Combined DiI and Antibody Labeling Reveals Complex Dysgenesis of Hippocampal Dendritic Spines in a Mouse Model of Fragile X Syndrome

Speranza, Luisa; Filiz, Kardelen Dalım; Goebel, Sarah; Perrone-Capano, Carla; Pulcrano, Salvatore; Volpicelli, Floriana; Francesconi, Anna

doi:10.3390/biomedicines10112692

Open AccessArticle

Combined DiI and Antibody Labeling Reveals Complex Dysgenesis of Hippocampal Dendritic Spines in a Mouse Model of Fragile X Syndrome

by

Luisa Speranza

^1,*,

Kardelen Dalım Filiz

²

,

Sarah Goebel

¹

,

Carla Perrone-Capano

²

,

Salvatore Pulcrano

³

,

Floriana Volpicelli

^2,†

and

Anna Francesconi

^1,*,†

¹

Dominick P. Purpura Department of Neuroscience, Albert Einstein College of Medicine, New York, NY 10461, USA

²

Department of Pharmacy, School of Medicine and Surgery, University of Naples Federico II, 80131 Naples, Italy

³

Institute of Genetics and Biophysics “A. Buzzati-Traverso”, C.N.R., 80131 Naples, Italy

^*

Authors to whom correspondence should be addressed.

^†

These authors contributed equally to this work.

Biomedicines 2022, 10(11), 2692; https://doi.org/10.3390/biomedicines10112692

Submission received: 30 August 2022 / Revised: 18 October 2022 / Accepted: 19 October 2022 / Published: 25 October 2022

(This article belongs to the Special Issue State-of-the-Art Neurologic Disease in Italy)

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Abstract

Structural, functional, and molecular alterations in excitatory spines are a common hallmark of many neurodevelopmental disorders including intellectual disability and autism. Here, we describe an optimized methodology, based on combined use of DiI and immunofluorescence, for rapid and sensitive characterization of the structure and composition of spines in native brain tissue. We successfully demonstrate the applicability of this approach by examining the properties of hippocampal spines in juvenile Fmr1 KO mice, a mouse model of Fragile X Syndrome. We find that mutant mice display pervasive dysgenesis of spines evidenced by an overabundance of both abnormally elongated thin spines and cup-shaped spines, in combination with reduced density of mushroom spines. We further find that mushroom spines expressing the actin-binding protein Synaptopodin—a marker for spine apparatus—are more prevalent in mutant mice. Previous work identified spines with Synaptopodin/spine apparatus as the locus of mGluR-LTD, which is abnormally elevated in Fmr1 KO mice. Altogether, our data suggest this enhancement may be linked to the preponderance of this subset of spines in the mutant. Overall, these findings demonstrate the sensitivity and versatility of the optimized methodology by uncovering a novel facet of spine dysgenesis in Fmr1 KO mice.

Keywords: DiIC₁₈; dendritic spines; excitatory synapses; synaptopodin; Fragile X Syndrome; Fmr1 knockout mouse; hippocampus

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MDPI and ACS Style

Speranza, L.; Filiz, K.D.; Goebel, S.; Perrone-Capano, C.; Pulcrano, S.; Volpicelli, F.; Francesconi, A. Combined DiI and Antibody Labeling Reveals Complex Dysgenesis of Hippocampal Dendritic Spines in a Mouse Model of Fragile X Syndrome. Biomedicines 2022, 10, 2692. https://doi.org/10.3390/biomedicines10112692

AMA Style

Speranza L, Filiz KD, Goebel S, Perrone-Capano C, Pulcrano S, Volpicelli F, Francesconi A. Combined DiI and Antibody Labeling Reveals Complex Dysgenesis of Hippocampal Dendritic Spines in a Mouse Model of Fragile X Syndrome. Biomedicines. 2022; 10(11):2692. https://doi.org/10.3390/biomedicines10112692

Chicago/Turabian Style

Speranza, Luisa, Kardelen Dalım Filiz, Sarah Goebel, Carla Perrone-Capano, Salvatore Pulcrano, Floriana Volpicelli, and Anna Francesconi. 2022. "Combined DiI and Antibody Labeling Reveals Complex Dysgenesis of Hippocampal Dendritic Spines in a Mouse Model of Fragile X Syndrome" Biomedicines 10, no. 11: 2692. https://doi.org/10.3390/biomedicines10112692

APA Style

Speranza, L., Filiz, K. D., Goebel, S., Perrone-Capano, C., Pulcrano, S., Volpicelli, F., & Francesconi, A. (2022). Combined DiI and Antibody Labeling Reveals Complex Dysgenesis of Hippocampal Dendritic Spines in a Mouse Model of Fragile X Syndrome. Biomedicines, 10(11), 2692. https://doi.org/10.3390/biomedicines10112692

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Combined DiI and Antibody Labeling Reveals Complex Dysgenesis of Hippocampal Dendritic Spines in a Mouse Model of Fragile X Syndrome

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