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Familial hypoparathyroidism

MedGen UID:
322005
Concept ID:
C1832648
Disease or Syndrome
Synonyms: CASR-Related Familial Isolated Hypoparathyroidism; Familial isolated hypoparathyroidism; GCM2-Related Familial Isolated Hypoparathyroidism; PTH-Related Familial Isolated Hypoparathyroidism
SNOMED CT: Familial isolated hypoparathyroidism (725036000)
Modes of inheritance:
Autosomal recessive inheritance
MedGen UID:
141025
Concept ID:
C0441748
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in individuals with two pathogenic alleles, either homozygotes (two copies of the same mutant allele) or compound heterozygotes (whereby each copy of a gene has a distinct mutant allele).
Autosomal dominant inheritance
MedGen UID:
141047
Concept ID:
C0443147
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in heterozygotes. In the context of medical genetics, an autosomal dominant disorder is caused when a single copy of the mutant allele is present. Males and females are affected equally, and can both transmit the disorder with a risk of 50% for each child of inheriting the mutant allele.
X-linked recessive inheritance
MedGen UID:
375779
Concept ID:
C1845977
Finding
Source: Orphanet
A mode of inheritance that is observed for recessive traits related to a gene encoded on the X chromosome. In the context of medical genetics, X-linked recessive disorders manifest in males (who have one copy of the X chromosome and are thus hemizygotes), but generally not in female heterozygotes who have one mutant and one normal allele.
 
Monarch Initiative: MONDO:0016390
OMIM® Phenotypic series: PS146200
Orphanet: ORPHA2238

Definition

A rare heterogeneous group of metabolic disorders with abnormal calcium metabolism due to deficient secretion of parathormone (PTH) without other endocrine disorders or developmental defects. It can occur at any age (from the newborn period to adulthood) but generally starts within the first decade of life. The disease may be due to an activating mutation of the calcium-sensing receptor (CASR) gene. This is the most common genetic cause and is transmitted as an autosomal dominant trait. It represents 42% of isolated hypoparathyroidism cases. Thirteen mutations have been described in familial or sporadic cases. In three families, mutations in the PTH gene have been identified. One family has been reported with a mutation in the gene encoding the glial cells missing homolog b (GCMB) transcription factor. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVFamilial hypoparathyroidism
Follow this link to review classifications for Familial hypoparathyroidism in Orphanet.

Professional guidelines

Recent clinical studies

Etiology

Stock JL, Brown RS, Baron J, Coderre JA, Mancilla E, De Luca F, Ray K, Mericq MV
J Clin Endocrinol Metab 1999 Sep;84(9):3036-40. doi: 10.1210/jcem.84.9.5977. PMID: 10487661

Diagnosis

Mannstadt M, Cianferotti L, Gafni RI, Giusti F, Kemp EH, Koch CA, Roszko KL, Yao L, Guyatt GH, Thakker RV, Xia W, Brandi ML
J Bone Miner Res 2022 Dec;37(12):2615-2629. Epub 2022 Nov 14 doi: 10.1002/jbmr.4667. PMID: 36375809
Gild ML, Bullock M, Luxford C, Field M, Clifton-Bligh RJ
J Clin Endocrinol Metab 2020 Jul 1;105(7) doi: 10.1210/clinem/dgaa279. PMID: 32421798
Pillar N, Pleniceanu O, Fang M, Ziv L, Lahav E, Botchan S, Cheng L, Dekel B, Shomron N
Hum Genet 2017 Jul;136(7):835-845. Epub 2017 Apr 25 doi: 10.1007/s00439-017-1804-9. PMID: 28444561Free PMC Article
Meena RL, Maloo SK, Samar N, Ruhela A, Saini S
J Assoc Physicians India 2015 Jun;63(6):73-5. PMID: 26710407
Bandyopadhyay SK, Moulick A, Chakrabarti N, Dutta A
J Assoc Physicians India 2010 Feb;58:115-7. PMID: 20653155

Therapy

Mukhtar N, Alghamdi B, Alswailem M, Alsagheir A, Alzahrani AS
Front Endocrinol (Lausanne) 2024;15:1415639. Epub 2024 Oct 7 doi: 10.3389/fendo.2024.1415639. PMID: 39435356Free PMC Article
Angurana SK, Muralidharan J, Dayal D, Ismail J
Indian J Pediatr 2017 May;84(5):405-407. Epub 2017 Feb 4 doi: 10.1007/s12098-017-2295-3. PMID: 28160154
Bandyopadhyay SK, Moulick A, Chakrabarti N, Dutta A
J Assoc Physicians India 2010 Feb;58:115-7. PMID: 20653155
Tank SJ, Malik S, Joshi SM, Gaval MA, Varthakavi P
Indian Pediatr 1999 Jul;36(7):714-7. PMID: 10740313
Richter PL, Chutorian AM
Neurology 1968 Jan;18(1 Pt 1):75-80. doi: 10.1212/wnl.18.1_part_1.75. PMID: 5688796

Prognosis

Mannstadt M, Cianferotti L, Gafni RI, Giusti F, Kemp EH, Koch CA, Roszko KL, Yao L, Guyatt GH, Thakker RV, Xia W, Brandi ML
J Bone Miner Res 2022 Dec;37(12):2615-2629. Epub 2022 Nov 14 doi: 10.1002/jbmr.4667. PMID: 36375809
Cranston T, Boon H, Olesen MK, Ryan FJ, Shears D, London R, Rostom H, Elajnaf T, Thakker RV, Hannan FM
Eur J Endocrinol 2022 May 24;187(1):111-122. doi: 10.1530/EJE-21-0730. PMID: 35521792Free PMC Article
Gild ML, Bullock M, Luxford C, Field M, Clifton-Bligh RJ
J Clin Endocrinol Metab 2020 Jul 1;105(7) doi: 10.1210/clinem/dgaa279. PMID: 32421798
Chiu WY, Chen HW, Chao HW, Yann LT, Tsai KS
J Clin Endocrinol Metab 2006 Nov;91(11):4587-92. Epub 2006 Aug 15 doi: 10.1210/jc.2006-0864. PMID: 16912130

Clinical prediction guides

Mannstadt M, Cianferotti L, Gafni RI, Giusti F, Kemp EH, Koch CA, Roszko KL, Yao L, Guyatt GH, Thakker RV, Xia W, Brandi ML
J Bone Miner Res 2022 Dec;37(12):2615-2629. Epub 2022 Nov 14 doi: 10.1002/jbmr.4667. PMID: 36375809
Cranston T, Boon H, Olesen MK, Ryan FJ, Shears D, London R, Rostom H, Elajnaf T, Thakker RV, Hannan FM
Eur J Endocrinol 2022 May 24;187(1):111-122. doi: 10.1530/EJE-21-0730. PMID: 35521792Free PMC Article
Gild ML, Bullock M, Luxford C, Field M, Clifton-Bligh RJ
J Clin Endocrinol Metab 2020 Jul 1;105(7) doi: 10.1210/clinem/dgaa279. PMID: 32421798
Pillar N, Pleniceanu O, Fang M, Ziv L, Lahav E, Botchan S, Cheng L, Dekel B, Shomron N
Hum Genet 2017 Jul;136(7):835-845. Epub 2017 Apr 25 doi: 10.1007/s00439-017-1804-9. PMID: 28444561Free PMC Article
Sarin R, Tomar N, Ray D, Gupta N, Sharma YD, Goswami R
Clin Endocrinol (Oxf) 2006 Sep;65(3):359-63. doi: 10.1111/j.1365-2265.2006.02604.x. PMID: 16918956

Recent systematic reviews

Mannstadt M, Cianferotti L, Gafni RI, Giusti F, Kemp EH, Koch CA, Roszko KL, Yao L, Guyatt GH, Thakker RV, Xia W, Brandi ML
J Bone Miner Res 2022 Dec;37(12):2615-2629. Epub 2022 Nov 14 doi: 10.1002/jbmr.4667. PMID: 36375809

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