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. 2022 Jul 20;17(7):e0271334. doi: 10.1371/journal.pone.0271334

“Where am I?” A snapshot of the developmental topographical disorientation among young Italian adults

Laura Piccardi 1,2,*, Massimiliano Palmiero 3, Vincenza Cofini 4, Paola Verde 5,*, Maddalena Boccia 1,6, Liana Palermo 7, Cecilia Guariglia 1,6, Raffaella Nori 8
Editor: David Giofrè9
PMCID: PMC9299294  PMID: 35857777

Abstract

In the last decade, several cases affected by Developmental Topographical Disorientation (DTD) have been described. DTD consists of a neurodevelopmental disorder affecting the ability to orient in the environment despite well-preserved cognitive functions, and in the absence of a brain lesion or other neurological or psychiatric conditions. Described cases showed different impairments in navigational skills ranging from topographic memory deficits to landmark agnosia. All cases lacked a mental representation of the environment that would allow them to use high-order spatial orientation strategies. In addition to the single case studies, a group study performed in Canada showed that the disorder is more widespread than imagined. The present work intends to investigate the occurrence of the disorder in 1,698 young Italian participants. The sample is deliberately composed of individuals aged between 18 and 35 years to exclude people who could manifest the loss of the ability to navigate as a result of an onset of cognitive decline. The sample was collected between 2016 and 2019 using the Qualtrics platform, by which the Familiarity and Spatial Cognitive Style Scale and anamnestic interview were administered. The data showed that the disorder is present in 3% of the sample and that the sense of direction is closely related to town knowledge, navigational strategies adopted, and gender. In general, males use more complex navigational strategies than females, although DTD is more prevalent in males than in females, in line with the already described cases. Finally, the paper discusses which protective factors can reduce DTD onset and which intervention measures should be implemented to prevent the spread of navigational disorders, which severely impact individuals’ autonomy and social relationships.

Introduction

Navigation is the ability to move from one location to the next, following habitual routes and avoiding getting lost [1] in new and familiar environments. Due to its importance, it is not surprising that many cognitive processes (i.e., memory, mental imagery, attention to landmarks and other features, and our perception of directions and distances as well as decision-making, planning, and problem-solving) [26], and multiple brain regions [711] are involved in successful navigation. Specifically, the success of this process also depends on internal and external factors to the individual. The internal factors are of greater interest because they directly affect navigational competence; by consequence, it is possible to better intervene on them in order to plan prevention programs related to navigational disorders. The most important internal factors for navigation are: a) the cognitive predisposition to grasp certain environmental information rather than others [1216]; b) gender [1724]; c) age [2530]; d) professional experience [3136]; e) familiarity with the environment [3740], reflecting the result of repeated exposure to a stimulus or an environment [41, 42]; f) navigational strategies used during navigation [36, 43]; g) psychiatric (e.g., spatial anxiety, depression, agoraphobia: [4446]) and neurologic diseases (Alzheimer’s disease and brain lesions in the navigational brain network: [4750]).

In the present study we focused on an indirect measure of navigation, namely the Sense of Direction (SOD), which is our own perception about navigation ability, in order to understand the critical internal variables that affect it: the demographic factors (e.g., age, gender and education), the degree of familiarity with the environment (e.g., town knowledge), and navigational strategies, which include not only cognitive styles (e.g., landmark, route and survey), but also the preferential mean to explore the environment, that is to say, means of travelling (MoT: active, passive). In addition, we also considered the right-left confusion (RLC), with reflects a pathological condition that can be associated or not with navigational disorders.

At the aim, Siegel and White’s model [51] is used as the theoretical framework. This model suggests that environmental knowledge occurs in three distinct hierarchical steps, namely Landmark (i.e., figurative memory of environmental objects), Route (i.e., a sequence memory of the path connecting environmental objects in an egocentric perspective), and Survey (i.e., map-like representation in an allocentric perspective) knowledge. Following this model, people can be classified in three categories according to the navigational strategies they adopt: landmark (less skilled and get lost easily), route (more skilled at connecting landmarks by verbal labels, such as right, left, behind, ahead, etc.., but less able at finding shortcuts or changing routes after environmental changes), and survey (the most navigational skilled, excellent visual-spatial abilities) users [52, 53]. Notably, both egocentric (route representations) and allocentric (survey representations) frames of reference are needed to specify categorical (non-metric) and coordinate (metric) spatial encodings, which are two different but complementary aspects of spatial cognition [5456]. According to Montello [5], even though familiarity (the repetitive exposure to a particular environment) is more associated with a survey format, spatial knowledge would be not hierarchically organized, but rather would occur in a parallel fashion, depending on the situation [57]. This means that navigation is very complex and that different difficulties can arise when moving through the environment. Furthermore, in the Environmental Knowledge Model [37] emerges as the environmental familiarity allows to perform navigational tasks requiring higher spatial cognitive strategies (e.g. environmental map; perspective changing) even in those individuals with poor spatial orientation skills. In this vein, familiarity with environment enhances wayfinding and reduces wandering and topographical disorientation in individuals suffering from neurocognitive decline [39, 58]. The effect due to familiarity emerges also in healthy ageing given that older people may be better than younger people [59].

Amongst others, the right-left confusion can explain same individual differences in term of SOD [6062]. Indeed, the ability to discriminate left from right from one’s own perspective is useful when individuals explore and recall the environment [60]. In addition, the ways of travelling can also affect the ability to orient oneself in space. Specifically, active travel (e.g., moving by car or bike or on foot) often indicates a better SOD and leads to a better representation of one’s surroundings [63], whereas passive travel (e.g., moving as a passenger in a car, but, taxi, etc…) is likely related to less navigation ability [64]. Yet, Bocchi et al. [12] found that navigational strategies can also affect SOD. Indeed, landmark users can show more difficulties, whereas survey users are the most skilled in solving navigational problems and in travel planning [53, 6567].

Most importantly for the purpose of the present study, navigational difficulties can be associated to a neurodevelopmental disorder that specifically undermines navigational skills. This disorder was described for the first time by Iaria et al. [68] and was named ‘Developmental Topographical Disorientation (DTD)’. Afterwards, numerous people worldwide have been identified as suffering from this condition [6975]. Iaria and Barton [74] demonstrated that DTD is widespread in the Canadian population, finding 120 individuals fulfilling the criteria for a diagnosis of DTD. This led to conclude that DTD is rather widespread in the population and requires targeted interventions by clinical services. In general, people with DTD have normal memory and neuropsychological profiles but show a major cognitive deficit in spatial cognition, and complain of severe problems in navigating on an everyday basis. Specifically, they are unable to use cognitive maps or place-based navigation strategies to find their way around familiar and novel environments. In general, individuals with DTD show a higher impairment of metric (coordinates) than nonmetric (categorical) spatial encoding, and, basing on Siegel and White’s model, they hardly reach a level of route knowledge of the environment, often stopping at a landmark knowledge, highlighting a lack of allocentric representation capacity [76]. By analysing single cases reports, subjects are characterised by a different degree in terms of severity of illness and in terms of navigational impairment. Specifically, Case one had a severe deficit in the formation of the environment mental map [66]; FG had a normal acquisition of environmental information but a specific impairment in the retrieval with a loss of information after 5 minutes [69]; Dr. WAI and LA had both normal acquisition and retrieval of environmental information [70, 73]. FG, LA, and Dr. WAI had deficits in mental representation, mental rotation and mental generation of environmental images. Nobody had difficulty in landmark recognition. LG, instead, was the first case of Landmark Agnosia Development, showing a selective deficit in recognizing landmarks allowing spatial orientation [77]. CF [75] was fully effective in learning and following routes and in building up cognitive maps as well as in recognizing landmarks. However, she performed significantly worse than age and gender-matched controls on the map-following task, namely when she was required to use a map to navigate in a novel environment. In terms of neural correlates, the first studies on DTD cases showed no clear brain structural abnormalities (i.e., [68, 69]). However, using an fMRI landmark sequencing task, Nemmi and co-workers [71] demonstrated that DTD individuals did not show any activation in the navigation brain network, whereas prefrontal areas, known to be involved in processing the sequential order of everyday life actions [78, 79], were normally activated. The decreased functional connectivity between the hippocampus and the prefrontal cortex has also been described [80] and interpreted as a defective functioning of two crucial areas for navigation and decision making. In addition, the rs-fMRI experiment demonstrated aberrant functional connectivity between regions within the default-mode network (DMN), specifically between the medial prefrontal cortex and the posterior cingulate cortex, the medial parietal and temporal cortices.

Thus, the objective of the present study was: to estimate the percentage of the DTD among a convenience sample of Italian adults aged 18–35 years to define soon clinical lines of intervention and a protocol of investigation shared on the national network. To this purpose we investigated the SOD and its correlates. We hypothesize that some internal factors may be correlated to the presence of DTD. Specifically, we expect the gender distribution to be different between males and females: in line with previous described cases we expect to find more males suffering from DTD than females [69, 70, 73, 75, 77]. Moreover, we hypothesize that DTD correlates differently to navigational strategies: we expect that individuals with Survey abilities show higher navigational skills [36] and less probability to have DTD symptoms with respect to people with lower navigational skills (Landmark and Route users). In this vein, we also hypothesize that individuals with DTD show higher right/left confusion and a passive use of means of transport. Consistent with the familiarity effect on SOD [37, 39, 40, 58, 59], we also hypothesize that familiarity, measured as town knowledge, may affect SOD favouring navigation, and could be a protective factor for individuals with DTD who may be able to perform certain spatial orientation tasks in a familiar environment.

Methods

Study design

For this study we conducted an online survey among Italian young people in order to investigate the presence of the DTD and its individual correlates.

Study population

Participants

The eligible study population consisted of all those people without neurological disorders and with an age between 18 and 35 years. A sample of 1,698 participants took part in the experiment. Participants had a full-time education, ranging from 8 to 18 years (mean = 14.80 years, SD = 2.83 years). They were not all university students. Specifically, 81 (4.79%) participants achieved: a secondary school diploma; 842 (49.76%) a high school diploma; 769 (45.45%) a degree or post-degrees. Demographic data of all participants are reported in Table 1. The study was performed according to the ethical principles expressed in the Declaration of Helsinki and it was approved by the Local Ethics Committee (Department of Psychology, University of Bologna, Italy).

Table 1. Demographic data of participants.
N. total 1,698
N. males 635
N. females 1,063
Age Total (years) 24.89 (4.08)
Age males (years) 25.56 (4.25)
Age females (years) 24.48 (3.92)
Education Total (years) 14.80 (2.83)
Males Education (years) 14.39 (2.08)
Females Education (years) 15.04 (2.82)

Note: means (Standard Deviations).

Data collection

Participants from all Italian regions (from North to South, including the Islands) were recruited between 2016 and 2019 using notices on social networks and on bulletin boards of researchers. The advices about the survey were basically spread out by word of mouth and flyers, that were distributed in community meeting points, such as bookshops, cafeterias, public library, and sport clubs. The software Qualtrics (First release: 2005, Provo, Utah, USA, Available at: https://www.qualtrics.com) was used. All participants gave their informed consent before their inclusion in the study.

Measures

A. Anamnesis questionnaire

Participants had to fill in some questions about problems of spatial orientation from an early age, neurological or major psychiatric illness, previous traumatic brain injury, history of learning disabilities, alcohol or drug abuse.

Specifically, with regard to neurological outcomes, participants were asked to specify whether they had experienced head trauma, ischaemic attacks, encephalitis, brain infections, pre-perinatal complications. For the psychiatric side, whether they had suffered or were suffering from depression, anxiety, psychosis, obsessive-compulsive disorder, eating disorder, post-traumatic stress disorder, schizophrenia, phobias. When he/she suffered from it and whether he/she was treated pharmacologically and/or is currently on medication. For substance use, we investigated whether he or she uses alcohol, if so how often, and whether he or she uses or has used drugs (cannabis, amphetamines, cocaine etc.) if so when did he or she use them, how often and which substances.

B. Familiarity and spatial cognitive style scale [81, 82]

Participants had to fill in a series of questions: each item of the scale was a self-referential statement about some aspect of environmental spatial cognition. At the beginning of the scale participants were asked to report demographic information (age, gender and educational level), as well as how they moved around the environment, that is whether they used active or passive means of transport. Specifically, we investigated the use of means of transport, distinguishing active means of transport in which the participant actively drives and moves around the environment (i.e., driving a car; riding a moped; riding a bicycle; riding a motorbike; walking), and passive means of transport in which the participant is led around the environment by others (i.e., being a passenger in a car; using a taxi; using a bus; using a train). For each choice of means of transport, the participant indicated on a scale of 1 to 5 how often they used it. Based on the frequency of use of the various means, the prevalence of active or passive use of the means of transport was defined.

In addition, they were also asked to indicate in the section ‘town knowledge’ to think of a town they knew well even if it was different from the one they lived in.

The scale was divided into the following subscales:

  1. Sense of Direction (SOD) was the summed rating for items concerning the sense of direction, Items 1, 2, 3, 4, 6, 7, 9, 10, 11, and 22 (e.g., Item 1: “How is your ability to read a map?”);

  2. Town Knowledge (TK) was the summed ratings of Items 8, 12, 13, 14, 15, 16, 19, 20, and 21 [e.g., Item 12: “How well do you know (insert the name of the city where you live in)?”];

  3. Spatial cognitive style. To evaluate individual spatial cognitive style, two items were used [Items 17 (a, b, c) and 18 (a, b, c)]. (e.g., given the following item “Try to imagine a route you usually take (e.g., home to work, college to cafeteria…”), participants were asked to evaluate each of the following strategies: a) Landmark—Do you visualize only the landmarks (e.g., your home, the cafeteria. . .)? b) Route—Do you visualize both the landmarks and the route leading to your destination? c) Survey—Do you ever imagine the route as if it were on a map?).

  4. Right-left confusion (RLC). Confusion of right and left self-referents was obtained by Item 5, “In everyday life, do you confound right and left?”.

For each item, participants should circle a number from 1 to 5 to indicate their response: higher numbers correspond to higher ability. In previous works [8284], the overall value of the Cronbach’s alpha for the total scale ranged between .79. and .74, which is good [84], as the test-retest reliability as reported by Nori and Piccardi [81]. In the present sample, Cronbach’s alpha for the total scale was = .71. In Piccardi, Risetti, Nori [82] the internal consistency of SOD and TK was estimated as a combined score (.74). In the present study, the internal consistencies of SOD (.73) and TK (.71) were estimated separately. The full Familiarity and Spatial Cognitive Style Scale is available in the appendix of Piccardi, Risetti and Nori [82].

In this study, the final scores were the means of the item scores for SOD, TK and spatial cognitive styles (landmark, route and survey), whereas ‘right-left confusion’ and the ‘ways of travelling’ were analysed as dichotomous variables. First of all, regarding the item ‘right-left confusion’ (hereafter RLC) we dichotomised as follows: ‘yes’ if the subject responded ‘sometimes’, ‘often’ or ‘always’; ‘no’ if the subject responded ‘never’ or ‘rarely’. Secondly, the ways of travelling, that is the Means of Transport (hereafter MoT) was computed from 5 items: 1) drove a car; 2) other MoT (e.g., bike); 3) only travelled as a passenger; 4) took public transport; 5) only moved on foot. Thus, a subject was defined as an ‘active traveller’ if he/she indicated to move mainly by car, motorbike, bicycle or on foot; a subject was defined as a ‘passive traveller’ if he/she indicated to move mainly as a passenger in a car, taxi, coach, bus, train, air, etc.

Specifically, we classified participants with DTD if they reported a total SOD of 2 Standard Deviations (SD) below the mean (95% CI), as calculated from data collected by Nori and Piccardi [79]. Furthermore, on the basis of the anamnesis questionnaire we also considered the four following diagnostic criteria suggested by Iaria and Barton in [74]: i) getting lost daily or often (1 to 5 times a week) in the most familiar environments; ii) the problem of spatial orientation must be present from an early age; iii) no other cognitive difficulties that may affect daily life activities, and as the last criterion; iv) no known brain lesions, malformation or any condition affecting the central nervous system, with the exception of migraine. In addition, we also took into account for two adding criteria: v) no psychiatric disorders and psychotropic drug use, and vi) substance abuse behaviour.

Statistical methods

Continuous variables were reported as means (Standard Deviation = SD), and categorical factors were reported as percentages. To identify the variables that were significantly related to SOD, the Spearman correlation coefficient was computed for variables measured at least on ordinal scale: Age, Educational Level (low: people with at most a high school diploma; high: degree or post-degrees), TK (score), Landmark (score), Route (score) and Survey (score); and the Point-Biserial Correlation Coefficient for the nominal variables: Gender, RLC (yes/no) and MoT (active/passive). When the correlation coefficients were statistically significant (p < .05) the variables were introduced into a Generalised Linear Regression model (Glm). For the Glm model, an identity link function and a normal family distribution were specified as a linear model, with SOD as the continuous dependent variable and the following model terms as independent variables: Age, TK (score), Landmark (score), Route (score) and Survey (score) as centred covariates; Gender (M/F), RLC (yes/no) and MoT (active/passive) as dummy factors. All interactions with categorical variables were introduced because they explained more variability of SOD (R2 = .337).

In addition, to study the associations between DTD and each socio-demographic variable and each different subscales of the Family and Spatial Cognitive Style Scale, the Chi-square test and One-way analysis of variance, for categorical or continuous data respectively, were used. Specifically, the univariate Logistic regression model, taking DTD as binary dependent variable (yes/no) and one independent variable at a time (Gender, Age, Educational level, RLC, MoT, TK, Landmark, Route, Survey) was carried out to estimate significant predictors, reporting the unadjusted odds ratio (OR) with their 95% Confidence Intervals (95% CI). The significant predictors (p < .05), estimated by univariate analysis, were introduced into a multivariable logistic model, in order to both estimate the odds ratio controlling for the other covariates (adjusted Odds ratios: ORadj), and identify significant protective or risk factors for DTD.

The analyses were performed by using STATA/MP14 software and the jamovi project (2021): jamovi (Version 1.6) Computer-Software, setting alpha to .05. All tests were two-tailed.

Results

General characteristics

Participants reported higher means for the Route scale (3.87, SD = 4), the Landmark (3.58, SD = 3.5) scale, and the SOD scale (3.14, SD = .6), while for the TK scale and the Survey scale the mean values were 2.79, SD = 2.9 and 2.55, SD = 2.5 respectively. Fig 1 shows the different score levels measured by the different subscales.

Fig 1. Box plot of SOD scale and TK, Landmark, Route and Survey subscales.

Fig 1

Correlation between factors investigated and SOD

As reported in Table 2, Spearman correlation analysis showed that SOD was negatively correlated with a weak magnitude level to gender, RLC and Educational level. In addition, SOD was positively correlated with a magnitude ranging from weak to moderate level to Landmark, Route and Survey scores. Moreover, SOD was also positively correlated with a weak magnitude level to Age, MoT and TK. (see [85, 86] for correlation magnitude).

Table 2. Spearman correlation between SOD and investigated factors.

Parameter n Correlation coefficients (rho) degrees of freedom p-value
Age (years) 1698 .069 1696 .004
Gender (M/F) 1698 -.289 1696 < .001
Educational Level (Low/High) 1692 -.045 1690 .066
RLC (yes/no) 1697 -.163 1695 < .001
MoT (active/passive) 1392 .064 1390 .017
TK (score) 1697 .290 1695 < .001
Landmark (score) 1697 .119 1695 < .001
Route (score) 1697 .276 1695 < .001
Survey (score) 1698 .506 1698 < .001

Note: RLC = Right-Left confusion; MoT = Means of Travelling; TK = Town Knowledge.

Using the generalised linear model, the multivariable analysis showed that SOD was related to gender and spatial abilities. In particular, SOD increased because of Survey ability (Beta coefficient = .241; p < .01), whereas decreased in subjects with RCL (Beta coefficient = -.555; p = .009). The model showed that the investigated interactions were not significant (Table 3).

Table 3. Factors related with SOD (multivariable analysis).

95%CI
Independent variables Estimate SE Beta lower upper z p
(Intercept) 3.225 .0636 .098 -.11 .30 50.670 < .001
Gender (male) -.179 .0827 -.293 -.56 -.03 -2.157 .031
TK .199 .0310 .147 .10 .19 6.424 < .001
Landmark .055 .0164 .078 .03 .12 3.366 < .001
Route .082 .0195 .104 .06 .15 4.227 < .001
Survey .241 .0152 .389 .34 .44 15.823 < .001
MoT (active) .082 .0683 .134 -.08 .35 1.200 .230
Age .001 .0034 .002 -.00 .01 .358 .720
RLC (no) -.555 .2122 -.909 -1.59 -.23 -2.610 .009
Gender * MoT -.045 .0888 -.074 -.36 .21 -.510 .610
Gender * RLC .425 .2343 .697 -.06 1.45 1.815 .070
MoT* RLC .418 .2312 .684 -.06 1.43 1.806 .071
Gender*MoT* RLC -.777 .2547 -.617 -1.43 .20 -1.479 .139

Note: RLC = Right-Left confusion; MoT = Means of Travelling; TK = Town Knowledge.

Fifty-four participants met the criteria for DTD, then the percentage of DTD was 3% of our sample (54/1698: 95% CI: 2.4% - 4.0%). The logistic univariate analysis showed that DTD was associated with gender: the males showed a higher risk than females to have DTD (OR: 2.39; 95% CI: 1.2–4.7; p = .009). The risk of DTD was lower in people with higher scores in TK, Route, and Survey scales (Table 4).

Table 4. Factors related to DTD (univariate logistic regression).

DTD
No (n = 1644) Yes (n = 54) P* OR 95%CI
Factors N (%) or mean (SD) N (%) or mean (SD)
Gender
Females 624 (98%) 11 (2%) 1
Males 1020 (96%) 43 (4%) .009 2.39 1.2–4.7
Age (yrs.) 24.9 (4.1) 24.8 (3.9) .9429 1.00 .9–1.1
Educational Level
Low 895 (97%) 28 (3%) 1
High 743 (97%) 26 (3%) .686 1.1 .6–1.9
RLC
No 1276 (97%) 37 (3%) 1
yes 367 (96%) 17 (4%) .114 1.6 .9–2.9
MoT
Passive 184 (96%) 7 (4%) 1
Active 1167 (97%) 34 (3%) .527 .8 .3–1.7
TK (score) -.01 (1.00) -.42 (1.13) .0058 .4 .2–0.8
LANDMARK (score) .03 (0.98) -.11 (0.80) .054 .8 .6–1.1
ROUTE (score) .06 (0.96) -.51 (0.97) < .001 .5 .3-.7
SURVEY (score) .02 (0.99) -.99 (0.70) < .001 .2 .1-.3

*Chi square or Anova test

Note: Sub-totals are not 1,698 because some participants did not fill in some questions. RLC = Right-Left confusion; MoT = Means of Travelling; TK = Town Knowledge.

The multivariable logistic regression analysis confirmed that the risk of DTD was statistically lower in subjects with a higher Survey Ability (ORadj = .26; 95% CI: .16 - .40), adjusted for all factors investigated (Fig 2).

Fig 2. Multivariable association between five risk factors and DTD.

Fig 2

Note: error bars are 95% confidence intervals.

Discussion

The present online investigation aimed to estimate the presence of DTD in a large young Italian sample to better understand the spread of this condition in the population. Thus, we considered the percentage of the DTD in a sample of 1,698 participants, using as a basic criterion 2 standard deviations below the means of the SOD. Then, we also investigated the critical factors predictive of both the SOD and DTD. As in Iaria and Barton’s study [74], we confirmed that the rate of occurrence of the disorder is not a rare condition; rather, it affects 3% of young people undermining their autonomy and ability to work away from family boundaries.

As concerns the predictive factors of SOD, the analyses showed that gender more than educational level is strictly related to SOD. In particular, although females use more landmark-based navigational strategies and complain more difficulties in SOD, males show a higher risk of DTD than females. Indeed, given a glance at the DTD literature, the most of the single cases described are males; this means that although males have better visuospatial and navigational skills than females, they are also the most fragile [69, 70, 72, 75, 77]. Moreover, SOD was related to TK and Survey competencies. Iaria and co-workers [87] clarified that normal navigators can switch from one strategy to another by increasing their familiarity with the environment. This means that it is possible to implement higher navigational skills by acting on environmental knowledge. Also in Nori and Piccardi [37] emerged that when the individual was familiar with the environment, even if s/he generally preferred low navigational strategies, s/he was able to perform more complex navigational tasks in that specific environment, therefore, citizens of Bologna who were familiar with a neighbourhood of the city were able to recognize rotated monuments of the city even though their ability to mentally rotate an object was low [37, 84].

Consistent with these results, we found that TK and the Survey strategy are negatively related to DTD, suggesting that they can be protective factors in counteracting the onset of DTD. In this vein, Bartonek and co-workers [8890] showed that children with cerebral palsy and motor disability manifested differences in topographic working memory as a function of the degree of autonomy to explore the environment, regardless of motor impairment. Our results also showed that the use of advanced navigational strategies (survey strategies) is not associated with the presence of DTD. In line with this finding, we also showed that individuals who achieve high spatial skills do not complain of RLC, which is generally involved in navigational disorders [60]. Consistent with this result, Giancola et al. [36] showed that survey strategies also characterise samples of experienced navigators, such as military pilots. According to Verde et al. [32, 33, 91, 92] this professional category is already selected on the basis of spatial and navigational skills. In addition, the military training would reinforce the survey strategy even more, including all those cognitive processes related to navigation, such as the ability to mentally rotate two and three-dimensional objects [93] or the ability to make directional and metric judgments.

Undoubtedly, the presence of a navigational deficit can also make the subject anxious and more reluctant to explore the environment, so it is difficult to determine how much one comes before the other. This result is particularly interesting in light of Lopez et al.’s [94] study, which showed that the role of the direct experience with exploring hometown on spatial mental representations appeared to be more important in the elderly than in young people. Our sample includes young people, therefore we can imagine that TK and the Survey strategy protects not only seniors from the detrimental effects of ageing on spatial mental representations but also young people in acquiring spatial competence by reducing the risk of navigational disorders.

Given the key role of spatial strategies, our results imply navigational training starting with pre-schoolers, in order to prevent the DTD, such as the one already used in Boccia et al. [95], which allows implementing spatial orientation and autonomy skills from the earliest years of life, starting in kindergarten. The introduction of navigational training in education settings may be useful not only for healthy children but also for children with different types of disabilities (e.g. sensory-motor impairments or acquired brain-damaged or ADHD: [96]), who show several navigational disorders. Furthermore, given that simply enhancing cognitive performance is insufficient to reduce a sense of inadequacy about one’s ability [97], the introduction of training activities specifically designed to improve metacognition would improve self-efficacy in individuals with respect to their SOD and related activities, and by consequence would reduce the risk of DTD. In this vein, De Lisi and Wolford [98] showed children improvements in mental rotation through the daily practice of the popular video game Tetris. This policy of intervention could have important spin-offs increasing also social life.

The current research is not without limitations. First, the study was conducted using an online self-reported survey. In the future it will be important to investigate DTD in presence using a battery of navigational tests. For example, no significant result was found in terms of MoT (active and/or passive movements in the environment). Probably this aspect should be investigated differently by quantifying more precisely the movements and their duration and stratifying the sample by strategies developed. Future work should investigate this component along with environmental characteristics (size of the place where one lives; the presence of distant landmarks; need to travel far to receive medical care or to use school services). Second, individuals with DTD were not tested for other cognitive deficits using a battery of neuropsychological tests, but were only asked to report if they suffered from cognitive disorders. Third, the diagnosis of DTD was not supported by structural imaging data in order to exclude the presence of any brain lesions.

In conclusion, the present study allowed us to identify in a large sample of young Italians the presence of DTD and its occurrence. It has also allowed us to observe protective factors that are associated with good navigational skills and that in the future can be used within protocols for the prevention of the development of spatial orientation disorders, as well as to promote these skills by reducing the gender gap that still emerged in this sample.

Data Availability

All data collected are available at the following link: https://osf.io/qwapm/?view_only=2da5876783c842779a4d1a9380784688 as reported also in the paper.

Funding Statement

The authors received no specific funding for this work.

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Decision Letter 0

David Giofrè

8 Mar 2022

PONE-D-22-02928Where am I? An Italian study on the prevalence in young adults of Developmental Topographical Disorientation.PLOS ONE

Dear Dr. Piccardi,

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3. We noticed you have some minor occurrence of overlapping text with the following previous publication(s), which needs to be addressed:

- https://www.frontiersin.org/articles/10.3389/fphar.2017.00496/full

The text that needs to be addressed involves the Introduction.

In your revision ensure you cite all your sources (including your own works), and quote or rephrase any duplicated text outside the methods section. Further consideration is dependent on these concerns being addressed.

Additional Editor Comments:

Dear dr. Piccardi,

I have now received comments from two reviewers, who are suggesting some more information and clarifications. Reviewer 1 suggested some minor changes, whereas comments from the reviewer 2 are substantial. There are several parts that were not entirely clear and that require some further clarifications, including missing information about your sample and materials.

I also had the opportunity to revise your manuscript, and I also have some suggestions as well.

Generally, please omit the 0 from correlations and from p-values

Table 1. N of males 635 + 1,062 = 1697, one participant is missing and this should be specified.

Line 214 “Spearman correlation analysis”, which is fine, but the authors might also want to explain why they decided to use rank order correlations.

Line 215, to the one hand a non-parametric approach was used, but few lines below generalized linear mixed models were used, which assume linearity. I think that some clarification is needed here.

Line 229 “multivariable logistic model”, this is ok, but I would probably suggest to include more information about this model.

Line 225 and subsequent (3.87±4) I assume that those are SDs, but this is not particularly clear and should be clarified.

Line 230 and subsequent, the statistical approach relies entirely on NHST, while correlations should also be interpreted in terms of magnitude.

Line 235 “and it was not correlated” this is statistically inaccurate, not statistically significant does not necessarily mean no correlation. Please also note that (rho=-0.045; p=0.066) is statistically significant if one tail. Here again the author might want to discuss the correlation in terms of magnitude, .045 is particularly small.

Line 238. Table 1 should be Table 2 (please amend all subsequent tables). Please also report the degrees of freedom for each correlation if the N is different across different measures.

Line 240 “Multivariable analysis” more information are required here, which multivariable analysis? Which kind of regression has been used here? Why interaction terms have been included, is there a rationale for this inclusion? Also, please report CIs and standardized betas for the regression model.

Line 247 “the prevalence of DTD was 3% (54/168:…” Is this accurate? 54/168 = 32.14%

Line 248 "The univariate analysis showed that DTD was associated with gender: the males had a higher risk than females (OR: 2.39; 95%CI:1.2-4.7)," which univariate analysis? Are those logistic regressions? If so, why did you decide to perform univariate analyses rather than including everything in the same model? I think that this might be ok, but some clarification is needed here.

Line 256 “Multivariable analysis” here again please provide some more information.

Line 256 “Survey (ORadj=” Please also clarify why you decided to used adjusted values.

Line 254 “Totals are not 1,698 because of missing data” this is quite unclear and should be clarified.

Figure 2. I guess that error bars are confidence intervals but this should be specified.

[Note: HTML markup is below. Please do not edit.]

Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

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Reviewer #1: Yes

Reviewer #2: Partly

**********

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Reviewer #1: Yes

Reviewer #2: Yes

**********

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The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: Yes

Reviewer #2: No

**********

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PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

Reviewer #2: Yes

**********

5. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: The ms. entitled "Where am I? An Italian study on the prevalence in young adults of Developmental

Topographical Disorientation" is interesting and well written.

Anyway I suggest some integrations as follows.

Review the introduction:

I suggest to add some information regarding how the individuals encode categorical and coordinate spatial information (please see 10.1111/sjop.12633; 10.1016/j.jenvp.2020.101392), and their role in the development of the disorder.

Review the method:

Line 171 pg 8 "All participants gave their informed consent before their inclusion in the study" seems to me a repetition

Please add a table with the items of Familiarity and Spatial Cognitive Style Scale as supplementary materials.

I have not concernes about statistical analysis and results.

Review the Discussion:

Please clarify this sentence:"For such a reason, preferred navigational strategies may not be attributed exclusively to

environmental characteristics, even if they may affect the development of navigational strategies".

Please add the limitations of the study.

Please check some typos throughout the manuscript.

Reviewer #2: 1,698 fill out a questionnaire investigating demographic characteristics, sense of direction, spatial cognitive styles, city of residence knowledge, left-right confusion, way of travelling.

They found that 3% of people met the criteria for Developmental Topographical Disorientation

I think the paper addresses an interesting topic. However, the introduction lack of a coherent line of reasoning, the rationale for the study aims, and material is not completely clear.

Specific points:

INTRODUCTION

Row 49. “Sense of Direction (SOD) is the ability”. I would speak of “Navigation is the ability”. Sense of direction is an indirect measure, it is our own perception about navigation ability.

Row 62. Why are internal factors of greater interest in the present study?

Row 64. I suggest dividing “professional experience” and “familiarity with the environment”. They are both individual factors associated with navigation ability, but I would consider them two distinct factors.

Rows 88-92. I found this paragraph confusing. Did you mean to introduce the “familiarity” with the environment factor? If so, I suggest rephrasing and giving more explanation of the point. If not, please make this paragraph clearer and linked with the line of reasoning.

AIMS AND HYPOTHESES

Row 149. The first aim: “(i) to investigate the Sense of Direction (SOD) and its correlates” is not clear to me. I think this was not introduced well from the title, to the abstract, and to the literature.

Did you mean you wanted to investigate people demographic characteristics, knowledge about their environment, cognitive style with their self-reported sense of direction? And which is the rationale of this choice? Why not considering spatial cognitive abilities for instance.

Furthermore, if this was the aim, the presentation of previous findings on the association between familiar environment knowledge and other factors with SOD merit to be better introduced.

Hypotheses are completely missing. What were you expecting?

Row 155. “Study Design It was a national online survey on the DTD.” What do you mean with national?

PARTICIPANTS

You spoke about prevalence, but is your sample representative of the young population?

I think more information about sample characteristics is necessary.

For instance

- Years of education. Are they all university students? I suggest adding more information on this topic.

- Recruitment. How was the sample recruited? You stated “awareness campaign”. But are many of them university (psychology) students?

- The provenance of participants? You briefly stated this in discussion, I think information should be added in Participants section

MATERIAL

You used a questionnaire asking about demographic variables, sense of direction, their own city of residence knowledge, spatial cognitive styles, left-right confusion. Then you mentioned also the way of travelling.

I am missing some points, and I think this deserves to be more clearly presented.

- What are the Cronbach alphas of the current sample?

- Does the sense of direction refer to both new and familiar environments?

- City knowledge also included how many years they lived in?

- Which is the rationale to measure left-right confusion? And ways of travelling? They are not presented in the introduction.

- Information about scoring? (Figure 1 depict a 1-5 point scale)

Row 235. “disorientation”. What do you mean?

Row 247. I am missing a point, what are the criteria?

Rows 300-305 Why are you referring to older adults? Please make this point clearer

**********

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PLoS One. 2022 Jul 20;17(7):e0271334. doi: 10.1371/journal.pone.0271334.r002

Author response to Decision Letter 0


19 May 2022

In the manuscript, all changes are highlighted in yellow.

PONE-D-22-02928

Where am I? An Italian study on the prevalence in young adults of Developmental Topographical Disorientation.

PLOS ONE

Dear Dr. Piccardi,

Thank you for submitting your manuscript to PLOS ONE. After careful consideration, we feel that it has merit but does not fully meet PLOS ONE’s publication criteria as it currently stands. Therefore, we invite you to submit a revised version of the manuscript that addresses the points raised during the review process. Please pay particular attention to my comments below.

Please submit your revised manuscript by Apr 22 2022 11:59PM. If you will need more time than this to complete your revisions, please reply to this message or contact the journal office at plosone@plos.org. When you're ready to submit your revision, log on to https://www.editorialmanager.com/pone/ and select the 'Submissions Needing Revision' folder to locate your manuscript file.

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If you would like to make changes to your financial disclosure, please include your updated statement in your cover letter. Guidelines for resubmitting your figure files are available below the reviewer comments at the end of this letter.

If applicable, we recommend that you deposit your laboratory protocols in protocols.io to enhance the reproducibility of your results. Protocols.io assigns your protocol its own identifier (DOI) so that it can be cited independently in the future. For instructions see: https://journals.plos.org/plosone/s/submission-guidelines#loc-laboratory-protocols. Additionally, PLOS ONE offers an option for publishing peer-reviewed Lab Protocol articles, which describe protocols hosted on protocols.io. Read more information on sharing protocols at https://plos.org/protocols?utm_medium=editorial-email&utm_source=authorletters&utm_campaign=protocols.

We look forward to receiving your revised manuscript.

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David Giofrè, Ph.D.

Academic Editor

PLOS ONE

Journal Requirements:

When submitting your revision, we need you to address these additional requirements.

1. Please ensure that your manuscript meets PLOS ONE's style requirements, including those for file naming. The PLOS ONE style templates can be found at

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2. We note that you have stated that you will provide repository information for your data at acceptance. Should your manuscript be accepted for publication, we will hold it until you provide the relevant accession numbers or DOIs necessary to access your data. If you wish to make changes to your Data Availability statement, please describe these changes in your cover letter and we will update your Data Availability statement to reflect the information you provide.

3. We noticed you have some minor occurrence of overlapping text with the following previous publication(s), which needs to be addressed:

- https://www.frontiersin.org/articles/10.3389/fphar.2017.00496/full

The text that needs to be addressed involves the Introduction.

In your revision ensure you cite all your sources (including your own works), and quote or rephrase any duplicated text outside the methods section. Further consideration is dependent on these concerns being addressed.

Revisiting the introduction following the Reviewers’ comments we solved the issue about of the overlapping text. We also checked the duplicated text outside the method section.

Additional Editor Comments:

Dear dr. Piccardi,

I have now received comments from two reviewers, who are suggesting some more information and clarifications. Reviewer 1 suggested some minor changes, whereas comments from the reviewer 2 are substantial. There are several parts that were not entirely clear and that require some further clarifications, including missing information about your sample and materials.

I also had the opportunity to revise your manuscript, and I also have some suggestions as well.

Generally, please omit the 0 from correlations and from p-values

Done.

Table 1. N of males 635 + 1,062 = 1697, one participant is missing and this should be specified.

We apologize for this, there was a distraction error. We reported the correct number of females.

Line 214 “Spearman correlation analysis”, which is fine, but the authors might also want to explain why they decided to use rank order correlations.

Thank you for your suggestion. We tried to better report the choice of the correlation’s coefficients used. Our choice is related to the type of the investigated variables. Some of them in fact are binary or ordinal variables and other continuous. We used the Spearman's rank-order correlation, that is the nonparametric version of the Pearson product-moment correlation, to analyze the relationships between two variables measured on at least an ordinal scale. We specify the choice on the manuscript, also reporting that we used the point-biserial correlation to investigate the correlation between binary variables (the results were the same using Spearman).

Line 215, to the one hand a non-parametric approach was used, but few lines below generalized linear mixed models were used, which assume linearity. I think that some clarification is needed here.

Please, as reported above we explored the relationship between SOD and the other variables using non-parametric approaches (correlations) with respect to the type of the variables (continuous, ordinal and categorical). However, please consider that we did not use the generalized linear mixed models. In order to assess which independent variable to enter into the model used (Generalized Linear Model), we investigated the relation between the SOD variable and the others variables using the p-values criterion (p<0.05). “Generalized Linear Model” is a generalization of the general linear model. The general linear model assumes linearity, whereas the relationship in the generalized linear model between dependent variable and independent variables can be non-linear [Generalized Linear Models and Extensions, Fourth Edition. James W. Hardin and Joseph M. Hilbe.STATA PRESS, 2018].

We used generalized linear model specifying an identity link function and a normal family distribution than it is equivalent to a (general) linear model, so we calculated the coefficients , adding all interactions with categorical factors (gender, travel and disorientation) because the interactions helped explaining more variability of Y (R2=.334 of the model with the main effects only vs R2 =.337 of the model with interactions).

We included this information in the manuscript.

Line 229 “multivariable logistic model”, this is ok, but I would probably suggest to include more information about this model.

Thank you for suggestion we reported it on the manuscript as you asked.

Line 225 and subsequent (3.87±4) I assume that those are SDs, but this is not particularly clear and should be clarified.

Thank you for your suggestion; we now reported standard deviations into parenthesis and we deleted the symbol ±.

Line 230 and subsequent, the statistical approach relies entirely on NHST, while correlations should also be interpreted in terms of magnitude.

WE investigate the relation between the SOD variable and the others using the p-values (p<.05) to assess the statistical significance of the result to enter the independent variables into the glm model.

However, as suggested by the Reviewer we added information about the magnitude of correlations in the manuscript.

Line 235 “and it was not correlated” this is statistically inaccurate, not statistically significant does not necessarily mean no correlation. Please also note that (rho=-0.045; p=0.066) is statistically significant if one tail. Here again the author might want to discuss the correlation in terms of magnitude, .045 is particularly small.

Thank you for your comment. We agree with you that not statistically significant does not necessarily mean no correlation, and the correlation should be discussed in terms of magnitude, but we used the p-values approach to choose the independent variables in our model. All tests were two-sided, we reported it on the manuscript

However, we included in the manuscript both significance and the magnitude issues, in the correlation section.

Line 238. Table 1 should be Table 2 (please amend all subsequent tables). Please also report the degrees of freedom for each correlation if the N is different across different measures.

Thank you, We reported consecutive numbers for all tables, and we reported n and degrees of freedom for each correlation in table 2

Line 240 “Multivariable analysis” more information are required here, which multivariable analysis? Which kind of regression has been used here? Why interaction terms have been included, is there a rationale for this inclusion? Also, please report CIs and standardized betas for the regression model.

We have integrated the information about glm as you requested in method’s section reporting standardized beta and CIs on table 3 and specifying the type of regression model in the manuscript.

Line 247 “the prevalence of DTD was 3% (54/168:…” Is this accurate? 54/168 = 32.14%

We apologize for this, the percentage is 3%: 54/1698, there was a typing error. We have corrected it.

Line 248 "The univariate analysis showed that DTD was associated with gender: the males had a higher risk than females (OR: 2.39; 95%CI:1.2-4.7)," which univariate analysis? Are those logistic regressions? If so, why did you decide to perform univariate analyses rather than including everything in the same model? I think that this might be ok, but some clarification is needed here.

Thank you for your suggestions, we revised the method’s section reporting more information about it. First, we used logistic univariate regression, to reduce the number of independent variables to include in our model. We included in the multiple logistic model only the factors (predictors) that were statistically significant (p<0.05 and Odds ratio different from 1, that is called the “crude OR” or unadjusted OR). An adjusted odds ratio is an odds ratio that has been adjusted to account for other predictor variables in a model.

Line 256 “Multivariable analysis” here again please provide some more information.

Thank you for your suggestion. We added it.

Line 256 “Survey (ORadj=” Please also clarify why you decided to used adjusted values.

Thank you. We have attempted to clarify it in the text.

Line 254 “Totals are not 1,698 because of missing data” this is quite unclear and should be clarified.

We clarified in the note of the table 4 that some subtotals are different from total count because some participants did not fill in all questions, which is why the subtotal for some variables is different. For correctness we reported this information.

Figure 2. I guess that error bars are confidence intervals but this should be specified.

Done, thank you.

Reviewer 2:

Review the introduction:

I suggest to add some information regarding how the individuals encode categorical and coordinate spatial information (please see 10.1111/sjop.12633; 10.1016/j.jenvp.2020.101392), and their role in the development of the disorder.

We added the reviewer’s suggestion, including more information about individuals’ categorical and coordinate spatial encoding, as well as an interpretation of the role of these types of encoding in the development of the DTD. Indeed, in general, individuals with DTD show a higher impairment of metric (coordinates) than nonmetric (categorical) spatial encoding, and, basing on Siegel and White's model, they hardly reach a level of route knowledge of the environment, often stopping at a landmark knowledge, highlighting a lack of allocentric representation capacity.

Review the method:

Line 171 pg 8 "All participants gave their informed consent before their inclusion in the study" seems to me a repetition

Done

Please add a table with the items of Familiarity and Spatial Cognitive Style Scale as supplementary materials.

The full items of Familiarity and Spatial Cognitive Style Scale are already published in the Appendix of the paper “Piccardi, L., Risetti, M., & Nori, R. (2011). Familiarity and environmentalrepresentations of a city: a self-report study. Psychological reports, 109(1), 309-326.” Therefore, wecouldnotreproduceitagain, butwe indicate this in the method.

I have not concernes about statistical analysis and results.

Review the Discussion:

Please clarify this sentence:"For such a reason, preferred navigational strategies may not be attributed exclusively to environmental characteristics, even if they may affect the development of navigational strategies".

We dropped the sentence because we realized that it was not clear and confounded the reading of the manuscript.

Please add the limitations of the study.

We added the limitations of the study as follows, before the last paragraph: ‘The current research is not without limitations. First, the study was conducted using an online self-reported survey. In the future it will be important to investigate DTD in presence using a battery of navigational tests. Second, individuals with DTD were not tested for other cognitive deficits using a battery of neuropsychological tests, but were only asked to report if they suffered from cognitive disorders. Third, the diagnosis of DTD was not supported by structural imaging data in order to exclude the presence of any brain lesions’.

Please check some typos throughout the manuscript.

We checked and corrected all typos throughout the manuscript.

Reviewer #2:

I think the paperaddresses an interestingtopic. However, the introductionlack of a coherent line of reasoning, the rationale for the studyaims, and materialisnotcompletelyclear.

Following the Reviewers’ directionswefullyreviseted the introduction and the rationale and weclarified the material.

Specific points:

INTRODUCTION

Row 49. “Sense of Direction (SOD) is the ability”. I would speak of “Navigation is the ability”. Sense of direction is an indirect measure, it is our own perception about navigation ability.

We corrected this point and specified that SOD is an indirect measure of the navigational ability.

Row 62. Why are internal factors of greater interest in the present study?

We specified at the beginning of the introduction that internal factors are important because they affect navigational competence; by consequence it is possible to better intervene on them in order to plan prevention programs related to navigational disorders. We then clarified which internal factors were used for our study, namely demographic factors (e.g., age, gender and education), the degree of familiarity with the environment (e.g., town knowledge), and navigational strategies, which include not only cognitive styles (e.g., landmark, route and survey), but also the preferential mean of movement (active, passive). In addition, we also considered the right-left confusion (RLC) as a pathological factor that can be associated or not with navigational disorders.

Row 64. I suggest dividing “professional experience” and “familiarity with the environment”. They are both individual factors associated with navigation ability, but I would consider them two distinct factors.

As suggested by the reviewer, when listing the internal factors, we divided ‘professional experience’ from ‘familiarity with the environment’, supporting the two factors with specific bibliography.

Rows 88-92. I found this paragraph confusing. Did you mean to introduce the “familiarity” with the environment factor? If so, I suggest rephrasing and giving more explanation of the point. If not, please make this paragraph clearer and linked with the line of reasoning.

Well, no, we refer to familiarity still as an internal factor. Indeed, following Craig et al. (2012, p. 2), ‘Intuitively, familiarity is simply the result of repeated exposure to a particular stimulus or environment. Indeed, this intuitive interpretation of familiarity is the basis of the mere-exposure effect put forward by Zajonc [10], which theorises that preference for objects can be induced by repeated exposure. This kind of familiarity might be called (after Zajonc), “objective familiarity,” or “actual familiarity”—a simple correlate of the number of times a person has seen a particular object or scene’. Then, we corrected the sentence as follows in order to avoid confusion: ‘Montello [5] reported that familiarity is more associated with a survey format (similar to cognitive maps)’. In addition, given the general restructuration of the introduction, as suggested in a previous comment, we better connected the paragraph to the rest of the text.

AIMS AND HYPOTHESES

Row 149. The first aim: “(i) to investigate the Sense of Direction (SOD) and its correlates” is not clear to me. I think this was not introduced well from the title, to the abstract, and to the literature.

Did you mean you wanted to investigate people demographic characteristics, knowledge about their environment, cognitive style with their self-reported sense of direction? And which is the rationale of this choice? Why not considering spatial cognitive abilities for instance.

Furthermore, if this was the aim, the presentation of previous findings on the association between familiar environment knowledge and other factors with SOD merit to be better introduced.

Thank to the Reviewer’s observation, we clarified the objective,which is the investigation of DTD, and accordingly we introduced the hypotheses. Concerning spatial abilities, we did not test them through paper and pencil tests because we were interested to detect DTD population, which is assessed using the Sense of Direction measure, and not by spatial tasks measuring spatial abilities.As described in the paper the DTD is measured considering 2 standard deviations below the mean of the sense of direction. Nevertheless,we believe that assessing spatial tasks in DTD deserves further studies.

Hypotheses are completely missing. What were you expecting?

We thank Reviewer for his/her suggestions, we now add at the end of the introduction the objectives also the hypotheses.Specifically, our objective and hypotheses are related to the DTD.

Row 155. “Study Design It was a national online survey on the DTD.” What do you mean with national?

Thank you for this comment, we meant a survey conducted by the Italian Population. We corrected this point also in the manuscript.

PARTICIPANTS

You spoke about prevalence, but is your sample representative of the young population?

Thanks to the Reviewer’s suggestion we decided to substitute the term ‘prevalence’ with the term ‘percentage’, which is more correct considering the size of our sample.

I think more information about sample characteristics is necessary.

For instance

- Years of education. Are they all university students? I suggest adding more information on this topic.

We specified in the manuscript the % of their educational levels as follows: Participants had a full-time education, ranging from 8 to 18 years (mean = 14.80 years, SD = 2.83 years). This means that they were not all university students. Specifically, 4.79% of the participants achieved a secondary school diploma, 49.76% achieved a high school diploma, and 45.45% achieved a degree or post-degree education level.

- Recruitment. How was the sample recruited? You stated “awareness campaign”. But are many of them university (psychology) students?

Participants were recruited through notices on social networks and on bulletin boards of researchers. We specified that ‘the advices about the survey were basically spread out by word of mouth and flyers, that were distributed in community meeting points, such as bookshops, cafeterias, public library, and sport clubs’.

The provenance of participants? You briefly stated this in discussion, I think information should be added in Participants section

We recruited participants from all Italian regions, including the Islands. We specified it in the participants’ section.

MATERIAL

You used a questionnaire asking about demographic variables, sense of direction, their own city of residence knowledge, spatial cognitive styles, left-right confusion. Then you mentioned also the way of travelling.

In the same questionnaire along with demographic characteristics, the participants were also asked how they moved around the environment, whether they used active or passive means of transport. Specifically, we investigated the use of means of transport, distinguishing active means of transport in which the participant actively drives and moves around the environment and passive means of transport in which the participant is led around the environment by others. We have replaced the term travel with active and/or passive means of transport to avoid misunderstandings.

I am missing some points, and I think this deserves to be more clearly presented.

- What are the Cronbach alphas of the current sample?

We reported the Cronbach’s alpha for the total scale for the sample we used, and it was = .71.

- Does the sense of direction refer to both new and familiar environments?

Yes, it refers to both new and familiar environments. We specified it in the manuscript the first time we introduced the concept of SOD.

- City knowledge also included how many years they lived in?

When we refer to the city (town) knowledge, all questions concern a town that the subject knows well. In fact, at the beginning of the questionnaire they were expressly asked to imagine a very familiar town and to indicate its name, while for the SOD the questions concern more or less familiar places.

- Which is the rationale to measure left-right confusion? And ways of travelling? They are not presented in the introduction.

Regarding the right/left confusion, the rational is that it is one of the visuo-spatial disorders and in our case is a kind of anamnestic question because we believe that this disorder affects the ability to verbally label left and right correctly but does not have an effect on the mental representation of space.

The ways of traveling, instead, are associated with the ability to orient oneself in space, which improves with practice. Indeed, active travel often indicates a better sense of direction and leads to a better representation of one's surroundings. In general, people with less navigation ability tend to move less and move with others in a passive manner.

We included this rationale in the introduction.

- Information about scoring? (Figure 1 depict a 1-5 point scale)

We reported in the ‘data collection’ section that ‘for each item, participants should circle a number from 1 to 5 to indicate their response: higher numbers correspond to higher ability. Then, the final scores were the means of the item scores’. Thus, the scoring involved the average of the item scores for each subscale (route, survey, landmark, SOD and TK).

Row 235. “disorientation”. What do you mean?

We mean ‘right-left confusion’. We Specified it throughout the manuscript.

Row 247. I am missing a point, what are the criteria?

We thank Reviewer for his/her comment. Indeed, we had not been clear in making explicit the diagnostic criteria for the disorder. Now, in addition to pointing out what score the subjects had to obtain in order to be considered affected by DTD, we have explicitly listed the international criteria (Iaria and Barton 2010) for the disorder and the two additional criteria that we have considered in this paper.

Rows 300-305 Why are you referring to older adults? Please make this point clearer

By making revisions to the manuscript, this point is no longer present.

________________________________________

Attachment

Submitted filename: Response to Reviewers.docx

Decision Letter 1

David Giofrè

21 Jun 2022

PONE-D-22-02928R1“Where am I?” A snapshot of the Developmental Topographical Disorientation among young Italian adults.PLOS ONE

Dear Dr. Piccardi,

Thank you for submitting your manuscript to PLOS ONE. After careful consideration, we feel that it has merit but does not fully meet PLOS ONE’s publication criteria as it currently stands. Therefore, we invite you to submit a revised version of the manuscript that addresses the points raised during the review process.

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We look forward to receiving your revised manuscript.

Kind regards,

David Giofrè, Ph.D.

Academic Editor

PLOS ONE

Journal Requirements:

Please review your reference list to ensure that it is complete and correct. If you have cited papers that have been retracted, please include the rationale for doing so in the manuscript text, or remove these references and replace them with relevant current references. Any changes to the reference list should be mentioned in the rebuttal letter that accompanies your revised manuscript. If you need to cite a retracted article, indicate the article’s retracted status in the References list and also include a citation and full reference for the retraction notice.

Additional Editor Comments:

Dear Dr. Piccardi,

you can see that the reviewers have now commented on your paper. One reviewer suggests accepting the paper as it stands now, while the other raises some concerns. By my own reading of the paper, I noticed that the manuscript has greatly improved. Therefore, I am encouraging you to revise the paper according to the instructions provided by the second reviewer.

Best wishes,

David Giofrè

[Note: HTML markup is below. Please do not edit.]

Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation.

Reviewer #1: All comments have been addressed

Reviewer #2: (No Response)

**********

2. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: Yes

Reviewer #2: Partly

**********

3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

Reviewer #2: Yes

**********

4. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: Yes

Reviewer #2: Yes

**********

5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

Reviewer #2: Yes

**********

6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: The authors replied all the questions raised. The manuscript can be accepted in the present form.

Reviewer #2: The authors have addressed almost all my previous concern sufficiently well.

I have a few additional comments.

- I found the description of the questionnaires a bit confusing. You present them as part of a unique “familiarity and spatial cognitive style scale” questionnaire (pages 8-9). If I am understanding, this questionnaire was used as a total score in other studies, while here you considered the SOD part as your dependent variable, and town knowledge, cognitive styles, left/right confusion, and means of transport as your predictors.

Therefore, I suggest presenting the materials as follows

A) Anamnesis Questionnaire.

B) Demographic questionnaire (age, gender and educational level)

C) Means of transport questions (they are not numbered items of the scale, so I suppose they can be described as separately)

D) And then the “familiarity and spatial cognitive style scale” investigating in the order: sense of direction, left/right confusion, town knowledge, spatial cognitive styles.

- And for each part include all the information together (e.g., for the means of transport, you stated the items below in the text, but I would organize a dedicated paragraph in which finding all information about the measure, including scoring)

- For the Anamnesis Questionnaire could you add more explicitly what you asked the participants?

- Cronbach’s alphas of the subscales (SOD, town knowledge, spatial cognitive styles, ..) should be reported given the total score is not used here.

- Which is the rationale of considering the right/left dichotomous given data are collected in a 5-point Likert scale? Did the analyses differ when considering it not dichotomic?

- Familiarity. In the first revision, I pointed out that “Furthermore, if this was the aim, the presentation of previous findings on the association between familiar environment knowledge and other factors with SOD merit to be better introduced.”

But I do not think the authors have addressed this point. Familiarity is only described as part of the Siegel and White model to reach survey knowledge, but neither specific rationale (supported by literature) for measuring it and hypothesis are presented.

Specifically, why consider town knowledge as a predictor of SOD? This could be ok, if supported.

But I was also wondering, why not add the town knowledge score together with SOD, given that DTD involves impaired orientation both in familiar and unfamiliar environments? (as you stated: “In general, people with DTD have normal memory and neuropsychological profiles, but show a major cognitive deficit in spatial cognition and complain of severe problems in navigation on an everyday basis. Specifically, they are unable to use cognitive maps or place-based navigation strategies to find their way around familiar and novel environments”).

**********

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Reviewer #1: No

Reviewer #2: No

**********

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PLoS One. 2022 Jul 20;17(7):e0271334. doi: 10.1371/journal.pone.0271334.r004

Author response to Decision Letter 1


25 Jun 2022

In the manuscript, all changes are highlighted in yellow.

PONE-D-22-02928R1

“Where am I?” A snapshot of the Developmental Topographical Disorientation among young Italian adults.

Journal Requirements:

Please review your reference list to ensure that it is complete and correct. If you have cited papers that have been retracted, please include the rationale for doing so in the manuscript text, or remove these references and replace them with relevant current references. Any changes to the reference list should be mentioned in the rebuttal letter that accompanies your revised manuscript. If you need to cite a retracted article, indicate the article’s retracted status in the References list and also include a citation and full reference for the retraction notice.

Reply: the reference list has been checked.

Additional Editor Comments:

Dear Dr. Piccardi,

you can see that the reviewers have now commented on your paper. One reviewer suggests accepting the paper as it stands now, while the other raises some concerns. By my own reading of the paper, I noticed that the manuscript has greatly improved. Therefore, I am encouraging you to revise the paper according to the instructions provided by the second reviewer.

Best wishes,

David Giofrè

Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation.

Reviewer #1: All comments have been addressed

Reviewer #2: (No Response)

________________________________________

2. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #1: Yes

Reviewer #2: Partly

________________________________________

3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #1: Yes

Reviewer #2: Yes

________________________________________

4. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #1: Yes

Reviewer #2: Yes

________________________________________

5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #1: Yes

Reviewer #2: Yes

________________________________________

6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #1: The authors replied all the questions raised. The manuscript can be accepted in the present form.

Reviewer #2: The authors have addressed almost all my previous concern sufficiently well.

I have a few additional comments.

- I found the description of the questionnaires a bit confusing. You present them as part of a unique “familiarity and spatial cognitive style scale” questionnaire (pages 8-9). If I am understanding, this questionnaire was used as a total score in other studies, while here you considered the SOD part as your dependent variable, and town knowledge, cognitive styles, left/right confusion, and means of transport as your predictors.

Reply: We would like to thank the Reviewer who allowed us to clarify the description of the instrument in the manuscript. First, we substituted the word “scale” for “questionnaire” and “subscales” instead of “sections”. We also add some more information about MoT and reliability of the subscales as asked by the Reviewer in the following concerns. Concerning the scale is always administered in its entirety and provides a total score, but it also provides scores for SOD; town knowledge; right/left confusion and spatial cognitive styles which were particularly useful in the present work to better describe the navigational skills of the population under investigation. For this reason, we considered these aspects in the analyses. They have also been taken into account in other works (e.g., Piccardi et al 2011; Nori and Piccardi 2012), while in other studies only SOD was considered in order to exclude the presence of individuals with navigational disorders from the sample.

Therefore, I suggest presenting the materials as follows

A) Anamnesis Questionnaire.

B) Demographic questionnaire (age, gender and educational level)

C) Means of transport questions (they are not numbered items of the scale, so I suppose they can be described as separately)

D) And then the “familiarity and spatial cognitive style scale” investigating in the order: sense of direction, left/right confusion, town knowledge, spatial cognitive styles.

- And for each part include all the information together (e.g., for the means of transport, you stated the items below in the text, but I would organize a dedicated paragraph in which finding all information about the measure, including scoring)

Reply: We have now divided the instruments into A (Anamnesis Questionnaire) and B (familiarity and spatial cognitive style scale). With respect to demographic information and means of transport are part of the scale and cannot be described separately. However, we have added information to help the reader better understand how they are articulated, then in the text we also refer to the scale that is in the appendix of the work of Piccardi et al 2011, which is the reason why we cannot include it in the supplementary materials of this work. We hope that the added information can make the scale clearer.

- For the Anamnesis Questionnaire could you add more explicitly what you asked the participants?

Reply: We add some more information about questions we asked participants

- Cronbach’s alphas of the subscales (SOD, town knowledge, spatial cognitive styles, ..) should be reported given the total score is not used here.

Reply: We now provided the internal consistencies of SOD and TK to compare the values with the original studies (i.e. Piccardi, Risetti and Nori 2011). Both in Nori and Piccardi (2012) and Piccardi, Risetti and Nori (2011) the only reliability measures concerned the total score and TK and SOD, but not the other subscales, for this reason, lacking previous comparative values, we did not proceed to report the internal consistency of spatial cognitive styles.

- Which is the rationale of considering the right/left dichotomous given data are collected in a 5-point Likert scale? Did the analyses differ when considering it not dichotomic?

Reply: We checked the Spearman correlation between the right/left confusion and SOD using RLC as a continuous variable, and we found a similar result (Rho= -.12; p< .001) obtained using dichotomic classification. This let us to assume that also the subsequent analyses reported in the paper yield similar results. However, we have decided to transform RLC from continuous to dichotomic because representing the presence/absence of a disorder the dichotomic subdivision makes stronger the result.

- Familiarity. In the first revision, I pointed out that “Furthermore, if this was the aim, the presentation of previous findings on the association between familiar environment knowledge and other factors with SOD merit to be better introduced.”

But I do not think the authors have addressed this point. Familiarity is only described as part of the Siegel and White model to reach survey knowledge, but neither specific rationale (supported by literature) for measuring it and hypothesis are presented.

Specifically, why consider town knowledge as a predictor of SOD? This could be ok, if supported.

Reply: We thank Reviewer for raising this point that we missed. We now explained better the reason why we insert TK as predictor, supporting our hypothesis with literature and a theoretical model.

But I was also wondering, why not add the town knowledge score together with SOD, given that DTD involves impaired orientation both in familiar and unfamiliar environments? (as you stated: “In general, people with DTD have normal memory and neuropsychological profiles, but show a major cognitive deficit in spatial cognition and complain of severe problems in navigation on an everyday basis. Specifically, they are unable to use cognitive maps or place-based navigation strategies to find their way around familiar and novel environments”).

Reply: We keep SOD and TK separate because they represent two independent albeit closely related constructs, i.e. it is true that increased familiarity with the environment produces positive effects on orientation, but as the Reviewer rightly points out in individuals with DTD the disorientation also occurs in highly familiar environments. It is now reasonable to see whether it can also represent a protective effect for them as we have now pointed out in the working hypothesis. Combining the two constructs, however, risks losing important information.

________________________________________

Attachment

Submitted filename: Rebuttal Letter Rev 2#.docx

Decision Letter 2

David Giofrè

29 Jun 2022

“Where am I?” A snapshot of the Developmental Topographical Disorientation among young Italian adults.

PONE-D-22-02928R2

Dear Dr. Piccardi,

We’re pleased to inform you that your manuscript has been judged scientifically suitable for publication and will be formally accepted for publication once it meets all outstanding technical requirements.

Within one week, you’ll receive an e-mail detailing the required amendments. When these have been addressed, you’ll receive a formal acceptance letter and your manuscript will be scheduled for publication.

An invoice for payment will follow shortly after the formal acceptance. To ensure an efficient process, please log into Editorial Manager at http://www.editorialmanager.com/pone/, click the 'Update My Information' link at the top of the page, and double check that your user information is up-to-date. If you have any billing related questions, please contact our Author Billing department directly at authorbilling@plos.org.

If your institution or institutions have a press office, please notify them about your upcoming paper to help maximize its impact. If they’ll be preparing press materials, please inform our press team as soon as possible -- no later than 48 hours after receiving the formal acceptance. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information, please contact onepress@plos.org.

Kind regards,

David Giofrè, Ph.D.

Academic Editor

PLOS ONE

Additional Editor Comments (optional):

Reviewers' comments:

Reviewer's Responses to Questions

Comments to the Author

1. If the authors have adequately addressed your comments raised in a previous round of review and you feel that this manuscript is now acceptable for publication, you may indicate that here to bypass the “Comments to the Author” section, enter your conflict of interest statement in the “Confidential to Editor” section, and submit your "Accept" recommendation.

Reviewer #2: All comments have been addressed

**********

2. Is the manuscript technically sound, and do the data support the conclusions?

The manuscript must describe a technically sound piece of scientific research with data that supports the conclusions. Experiments must have been conducted rigorously, with appropriate controls, replication, and sample sizes. The conclusions must be drawn appropriately based on the data presented.

Reviewer #2: Yes

**********

3. Has the statistical analysis been performed appropriately and rigorously?

Reviewer #2: Yes

**********

4. Have the authors made all data underlying the findings in their manuscript fully available?

The PLOS Data policy requires authors to make all data underlying the findings described in their manuscript fully available without restriction, with rare exception (please refer to the Data Availability Statement in the manuscript PDF file). The data should be provided as part of the manuscript or its supporting information, or deposited to a public repository. For example, in addition to summary statistics, the data points behind means, medians and variance measures should be available. If there are restrictions on publicly sharing data—e.g. participant privacy or use of data from a third party—those must be specified.

Reviewer #2: Yes

**********

5. Is the manuscript presented in an intelligible fashion and written in standard English?

PLOS ONE does not copyedit accepted manuscripts, so the language in submitted articles must be clear, correct, and unambiguous. Any typographical or grammatical errors should be corrected at revision, so please note any specific errors here.

Reviewer #2: Yes

**********

6. Review Comments to the Author

Please use the space provided to explain your answers to the questions above. You may also include additional comments for the author, including concerns about dual publication, research ethics, or publication ethics. (Please upload your review as an attachment if it exceeds 20,000 characters)

Reviewer #2: The paper has improved, the authors have addressed my previous issues and I have no more suggestions.

**********

7. PLOS authors have the option to publish the peer review history of their article (what does this mean?). If published, this will include your full peer review and any attached files.

If you choose “no”, your identity will remain anonymous but your review may still be made public.

Do you want your identity to be public for this peer review? For information about this choice, including consent withdrawal, please see our Privacy Policy.

Reviewer #2: No

**********

Acceptance letter

David Giofrè

7 Jul 2022

PONE-D-22-02928R2

“Where am I?” A snapshot of the Developmental Topographical Disorientation among young Italian adults.

Dear Dr. Piccardi:

I'm pleased to inform you that your manuscript has been deemed suitable for publication in PLOS ONE. Congratulations! Your manuscript is now with our production department.

If your institution or institutions have a press office, please let them know about your upcoming paper now to help maximize its impact. If they'll be preparing press materials, please inform our press team within the next 48 hours. Your manuscript will remain under strict press embargo until 2 pm Eastern Time on the date of publication. For more information please contact onepress@plos.org.

If we can help with anything else, please email us at plosone@plos.org.

Thank you for submitting your work to PLOS ONE and supporting open access.

Kind regards,

PLOS ONE Editorial Office Staff

on behalf of

Dr. David Giofrè

Academic Editor

PLOS ONE

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    Submitted filename: Response to Reviewers.docx

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    Submitted filename: Rebuttal Letter Rev 2#.docx

    Data Availability Statement

    All data collected are available at the following link: https://osf.io/qwapm/?view_only=2da5876783c842779a4d1a9380784688 as reported also in the paper.


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