Genetic disruption of CYP26B1 severely affects development of neural crest derived head structures, but does not compromise hindbrain patterning

MacLean, Glenn; Dollé, Pascal; Petkovich, Martin

doi:10.1002/dvdy.21878

Cited by 77 publications

(100 citation statements)

References 57 publications

Supporting

Mentioning

Contrasting

Unclassified

Order By: Relevance

“…All Cyp26b1À/À fetuses examined had a complete cleft of the secondary palate (30 of 30 Cyp26b1À/À fetuses at E18.5). Cyp26b1À/À mice at E18.5 exhibit a wide palate defect and die after birth, as previously reported (Yashiro et al, 2004;Maclean et al, 2009) (Fig. 2A).…”

Section: Cyp26b12/2 Mouse Fetuses Have Cleft Palatesupporting

confidence: 66%

“…Scale bars ¼ 100 mm in A-D and 200 mm in H and I. that either their origin or insertion is located on the hyoid bone and all of them play an important role for depressing the tongue and mandibular primordia. Since the hyoid bone is absent in Cyp26b1À/À mice (Maclean et al, 2009), it is very plausible that the origin or insertion of these muscles is severely disrupted. The significantly greater height of the Cyp26b1À/À tongue compared to the WT tongue would be consistent with defective function of these tongue muscles in Cyp26b1À/À mice.…”

Section: The Effects Of Deletion Of Cyp26b1 On Extrinsic Forcesmentioning

confidence: 99%

See 1 more Smart Citation

The regulation of endogenous retinoic acid level through CYP26B1 is required for elevation of palatal shelves

Okano

Kimura

Papaionnou

et al. 2012

Developmental Dynamics

View full text Add to dashboard Cite

Background: In Parkinson's disease, sleep disturbance is a common occurrence. Methods: We evaluated sleep in 10 patients with Parkinson's disease (age, 57.5 ± 9.8 years; disease duration, 12.3 ± 2.7 years) before and after subthalamic nucleus deep brain stimulation using the Parkinson's disease sleep scale and polysomnography. Results: Their total sleep scale scores and daytime sleepiness subscale scores significantly improved after subthalamic nucleus‐deep brain stimulation. The novel findings from this study significantly increased normal rapid eye movement sleep, and decreased abnormal rapid eye movement sleep without atonia after deep brain stimulation in patients with Parkinson's disease. The improved total sleep scale score correlated with decreased wakefulness after sleep onset. Moreover, improved daytime sleepiness correlated with increased normal rapid eye movement sleep time. Sleep improvement did not significantly correlate with resolution of motor complication or reduced dopaminergic dosages. Conclusions: Subthalamic nucleus‐deep brain stimulation may have beneficial effects on sleep disturbance in advanced Parkinson's disease by restoring sleep architecture and normal rapid eye movement sleep. © 2011 Movement Disorder Society

show abstract

Section: Cyp26b12/2 Mouse Fetuses Have Cleft Palatesupporting

confidence: 66%

Section: The Effects Of Deletion Of Cyp26b1 On Extrinsic Forcesmentioning

confidence: 99%

The regulation of endogenous retinoic acid level through CYP26B1 is required for elevation of palatal shelves

Okano

Kimura

Papaionnou

et al. 2012

Developmental Dynamics

View full text Add to dashboard Cite

show abstract

“…8). Treatment of wild-type zebrafish with RA or the Cyp26 inhibitor before suture establishment led to reduced calvarial growth and reduced plate sizes and thicknesses, mimicking the defects of human and mouse CYP26B1 amorphs (Maclean et al, 2009;Laue et al, 2011). In addition, osteogenic fronts displayed a loss of osteoblast markers and a gain of preosteocyte markers (Fig.…”

Section: Discussionmentioning

confidence: 87%

Retinoic acid-induced premature osteoblast-to-preosteocyte transitioning has multiple effects on calvarial development

Jeradi

Hammerschmidt

2016

Development

View full text Add to dashboard Cite

We have previously shown that, in human and zebrafish, hypomorphic mutations of the gene encoding the retinoic acid (RA)-metabolizing enzyme Cyp26b1 result in coronal craniosynostosis, caused by an RA-induced premature transitioning of suture osteoblasts to preosteocytes, inducing ectopic mineralization of the suture's osteoid matrix. In addition, we showed that human CYP26B1 null patients have more severe and seemingly opposite skull defects, characterized by smaller and fragmented calvaria, but the cellular basis of these defects remained largely unclear. Here, by treating juvenile zebrafish with exogenous RA or a chemical Cyp26 inhibitor in the presence or absence of osteogenic cells or bone-resorbing osteoclasts, we demonstrate that both reduced calvarial size and calvarial fragmentation are also caused by RA-induced premature osteoblast-to-preosteocyte transitioning. During calvarial growth, the resulting osteoblast deprival leads to decreased osteoid production and thereby smaller and thinner calvaria, whereas calvarial fragmentation is caused by increased osteoclast stimulation through the gained preosteocytes. Together, our data demonstrate that RAinduced osteoblast-to-preosteocyte transitioning has multiple effects on developing bone in Cyp26b1 mutants, ranging from gain to loss of bone, depending on the allelic strength, the developmental stage and the cellular context.

show abstract

“…It may represent an important mechanism for driving morphological changes in facial architecture during evolution. Such a mechanism is reinforced by experiments implicating elevation of RA that also lead to skull and face bone defects (Vieux-Rochas et al, 2007;Maclean et al, 2009). In both Rara/g-and Rara/b-null mutants, the alisphenoid and incus, which are derived from the maxillary process of BA1, are fused through a supernumerary piece of cartilage to form a pterygoquadrate element that has been lost during mammalian evolution .…”

Section: Rars Are Largely Dispensable In Ncc For the Early Morphogenementioning

confidence: 97%

Retinoic acid receptors exhibit cell‐autonomous functions in cranial neural crest cells

Dupé

Pellerin

2009

Developmental Dynamics

View full text Add to dashboard Cite

Previous work has emphasized the crucial role of retinoic acid (RA) in the ontogenesis of the vast majority of mesenchymal structures derived from the neural crest cells (NCC), which migrate through, or populate, the frontonasal process and branchial arches. Using somatic mutagenesis in the mouse, we have selectively ablated two or three retinoic acid receptors (i.e., RARa/RARb, RARa/RARc and RARa/RARb/ RARc) in NCC. By rigorously analyzing these mutant mice, we found that survival and migration of NCC is normal until gestational day 10.5, suggesting that RAR-dependent signaling is not intrinsically required for the early steps of NCC development. However, ablation of Rara and Rarg genes in NCC yields an agenesis of the median portion of the face, demonstrating that RARa and RARc act cell-autonomously in postmigratory NCC to control the development of structures derived from the frontonasal process. In contrast, ablation of the three Rar genes in NCC leads to less severe defects of the branchial arches derived structures compared with Rar compound null mutants. Therefore, RARs exert a function in the NCC as well as in a separated cell population. This work demonstrates that RARs use distinct mechanisms to pattern cranial NCC. INTRODUCTIONThe morphogenesis of the craniofacial region results from complex developmental processes involving the migration of neural crest cells (NCC) and their subsequent differentiation through interactions with endoderm and ectoderm (Veitch et al., 1999;Trainor and Krumlauf, 2001). Cranial NCC arise from fore-, mid-, and hindbrain in distinct migratory streams. Fore-and midbrain NCC give rise to the frontonasal skeleton, whereas pre-otic hindbrain populations colonize the first two branchial arches (BAs), where they differentiate to form skeletal elements of the jaw, middle ear and tongue. Cardiac NCC participate in the development of aortic arch arteries, as well as thymus, thyroid, and parathyroid glands (Le Douarin et al., 1993;Santagati and Rijli, 2003). Because patterning and morphogenesis of these craniofacial structures require a finely orchestrated series of tissue interactions (Graham and Smith, 2001;Trainor and Krumlauf, 2001), perturbation of different processes may yield similar developmental abnormalities. For instance, inactivation of genes expressed in either NCC, endoderm, or ectoderm, results in similar cardiovascular defects, as remodeling of BA arteries into the definitive arterial vessels and heart outflow tract requires coordinated communication between NCC, endoderm and ectoderm (Depew ABBREVIATIONS BA branchial arch E embryonic day NCC neural crest cells RA retinoic acid ALDH1A retinaldehyde dehydrogenase RAR retinoic acid receptor RXR rexinoid receptor VAD vitamin A-deficient

show abstract

Genetic disruption of CYP26B1 severely affects development of neural crest derived head structures, but does not compromise hindbrain patterning

Cited by 77 publications

References 57 publications

The regulation of endogenous retinoic acid level through CYP26B1 is required for elevation of palatal shelves

The regulation of endogenous retinoic acid level through CYP26B1 is required for elevation of palatal shelves

Retinoic acid-induced premature osteoblast-to-preosteocyte transitioning has multiple effects on calvarial development

Retinoic acid receptors exhibit cell‐autonomous functions in cranial neural crest cells

Contact Info

Product

Resources

About