bluuebunny/crossref_metadata_2025_split huggingface dataset:
Calciphylaxis in Association With Hungry Bone Syndrome. A Possible Risk Factor?
Authors: Arreaza Javier , Kim Ga Hee , Kim Andrew J , Dulal Jeewendra , Goulden Peter Anthony 
Published: May 2021

Abstract
Background:
Calciphylaxis (calcific uremic arteriolopathy) is a rare complication seen in, although not limited to, patients with end-stage renal disease (ESRD). The abnormal regulation of calcium (Ca) and phosphorus (P) homeostasis in this patient group results in intravascular Ca deposition. These patients often develop secondary/tertiary hyperparathyroidism, presenting unique treatment challenges. When patients do not respond to medical therapy, parathyroidectomy is an option that may be complicated by hungry bone syndrome (HBS). We present a case of a patient with calciphylaxis with HBS post-parathyroidectomy.
Case Report:
The patient is a 41 y.o. male with ESRD on hemodialysis who presented with lower extremity ulcers complicated by calciphylaxis. On admission, the PTH was elevated at 2200 U with a normal corrected Ca 8.7 and P 8.5. He was found to have a non-displaced pathologic fracture of the right femoral neck. CT scan of the neck revealed nodular parathyroid hyperplasia affecting all four glands. His hyperparathyroidism was managed medically with cinacalcet, phosphate binder, and sodium thiosulfate to optimize his condition before surgery.
He underwent a subtotal parathyroidectomy, with post-op course complicated by HBS. Repeat labs showed PTH 444, P 6.1, corrected Ca 7.4, and ionized Ca &lt;4. Despite frequent repletion with IV Ca and addition of calcitriol, he remained persistently hypocalcemic with symptoms (paresthesia, perioral numbness) and prolonged QTc (560 ms) on 12-lead EKG. After weighing the more imminent risk of unstable arrhythmia versus exacerbation of HBS by targeting too high of a Ca level post-op, ICU admission was decided where Ca levels could be more closely monitored and titrated. The patient eventually demonstrated an improvement in his Ca level and symptoms.
Discussion:
This patient with ESRD complicated by hyperparathyroidism presented with ulcers and calciphylaxis refractory to medical therapy, requiring parathyroidectomy. HBS was a concern in this particular patient given his risk factors.
Hypocalcemia is an expected electrolyte imbalance that usually resolves within 2–4 days after parathyroidectomy. Severe and persistent hypocalcemia on day 4 after the procedure should raise concern for HBS. It has been shown that the risk is higher in patients who have secondary hyperparathyroidism, prolonged duration of elevated PTH, age &gt;60, and radiologic evidence of bone disease.
ConclusionCalciphylaxis is an uncommon but potentially fatal illness seen in ESRD patients associated with Ca and P abnormalities. Whether this may increase the risk of HBS status-post parathyroidectomy is unknown; however, HBS requiring ICU management to treat refractory hypocalcemia has been reported in this group1. It is arguable that calciphylaxis may serve to identify such patients.
(1) Hassanein M, et al. BMJ Case Rep 2018;11:e226696. doi:10.1136/bcr-2018–226696
Calcitriol Induced Hyper Calcemia in Stroma Uterine Cancer Independent From PTHrp
Authors: Alam Mustafa , Lababidi Hashem , Mahad Alam , Horani Mohamad Hosam 
Published: May 2021

Abstract
Case Presentation: A 70-year-old female with a history of breast cancer presented with an intra-abdominal mass. The biopsy of the mass was positive for endometrial stromal sarcoma. During hospital admission, the patient was noted to have hypercalcemia attributable to her malignancy. This hypercalcemia responded positively with IV fluids with calcitonin, and the patient was discharged with oncology follow-up for chemotherapy. The patient was referred back upon repeat labs by oncology showing a calcium of 13.4 mg/dL. The physical exam at this time was unremarkable. However, labs were significant for a RDW of 16.7%, Monocyte count of 17.7 mcg/L, Creatinine of 1.24 mg/dL, Calcium of 12.8 mg/dL, and Globulin of 4.2 g/dL. Special chemistry tests came back significant for a 1,25 Dihydroxy Vitamin D level of 256.8 pg/mL, PTHrP level of 3.7 pmol/L, PTH of 18.6 pg/mL, and free kappa light chains of 47.18 mg/dL. The patient was then placed on Prednisone 20mg daily with Zometa as needed with GYN oncology follow-up.
Discussion: Paraneoplastic hypercalcemia is a relatively common finding in malignancy, affecting up to 44% of patients (Mirrakhimov, 2015). However, the etiology behind hypercalcemia ranges from increased PTHrP, osteolytic metastases, parathyroid related tumors, and uncommonly from 1,25 Hydroxy Vitamin D (Mirrakhimov, 2015). The patient presented with hypercalcemia secondary to an endometrial stromal sarcoma. The hypercalcemia was presumed to be related to PTHrP, however, serum levels of PTHrP and PTH were at the upper limits of normal, thus not sufficient to explain hypercalcemia. The elevated Vitamin D level was suspect for a calcitriol mediated hypercalcemia, a rare finding with a few case reports relating to gastrointestinal stromal tumors (Betlachin, Kim, & Oxman, 2020). The diagnosis is further backed up by the patient’s response to prednisone, which reduces 1 alpha hydroxylase activity, reducing the calcium levels. Calcitriol mediated hypercalcemia is a phenomenon that is typical of certain granulomatous diseases, but not as well defined in the literature for cancer (Kallas et al., 2010). Thus, it is important to consider calcitriol related hypercalcemia in malignancies with reduced PTH or PTHrP for proper treatment.
Mirrakhimov A. E. (2015). Hypercalcemia of Malignancy: An Update on Pathogenesis and Management. North American journal of medical sciences, 7(11), 483
Anna Betlachin, MD, Sarah Sangnim Rhee Kim, MD, Rachael Oxman, MD, MPH, Calcitriol-Mediated Hypercalcemia in a Patient With Metastatic Gastrointestinal Stromal Tumor, Journal of the Endocrine Society, Volume 4, Issue Supplement_1, April-May 2020,
Melissa Kallas, Francis Green, Martin Hewison, Christopher White, Gregory Kline, Rare Causes of Calcitriol-Mediated Hypercalcemia: A Case Report and Literature Review, The Journal of Clinical Endocrinology & Metabolism, Volume 95, Issue 7, 1 July 2010, Pages 3111.
Clinical Presentation and Management Approach in a Case of Familial Hypocalciuric Hypercalcemia Type 3 Due to APS21 Gene Mutation
Authors: Elsheikh Sahar A , Blunk Henry M , Wilhelm Scott 
Published: May 2021

Abstract
Familial hypocalciuric hypercalcemia (FHH) is a genetic disorder caused by dysfunctional calcium homeostasis. Thus far, three types of FHH are known to be caused by mutations inCASR (FHH1), GNA11 (FHH2), and AP2S1 (FHH3). The patient in this case report is a 36-year old male that initially presented for a second opinion after being diagnosed with Primary Hyperparathyroidism(PHPT) with subsequent parathyroidectomy done at another institute, and developed recurrent symptomatic hypercalcemia. Prior to considering this patient for further surgical options, he underwent genetic testing, which revealed he had c.43C&gt;T (p.Arg15Cys) mutation in the AP2S1 gene diagnostic of Familial Hypocalciuric Hypercalcemia Type 3 (FHH3). The patient’s father and sister also have hypercalcemia, and have been offered genetic testing. There have been cases reported of patients with FHH3 that have symptomatic hypercalcemia and that have associated cognitive issues. Many patients with FHH can be misdiagnosed and may undergo unnecessary parathyroidectomy. This case report further elucidates the need to raise awareness of FHH.
Extreme Secretion of PTHrP From a Pancreatic Neuroendocrine Tumor, A Shift From Severe Hypercalcemia to Hypocalcemia
Authors: Nachawi Noura , Paredes Stephania , Seibert Stephen , McShane Adam , Pantalone Kevin M 
Published: May 2021

Abstract
Background: PTHrP-secreting pancreatic neuroendocrine tumors (PNET) are a recognized cause of malignancy associated hypercalcemia. Herein, we report a case of severe hypercalcemia due to an extreme elevation of PTHrP from a PNET, where after treatment of the hypercalcemia, symptomatic hypocalcemia occurred. Clinical Case: A 59 year-old-woman with a recurrent PNET with liver metastases undergoing an evaluation for multi-visceral transplant presented with acute confusion, nausea and vomiting. Diagnostic testing identified an extreme elevation of total calcium (Ca) [&gt;20.1 mg/dL (8.5 - 10.2)] from two different samples [serum albumin 4.1 g/dL (3.9 - 4.9)]. The total Ca level one month earlier was 8.3 mg/dL with a serum albumin of 3.1 g/dL. Total Ca measurements were performed with the Ca Gen.2 assay on a cobas c702 chemistry analyzer (Roche Diagnostics). Results greater than the analytical measurement range (0.8 – 20.1 mg/dL) were diluted with saline and confirmed (22.6 mg/dL). A Radiometer ABL 800 Flex blood gas analyzer was used to determine the ionized Ca concentration [2.94 mmol/L (1.08 - 1.30)]. Upon presentation the serum creatinine (Cr) was 2.07 mg/dL (0.58 - 0.96); eGFR utilizing the MDRD equation 24 mL/min/1.73m2; baseline serum Cr 0.78 mg/dL. Her serum 25-OH vitamin D was 31 ng/mL (31.0 - 80.0), PTH 12 pg/mL (15 - 65), phosphate 4.3 mg/dL (2.7 - 4.8) and 1, 25-OH vitamin D 39.1 (15.0 - 60.0). PTHrP measurements were performed by ARUP Laboratories via liquid chromatography tandem mass spectrometry (LC-MS/MS) and resulted in a reported value of &gt;2500 pmol/L (0.0 - 3.4). Her symptoms resolved and the corrected Ca gradually decreased to 8 mg/dL after treatment with IV fluids, calcitonin 200 units sc every 12 hours for 48 hours, 60 mg IV pamidronate, and five sessions of hemodialysis. Within thirteen days of receiving pamidronate, her corrected Ca slowly increased to 12mg/dL; thus, she received a single dose of 120 mg sc denosumab. Nine days later, the patient developed symptomatic hypocalcemia (7.3 mg/dL) manifested by paresthesia in the hands and feet and perioral numbness. She then received multiple doses of oral and intravenous Ca along with 50,000 units of oral ergocalciferol twice weekly. The corrected Ca normalized (8.1 mg/dL) and symptoms resolved. The patient was discharged with plans for future treatment of her underlying malignancy. Conclusion: This is the first report of a PNET producing an extreme elevation of PTHrP of higher than 2500 pmol/L, resulting in a concordant extreme elevation of total calcium within a month of documented normocalcemia. Treatment of hypercalcemia with denosumab may result in the development of hypocalcemia requiring treatment.
Hypercalcemia- A Common Abnormality With Uncommon Etiologies
Authors: Kapoor Ashutosh , Basu Ambar 
Published: May 2021

Abstract
Introduction: Hypercalcemia is a relatively common electrolyte abnormality which is generally associated with Parathyroid related disorders or Cancers. Rare aetiologies comprise of granulomatous disorders and haematological malignancies namely Sarcoidosis and Lymphoma. Parathyroid hormone related Peptide (PTH-rp) and Calcitriol are implicated in causing Hypercalcemia in mentioned aetiologies.
Case Details: A 57 year gentleman was admitted following a 10 day history of feeling unwell, unsteadiness in gait, constipation and mild shortness of breath (SOB) on walking uphill. His medical history comprised of Type 1 Diabetes Mellitus (DM) and Hypertension (HTN). Regular medications did not comprise of any drugs that could lead to Hypercalcemia. Biochemic he was Hypercalcemic at 3.14 (2.20–2.60). Parathyroid Hormone levels (PTH) was 0.6 (1.1–4.7 pmol/l) which excluded Parathyroid driven aetiologies. ACE levels were normal and Myeloma screen was negative. 25-OH Vitamin D levels were within Normal limits. Management: Hypercalcemia was managed with Intravenous Fluids and optimum hydration which improved his Calcium levels, followed by subsequent normalisation. Given his suppressed PTH levels, he underwent whole body imaging to assess for evidence of underlying occult malignancy. The CT imaging of his chest, abdomen and pelvis revealed widespread Lymphadenopathy and hypodense lesions in the Spleen. The report stated likely Sarcoidosis accompanied by Splenic involvement, with the main differentials being Sarcoidois versus Lymphoproliferative disorder. This was discussed with the respiratory team and Steroids were not commenced taking into account the improvement in his Calcium levels following adequate hydration and fluids. The patient has been kept under close follow up with ongoing monitoring of his clinical status.
Summary: Hypercalcemia has been described in patients with Granulomatous disorders and Haematological malignancies. The incidence of hypercalcemia in Sarcoidosis is estimated to be approximately 10%, with no association with a specific demographic. Granulomatous disorders and Haematological malignancies constitute the rare aetiologies of hypercalcemia and are characterised by a low-normal or suppressed PTH level. Diagnosis of Sarcoidosis is often challenging with the lack of gold standard tests. Serum ACE levels can be within normal limits in people with Sarcoidosis despite not being on Steroid therapy. Evaluation of serum PTH-rp and Calcitriol can aid in diagnosis and more importantly assisting in prognostication, thus guiding further management.
Intravenous Iron Induced Severe Hypophosphatemia
Authors: Guo Rong R , Sunny Sonie S , Correa Ricardo R 
Published: May 2021

Abstract
Introduction: Hypophosphatemia is a recently recognized adverse effect of certain intravenous iron formulations. It was previously thought to be asymptomatic and transient, however it can cause severe hypophosphatemia associated with renal phosphate wasting. We present a case of severe hypophosphatemia following intravenous ferric carboxymaltose administration.
Clinical Case: A 40-year-old female with history of iron deficiency anemia, Hashimoto’s hypothyroidism and multinodular goiter status post thyroidectomy and obesity status post gastric sleeve presents to the hospital due to dizziness, generalized weakness, bone pain and severe myalgia right after 2nd dose of 750 mg of ferric carboxymaltose administration. She received 1st dose of 750 mg of ferric carboxymaltose a week ago prior to the presentation. She was found to have severe hypophosphatemia with a phosphorus level 1.0 mg/dl (2.4 - 4.8 mg/dl), hypocalcemia with calcium level at 8.7 mg/dl, normal PTH at 40 pg/ml and normal 25-OH Vitamin D level at 59.1 ng/ml. The fractional excretion of filtered phosphate (FEPO4) was 19.5%, supporting renal phosphate wasting. She was hospitalized for 5 days and received intravenous phosphate 45 mmol on hospitalization day 1, 15 mmol on day 2 and 30 mmol on day 3. She was discharged on K-phos Neutral 250 mg TID, Calcium carbonate/Vitamin D3 600 mg/500 IU 2 tablets TID and Calcitriol 0.5 mcg daily. While she was taking K-phos Neutral, Calcitriol and Calcium carbonate/Vitamin D3, her phosphorous level was down to 1.3 mg/dl, which was 4 weeks after 1st dose of ferric carboxymatose administration. She presented to the ED and received intravenous phosphate 15 mmol. She is currently taking K-phos Neutral 250 mg QID, Calcitriol 0.5 mcg daily and Calcium carbonate/Vitamin D3 600 mg/500 IU 2 tablets TID. Her phosphorous levels ranged at 2.5 - 3.0 mg/dl. Her bone pain and myalgia have gradually improved.
Conclusion: Clinician should be aware of the side effect of hypophosphatemia following ferric carboxymaltose administration. Recent studies showed that incidence of hypophosphatemia associated with intravenous iron administration was significantly higher in ferric carboxymatose group compared with either iron isomaltoside group or ferumoxytol group. The impact of severe hypophosphatemia should be considered when choosing an intravenous iron medication.
Isolated Hepatic Sarcoidosis Presenting With Severe Hypercalcemia
Authors: Antony Mc Anto , Verma Vipin , Bello Diego , Surabhi Ritika R , Kant Ravi 
Published: May 2021

Abstract
Introduction:
Sarcoidosis is an inflammatory disorder of unknown etiology that can affect various organs. Lungs, intra thoracic lymph nodes and skin are the most commonly affected organs. The prevalence of hepatic sarcoidosis ranges between 5 to 30%. However, isolated hepatic sarcoidosis is rare. Hypercalcemia in sarcoidosis varies considerably due to the varying disease course and is reported to occur between 2 to 63% cases. We report a unique care of isolated liver sarcoidosis that presented with severe parathyroid hormone (PTH) independent hypercalcemia.
Case:
A 58 years old woman of Asian ethnicity with a past medical history of type 1 diabetes, hypothyroidism and chronic kidney disease presented to emergency department with headaches and altered mental status. The headaches were present since 4 weeks and was associated with polyuria and polydipsia. Non contrast CT scan of brain was negative for acute intracranial process. Physical examination revealed unremarkable vital signs and physical findings except for the altered mental status on neurological exam (orientation to self only). Initial laboratory testing revealed high corrected serum calcium 12.2 (8.0- 10.1 mg/dl), acute renal failure with high serum creatinine 2.90 (0.57–1.0 mg/dl), abnormal liver panel with elevated AST 84 (5-32U/L), ALT 85 (5–33 U/L), Alkaline Phosphatase 151 (35-104U/L). Repeat corrected serum calcium was still high at 12.4 mg/dl which prompted further evaluation to search for the etiology of hypercalcemia and testing revealed 1, 25 dihydroxy vitamin D (1, 25 vit D) mediated hypercalcemia. Labs showed low PTH 14.6 (15–65 pg/ml), normal serum protein electrophoresis, low PTH-related peptide &lt;2.0 pmol/L, low 25 hydroxy vitamin D 20 (30–100 ng/ml) and high 1, 25 vit D 96.5 (19.9–79.3 pg/ml). Imaging evaluation revealed multiple hypodense nodules in both lobes of the liver seen on ultrasound, and CT scan of chest, abdomen and pelvis was unremarkable except for the similar liver findings. Biopsy of the liver lesion revealed non-caseating granulomas with no evidence for lymphoma and negative for acid fast bacteria and fungal organisms. Patient was treated with intravenous fluids, zoledronic acid and initiated on a course on oral prednisone that was tapered over a period of 6 months. There was a tremendous improvement in overall clinical condition. Serum calcium and 1, 25 vit D levels normalized. CT scan performed 3 months later showed complete resolution of all liver lesions.
Conclusion:
While most cases of hepatic sarcoidosis are asymptomatic and are incidentally found due to abnormal liver function tests or imaging done for other causes, we present a case of isolated hepatic sarcoidosis diagnosed due to symptomatic severe PTH independent hypercalcemia. Even though rare, extra-pulmonary sarcoidosis should be in differentials for 1, 25 vit D mediated hypercalcemia even if thoracic imaging are unremarkable.
Late-Onset Neonatal Hypocalcemia Due to Transient Hypoparathyroidism in Infant of Mother With COVID-19 at Delivery
Authors: Grundman Jody B , Persky Rebecca W , Shankar Roopa Kanakatti 
Published: May 2021

Abstract
Introduction: Neonatal hypocalcemia is an uncommon condition but clinical presentation can include severe manifestations such as neuromuscular irritability, tetany, seizure or cardiac conduction abnormalities that require prompt intervention to normalize calcium levels. To our knowledge there have been no reports of neonatal hypocalcemia following maternal SARS-CoV2 infection at the time of birth. Here we report a case of transient late-onset neonatal hypocalcemia complicated by maternal SARS-CoV-2 infection at the time of delivery.
Clinical Case: A 13-day old full-term appropriate for gestational age female was born to an asymptomatic mother who tested positive for SARS-CoV2 at the time of delivery. Caretakers noticed minor twitching movements in the first few days of life that were progressively worsening. Initial ionized calcium was 0.6 mmol/L, and labs at time of transfer to our hospital were notable for total calcium of 5.5mg/dL(n 8.9–9.9), and ionized calcium of 0.67mmol/L (n 1.12–1.37 mmol/L). Calcium levels improved after she received IV calcium boluses and was started on continuous IV calcium infusion. Initial phosphorus was 8.3 mg/dL (n 3.2–7.4 mg/dL) and magnesium was 1.2 mg/dL (n 1.5–2.2 mg/dL), while intact PTH was inappropriately low at 12.2 pg/mL (n 10–65 pg/mL), and urine calcium to urine creatinine ratio was below the limits of assay detection (n &lt;0.86), consistent with a diagnosis of neonatal hypoparathyroidism. The 25-hydroxy vitamin D was 11.1 ng/mL (n 30–100 ng/mL), which may have also been a contributing factor. She had no dysmorphic features on examination concerning for 22q deletion syndrome, and tested negative for SARS-CoV2. Chromosomal microarray did not reveal clinically relevant copy number alterations or areas of homozygosity. Calcium stabilized on enteral calcium carbonate, calcitriol, and cholecalciferol, and she was weaned off of all supplementation. Prior to discharge PTH recovered to 36.9 pg/ml with a calcium of 9.9 mg/dL, phosphorus 7.7 mg/dL, normal magnesium and 25-hydroxy vitamin D (31.3 ng/ml). She was discharged on Similac PM 60/40 formula to reduce her dietary phosphorus content, and 400 IU of cholecalciferol.
Conclusions: Hypocalcemia in patients with severe COVID-19 is being increasingly reported. There is minimal data on the effect of perinatal SARS-CoV2 infections on neonatal health. While this report does not establish causation, expanding awareness of neonatal abnormalities following maternal SARS-CoV2 infection at delivery will help to recognize causal associations and improve patient care.