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Osteopetrosis with renal tubular acidosis(OPTB3)

MedGen UID:
91042
Concept ID:
C0345407
Disease or Syndrome
Synonyms: Autosomal recessive osteopetrosis type 3; Carbonic anhydrase 2 deficiency; Guibaud Vainsel syndrome; Marble brain disease; OPTB3; Osteopetrosis, autosomal recessive 3, with renal tubular acidosis
SNOMED CT: Carbonic anhydrase II deficiency (254122007); CA2 - Osteopetrosis with renal tubular acidosis (254122007); Osteopetrosis with renal tubular acidosis (254122007)
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).
 
Gene (location): CA2 (8q21.2)
 
Monarch Initiative: MONDO:0009818
OMIM®: 259730
Orphanet: ORPHA2785

Definition

Osteopetrosis is a bone disease that makes bone tissue abnormally compact and dense and also prone to breakage (fracture). Researchers have described several major types of osteopetrosis, which are usually distinguished by their pattern of inheritance: autosomal dominant or autosomal recessive. The different types of the disorder can also be distinguished by the severity of their signs and symptoms.

Autosomal dominant osteopetrosis (ADO), which is also called Albers-Schönberg disease, is typically the mildest type of the disorder. Some affected individuals have no symptoms. In affected people with no symptoms, the unusually dense bones may be discovered by accident when an x-ray is done for another reason. 

In individuals with ADO who develop signs and symptoms, the major features of the condition include multiple bone fractures after minor injury, abnormal side-to-side curvature of the spine (scoliosis) or other spinal abnormalities, arthritis in the hips, and a bone infection called osteomyelitis. These problems usually become apparent in late childhood or adolescence.

Autosomal recessive osteopetrosis (ARO) is a more severe form of the disorder that becomes apparent in early infancy. Affected individuals have a high risk of bone fracture resulting from seemingly minor bumps and falls. Their abnormally dense skull bones pinch nerves in the head and face (cranial nerves), often resulting in vision loss, hearing loss, and paralysis of facial muscles. Dense bones can also impair the function of bone marrow, preventing it from producing new blood cells and immune system cells. As a result, people with severe osteopetrosis are at risk of abnormal bleeding, a shortage of red blood cells (anemia), and recurrent infections. In the most severe cases, these bone marrow abnormalities can be life-threatening in infancy or early childhood.

Other features of autosomal recessive osteopetrosis can include slow growth and short stature, dental abnormalities, and an enlarged liver and spleen (hepatosplenomegaly). Depending on the genetic changes involved, people with severe osteopetrosis can also have brain abnormalities, intellectual disability, or recurrent seizures (epilepsy).

A few individuals have been diagnosed with intermediate autosomal osteopetrosis (IAO), a form of the disorder that can have either an autosomal dominant or an autosomal recessive pattern of inheritance. The signs and symptoms of this condition become noticeable in childhood and include an increased risk of bone fracture and anemia. People with this form of the disorder typically do not have life-threatening bone marrow abnormalities. However, some affected individuals have had abnormal calcium deposits (calcifications) in the brain, intellectual disability, and a form of kidney disease called renal tubular acidosis. [from MedlinePlus Genetics]

Clinical features

From HPO
Short stature
MedGen UID:
87607
Concept ID:
C0349588
Finding
A height below that which is expected according to age and gender norms. Although there is no universally accepted definition of short stature, many refer to "short stature" as height more than 2 standard deviations below the mean for age and gender (or below the 3rd percentile for age and gender dependent norms).
Hepatosplenomegaly
MedGen UID:
9225
Concept ID:
C0019214
Sign or Symptom
Simultaneous enlargement of the liver and spleen.
Optic nerve compression
MedGen UID:
78765
Concept ID:
C0271344
Disease or Syndrome
Intellectual disability
MedGen UID:
811461
Concept ID:
C3714756
Mental or Behavioral Dysfunction
Intellectual disability, previously referred to as mental retardation, is characterized by subnormal intellectual functioning that occurs during the developmental period. It is defined by an IQ score below 70.
Periodic hypokalemic paresis
MedGen UID:
867195
Concept ID:
C4021553
Disease or Syndrome
Episodes of muscle weakness associated with reduced levels of potassium in the blood.
Anemia
MedGen UID:
1526
Concept ID:
C0002871
Disease or Syndrome
A reduction in erythrocytes volume or hemoglobin concentration.
Extramedullary hematopoiesis
MedGen UID:
392862
Concept ID:
C2613439
Disease or Syndrome
The process of hematopoiesis occurring outside of the bone marrow (in the liver, thymus, and spleen) in the postnatal organisms.
Osteopetrosis
MedGen UID:
18223
Concept ID:
C0029454
Finding
Abnormally increased formation of dense trabecular bone tissue. Despite the increased density of bone tissue, osteopetrotic bones tend to be more fracture-prone than normal.
Cerebral calcification
MedGen UID:
124360
Concept ID:
C0270685
Finding
The presence of calcium deposition within the cerebrum.
Basal ganglia calcification
MedGen UID:
234651
Concept ID:
C1389280
Pathologic Function
The presence of calcium deposition affecting one or more structures of the basal ganglia.
Cranial hyperostosis
MedGen UID:
318629
Concept ID:
C1832451
Finding
Excessive growth of the bones of cranium, i.e., of the skull.
Diaphyseal sclerosis
MedGen UID:
1631208
Concept ID:
C4551853
Finding
An elevation in bone density in one or more diaphyses. Sclerosis is normally detected on a radiograph as an area of increased opacity.
Distal renal tubular acidosis
MedGen UID:
853429
Concept ID:
C1704380
Disease or Syndrome
A type of renal tubular acidosis characterized by a failure of acid secretion by the alpha intercalated cells of the cortical collecting duct of the distal nephron. The urine cannot be acidified below a pH of 5.3, associated with acidemia and hypokalemia.
Dental malocclusion
MedGen UID:
9869
Concept ID:
C0024636
Anatomical Abnormality
Dental malocclusion refers to an abnormality of the occlusion, or alignment, of the teeth and the way the upper and lower teeth fit together, resulting in overcrowding of teeth or in abnormal bite patterns.
Visual impairment
MedGen UID:
777085
Concept ID:
C3665347
Finding
Visual impairment (or vision impairment) is vision loss (of a person) to such a degree as to qualify as an additional support need through a significant limitation of visual capability resulting from either disease, trauma, or congenital or degenerative conditions that cannot be corrected by conventional means, such as refractive correction, medication, or surgery.
Visual loss
MedGen UID:
784038
Concept ID:
C3665386
Finding
Loss of visual acuity (implying that vision was better at a certain time point in life). Otherwise the term reduced visual acuity should be used (or a subclass of that).
Elevated serum acid phosphatase
MedGen UID:
326597
Concept ID:
C1839866
Finding

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVOsteopetrosis with renal tubular acidosis

Recent clinical studies

Etiology

Shamsian BS, Momtazmanesh N, Saneifard H, Tabatabaei SMTH, Jafari M, Pour ZK, Al-Hussieni KJMR, Jamee M, Kamfar S
Pediatr Transplant 2024 May;28(3):e14689. doi: 10.1111/petr.14689. PMID: 38655726

Diagnosis

Whyte MP
Bone 2023 Apr;169:116684. Epub 2023 Jan 27 doi: 10.1016/j.bone.2023.116684. PMID: 36709914
Kelly DM, O'Hara PV, Kelleher EM, Casserly LF
Kidney Int 2018 Apr;93(4):1020. doi: 10.1016/j.kint.2017.10.012. PMID: 29571443
Batlle D, Haque SK
Nephrol Dial Transplant 2012 Oct;27(10):3691-704. doi: 10.1093/ndt/gfs442. PMID: 23114896
Shah GN, Bonapace G, Hu PY, Strisciuglio P, Sly WS
Hum Mutat 2004 Sep;24(3):272. doi: 10.1002/humu.9266. PMID: 15300855
Whyte MP
Clin Orthop Relat Res 1993 Sep;(294):52-63. PMID: 8358947

Therapy

Alkhayal Z, Shinwari Z, Gaafar A, Alaiya A
Int J Mol Sci 2023 Sep 8;24(18) doi: 10.3390/ijms241813841. PMID: 37762144Free PMC Article
Sly WS, Whyte MP, Krupin T, Sundaram V
Pediatr Res 1985 Oct;19(10):1033-6. doi: 10.1203/00006450-198510000-00017. PMID: 3932950
Krupin T, Sly WS, Whyte MP, Dodgson SJ
Am J Ophthalmol 1985 Apr 15;99(4):396-9. doi: 10.1016/0002-9394(85)90004-2. PMID: 3920916

Prognosis

Shamsian BS, Momtazmanesh N, Saneifard H, Tabatabaei SMTH, Jafari M, Pour ZK, Al-Hussieni KJMR, Jamee M, Kamfar S
Pediatr Transplant 2024 May;28(3):e14689. doi: 10.1111/petr.14689. PMID: 38655726
Shaik NA, Bokhari HA, Masoodi TA, Shetty PJ, Ajabnoor GMA, Elango R, Banaganapalli B
J Biomol Struct Dyn 2020 Sep;38(14):4067-4080. Epub 2019 Oct 8 doi: 10.1080/07391102.2019.1671899. PMID: 31542996
Pang Q, Qi X, Jiang Y, Wang O, Li M, Xing X, Dong J, Xia W
Metab Brain Dis 2015 Aug;30(4):989-97. Epub 2015 Feb 27 doi: 10.1007/s11011-015-9660-6. PMID: 25720518
Sh Ali AA, Al-Mashta SA
Saudi J Kidney Dis Transpl 2013 May;24(3):561-5. doi: 10.4103/1319-2442.111067. PMID: 23640632

Clinical prediction guides

Shaik NA, Bokhari HA, Masoodi TA, Shetty PJ, Ajabnoor GMA, Elango R, Banaganapalli B
J Biomol Struct Dyn 2020 Sep;38(14):4067-4080. Epub 2019 Oct 8 doi: 10.1080/07391102.2019.1671899. PMID: 31542996
Sh Ali AA, Al-Mashta SA
Saudi J Kidney Dis Transpl 2013 May;24(3):561-5. doi: 10.4103/1319-2442.111067. PMID: 23640632
Felix R, Hofstetter W, Cecchini MG
Eur J Endocrinol 1996 Feb;134(2):143-56. doi: 10.1530/eje.0.1340143. PMID: 8630510
Sly WS, Whyte MP, Sundaram V, Tashian RE, Hewett-Emmett D, Guibaud P, Vainsel M, Baluarte HJ, Gruskin A, Al-Mosawi M
N Engl J Med 1985 Jul 18;313(3):139-45. doi: 10.1056/NEJM198507183130302. PMID: 3925334
Sly WS, Hewett-Emmett D, Whyte MP, Yu YS, Tashian RE
Proc Natl Acad Sci U S A 1983 May;80(9):2752-6. doi: 10.1073/pnas.80.9.2752. PMID: 6405388Free PMC Article

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