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Familial expansile osteolysis(FEO)

MedGen UID:
96593
Concept ID:
C0432292
Congenital Abnormality
Synonyms: FEO; Mccabe disease; Polyostotic osteolytic dysplasia, hereditary expansile
SNOMED CT: Familial expansile osteolysis (254153009)
Modes of inheritance:
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.
 
Gene (location): TNFRSF11A (18q21.33)
 
Monarch Initiative: MONDO:0008275
OMIM®: 174810
Orphanet: ORPHA85195

Definition

Familial expansile osteolysis is an autosomal dominant bone dysplasia characterized by increased bone remodeling with osteolytic lesions mainly affecting the appendicular skeleton. There is medullary and cortical expansion of the bone without sclerosis, leading to painful and disabling deformities and tendency to pathologic fracture. Clinical features include onset of conductive hearing loss in childhood, premature loss of teeth, and variably increased serum alkaline phosphatase (summary by Palenzuela et al., 2002 and Elahi et al., 2007). [from OMIM]

Clinical features

From HPO
Bone pain
MedGen UID:
57489
Concept ID:
C0151825
Sign or Symptom
An unpleasant sensation characterized by physical discomfort (such as pricking, throbbing, or aching) localized to bone.
Hydroxyprolinuria
MedGen UID:
215298
Concept ID:
C0948585
Finding
An increased concentration of 4-hydroxy-L-proline in the urine.
Conductive hearing impairment
MedGen UID:
9163
Concept ID:
C0018777
Disease or Syndrome
An abnormality of vibrational conductance of sound to the inner ear leading to impairment of sensory perception of sound.
Pathologic fracture
MedGen UID:
42095
Concept ID:
C0016663
Pathologic Function
A pathologic fracture occurs when a bone breaks in an area that is weakened secondarily to another disease process such as tumor, infection, and certain inherited bone disorders. A pathologic fracture can occur without a degree of trauma required to cause fracture in healthy bone.
Thin bony cortex
MedGen UID:
318844
Concept ID:
C1833325
Finding
Abnormal thinning of the cortical region of bones.
Bowing of the long bones
MedGen UID:
340849
Concept ID:
C1855340
Congenital Abnormality
A bending or abnormal curvature of a long bone.
Osteolysis
MedGen UID:
1648424
Concept ID:
C4721411
Pathologic Function
Osteolysis refers to the destruction of bone through bone resorption with removal or loss of calcium.
Elevated circulating alkaline phosphatase concentration
MedGen UID:
727252
Concept ID:
C1314665
Finding
Abnormally increased serum levels of alkaline phosphatase activity.
Premature loss of teeth
MedGen UID:
66678
Concept ID:
C0232513
Finding
Exfoliation of a tooth more than 2 SD earlier than the normal age for the deciduous teeth and not related to traume or neglect. Exfoliation of a permanent tooth is per se abnormal.
Fragile teeth
MedGen UID:
382413
Concept ID:
C2674620
Finding
A tendency of teeth to fracture as manifested by a history of repeated fracture of the dental enamel without adequate trauma.

Term Hierarchy

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

Professional guidelines

PubMed

Skowrońska-Jóźwiak E, Lorenc RS
Treat Endocrinol 2006;5(5):297-318. doi: 10.2165/00024677-200605050-00004. PMID: 17002489

Recent clinical studies

Etiology

Lucas GJ, Daroszewska A, Ralston SH
J Bone Miner Res 2006 Dec;21 Suppl 2:P31-7. doi: 10.1359/jbmr.06s206. PMID: 17229006
Kantaputra PN, Limwongse C, Koolvisoot A, Ausawamongkolkul A, Tayavitit S
Am J Med Genet A 2006 Dec 1;140(23):2640-5. doi: 10.1002/ajmg.a.31373. PMID: 17001672
Daroszewska A, Ralston SH
Clin Sci (Lond) 2005 Sep;109(3):257-63. doi: 10.1042/CS20050053. PMID: 16104845
Hansen MF, Nellissery MJ, Bhatia P
J Bone Miner Res 1999 Oct;14 Suppl 2:39-44. doi: 10.1002/jbmr.5650140209. PMID: 10510212
Olsen CB, Tangchaitrong K, Chippendale I, Graham HK, Dahl HM, Stockigt JR
Pediatr Dent 1999 Sep-Oct;21(6):363-7. PMID: 10509339

Diagnosis

Macaraeg K, Lee SM, Kalra L, Velasco M, Hajishengallis E
Pediatr Dent 2020 Jan 15;42(1):62-65. PMID: 32075714
Ralston SH, Taylor JP
Calcif Tissue Int 2019 May;104(5):501-516. Epub 2019 Feb 13 doi: 10.1007/s00223-019-00520-5. PMID: 30756140Free PMC Article
Kantaputra PN, Limwongse C, Koolvisoot A, Ausawamongkolkul A, Tayavitit S
Am J Med Genet A 2006 Dec 1;140(23):2640-5. doi: 10.1002/ajmg.a.31373. PMID: 17001672
Crone MD, Wallace RG
Skeletal Radiol 1990;19(4):245-50. doi: 10.1007/BF00191665. PMID: 2353208
Osterberg PH, Wallace RG, Adams DA, Crone RS, Dickson GR, Kanis JA, Mollan RA, Nevin NC, Sloan J, Toner PG
J Bone Joint Surg Br 1988 Mar;70(2):255-60. doi: 10.1302/0301-620X.70B2.3346299. PMID: 3346299

Therapy

Whyte MP, Tau C, McAlister WH, Zhang X, Novack DV, Preliasco V, Santini-Araujo E, Mumm S
Bone 2014 Nov;68:153-61. Epub 2014 Jul 23 doi: 10.1016/j.bone.2014.07.019. PMID: 25063546Free PMC Article
Riches PL, Imanishi Y, Nakatsuka K, Ralston SH
Calcif Tissue Int 2008 Oct;83(4):272-5. Epub 2008 Oct 4 doi: 10.1007/s00223-008-9177-7. PMID: 18836673
Skowrońska-Jóźwiak E, Lorenc RS
Treat Endocrinol 2006;5(5):297-318. doi: 10.2165/00024677-200605050-00004. PMID: 17002489
Whyte MP, Reinus WR, Podgornik MN, Mills BG
Medicine (Baltimore) 2002 Mar;81(2):101-21. doi: 10.1097/00005792-200203000-00002. PMID: 11889411
Wallace RG, Barr RJ, Osterberg PH, Mollan RA
Clin Orthop Relat Res 1989 Nov;(248):265-77. PMID: 2530018

Prognosis

Craven M, Vajravelu ME, Shekdar KV, Levine MA, Mumm S, Whyte MP, Mancilla EE
Bone 2023 May;170:116698. Epub 2023 Feb 3 doi: 10.1016/j.bone.2023.116698. PMID: 36740137Free PMC Article
Marik I, Marikova A, Hyankova E, Kozlowski K
Skeletal Radiol 2006 Nov;35(11):872-5. Epub 2006 Feb 10 doi: 10.1007/s00256-005-0077-x. PMID: 16470392
Daneshi A, Shafeghati Y, Karimi-Nejad MH, Khosravi A, Farhang F
Otol Neurotol 2005 Mar;26(2):237-40. doi: 10.1097/00129492-200503000-00018. PMID: 15793411
Olsen CB, Tangchaitrong K, Chippendale I, Graham HK, Dahl HM, Stockigt JR
Pediatr Dent 1999 Sep-Oct;21(6):363-7. PMID: 10509339
Mitchell CA, Kennedy JG, Wallace RG
Oral Surg Oral Med Oral Pathol 1990 Sep;70(3):301-7. doi: 10.1016/0030-4220(90)90145-i. PMID: 2216357

Clinical prediction guides

Whyte MP, Tau C, McAlister WH, Zhang X, Novack DV, Preliasco V, Santini-Araujo E, Mumm S
Bone 2014 Nov;68:153-61. Epub 2014 Jul 23 doi: 10.1016/j.bone.2014.07.019. PMID: 25063546Free PMC Article
Marik I, Marikova A, Hyankova E, Kozlowski K
Skeletal Radiol 2006 Nov;35(11):872-5. Epub 2006 Feb 10 doi: 10.1007/s00256-005-0077-x. PMID: 16470392
Johnson-Pais TL, Singer FR, Bone HG, McMurray CT, Hansen MF, Leach RJ
J Bone Miner Res 2003 Feb;18(2):376-80. doi: 10.1359/jbmr.2003.18.2.376. PMID: 12568416
Cody JD, Singer FR, Roodman GD, Otterund B, Lewis TB, Leppert M, Leach RJ
Am J Hum Genet 1997 Nov;61(5):1117-22. doi: 10.1086/301601. PMID: 9345096Free PMC Article
Hughes AE, Shearman AM, Weber JL, Barr RJ, Wallace RG, Osterberg PH, Nevin NC, Mollan RA
Hum Mol Genet 1994 Feb;3(2):359-61. doi: 10.1093/hmg/3.2.359. PMID: 7911698

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