PMID- 18414463
OWN - NLM
STAT- MEDLINE
DCOM- 20080701
LR  - 20181201
IS  - 1745-8374 (Electronic)
IS  - 1745-8366 (Linking)
VI  - 4
IP  - 6
DP  - 2008 Jun
TI  - A patient with MEN1-associated hyperparathyroidism, responsive to cinacalcet.
PG  - 351-7
LID - 10.1038/ncpendmet0816 [doi]
AB  - BACKGROUND: A 30-year-old woman with suspected multiple endocrine neoplasia type 
      1 (MEN1) was referred to our center in 2001 with primary hyperparathyroidism 
      caused by a multiglandular parathyroid adenoma. The patient also had 
      hyperprolactinemia caused by an anterior pituitary macroadenoma. The patient 
      underwent a parathyroidectomy with autotransplantation of parathyroid fragments 
      into the nondominant forearm, resulting in resolution of the primary 
      hyperparathyroidism. MEN1 was confirmed by analysis of the MEN1 gene, which 
      revealed a 1555insG frameshift mutation. In 2006 serum calcium and parathyroid 
      hormone (PTH) levels were again found to be high. INVESTIGATIONS: After 
      parathyroidectomy in 2001, the patient underwent regular measurements of PTH 
      levels from both forearms, of serum calcium, prolactin and phosphate levels, and 
      of urinary calcium and phosphate levels. When serum calcium and PTH levels were 
      found to be elevated in 2006, circulating PTH levels were similar in both 
      forearms. Ultrasound scan and technetium-99m-labeled 
      hexakis-2-methoxyisobutylisonitrile ((99m)Tc MIBI) scintigraphy evidenced a 
      metabolically active parathyroid nodule in the neck. DIAGNOSIS: Local recurrence 
      of a parathyroid adenoma associated with MEN1. MANAGEMENT: Because the patient 
      refused a further operation, we decided to initiate pharmacological treatment 
      with cinacalcet. After 1 month of therapy, serum calcium and PTH levels returned 
      to normal. The patient has now been closely monitored for 1 year. During this 
      time calcium and PTH levels remained normal, morphologically the parathyroid 
      nodular lesion remained unchanged and cinacalcet was well tolerated without the 
      occurrence of adverse events. Cinacalcet could represent an important 
      pharmacological intervention in MEN1-associated primary hyperparathyroidism 
      before surgery and in postsurgical recurrences.
FAU - Falchetti, Alberto
AU  - Falchetti A
AD  - University Hospital of Careggi, Florence, Italy.
FAU - Cilotti, Antonio
AU  - Cilotti A
FAU - Vaggelli, Luca
AU  - Vaggelli L
FAU - Masi, Laura
AU  - Masi L
FAU - Amedei, Antonietta
AU  - Amedei A
FAU - Cioppi, Federica
AU  - Cioppi F
FAU - Tonelli, Francesco
AU  - Tonelli F
FAU - Brandi, Maria Luisa
AU  - Brandi ML
LA  - eng
PT  - Case Reports
PT  - Journal Article
PT  - Research Support, Non-U.S. Gov't
DEP - 20080415
PL  - England
TA  - Nat Clin Pract Endocrinol Metab
JT  - Nature clinical practice. Endocrinology & metabolism
JID - 101261798
RN  - 0 (Naphthalenes)
RN  - 0 (Parathyroid Hormone)
RN  - SY7Q814VUP (Calcium)
RN  - UAZ6V7728S (Cinacalcet)
SB  - IM
EIN - Nat Clin Pract Endocrinol Metab. 2008 Jul;4(7):420. Vagelli, Luca [corrected to 
      Vaggelli, Luca]
MH  - Adult
MH  - Calcium/blood
MH  - Cinacalcet
MH  - Female
MH  - Humans
MH  - Hyperparathyroidism, Primary/blood/*drug therapy
MH  - Multiple Endocrine Neoplasia Type 1/*complications
MH  - Naphthalenes/*therapeutic use
MH  - Parathyroid Hormone/blood
EDAT- 2008/04/17 09:00
MHDA- 2008/07/02 09:00
CRDT- 2008/04/17 09:00
PHST- 2007/12/19 00:00 [received]
PHST- 2008/03/04 00:00 [accepted]
PHST- 2008/04/17 09:00 [pubmed]
PHST- 2008/07/02 09:00 [medline]
PHST- 2008/04/17 09:00 [entrez]
AID - ncpendmet0816 [pii]
AID - 10.1038/ncpendmet0816 [doi]
PST - ppublish
SO  - Nat Clin Pract Endocrinol Metab. 2008 Jun;4(6):351-7. doi: 10.1038/ncpendmet0816. 
      Epub 2008 Apr 15.