Association of bronchopulmonary sequestration with expression of the homeobox protein Hoxb-5

Volpe, MaryAnn V.; Archavachotikul, Kwanchai; Bhan, Ina; Lessin, Marc S.; Nielsen, Heber C.

doi:10.1053/jpsu.2000.19266

Cited by 25 publications

(10 citation statements)

References 18 publications

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“…Even though no lung phenotype was reported in Hoxb5 mutant mice, Hoxb5 lung expression, in vitro studies, and expression data in human lung diseases suggest a role for Hoxb5 in lung development (6,43,52,53). The severity of the Hoxa5 lung phenotype indicates that Hoxa5 function is less subject to rescue by other genes.…”

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confidence: 99%

Partial functional redundancy betweenHoxa5andHoxb5paralog genes during lung morphogenesis

Boucherat

Montaron

Bérubé-Simard

et al. 2013

American Journal of Physiology-Lung Cellular and Molecular Phys

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Hox genes encode transcription factors governing complex developmental processes in several organs. A subset of Hox genes are expressed in the developing lung. Except for Hoxa5, the lack of overt lung phenotype in single mutants suggests that Hox genes may not play a predominant role in lung ontogeny or that functional redundancy may mask anomalies. In the Hox5 paralog group, both Hoxa5 and Hoxb5 genes are expressed in the lung mesenchyme whereas Hoxa5 is also expressed in the tracheal mesenchyme. Herein, we generated Hoxa5;Hoxb5 compound mutant mice to evaluate the relative contribution of each gene to lung development. Hoxa5;Hoxb5 mutants carrying the four mutated alleles displayed an aggravated lung phenotype, resulting in the death of the mutant pups at birth. Characterization of the phenotype highlighted the role of Hoxb5 in lung formation, the latter being involved in branching morphogenesis, goblet cell specification, and postnatal air space structure, revealing partial functional redundancy with Hoxa5. However, the Hoxb5 lung phenotypes were less severe than those seen in Hoxa5 mutants, likely because of Hoxa5 compensation. New specific roles for Hoxa5 were also unveiled, demonstrating the extensive contribution of Hoxa5 to the developing respiratory system. The exclusive expression of Hoxa5 in the trachea and the phrenic motor column likely underlies the Hoxa5-specific trachea and diaphragm phenotypes. Altogether, our observations establish that the Hoxa5 and Hoxb5 paralog genes shared some functions during lung morphogenesis, Hoxa5 playing a predominant role.

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confidence: 99%

Partial functional redundancy betweenHoxa5andHoxb5paralog genes during lung morphogenesis

Boucherat

Montaron

Bérubé-Simard

et al. 2013

American Journal of Physiology-Lung Cellular and Molecular Phys

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“…HoxB5 is a homeobox-containing gene that plays an important role in the patterning of airway branches 11,23,24. Strong expression of this gene is found in lung mesenchyme during branching morphogenesis, and its expression falls in the saccular stage11 and after birth it is expressed only in alveolar type I cells.…”

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confidence: 99%

The expression of HoxB5 and SPC in neonatal rat lung at exposure to fluoxetine

Taghizadeh

Taghipour

Karimi

et al. 2016

DDDT

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ObjectiveApproximately 10% of pregnant women suffer from pregnancy-associated depression. Fluoxetine, as a selective serotonin reuptake inhibitor, is being employed as a therapy for depressive disorders. The present study aimed to determine the effects of fluoxetine on neonatal lung development.MethodsThirty pregnant Wistar rats (weighing 200–250 g) were treated daily with 7 mg/kg fluoxetine from gestation day 0 to gestation day 21, via gavage. The control group received a similar volume of distilled water only. Following delivery, the newborns and their lungs were immediately weighed in both of the groups. The right lung was fixed for histological assessments while the left lung was used for evaluation of the expression of SPC and HoxB5 by the real-time polymerase chain reaction method.ResultsResults have indicated that even though the body weight and the number of neonatal rats in both groups were the same, the lung weight of neonates exposed to fluoxetine was significantly different compared to the control group (P<0.05). Expression of both genes was increased, nonetheless, only elevation of HoxB5 was significant (P<0.05). Histological studies demonstrated that lung tissue in the fluoxetine treatment group morphologically appears to be similar to the pseudoglandular phase, whereas the control group lungs experienced more development.ConclusionAccording to the upregulated expression of HoxB5 concerning histological findings, results of the present study showed that fluoxetine can influence lung growth and may in turn lead to delay in lung development. So establishment of studies to identify the effects of antidepressant drugs during pregnancy is deserved.

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“…It occurs within a lobe of the lung, and unlike extralobar sequestrations, has no separate pleural investment. Unlike extralobar sequestrations which may be due to abnormal expression of homeobox genes, there is evidence that intralobar sequestrations are acquired lesions with a late presentation caused by recurrent bronchial obstruction and distal infection leading to scarring and fibrosis around the airway and ultimate isolation of the involved parenchyma [Volpe et al, 2000; Mendeloff, 2004].…”

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confidence: 99%

Aicardi syndrome associated with hepatoblastoma and pulmonary sequestration

Kamien

Gabbett

2009

American J of Med Genetics Pt A

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We present a female with Aicardi syndrome (AS) who had infantile spasms, characteristic chorioretinal lacunae, partial agenesis of the corpus callosum, and neuronal migration abnormalities. Our patient was also diagnosed with a composite lung lesion of intralobar pulmonary sequestration and congenital cystic adenomatoid malformation (CCAM) and developed a hepatoblastoma. This is the second report that describes the association of hepatoblastoma and AS, and the first report of pulmonary sequestration or CCAM and AS.The propositus was female and the second child born to healthy non-consanguineous Caucasian parents. The family history was unremarkable; her older sister was well and developmentally normal. An antenatal ultrasound performed at 20 weeks gestation demonstrated a pulmonary sequestration. The pregnancy proceeded to an uneventful spontaneous vaginal delivery at term. Computerized tomography performed at age 11 days to further investigate the pulmonary sequestration, demonstrated a superior hepatic mass (Fig. 1). Further investigation revealed this to be a hepatoblastoma (Fig. 2). The hepatoblastoma was successfully treated with debulking chemotherapy followed by complete resection.Infantile spasms commenced at age 5 months. Management included high dose steroids, later changed to vigabatrin. Cranial MRI revealed dysgenesis of the corpus callosum, a forme fruste of schizencephaly involving the mid body of the right lateral ventricle, foci of nodular heterotopic gray matter marginating both lateral ventricles, and polymicrogyria of the right frontal lobe. The cerebellum had a normal appearance.On examination at 6 months of age, her weight was 6.7 kg (75th centile), length was 63 cm (75th centile), and head circumference was 40 cm (25th centile). She had mild Cushing syndrome-like features, attributed to high dose steroids. She had upslanting palpebral fissures, an upturned nasal tip and a deep philtrum. She had relatively large ears and sparse eyebrows (Fig. 3).Normal investigations included a G-banded karyotype and chest X-ray demonstrating normal ribs and spine. A specialist ophthalmology opinion was sought and the presence of characteristic chorioretinal lacunae confirmed the diagnosis of AS. There was no evidence of microphthalmia, coloboma, or other optic nerve abnormalities.

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Association of bronchopulmonary sequestration with expression of the homeobox protein Hoxb-5

Cited by 25 publications

References 18 publications

Partial functional redundancy betweenHoxa5andHoxb5paralog genes during lung morphogenesis

Partial functional redundancy betweenHoxa5andHoxb5paralog genes during lung morphogenesis

The expression of HoxB5 and SPC in neonatal rat lung at exposure to fluoxetine

Aicardi syndrome associated with hepatoblastoma and pulmonary sequestration

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