Orphan or Rare Diseases

Carer burden in rare inherited diseases: a literature review and conceptual model

Fri, 2022-12-09 06:00

Orphanet J Rare Dis. 2022 Dec 9;17(1):428. doi: 10.1186/s13023-022-02561-w.

NO ABSTRACT

PMID:36494728 | PMC:PMC9733280 | DOI:10.1186/s13023-022-02561-w

Categories: Literature Watch

Case report: Moyamoya disease, the culprit in an adult female presenting with left sided numbness

Fri, 2022-12-09 06:00

Medicine (Baltimore). 2022 Dec 2;101(48):e32160. doi: 10.1097/MD.0000000000032160.

NO ABSTRACT

PMID:36482624 | DOI:10.1097/MD.0000000000032160

Categories: Literature Watch

Efficacy and safety clinical trial with efavirenz in patients diagnosed with adult Niemann-pick type C with cognitive impairment

Fri, 2022-12-09 06:00

Medicine (Baltimore). 2022 Dec 2;101(48):e31471. doi: 10.1097/MD.0000000000031471.

NO ABSTRACT

PMID:36482560 | DOI:10.1097/MD.0000000000031471

Categories: Literature Watch

16p13.11 microdeletion/microduplication in fetuses: investigation of associated ultrasound phenotypes, genetic anomalies, and pregnancy outcome follow-up

Thu, 2022-12-08 06:00

BMC Pregnancy Childbirth. 2022 Dec 7;22(1):913. doi: 10.1186/s12884-022-05267-w.

NO ABSTRACT

PMID:36476185 | DOI:10.1186/s12884-022-05267-w

Categories: Literature Watch

Eradication of early MRSA infection in cystic fibrosis: a novel study design for the STAR-ter trial

Thu, 2022-12-08 06:00

ERJ Open Res. 2022 Dec 5;8(4):00190-2022. doi: 10.1183/23120541.00190-2022. eCollection 2022 Oct.

NO ABSTRACT

PMID:36478917 | PMC:PMC9720546 | DOI:10.1183/23120541.00190-2022

Categories: Literature Watch

Corneal neurotization in the management of neurotrophic keratopathy: A review of the literature

Tue, 2022-12-06 06:00

J Fr Ophtalmol. 2023 Jan;46(1):83-96. doi: 10.1016/j.jfo.2022.09.007. Epub 2022 Dec 5.

ABSTRACT

Neurotrophic keratopathy (NK) is a rare degenerative disease in which damage to the corneal nerves leads to corneal hypoesthesia or anesthesia. Neurotrophic corneal ulcers are notoriously difficult to treat and can lead to blindness. Corneal neurotization (CN) is a recent surgical technique aimed at restoring corneal sensation and may offer a definitive treatment in the wake of NK. Herein, we review the surgical techniques utilized in direct and indirect CN. Technical considerations, outcomes, current limitations and future perspectives are also discussed. This article highlights the key points of this promising procedure and biological aspects that will help provide the best treatment options for patients with severe NK.

PMID:36473789 | DOI:10.1016/j.jfo.2022.09.007

Categories: Literature Watch

Cannabidiol reveals a disruptive strategy for 21st century epilepsy drug discovery

Tue, 2022-12-06 06:00

Exp Neurol. 2022 Nov 26;360:114288. doi: 10.1016/j.expneurol.2022.114288. Online ahead of print.

NO ABSTRACT

PMID:36471511 | DOI:10.1016/j.expneurol.2022.114288

Categories: Literature Watch

Federated learning enables big data for rare cancer boundary detection

Mon, 2022-12-05 06:00

Nat Commun. 2022 Dec 5;13(1):7346. doi: 10.1038/s41467-022-33407-5.

NO ABSTRACT

PMID:36470898 | DOI:10.1038/s41467-022-33407-5

Categories: Literature Watch

A novel splice-site variant of the LAMB3 gene is associated with junctional epidermolysis bullosa

Mon, 2022-12-05 06:00

Eur J Dermatol. 2022 Sep 1;32(5):632-636. doi: 10.1684/ejd.2022.4318.

NO ABSTRACT

PMID:36468719 | DOI:10.1684/ejd.2022.4318

Categories: Literature Watch

Palpebral marginoplasty in the management of distichiasis: About 104 eyes

Sat, 2022-12-03 06:00

J Fr Ophtalmol. 2023 Jan;46(1):65-71. doi: 10.1016/j.jfo.2022.05.024. Epub 2022 Nov 30.

ABSTRACT

Distichiasis is an anomaly of the of the eyelid margin characterized by the presence of a second row of eyelashes arising from the posterior lamella. It is a rare condition whose severity is related to corneal complications. There are two forms: congenital and acquired, the latter of which is the most frequent in our population, resulting mainly from chronic inflammation. There are several treatment modalities, the choice of which is up to the practitioner depending on the means at his or her disposal and the risk-benefit ratio. We report a series of 104 eyes (90 patients) undergoing surgery for distichiasis using the marginoplasty technique with labial mucosal autograft, collected between January 2000 and September 2021. The main indication was any distichiasis with more than seven lashes per eyelid. The mean age of the patients was 65.5 years. With a mean follow-up of over 24 months, a reduction in functional signs was noted in over 95% of cases. The success rate after marginoplasty with labial mucosal autograft alone was 82.7%. The recurrence rate was 17.3%, which patients underwent additional treatment by argon laser photoablation with very good final results (96% final success rate). Marginoplasty with labial mucosal autograft represents a technique of choice in distichiasis threatening the cornea, particularly in cases of distichiasis with a significant number of eyelashes to be treated.

PMID:36462946 | DOI:10.1016/j.jfo.2022.05.024

Categories: Literature Watch

Natural History and Real-World Data in Rare Diseases: Applications, Limitations, and Future Perspectives

Sat, 2022-12-03 06:00

J Clin Pharmacol. 2022 Dec;62 Suppl 2:S38-S55. doi: 10.1002/jcph.2134.

NO ABSTRACT

PMID:36461748 | DOI:10.1002/jcph.2134

Categories: Literature Watch

Overview of Clinical Pharmacology Packages of New Drug Applications Approved for the Treatment of Rare Diseases

Sat, 2022-12-03 06:00

J Clin Pharmacol. 2022 Dec;62 Suppl 2:S72-S78. doi: 10.1002/jcph.2167.

NO ABSTRACT

PMID:36461747 | DOI:10.1002/jcph.2167

Categories: Literature Watch

Immunogenicity Considerations for Therapeutic Modalities Used in Rare Diseases

Sat, 2022-12-03 06:00

J Clin Pharmacol. 2022 Dec;62 Suppl 2:S110-S118. doi: 10.1002/jcph.2166.

NO ABSTRACT

PMID:36461746 | DOI:10.1002/jcph.2166

Categories: Literature Watch

Until My Son Calls Me Dad: Quest to Find a Treatment for Ultra-Rare Disease

Sat, 2022-12-03 06:00

J Clin Pharmacol. 2022 Dec;62 Suppl 2:S12-S14. doi: 10.1002/jcph.2154.

NO ABSTRACT

PMID:36461745 | DOI:10.1002/jcph.2154

Categories: Literature Watch

Model-Informed Approach Supporting Drug Development and Regulatory Evaluation for Rare Diseases

Sat, 2022-12-03 06:00

J Clin Pharmacol. 2022 Dec;62 Suppl 2:S27-S37. doi: 10.1002/jcph.2143.

NO ABSTRACT

PMID:36461744 | DOI:10.1002/jcph.2143

Categories: Literature Watch

Bayesian Complex Innovative Trial Designs (CIDs) and Their Use in Drug Development for Rare Disease

Sat, 2022-12-03 06:00

J Clin Pharmacol. 2022 Dec;62 Suppl 2:S56-S71. doi: 10.1002/jcph.2132.

NO ABSTRACT

PMID:36461743 | DOI:10.1002/jcph.2132

Categories: Literature Watch

Think Rare, Think Inside and Out: Simple Question-Based Approach to Complex Rare Disease Drug Development

Sat, 2022-12-03 06:00

J Clin Pharmacol. 2022 Dec;62 Suppl 2:S6-S11. doi: 10.1002/jcph.2173.

NO ABSTRACT

PMID:36461741 | DOI:10.1002/jcph.2173

Categories: Literature Watch

Bridging the Gap With Clinical Pharmacology in Innovative Rare Disease Treatment Modalities: Targeting DNA to RNA to Protein

Sat, 2022-12-03 06:00

J Clin Pharmacol. 2022 Dec;62 Suppl 2:S95-S109. doi: 10.1002/jcph.2172.

NO ABSTRACT

PMID:36461740 | DOI:10.1002/jcph.2172

Categories: Literature Watch

Regulatory Framework for Drug Development in Rare Diseases

Sat, 2022-12-03 06:00

J Clin Pharmacol. 2022 Dec;62 Suppl 2:S15-S26. doi: 10.1002/jcph.2171.

NO ABSTRACT

PMID:36461739 | DOI:10.1002/jcph.2171

Categories: Literature Watch

Superior mesenteric artery syndrome and disseminated tuberculosis: "Double troubleˮ

Fri, 2022-12-02 06:00

Indian J Tuberc. 2022 Oct;69(4):702-705. doi: 10.1016/j.ijtb.2021.08.019. Epub 2021 Aug 19.

NO ABSTRACT

PMID:36460412 | DOI:10.1016/j.ijtb.2021.08.019

Categories: Literature Watch

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