Processes that regulate size and patterning along an axis must be highly integrated to generate robust shapes; relative changes in these processes underlie both congenital disease and evolutionary change. Fin length mutants in zebrafish have provided considerable insight into the pathways regulating fin size, yet signals underlying patterning have remained less clear. The bony rays of the fins possess distinct patterning along the proximodistal axis, reflected in the location of ray bifurcations and the lengths of ray segments, which show progressive shortening along the axis. Here, we show that thyroid hormone (TH) regulates aspects of proximodistal patterning of the caudal fin rays, regardless of fin size. TH promotes distal gene expression patterns, coordinating ray bifurcations and segment shortening with skeletal outgrowth along the proximodistal axis. This distalizing role for TH is conserved between development and regeneration, in all fins (paired and medial), and between Danio species as well as distantly related medaka. During regenerative outgrowth, TH acutely induces Shh-mediated skeletal bifurcation. Zebrafish have multiple nuclear TH receptors, and we found that unliganded Thrab—but not Thraa or Thrb—inhibits the formation of distal features. Broadly, these results demonstrate that proximodistal morphology is regulated independently from size-instructive signals. Modulating proximodistal patterning relative to size—either through changes to TH metabolism or other hormone-independent pathways—can shift skeletal patterning in ways that recapitulate aspects of fin ray diversity found in nature.

Neuroimaging studies of the functional organization of human auditory cortex have focused on group-level analyses to identify tendencies that represent the typical brain. Here, we mapped auditory areas of the human superior temporal cortex (STC) in 30 participants (15 women) by combining functional network analysis and 1-mm isotropic resolution 7T functional magnetic resonance imaging (fMRI). Two resting-state fMRI sessions, and one or two auditory and audiovisual speech localizer sessions, were collected on 3–4 separate days. We generated a set of functional network-based parcellations from these data. Solutions with 4, 6, and 11 networks were selected for closer examination based on local maxima of the Dice coefficients and Silhouette values. The resulting parcellation of auditory cortices showed intraindividual reproducibility of 69–78% between resting-state sessions and 62–73% between resting-state and task sessions, indicating moderate reproducibility. The interindividual variability was significantly larger than intraindividual variability (Dice coefficient: 57%–68%, p < 0.001), indicating that the parcellations also captured meaningful interindividual variability. The individual-specific parcellations yielded the highest alignment with task response topographies, suggesting that individual variability in parcellations reflects individual variability in auditory function. Connectional homogeneity within networks was also highest for the individual-specific parcellations. Furthermore, the similarity in the functional parcellations was not explainable by the similarity of macroanatomical properties of the auditory cortex. Together, our results show that auditory areas in STC can be segmented into functional subareas based on functional connectivity. Our findings also suggest that individual-level parcellations capture meaningful idiosyncrasies in auditory cortex organization.

Introduction: Extrarenal Wilms tumors (ERWTs) (i.e., nephroblastoma) are exceptionally rare tumors that have only been reported approximately 100 times in the literature. These tumors necessitate histology (rather than imaging) for proper identification, often resulting in a postoperative diagnosis. Case Presentation: At 20 weeks of gestation, a female fetus was diagnosed with a subcutaneous lumbosacral mass by prenatal ultrasound (US). Days after birth, the mass was resected and pathologically determined to be an ERWT. Specifically, the excised mass had a triphasic histologic pattern, including blastemal, stromal, and primitive epithelial components. Centrally, the lesion demonstrated cystic and pseudopapillary architectural features, while peripherally, the lesion was more solid with the morphologic appearance of Wilms tumor (WT). This case is unique due to (1) the unusual lumbosacral location, (2) the presence of normal bilateral kidneys on US, and (3) the detection of the mass on in utero imaging studies. Literature Review: Including our report, ERWT has been reported 16 times in the (para)spinal/vertebral region and range from T9 to the coccyx. Most patients initially receive surgery (gross total resection, if feasible) followed by chemotherapy (vincristine and dactinomycin) and radiation depending on final pathology and additional case considerations. Discussion: Due to the rarity of ERWTs, there is no standardized treatment. Complete excision with adjuvant chemotherapy (vincristine and dactinomycin) is most often suggested as a best/most appropriate approach to treatment, with the addition of radiotherapy for recurrence and/or metastasis. Conclusion: This case report and literature review highlights the necessity of considering ERWT as a potential diagnosis when faced with a patient who has a lumbosacral paraspinal/spinal mass, even if other clinicoradiographic features typical of WT are not identified. We also call to light the need for a standardized treatment regimen for ERWT.

Background Esthesioneuroblastoma (ENB) is a rare neuroectodermal tumor arising from the ol-factory epithelium. This study aimed to identify survival determinants and construct a prognostic web-based nomogram using the Surveillance, Epidemiology, and End Results (SEER) database records. Methods The SEER database, including 18 registries across the nation, was queried for patients diagnosed with ENB between 2000 and 2020. Patient demographics, tumor characteristics, and treatment data were analyzed using Cox Proportional Hazard models and Kaplan-Meier based survival plots. A web-based nomogram was constructed to predict 5- and 10-year survival proba-bilities individualized to a specific patient. Results 941 patients were included in this analysis. Overall survival rates at 1-, 2-, 5-, and 10-years were 89%, 85%, 75%, and 58%, respectively. Multivariable analysis showed worse OS associated with age > 50 (p< 0.001) and tumor size >6.0 cm (p< 0.01). Compared to patients treated with gross total resection (GTR), worse OS was observed in those treated with chemotherapy (CT) only (p< 0.01), CT and radiation (RT, p< 0.01), GTR + CT (p< 0.05), RT only (p< 0.01), or no treatment (p< 0.001). No statistically significant differences in OS were observed for patients treated with GTR + RT, GTR + RT + CT, subtotal resection (STR), or STR with adjuvant therapies when compared to GTR alone. Our web-based nomogram is publicly available (link). This model demonstrated a strong predictive performance, with a C-index of 0.733±0.041 and AUC of 0.747. Conclusion This is currently the largest retrospective cohort study to analyze survival determinants in patients with ENB. This is the first nomogram-based app developed for this specific patient population. We hope this will help guide clinicians to have more meaningful discussions with their patients and employ evidence-based practices.

Background: Esthesioneuroblastoma (ENB) is a rare neuroectodermal tumor arising from the olfactory epithelium. This study aimed to identify survival determinants and construct a prognostic web-based nomogram to provide individualized survival predictions for ENB patients. Methods: Patients diagnosed with ENB between 2000 and 2022 were identified using the Surveillance, Epidemiology, and End Results (SEER) database. Patient demographics, tumor characteristics, and treatment data were analyzed using Cox proportional hazards models and Kaplan–Meier survival curves. A web-based nomogram was constructed to predict 5- and 10-year survival probabilities individualized to a specific patient. Results: Nine hundred and forty-one ENB patients were analyzed. Overall survival (OS) rates at 1-, 2-, 5-, and 10-years were 89%, 85%, 75%, and 58%, respectively. Multivariable analysis revealed that age over 50 (p < 0.001) and tumor size over 6.0 cm (p < 0.01) were independent predictors of worse OS. Compared to patients treated with gross total resection (GTR) alone, worse OS was observed in those treated with chemotherapy (CT) only (p < 0.01), CT and radiation (RT, p < 0.01), GTR + CT (p < 0.05), RT only (p < 0.01), or no treatment (p < 0.001). No statistically significant differences in OS were found between GTR alone and GTR + RT, GTR + RT + CT, subtotal resection (STR), or STR with adjuvant therapies. The resulting ENB prognostic nomogram demonstrated acceptable predictive performance, with a C-index of 0.733 ± 0.041 and AUC of 0.749. Conclusions: Age and tumor size are critical survival determinants in ENB, and GTR continues to be the foundational treatment. The developed prognostic web-based nomogram successfully quantifies individualized survival probabilities, providing clinicians with a tailored tool to guide evidence-based counseling and management for ENB patients.