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Lobar holoprosencephaly

MedGen UID:
96559
Concept ID:
C0431362
Congenital Abnormality
Synonyms: Holoprosencephalies, Lobar; Holoprosencephaly, Lobar; Lobar Holoprosencephalies; Lobar Holoprosencephaly
SNOMED CT: Lobar holoprosencephaly (253136007); Olfactory aplasia (253136007); Cingulosynapsis (253136007)
Modes of inheritance:
Non-Mendelian inheritance
MedGen UID:
109109
Concept ID:
C0600599
Genetic Function
Source: Orphanet
A mode of inheritance that depends on a mixture of major and minor genetic determinants possibly together with environmental factors. Diseases inherited in this manner are termed complex diseases.
Not genetically inherited
MedGen UID:
988794
Concept ID:
CN307044
Finding
Source: Orphanet
clinical entity without genetic inheritance.
 
HPO: HP:0006870
Monarch Initiative: MONDO:0019756
Orphanet: ORPHA93924

Definition

A type of holoprosencephaly in which most of the right and left cerebral hemispheres and lateral ventricles are separated but the most rostral aspect of the telencephalon, the frontal lobes, are fused, especially ventrally. [from HPO]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • Lobar holoprosencephaly
Follow this link to review classifications for Lobar holoprosencephaly in Orphanet.

Conditions with this feature

Hartsfield-Bixler-Demyer syndrome
MedGen UID:
335111
Concept ID:
C1845146
Congenital Abnormality
FGFR1-related Hartsfield syndrome comprises two core features: holoprosencephaly (HPE) spectrum disorder and ectrodactyly spectrum disorder. HPE spectrum disorder, resulting from failed or incomplete forebrain division early in gestation, includes alobar, semilobar, or lobar HPE. Other observed midline brain malformations include corpus callosum agenesis, absent septum pellucidum, absent olfactory bulbs and tracts, and vermian hypoplasia. Other findings associated with the HPE spectrum such as craniofacial dysmorphism, neurologic issues (developmental delay, spasticity, seizures, hypothalamic dysfunction), feeding problems, and endocrine issues (hypogonadotropic hypogonadism and central insipidus diabetes) are common. Ectrodactyly spectrum disorders are unilateral or bilateral malformations of the hands and/or feet characterized by a median cleft of hand or foot due to absence of the longitudinal central rays (also called split-hand/foot malformation). The number of digits on the right and left can vary. Polydactyly and syndactyly can also be seen.
Holoprosencephaly 5
MedGen UID:
355304
Concept ID:
C1864827
Disease or Syndrome
Holoprosencephaly associated with mutations in the ZIC2 gene.
Cornelia de Lange syndrome 4
MedGen UID:
766431
Concept ID:
C3553517
Disease or Syndrome
Cornelia de Lange syndrome (CdLS) encompasses a spectrum of findings from mild to severe. Severe (classic) CdLS is characterized by distinctive facial features, growth restriction (prenatal onset; <5th centile throughout life), hypertrichosis, and upper-limb reduction defects that range from subtle phalangeal abnormalities to oligodactyly (missing digits). Craniofacial features include synophrys, highly arched and/or thick eyebrows, long eyelashes, short nasal bridge with anteverted nares, small widely spaced teeth, and microcephaly. Individuals with a milder phenotype have less severe growth, cognitive, and limb involvement, but often have facial features consistent with CdLS. Across the CdLS spectrum IQ ranges from below 30 to 102 (mean: 53). Many individuals demonstrate autistic and self-destructive tendencies. Other frequent findings include cardiac septal defects, gastrointestinal dysfunction, hearing loss, myopia, and cryptorchidism or hypoplastic genitalia.

Professional guidelines

PubMed

Vinurel N, Van Nieuwenhuyse A, Cagneaux M, Garel C, Quarello E, Brasseur M, Picone O, Ferry M, Gaucherand P, des Portes V, Guibaud L
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Recent clinical studies

Etiology

Hu Y, Sun L, Feng L, Wang J, Zhu Y, Wu Q
BMC Pregnancy Childbirth 2023 May 3;23(1):312. doi: 10.1186/s12884-023-05644-z. PMID: 37138220Free PMC Article
Fujino S, Enokizono M, Ihara S, Kono T, Miyama S
BMC Neurol 2022 Aug 25;22(1):316. doi: 10.1186/s12883-022-02764-5. PMID: 36008788Free PMC Article
Jacquinet A, Brown L, Sawkins J, Liu P, Pugash D, Van Allen MI, Patel MS
Eur J Med Genet 2018 May;61(5):257-261. Epub 2017 Dec 24 doi: 10.1016/j.ejmg.2017.12.011. PMID: 29278735
Takenouchi T, Okuno H, Kosaki R, Ariyasu D, Torii C, Momoshima S, Harada N, Yoshihashi H, Takahashi T, Awazu M, Kosaki K
Am J Med Genet A 2012 Oct;158A(10):2537-41. Epub 2012 Aug 10 doi: 10.1002/ajmg.a.35465. PMID: 22887648
Cohen MM Jr
Birth Defects Res A Clin Mol Teratol 2006 Sep;76(9):658-73. doi: 10.1002/bdra.20295. PMID: 17001700

Diagnosis

Daly T, Roberts A, Yang E, Mochida GH, Bodamer O
Am J Med Genet A 2020 Mar;182(3):441-445. Epub 2019 Dec 17 doi: 10.1002/ajmg.a.61454. PMID: 31846209
Kakar N, Ahmad J, Morris-Rosendahl DJ, Altmüller J, Friedrich K, Barbi G, Nürnberg P, Kubisch C, Dobyns WB, Borck G
Hum Genet 2015 Jan;134(1):45-51. Epub 2014 Sep 14 doi: 10.1007/s00439-014-1487-4. PMID: 25218063
Huss S, Fronhoffs F, Gembruch U, Loeffler K, Born M, Gessi M, Kuchelmeister K, Müller AM
Pediatr Dev Pathol 2011 Sep-Oct;14(5):411-7. Epub 2011 Apr 13 doi: 10.2350/10-09-0916-CR.1. PMID: 21488711
Fitz CR
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Osaka K, Matsumoto S
J Neurosurg 1978 May;48(5):787-803. doi: 10.3171/jns.1978.48.5.0787. PMID: 305951

Therapy

Chuen-im P, Smyth MD, Segura B, Ferkol T, Rivera-Spoljaric K
Pediatr Pulmonol 2012 Jan;47(1):91-5. Epub 2011 Aug 24 doi: 10.1002/ppul.21510. PMID: 22170808
Pasquier L, Marcorelles P, Loget P, Pelluard F, Carles D, Perez MJ, Bendavid C, de La Rochebrochard C, Ferry M, David V, Odent S, Laquerrière A
Acta Neuropathol 2009 Feb;117(2):185-200. Epub 2008 Dec 5 doi: 10.1007/s00401-008-0469-9. PMID: 19057916
Kotzot D, Weigl J, Huk W, Rott HD
Teratology 1993 Jul;48(1):15-9. doi: 10.1002/tera.1420480105. PMID: 8351644

Prognosis

Steinberg JP, Brady CM, Burstein FD
J Craniofac Surg 2016 Mar;27(2):480-3. doi: 10.1097/SCS.0000000000002420. PMID: 26967082
Gawrych E, Janiszewska-Olszowska J, Walecka A, Syryńska M, Chojnacka H
Cleft Palate Craniofac J 2009 Sep;46(5):549-54. Epub 2009 Mar 2 doi: 10.1597/08-059.1. PMID: 19929095
Sztriha L, Várady E, Hertecant J, Nork M
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Pensler JM, Giese S, Charrow J
J Craniofac Surg 1993 Jan;4(1):2-5; discussion 6-7. doi: 10.1097/00001665-199301000-00002. PMID: 8467017
Shanks DE, Wilson WG
Dev Med Child Neurol 1988 Jun;30(3):383-6. doi: 10.1111/j.1469-8749.1988.tb14565.x. PMID: 3402680

Clinical prediction guides

Fujino S, Enokizono M, Ihara S, Kono T, Miyama S
BMC Neurol 2022 Aug 25;22(1):316. doi: 10.1186/s12883-022-02764-5. PMID: 36008788Free PMC Article
Galante D, Fortarezza D, Caggiano M, de Francisci G, Pedrotti D, Caruselli M
Braz J Anesthesiol 2015 Sep-Oct;65(5):379-83. Epub 2014 Oct 25 doi: 10.1016/j.bjane.2014.07.003. PMID: 26323737
Takenouchi T, Okuno H, Kosaki R, Ariyasu D, Torii C, Momoshima S, Harada N, Yoshihashi H, Takahashi T, Awazu M, Kosaki K
Am J Med Genet A 2012 Oct;158A(10):2537-41. Epub 2012 Aug 10 doi: 10.1002/ajmg.a.35465. PMID: 22887648
Bertolacini CD, Ribeiro-Bicudo LA, Petrin A, Richieri-Costa A, Murray JC
Clin Genet 2012 Jan;81(1):70-5. Epub 2011 Jan 19 doi: 10.1111/j.1399-0004.2010.01606.x. PMID: 21204792Free PMC Article
Shanks DE, Wilson WG
Dev Med Child Neurol 1988 Jun;30(3):383-6. doi: 10.1111/j.1469-8749.1988.tb14565.x. PMID: 3402680

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