FNA). In this case, no evidence for potential pathogenic association between parathyroid cyst and SLE was uncovered. However, the recognition of this association is very important because the therapeutical strategy is completely different. Operative management is usually straightforward and alleviates symptoms and any biochemical abnormalities caused by the cyst.

Learning points

  • Functional parathyroid cysts are the rare cause of primary hyperparathyroidism and are often mistaken for thyroid cysts.

  • SLE is also a very rare cause of hypercalcemia.

  • Ultrasound-guided FNA of cystic fluid with assay for PTH level is an accurate method of differentiating parathyroid cyst from thyroid cyst.

  • Appropriate management of functional parathyroid cysts is surgical excision.

Open access